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• What Is a Case Study? | Definition, Examples & Methods

What Is a Case Study? | Definition, Examples & Methods

Published on May 8, 2019 by Shona McCombes . Revised on November 20, 2023.

A case study is a detailed study of a specific subject, such as a person, group, place, event, organization, or phenomenon. Case studies are commonly used in social, educational, clinical, and business research.

A case study research design usually involves qualitative methods , but quantitative methods are sometimes also used. Case studies are good for describing , comparing, evaluating and understanding different aspects of a research problem .

Table of contents

When to do a case study, step 1: select a case, step 2: build a theoretical framework, step 3: collect your data, step 4: describe and analyze the case, other interesting articles.

A case study is an appropriate research design when you want to gain concrete, contextual, in-depth knowledge about a specific real-world subject. It allows you to explore the key characteristics, meanings, and implications of the case.

Case studies are often a good choice in a thesis or dissertation . They keep your project focused and manageable when you don’t have the time or resources to do large-scale research.

You might use just one complex case study where you explore a single subject in depth, or conduct multiple case studies to compare and illuminate different aspects of your research problem.

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Once you have developed your problem statement and research questions , you should be ready to choose the specific case that you want to focus on. A good case study should have the potential to:

• Provide new or unexpected insights into the subject
• Challenge or complicate existing assumptions and theories
• Propose practical courses of action to resolve a problem
• Open up new directions for future research

TipIf your research is more practical in nature and aims to simultaneously investigate an issue as you solve it, consider conducting action research instead.

Unlike quantitative or experimental research , a strong case study does not require a random or representative sample. In fact, case studies often deliberately focus on unusual, neglected, or outlying cases which may shed new light on the research problem.

Example of an outlying case studyIn the 1960s the town of Roseto, Pennsylvania was discovered to have extremely low rates of heart disease compared to the US average. It became an important case study for understanding previously neglected causes of heart disease.

However, you can also choose a more common or representative case to exemplify a particular category, experience or phenomenon.

Example of a representative case studyIn the 1920s, two sociologists used Muncie, Indiana as a case study of a typical American city that supposedly exemplified the changing culture of the US at the time.

While case studies focus more on concrete details than general theories, they should usually have some connection with theory in the field. This way the case study is not just an isolated description, but is integrated into existing knowledge about the topic. It might aim to:

• Exemplify a theory by showing how it explains the case under investigation
• Expand on a theory by uncovering new concepts and ideas that need to be incorporated
• Challenge a theory by exploring an outlier case that doesn’t fit with established assumptions

To ensure that your analysis of the case has a solid academic grounding, you should conduct a literature review of sources related to the topic and develop a theoretical framework . This means identifying key concepts and theories to guide your analysis and interpretation.

There are many different research methods you can use to collect data on your subject. Case studies tend to focus on qualitative data using methods such as interviews , observations , and analysis of primary and secondary sources (e.g., newspaper articles, photographs, official records). Sometimes a case study will also collect quantitative data.

Example of a mixed methods case studyFor a case study of a wind farm development in a rural area, you could collect quantitative data on employment rates and business revenue, collect qualitative data on local people’s perceptions and experiences, and analyze local and national media coverage of the development.

The aim is to gain as thorough an understanding as possible of the case and its context.

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In writing up the case study, you need to bring together all the relevant aspects to give as complete a picture as possible of the subject.

How you report your findings depends on the type of research you are doing. Some case studies are structured like a standard scientific paper or thesis , with separate sections or chapters for the methods , results and discussion .

Others are written in a more narrative style, aiming to explore the case from various angles and analyze its meanings and implications (for example, by using textual analysis or discourse analysis ).

In all cases, though, make sure to give contextual details about the case, connect it back to the literature and theory, and discuss how it fits into wider patterns or debates.

If you want to know more about statistics , methodology , or research bias , make sure to check out some of our other articles with explanations and examples.

• Normal distribution
• Degrees of freedom
• Null hypothesis
• Discourse analysis
• Control groups
• Mixed methods research
• Non-probability sampling
• Quantitative research
• Ecological validity

Research bias

• Rosenthal effect
• Implicit bias
• Cognitive bias
• Selection bias
• Negativity bias
• Status quo bias

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How to write a case study — examples, templates, and tools

It’s a marketer’s job to communicate the effectiveness of a product or service to potential and current customers to convince them to buy and keep business moving. One of the best methods for doing this is to share success stories that are relatable to prospects and customers based on their pain points, experiences, and overall needs.

That’s where case studies come in. Case studies are an essential part of a content marketing plan. These in-depth stories of customer experiences are some of the most effective at demonstrating the value of a product or service. Yet many marketers don’t use them, whether because of their regimented formats or the process of customer involvement and approval.

A case study is a powerful tool for showcasing your hard work and the success your customer achieved. But writing a great case study can be difficult if you’ve never done it before or if it’s been a while. This guide will show you how to write an effective case study and provide real-world examples and templates that will keep readers engaged and support your business.

In this article, you’ll learn:

What is a case study?

How to write a case study, case study templates, case study examples, case study tools.

A case study is the detailed story of a customer’s experience with a product or service that demonstrates their success and often includes measurable outcomes. Case studies are used in a range of fields and for various reasons, from business to academic research. They’re especially impactful in marketing as brands work to convince and convert consumers with relatable, real-world stories of actual customer experiences.

The best case studies tell the story of a customer’s success, including the steps they took, the results they achieved, and the support they received from a brand along the way. To write a great case study, you need to:

• Celebrate the customer and make them — not a product or service — the star of the story.
• Craft the story with specific audiences or target segments in mind so that the story of one customer will be viewed as relatable and actionable for another customer.
• Write copy that is easy to read and engaging so that readers will gain the insights and messages intended.
• Follow a standardized format that includes all of the essentials a potential customer would find interesting and useful.
• Support all of the claims for success made in the story with data in the forms of hard numbers and customer statements.

Case studies are a type of review but more in depth, aiming to show — rather than just tell — the positive experiences that customers have with a brand. Notably, 89% of consumers read reviews before deciding to buy, and 79% view case study content as part of their purchasing process. When it comes to B2B sales, 52% of buyers rank case studies as an important part of their evaluation process.

Telling a brand story through the experience of a tried-and-true customer matters. The story is relatable to potential new customers as they imagine themselves in the shoes of the company or individual featured in the case study. Showcasing previous customers can help new ones see themselves engaging with your brand in the ways that are most meaningful to them.

Besides sharing the perspective of another customer, case studies stand out from other content marketing forms because they are based on evidence. Whether pulling from client testimonials or data-driven results, case studies tend to have more impact on new business because the story contains information that is both objective (data) and subjective (customer experience) — and the brand doesn’t sound too self-promotional.

Case studies are unique in that there’s a fairly standardized format for telling a customer’s story. But that doesn’t mean there isn’t room for creativity. It’s all about making sure that teams are clear on the goals for the case study — along with strategies for supporting content and channels — and understanding how the story fits within the framework of the company’s overall marketing goals.

Here are the basic steps to writing a good case study.

1. Identify your goal

Start by defining exactly who your case study will be designed to help. Case studies are about specific instances where a company works with a customer to achieve a goal. Identify which customers are likely to have these goals, as well as other needs the story should cover to appeal to them.

The answer is often found in one of the buyer personas that have been constructed as part of your larger marketing strategy. This can include anything from new leads generated by the marketing team to long-term customers that are being pressed for cross-sell opportunities. In all of these cases, demonstrating value through a relatable customer success story can be part of the solution to conversion.

2. Choose your client or subject

Who you highlight matters. Case studies tie brands together that might otherwise not cross paths. A writer will want to ensure that the highlighted customer aligns with their own company’s brand identity and offerings. Look for a customer with positive name recognition who has had great success with a product or service and is willing to be an advocate.

The client should also match up with the identified target audience. Whichever company or individual is selected should be a reflection of other potential customers who can see themselves in similar circumstances, having the same problems and possible solutions.

Some of the most compelling case studies feature customers who:

• Switch from one product or service to another while naming competitors that missed the mark.
• Experience measurable results that are relatable to others in a specific industry.
• Represent well-known brands and recognizable names that are likely to compel action.
• Advocate for a product or service as a champion and are well-versed in its advantages.

Whoever or whatever customer is selected, marketers must ensure they have the permission of the company involved before getting started. Some brands have strict review and approval procedures for any official marketing or promotional materials that include their name. Acquiring those approvals in advance will prevent any miscommunication or wasted effort if there is an issue with their legal or compliance teams.

3. Conduct research and compile data

Substantiating the claims made in a case study — either by the marketing team or customers themselves — adds validity to the story. To do this, include data and feedback from the client that defines what success looks like. This can be anything from demonstrating return on investment (ROI) to a specific metric the customer was striving to improve. Case studies should prove how an outcome was achieved and show tangible results that indicate to the customer that your solution is the right one.

This step could also include customer interviews. Make sure that the people being interviewed are key stakeholders in the purchase decision or deployment and use of the product or service that is being highlighted. Content writers should work off a set list of questions prepared in advance. It can be helpful to share these with the interviewees beforehand so they have time to consider and craft their responses. One of the best interview tactics to keep in mind is to ask questions where yes and no are not natural answers. This way, your subject will provide more open-ended responses that produce more meaningful content.

4. Choose the right format

There are a number of different ways to format a case study. Depending on what you hope to achieve, one style will be better than another. However, there are some common elements to include, such as:

• An engaging headline
• A subject and customer introduction
• The unique challenge or challenges the customer faced
• The solution the customer used to solve the problem
• The results achieved
• Data and statistics to back up claims of success
• A strong call to action (CTA) to engage with the vendor

It’s also important to note that while case studies are traditionally written as stories, they don’t have to be in a written format. Some companies choose to get more creative with their case studies and produce multimedia content, depending on their audience and objectives. Case study formats can include traditional print stories, interactive web or social content, data-heavy infographics, professionally shot videos, podcasts, and more.

5. Write your case study

We’ll go into more detail later about how exactly to write a case study, including templates and examples. Generally speaking, though, there are a few things to keep in mind when writing your case study.

• Be clear and concise. Readers want to get to the point of the story quickly and easily, and they’ll be looking to see themselves reflected in the story right from the start.
• Provide a big picture. Always make sure to explain who the client is, their goals, and how they achieved success in a short introduction to engage the reader.
• Construct a clear narrative. Stick to the story from the perspective of the customer and what they needed to solve instead of just listing product features or benefits.
• Leverage graphics. Incorporating infographics, charts, and sidebars can be a more engaging and eye-catching way to share key statistics and data in readable ways.
• Offer the right amount of detail. Most case studies are one or two pages with clear sections that a reader can skim to find the information most important to them.
• Include data to support claims. Show real results — both facts and figures and customer quotes — to demonstrate credibility and prove the solution works.

6. Promote your story

Marketers have a number of options for distribution of a freshly minted case study. Many brands choose to publish case studies on their website and post them on social media. This can help support SEO and organic content strategies while also boosting company credibility and trust as visitors see that other businesses have used the product or service.

Marketers are always looking for quality content they can use for lead generation. Consider offering a case study as gated content behind a form on a landing page or as an offer in an email message. One great way to do this is to summarize the content and tease the full story available for download after the user takes an action.

Sales teams can also leverage case studies, so be sure they are aware that the assets exist once they’re published. Especially when it comes to larger B2B sales, companies often ask for examples of similar customer challenges that have been solved.

Now that you’ve learned a bit about case studies and what they should include, you may be wondering how to start creating great customer story content. Here are a couple of templates you can use to structure your case study.

Template 1 — Challenge-solution-result format

• Start with an engaging title. This should be fewer than 70 characters long for SEO best practices. One of the best ways to approach the title is to include the customer’s name and a hint at the challenge they overcame in the end.
• Create an introduction. Lead with an explanation as to who the customer is, the need they had, and the opportunity they found with a specific product or solution. Writers can also suggest the success the customer experienced with the solution they chose.
• Present the challenge. This should be several paragraphs long and explain the problem the customer faced and the issues they were trying to solve. Details should tie into the company’s products and services naturally. This section needs to be the most relatable to the reader so they can picture themselves in a similar situation.
• Share the solution. Explain which product or service offered was the ideal fit for the customer and why. Feel free to delve into their experience setting up, purchasing, and onboarding the solution.
• Explain the results. Demonstrate the impact of the solution they chose by backing up their positive experience with data. Fill in with customer quotes and tangible, measurable results that show the effect of their choice.
• Ask for action. Include a CTA at the end of the case study that invites readers to reach out for more information, try a demo, or learn more — to nurture them further in the marketing pipeline. What you ask of the reader should tie directly into the goals that were established for the case study in the first place.

Template 2 — Data-driven format

• Start with an engaging title. Be sure to include a statistic or data point in the first 70 characters. Again, it’s best to include the customer’s name as part of the title.
• Create an overview. Share the customer’s background and a short version of the challenge they faced. Present the reason a particular product or service was chosen, and feel free to include quotes from the customer about their selection process.
• Present data point 1. Isolate the first metric that the customer used to define success and explain how the product or solution helped to achieve this goal. Provide data points and quotes to substantiate the claim that success was achieved.
• Present data point 2. Isolate the second metric that the customer used to define success and explain what the product or solution did to achieve this goal. Provide data points and quotes to substantiate the claim that success was achieved.
• Present data point 3. Isolate the final metric that the customer used to define success and explain what the product or solution did to achieve this goal. Provide data points and quotes to substantiate the claim that success was achieved.
• Summarize the results. Reiterate the fact that the customer was able to achieve success thanks to a specific product or service. Include quotes and statements that reflect customer satisfaction and suggest they plan to continue using the solution.
• Ask for action. Include a CTA at the end of the case study that asks readers to reach out for more information, try a demo, or learn more — to further nurture them in the marketing pipeline. Again, remember that this is where marketers can look to convert their content into action with the customer.

While templates are helpful, seeing a case study in action can also be a great way to learn. Here are some examples of how Adobe customers have experienced success.

Juniper Networks

One example is the Adobe and Juniper Networks case study , which puts the reader in the customer’s shoes. The beginning of the story quickly orients the reader so that they know exactly who the article is about and what they were trying to achieve. Solutions are outlined in a way that shows Adobe Experience Manager is the best choice and a natural fit for the customer. Along the way, quotes from the client are incorporated to help add validity to the statements. The results in the case study are conveyed with clear evidence of scale and volume using tangible data.

The story of Lenovo’s journey with Adobe is one that spans years of planning, implementation, and rollout. The Lenovo case study does a great job of consolidating all of this into a relatable journey that other enterprise organizations can see themselves taking, despite the project size. This case study also features descriptive headers and compelling visual elements that engage the reader and strengthen the content.

Tata Consulting

When it comes to using data to show customer results, this case study does an excellent job of conveying details and numbers in an easy-to-digest manner. Bullet points at the start break up the content while also helping the reader understand exactly what the case study will be about. Tata Consulting used Adobe to deliver elevated, engaging content experiences for a large telecommunications client of its own — an objective that’s relatable for a lot of companies.

Case studies are a vital tool for any marketing team as they enable you to demonstrate the value of your company’s products and services to others. They help marketers do their job and add credibility to a brand trying to promote its solutions by using the experiences and stories of real customers.

When you’re ready to get started with a case study:

• Think about a few goals you’d like to accomplish with your content.
• Make a list of successful clients that would be strong candidates for a case study.
• Reach out to the client to get their approval and conduct an interview.
• Gather the data to present an engaging and effective customer story.

Adobe can help

There are several Adobe products that can help you craft compelling case studies. Adobe Experience Platform helps you collect data and deliver great customer experiences across every channel. Once you’ve created your case studies, Experience Platform will help you deliver the right information to the right customer at the right time for maximum impact.

To learn more, watch the Adobe Experience Platform story .

Keep in mind that the best case studies are backed by data. That’s where Adobe Real-Time Customer Data Platform and Adobe Analytics come into play. With Real-Time CDP, you can gather the data you need to build a great case study and target specific customers to deliver the content to the right audience at the perfect moment.

Watch the Real-Time CDP overview video to learn more.

Finally, Adobe Analytics turns real-time data into real-time insights. It helps your business collect and synthesize data from multiple platforms to make more informed decisions and create the best case study possible.

Request a demo to learn more about Adobe Analytics.

https://business.adobe.com/blog/perspectives/b2b-ecommerce-10-case-studies-inspire-you

https://business.adobe.com/blog/basics/business-case

https://business.adobe.com/blog/basics/what-is-real-time-analytics

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What Is a Case Study?

Weighing the pros and cons of this method of research

Kendra Cherry, MS, is a psychosocial rehabilitation specialist, psychology educator, and author of the "Everything Psychology Book."

Cara Lustik is a fact-checker and copywriter.

Verywell / Colleen Tighe

• Pros and Cons

What Types of Case Studies Are Out There?

Where do you find data for a case study, how do i write a psychology case study.

A case study is an in-depth study of one person, group, or event. In a case study, nearly every aspect of the subject's life and history is analyzed to seek patterns and causes of behavior. Case studies can be used in many different fields, including psychology, medicine, education, anthropology, political science, and social work.

The point of a case study is to learn as much as possible about an individual or group so that the information can be generalized to many others. Unfortunately, case studies tend to be highly subjective, and it is sometimes difficult to generalize results to a larger population.

While case studies focus on a single individual or group, they follow a format similar to other types of psychology writing. If you are writing a case study, we got you—here are some rules of APA format to reference.  

At a Glance

A case study, or an in-depth study of a person, group, or event, can be a useful research tool when used wisely. In many cases, case studies are best used in situations where it would be difficult or impossible for you to conduct an experiment. They are helpful for looking at unique situations and allow researchers to gather a lot of˜ information about a specific individual or group of people. However, it's important to be cautious of any bias we draw from them as they are highly subjective.

What Are the Benefits and Limitations of Case Studies?

A case study can have its strengths and weaknesses. Researchers must consider these pros and cons before deciding if this type of study is appropriate for their needs.

One of the greatest advantages of a case study is that it allows researchers to investigate things that are often difficult or impossible to replicate in a lab. Some other benefits of a case study:

• Allows researchers to capture information on the 'how,' 'what,' and 'why,' of something that's implemented
• Gives researchers the chance to collect information on why one strategy might be chosen over another
• Permits researchers to develop hypotheses that can be explored in experimental research

On the other hand, a case study can have some drawbacks:

• It cannot necessarily be generalized to the larger population
• Cannot demonstrate cause and effect
• It may not be scientifically rigorous
• It can lead to bias

Researchers may choose to perform a case study if they want to explore a unique or recently discovered phenomenon. Through their insights, researchers develop additional ideas and study questions that might be explored in future studies.

It's important to remember that the insights from case studies cannot be used to determine cause-and-effect relationships between variables. However, case studies may be used to develop hypotheses that can then be addressed in experimental research.

Case Study Examples

There have been a number of notable case studies in the history of psychology. Much of  Freud's work and theories were developed through individual case studies. Some great examples of case studies in psychology include:

• Anna O : Anna O. was a pseudonym of a woman named Bertha Pappenheim, a patient of a physician named Josef Breuer. While she was never a patient of Freud's, Freud and Breuer discussed her case extensively. The woman was experiencing symptoms of a condition that was then known as hysteria and found that talking about her problems helped relieve her symptoms. Her case played an important part in the development of talk therapy as an approach to mental health treatment.
• Phineas Gage : Phineas Gage was a railroad employee who experienced a terrible accident in which an explosion sent a metal rod through his skull, damaging important portions of his brain. Gage recovered from his accident but was left with serious changes in both personality and behavior.
• Genie : Genie was a young girl subjected to horrific abuse and isolation. The case study of Genie allowed researchers to study whether language learning was possible, even after missing critical periods for language development. Her case also served as an example of how scientific research may interfere with treatment and lead to further abuse of vulnerable individuals.

Such cases demonstrate how case research can be used to study things that researchers could not replicate in experimental settings. In Genie's case, her horrific abuse denied her the opportunity to learn a language at critical points in her development.

This is clearly not something researchers could ethically replicate, but conducting a case study on Genie allowed researchers to study phenomena that are otherwise impossible to reproduce.

There are a few different types of case studies that psychologists and other researchers might use:

• Collective case studies : These involve studying a group of individuals. Researchers might study a group of people in a certain setting or look at an entire community. For example, psychologists might explore how access to resources in a community has affected the collective mental well-being of those who live there.
• Descriptive case studies : These involve starting with a descriptive theory. The subjects are then observed, and the information gathered is compared to the pre-existing theory.
• Explanatory case studies : These   are often used to do causal investigations. In other words, researchers are interested in looking at factors that may have caused certain things to occur.
• Exploratory case studies : These are sometimes used as a prelude to further, more in-depth research. This allows researchers to gather more information before developing their research questions and hypotheses .
• Instrumental case studies : These occur when the individual or group allows researchers to understand more than what is initially obvious to observers.
• Intrinsic case studies : This type of case study is when the researcher has a personal interest in the case. Jean Piaget's observations of his own children are good examples of how an intrinsic case study can contribute to the development of a psychological theory.

The three main case study types often used are intrinsic, instrumental, and collective. Intrinsic case studies are useful for learning about unique cases. Instrumental case studies help look at an individual to learn more about a broader issue. A collective case study can be useful for looking at several cases simultaneously.

The type of case study that psychology researchers use depends on the unique characteristics of the situation and the case itself.

There are a number of different sources and methods that researchers can use to gather information about an individual or group. Six major sources that have been identified by researchers are:

• Archival records : Census records, survey records, and name lists are examples of archival records.
• Direct observation : This strategy involves observing the subject, often in a natural setting . While an individual observer is sometimes used, it is more common to utilize a group of observers.
• Documents : Letters, newspaper articles, administrative records, etc., are the types of documents often used as sources.
• Interviews : Interviews are one of the most important methods for gathering information in case studies. An interview can involve structured survey questions or more open-ended questions.
• Participant observation : When the researcher serves as a participant in events and observes the actions and outcomes, it is called participant observation.
• Physical artifacts : Tools, objects, instruments, and other artifacts are often observed during a direct observation of the subject.

If you have been directed to write a case study for a psychology course, be sure to check with your instructor for any specific guidelines you need to follow. If you are writing your case study for a professional publication, check with the publisher for their specific guidelines for submitting a case study.

Here is a general outline of what should be included in a case study.

Section 1: A Case History

This section will have the following structure and content:

Background information : The first section of your paper will present your client's background. Include factors such as age, gender, work, health status, family mental health history, family and social relationships, drug and alcohol history, life difficulties, goals, and coping skills and weaknesses.

Description of the presenting problem : In the next section of your case study, you will describe the problem or symptoms that the client presented with.

Describe any physical, emotional, or sensory symptoms reported by the client. Thoughts, feelings, and perceptions related to the symptoms should also be noted. Any screening or diagnostic assessments that are used should also be described in detail and all scores reported.

Your diagnosis : Provide your diagnosis and give the appropriate Diagnostic and Statistical Manual code. Explain how you reached your diagnosis, how the client's symptoms fit the diagnostic criteria for the disorder(s), or any possible difficulties in reaching a diagnosis.

Section 2: Treatment Plan

This portion of the paper will address the chosen treatment for the condition. This might also include the theoretical basis for the chosen treatment or any other evidence that might exist to support why this approach was chosen.

• Cognitive behavioral approach : Explain how a cognitive behavioral therapist would approach treatment. Offer background information on cognitive behavioral therapy and describe the treatment sessions, client response, and outcome of this type of treatment. Make note of any difficulties or successes encountered by your client during treatment.
• Humanistic approach : Describe a humanistic approach that could be used to treat your client, such as client-centered therapy . Provide information on the type of treatment you chose, the client's reaction to the treatment, and the end result of this approach. Explain why the treatment was successful or unsuccessful.
• Psychoanalytic approach : Describe how a psychoanalytic therapist would view the client's problem. Provide some background on the psychoanalytic approach and cite relevant references. Explain how psychoanalytic therapy would be used to treat the client, how the client would respond to therapy, and the effectiveness of this treatment approach.
• Pharmacological approach : If treatment primarily involves the use of medications, explain which medications were used and why. Provide background on the effectiveness of these medications and how monotherapy may compare with an approach that combines medications with therapy or other treatments.

This section of a case study should also include information about the treatment goals, process, and outcomes.

When you are writing a case study, you should also include a section where you discuss the case study itself, including the strengths and limitiations of the study. You should note how the findings of your case study might support previous research. 

In your discussion section, you should also describe some of the implications of your case study. What ideas or findings might require further exploration? How might researchers go about exploring some of these questions in additional studies?

Need More Tips?

Here are a few additional pointers to keep in mind when formatting your case study:

• Never refer to the subject of your case study as "the client." Instead, use their name or a pseudonym.
• Read examples of case studies to gain an idea about the style and format.
• Remember to use APA format when citing references .

Crowe S, Cresswell K, Robertson A, Huby G, Avery A, Sheikh A. The case study approach .  BMC Med Res Methodol . 2011;11:100.

Crowe S, Cresswell K, Robertson A, Huby G, Avery A, Sheikh A. The case study approach . BMC Med Res Methodol . 2011 Jun 27;11:100. doi:10.1186/1471-2288-11-100

Gagnon, Yves-Chantal.  The Case Study as Research Method: A Practical Handbook . Canada, Chicago Review Press Incorporated DBA Independent Pub Group, 2010.

Yin, Robert K. Case Study Research and Applications: Design and Methods . United States, SAGE Publications, 2017.

By Kendra Cherry, MSEd Kendra Cherry, MS, is a psychosocial rehabilitation specialist, psychology educator, and author of the "Everything Psychology Book."

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• Methodology
• Case Study | Definition, Examples & Methods

Case Study | Definition, Examples & Methods

Published on 5 May 2022 by Shona McCombes . Revised on 30 January 2023.

A case study is a detailed study of a specific subject, such as a person, group, place, event, organisation, or phenomenon. Case studies are commonly used in social, educational, clinical, and business research.

A case study research design usually involves qualitative methods , but quantitative methods are sometimes also used. Case studies are good for describing , comparing, evaluating, and understanding different aspects of a research problem .

Table of contents

When to do a case study, step 1: select a case, step 2: build a theoretical framework, step 3: collect your data, step 4: describe and analyse the case.

A case study is an appropriate research design when you want to gain concrete, contextual, in-depth knowledge about a specific real-world subject. It allows you to explore the key characteristics, meanings, and implications of the case.

Case studies are often a good choice in a thesis or dissertation . They keep your project focused and manageable when you don’t have the time or resources to do large-scale research.

You might use just one complex case study where you explore a single subject in depth, or conduct multiple case studies to compare and illuminate different aspects of your research problem.

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Once you have developed your problem statement and research questions , you should be ready to choose the specific case that you want to focus on. A good case study should have the potential to:

• Provide new or unexpected insights into the subject
• Challenge or complicate existing assumptions and theories
• Propose practical courses of action to resolve a problem
• Open up new directions for future research

Unlike quantitative or experimental research, a strong case study does not require a random or representative sample. In fact, case studies often deliberately focus on unusual, neglected, or outlying cases which may shed new light on the research problem.

If you find yourself aiming to simultaneously investigate and solve an issue, consider conducting action research . As its name suggests, action research conducts research and takes action at the same time, and is highly iterative and flexible. 

However, you can also choose a more common or representative case to exemplify a particular category, experience, or phenomenon.

While case studies focus more on concrete details than general theories, they should usually have some connection with theory in the field. This way the case study is not just an isolated description, but is integrated into existing knowledge about the topic. It might aim to:

• Exemplify a theory by showing how it explains the case under investigation
• Expand on a theory by uncovering new concepts and ideas that need to be incorporated
• Challenge a theory by exploring an outlier case that doesn’t fit with established assumptions

To ensure that your analysis of the case has a solid academic grounding, you should conduct a literature review of sources related to the topic and develop a theoretical framework . This means identifying key concepts and theories to guide your analysis and interpretation.

There are many different research methods you can use to collect data on your subject. Case studies tend to focus on qualitative data using methods such as interviews, observations, and analysis of primary and secondary sources (e.g., newspaper articles, photographs, official records). Sometimes a case study will also collect quantitative data .

The aim is to gain as thorough an understanding as possible of the case and its context.

In writing up the case study, you need to bring together all the relevant aspects to give as complete a picture as possible of the subject.

How you report your findings depends on the type of research you are doing. Some case studies are structured like a standard scientific paper or thesis, with separate sections or chapters for the methods , results , and discussion .

Others are written in a more narrative style, aiming to explore the case from various angles and analyse its meanings and implications (for example, by using textual analysis or discourse analysis ).

In all cases, though, make sure to give contextual details about the case, connect it back to the literature and theory, and discuss how it fits into wider patterns or debates.

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What the Case Study Method Really Teaches

• Nitin Nohria

Seven meta-skills that stick even if the cases fade from memory.

It’s been 100 years since Harvard Business School began using the case study method. Beyond teaching specific subject matter, the case study method excels in instilling meta-skills in students. This article explains the importance of seven such skills: preparation, discernment, bias recognition, judgement, collaboration, curiosity, and self-confidence.

During my decade as dean of Harvard Business School, I spent hundreds of hours talking with our alumni. To enliven these conversations, I relied on a favorite question: “What was the most important thing you learned from your time in our MBA program?”

• Nitin Nohria is the George F. Baker Jr. Professor at Harvard Business School and the former dean of HBS.

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• Open access
• Published: 06 April 2016

How to review a case report

• Rakesh Garg 1 ,
• Shaheen E. Lakhan 2 &
• Ananda K. Dhanasekaran 3  

Journal of Medical Case Reports volume  10 , Article number:  88 ( 2016 ) Cite this article

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Peer Review reports

Introduction

Sharing individual patient experiences with clinical colleagues is an essential component of learning from each other. This sharing of information may be made global by reporting in a scientific journal. In medicine, patient management decisions are generally based on the evidence available for use of a particular investigation or technology [ 1 ]. The hierarchical rank of the evidence signifies the probability of bias. The higher up the hierarchy, the better its reliability and thus its clinical acceptance (Table  1 ). Though case reports remain lowest in the hierarchy of evidence, with meta-analysis representing the highest level, they nevertheless constitute important information with regard to rare events and may be considered as anecdotal evidence [ 2 ] (Table  1 ). Case reports may stimulate the generation of new hypotheses, and thus may support the emergence of new research.

The definition of a case report or a case series is not well defined in the literature and has been defined variously by different journals and authors. However, the basic definition of a case report is the detailed report of an individual including aspects like exposure, symptoms, signs, intervention, and outcome. It has been suggested that a report with more than four cases be called a case series and those with fewer than four a case report [ 3 ]. A case series is descriptive in design. Other authors describe “a collection of patients” as a case series and “a few patients” as a case report [ 4 ]. We suggest that should more than one case be reported, it may be defined as a case series—a concept proposed by other authors [ 5 ].

The importance of case reports

A case report may describe an unusual etiology, an unusual or unknown disorder, a challenging differential diagnosis, an unusual setting for care, information that can not be reproduced due to ethical reasons, unusual or puzzling clinical features, improved or unique technical procedures, unusual interactions, rare or novel adverse reactions to care, or new insight into the pathogenesis of disease [ 6 , 7 ]. In recent years, the publication of case reports has been given low priority by many high impact factor journals. However, the need for reporting such events remains. There are some journals dedicated purely to case reports, such as the Journal of Medical Case Reports , emphasizing their importance in modern literature. In the past, isolated case reports have led to significant advancements in patient care. For example, case reports concerning pulmonary hypertension and anorexic agents led to further trials and the identification of the mechanism and risk factors associated with these agents [ 2 , 8 ].

Reporting and publishing requirements

The reporting of cases varies for different journals. The authors need to follow the instructions for the intended publication. Owing to significant variability, it would be difficult to have uniform publication guidelines for case reports. A checklist called the CARE guidelines is useful for authors writing case reports [ 9 , 10 ]. However, it would be universally prudent to include a title, keywords, abstract, introduction, patient information, clinical findings, timeline, diagnostic assessment, therapeutic interventions, follow-up and outcomes, discussion, patient perspective, and informed consent.

Peer review process

The peer review process is an essential part of ethical and scientific writing. Peer review ultimately helps improve articles by providing valuable feedback to the author and helps editors make a decision regarding publication. The peer reviewer should provide unbiased, constructive feedback regarding the manuscript. They may also highlight the strengths and weaknesses of the report. When reviewing an article, it is prudent to read the entire manuscript first to understand the overall content and message. The reviewer than may read section-wise and provide comments to the authors and editorial team accordingly. The reviewer needs to consider the following important points when reviewing a case for possible publication [ 8 , 9 ] (summarized in Table  2 ).

Novelty remains the foremost important aspect of a case. The case report should introduce novel aspects of patient evaluation, investigation, treatment, or any other aspect related to patient care. The relevant information becomes a hypothesis generator for further study. The novelty may at times be balanced with some important information like severe adverse effects, even if they have been reported earlier. Reporting adverse events remains important so that information on cumulative adverse effects can be gathered globally, which helps in preparing a policy or guideline or a warning note for its use in patients. The data related to adverse effects include not only the impact but also the number of patients affected. This becomes more important for serious adverse effects. In the absence of an international registry for adverse effects, published case reports are important pieces of information. Owing to ethical concerns, formal evaluation may not be feasible in the format of prospective study.

Essential description

The case needs to have all essential details to allow a useful conclusion to emerge. For example, if a case is being reported for hemodynamic variability due to a drug, then the drug dose and timing along with timed vital signs need to be described.

Authenticity and genuineness

Honesty remains the most important basic principle of all publications. This remains a primary responsibility of the authors. However, if there is any doubt, reviewers may seek clarification. This doubt may result from some discordance in the case description. At times, a lack of correlation between the figures and description may act as “red flags.” For instance, authors may discuss a technique for dealing with a difficult airway, but the figure is of a normal-appearing airway. Another example would be where the data and figure do not correlate in a hemodynamic response related to a drug or a technique, with the graphical picture or screenshot of hemodynamics acting as an alert sign. Such cause for concern may be communicated in confidence to the editor.

Ethical or competing interests

Ethical issues need to be cautiously interpreted and communicated. The unethical use of a drug or device is not desirable and often unworthy of publication. This may relate to the route or dose of the drug administered. The off-label use of drugs where known side effects are greater than potential benefit needs to be discouraged and remains an example of unethical use. This use may be related to the drug dose, particularly when the drug dose exceeds the routine recommended dose, or to the route of administration. As an example, the maximal dose of acetaminophen (paracetamol) is 4g/day, and if an author reports exceeding this dose, it should be noted why a greater than recommended dose was used. Ultimately, the use of a drug or its route of administration needs to be justified in the manuscript. The reviewers need to serve as content experts regarding the drugs and other technologies used in the case. A literature search by the reviewer provides the data to comment on this aspect.

Competing interests (or conflicts of interest) are concerns that interfere or potentially interfere with presentation, review, or publication. They must be declared by the authors. Conflicts can relate to patient-related professional attributes (like the use of a particular procedure, drug, or instrument) being affected by some secondary gains (financial, non-financial, professional, personal). Financial conflict may be related to ownership, paid consultancy, patents, grants, honoraria, and gifts. Non-financial conflicts may be related to memberships, relationships, appearance as an expert witness, or personal convictions. At times, the conflict may be related to the author’s relationship with an organization or another person. A conflict may influence the interpretation of the outcome in an inappropriate and unscientific manner. Although conflicts may not be totally abolished, they must be disclosed when they reasonably exist. This disclosure should include information such as funding sources, present membership, and patents pending. Reviewers should cautiously interpret any potential bias regarding the outcome of the case based on the reported conflicts. This is essential for transparent reporting of research. At times, competing interests may be discovered by a reviewer and should be included in comments to the editorial team. Such conflicts may again be ascertained when the reviewer reviews the literature during the peer review process. The reviewer should also disclose their own conflicts related to the manuscript review when sending their report to the editorial team.

Impact on clinical practice

This is an important aspect for the final decision of whether to publish a case report. The main thrust or carry-home message needs to be emphasized clearly. It needs to be elaborated upon in concluding remarks.

Patient anonymity, consent, and ethical approval

When reviewing the manuscript of a case report, reviewers should ensure that the patient’s anonymity and confidentiality is protected. The reviewers should check that patient identifiers have been removed or masked from all aspects of the manuscript, whether in writing or within photograph. Identifiers can include things like the name of the patient, geographical location, date of birth, phone numbers, email of the patient, medical record numbers, or biometric identifiers. Utmost care needs to be taken to provide full anonymity for the patient.

Consent is required to participate in research, receive a certain treatment, and publish identifiable details. These consents are for different purposes and need to be explained separately to the patient. A patient’s consent to participate in the research or for use of the drug may not extend to consent for publication. All these aspects of consent must be explained to the patient, written explicitly in the patient’s own language, understood by the patient, and signed by the patient. For the purpose of the case, the patient must understand and consent for any new technique or drug (its dose, route, and timing) being used. In the case of a drug being used for a non-standard indication or route, consent for use must also be described. Patient consent is essential for the publication of a case if patient body parts are displayed in the article. This also includes any identifiers that can reveal the identity of the patient, such as the patient’s hospital identification number, address, and any other unique identifier. In situations where revealing the patient’s identity cannot be fully avoided, for example if the report requires an image of an identifiable body part like the face, then this should be explained to the patient, the image shown to them, and consent taken. Should the patient die, then consent must be obtained from next of kin or legal representative.

With case series, securing individual patient consent is advised and preferable. The authors may also need institutional review board (IRB) approval to publish a case series. IRBs can waive the need for consent if a study is conducted retrospectively and data are collected from patient notes for the purpose of research, usually in an anonymized way. However, wherever possible, individual patient consent is preferable, even for a retrospective study. Consent is mandatory for any prospective data collection for the purpose of publication as a case series. Consent and/or IRB approval must be disclosed in the case report and reasons for not obtaining individual consent may be described, if applicable.

There may be situations in which publishing patient details without their consent is justified, but this is a decision that should be made by the journal editor, who may decide to discuss the case with the Committee on Publication Ethics. Reviewers need to emphasize the issue to the editor when submitting their comments.

Manuscript writing

The CARE guidelines provide a framework that supports transparency and accuracy in the publication of case reports and the reporting of information from patient encounters. The acronym CARE was created from CA (the first two letters in “case”) and RE (the first two letters in “reports”). The initial CARE tools are the CARE checklist and the Case Report Writing Templates. These tools support the writing of case reports and provide data that inform clinical practice guidelines and provide early signals of effectiveness, harms, and cost [ 10 ].

The presentation of the case and its interpretation should be comprehensive and related. The various components of the manuscript should have sufficient information for understanding the key message of the case. The reviewer needs to comment on the relevant components of the manuscript. The reviewer should ascertain that the title of the case manuscript is relevant and includes keywords related to the case. The title should be short, descriptive, and interesting. The abstract should be brief, without any abbreviations, and include keywords. It is preferable to use Medical Subject Headings (MeSH) keywords. Reviewers must ensure that the introduction emphasizes the context of the case and describes the relevance and its importance in a concise and comprehensive manner. The case description should be complete and should follow basic rules of medical communication. The details regarding patient history, physical examination, investigations, differential diagnosis, management, and outcome should be described in chronological order. If repeated observations are present, then they may be tabulated. The use of graphs and figures helps the readers to better understand the case. Interpretation or inferences based on the outcomes should be avoided in this section and should be considered a part of the discussion. The discussion should highlight important aspects of the case, with its interpretation within the context of the available literature. References should be formatted as per the journal style. They should be complete and preferably of recent publications.

Reviewer responsibility

The reviewer’s remarks are essential not only for the editorial team but also for authors. A good peer review requires honesty, sincerity, and punctuality. Even if a manuscript is rejected, the authors should receive learning points from peer review commentary. The best way to review a manuscript is to read the manuscript in full for a gross overview and develop general comments. Thereafter, the reviewer should address each section of the manuscript separately and precisely. This may be done after a literature search if the reviewer needs to substantiate his/her commentary.

Constructive criticism

The reviewer’s remarks should be constructive to help the authors improve the manuscript for further consideration. If the manuscript is rejected, the authors should have a clear indication for the rejection. The remarks may be grouped as major and minor comments. Major comments likely suggest changes to the whole presentation, changing the primary aim of the case report, or adding images. Minor comments may include grammatical errors or getting references for a statement. The editorial team must be able to justify their decision on whether or not to accept an article for publication, often by citing peer review feedback. It is also good style to tabulate a list of the strengths and weaknesses of the manuscript.

Fixed time for review

Reviewer remarks should be submitted within a specified timeframe. If any delay is expected, it should be communicated to the editorial team. Reviewers should not rush to submit feedback without sufficient time to adequately review the paper and perform any necessary literature searches. Should a reviewer be unable to submit the review within the specified timeframe, they should reply to the review invitation to decline at their earliest convenience. If, after accepting a review invitation, the reviewer realizes they do not have time to perform the review, this must be communicated to the editorial team.

Conflict of interest

The reviewer’s conflicts of interest should be included along with the review. The conflicts may be related to the contents of the case, drugs, or devices pertaining to the case; the author(s); or the affiliated institution(s) of the author(s).

Lack of expertise

The reviewer may decline to review the manuscript if they think the topic is out of their area of expertise. If, after accepting an invitation to review, the reviewer realizes they are unable to review the manuscript owing to a lack of expertise in that particular field, they should disclose the fact to the editorial team.

Confidentiality

The reviewer should keep the manuscript confidential and should not use the contents of the unpublished manuscript in any form. Discussing the manuscript among colleagues or any scientific forum or meetings is inappropriate.

Review of revised manuscript

At times, a manuscript is sent for re-review to the reviewer. The reviewer should read the revised manuscript, the author’s response to the previous round of peer review, and the editorial comments. Sometimes, the authors may disagree with the reviewer’s remarks. This issue needs to be elaborated on and communicated with the editor. The reviewer should support their views with appropriate literature references. If the authors justify their reason for disagreeing with the viewer, then their argument should be considered evidence-based. However, if the reviewer still requests the revision, this may be politely communicated to the author and editor with justification for the same. In response to reviewers remarks, authors may not agree fully and provide certain suggestion in the form of clarification related to reviewers remarks. The reviewers should take these clarifications judiciously and comment accordingly with the intent of improving the manuscript further.

Peer reviewers have a significant role in the dissemination of scientific literature. They act as gatekeepers for science before it is released to society. Their sincerity and dedication is paramount to the success of any journal. The reviewers should follow a scientific and justifiable methodology for reviewing a case report for possible publication. Their comments should be constructive for the overall improvement of the manuscript and aid the editorial team in making a decision on publication. We hope this article will help reviewers to perform their important role in the best way possible. We send our best wishes to the reviewer community and, for those who are inspired to become reviewers after reading this article, our warm welcome to the reviewers’ club.

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Department of Anaesthesiology, Pain and Palliative Care, DR BRAIRCH, AIIMS, Ansari Nagar, New Delhi, 110029, India

Rakesh Garg

Neurology and Medical Education, California University of Science and Medicine - School of Medicine, Colton, CA, USA

Shaheen E. Lakhan

Sandwell & West Birmingham Hospitals, NHS Trust, Birmingham, UK

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Garg, R., Lakhan, S.E. & Dhanasekaran, A.K. How to review a case report. J Med Case Reports 10 , 88 (2016). https://doi.org/10.1186/s13256-016-0853-3

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Definition and Introduction

Case analysis is a problem-based teaching and learning method that involves critically analyzing complex scenarios within an organizational setting for the purpose of placing the student in a “real world” situation and applying reflection and critical thinking skills to contemplate appropriate solutions, decisions, or recommended courses of action. It is considered a more effective teaching technique than in-class role playing or simulation activities. The analytical process is often guided by questions provided by the instructor that ask students to contemplate relationships between the facts and critical incidents described in the case.

Cases generally include both descriptive and statistical elements and rely on students applying abductive reasoning to develop and argue for preferred or best outcomes [i.e., case scenarios rarely have a single correct or perfect answer based on the evidence provided]. Rather than emphasizing theories or concepts, case analysis assignments emphasize building a bridge of relevancy between abstract thinking and practical application and, by so doing, teaches the value of both within a specific area of professional practice.

Given this, the purpose of a case analysis paper is to present a structured and logically organized format for analyzing the case situation. It can be assigned to students individually or as a small group assignment and it may include an in-class presentation component. Case analysis is predominately taught in economics and business-related courses, but it is also a method of teaching and learning found in other applied social sciences disciplines, such as, social work, public relations, education, journalism, and public administration.

Ellet, William. The Case Study Handbook: A Student's Guide . Revised Edition. Boston, MA: Harvard Business School Publishing, 2018; Christoph Rasche and Achim Seisreiner. Guidelines for Business Case Analysis . University of Potsdam; Writing a Case Analysis . Writing Center, Baruch College; Volpe, Guglielmo. "Case Teaching in Economics: History, Practice and Evidence." Cogent Economics and Finance 3 (December 2015). doi:https://doi.org/10.1080/23322039.2015.1120977.

How to Approach Writing a Case Analysis Paper

The organization and structure of a case analysis paper can vary depending on the organizational setting, the situation, and how your professor wants you to approach the assignment. Nevertheless, preparing to write a case analysis paper involves several important steps. As Hawes notes, a case analysis assignment “...is useful in developing the ability to get to the heart of a problem, analyze it thoroughly, and to indicate the appropriate solution as well as how it should be implemented” [p.48]. This statement encapsulates how you should approach preparing to write a case analysis paper.

Before you begin to write your paper, consider the following analytical procedures:

• Review the case to get an overview of the situation . A case can be only a few pages in length, however, it is most often very lengthy and contains a significant amount of detailed background information and statistics, with multilayered descriptions of the scenario, the roles and behaviors of various stakeholder groups, and situational events. Therefore, a quick reading of the case will help you gain an overall sense of the situation and illuminate the types of issues and problems that you will need to address in your paper. If your professor has provided questions intended to help frame your analysis, use them to guide your initial reading of the case.
• Read the case thoroughly . After gaining a general overview of the case, carefully read the content again with the purpose of understanding key circumstances, events, and behaviors among stakeholder groups. Look for information or data that appears contradictory, extraneous, or misleading. At this point, you should be taking notes as you read because this will help you develop a general outline of your paper. The aim is to obtain a complete understanding of the situation so that you can begin contemplating tentative answers to any questions your professor has provided or, if they have not provided, developing answers to your own questions about the case scenario and its connection to the course readings,lectures, and class discussions.
• Determine key stakeholder groups, issues, and events and the relationships they all have to each other . As you analyze the content, pay particular attention to identifying individuals, groups, or organizations described in the case and identify evidence of any problems or issues of concern that impact the situation in a negative way. Other things to look for include identifying any assumptions being made by or about each stakeholder, potential biased explanations or actions, explicit demands or ultimatums , and the underlying concerns that motivate these behaviors among stakeholders. The goal at this stage is to develop a comprehensive understanding of the situational and behavioral dynamics of the case and the explicit and implicit consequences of each of these actions.
• Identify the core problems . The next step in most case analysis assignments is to discern what the core [i.e., most damaging, detrimental, injurious] problems are within the organizational setting and to determine their implications. The purpose at this stage of preparing to write your analysis paper is to distinguish between the symptoms of core problems and the core problems themselves and to decide which of these must be addressed immediately and which problems do not appear critical but may escalate over time. Identify evidence from the case to support your decisions by determining what information or data is essential to addressing the core problems and what information is not relevant or is misleading.
• Explore alternative solutions . As noted, case analysis scenarios rarely have only one correct answer. Therefore, it is important to keep in mind that the process of analyzing the case and diagnosing core problems, while based on evidence, is a subjective process open to various avenues of interpretation. This means that you must consider alternative solutions or courses of action by critically examining strengths and weaknesses, risk factors, and the differences between short and long-term solutions. For each possible solution or course of action, consider the consequences they may have related to their implementation and how these recommendations might lead to new problems. Also, consider thinking about your recommended solutions or courses of action in relation to issues of fairness, equity, and inclusion.
• Decide on a final set of recommendations . The last stage in preparing to write a case analysis paper is to assert an opinion or viewpoint about the recommendations needed to help resolve the core problems as you see them and to make a persuasive argument for supporting this point of view. Prepare a clear rationale for your recommendations based on examining each element of your analysis. Anticipate possible obstacles that could derail their implementation. Consider any counter-arguments that could be made concerning the validity of your recommended actions. Finally, describe a set of criteria and measurable indicators that could be applied to evaluating the effectiveness of your implementation plan.

Use these steps as the framework for writing your paper. Remember that the more detailed you are in taking notes as you critically examine each element of the case, the more information you will have to draw from when you begin to write. This will save you time.

NOTE : If the process of preparing to write a case analysis paper is assigned as a student group project, consider having each member of the group analyze a specific element of the case, including drafting answers to the corresponding questions used by your professor to frame the analysis. This will help make the analytical process more efficient and ensure that the distribution of work is equitable. This can also facilitate who is responsible for drafting each part of the final case analysis paper and, if applicable, the in-class presentation.

Framework for Case Analysis . College of Management. University of Massachusetts; Hawes, Jon M. "Teaching is Not Telling: The Case Method as a Form of Interactive Learning." Journal for Advancement of Marketing Education 5 (Winter 2004): 47-54; Rasche, Christoph and Achim Seisreiner. Guidelines for Business Case Analysis . University of Potsdam; Writing a Case Study Analysis . University of Arizona Global Campus Writing Center; Van Ness, Raymond K. A Guide to Case Analysis . School of Business. State University of New York, Albany; Writing a Case Analysis . Business School, University of New South Wales.

Structure and Writing Style

A case analysis paper should be detailed, concise, persuasive, clearly written, and professional in tone and in the use of language . As with other forms of college-level academic writing, declarative statements that convey information, provide a fact, or offer an explanation or any recommended courses of action should be based on evidence. If allowed by your professor, any external sources used to support your analysis, such as course readings, should be properly cited under a list of references. The organization and structure of case analysis papers can vary depending on your professor’s preferred format, but its structure generally follows the steps used for analyzing the case.

Introduction

The introduction should provide a succinct but thorough descriptive overview of the main facts, issues, and core problems of the case . The introduction should also include a brief summary of the most relevant details about the situation and organizational setting. This includes defining the theoretical framework or conceptual model on which any questions were used to frame your analysis.

Following the rules of most college-level research papers, the introduction should then inform the reader how the paper will be organized. This includes describing the major sections of the paper and the order in which they will be presented. Unless you are told to do so by your professor, you do not need to preview your final recommendations in the introduction. U nlike most college-level research papers , the introduction does not include a statement about the significance of your findings because a case analysis assignment does not involve contributing new knowledge about a research problem.

Background Analysis

Background analysis can vary depending on any guiding questions provided by your professor and the underlying concept or theory that the case is based upon. In general, however, this section of your paper should focus on:

• Providing an overarching analysis of problems identified from the case scenario, including identifying events that stakeholders find challenging or troublesome,
• Identifying assumptions made by each stakeholder and any apparent biases they may exhibit,
• Describing any demands or claims made by or forced upon key stakeholders, and
• Highlighting any issues of concern or complaints expressed by stakeholders in response to those demands or claims.

These aspects of the case are often in the form of behavioral responses expressed by individuals or groups within the organizational setting. However, note that problems in a case situation can also be reflected in data [or the lack thereof] and in the decision-making, operational, cultural, or institutional structure of the organization. Additionally, demands or claims can be either internal and external to the organization [e.g., a case analysis involving a president considering arms sales to Saudi Arabia could include managing internal demands from White House advisors as well as demands from members of Congress].

Throughout this section, present all relevant evidence from the case that supports your analysis. Do not simply claim there is a problem, an assumption, a demand, or a concern; tell the reader what part of the case informed how you identified these background elements.

Identification of Problems

In most case analysis assignments, there are problems, and then there are problems . Each problem can reflect a multitude of underlying symptoms that are detrimental to the interests of the organization. The purpose of identifying problems is to teach students how to differentiate between problems that vary in severity, impact, and relative importance. Given this, problems can be described in three general forms: those that must be addressed immediately, those that should be addressed but the impact is not severe, and those that do not require immediate attention and can be set aside for the time being.

All of the problems you identify from the case should be identified in this section of your paper, with a description based on evidence explaining the problem variances. If the assignment asks you to conduct research to further support your assessment of the problems, include this in your explanation. Remember to cite those sources in a list of references. Use specific evidence from the case and apply appropriate concepts, theories, and models discussed in class or in relevant course readings to highlight and explain the key problems [or problem] that you believe must be solved immediately and describe the underlying symptoms and why they are so critical.

Alternative Solutions

This section is where you provide specific, realistic, and evidence-based solutions to the problems you have identified and make recommendations about how to alleviate the underlying symptomatic conditions impacting the organizational setting. For each solution, you must explain why it was chosen and provide clear evidence to support your reasoning. This can include, for example, course readings and class discussions as well as research resources, such as, books, journal articles, research reports, or government documents. In some cases, your professor may encourage you to include personal, anecdotal experiences as evidence to support why you chose a particular solution or set of solutions. Using anecdotal evidence helps promote reflective thinking about the process of determining what qualifies as a core problem and relevant solution .

Throughout this part of the paper, keep in mind the entire array of problems that must be addressed and describe in detail the solutions that might be implemented to resolve these problems.

Recommended Courses of Action

In some case analysis assignments, your professor may ask you to combine the alternative solutions section with your recommended courses of action. However, it is important to know the difference between the two. A solution refers to the answer to a problem. A course of action refers to a procedure or deliberate sequence of activities adopted to proactively confront a situation, often in the context of accomplishing a goal. In this context, proposed courses of action are based on your analysis of alternative solutions. Your description and justification for pursuing each course of action should represent the overall plan for implementing your recommendations.

For each course of action, you need to explain the rationale for your recommendation in a way that confronts challenges, explains risks, and anticipates any counter-arguments from stakeholders. Do this by considering the strengths and weaknesses of each course of action framed in relation to how the action is expected to resolve the core problems presented, the possible ways the action may affect remaining problems, and how the recommended action will be perceived by each stakeholder.

In addition, you should describe the criteria needed to measure how well the implementation of these actions is working and explain which individuals or groups are responsible for ensuring your recommendations are successful. In addition, always consider the law of unintended consequences. Outline difficulties that may arise in implementing each course of action and describe how implementing the proposed courses of action [either individually or collectively] may lead to new problems [both large and small].

Throughout this section, you must consider the costs and benefits of recommending your courses of action in relation to uncertainties or missing information and the negative consequences of success.

The conclusion should be brief and introspective. Unlike a research paper, the conclusion in a case analysis paper does not include a summary of key findings and their significance, a statement about how the study contributed to existing knowledge, or indicate opportunities for future research.

Begin by synthesizing the core problems presented in the case and the relevance of your recommended solutions. This can include an explanation of what you have learned about the case in the context of your answers to the questions provided by your professor. The conclusion is also where you link what you learned from analyzing the case with the course readings or class discussions. This can further demonstrate your understanding of the relationships between the practical case situation and the theoretical and abstract content of assigned readings and other course content.

Problems to Avoid

The literature on case analysis assignments often includes examples of difficulties students have with applying methods of critical analysis and effectively reporting the results of their assessment of the situation. A common reason cited by scholars is that the application of this type of teaching and learning method is limited to applied fields of social and behavioral sciences and, as a result, writing a case analysis paper can be unfamiliar to most students entering college.

After you have drafted your paper, proofread the narrative flow and revise any of these common errors:

• Unnecessary detail in the background section . The background section should highlight the essential elements of the case based on your analysis. Focus on summarizing the facts and highlighting the key factors that become relevant in the other sections of the paper by eliminating any unnecessary information.
• Analysis relies too much on opinion . Your analysis is interpretive, but the narrative must be connected clearly to evidence from the case and any models and theories discussed in class or in course readings. Any positions or arguments you make should be supported by evidence.
• Analysis does not focus on the most important elements of the case . Your paper should provide a thorough overview of the case. However, the analysis should focus on providing evidence about what you identify are the key events, stakeholders, issues, and problems. Emphasize what you identify as the most critical aspects of the case to be developed throughout your analysis. Be thorough but succinct.
• Writing is too descriptive . A paper with too much descriptive information detracts from your analysis of the complexities of the case situation. Questions about what happened, where, when, and by whom should only be included as essential information leading to your examination of questions related to why, how, and for what purpose.
• Inadequate definition of a core problem and associated symptoms . A common error found in case analysis papers is recommending a solution or course of action without adequately defining or demonstrating that you understand the problem. Make sure you have clearly described the problem and its impact and scope within the organizational setting. Ensure that you have adequately described the root causes w hen describing the symptoms of the problem.
• Recommendations lack specificity . Identify any use of vague statements and indeterminate terminology, such as, “A particular experience” or “a large increase to the budget.” These statements cannot be measured and, as a result, there is no way to evaluate their successful implementation. Provide specific data and use direct language in describing recommended actions.
• Unrealistic, exaggerated, or unattainable recommendations . Review your recommendations to ensure that they are based on the situational facts of the case. Your recommended solutions and courses of action must be based on realistic assumptions and fit within the constraints of the situation. Also note that the case scenario has already happened, therefore, any speculation or arguments about what could have occurred if the circumstances were different should be revised or eliminated.

Bee, Lian Song et al. "Business Students' Perspectives on Case Method Coaching for Problem-Based Learning: Impacts on Student Engagement and Learning Performance in Higher Education." Education & Training 64 (2022): 416-432; The Case Analysis . Fred Meijer Center for Writing and Michigan Authors. Grand Valley State University; Georgallis, Panikos and Kayleigh Bruijn. "Sustainability Teaching using Case-Based Debates." Journal of International Education in Business 15 (2022): 147-163; Hawes, Jon M. "Teaching is Not Telling: The Case Method as a Form of Interactive Learning." Journal for Advancement of Marketing Education 5 (Winter 2004): 47-54; Georgallis, Panikos, and Kayleigh Bruijn. "Sustainability Teaching Using Case-based Debates." Journal of International Education in Business 15 (2022): 147-163; .Dean,  Kathy Lund and Charles J. Fornaciari. "How to Create and Use Experiential Case-Based Exercises in a Management Classroom." Journal of Management Education 26 (October 2002): 586-603; Klebba, Joanne M. and Janet G. Hamilton. "Structured Case Analysis: Developing Critical Thinking Skills in a Marketing Case Course." Journal of Marketing Education 29 (August 2007): 132-137, 139; Klein, Norman. "The Case Discussion Method Revisited: Some Questions about Student Skills." Exchange: The Organizational Behavior Teaching Journal 6 (November 1981): 30-32; Mukherjee, Arup. "Effective Use of In-Class Mini Case Analysis for Discovery Learning in an Undergraduate MIS Course." The Journal of Computer Information Systems 40 (Spring 2000): 15-23; Pessoa, Silviaet al. "Scaffolding the Case Analysis in an Organizational Behavior Course: Making Analytical Language Explicit." Journal of Management Education 46 (2022): 226-251: Ramsey, V. J. and L. D. Dodge. "Case Analysis: A Structured Approach." Exchange: The Organizational Behavior Teaching Journal 6 (November 1981): 27-29; Schweitzer, Karen. "How to Write and Format a Business Case Study." ThoughtCo. https://www.thoughtco.com/how-to-write-and-format-a-business-case-study-466324 (accessed December 5, 2022); Reddy, C. D. "Teaching Research Methodology: Everything's a Case." Electronic Journal of Business Research Methods 18 (December 2020): 178-188; Volpe, Guglielmo. "Case Teaching in Economics: History, Practice and Evidence." Cogent Economics and Finance 3 (December 2015). doi:https://doi.org/10.1080/23322039.2015.1120977.

Writing Tip

Ca se Study and Case Analysis Are Not the Same!

Confusion often exists between what it means to write a paper that uses a case study research design and writing a paper that analyzes a case; they are two different types of approaches to learning in the social and behavioral sciences. Professors as well as educational researchers contribute to this confusion because they often use the term "case study" when describing the subject of analysis for a case analysis paper. But you are not studying a case for the purpose of generating a comprehensive, multi-faceted understanding of a research problem. R ather, you are critically analyzing a specific scenario to argue logically for recommended solutions and courses of action that lead to optimal outcomes applicable to professional practice.

To avoid any confusion, here are twelve characteristics that delineate the differences between writing a paper using the case study research method and writing a case analysis paper:

• Case study is a method of in-depth research and rigorous inquiry ; case analysis is a reliable method of teaching and learning . A case study is a modality of research that investigates a phenomenon for the purpose of creating new knowledge, solving a problem, or testing a hypothesis using empirical evidence derived from the case being studied. Often, the results are used to generalize about a larger population or within a wider context. The writing adheres to the traditional standards of a scholarly research study. A case analysis is a pedagogical tool used to teach students how to reflect and think critically about a practical, real-life problem in an organizational setting.
• The researcher is responsible for identifying the case to study; a case analysis is assigned by your professor . As the researcher, you choose the case study to investigate in support of obtaining new knowledge and understanding about the research problem. The case in a case analysis assignment is almost always provided, and sometimes written, by your professor and either given to every student in class to analyze individually or to a small group of students, or students select a case to analyze from a predetermined list.
• A case study is indeterminate and boundless; a case analysis is predetermined and confined . A case study can be almost anything [see item 9 below] as long as it relates directly to examining the research problem. This relationship is the only limit to what a researcher can choose as the subject of their case study. The content of a case analysis is determined by your professor and its parameters are well-defined and limited to elucidating insights of practical value applied to practice.
• Case study is fact-based and describes actual events or situations; case analysis can be entirely fictional or adapted from an actual situation . The entire content of a case study must be grounded in reality to be a valid subject of investigation in an empirical research study. A case analysis only needs to set the stage for critically examining a situation in practice and, therefore, can be entirely fictional or adapted, all or in-part, from an actual situation.
• Research using a case study method must adhere to principles of intellectual honesty and academic integrity; a case analysis scenario can include misleading or false information . A case study paper must report research objectively and factually to ensure that any findings are understood to be logically correct and trustworthy. A case analysis scenario may include misleading or false information intended to deliberately distract from the central issues of the case. The purpose is to teach students how to sort through conflicting or useless information in order to come up with the preferred solution. Any use of misleading or false information in academic research is considered unethical.
• Case study is linked to a research problem; case analysis is linked to a practical situation or scenario . In the social sciences, the subject of an investigation is most often framed as a problem that must be researched in order to generate new knowledge leading to a solution. Case analysis narratives are grounded in real life scenarios for the purpose of examining the realities of decision-making behavior and processes within organizational settings. A case analysis assignments include a problem or set of problems to be analyzed. However, the goal is centered around the act of identifying and evaluating courses of action leading to best possible outcomes.
• The purpose of a case study is to create new knowledge through research; the purpose of a case analysis is to teach new understanding . Case studies are a choice of methodological design intended to create new knowledge about resolving a research problem. A case analysis is a mode of teaching and learning intended to create new understanding and an awareness of uncertainty applied to practice through acts of critical thinking and reflection.
• A case study seeks to identify the best possible solution to a research problem; case analysis can have an indeterminate set of solutions or outcomes . Your role in studying a case is to discover the most logical, evidence-based ways to address a research problem. A case analysis assignment rarely has a single correct answer because one of the goals is to force students to confront the real life dynamics of uncertainly, ambiguity, and missing or conflicting information within professional practice. Under these conditions, a perfect outcome or solution almost never exists.
• Case study is unbounded and relies on gathering external information; case analysis is a self-contained subject of analysis . The scope of a case study chosen as a method of research is bounded. However, the researcher is free to gather whatever information and data is necessary to investigate its relevance to understanding the research problem. For a case analysis assignment, your professor will often ask you to examine solutions or recommended courses of action based solely on facts and information from the case.
• Case study can be a person, place, object, issue, event, condition, or phenomenon; a case analysis is a carefully constructed synopsis of events, situations, and behaviors . The research problem dictates the type of case being studied and, therefore, the design can encompass almost anything tangible as long as it fulfills the objective of generating new knowledge and understanding. A case analysis is in the form of a narrative containing descriptions of facts, situations, processes, rules, and behaviors within a particular setting and under a specific set of circumstances.
• Case study can represent an open-ended subject of inquiry; a case analysis is a narrative about something that has happened in the past . A case study is not restricted by time and can encompass an event or issue with no temporal limit or end. For example, the current war in Ukraine can be used as a case study of how medical personnel help civilians during a large military conflict, even though circumstances around this event are still evolving. A case analysis can be used to elicit critical thinking about current or future situations in practice, but the case itself is a narrative about something finite and that has taken place in the past.
• Multiple case studies can be used in a research study; case analysis involves examining a single scenario . Case study research can use two or more cases to examine a problem, often for the purpose of conducting a comparative investigation intended to discover hidden relationships, document emerging trends, or determine variations among different examples. A case analysis assignment typically describes a stand-alone, self-contained situation and any comparisons among cases are conducted during in-class discussions and/or student presentations.

The Case Analysis . Fred Meijer Center for Writing and Michigan Authors. Grand Valley State University; Mills, Albert J. , Gabrielle Durepos, and Eiden Wiebe, editors. Encyclopedia of Case Study Research . Thousand Oaks, CA: SAGE Publications, 2010; Ramsey, V. J. and L. D. Dodge. "Case Analysis: A Structured Approach." Exchange: The Organizational Behavior Teaching Journal 6 (November 1981): 27-29; Yin, Robert K. Case Study Research and Applications: Design and Methods . 6th edition. Thousand Oaks, CA: Sage, 2017; Crowe, Sarah et al. “The Case Study Approach.” BMC Medical Research Methodology 11 (2011):  doi: 10.1186/1471-2288-11-100; Yin, Robert K. Case Study Research: Design and Methods . 4th edition. Thousand Oaks, CA: Sage Publishing; 1994.

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Case Study by Graeme Macrae Burnet review – unstable identities

This wry look at 1960s counterculture focuses on an enfant terrible of the anti-psychiatry movement to explore the gaps between appearance and reality

T he Booker-shortlisted 2015 novel His Bloody Project employed a range of narrative techniques to prod at the truth surrounding a murder in a 19th-century Scottish crofting community. Graeme Macrae Burnet’s concern was not so much with who committed the crime – we know that from the outset – but with the moral ambiguity inherent in assigning blame. His new novel, Case Study, is different in tone, though an interest in exploring complex psychological dramas through intricate narrative structures takes centre stage once again.

One of the key voices in His Bloody Project belongs to the prison doctor, charged with determining whether the accused is mentally fit to stand trial. The narrative spotlight in Case Study is focused on psychiatry itself, and how those who practise it are not always best qualified to pass judgment on the sanity or otherwise of those they purport to treat. The novel presents itself as the work of one “GMB”, a writer who has become interested in Collins Braithwaite, enfant terrible of the 1960s anti-psychiatry movement. After stumbling on Braithwaite’s “salacious, iconoclastic and compelling” collection of case studies, Untherapy, in a Glasgow bookshop, GMB toys with the idea of writing his biography. Although the plan meets with little enthusiasm from his agent and publisher, GMB’s fascination with Braithwaite is redoubled when he is contacted by a Mr Martin Grey, offering him six notebooks containing the journal of his cousin, whom Grey claims was a patient of Braithwaite. The notebooks contain “certain allegations” he is sure GMB will find of interest.

The notebooks are presented in full, interspersed with GMB’s biographical commentary. After giving up his studies at Oxford, Braithwaite spends a brief period working under RD Laing before pursuing his own more unorthodox path, later accusing his mentor of stealing his ideas. Railing against Laing’s success and unearned celebrity, Braithwaite sets himself up in practice near Primrose Hill, north London, an enterprise that seems doomed to failure until a chance encounter with Dirk Bogarde brings him an ever-expanding roster of celebrity clients. Braithwaite’s success is not to last, however, as his increasingly outrageous behaviour and monstrous egotism put him on a collision course with the law.

The six “Grey” notebooks offer the first-person account of an unnamed narrator, a young woman from a comfortable middle-class background whose older sister, Veronica, has recently killed herself. She believes the ultimate blame for Veronica’s death must lie with her psychotherapist, notorious “quack” Collins Braithwaite. Under the name Rebecca Smyth, the young woman books herself a consultation with Braithwaite, determined to discover the truth.

In his preface to the main text, GMB puts forward certain minor inaccuracies in the notebooks as grounds for questioning their authenticity, and as readers we would be advised to be equally suspicious. Those already familiar with Burnet’s writing have met GMB before, not only as the writer and researcher who claims distant kinship with the teenage murderer Roddy Macrae in His Bloody Project, but also as the translator of Burnet’s two “Raymond Brunet” crime novels. The defining essence of Burnet’s work to date is to be found in this kind of literary gamesmanship, a brand of metatextuality that is as much about exploiting the possibilities of the novel form as it is about blurring the boundaries between appearance and reality. In throwing us into doubt about which – and more crucially whose – story we are supposed to be following, Burnet encourages us to look more closely at the inherent instability of fiction itself. The painstakingly assembled, predominantly mimetic fiction of the 19th century has trained us to trust the author; Burnet has always delighted in undermining such easy assumptions, and in Case Study he ups the stakes still further, providing a veritable layer cake of possible realities to get lost in.

“Rebecca Smyth” tells us that in her sessions with Braithwaite he constantly questions her account of things, accusing her not only of inventing whole tracts of her past, but presenting him with an identity that is itself a construction. We know that in this at least Braithwaite is right, but with only the fictitious GMB’s word to go on that Braithwaite exists, it would be foolish for us to trust his suggestions or his analysis. The harder we tug on Burnet’s narrative threads, the more Veronica, her sister, and even Braithwaite himself start to look like different aspects of an unsteady unity.

In his rendering of the six notebooks, Burnet has cited the copious amounts of research he has undertaken, looking to the women’s magazines and journals of the 1950s and 60s in search of authenticity. While such publications might well reflect the moral tone and societal attitudes of the time, they are not necessarily an accurate representation of how young women in postwar England thought and felt. If we take the notebooks at face value, their shallowness and internalised misogyny quickly become irritating as well as unconvincing. If we choose to see them as satire, as part of the novel’s plot in a larger sense, they become something rather different.

As the notebooks progress, their unnamed narrator becomes ever more confused about her own identity. In wishing she was more like her invented alter ego, she begins to see Rebecca almost literally as a separate person, an uncanny simulacrum who can usurp her position and control her behaviour. In the biographical segments, GMB augments this with some interesting discursions on doubles in literature and Braithwaite’s Kierkegaard-inflected theories on the self. As the notebooks’ narrator slides further towards dissociation and depression, Case Study finally becomes a genuinely affecting discourse on mental health, the gulf between societal expectations and inward reality.

In pointed contrast to the gritty true-crime ambience of His Bloody Project, Case Study is above all a very funny book, a wry look back at 60s counterculture in which Burnet’s inventions rub shoulders with real personalities. But much as Braithwaite’s outlandish behaviour and performative rudeness might raise a knowing smile, his theories on identity and selfhood, appearance and reality are never as bonkers as we pretend they are. If Burnet’s aim in writing Case Study was to force us up against the contradictions of our conflicted selves, he has surely succeeded. This is a novel that is entertaining and mindfully engrossing in equal measure.

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The effect of “typical case discussion and scenario simulation” on the critical thinking of midwifery students: Evidence from China

• Yuji Wang 1   na1 ,
• Yijuan Peng 1   na1 &
• Yan Huang 1  

BMC Medical Education volume  24 , Article number:  340 ( 2024 ) Cite this article

Metrics details

Assessment ability lies at the core of midwives’ capacity to judge and treat clinical problems effectively. Influenced by the traditional teaching method of “teacher-led and content-based”, that teachers involve imparting a large amount of knowledge to students and students lack active thinking and active practice, the clinical assessment ability of midwifery students in China is mostly at a medium or low level. Improving clinical assessment ability of midwifery students, especially critical thinking, is highly important in practical midwifery education. Therefore, we implemented a new teaching program, “typical case discussion and scenario simulation”, in the Midwifery Health Assessment course. Guided by typical cases, students were organized to actively participate in typical case discussions and to promote active thinking and were encouraged to practice actively through scenario simulation. In this study, we aimed to evaluate the effect of this strategy on the critical thinking ability of midwifery students.

A total of 104 midwifery students in grades 16–19 at the West China School of Nursing, Sichuan University, were included as participants through convenience sampling. All the students completed the Midwifery Health Assessment course in the third year of university. Students in grades 16 and 17 were assigned to the control group, which received routine teaching in the Midwifery Health Assessment, while students in grades 18 and 19 were assigned to the experimental group, for which the “typical case discussion and scenario simulation” teaching mode was employed. The Critical Thinking Disposition Inventory-Chinese Version (CTDI-CV) and Midwifery Health Assessment Course Satisfaction Questionnaire were administered after the intervention.

After the intervention, the critical thinking ability of the experimental group was greater than that of the control group (284.81 ± 27.98 and 300.94 ± 31.67, p  = 0.008). Furthermore, the experimental group exhibited higher scores on the four dimensions of Open-Mindedness (40.56 ± 5.60 and 43.59 ± 4.90, p  = 0.005), Analyticity (42.83 ± 5.17 and 45.42 ± 5.72, p  = 0.020), Systematicity (38.79 ± 4.70 and 41.88 ± 6.11, p  = 0.006), and Critical Thinking Self-Confidence (41.35 ± 5.92 and 43.83 ± 5.89, p  = 0.039) than did the control group. The course satisfaction exhibited by the experimental group was greater than that exhibited by the control group (84.81 ± 8.49 and 90.19 ± 8.41, p  = 0.002).

The “typical case discussion and scenario simulation” class mode can improve the critical thinking ability of midwifery students and enhance their curriculum satisfaction. This approach carries a certain degree of promotional significance in medical education.

Typical case discussion and scenario simulation can improve midwifery students’ critical thinking ability.

Typical case discussion and scenario simulation can enhance students’ learning interest and guide students to learn independently.

Midwifery students were satisfied with the new teaching mode.

Peer Review reports

Maternal and neonatal health are important indicators to measure of the level of development of a country’s economy, culture and health care. The positive impact of quality midwifery education on maternal and newborn health is acknowledged in the publication framework for action strengthening quality midwifery education issued by the World Health Organization (WHO) [ 1 ]. Extensive evidence has shown that skilled midwifery care is crucial for reducing preventable maternal and neonatal mortality [ 2 , 3 , 4 ]. Clinical practice features high requirements for the clinical thinking ability of midwives, which refers to the process by which medical personnel analyze and integrate data with professional medical knowledge in the context of diagnosis and treatment as well as discover and solve problems through logical reasoning [ 5 ]. Critical thinking is a thoughtful process that is purposeful, disciplined, and self-directed and that aims to improve decisions and subsequent actions [ 6 ]. In 1986, the American Association of Colleges of Nursing formulated the “Higher Education Standards for Nursing Specialty”, which emphasize the fact that critical thinking is the primary core competence that nursing graduates should possess [ 7 ]. Many studies have shown that critical thinking can help nurses detect, analyze and solve problems creatively in clinical work and is a key factor in their ability to make correct clinical decisions [ 8 , 9 , 10 ].

However, the traditional teaching method used for midwifery students in China is “teacher-led and content-based”, and it involves efficiently and conveniently imparting a large amount of knowledge to students over a short period. Students have long failed to engage in active thinking and active practice, and the cultivation of critical thinking has long been ignored [ 5 ]. As a result, the critical thinking ability of midwifery students in China is mostly at a medium or low level [ 5 ]. Therefore, it is necessary to develop a new teaching mode to improve the critical thinking ability of midwifery students.

In 2014, Professor Xuexin Zhang of Fudan University, Shanghai, China, proposed a novel teaching method: the divided class mode. The basic idea of this approach is to divide the class time into two parts. The teachers explain the theoretical knowledge in the first lesson, and the students discuss that knowledge in the second lesson. This approach emphasizes the guiding role of teachers and encourages and empowers students to take responsibility for their studies [ 11 ]. Research has shown that the divided class mode can improve students’ enthusiasm and initiative as well as teaching effectiveness [ 12 ].

The problem-originated clinical medical curriculum mode of teaching was first established at McMaster University in Canada in 1965. This model is based on typical clinical cases and a problem-oriented heuristic teaching model [ 13 ]. The process of teaching used in this approach is guided by typical cases with the goal of helping students combine theoretical knowledge and practical skills. This approach can enhance the enthusiasm and initiative of students by establishing an active learning atmosphere. Students are encouraged to discuss and analyze typical cases to promote their ability to digest and absorb theoretical knowledge. Research has shown that the problem-originated clinical medical curriculum teaching mode can enhance students’ confidence and improve their autonomous learning and exploration ability. Scenario simulation teaching can provide students with real scenarios, allowing them to practice and apply their knowledge in a safe environment [ 14 ], which can effectively improve their knowledge and clinical skills and enhance their self-confidence [ 15 , 16 ].

Based on the teaching concept of divided classes, our research team established a new teaching model of “typical case discussion and scenario simulation”. Half of the class time is allocated for students to discuss typical cases and carry out scenario simulations to promote their active thinking and active practice. The Midwifery Health Assessment is the final professional core course that midwifery students must take in our school before clinical practice. All students must complete the course in Grade 3. Teaching this course is important for cultivating the critical thinking and clinical assessment ability of midwifery students. Therefore, our team adopted the new teaching mode of "typical case discussion and scenario simulation" in the teaching of this course. This study explored the teaching mode’s ability to improve the critical thinking ability of midwifery students.

Study design

The study employed a semiexperimental design.

Participants

A convenience sample of 104 third-year midwifery students who were enrolled in the Midwifery Health Assessment course volunteered to participate in this research at a large public university in Sichuan Province from February 2019 to June 2022 (grades 16 to 19). All the students completed the course in the third year of university. Students in grades 16 and 17 were assigned to the control group, which received the traditional teaching mode. Students in grades 18 and 19 were assigned to the experimental group, in which context the “typical case discussion and scenario simulation” class mode was used. The exclusion criteria for midwifery students were as follows: (1) dropped out of school during the study, (2) took continuous leave from school for more than two weeks, or (3) were unable to complete the questionnaire. The elimination criterion for midwifery students was that all the items were answered in the same way. No significant differences in students’ scores in their previous professional courses (Midwifery) were observed between the two groups. Textbooks, teachers, and teaching hours were the same for both groups.

Development of the “typical case discussion and scenario simulation” class mode

This study is based on the implementation of the new century higher education teaching reform project at Sichuan University. With the support of Sichuan University, we first established a “typical case discussion and scenario simulation” class mode team. The author of this paper was the head of the teaching reform project and served as a consultant, and the first author is responsible for supervising the implementation of the project. Second, the teaching team discussed and developed a standard process for the “typical case discussion and scenario simulation” class mode. Third, the entire team received intensive training in the standard process for the “typical case discussion and scenario simulation” class mode.

Implementation of the “typical case discussion and scenario simulation” class mode

Phase i (before class).

Before class, in accordance with the requirements for evaluating different periods of pregnancy, the teacher conceptualized typical cases and then discussed those cases with the teaching team and made any necessary modifications. After the completion of the discussion, the modified cases were released to the students through the class group. To ensure students’ interest, they were guided through the task of discovering and solving relevant problems using an autonomous learning approach.

Phase II (the first week)

Typical case discussion period. The Midwifery Health Assessment course was taught by 5 teachers and covered 5 health assessment periods, namely, the pregnancy preparation, pregnancy, delivery, puerperium and neonatal periods. The health assessment course focused on each period over 2 consecutive teaching weeks, and 2 lessons were taught per week. The first week focused on the discussion of typical cases. In the first lesson, teachers introduced typical cases, taught key knowledge or difficult evaluation content pertaining to the different periods, and explored the relevant knowledge framework. In the second lesson, teachers organized group discussions, case analyses and intergroup communications for the typical cases. They were also responsible for coordinating and encouraging students to participate actively in the discussion. After the discussion, teachers and students reviewed the definitions, treatments and evaluation points associated with the typical cases. The teachers also encouraged students to internalize knowledge by engaging in a process of summary and reflection to achieve the purpose of combining theory with practice.

Phase III (the second week)

Scenario simulation practice period. The second week focused on the scenario simulation practice period. In the first lesson, teachers reviewed the focus of assessment during the different periods and answered students’ questions. In the second lesson, students performed typical case assessment simulations in subgroups. After the simulation, the teachers commented on and summarized the students’ simulation evaluation and reviewed the evaluation points of typical cases to improve the students’ evaluation ability.

The organizational structure and implementation of the “typical case discussion and scenario simulation” class mode showed in Fig.  1 .

“Typical case discussion and scenario simulation” teaching mode diagram

A demographic questionnaire designed for this purpose was used to collect relevant information from participants, including age, gender, single-child status, family location, experience with typical case discussion or scenario simulation and scores in previous professional courses (Midwifery).

The Critical Thinking Disposition Inventory-Chinese Version (CTDI-CV) was developed by Peng et al. to evaluate the critical thinking ability of midwifery students [ 17 ]. The scale contains 70 items across a total of seven dimensions, namely, open-mindedness, truth-seeking, analytical ability, systematic ability, self-confidence in critical thinking, thirst for knowledge, and cognitive maturity. Each dimension is associated with 10 items, and each item is scored on a 6-point Likert scale, with 1 indicating “extremely agree” and 6 representing “extremely disagree”. The scale includes 30 positive items, which receive scores ranging from “extremely agree” to “extremely disagree” on a scale of 6 to 1, and 40 negative items, which receive scores ranging from “extremely agree” to “extremely disagree” on a scale of 1 to 6. A total score less than 210 indicates negative critical thinking ability, scores between 211 and 279 indicate an unclear meaning, scores of 280 or higher indicate positive critical thinking ability, and scores of 350 or higher indicate strong performance. The score range of each trait is 10–60 points; a score of 30 points or fewer indicates negative trait performance, scores between 31 and 39 points indicate that the trait meaning is incorrect, scores of 40 points or higher indicate positive trait performance, and scores of 50 points or higher indicate extremely positive trait performance. The Cronbach’s α coefficient of the scale was 0.90, thus indicating good content validity and structure. The higher an individual’s score on this measure is, the better that individual’s critical thinking ability.

The evaluation of teaching results was based on a questionnaire used to assess undergraduate course satisfaction, and the researchers deleted and modified items in the questionnaire to suit the context of the “typical case discussion and scenario simulation” teaching mode. Two rounds of discussion were held within the study group to form the final version of the Midwifery Health Assessment satisfaction questionnaire. The questionnaire evaluates the effect of teaching in terms of three dimensions, namely, curriculum content, curriculum teaching and curriculum evaluation. The questionnaire contains 21 items, each of which is scored on a 5-point Likert scale, with 1 indicating “extremely disagree” and 5 representing “extremely agree”. The higher the score is, the better the teaching effect.

Data collection and statistical analysis

We input the survey data into the “Wenjuanxing” platform ( https://www.wjx.cn/ ), which specializes in questionnaire services. At the beginning of the study, an electronic questionnaire was distributed to the students in the control group via student WeChat and QQ groups for data collection. After the intervention, an electronic questionnaire was distributed to the students in the experimental group for data collection in the final class of the Midwifery Health Assessment course. All the data were collected by the first author (Yuji Wang). When students had questions about the survey items, the first author (Yuji Wang) immediately explained the items in detail. To ensure the integrity of the questionnaire, the platform required all the items to be answered before submission.

Statistical Package for Social Sciences Version 26.0 (SPSS 26.0) software was used for data analysis. The Shapiro‒Wilk test was used to test the normality of the data. The measurement data are expressed as the mean ± standard deviation (X ± S), and an independent sample t test was used for comparisons among groups with a normal distribution. The data presented as the number of cases (%), and the chi-square test was performed. A P value < 0.05 indicated that a difference was statistically significant.

Ethical considerations

The study was funded by the New Century Teaching Reform Project of Sichuan University and passed the relevant ethical review. Oral informed consent was obtained from all individual participants in the study.

Characteristics of the participants

A total of 104 third-year midwifery students were enrolled from February 2019 to June 2022, and 98.1% (102/144) of these students completed the survey. Two invalid questionnaires that featured the same answers for each item were eliminated. A total of 100 participants were ultimately included in the analysis. Among the participants, 48 students were assigned to the control group, and 52 students were assigned to the experimental group. The age of the students ranged from 19 to 22 years, and the mean age of the control group was 20.50 years (SD = 0.61). The mean age of the experimental group was 20.63 years (SD = 0.65). Of the 100 students who participated in the study, the majority (96.0%) were women. No significant differences were observed between the intervention and control groups in terms of students’ demographic information (i.e., age, gender, status as an only child, or family location), experience with scenario simulation or typical case discussion and scores in previous Midwifery courses (Table  1 ).

Examining the differences in critical thinking ability between the two groups

The aim of this study was to evaluate the effect of the new teaching mode of “typical case discussion and scenario simulation” on improving the critical thinking ability of midwifery students. Independent sample t tests were used to examine the differences in critical thinking ability between the two groups (Table  2 ). The results showed that the total critical thinking scores obtained by the experimental group were greater than those obtained by the control group (284.81 ± 27.98 and 300.94 ± 31.67, p  = 0.008). The differences in four dimensions (Open-Mindedness (40.56 ± 5.60 and 43.59 ± 4.90, p  = 0.005), Analyticity (42.83 ± 5.17 and 45.42 ± 5.72, p  = 0.020), Systematicity (38.79 ± 4.70 and 41.88 ± 6.11, p  = 0.006), and Critical Thinking Self-Confidence (41.35 ± 5.92 and 43.83 ± 5.89, p  = 0.039)) were statistically significant.

Examining the differences in curriculum satisfaction between the two groups

To evaluate the effect of the new teaching mode of “the typical case discussion and scenario simulation” on the course satisfaction of midwifery students. Independent sample t tests were used to examine the differences in course satisfaction between the two groups (Table  3 ). The results showed that the curriculum satisfaction of the experimental group was greater than that of the control group (84.81 ± 8.49 and 90.19 ± 8.41, p  = 0.002). Independent sample t tests were used to examine the differences in the three dimensions of curriculum satisfaction between the two groups (Table  3 ). The results showed that the average scores of the intervention group on the three dimensions were significantly greater than those of the control group (curricular content: 20.83 ± 1.96 and 22.17 ± 2.23, p  = 0.002; curriculum teaching: 34.16 ± 3.89 and 36.59 ± 3.66, p  = 0.002; curriculum evaluation: 29.81 ± 3.27 and 31.42 ± 3.19, p  = 0.015).

Midwifery is practical and intensive work. To ensure maternal and child safety, midwives must make decisions and take action quickly. Therefore, midwives should have both critical thinking ability and clinical decision-making ability [ 18 ]. In addition, the Australian Nursing and Midwifery Accreditation Council (ANMAC) regulates the educational requirements for the programs required for registration as a midwife. According to these standards, education providers must incorporate learning activities into curricula to encourage the development and application of critical thinking and reflective practice [ 19 ]. Therefore, the challenge of cultivating the critical thinking ability of midwifery students is an urgent problem that must be solved. However, influenced by the traditional teaching method of “teacher-led and content-based”, the critical thinking ability of midwifery students in China is mostly at a medium or low level. In order to improve the critical thinking ability of midwifery students. Our research team has established a new teaching model, the “typical case discussion and scenario simulation” class model. And applied to the midwifery core curriculum Midwifery Health Assessment. This study aimed to investigate the implementation of a novel systematic and structured teaching model for midwifery students and to provide evidence regarding how to improve the critical thinking ability of midwives.

The results showed that the total CTDI-CV score obtained for the experimental group was greater than that obtained for the control group. These findings indicate that the “typical case discussion and scenario simulation” class mode had a positive effect on the cultivation of students’ critical thinking ability, a conclusion which is similar to the findings of Holdsworth et al. [ 20 ], Lapkin et al. [ 21 ] and Demirören M et al. [ 22 ]. We indicate the following reasons that may explain these results.The core aim of the typical case discussion teaching mode is to raise questions based on typical clinical cases and to provide heuristic teaching to students [ 23 ]. This approach emphasizes asking questions based on specific clinical cases, which enables students to engage in targeted learning. Moreover, scenario simulation allows students to attain certain inner experiences and emotions and actively participate in curriculum practice, which can enhance their ability to remember and understand knowledge [ 24 ]. Through the divided class mode, half of the class time was divided into the students. This method emphasizes the guiding role of teachers and encourages and empowers students to assume learning responsibilities. In addition, students can think, communicate and discuss actively [ 22 , 23 ]. Furthermore, this approach created opportunities for students to analyze and consider problems independently and give students sufficient time to internalize and absorb knowledge and deepen their understanding of relevant knowledge, which can increase their confidence in their ability to address such problems and improve their critical thinking ability [ 12 , 25 , 26 ].

In addition, the results showed that except for Truth-Seeking and Systematicity, the other five dimensions were all positive. These findings are similar to the results reported by Atakro et al.. and Sun et al. [ 27 , 28 ]. Through the intervention, the Systematicity scores became positive, suggesting that the new teaching mode can help students deal with problems in an organized and purposeful way. However, Truth-Seeking still did not become positive; this notion focuses on intellectual honesty, i.e., the disposition to be courageous when asking questions and to be honest and objective in the pursuit of knowledge even when the topics under investigation do not support one’s self-interest [ 29 ]. Studies have shown that this factor is related to the traditional teaching mode used [ 30 ]. The traditional teaching mode focuses on knowledge infusion, helps students remember the greatest possible amount of knowledge in a short time, and does not focus on guiding students to seek knowledge with sincerity and objectivity. Therefore, in future educational practice, we should focus on cultivating students’ ability to seek truth and engage in systematization.

Student evaluative feedback is an important way to test the effectiveness teaching mode. Therefore, understanding students’ evaluations of the effects of classroom teaching is key to promoting teaching reform and improving teaching quality. Therefore, we distributed a satisfaction questionnaire pertaining to the midwifery health assessment curriculum, which was based on the “typical case discussion and scenario simulation” class mode, with the goal of investigating curriculum satisfaction in terms of three dimensions (curriculum content, curriculum teaching and curriculum evaluation). The results showed that the satisfaction scores for each dimension increased significantly. This finding suggests that the new teaching method can enrich the teaching content, diversify the teaching mode and improve students’ curriculum evaluations.

In summary, the “typical case discussion and scenario simulation” class mode focuses on typical cases as its main content. Students’ understanding of this content is deepened through group discussion and scenario simulation. The subjectivity of students in curriculum learning should be accounted for. Students can be encouraged to detect, analyze and solve problems with the goal of improving their critical thinking ability. Moreover, this approach can also enhance curriculum satisfaction. It is recommended that these tools should be used continuously in future curriculum teaching.

This study has several limitations. First, the representativeness of the sample may be limited since the participants were recruited from specific universities in China. Second, we used historical controls, which are less effective than simultaneous controlled trials. Third, online self-report surveys are susceptible to response biases, although we included quality control measurements in the process of data collection. Fourth, we did not use the same critical thinking instrument, CTDI-CV, to investigate the critical thinking of the students in the experimental group or the control group before intervention but used professional course grades from the Midwifery for substitution comparison. This may not be a sufficient substitute. However, these comparisons could be helpful since those grades included some sort of evaluation of critical thinking. In light of these limitations, future multicenter simultaneous controlled studies should be conducted. Nonetheless, this study also has several strengths. First, no adjustment of teachers or change in learning materials occurred since the start of the midwifery health assessment, thus ensuring that the experimental and control groups featured the same teaching materials, teachers and teaching hours. In addition, to ensure the quality of the research, the first author of this paper participated in the entirety of the course teaching.

The “typical case discussion and scenario simulation” class mode can improve the critical thinking of midwifery students, which is helpful for ensuring maternal and child safety. Students are highly satisfied with the new teaching mode, and this approach has a certain degree of promotional significance. However, this approach also entails higher requirements for both teachers and students.

Availability of data and materials

The datasets used and/or analyzed during the current study are available from the corresponding author on reasonable request.

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The study was supported by Sichuan University’s New Century Education and Teaching Reform Project (SCU9316).

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Department of Nursing, West China Second University Hospital, Sichuan University/West China School of Nursing, Sichuan University/Key Laboratory of Birth Defects and Related Diseases of Women and Children (Sichuan University), No. 20 Third Section, Renmin South Road, Chengdu, Sichuan Province, 610041, China

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All authors contributed to the study conception and design. Material preparation, data collection and analysis were performed by Yuji Wang, Yijuan Peng and Yan Huang. The first draft of the manuscript were written by Yuji Wang and Yijuan Peng, and all authors commented on previous versions of the manuscript.

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Correspondence to Yan Huang .

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This study was supported by Sichuan University. And it was approved by the Ethics Review Committee of West China School of Nursing, Sichuan University. As it is a teaching research with no harm to samples, we only obtained oral informed consents from the participants including teachers and midwifery students and it was approved by the Ethics Review Committee of West China School of Nursing, Sichuan University(approval number 2021220). We comfirm that all methods were performed in accordance with the relevant guidelines and regulations in Ethics Approval and Consent to participate in Declarations.

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Wang, Y., Peng, Y. & Huang, Y. The effect of “typical case discussion and scenario simulation” on the critical thinking of midwifery students: Evidence from China. BMC Med Educ 24 , 340 (2024). https://doi.org/10.1186/s12909-024-05127-5

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Knowledge of cytology results affects the performance of colposcopy: a crossover study

• Eva Lalande 1 , 2   na1 ,
• Holly Clarke 1   na1 ,
• Manuela Undurraga 1 ,
• Vu Quoc Huy Nguyen 3 ,
• Cyril Jaksic 4 ,
• Frederic Goffin 5 ,
• Marc Arbyn 6 , 7 ,
• Jose Jeronimo 8 ,
• Jean-Christophe Tille 9 ,
• Essia Saiji 9 ,
• Pierre Vassilakos 1 , 10 &
• Patrick Petignat 1  

BMC Women's Health volume  24 , Article number:  189 ( 2024 ) Cite this article

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Metrics details

To determine whether knowledge of cytology affects the colposcopist’s diagnostic accuracy in the identification of cervical intraepithelial neoplasia grade 2 and worse (≥ CIN2).

In this cross-over study, healthcare professionals interpreted colposcopy images from 80 patient cases with known histological diagnoses. For each case, 2 images taken with a colposcope were provided (native and after acetic acid application). Inclusion criteria consisted of women with a transformation zone type 1 or 2, who had both a cytological and histological diagnosis. Cases were distributed across two online surveys, one including and one omitting the cytology. A wash-out period of six weeks between surveys was implemented. Colposcopists were asked to give their diagnosis for each case as < CIN2 or ≥ CIN2 on both assessments. Statistical analysis was conducted to compare the two interpretations.

Knowledge of cytology significantly improved the sensitivity when interpreting colposcopic images, from 51.1% [95%CI: 39.3 to 62.8] to 63.7% [95%CI: 52.1 to 73.9] and improved the specificity from 63.5% [95%CI: 52.3 to 73.5] to 76.6% [95%CI: 67.2 to 84.0]. Sensitivity was higher by 38.6% when a high-grade cytology (ASC-H, HSIL, AGC) was communicated compared to a low-grade cytology (inflammation, ASC-US, LSIL). Specificity was higher by 31% when a low-grade cytology was communicated compared to a high-grade.

Conclusions

Our data suggests that knowledge of cytology increases sensitivity and specificity for diagnosis of ≥ CIN2 lesions at colposcopy. Association between cytology and histology may have contributed to the findings.

Peer Review reports

Introduction

Cervical cancer affects a large number of women worldwide, and represents the fourth most frequent cancer diagnosed in this population [ 1 , 2 ]. In recent years, the incidence of cervical cancer in high-income countries has significantly declined, mainly due to effective prevention through vaccination, screening, diagnosis and treatment of pre-cancerous lesions [ 1 ]. Cytological screening contributes to early diagnosis of cervical precancerous lesions. When combined with colposcopy for women with an abnormal screen, it has been shown to be an effective standard for ≥ CIN2 (≥ cervical intra-epithelial neoplasia grade 2) diagnosis [ 3 ].

The colposcopy exam includes the observation of the native cervix as well as the cervix after application of 3–5% acetic acid solution to determine the presence of acetowhite lesions. Colposcopy allows localization of the lesion(s), evaluation of the lesion(s) severity, and facilitates directed biopsy for diagnosis. Results from this exam determine whether patients require biopsy, or if they can be managed conservatively with follow-up. In countries with screening programs, gynecologists may carry out colposcopies in diverse settings such as primary care, regional and tertiary hospitals. Some perform exams sporadically whilst others see large volumes, frequently in colposcopy clinics. Therefore, there is likely to be a great variability in the experience and training of colposcopists [ 4 ]. The diagnostic performance of colposcopy depends on the experience of the observers, and their skill in recognizing the acetowhitening of the epithelium after acetic acid application (thickness, color, border irregularity). Inconsistencies in performance for colposcopy have been demonstrated in the literature [ 5 , 6 ] particularly for the detection of high-grade abnormalities [ 7 ]. Reported sensitivity varies from 58 to 99% [ 8 , 9 , 10 , 11 , 12 , 13 ] while specificity ranges from 23 to 93% [ 8 , 9 , 10 , 11 , 12 , 13 ] depending also on the grade of the lesions. This range of values demonstrates the inherent subjectivity of this method, limited by the clinician’s capacity to distinguish lesions, appreciate their characteristics [ 6 ] and differentiate precancerous from benign appearances, leading in some cases to a misdiagnosis. To overcome the weakness of colposcopy, investigators have suggested systematically performing cervical biopsy even if the cervix appears normal. One study demonstrated that the sensitivity for detecting ≥ CIN2 increased from 61% (95%CI: 55–67) in a single biopsy to 86% (95%CI: 80–90) with two biopsies and to 96% (95%CI: 91–99) with three biopsies [ 14 ].

Colposcopy is generally conducted with knowledge of the cytology results, but it is still unclear if the sensitivity and specificity of colposcopy is significantly impacted when interpreted in the presence of the cytological result. Alongside cytology, colposcopy may also be influenced by the knowledge of other results or demographic parameters such as HPV status, age or prior histopathology [ 15 ].

Our study’s aim was to determine whether knowledge of cytology influences the colposcopic diagnosis of ≥ CIN2 lesions.

Case selection

A total of 80 cases were collected from a cohort of women already recruited to a study entitled ‘Use of a smartphone-based Artificial Intelligence classifier as an adjunct to colposcopy for identifying cervical pre-cancer and cancer: Proof of concept’ (study number 2020 − 00868) Fig.  1 . Patients were consented for this aforementioned study at the colposcopy clinic, gynecologic division of the University Hospital of Geneva, between June 2021 and June 2022, and all patients signed an informed consent. Inclusion criteria were women (i) aged 18–75, (ii) having a transformation zone type 1 or 2 and having (iii) a cytology result (iv), histopathology result and (v) colposcope images of sufficient quality for use. All cases addressed in our colposcopy setting that fulfilled inclusion criteria were considered in a chronological manner.

Flowchart of participants recruited for the online surveys

Case interpretation

The study relied on specialists in colposcopy analyzing two images for each of the 80 cases, one native and one after acetic acid application. The interpretation was recorded in the form of a JotForm web-based survey. The same images were analyzed twice on two separate sessions with at least a six-week interval between the two sessions. In one of the sessions the images without cytology were analysed, while in another session the images were complemented by the cytological status. Patients were initially referred in the context of primary cytology screening, and participating colposcopists had no underlying knowledge of HPV results or other information such as the age of the patient. Each participant was asked to categorize the image as “<CIN2” or “≥CIN 2”. Overall, the study involved 40 patients with < CIN2 histopathology results, and 40 patients with ≥ CIN2 histopathology, but participants were unaware of the ratio of positive and negative cases.

Colposcopists and interpretation rounds

A web-based Lime-survey link was distributed to multiple gynecology departments, inviting colposcopists to enroll and participate in the study. Initially, 71 participants enlisted, and ultimately, 38 of them completed both surveys and were divided into groups A and B. In keeping with the crossover design, group A received the survey without the cytology result, and then the survey with the cytology result six weeks later. Group B received the surveys in the opposite order. The participants were not aware of each other’s responses. A 6-week washout period was chosen as in the literature this has been demonstrated to confer a reduced rate of residuary recall, thus minimizing risks associated with intra-observer studies [ 16 ]. Empirically, multiple studies assessing whole-slide imaging (WSI) in pathology have used a minimum of 3 weeks as a washout period in their method [ 17 , 18 ].

Reference standard

The histological assessment of biopsy constituted the reference standard diagnosis. Patients with a ≥ CIN2 diagnosis based on biopsy underwent confirmation of the diagnosis through cone biopsy. Colposcopy-directed biopsies from all suspicious areas on examination had been undertaken for all women with histology that was revealed to be < CIN2. <CIN2 patients also had a colposcopy follow-up visit at 6 months with cytology +/-biopsy if lesions were seen. During the colposcopy examination, native images of the cervix and images after application of acetic acid were taken. All patient data was gathered retrospectively.

Cytological and histological interpretation

The cytological results were classified according to the Bethesda system 2015 [ 19 ]. Namely: negative for intraepithelial lesions or malignancy; atypical squamous cells of undetermined significance (ASC-US); atypical squamous cells of undetermined significance that cannot exclude high-grade squamous intraepithelial lesions (ASC-H); low-grade squamous intraepithelial lesions (LSIL); and high-grade squamous intraepithelial lesions (HSIL). Cervical biopsy specimens were interpreted using the WHO classification of Tumors 2020, which describes: low grade dysplasia (CIN1); and high-grade dysplasia including moderate (CIN2) and severe (CIN3) dysplasia [ 20 ].

Statistical method

Sample size calculation: We expected the sensitivity of colposcopy in isolation to be around 60% to detect high grade lesions. Few articles have evaluated the sensitivity of colposcopy alone, therefore an approximate average of values mentioned in available literature on colposcopy was used [ 8 , 9 , 10 , 11 , 12 , 13 ]. A simulation study was conducted to assess the smallest improvement in sensitivity with the cytology for which the statistical power was 90% with the above-mentioned sample size. The simulation study was generated under a two-sided risk alpha of 5% and crossed random effects (standard deviation of 0.40). Ultimately, with the planed sample size, the power is 90% or more to detect an absolute difference in sensitivity of 7% or more (60% for colposcopy in isolation versus 67% for colposcopy and cytology). The power calculation was similar for specificity.

Statistical analyses

We used mixed effects logistic regression models with two crossed random effects on the intercept: one random effect models the between-readers variability, and one random effect models the between-cases variability. We expect that the sensitivity varied across readers but also across cases as some cases may be more difficult to interpret than others. Statistical analyses were conducted with software R (R Core Team (2022)).

Patient characteristics

We included a total number of 80 patients who were referred to our colposcopy clinic. The principal reason for referral was an abnormal pap smear. The mean age of the women included at the time of colposcopy was 33.4 years (standard deviation 7.3 years). Twenty-two women (27.5%) were aged under 30, and 58 (72.5%) were aged over 30. For the 40 patients classified as “<CIN2”, 13 had a normal histology result, 12 had inflammation or metaplasia, and 15 had a CIN1 histology result. Amongst the 40 patients classified as “≥CIN2”, 9 had a CIN2 histology result, 28 had a CIN3 histology result, and 3 had AIS cytology result. Regarding the cytology, 18 had normal or inflammation results, 10 had ASC-US, 18 had LSIL, 15 had ASC-H, 15 had HSIL, 1 had an AGC result and 3 had an AIS result.

Colposcopist characteristics

The average experience in colposcopy of the participants was 10.9 years with a standard deviation of 9.5 years, and range of 1 to 35 years. Overall, 37,8% ( n  = 14) of colposcopists performed less than 50 colposcopies per year, and 63,2% ( n  = 24) performed more than 50 colposcopies per year. The washout period between the two rounds was 9.4 weeks on average.

Diagnostic performance: with cytology versus without cytology

The overall sensitivity of colposcopy without cytology was 51.1% [95%CI: 39.3 to 62.8], and 63.7% [95%CI: 52.1 to 73.9] with knowledge of cytology. The presence of cytology increased the chance of correctly identifying a patient as positive with an odds ratio (OR) of 1.68 [95%CI: 1.42 to 1.99]. The overall specificity of colposcopy without cytology was 63.5% [95%CI: 52.3 to 73.5], and 76.7% [95%CI: 67.2 to 84.0] with knowledge of cytology. Knowing the cytological result increased the chance of correctly identifying a patient as negative with an OR of 1.88 [95%CI: 1.57 to 2.24]. This is illustrated in Fig.  2 which represents the overall sensitivity and specificity of each colposcopist in both assessments of the cases.

Sensitivity and 1-specificity per rater, with (orange circles) and without (purple circles) cytology. The horizontal (respectively vertical) lines represent the 95% confidence intervals of the overall sensitivities and specificities with and without cytology

Participant’s experience associated with performance

For colposcopists who performed fewer than 50 colposcopies per year, the sensitivity was 55.9% [95%CI: 44.0 to 67.1] without cytology and 66.8% [95%CI: 55.5 to 76.5] with cytology. Having the cytological result available increased the likelihood of correctly identifying a patient as positive, with an OR of 1.59 [95%CI: 1.21 to 2.08]. In this subgroup, the specificity without cytology was 52.1% [95%CI: 41.1 to 62.6], which improved to 72.7% [95%CI: 63.2 to 80.5] with cytology. The presence of cytology increased the chance of correctly identifying a patient as negative, with an OR of 2.45 [95%CI: 1.86 to 3.22]. For colposcopists who performed more than 50 colposcopies per year, the sensitivity without cytology was 48.4% [95%CI: 33.6 to 63.6], and 62.7% [95%CI: 47.5 to 75.8] with cytology. In this group, the inclusion of cytology increased the likelihood of correctly identifying a patient as positive, with an OR of 1.79 [95%CI: 1.44 to 2.23]. The specificity without cytology was 71.9% [95%CI: 57.6 to 82.8], and 80.1% [95%CI: 68.0 to 88.4] with knowledge of cytology. Having the cytological result available increased the chance of correctly identifying a patient as negative, with an OR of 1.57 [95%CI: 1.24 to 1.99].

Association between colposcopic diagnosis and histology and cytology grades

In Table  1 , sensitivity and specificity were assessed in histological sub-groups. Specificity for detection of benign appearances of the cervix was increased by 12.8% when cytology was known. Specificity for detection of CIN1 was increased by 13.8% when cytology was known. For the detection of CIN2 lesions, sensitivity was increased by 17.7%, and for the detection of CIN3 lesions sensitivity was increased by 7.7%. Sensitivity and specificity were also analysed in cytological sub-groups, presented in Table  2 . The cytology information communicated to the colposcopists was binarised into ‘benign/borderline’ (normal, inflammation, ASC-US, LSIL) and ‘high-grade’ (ASC-H, HSIL, AGC, AIS). Sensitivity when cytology was reported as high-grade was 75.5% [95%CI: 63.3 to 84.6] compared to 36.9% [95%CI: 21.0 to 56.3] when cytology was reported benign. A high-grade cytology increased the chance of correctly identifying a patient as positive by an OR of 5.25 [95%CI: 2.11 to 13.12]. Specificity when cytology was reported as benign was 81.5% [95%CI: 72.7 to 88.0] compared to 50.5% [95%CI: 28.0 to 72.8] when cytology was reported as high-grade. A benign cytology increased the chance of correctly identifying a patient as negative by an OR of 4.33 [95%CI: 1.54 to 12.14].

Our study was designed to evaluate to what extent knowledge of cytology results affects interpretation of colposcopy. Our main finding supports that a known cytology influences the colposcopist’s diagnosis. Our study showed an improvement in sensitivity for ≥ CIN2 detection when cytology was available (51.1% vs. 63.7%). Information offered by cytology, particularly in the case of women with a high-grade result, may make the colposcopist more attentive to possible cervical abnormalities that would have otherwise been overlooked. This allows disease that would be missed to be correctly identified and may therefore reduce morbidity and mortality by facilitating disease treatment at an early stage. Equally, a benign or borderline cytology increased the chances of correctly identify a patient as negative. The improved specificity demonstrated in this study (63.5% vs. 76.6%) has important implications, namely reducing the chances of women having unnecessary cervical biopsy and prolonged follow-up which may be associated with discomfort, emotional distress, and extra financial costs.

Since the initial cytology had contributed to the diagnostic outcome (< CIN2 and ≥ CIN2), some level of inflation of the contrast between colposcopy interpretation with and without knowledge of cytology cannot be excluded.

The principal strength of our study is the large number of colposcopists who took part, increasing statistical power to elucidate the effect of cytology on the overall performance of colposcopy. Additionally, the use of a crossover design to account for individual variation and possible confounding factors strengthens the validity of the results. Finally, the use of a logistic regression mixed model to calculate sensitivities and specificities also enhances the statistical rigour. A relative weakness of this study is that static images were used which does not completely reflect the process of colposcopy. Static images do not convey the ability of the colposcopist to manipulate the cervix in order to visualise the entire squamocolumnar junction, to identify lesions that may be partially hidden inside the os. Another important difference is that when evaluating static images, the interpreter loses the potential to assess the dynamic acetowhite character of cervical tissue. The dynamic nature of acetowhite changes have been proposed as an important factor in determining the likelihood of ≥ CIN2 pathology [ 21 ]. Despite this, studies have shown that sensitivity of diagnosing ≥ CIN2 can be very high even when using solely static images [ 22 ].

Considering that cytology results are typically available at the point of colposcopy in high income settings worldwide, there is very little literature examining how cytology influences the colposcopist, and therefore to what extent it is beneficial to know the cytology result during colposcopy. One notable study showed, like us, that ≥ CIN2 lesions were more frequently missed when the referral PAP was negative [ 15 ]. However, after analysis, the investigators concluded that this was because these lesions were smaller at colposcopy, and that referral PAP result had no influence when this was taken into account. Our study did not evaluate the effect of the size of the lesion during colposcopy. This study added to existing literature suggesting that, as a tool in isolation, examination of cervical appearances after acetic acid application may have relatively poor sensitivity [ 23 ]. Our reported sensitivity of 51% for the detection of ≥ CIN2 implies that there are nearly equal numbers of true positives and false negatives, and therefore colposcopy alone likely represents an insufficient measure when determining whether there is significant pathology present.

An interesting result generated by this study was that healthcare professionals with greater recent experience (> 50 colposcopies per year) had a worse sensitivity for the detection of ≥ CIN2 (48.4% without cytology and 62.7% with cytology) than their colleagues undertaking fewer colposcopies (55.9% without cytology and 66.8% with cytology). They did, however, record a superior specificity. This is consistent with other literature in the field which reports that increasing colposcopic experience does not always equate to better sensitivity for the diagnosis of ≥ CIN2 at colposcopy [ 24 ]. The population size of this study could have impacted on the difference in sensitivities and may explain this result. A different interpretation of this could be that senior healthcare workers - who have greater overall experience - may do fewer colposcopies each year than their junior counterparts, as they have more responsibilities outside of the direct clinical environment.

In cervical cytology, also a rather subjective diagnostic activity, the cytologist’s pre-knowledge of HPV status has been reported to influence its accuracy [ 25 ]. With screening based on HPV test results becoming commonplace, the role of HPV testing in colposcopy, and its sensitivity and specificity in detecting ≥ CIN2 lesions should be investigated. In regards to our study, HPV data analysis was not conducted. However, the effect of knowing HPV-status on the overall performance of colposcopy, paired with knowledge of cytology or alone, will require investigation and could be conducted in a format similar to this study.

In conclusion, our findings support that knowledge of cytology results significantly influences the interpretation of colposcopy, leading to improved sensitivity for detecting ≥ CIN2 lesions. In cases of high-grade results, colposcopist performance is improved to a greater degree. This may be attributed to the association between cytology and colposcopy results.

Data availability

The datasets used and/or analysed during the current study are available from the corresponding author on reasonable request.

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Acknowledgements

We are grateful to Huma Farid, Dorothy Huang, Aisha Ali Issaka, Alice Fromont, Madhuri Chandra, Farida Akhoundova, Judith Norman, Francis Abihanna, Yveline Ansaldi, Athanasios Kakkos, Rosa Pinto Catarino, Akon Marcelline Ijang, Doan Tu Tran, Joëlle Desreux, Glenn Vergauwen, Françoise Nzoogou Yemelong, Esther Abatsong, Martine Jacot-Guillarmod, Cedric Tarek, Sarah Ongaro, Gino Venegas, Ha Hieu Thao and Yvonne Nokam for completing the surveys. Many thanks also to Christophe Combescure for his support with data analysis.

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Open access funding provided by University of Geneva

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Eva Lalande and Holly Clarke contributed equally to this work.

Authors and Affiliations

Gynecology Division, Department of Pediatrics, Gynecology and Obstetrics, University Hospitals of Geneva, Geneva, Switzerland

Eva Lalande, Holly Clarke, Manuela Undurraga, Pierre Vassilakos & Patrick Petignat

Faculty of Medicine, University of Geneva, Geneva, Switzerland

Eva Lalande

Department of Obstetrics and Gynecology, Hue University of Medicine and Pharmacy, Hué, Vietnam

Vu Quoc Huy Nguyen

Clinical Research Center, Faculty of Medicine, Geneva University Hospitals, University of Geneva, Geneva, Switzerland

Cyril Jaksic

Department of Obstetrics and Gynecology, University of Liege, Liege, Belgium

Frederic Goffin

Unit of Cancer Epidemiology, Belgian Cancer Centre, Sciensano, Brussels, Belgium

Department of Human Structure and Repair, Faculty of Medicine and Health Sciences, University Ghent, Ghent, Belgium

National Cancer Institute, Bethesda, MD, USA

Jose Jeronimo

Diagnostic Department, Division of clinical pathology, Geneva University Hospitals, Geneva, Switzerland

Jean-Christophe Tille & Essia Saiji

Geneva Foundation for Medical Education and Research, Geneva, Switzerland

Pierre Vassilakos

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Contributions

EL and HC should be credited as joint first author as they contributed equally to the work. Conception and design: PP, HC, EL, PV. Creation of Jotform web survey: HC, EL. Organization and/or dissemination of Jotform web surveys: EL, HN, FG, MA, JJ. Cytological and histological analysis: JC-T, ES. Statistical analysis: CJ. Interpretation of results, writing and review of final manuscript: EL, HC, MU, HN, CJ, FG, MA, JJ, JC-T, ES, PV, PP.

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Correspondence to Holly Clarke .

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Ethics approval and consent to participate.

Participants were retrospectively taken from a cohort of patients recruited to the study: ‘Use of a smartphone-based Artificial Intelligence classifier as an adjunct to colposcopy for identifying cervical pre-cancer and cancer: Proof of concept’ (study number 2020 − 00868). Informed consent was obtained from all the participants. The study was approved by the Ethics committee: Cantonal Commission for Research Ethics, Geneva.

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MA is supported by the Horizon 2020 Framework Programme for Research and Innovation of the European Commission, through the RISCC Network (Grant No. 847845) and all the other author’s declare no competing interest.

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Lalande, E., Clarke, H., Undurraga, M. et al. Knowledge of cytology results affects the performance of colposcopy: a crossover study. BMC Women's Health 24 , 189 (2024). https://doi.org/10.1186/s12905-024-03025-y

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Evaluating the association between duration of breastfeeding and fine motor development among children aged 20 to 24 months in Butajira, Ethiopia: a case-control study

• Rediate Shiferaw 1 ,
• Robel Yirgu 2 &
• Yalemwork Getnet 2  

BMC Pediatrics volume  24 , Article number:  216 ( 2024 ) Cite this article

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A Suitable environment and proper child nutrition are paramount to a child’s physical and mental development. Different environmental factors contribute to proper child development. Breast milk is an important source of nutrition during the early years of life and contains essential nutrients that are the building blocks for growth and development.

To assess the association between the duration of breastfeeding and fine motor development among children aged 20 to 24 months living in Butajira, southern Ethiopia.

Community-based case-control study design was employed among mother-child dyads of children aged 20 to 24 months in Butajira Southern Ethiopia. Children were screened for fine motor delay using the Denver II developmental screening and identified as cases and controls. A repeated visit was done to gather the rest of the information and 332 samples, 83 cases, and 249 controls were available and assessed. Epi-data version 4.4.2.1 software was used to prepare a data entry template, which was later exported to and analyzed using STATA version 14 statistical software. Finally, a Multivariable logistic regression model was used to adjust for confounders and estimate the independent effect of breastfeeding duration on fine motor development.

We didn’t find a significant association between the duration of breastfeeding from 21 to 24 months and fine motor delay compared to children who were breastfed less than 18 months[AOR: 0.86, 95% CI: (0.36, 2.05)]. Children who have mothers > 35 years of age were 78% less likely than children who had mothers younger than 25 years, Children who had mothers in secondary school and above were 77% less likely than mothers who didn’t have formal education, Females were 1.86 times more likely than males, and Children who scored 20–29 on the Home score were 51% less likely than Children who scored < 20 to have fine motor delay.

Duration of breastfeeding was not significantly associated with fine motor delay for children aged 20 to 24 months old. The age of the mother, the educational status of the mother, being female, and Home score were identified to have a significant association with fine motor delay. Improving the educational status and empowerment of women is essential. Further work should be done on avoiding gender differences starting from a young age and creating a conducive environment for child development is crucial.

Peer Review reports

The first 2 years can predict the quality of life a child can have. Appropriate connections are formed and wired in this window if this stage is passed then it is hard to rewire the brain connections [ 1 ]. Child development is a dynamic process that is a result of the interaction between biological and environmental factors [ 2 ]. Motor development is also seen as an indicator of global child development [ 3 ]. Motor development is the development of the child’s bones, muscles, and ability to move around and manipulate their environment [ 4 ]. Motor development is a critical factor in child behavior, being associated with the foundation of cognitive and social-emotional development[ 3 ]. Fine motor development is very important for the development of gross motor skills and is connected to how a child performs later on other cognitive tasks, reading and solving mathematical problems [ 5 ]. Fine motor skill is the ability to control movement through activities and coordination of the nervous system, fibril, and muscles such as fingers and hands [ 6 ]. Fine motor skills are important to do certain activities such as eating and handwriting. The United Nations has set sustainable developmental Goals to improve early child development by 2030. Goal 4 Target 4.2 supplies all children to get access to quality early childhood development so they are ready for primary education [ 7 ].

Child development is a dynamic process that is a result of the interaction between biological and environmental factors. Although infant development is influenced by several factors, The centrality of good nutrition cannot be ignored by providing the important building blocks for development [ 2 ]. Breastfeeding is the main source of important nutrients for children at this age. Breastfeeding has been identified by the World Health Organization (WHO) as an ideal source that contains important nutrients that can help for the optimal growth and development of children. WHO recommends continuing to breastfeed for up to 2 years with additional complementary foods [ 8 ]. Especially fatty acids Docosahexaenoic acid and Arachidonic acid in breast milk are important for brain growth and development and the formation of important synapses or connections in the brain. When a child is adequately nourished with important nutrients in the foundational period it creates a base for lifetime proper brain function [ 9 ]. Motor skills are also affected by factors such as caregiving practice, and stimulating environments [ 1 ]. Nutritional supplementation and psychosocial stimulation together result in greater improvements in child development than either intervention alone [ 9 ]. Determining the solo influence of breastfeeding on child development is difficult because child development is interrelated and associated with different environmental and biological factors. The complexity of child development makes it difficult to evaluate these effects [ 10 ]. The effects of environmental factors are pronounced in areas with limited access to the requirements for development [ 11 ]. Especially in developing countries, the problem can be worse due to limited resources in the environment that can aggravate the problem [ 12 ]. In resource-limited environments, limited resources such as poor stimulating environments and poor nutrition can limit the developmental potential of the children. Therefore, we need to study the effect of multiple environmental factors and nutrition on child development especially in a developing country context. To our best knowledge there are limited studies regarding developmental delays and also the practice of assessing child development in Ethiopia is low. Therefore, knowing the current status and assessing the impact is helpful for early intervention to prevent different adverse outcomes.

Materials and methods

Study design, area, and period.

Community-based Case-control study was conducted from March to May 2019 among children aged 20 to 24 months living in Butajira Health and demographic surveillance site located in Southern Nations and Nationalities (SNNP), Ethiopia. The area is located 135 km south of Addis Ababa and 50 km to the west of Zeway town in the Rift Valley, 8.2 o north latitude and 38.5 o east longitude. The climate varies from arid dry lowland areas at altitudes around 1,500 m (tropical climate) to cool mountainous areas up to 3,500 m above mean sea level (temperate climate). The livelihood of the residents is based on mixed farming. Khat (Catha edulis Forsk) and chili-peppers are the main cash crops, while maize and “false banana” or Ensete (Ensete ventricosun) are the main staples [ 13 ].

Source population

All children within the age group of 20 to 24 months living in Butajira HDSS are the Source population.

Study population

Children within the age group of 20 to 24 months living in Butajira HDSS have been identified as cases and controls based on the Denver developmental screening test.

Case definition

Cases were children who were identified as being suspect for fine motor delay and controls were children without fine motor delay.

Case (Suspect ): Two or more cautions (Item on which the age line fails or between the 75th and 90th percentile). This means 75% of the children can pass the test below the child’s age, and /or One or more delays (a child fails to perform an activity that fails completely to the left of the age line) using the Denver developmental screening test.

It is considered that a child fails to perform an item that 90% of children can perform at an earlier age.

Control (normal)

No delays and a maximum of one caution using the Denver developmental screening test.

Study variables

The outcome variable was Fine motor delay. The exposure variables were Nutritional factors (Breastfeeding duration, Dietary habits of the infant), Child characteristics (sex of the child, birth order), Socio-demographic variables (age of mother, occupation of mother, education status of mother, socioeconomic status), and Caregiving practice: (Home environment.)

Sample size calculation

The required sample size was calculated using EPI INFO 7 software using an unmatched case-control study using Proportion of Controls among those who breastfeed < 6 months P = 89.97% and Proportion of Cases among those who breastfeed < 6 months P = 76.4% and OR = 0.36 A study done in Taiwan [ 14 ].

At precision level of 5%, 95% confidence interval, and 80% power and using r = 3(ratio of cases to controls) and 10% for non-response compensation the sample size becomes 360 with 271 controls and 90 cases.

Sampling method

A survey (screening) was conducted from March to May 2019 in Butajira HDSS by obtaining a sampling frame from the Butajira HDSS. Participants were all children from 20 to 24 months living in the Butajira HDSS. The total population of children in Butajira HDSS from 20 to 24 months was 453.

After going into each Keble and household 376 children that were available were assessed using the Denver developmental screening test and identified as cases and controls. We found 85 cases and 291 controls. Then after identifying the households with cases and controls a repeated visit on the same household and on the same child was done to gather the rest of the information. After visiting the households 332 samples 332 samples 83 cases and 249 controls were available and were assessed using interviewer-administered questioners.

Operational definition

Caregiver (caretaker).

The people who look after infants and young children [ 15 ].

Breastfeeding less than 18 months

mothers while in the data collection period report that they have breastfed their babies less than 18 months.

Continue to breastfeed 18 to 20 months

mothers while in the data collection period report that they have breastfed their babies from 18 to 20 months and stopped.

Continue to breastfeed 21 to 24 months

mothers while in the data collection period report that they have breastfed their babies from 21 to 24 months.

• Fine motor development

The fine motor section of Denver II contains 33 items. Each test item on Denver II is presented on a chart by a horizontal bar partitioned into 25, 50, 75 and 90 percentile ages of passing the items. After calculating the exact age draw the age line after drawing the age line the child was asked to perform an activity to the left of the age line, this was done until the child was able to pass three or more consecutive items. Also, the child was tested for items above the age line until the child failed three or more consecutive items.

A child can pass-fail or refuse an item on which the age line fails.

By then identifying the child’s outcome using all the scores that the child has and finding the results will be carried out.

The scoring has 4 items.

“P” for pass – the child successfully performs the item or the caregiver reports (when appropriate) that the child does the item.

“F” for fail- the child does not successfully perform the item, or the caregiver reports (when appropriate) that the child does not do the item.

“N.O” for no opportunity- the child has not had the opportunity to perform the item, due to restrictions from the caregiver or other reasons. This score may only be used on “report” items.

“R” for refusal- the child refuses to attempt the item. Refusal can be minimized by telling the child what to do rather than asking. If given instruction in proper administration, the caregiver may administer the item. Report items cannot be scored as refusals.

no delays and a maximum of one caution.

Caution items are interpreted when a child fails or refuses an item on which the age line fails or between the 75th and 90th percentile. This means 75% of the children can pass the test below the child’s age. Delays are considered when a child fails to perform an activity that fails completely to the left of the age line. (Not on the item that the age line passes) It is considered that a child fails to perform an item that 90% of children can perform at an earlier age. This means 75% of the children can pass the test below the child’s age. When a child passes, fails, or refuses an item that is between the 25th and 75th percentile it is considered normal.

Delays suspect

Two or more cautions and /or one or more delays.

Caution items are interpreted when a child fails or refuses an item on which the age line fails or between the 75th and 90th percentile. This means 75% of the children can pass the test below the child’s age [ 16 ].

Adequate dietary diversity

Children who receive foods from 4 or more food groups using 24-hour recall [ 8 ].

Inadequate dietary diversity

Children who received foods less than four groups using 24-hour recall [ 8 ].

Household wealth index —is households’ living status and was constructed by using household asset data on housing conditions like the type of floor, the material of the wall, the material of roof; ownership of assets like radio, TV, telephone, vehicle; the presence of functional latrine, source of drinking water, ownership of domestic animals, ownership of farmland and amount of grain harvested in the last production year among others. After running principal components analysis (PCA) in STATA, the households’ wealth index was grouped into quintiles (lowest quintile, second quintile, middle quintile, fourth quintile, and highest quintile).

Data collection instrument and procedure

Development was assessed by the Denver developmental screening test which is designed to test the development of the child. The data collection started by Screening for a suspect for fine motor development. The fine motor was assessed using the Denver II developmental screening test. The tool contains different materials that help to examine the development of the child and a test form that contains all the developmental domains in sections. The Denver II tool was adapted in Jimma into a developing country context and was validated in Butajira Ethiopia [ 17 , 18 ]. The Denver II was assessed by a BSC nurse trained and certified for assessing children using the Denver developmental screening test.

The test was done in a natural and comfortable environment where the child could play with minimal disturbance in the presence of the caretaker. The test was started by informing the mother that the child is not expected to pass all the items.

The test contains a total of 125 items in four developmental domains: personal-social, fine motor, language, and gross motor. The fine motor section of Denver II contains 33 items. Each test item on Denver II is presented on a chart by a horizontal bar partitioned into 25, 50, 75 and 90 percentile ages of passing the items.

Draw the exact age without rounding off days, weeks, or months. Age scales are placed at the top and bottom of the page. Spaces between the age marks represent 1 month until 24 months. After carefully identifying the child’s age draw the age line using the age scales draw an age line from the top to the bottom of the form. After drawing the age line the child was asked to perform an activity to the left of the age line, this was done until the child was able to pass three or more consecutive items. Also, the child was tested for items above the age line until the child failed three or more consecutive items.

For each item, there are 25th, 50th, 75th and 90th percentile.

The age line, pass through the following tasks.

16. Dump coffee bean demonstrated.

Show the child 2 or 3 times how to dump the coffee bean out of the bottle. Then ask the child to get it out. (Do not use the word “dump.”)

Pass if the child dumps the coffee bean out of the bottle or rakes the coffee bean close to the opening and then dumps it out. Do not pass if the child removes the coffee bean with a finger.

17. Tower of cubes – 2, 4, 6, 8.

With the child sitting high enough at the table so that elbows are level with table top and hands are on the table, place the blocks on the table in front of the child. Encourage the child to stack them by demonstration and words. It may be helpful to hand the blocks to the child, one at a time. Three trials may be given.

Pass Tower of 2 cubes if the child puts one block on top of another so that it does not fall when he/she removes his/her hand.

Pass Tower of 4, 6, 8 cubes , depending upon the greatest number of blocks the child stacks in three trials.

A pass of 4, 6, or 8 cubes also passes the lower tower items (for example, passing Tower of 6 cubes also passes Tower of 2 and 4 cubes ).

“N.O” for no opportunity- the child has not had the opportunity to perform the item, due to restrictions from the caregiver or other reasons. This score may only be used on “report” items. “R” for refusal- the child refuses to attempt the item. Refusal can be minimized by telling the child what to do rather than asking. If given instruction in proper administration, the caregiver may administer the item. Report items cannot be scored as refusals.

By then identifying the child’s outcome using all the scores that the child has and finding the results were carried out.

no delays (the child successfully performs the action) and a maximum of one caution (between the 75th or 90th percentile).

two or more cautions and/or one or more delays (the child fails to perform an activity that fails completely to the left of the age line.)

refusal scores on one or more items completely to the left of the age line or on more than one item intersected by the age line in the 75-90% area.

Praise the child even for items that are failed. This will build the confidence of the child to attempt more difficult items.

Data on socio-demographic, breastfeeding, and nutritional histories were collected using interviewer-administered questions.

Total Breastfeeding duration was assessed from a study that assessed breastfeeding duration since birth [ 19 ]. It was taken by asking the mother to recall the total duration she breastfed her child. Whether she is still breastfeeding or to recall the time she stopped breastfeeding her child.

Complimentary food was assessed using WHO dietary diversity [ 8 ]. Dietary diversity was collected using dietary diversity scores adapted from the WHO standardized questionnaire for infant and young child feeding (IYCF). Mothers or caregivers were asked to recall all the food items that the child consumed during the past 24 h [ 8 ]. The home environment was assessed using the Home inventory used in different studies [ 20 ]. The Home score was assessed by interview-administered questionnaires. It was done by giving the mother a picture book and the mother will show the picture book to the child. Observation will be made on the interaction and the response the mother has towards her child. The interview was conducted in a free and friendly environment. The observation was made on the maternal and child interaction and maternal responses towards the child while asking other questions from the Home inventory.

The training was given to data collectors and supervisors regarding the objective and method of data collection and discussions were made for unclear questions in the questionnaire.

Data processing and analysis

Data were checked manually for completeness and entered into Epi-data version 4.2.2.1 statistical software and exported into STATA version 14 for data cleaning and analysis. Frequencies and summary statistics (median, interquartile range, percentage, and range) were used to describe the study population in relation to relevant variables.

Nutrition-related variables such as duration of breastfeeding were categorized based on the duration of breastfeeding in months and were grouped as breastfed less than 18 months, 18 to 20, and 21 to 24 months. Dietary diversity was also assessed using a Minimum dietary diversity score comparing children who had consumed four or more food groups and children who scored less than four groups using 24-hour recall. Socioeconomic status was analyzed based on the wealth index by using Principal component analysis (PCA). Childcare practices, maternal-child interaction were checked using the Home score.

Binary logistic regression was used to check for the association between the dependent, fine motor delay, and independent variables. Variables with P- value < 0.2 and which had clinical importance or subject matter were included in the multiple logistic regression. In the multiple logistic regressions, Variables with 95% CI of AOR which did not include 1 were considered to have significant association with the outcome variables. The goodness of fit test indicated (P = 0.0518) that the model was good enough to fit the data well.

Ethical consideration

Before data collection ethical clearance was obtained from Addis Ababa University School of Public Health Institutional Review Board (AAU-IRB). Written Informed consent was obtained from parents (legal guardians) before participating in the study. All study participants were informed about the purpose of the study, their right to deny participation, anonymity, and confidentiality of the information. All methods were carried out in accordance with relevant guidelines and regulations. The Denver II developmental screening test used in this study to measure the developmental milestone was assessed by a well-trained and certified data collector to ensure the safety of the children. It was conducted in a free and friendly environment. It was explained to the parents that the scale determines the child’s current developmental status and that it’s not an IQ test and the child is not expected to pass all the tests administered. The beneficence of the participants was assured by providing education to the participants about the benefits of breastfeeding, growth, and development. For Children identified with developmental delay, further education was given on methods of improving the motor skills of the Children. The confidentiality of the information of the participants was not disclosed.

Socio-demographic characteristics of the study participants

Mothers in the age group from 25 to 29 years were 35(42.17%) in the cases while 101(40.56%) were in the controls. The median age of the mothers was 28, IQR (25 33%). About 48(57.83%) of mothers in the cases and 93(37.65%) of mothers in the controls didn’t have any formal education. About 58(69.88%) of the cases and 185(74.60%) of mothers from the controls were Housewives. About 39(46.99%) fathers in the cases and 67(26.91%) in the controls didn’t have any formal education. About 23(27.71%) of the cases and 52(20.88%) in the controls were from the lowest quintile. About 69(83.13%) of the cases and 179(71.89%) of the controls were Rural residents (Table  1 ).

Child-related characteristics

The study included 168 male and 164 female children from the age group of 20–24 months. About 36(43.37%) males were cases while 132(53.01%) were in the controls (Table  2 ).

Delivery and nutritional characteristics of the study participants

Health facility delivery among the cases was 67(80.72%) and 213(85.54%) among the controls. Breastfeeding at least once was 81(97.59%) among the cases and 248(99.60%) among the controls. About 49(59.04%) mothers in the cases and 139(55.82%) in the controls reported that they are currently breastfeeding.

About 66(79.52%) children in the cases and 177(71.08%) children in the controls continued to be breastfed from 21 to 24 months. There was no significant variation among cases and controls by the duration of breastfeeding 95% CI (p = 0.234) (Table  3 ).

Dietary practices and nutritional characteristics of the children

About 46(55.42%) of children in the cases and 179(71.89%) in the controls started solid or semi-solid food within 6 to 8 months. There was a difference among cases and controls on children at the time of starting solids and semisolid foods (Table  4 ).

Caregiving practice

About 41(49.40%) of children in the cases and 172(69.08%) in the controls had a score between 20 and 29 on the Home score. The Home score had a minimum score of 13 and a maximum score of 32 (Table  5 ).

Association of different characteristics of children with suspect of fine motor delay

In the binary logistic regression variables with p-value < 0.2 or factors that had clinical importance were identified (Table  6 ).

After adjusting for these variables age of the mother, the educational status of the mother, the sex of the child, and the Home score were identified to have a significant association with fine motor delay.

We didn’t find a significant association between duration of breastfeeding and fine motor delay for children who were breastfed from 18 to 20 months [AOR: 0.45, 95% CI: (0.13, 1.56)] and for children who were breastfed from 21 to 24 months [AOR: 0.86, 95% CI: (0.36, 2.05)] compared to children who were breastfed less than 18 months. Children who have mothers > 35 years of age were 78% less likely to have fine motor delay than mothers who were < 25 years old [AOR: 0.22, 95% CI: (0.05, 0.87)]. Children who had mothers in primary school were 66% less likely [AOR: 0.34, 95% CI: (0.14, 0.81)] and children who had mothers in secondary school and above were 77% less likely [AOR 0. 23, 95% CI: (0.06, 0.80)] to have fine motor delay than mothers who didn’t have any formal education. Females were 1.86 times more likely to have fine motor delay than males [AOR: 1.86, 95% CI: (1.05, 3.28)]. Children who scored 20–29 on the Home score were 51% less likely to have fine motor delay than Children who scored < 20 [AOR: 0.49, 95% CI: (0.27, 0.88)] (Table  6 ).

Child development is an important aspect of human life. Development can be affected by different factors. Environmental factors and nutritional factors together play a significant role in child development. Nutritional factors have a great role in development but due to the adverse environmental and social factors, the outcome could be influenced by different factors, especially in developing countries [ 1 ].

Breastfeeding is known to have a significant effect on child growth and development [ 11 ] but in our study, We didn’t find a significant association between the duration of breastfeeding and fine motor delay for children who were breastfed from 18 to 20 and for children who were breastfed from 21 to 24 months compared to children who were breastfed less than 18 months.

Our findings are consistent with some studies that didn’t find a significant association between duration of breastfeeding and fine motor development [ 21 , 22 , 23 ]. All the studies acknowledged that breastfeeding is important for development but they suggested that other factors also have a role in influencing fine motor development. Similar to our study, A study in Singapore didn’t find a significant association between breastfeeding and fine motor development at 24 months [ 21 ]. Another study done in rural Brazil didn’t find a significant association between breastfeeding and fine motor development at 12 months and suggested home stimulation, maternal education, and income were influencing the outcome [ 22 ].

The study in Singapore suggested they have used specific research tools and have controlled for a large number of potential confounders and they didn’t find any relationship between breastfeeding on fine motor development [ 21 ]. The study in Brazil investigates the association between breastfeeding and mental and motor development, controlling for comprehensive measures of the child’s socioeconomic maternal, and environmental background, and nutritional status. They didn’t find a significant association between breastfeeding and motor development. They explained that the reason most studies have found an association between breastfeeding and development is that the studies have been done in relatively affluent populations where, in general, mothers who succeed in breastfeeding have higher socio-economic status, better educated with higher educational attainment. While In their study mothers who were breastfeeding longer had lower socioeconomic status, poorer education, and provided less stimulating home environments. They explained the reason that most studies found the association was due to incomplete adjustment for covariates, differences in methodological robustness, and types of tests used are likely to be contributory, which will result in an apparent breastfeeding benefit. To prevent this bias they controlled for different covariates. They suggested that no subgroup is differentially protected by breastfeeding, but rather that all groups benefit. The benefit of breastfeeding was an important factor that benefited all the comparison groups, while it has a beneficial effect, breastfeeding didn’t have a protective effect on fine motor development. The difference in the outcome was appreciated by other potential determinants. They found home stimulation and family income to be more important factors [ 22 ].

This is similar to our study finding that mothers who were breastfeeding longer had lower socio-economic status and poorer education. We also have found other environmental factors to be significantly associated. Similar to these studies environmental factors were playing a significant role in fine motor development.

All the studies acknowledged that breastfeeding is important for development but they suggested that other factors were influencing fine motor delay and we need to take into consideration other factors that could also affect or contribute to child development.

A systematic review also suggested that development is influenced by different environmental and psychological factors. Different factors need to be put into consideration that can affect the developmental potential of the children. Their analysis reveals that there are studies that have shown an apparent decrease in effect after multivariate analysis. Given that tight control of confounders resulted in a greater likelihood of the disappearance of the breastfeeding effect. Studies completed in middle-income and low-income countries were nearly twice as likely to find no association compared with studies set in developed countries. The fact that this relationship is less apparent in developing countries suggests that much of the observed relationship may be due to parental social advantage, confounding the choice to breastfeed [ 23 ].

In conclusion, the systematic review suggests that much of the reported effect of breastfeeding on child developmental abilities is due to maternal and socioeconomic effects. They suggested additional, future studies in this field are needed to rigorously control for all important confounders [ 23 ]. Development is not the solo effect of breastfeeding alone but a combination of different factors working together.

All these studies have used different developmental screening tools so the comparison should be done cautiously.

Contrary to our study A study in Malawi among children who breastfeed from 9 to 10 months found a small but significant protective effect on fine motor development at 12 to 18 months [ 24 ]. Studies in Western countries, a study done in Taiwan and Greece assessed the effect of duration of breastfeeding more than 6 months and fine motor assessed at 18 months. They found that any increase in the duration of breastfeeding was associated in decreasing in the odds of fine delay which persisted after controlling for different factors [ 25 , 26 ]. The Taiwan study has shown that mothers who breastfeed longer were older, had a university education, and were from a better socioeconomic class and suggested that the positive result could be due to the presence of these factors [ 25 ]. These factors were different in our setting, the majority of the mothers in this study who breastfeed for longer durations were less educated. Studies have shown that mothers who are more educated create a more favorable and stimulating environment and when breastfeeding is added to these factors there could be better results that can be helpful for child development [ 27 , 28 ]. This might be one of the reasons why we couldn’t find a significant association.

We have also found the age of the mother to have a significant association with the development of the child. We have found older mothers had more favorable outcomes than young mothers. Similar findings have suggested that older mothers tend to create a more favorable environment for child development and would also breastfeed for longer durations [ 29 – 31 ].

Also, we have found the education level of the mother to be significantly associated with fine motor delay. Children who had mothers in primary and secondary school were less likely to have fine motor delay than mothers who didn’t have formal education. Studies have shown that a mother’s education is important because as the educational level of the mother increases the level of stimuli the mother gives to her child also increases [ 27 ]. In addition to that, as the education level of the parents increases the socioeconomic status also could increase and will create a more favorable environment for the children [ 32 ].

Another factor that we found significant was the sex of the child. We have found females have greater odds of being affected by fine motor delay than males. Contrary to our study different studies have suggested females have a better score on fine motor and boys have a higher risk of having developmental delay [ 33 , 34 ]. While we cannot give a general conclusion other factors in the environment could affect the development of females. A study in India has shown that Girls are breastfed for shorter durations than boys due to the gender preferences of the mothers. Mothers will start early weaning for girls than boys to have another pregnancy and not to delay another pregnancy [ 35 ]. The gender preferences of the mother could affect the duration of breastfeeding and the care the child will have [ 36 ]. This gender preference could lead to a developmental delay in the female population.

We have found the Home score to have a significant association with fine motor development. Similarly, studies found the Home environment to have a significant association with fine motor development [ 37 , 38 ]. Motor development can be regulated critically by the home environment and maternal and child interaction [ 39 ]. The role of the mother or the caregiver has a protective role even for children growing up in limited environments such as low socioeconomic status, low levels of education, chronic illness, conflict, and mental health problems of caregivers. Mothers’ sensitivity is important because it creates a conducive environment for the development of the child [ 40 ]. A study done in Iran did not find a significant positive association between home motor affordances and motor development in their sample. They suggested that this could be due to the tool that they used was not sensitive enough to detect differences [ 3 ]. Home environment is a very important factor for childhood development a study has shown in nutrition-related interventions certain amount of stimulation from the environment was necessary and nutritional intervention alone was insufficient to bring brain development [ 40 ].

The Strengths of this study are the study was a community-based case-control study which is helpful to asses multiple exposure or risk factors. We have also used new cases that were identified at the time of collection which could prevent misclassification bias. We have used tools that are validated in our setting which can measure the case of interest in a better way.

The following limitation needs to be taken into account when interpreting the results. Most of the mothers in our study group had the practice of long-term breastfeeding durations and conducting the study where different information or different study groups are available would help to further strengthen the study finding. Our study was conducted in a rural setting but including the figure of urban mothers would further enrich the information that can be found. Even though birth weight is an important factor for development we didn’t have information on the birth weight of the children.

Conclusions

This study still supports that breastfeeding is important for child development. However, in our study, we didn’t find a significant association between the duration of breastfeeding from 18 to 20 months and for duration of breastfeeding from 21 to 24 months compared to children who were breastfed less than 18 months on fine motor development. Children from older mothers were less likely to be affected than young mothers. Children who had mothers in primary and secondary school were less likely to have fine motor delay than mothers who didn’t have formal education. Females have higher odds of being suspect of fine motor delay than males. Children who had better maternal care practices or Home scores were less likely to be affected than Children who had lower maternal care practices or lower Home scores.

Based on our findings we forward the following recommendations: Health care providers should be the first-line source of information to provide appropriate information to the mothers and the community during delivery or during any visit the mother makes to the health facility. They should educate the mothers and the community about the importance of child feeding and childcare and creating a conducive environment for child development. Older mothers tend to create more conducive environments for child development. Delaying early pregnancies is helpful to have physically and psychologically mature mothers. Since mothers are the primary caretakers improving maternal education and empowerment to improve developmental outcomes is helpful for child development. Therefore policymakers should work on improving the educational status and empowerment of women and work on avoiding gender differences starting from a young age. Assessment of Developmental delay in children should also be done routinely by Health care providers to catch delays during the early years and to have early interventions. Further studies should be done in a different setup to appreciate the difference and the effect of other environmental factors. Further follow-up studies should be done to prevent recall bias in a better way. Thus, overall, child development can be influenced by different factors in the environment, and having a holistic approach is mandatory to tackle the problem.

Data Availability

The datasets used and/or analyzed during the current study are available from the corresponding author upon reasonable request.

Abbreviations

Infant and Young Child Feeding

Arachidonic acid

Docosahexaenoic acid

Ethiopian Demographic and Health Survey

Health and Demographic Surveillance site

Nationalities and Peoples Regional State

World Health Organization

Adjusted Odds Ratio

Confidence Interval

Crude Odds Ratio

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Acknowledgements

We acknowledge the data collectors, study participants, and all those who were involved in the study. We would like to extend our gratitude to Professor Frances Abound for his comment, support, and advice. We would also like to thank Dr. Teklu Gemechu for his help and guidance and Miss. Mashresa Harisgo for her help and dedication during the data collection.

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Shiferaw, R., Yirgu, R. & Getnet, Y. Evaluating the association between duration of breastfeeding and fine motor development among children aged 20 to 24 months in Butajira, Ethiopia: a case-control study. BMC Pediatr 24 , 216 (2024). https://doi.org/10.1186/s12887-023-04391-6

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Use of progestogens and the risk of intracranial meningioma: national case-control study

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• Use of progestogens and the risk of intracranial meningioma: national case-control study - March 28, 2024
• Noémie Roland , general practitioner and epidemiologist 1 ,
• Anke Neumann , senior statistician 1 ,
• Léa Hoisnard , epidemiologist 2 ,
• Lise Duranteau , endocrinologist and gynaecologist 3 ,
• Sébastien Froelich , professor of neurosurgery 4 ,
• Mahmoud Zureik , professor of epidemiology and head of department 1 5 ,
• Alain Weill , senior epidemiologist and deputy director 1
• 1 EPI-PHARE Scientific Interest Group, French National Agency for Medicines and Health Products Safety, French National Health Insurance, Saint-Denis, France
• 2 EpiDermE Epidemiology in Dermatology and Evaluation of Therapeutics, EA7379, Paris Est Créteil University UPEC, Créteil, France
• 3 Department of Medical Gynaecology, Bicêtre Hospital, Assistance Publique-Hôpitaux de Paris, Paris Saclay University, 94270, Le Kremlin-Bicêtre, France
• 4 Department of Neurosurgery, Lariboisière University Hospital, Paris-Cité University, Assistance Publique-Hôpitaux de Paris, Paris, France
• 5 University Versailles St-Quentin-en-Yvelines, Montigny le Bretonneux, France
• Correspondence to: N Roland noemie.roland{at}assurance-maladie.fr (@NoemieRoland11 @EPIPHARE on X)
• Accepted 22 February 2024

Objective To assess the risk of intracranial meningioma associated with the use of selected progestogens.

Design National case-control study.

Setting French National Health Data System (ie, Système National des Données de Santé ).

Participants Of 108 366 women overall, 18 061 women living in France who had intracranial surgery for meningioma between 1 January 2009 and 31 December 2018 (restricted inclusion periods for intrauterine systems) were deemed to be in the case group. Each case was matched to five controls for year of birth and area of residence (90 305 controls).

Main outcome measures Selected progestogens were used: progesterone, hydroxyprogesterone, dydrogesterone, medrogestone, medroxyprogesterone acetate, promegestone, dienogest, and intrauterine levonorgestrel. For each progestogen, use was defined by at least one dispensation within the year before the index date (within three years for 13.5 mg levonorgestrel intrauterine systems and five years for 52 mg). Conditional logistic regression was used to calculate odds ratio for each progestogen meningioma association.

Results Mean age was 57.6 years (standard deviation 12.8). Analyses showed excess risk of meningioma with use of medrogestone (42 exposed cases/18 061 cases (0.2%) v 79 exposed controls/90 305 controls (0.1%), odds ratio 3.49 (95% confidence interval 2.38 to 5.10)), medroxyprogesterone acetate (injectable, 9/18 061 (0.05%) v 11/90 305 (0.01%), 5.55 (2.27 to 13.56)), and promegestone (83/18 061 (0.5%) v 225/90 305 (0.2 %), 2.39 (1.85 to 3.09)). This excess risk was driven by prolonged use (≥one year). Results showed no excess risk of intracranial meningioma for progesterone, dydrogesterone, or levonorgestrel intrauterine systems. No conclusions could be drawn concerning dienogest or hydroxyprogesterone because of the small number of individuals who received these drugs. A highly increased risk of meningioma was observed for cyproterone acetate (891/18 061 (4.9%) v 256/90 305 (0.3%), odds ratio 19.21 (95% confidence interval 16.61 to 22.22)), nomegestrol acetate (925/18 061 (5.1%) v 1121/90 305 (1.2%), 4.93 (4.50 to 5.41)), and chlormadinone acetate (628/18 061 (3.5%) v 946/90 305 (1.0%), 3.87 (3.48 to 4.30)), which were used as positive controls for use.

Conclusions Prolonged use of medrogestone, medroxyprogesterone acetate, and promegestone was found to increase the risk of intracranial meningioma. The increased risk associated with the use of injectable medroxyprogesterone acetate, a widely used contraceptive, and the safety of levonorgestrel intrauterine systems are important new findings.

Introduction

Meningiomas account for 40% of primary tumours of the central nervous system. 1 2 The incidence of meningioma in the United States is 9.5 per 100 000 person years. 2 Meningiomas are mostly slow growing, histologically benign tumours but can nevertheless compress adjacent brain tissue and thus patients may require surgical decompression. 3 The incidence of meningiomas increases with age, rising sharply after the age of 65 years. Conversely, meningiomas are rare before the age of 35. Other recognised risk factors for meningioma are being female, intracranial exposure to ionising radiation, neurofibromatosis type 2 2 , and, as shown only recently, prolonged use (≥one year) to high doses of three potent progestogens: cyproterone acetate, 4 5 chlormadinone acetate, 4 and nomegestrol acetate. 4

The link between female sexual hormones, in particular progesterone, and intracranial meningioma is biologically plausible. 6 Progesterone receptors are present in more than 60% of meningiomas 7 and the volume of these tumours has been observed to increase during pregnancy and to decrease post partum. 8 However, previous pregnancy does not appear to be an unequivocal risk factor for meningioma. 9 Studies have also shown a link, albeit a weak one, between breast cancer and meningiomas. 10

No significant association between exogenous female hormones and risk of meningioma has been shown to date for hormonal contraceptives (either combined or progestogen only pills). 11 12 Additionally, data for hormone replacement treatment for menopause are contradictory. Several studies have shown a slight excess risk of meningioma associated with the use of hormone replacement treatment for menopause, 11 13 whereas others have reported no deleterious effects of these molecules. 14 By contrast, the excess risk of meningioma observed with the use of high doses of cyproterone acetate among cis women, men, and trans women has been shown to be very high 5 15 16 and somewhat lower, but still substantial, for chlormadinone acetate and nomegestrol acetate. 4 Discontinuation of each of these three progestogens generally leads to a reduction in meningioma volume, 17 18 which avoids the need for surgery and its associated risk of complications for most patients.

Whether progestogens other than these three oral progestogens at high doses have a similar effect depending on their route of administration is still unknown. Our study aimed to assess the real-life risk of intracranial meningioma associated with the use of progestogens from an extensive list (progesterone, hydroxyprogesterone, dydrogesterone, medrogestone, medroxyprogesterone acetate, promegestone, dienogest, and levonorgestrel intrauterine systems) with different routes of administration (oral, percutaneous, intravaginal, intramuscular, and intrauterine). Although some of the progestogens studied are used in France (promegestone) or in only a few countries (medrogestone), others are widely used worldwide in various doses and for various indications (progesterone, levonorgestrel, hydroxyprogesterone, medroxyprogesterone) (supplementary table A). Certain progestogens may also be risky at some doses when used over a long period of time, but not at lower doses or when used for a short period of time. Our secondary objectives were to describe the characteristics of the women who were in the cases group (age, grade, and anatomical location of the meningiomas) and to approximate the number of surgically treated meningiomas attributable to the use of the concerned progestogens.

Study design and data source

This observational population based study used data derived from the French national health data system ( Système National des Données de Santé (SNDS)). Given the analysis of multiple exposure situations (different exposure definitions and lookback periods) in our study, we opted for a case-control design rather than a cohort study, thus including long term users of the considered medications. 19

The SNDS database contains information on all health spending reimbursements for over 99% of the population residing in France and is linked to the French hospital discharge database. 20 SNDS is currently one of the largest healthcare databases in the world and is widely used in pharmacoepidemiological studies. 4 5 21 22 23 24

Definition of cases and selection of controls

The eligible cases in this study were women residing in France of all ages who underwent surgery for intracranial meningioma between 1 January 2009, and 31 December 2018. For each case, the start date of the corresponding admission to hospital marked the index date. Women with a pregnancy beginning in the two years before the index date were excluded from the study (pregnancies were defined as those that had resulted in childbirth or medical termination of the pregnancy after 22 weeks of amenorrhoea).

Surgery for intracranial meningioma was defined by the simultaneous combination of the following diagnoses and procedures recorded for the same hospital stay: a meningeal tumour (codes D32, D42, or C70 according to the 10 th revision of the International Classification of Diseases (ICD-10)) coded as the main diagnosis of the admission to hospital and an intracranial surgery act (supplementary table B). These codes have already been used in our previous studies. 4 5

Five women in the control group were randomly matched to each woman in the case group for the year of birth and area of residence (“ département ”, a French geographical subdivision, n=101). Matching was based on the risk set sampling approach. 25 The traceability of the controls in the SNDS was ensured by selecting only women who had had at least one service reimbursed in the calendar year before the index date and the two to three calendar years preceding the index date. This criterion was also applied to the selection of cases.

For analyses relating to intrauterine systems, subsets of these cases and the matched controls were considered to ensure sufficiently long lookback periods. For the hormonal intrauterine systems containing 52 mg levonorgestrel and copper intrauterine devices, the cases and controls from the years 2011 to 2018 were retained. For the hormonal intrauterine systems containing 13.5 mg levonorgestrel, the inclusion period was restricted to 2017 to 2018 (start of commercialisation in France in 2013).

Definition of exposure

Exposure to the progestogen of interest was defined according to WHO’s anatomical, therapeutic, and chemical (ATC) classification. The list included progesterone (oral and intravaginal: 100, 200 mg (ATC code G03DA04); percutaneous: 25 mg per bar (G03DA04)), dydrogesterone (10 mg, or in association with oestrogen: 5 or 10 mg (G03DB01, G03FA14, G03FB08)), hydroxyprogesterone (500 mg (G03DA03)), medrogestone (5 mg (G03DB03)), promegestone (0.125, 0.25, or 0.5 mg (G03DB07)), medroxyprogesterone acetate (injectable contraceptive, 150 mg/3 mL (G03AC06, L02AB02 partially)), dienogest (in association with oestrogen, 2 mg (G03FA15)), levonorgestrel (52 mg intrauterine systems (G02BA03); 13.5 mg intrauterine systems (G02BA03)) (supplementary tables C and D). As drospirenone, which is a spironolactone derivative, is not reimbursed in France, we were unable to access data concerning its use. We therefore chose to study the use of spironolactone (25, 50, and 75 mg), even though its indications may be very different. The code used to identify spironolactone was C03DA01. The indications for these various progestogens in France are available in table 1 .

Main indications (marked as x), in France, for the progestogens under study

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For oral, intravaginal, percutaneous, or intramuscular progestogens, exposure was defined as at least one dispensation of the progestogen of interest in the 365 days before the index date. For intrauterine progestogens, a dispensation was sought within three years before the index date for levonorgestrel 13.5 mg (as the duration of efficacy of this intrauterine system is three years before any change or withdrawal of the device) and within five years before the index date for levonorgestrel 52 mg intrauterine systems (duration of contraceptive efficacy of five to six years according to current recommendations during the study period).

Exposure was described by three modes for each progestogen as follows: 1) exposure to the progestogen concerned, 2) exposure during the three years preceding the index date to at least one of the three high dose progestogens known to increase the risk of meningioma (ie, chlormadinone acetate, nomegestrol acetate, and cyproterone acetate), and 3) absence of exposure to the progestogen considered or to the three high dose progestogens (the reference for the analyses).

Definition of covariates

The description of sociodemographic and medical characteristics included age, area of residence, existence of neurofibromatosis type 2 (ICD-10 code Q85.1), and, for cases only, the year of surgery, anatomical site (anterior, middle, or posterior base of the skull, convexity, falx and tentorium, others; supplementary table C), and grade of severity of the meningioma (according to WHO’s classification 1 : benign, malignant, or atypical, supplementary table E).

Adjuvant radiotherapy was also sought from three months before the index date to six months after (supplementary table F). Additionally, all causes mortality at two and five years after the index date was assessed in cases, as well as the use of antiepileptic drugs in the third year after the index date (supplementary table G).

Statistical analysis

Logistic regression models conditioned on matched pairs were used to estimate odds ratios and their 95% confidence intervals (CIs) for the association between exposure to the progestogens of interest and meningioma (odds ratio of exposure relative to non-exposure). Additionally, the effect of history of neurofibromatosis type 2 on the risk of meningioma was estimated, as well as the effect of chlormadinone acetate, nomegestrol acetate, and cyproterone acetate exposure, all serving as positive controls for exposure to validate our results. In parallel, exposure to a copper intrauterine device was used as a negative control for exposure (codes in supplementary table H).

The risk of meningioma associated with progestogen use was also estimated for each oral, percutaneous, intravaginal, and intramuscular progestogen according to the duration of use: short term (at least one dispensation in the year before the index date but no dispensation in the second year before the index date) and prolonged use (at least one dispensation in the year before the index date and at least one dispensation in the second year before the index date).

The population attributable fraction was approximated from the odds ratio obtained for each progestogen. The formula used was as follows: population attributable fraction=p c (1-1/odds ratio), where p c is the prevalence of the use of the progestogen concerned (isolated exposure) among the cases. 26 Lastly, sensitivity analyses were performed. Analyses were stratified for age (<35 years, 35-44 years, 45-54 years, 55-64 years, and ≥65 years) and for the location and grade of severity of the tumours whenever a positive association was found between exposure to the considered progestogen and meningioma surgery.

Data were analysed using SAS software version 9.4 (SAS Institute Inc). A P value of less than 0.05 was considered statistically significant (two tailed tests).

The present study was authorised by decree 2016–1871 on 26 December 2016. 27 As an authorised permanent user of the SNDS, the author’s team was exempt from approval from the institutional review board. This work was declared, before implementation, on the register of studies of the EPI-PHARE Scientific Interest Group with register reference T-2023-01-437.

Patient and public involvement

The list of progestogens of interest (supplementary table B) was drawn up in consultation with a temporary scientific advisory board comprised of representatives of the French National Agency for Medicines and Health Products Safety, patient organisations, and healthcare professionals (neurosurgery, endocrinology, gynaecology, and general medicine).

Description of cases and controls

In total, 108 366 women were included in the study during the inclusion period of 2009 to 2018, consisting of 18 061 women in the case group were matched with 90 305 in the control group ( fig 1 ).

Flowchart for the analyses of oral, percutaneous, intravaginal, and intramuscular progestogens. Index date is defined as the date of hospital admission

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Among them, 15 162 cases and 75 810 controls were retained for the analyses of intrauterine systems and copper intrauterine devices using 52 mg of levonorgestrel (restricted inclusion period: 2011 to 2018) (supplementary figure A) and 4048 cases and their 20 240 controls for the analysis of intrauterine systems of 13.5 mg of levonorgestrel (2017-18) (supplementary figure B). Descriptions of cases and controls for the analyses of intrauterine devices are detailed in supplementary I and J.

The mean age of all women was 57.6 years (standard deviation 12.8 years). The most highly represented age groups were 45-54 (26.7%), 55-64 (26.4%), and 65-74 (21.5%) years ( table 2 ).

Description of the cases and controls (overall inclusion period 2009-18). Data are number of individuals (percentage), unless otherwise specified

The number of cases steadily increased from 1329 in 2009 to 2069 in 2018. Meningiomas requiring surgery were most frequently located at the base of the skull (a total of 10 046/18 061 cases (55.6%); anterior skull base: 3979/18 061 (22.0%), middle: 3911/18 061 (21.7%), posterior: 2156/18 061 (11.9%)), followed by the convexity (6468/18 061 (35.8%)). Concerning tumour grade, most meningioma cases were benign (16 662/18 061, 92.3%) and 1047/18 061 (5.8%) were classified as atypical and 352/18 061 (1.9%) as malignant. Among cases, 28.8% (5202/18 061) of women used antiepileptic drugs three years after the index date of surgery. Mortality was also higher among cases than controls: 502 cases/18 061 (2.8%) died within two years ( v 1.2% of controls) and 951/18 061 (5.3%) within five years ( v 3.4% of controls). Mortality was higher for the cases with malignant tumours, 12.5% of whom died within two years and 20.7% within five.

The comparison of the cases and controls in the subsets used to analyse hormonal intrauterine systems is included the supplementary data (supplementary tables I and J).

Progestogens (others than intrauterine)

Exposure among cases.

Among the 18 061 women admitted to hospital for meningioma surgery between 2009 and 2018, 329 (1.8%) had used oral or intravaginal progesterone, 90 (0.5%) percutaneous progesterone, zero hydroxyprogesterone, 156 (0.9%) dydrogesterone, 42 (0.2%) medrogestone, nine (<0.1%) medroxyprogesterone acetate, 83 (0.5%) promegestone, three (<0.1%) dienogest, and 264 (1.5%) spironolactone ( table 3 , supplementary figure C). These numbers excluded 2999 women who had been exposed to cyproterone acetate, nomegestrol acetate, or chlormadinone acetate, or a combination, within the previous three years (among these 2999 women, 68 had also been exposed to oral progesterone, 47 to percutaneous progesterone, 0 to hydroxyprogesterone, 43 to dydrogesterone, 10 to medrogestone, 0 to medroxyprogesterone acetate, 17 to promegestone, 1 to dienogest, and 56 to spironolactone). The median cumulative doses of progestogens for cases and exposed controls are shown in supplementary table K.

Associations between use of oral, percutaneous, intravaginal, and intramuscular progestogen and risk of surgically treated intracranial meningioma. Data are number of individuals (percentage), unless otherwise specified

Effect on meningioma risk

No significant association with an increased risk of intracranial meningioma surgery was noted with exposure to oral or intravaginal progesterone (odds ratio of 0.88 (95% CI 0.78 to 0.99)) or percutaneous progesterone (1.11 (0.89 to 1.40)), dydrogesterone (0.96 (0.81 to 1.14)), or spironolactone (0.95 (0.84 to 1.09)) ( table 3 , supplementary figure C). Exposure to dienogest was rare, with only 14 women who were exposed (3/18 061 among cases and 11/90 305 among controls) and, consequently, the estimated odds ratio had a very large confidence interval (1.48 (0.41 to 5.35)). Additionally, we could not assess the odds ratio concerning hydroxyprogesterone because no exposed cases were found ( fig 2 ).

Associations between various progestogens and risk of intracranial meningioma requiring surgery (case control design, 2009-18). Odds ratio in logarithmic scale. CI=confidential interval; LNG=levonorgestrel; SNDS=French National Health Data System ( Système National des Données de Santé ). *LNG had different denominators due to restricted inclusion periods (10/4048 cases, 48/20 240 controls; 566/15 162 cases, 3888/75 810 controls)

By contrast, an excess risk of meningioma was associated with the use of medrogestone (3.49 (2.38 to 5.10)), medroxyprogesterone acetate (5.55 (2.27 to 13.56)), and promegestone (2.39 (1.85 to 3.09)). As expected, an excess risk of meningioma for women with positive control exposure neurofibromatosis type 2 (18.93 (10.50 to 34.11)), as well as those exposed to chlormadinone acetate (3.87 (3.48 to 4.30)), nomegestrol acetate (4.93 (4.50 to 5.41)), and cyproterone acetate (19.21 (16.61 to 22.22)) was also noted ( fig 2 ).

The duration of exposure to medrogestone, medroxyprogesterone acetate, promegestone, chlormadinone, nomegestrol, and cyproterone acetate for exposed cases and controls is presented in supplementary table L. The results show that three quarters of the women in the cases group who had been exposed for more than a year had been exposed for more than three years. As for medrogestone, medroxyprogesterone acetate, and promegestone, the excess risk associated with prolonged use was higher than that measured for short term and prolonged exposure combined. Specifically, prolonged use of promegestone had an odds ratio of 2.74 (2.04 to 3.67) (versus 2.39 for all durations of exposure) and short term use an odds ratio of 1.62 (0.95 to 2.76). For prolonged use of medrogestone, the odds ratio was 4.08 (2.72 to 6.10) (versus 3.49 for all durations of exposure combined), and for medroxyprogesterone acetate, the odds ratio was 5.62 (2.19 to 14.42). No significant association was reported for either short or prolonged periods of use for any of the other progestogens studied.

Meningiomas before age 45 years were rare in cases of exposure to medrogestone (n=3/42), medroxyprogesterone acetate (n=3/9), or promegestone (n=10/83), and only one (medroxyprogesterone) was observed before the age of 35.

Concerning medrogestone, the most frequent locations of meningiomas in exposed cases were the base of the skull (n=21/42; 13 in the middle) and the convexity (n=19/42) (supplementary tables M, N and O). The excess risk of meningioma for the middle of the base of the skull was particularly high (odds ratio 8.30 (95% CI 3.70 to 18.63)). Additionally, the estimated excess risk among women aged 45-54 years was slightly higher than that in the main analysis (4.53 (2.73 to 7.53) v 3.49 (2.38 to 5.10)).

In women in the cases group who were exposed to promegestone, meningiomas were preferentially located at the front of the base of the skull (n=25/83), the convexity (n=25/83), and the middle of the base of the skull (n=22/83). The excess risk of meningioma linked to promegestone use was slightly higher in the group who were older than 65 years (odds ratio 3.21 (95% CI 1.39 to 7.43)) and for meningiomas located at the front or middle of the base of the skull (3.15 (1.95 to 5.10) and 3.03 (1.82 to 5.02), respectively).

We found no malignant grade tumours among cases exposed to medrogestone, medroxyprogesterone acetate, or promegestone (for information, the same analyses were carried out for chlormadinone acetate, nomegestrol acetate, and cyproterone acetate in supplementary table N).

Levonorgestrel intrauterine systems

In total, 566/15 162 users of hormonal levonorgestrel 52 mg were among the cases with meningioma surgery between 2011 and 2018 (3.7%) ( table 3 ). For the intrauterine systems with 13.5 mg of levonorgestrel, 10 of 4048 users were reported among the cases from 2017 and 2018 (0.2% of all cases). Again, women who had been exposed to cyproterone acetate, nomegestrol acetate, or cyproterone acetate, or a combination, within the previous three years were not counted (among them, 95 were exposed to the intrauterine systems of 52 mg levonorgestrel and three to intrauterine systems of 13.5 mg levonorgestrel).

No excess risk of meningioma was reported with the use of hormonal intrauterine systems containing 52 mg (odds ratio 0.94 (95% CI 0.86 to 1.04)) or 13.5 mg (1.39 (0.70 to 2.77)) of levonorgestrel ( fig 2).

Exposure to copper intrauterine devices, used as a negative control for exposure in this study, had an odds ratio of 1.13 (1.01 to 1.25).

Attributable cases

The population attributable fractions, which are relative to the observed overall number of surgically treated intracranial meningiomas, were 0.17% for exposure to medrogestone, 0.04% for medroxyprogesterone acetate, and 0.27% for promegestone. For comparison, they were calculated as 2.58% for chlormadinone acetate, 4.08% for nomegestrol acetate, and 4.68% for cyproterone acetate. The numbers for the attributable cases are presented in supplementary figure D.

Principal findings

Although the risk of meningioma was already known for three progestogens, this study is the first to assess the risk associated with progestogens that are much more widely used for multiple indications, such as contraception.

This population based study shows an association between the prolonged use of medrogestone (5 mg), medroxyprogesterone acetate injection (150 mg), and promegestone (0.125, 0.25, 0.5 mg) and a risk of intracranial meningioma requiring surgery. No such risk was reported for less than one year of use of these progestogens. However, we found no excess risk of meningioma with the use of progesterone (25, 100, 200 mg; oral, intravaginal, percutaneous), dydrogesterone (10 mg, combined with oestrogen: 5, 10 mg), or spironolactone (25, 50, 75 mg), neither with short term nor prolonged use, and with the use of levonorgestrel intrauterine systems (13.5, 52 mg). A small number of women were exposed to dienogest (2 mg, in association with oestrogen) and hydroxyprogesterone (500 mg), therefore we cannot draw any conclusions concerning the association between use of these progestogens and the risk of meningioma.

No malignant meningiomas were noted for women exposed to medrogestone, medroxyprogesterone acetate, or promegestone. Moreover, the number of cases of surgically treated intracranial meningioma attributable to use of these progestogens was much lower than the number of cases attributable to the intake of chlormadinone acetate, nomegestrol acetate, and, in particular, cyproterone acetate. This finding is explained by both a lower excess risk of meningioma (for medrogestone and promegestone) and lower rates of use in France (particularly low for medroxyprogesterone acetate, with less than 5000 women exposed each quarter during the inclusion period of the study of 2009-18).

Specific considerations on meningiomas

Meningioma is a predominantly benign tumour. Between 2011 and 2015, 80.5% of the meningiomas diagnosed in the United States were grade 1, 17.7% grade 2, and 1.7% grade 3. 1 Even in the absence of malignancy, meningiomas can cause potentially disabling symptoms. In such cases, first line treatment is surgery, even for the oldest patients, entailing a risk of complications and morbidity. 28 29

Age is an important factor both for the indication of progestogens and for considering intracranial surgery. In our study, the mean age of women in the cases group was 57.6 years. Medrogestone, medroxyprogesterone acetate, and promegestone can be used both by women of childbearing age and by premenopausal and postmenopausal women. In our study, only one user of these progestogens who had undergone meningioma surgery was younger than 35 years (medroxyprogesterone).

Postoperative complications are not uncommon for meningioma surgery. Depending on the exact location of meningiomas, the surgical risk varies but surgery may have severe neurological consequences due to the immediate proximity of highly functional cortical area and critical neurovascular structures. Cognitive function tends to improve after surgery for meningioma, 30 31 but several studies have suggested a potential for postoperative anxiety and depression and a high intake of antidepressants and sedatives in the medium term, 32 33 although other studies have reported conflicting findings for depression. 34 Seizures are also a possible short term complication of surgery, 35 leading to a need to take antiepileptic drugs in the years following the operation. In our study, almost three in 10 women (28.8% of cases) were using antiepileptic drugs three years after the operation, which was consistent with previously published findings. 36 Additionally, results showed that progestin related meningiomas tend to occur more frequently at the skull base and that surgery for lesions in this location is much more challenging. The recent evidence supporting stabilisation or regression of meningiomas after stopping chlormadinone acetate, nomegestrol acetate, and cyproterone acetate has reduced the surgical indications for these patients, thus avoiding potential complications. 17 18 A recent report showed that although the tissue portion of the meningioma most often regresses in size, the hyperostosis associated with meningiomas further increases, which may require surgical intervention, not for oncological purposes but only for decompression of the structures nerves and relief of symptoms. 37

Use of the studied progestogens in France and worldwide

Medrogestone is indicated in France for the treatment of menstrual cycle disorders and luteal insufficiency (eg, dysmenorrhea, functional menorrhagia or fibroid-related menorrhagia, premenstrual syndrome, and irregular cycles), endometriosis, mastodynia, and hormone replacement therapy for menopause. In the United States, medrogestone has never been approved by the US Food and Drug Administration. Outside of France, this molecule is also used in Germany, in combination with oestrogen (0.3 mg/5 mg, 0.6 mg/2 mg, 0.6 mg/5 mg). 38 The use of medrogestone increased significantly in France in 2019, notably as a result of postponements in the prescription of chlormadinone acetate, nomegestrol acetate, and cyproterone acetate, following the French and European recommendations to reduce the risk of meningioma attributable to these progestogens in 2018 and 2019. 39 40 As therapeutic alternatives have not shown an increased risk of meningioma, switching from products that notoriously increase this risk to medrogestone should be reconsidered.

Worldwide, in 2019, 3.9% of women of childbearing age were using injectable contraception (medroxyprogesterone), that is, 74 million users, but figures vary widely between world regions (from 1.8% in high income countries to 8.7% in low income countries). 41 This method of contraception is the most widely used in Indonesia (13 million women), 42 Ethiopia (4.6 million women), and South Africa (3.6 million women). 41 In the USA, medroxyprogesterone acetate is used in more than 2 million prescriptions in 2020 and more than one of five sexually active American women report having used injected medroxyprogesterone acetate (150 mg/3 mL) in their lifetime. 43 44 Injectable contraceptives are much less widely used in Europe (3.1% of women of childbearing age in the UK and 0.2% in France 41 ). Our results support preliminary findings from studies of meningioma cases exposed to chronic use of medroxyprogesterone acetate or cases of high dose administration. 45 46 47 48 49 In particular, our results show similarities with those of a retrospective review of 25 patients diagnosed with meningioma who had a history of chronic medroxyprogesterone acetate use and were treated at the University of Pittsburgh Medical Center between 2014 and 2021 concerning the characteristics of cases exposed to medroxyprogesterone acetate (women (mean age of 46 years) with meningiomas commonly located at the base of the skull). 48 In addition, medroxyprogesterone acetate used as an injected contraceptive is known to be prescribed to specific populations, especially people with mental illnesses. 50 The protection of these vulnerable populations from additional drug risks is particularly important. Depot medroxyprogesterone acetate (150 mg) is registered for use as a form of birth control in more than 100 countries worldwide. 41 In countries that have high numbers of people using medroxyprogesterone acetate, the number of meningiomas attributable to this progestogen may be potentially high. Furthermore, medroxyprogesterone (non-acetate) is also used orally, at lower doses, in some countries other than France (notably in the US), for which no data exists on a risk of meningioma so far.

Promegestone was only available in France (not marketed in any other country) and was withdrawn from the market in 2020. This drug was indicated for the relief of premenopausal symptoms and hormone replacement therapy for menopause. With the discontinuation of its marketing, some users could have switched to medrogestone in 2020, a molecule also implicated in the risk of meningioma in our results. Clinicians therefore must remain vigilant because meningioma risk could last beyond market withdrawal and a potential switch to another progestogen.

The FDA defines a therapeutic class as “all products (…) assumed to be closely related in chemical structure, pharmacology, therapeutic activity, and adverse reactions”. 51 52 Various subtypes of progestogens exist depending on the molecule from which the progestogen is derived (ie, progesterone, testosterone, and spironolactone) (supplementary table B). 53 Their chemical structures and pharmacological properties differ according to this classification, which explains why no class effect is reported for certain benefits and risks associated with their use (eg, breast cancer and cardiovascular risk). 54 55 56 57 Progestogens have distinct affinities for different target organ steroid receptors, which may vary even within a subclass, determining their activity.

Our study suggests that 17-OH-hydroprogesterone and 19-norprogesterone derivatives, both progesterone derivatives, have a class effect on meningioma risk. Four of five progestogens belonging to the 17-OH-hydroprogesterone group have shown an increase in the risk of meningioma (supplementary table R). However, the fact that we found different sizes of risk appears to be more a question of duration and cumulative dose than that of belonging to a progestogen class. We could not draw any conclusions about hydroxyprogesterone (due to a lack of power), the fifth progestogen in the subclass, but its main indication (assisted reproductive technology) corresponded to fewer women exposed and very short exposure (approximately 15 days), which could explain why this drug differs from the others. Finally, to date, at the doses considered in the study, no excess risk of meningioma associated with testosterone derivatives has been shown. However, the risk of meningioma associated with the use of these derivatives at other doses and in other regimens needs to be investigated.

Strengths and limitations

To our knowledge, this study of meningioma risk is the first to expand the list of progestogens of interest beyond chlormadinone acetate, nomegestrol acetate, and cyproterone acetate, detailing the risk associated with each progestogen, with different modes of administration. This study was conducted on a national scale for women of all ages for both the cases and their controls. The SNDS database allowed the use of exhaustive real-world data from over a period of 12 years (2006-18; postoperative information was searched even up to 2022), thus preventing recall bias.

The exclusion of women with a pregnancy beginning in the two years preceding the index date ensured that estimates of the risks associated with progestogen use were reliable. Pregnancy is a unique state, affecting exposure to progestogens (of endogenous or exogenous origin), the likelihood of a meningioma appearing or increasing in volume, 9 58 59 and the likelihood of admission to hospital for surgery (possibly with a lower surgery rate, depending on the symptoms, maternal and foetal health, and tumour characteristics). 59

Another potentially important confounding factor, use of chlormadinone acetate, nomegestrol acetate, or cyproterone acetate, was considered in the analyses by modelling exposure to each progestogen of interest with a separate mode of prior or simultaneous exposure to these drugs. Furthermore, the results obtained for the negative and positive control exposure, including exposure to chlormadinone acetate, nomegestrol acetate, and cyproterone acetate, support the appropriateness of the method chosen for this study.

However, this study also had several limitations. As a result of the scarcity of historical data in the SNDS (which began in 2006, and did not have information for some reimbursement schemes during the first few years), we have only three years of lookback period for the oldest meningioma cases (2009-06), and 12 years for the most recent. The SNDS does not provide information on non-reimbursed drugs, which obliged us to study dienogest in association with oestrogen rather than dienogest alone. Further studies will therefore be necessary. Similarly, we were unable to study other progestogens, such as norgestimate, gestodene, and norethisterone, contained in non-reimbursed products (supplementary table B). Conversely, desogestrel is available and reimbursed in France. Its dosage is much lower and, thus, we chose not to study the drug. Further study to assess a dose-response association in the event of prolonged use would be needed. The progestogen implants (etonogestrel) are also rarely used in France, and concern young women, for whom the risk of meningioma is probably very low. 60 61 We have also not studied the risk associated with the use of hormonal intrauterine systems containing 19.5 mg levonorgestrel because its marketing in France was too recent (2018). However, any excess risk associated with the use of the levonorgestrel 19.5 mg intrauterine systems is unlikely because this dose of levonorgestrel is lower than that of the levonorgestrel 52 mg intrauterine systems, for which we observed no risk.

Moreover, the SNDS does not provide information on all the clinical details and medical indications for which progestogens are prescribed. These missing data mean assessing the risk-benefit ratio of prescriptions is not possible, which could be favourable in the absence of an effective alternative, for example, in the case of relief of endometriosis symptoms. We only have some indirect idea of the indication, depending on the age of the user, and the molecule used (progesterone is not indicated for endometriosis, for example, and dydrogesterone is indicated for endometriosis but is rarely used in this indication). Nevertheless, a risk-benefit assessment was not the aim of our study and will require further studies using other sources of data for product efficacy. Furthermore, no evidence suggests that an increase in meningioma risk depends on the medical indication for the progestogen prescription. In the study of Weill and colleagues in 2021, the excess risk of meningioma associated with the use of cyproterone acetate was equivalent for men and women, who, nevertheless, use cyproterone acetate for radically different indications. 5

In this study, only admission to hospital for meningioma surgery was used as the outcome of interest. However, meningiomas can also be treated with radiotherapy (in rare cases) or simply monitored. Therefore, this study is highly likely to have underestimated the prevalence of meningiomas attributable to the use of progestogens by limiting itself solely to symptomatic tumours that require surgery. However, using admission to hospital for surgery as the outcome ensured diagnostic specificity and thus limited classification bias. The SNDS does not specify the histological characteristics of the meningiomas or the isolated or multiple nature of the tumour, both of which are important criteria in determining severity and the choice of appropriate treatment. Nevertheless, for the cases selected for this study, WHO’s severity grade of the meningioma is coded via the main diagnosis, which is entered according to the ICD-10 code at the end of the hospital stay after a reading of the pathology report. As such, we had indirect information about the histology of the tumours.

Our study has several confounding factors. The two main risk factors identified for meningioma, in addition to age (which was considered in this study) and being female (only women were included in this study), are genetic predisposition, attributed, in particular, to hereditary mutations of the neurofibromatosis type 2 gene, and medical or environmental exposure to high doses of ionising radiation. Radiotherapy for brain cancer (especially during childhood) is probably the most important of the possible medical reasons for intracranial radiation exposure, but only a small proportion of individuals in the general population had cerebral radiotherapy or a malignant brain tumour during childhood.

The progestogens investigated in our study that did not result in an increase to risk of meningioma should be considered under the specific conditions of use in France. These results may not be generalised to the use of these progestogens for other indications, increased doses, or increased duration of use. Similarly, the use of one or more of these progestogens might increase the meningioma risk, when the patient had previously received another type of progestogen.

Prescribers need to be aware of previous progestogen use of any kind and any changes in type of progestogen prescribed that may have occurred and should consider the cumulative dose of progestogens for each patient. The list of progestogens we studied is wide ranging, covering a variety of indications (summarised in table 1 ) for women of all ages (childbearing, premenopausal, and menopausal). As in hormone replacement therapy for menopause, progestogens can be easily substituted for each other, and thus progesterone appears to be the safest alternative. For endometriosis, however, therapeutic alternatives are much more limited, and each indication must be discussed on the basis of the personal benefit to risk ratio. If a high risk progestogen is to be continued, clinical and radiological monitoring and compliance with recommendations are essential.

Finally, we did not estimate the effect of concomitant oestrogen use on the risk of meningioma. In a previous report, having a concomitant oestrogen prescription was weakly but significantly associated with meningioma risk, with an age adjusted hazard ratio of 1.6 (95% CI 1.1 to 2.4) for use of cyproterone acetate. In our previous studies, the simultaneous prescription of oestrogen with chlormadinone acetate (hazard ratio 0.8 (0.5 to 1.3)) and nomegestrol acetate (1.0 (0.7 to 1.7)) was not significantly associated with a risk of meningioma. 28 62 In addition, in these two studies, which were cohort studies of women initiating treatment with the progestogen considered, the proportion of women with a simultaneous prescription of oestrogen at the initiation of progestogen treatment was relatively low (6.8%, and 5.0%, respectively per study).

Conclusions

Prolonged use of medrogestone, medroxyprogesterone acetate, and promegestone was found to be associated with an increased risk of meningioma. Future studies should further clarify the association between the duration of use and risk for the progestogens studied, and extend the discussion of meningioma risk to dienogest and hydroxyprogesterone. Finally, no excess risk of meningioma was associated with the use of progesterone, dydrogesterone, or spironolactone, or the hormonal intrauterine systems used worldwide, regardless of the dose of levonorgestrel they contained.

Further studies are also needed to assess the meningioma risk with the use of medroxyprogesterone acetate, which, in this study, was considered at a dose of 150 mg and corresponded to a second line injectable contraceptive that is rarely used in France. Studies from countries with a broader use of this product, which, furthermore, is often administered to vulnerable populations, are urgently needed to gain a better understanding of its dose-response association.

What is already known on this topic

Known risk factors for intracranial meningioma include age, female sex, neurofibromatosis type 2, exposure to ionising radiation, and use of high dose progestogens: nomegestrol, chlormadinone, and cyproterone acetate

Many other progestogens are widely used for multiple indications for which the risk of meningioma associated with their use has not been estimated individually

What this study adds

Prolonged use of medrogestone (5 mg, oral), medroxyprogesterone acetate (150 mg, injectable), and promegestone (0.125/0.5 mg, oral) was found to be associated with an excess risk of intracranial meningioma

In countries for which the use of medroxyprogesterone acetate for birth control is frequent (74 million users worldwide), the number of attributable meningiomas may be potentially high

The results for oral, intravaginal, and percutaneous progesterone, as well as dydrogesterone and levonorgestrel intrauterine systems, are reassuring, supporting the absence of excess meningioma risk

Ethics statements

Ethical approval.

The present study was authorised by decree 2016–1871 on December 26, 2016. 27 As a permanent user of the SNDS, the author’s team was exempt from approval from the institutional review board. This work was declared, before implementation, on the register of studies of the EPI-PHARE Scientific Interest Group requiring use of the SNDS (register reference: EP-0437).

Data availability statement

Under the terms of the SNDS data use agreement, the complete study data cannot be shared with other investigators ( https://www.snds.gouv.fr ). However, the authors try to share publication related data as much as possible: algorithms and other additional information are provided in the supplemental data; aggregated data can be supplied upon request by contacting the corresponding author at noemie.roland{at}assurance-maladie.fr .

Acknowledgments

We thank Bérangère Baricault and Pauline Dayani for their help in responding to the reviewers, and Sylvie Fontanel and Emmanuelle Mignaton for reviewing the manuscript. We also thank Alex Edelman and Associates for proofreading the English version.

Contributors: AW had the idea for the study. NR, AN, LH, and AW conceived and planned the study. NR and AN drafted the manuscript. AN and LH performed the data management. AN, LH, and NR performed the statistical analyses. AW and MZ ensured project and study management. All authors approved the final manuscript. The corresponding author (NR) attests that all listed authors meet the authorship criteria and that no others meeting the criteria have been omitted. AW is the guarantor.

Funding: This research was funded by the French National Health Insurance Fund (Cnam) and the French National Agency for Medicines and Health Products Safety (ANSM) via the Health Product Epidemiology Scientific Interest Group (ANSM-Cnam EPI-PHARE Scientific Interest Group). NR, AN, and AW are employees of the French National Health Insurance Fund, MZ is an employee of the French National Agency for Medicines and Health Products Safety. The funders had no role in considering the study design or in the collection, analysis, interpretation of data, writing of the report, or decision to submit the article for publication.

Competing interests: All authors have completed the ICMJE uniform disclosure form at https://www.icmje.org/disclosure-of-interest/ and declare: support from French National Health Insurance Fund (Cnam) and the Health Product Epidemiology Scientific Interest Group (ANSM-Cnam EPI-PHARE Scientific Interest Group) for the submitted work, no financial relationships with any organizations that might have an interest in the submitted work in the previous three years, and no other relationships or activities that could appear to have influenced the submitted work.

Transparency: The lead author affirms that the manuscript is an honest, accurate, and transparent account of the study being reported, that no important aspects of the study have been omitted, and that any discrepancies from the study as originally planned (and, if relevant, registered) have been explained.

Dissemination to participants and related patient and public communities: The results were presented for the first time on 12 June 2023, at a meeting organised by the French National Agency for Medicines and Health Products Safety to invited patient association representatives, gynaecologists, endocrinologists, neurosurgeons, and general practitioners. The report on this study (in French) was than published on 26 June 2023, on the EPI-PHARE, ANSM (Agence nationale de sécurité du médicament et des produits de santé), and Cnam (Caisse nationale de l’assurance maladie) websites and was sent to the European Medicine Agency.

Provenance and peer review: Not commissioned; externally peer reviewed.

This is an Open Access article distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/ .

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‘Opening Night’ Review: A Stylish Movie Becomes a Sludgy Travesty

Ivo van Hove’s stage adaptation of the 1977 John Cassavetes film, with music by Rufus Wainwright, turns a taut character study into a corny melodrama.

By Houman Barekat

The critic Houman Barekat saw “Opening Night” in London.

In a London auditorium, a work of art is being desecrated. “Opening Night,” John Cassavetes’s understatedly stylish 1977 movie about an actress struggling with midlife ennui, has been reimagined as a musical by the Belgian director Ivo van Hove, and the result is a travesty.

Its antiheroine, the Broadway superstar Myrtle Gordon (Sheridan Smith), has landed the lead role in a play about a middle-aged woman. But she isn’t feeling it: Though she is about 40, she insists she can’t relate. She stumbles through rehearsals, clashing with the director, Manny (Hadley Fraser), and the playwright, Sarah (Nicola Hughes), then goes rogue during previews, taking liberties with the script.

To compound matters, the actress develops a neurotic fixation on Nancy (Shira Haas), a 17-year-old fan killed in a car crash moments after getting Myrtle’s autograph. Convinced that Nancy is a cipher for her own lost youth, Myrtle intermittently hallucinates the dead girl’s ghost, and even converses with it. Myrtle is unraveling, but the show — somehow — must go on.

It’s a compelling story line, filled with dramatic possibilities, but “Opening Night,” which runs at the Gielgud Theater through July 27, is scuppered by a series of poor choices. Smith is miscast as Myrtle, for a start: Her onstage bearing exudes a homely approachability rather than high-strung poise or inscrutable aloofness.

Benjamin Walker is wooden as Maurice, Myrtle’s stage co-star and ex-partner, who Cassavetes himself played charmingly in the film. The estranged couple’s brittle onstage chemistry is an essential ingredient in the drama; here, they seem like actual strangers. Haas’s spectral Nancy is a disconcertingly cutesy symbol of youthful feminine vitality, a sprite-like figure who scurries around the stage in a short skirt, knee-high socks and platform boots — suggesting not so much a young woman as a pubescent child.

The songs, by Rufus Wainwright, are algorithmically bland. Several address aging, including the unsubtly titled “A Change of Life” (about menopause) and “Makes One Wonder,” a duet in which Myrtle and Sarah realize that, as women of a certain age, they may have more in common than they’d like to admit.

Others are about showbiz: “Magic” is an upbeat cabaret-style number about the wonder of the stage; “Moths to a Flame” is a somber, sentimental paean to the indefatigability of thespians everywhere. There is a brief foray into rock opera during an excruciating scene in which Myrtle, having figured out she must banish Nancy’s specter to get herself back on track, scuffles with the girl-child amid flashing strobe lights and 1980s-style power riffs. It’s so schlocky that it almost feels like a sendup.

Jan Versweyveld’s set is a theater within a theater. The rehearsal space occupies the foreground, and a row of vanity mirrors at the rear of the stage represents the backstage area. As in van Hove’s 2019 adaptation of “All About Eve ” — another story about the emotional travails of an aging actress — camera operators stalk its perimeter, transmitting close-up, real-time footage of the actors onto a big screen above the stage.

The idea is to ramp up the psychodrama by bringing us up close and personal, but there isn’t much intensity to intensify. The multiple angles add little to the experience. (The occasional bird’s-eye view is particularly unnecessary, unless you happen to have an interest in the topography of hairlines.) A screen caption at the start of the show informs us that a documentary film crew is recording the company’s rehearsals — a plot device that is supposed to make this camerawork feel less like a gratuitous gimmick, but so flimsily transparent that it has the opposite effect.

There are one or two good moments, including a tense rehearsal scene in which Myrtle objects to having to endure an onstage slap. She says it’s humiliating, but Manny insists it’s artistically necessary. Smith renders the standoff with a bleak comic pathos: At one point she even slaps herself to forestall the blow. (For van Hove, who is known for pushing his performers to the limit, this material is close to home.) Near the end, as the characters make their final preparations for opening night, the big screen cuts to recorded footage of theatergoers passing through the Gielgud foyer a couple of hours earlier — a clever touch that spurred a ripple of amused murmurs from the audience. But these are slim pickings.

As an artist yearning to take back control of her narrative, Myrtle should resonate at a time when questions of agency — for women and minorities, among others — are on many people’s minds. But van Hove’s corny treatment trivializes her suffering. Cassavetes’s movie had an elliptical quality that drew viewers in through the strength of its narrative artifice and the power of the actors’ performances; here, the story never comes to life, and the themes are labored. Van Hove has transformed a taut, subtly observed character study into a sludgy melodrama.

Opening Night Through July 27 at the Gielgud Theater in London; openingnightmusical.com .

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How to review a case report

Rakesh garg.

Department of Anaesthesiology, Pain and Palliative Care, DR BRAIRCH, AIIMS, Ansari Nagar, New Delhi, 110029 India

Shaheen E. Lakhan

Neurology and Medical Education, California University of Science and Medicine - School of Medicine, Colton, CA USA

Ananda K. Dhanasekaran

Sandwell & West Birmingham Hospitals, NHS Trust, Birmingham, UK

Introduction

Sharing individual patient experiences with clinical colleagues is an essential component of learning from each other. This sharing of information may be made global by reporting in a scientific journal. In medicine, patient management decisions are generally based on the evidence available for use of a particular investigation or technology [ 1 ]. The hierarchical rank of the evidence signifies the probability of bias. The higher up the hierarchy, the better its reliability and thus its clinical acceptance (Table  1 ). Though case reports remain lowest in the hierarchy of evidence, with meta-analysis representing the highest level, they nevertheless constitute important information with regard to rare events and may be considered as anecdotal evidence [ 2 ] (Table  1 ). Case reports may stimulate the generation of new hypotheses, and thus may support the emergence of new research.

Levels of evidence

Produced by Bob Phillips, Chris Ball, Dave Sackett, Doug Badenoch, Sharon Straus, Brian Haynes, Martin Dawes since November 1998. Updated by Jeremy Howick March 2009. Assessed from: http://www.cebm.net/oxford-centre-evidence-based-medicine-levels-evidence-march-2009. Assessed on 1 Nov 2015

The definition of a case report or a case series is not well defined in the literature and has been defined variously by different journals and authors. However, the basic definition of a case report is the detailed report of an individual including aspects like exposure, symptoms, signs, intervention, and outcome. It has been suggested that a report with more than four cases be called a case series and those with fewer than four a case report [ 3 ]. A case series is descriptive in design. Other authors describe “a collection of patients” as a case series and “a few patients” as a case report [ 4 ]. We suggest that should more than one case be reported, it may be defined as a case series—a concept proposed by other authors [ 5 ].

The importance of case reports

A case report may describe an unusual etiology, an unusual or unknown disorder, a challenging differential diagnosis, an unusual setting for care, information that can not be reproduced due to ethical reasons, unusual or puzzling clinical features, improved or unique technical procedures, unusual interactions, rare or novel adverse reactions to care, or new insight into the pathogenesis of disease [ 6 , 7 ]. In recent years, the publication of case reports has been given low priority by many high impact factor journals. However, the need for reporting such events remains. There are some journals dedicated purely to case reports, such as the Journal of Medical Case Reports , emphasizing their importance in modern literature. In the past, isolated case reports have led to significant advancements in patient care. For example, case reports concerning pulmonary hypertension and anorexic agents led to further trials and the identification of the mechanism and risk factors associated with these agents [ 2 , 8 ].

Reporting and publishing requirements

The reporting of cases varies for different journals. The authors need to follow the instructions for the intended publication. Owing to significant variability, it would be difficult to have uniform publication guidelines for case reports. A checklist called the CARE guidelines is useful for authors writing case reports [ 9 , 10 ]. However, it would be universally prudent to include a title, keywords, abstract, introduction, patient information, clinical findings, timeline, diagnostic assessment, therapeutic interventions, follow-up and outcomes, discussion, patient perspective, and informed consent.

Peer review process

The peer review process is an essential part of ethical and scientific writing. Peer review ultimately helps improve articles by providing valuable feedback to the author and helps editors make a decision regarding publication. The peer reviewer should provide unbiased, constructive feedback regarding the manuscript. They may also highlight the strengths and weaknesses of the report. When reviewing an article, it is prudent to read the entire manuscript first to understand the overall content and message. The reviewer than may read section-wise and provide comments to the authors and editorial team accordingly. The reviewer needs to consider the following important points when reviewing a case for possible publication [ 8 , 9 ] (summarized in Table  2 ).

Checklist for case report reviewer

Novelty remains the foremost important aspect of a case. The case report should introduce novel aspects of patient evaluation, investigation, treatment, or any other aspect related to patient care. The relevant information becomes a hypothesis generator for further study. The novelty may at times be balanced with some important information like severe adverse effects, even if they have been reported earlier. Reporting adverse events remains important so that information on cumulative adverse effects can be gathered globally, which helps in preparing a policy or guideline or a warning note for its use in patients. The data related to adverse effects include not only the impact but also the number of patients affected. This becomes more important for serious adverse effects. In the absence of an international registry for adverse effects, published case reports are important pieces of information. Owing to ethical concerns, formal evaluation may not be feasible in the format of prospective study.

Essential description

The case needs to have all essential details to allow a useful conclusion to emerge. For example, if a case is being reported for hemodynamic variability due to a drug, then the drug dose and timing along with timed vital signs need to be described.

Authenticity and genuineness

Honesty remains the most important basic principle of all publications. This remains a primary responsibility of the authors. However, if there is any doubt, reviewers may seek clarification. This doubt may result from some discordance in the case description. At times, a lack of correlation between the figures and description may act as “red flags.” For instance, authors may discuss a technique for dealing with a difficult airway, but the figure is of a normal-appearing airway. Another example would be where the data and figure do not correlate in a hemodynamic response related to a drug or a technique, with the graphical picture or screenshot of hemodynamics acting as an alert sign. Such cause for concern may be communicated in confidence to the editor.

Ethical or competing interests

Ethical issues need to be cautiously interpreted and communicated. The unethical use of a drug or device is not desirable and often unworthy of publication. This may relate to the route or dose of the drug administered. The off-label use of drugs where known side effects are greater than potential benefit needs to be discouraged and remains an example of unethical use. This use may be related to the drug dose, particularly when the drug dose exceeds the routine recommended dose, or to the route of administration. As an example, the maximal dose of acetaminophen (paracetamol) is 4g/day, and if an author reports exceeding this dose, it should be noted why a greater than recommended dose was used. Ultimately, the use of a drug or its route of administration needs to be justified in the manuscript. The reviewers need to serve as content experts regarding the drugs and other technologies used in the case. A literature search by the reviewer provides the data to comment on this aspect.

Competing interests (or conflicts of interest) are concerns that interfere or potentially interfere with presentation, review, or publication. They must be declared by the authors. Conflicts can relate to patient-related professional attributes (like the use of a particular procedure, drug, or instrument) being affected by some secondary gains (financial, non-financial, professional, personal). Financial conflict may be related to ownership, paid consultancy, patents, grants, honoraria, and gifts. Non-financial conflicts may be related to memberships, relationships, appearance as an expert witness, or personal convictions. At times, the conflict may be related to the author’s relationship with an organization or another person. A conflict may influence the interpretation of the outcome in an inappropriate and unscientific manner. Although conflicts may not be totally abolished, they must be disclosed when they reasonably exist. This disclosure should include information such as funding sources, present membership, and patents pending. Reviewers should cautiously interpret any potential bias regarding the outcome of the case based on the reported conflicts. This is essential for transparent reporting of research. At times, competing interests may be discovered by a reviewer and should be included in comments to the editorial team. Such conflicts may again be ascertained when the reviewer reviews the literature during the peer review process. The reviewer should also disclose their own conflicts related to the manuscript review when sending their report to the editorial team.

Impact on clinical practice

This is an important aspect for the final decision of whether to publish a case report. The main thrust or carry-home message needs to be emphasized clearly. It needs to be elaborated upon in concluding remarks.

Patient anonymity, consent, and ethical approval

When reviewing the manuscript of a case report, reviewers should ensure that the patient’s anonymity and confidentiality is protected. The reviewers should check that patient identifiers have been removed or masked from all aspects of the manuscript, whether in writing or within photograph. Identifiers can include things like the name of the patient, geographical location, date of birth, phone numbers, email of the patient, medical record numbers, or biometric identifiers. Utmost care needs to be taken to provide full anonymity for the patient.

Consent is required to participate in research, receive a certain treatment, and publish identifiable details. These consents are for different purposes and need to be explained separately to the patient. A patient’s consent to participate in the research or for use of the drug may not extend to consent for publication. All these aspects of consent must be explained to the patient, written explicitly in the patient’s own language, understood by the patient, and signed by the patient. For the purpose of the case, the patient must understand and consent for any new technique or drug (its dose, route, and timing) being used. In the case of a drug being used for a non-standard indication or route, consent for use must also be described. Patient consent is essential for the publication of a case if patient body parts are displayed in the article. This also includes any identifiers that can reveal the identity of the patient, such as the patient’s hospital identification number, address, and any other unique identifier. In situations where revealing the patient’s identity cannot be fully avoided, for example if the report requires an image of an identifiable body part like the face, then this should be explained to the patient, the image shown to them, and consent taken. Should the patient die, then consent must be obtained from next of kin or legal representative.

With case series, securing individual patient consent is advised and preferable. The authors may also need institutional review board (IRB) approval to publish a case series. IRBs can waive the need for consent if a study is conducted retrospectively and data are collected from patient notes for the purpose of research, usually in an anonymized way. However, wherever possible, individual patient consent is preferable, even for a retrospective study. Consent is mandatory for any prospective data collection for the purpose of publication as a case series. Consent and/or IRB approval must be disclosed in the case report and reasons for not obtaining individual consent may be described, if applicable.

There may be situations in which publishing patient details without their consent is justified, but this is a decision that should be made by the journal editor, who may decide to discuss the case with the Committee on Publication Ethics. Reviewers need to emphasize the issue to the editor when submitting their comments.

Manuscript writing

The CARE guidelines provide a framework that supports transparency and accuracy in the publication of case reports and the reporting of information from patient encounters. The acronym CARE was created from CA (the first two letters in “case”) and RE (the first two letters in “reports”). The initial CARE tools are the CARE checklist and the Case Report Writing Templates. These tools support the writing of case reports and provide data that inform clinical practice guidelines and provide early signals of effectiveness, harms, and cost [ 10 ].

The presentation of the case and its interpretation should be comprehensive and related. The various components of the manuscript should have sufficient information for understanding the key message of the case. The reviewer needs to comment on the relevant components of the manuscript. The reviewer should ascertain that the title of the case manuscript is relevant and includes keywords related to the case. The title should be short, descriptive, and interesting. The abstract should be brief, without any abbreviations, and include keywords. It is preferable to use Medical Subject Headings (MeSH) keywords. Reviewers must ensure that the introduction emphasizes the context of the case and describes the relevance and its importance in a concise and comprehensive manner. The case description should be complete and should follow basic rules of medical communication. The details regarding patient history, physical examination, investigations, differential diagnosis, management, and outcome should be described in chronological order. If repeated observations are present, then they may be tabulated. The use of graphs and figures helps the readers to better understand the case. Interpretation or inferences based on the outcomes should be avoided in this section and should be considered a part of the discussion. The discussion should highlight important aspects of the case, with its interpretation within the context of the available literature. References should be formatted as per the journal style. They should be complete and preferably of recent publications.

Reviewer responsibility

The reviewer’s remarks are essential not only for the editorial team but also for authors. A good peer review requires honesty, sincerity, and punctuality. Even if a manuscript is rejected, the authors should receive learning points from peer review commentary. The best way to review a manuscript is to read the manuscript in full for a gross overview and develop general comments. Thereafter, the reviewer should address each section of the manuscript separately and precisely. This may be done after a literature search if the reviewer needs to substantiate his/her commentary.

Constructive criticism

The reviewer’s remarks should be constructive to help the authors improve the manuscript for further consideration. If the manuscript is rejected, the authors should have a clear indication for the rejection. The remarks may be grouped as major and minor comments. Major comments likely suggest changes to the whole presentation, changing the primary aim of the case report, or adding images. Minor comments may include grammatical errors or getting references for a statement. The editorial team must be able to justify their decision on whether or not to accept an article for publication, often by citing peer review feedback. It is also good style to tabulate a list of the strengths and weaknesses of the manuscript.

Fixed time for review

Reviewer remarks should be submitted within a specified timeframe. If any delay is expected, it should be communicated to the editorial team. Reviewers should not rush to submit feedback without sufficient time to adequately review the paper and perform any necessary literature searches. Should a reviewer be unable to submit the review within the specified timeframe, they should reply to the review invitation to decline at their earliest convenience. If, after accepting a review invitation, the reviewer realizes they do not have time to perform the review, this must be communicated to the editorial team.

Conflict of interest

The reviewer’s conflicts of interest should be included along with the review. The conflicts may be related to the contents of the case, drugs, or devices pertaining to the case; the author(s); or the affiliated institution(s) of the author(s).

Lack of expertise

The reviewer may decline to review the manuscript if they think the topic is out of their area of expertise. If, after accepting an invitation to review, the reviewer realizes they are unable to review the manuscript owing to a lack of expertise in that particular field, they should disclose the fact to the editorial team.

Confidentiality

The reviewer should keep the manuscript confidential and should not use the contents of the unpublished manuscript in any form. Discussing the manuscript among colleagues or any scientific forum or meetings is inappropriate.

Review of revised manuscript

At times, a manuscript is sent for re-review to the reviewer. The reviewer should read the revised manuscript, the author’s response to the previous round of peer review, and the editorial comments. Sometimes, the authors may disagree with the reviewer’s remarks. This issue needs to be elaborated on and communicated with the editor. The reviewer should support their views with appropriate literature references. If the authors justify their reason for disagreeing with the viewer, then their argument should be considered evidence-based. However, if the reviewer still requests the revision, this may be politely communicated to the author and editor with justification for the same. In response to reviewers remarks, authors may not agree fully and provide certain suggestion in the form of clarification related to reviewers remarks. The reviewers should take these clarifications judiciously and comment accordingly with the intent of improving the manuscript further.

Peer reviewers have a significant role in the dissemination of scientific literature. They act as gatekeepers for science before it is released to society. Their sincerity and dedication is paramount to the success of any journal. The reviewers should follow a scientific and justifiable methodology for reviewing a case report for possible publication. Their comments should be constructive for the overall improvement of the manuscript and aid the editorial team in making a decision on publication. We hope this article will help reviewers to perform their important role in the best way possible. We send our best wishes to the reviewer community and, for those who are inspired to become reviewers after reading this article, our warm welcome to the reviewers’ club.

Competing interests

The authors declare that they have no competing interests.

Authors’ contributions

All authors read and approved the final manuscript.

Scientists try to unravel the case of 1,300 mysteriously preserved human brains

Study documents discoveries of long decayed human remains with remarkably intact brains.

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Oxford University's Alexandra Morton-Hayward spends her days surrounded by brains — literally. 

The undertaker-turned-scientist is trying to unravel why some human brains remain remarkably well-preserved after death, sometimes for thousands of years, even when all other soft tissue has long decayed.

And anyone visiting her at work would be well-advised not to go looking for a snack. 

"We do have four and a half fridges, so you'd think they might be in with a good chance of a meal," Morton-Hayward told As It Happens host Nil Köksal. 

"But no, they are stuffed to the brim with preserved ancient brains."

Morton-Hayward, a postgraduate researcher with the university's earth sciences department, has co-authored a study about the phenomenon. The findings were published this week in the journal Proceedings of the Royal Society B: Biological Sciences.

'Just a brain rattling around in a skull'

In forensics, it's common wisdom that when a body decomposes, the brain is the first to go, Morton-Hayward says.

But sometimes, she says, the opposite is true. 

She and her colleagues have documented 4,400 cases around the world, dating back to the 1600s, where preserved human brains have been discovered. 

In most cases, there's a reasonable explanation. The brains and other soft tissues were preserved either intentionally through death rituals, or naturally by environmental factors.

But one third — roughly 1,300 — remain a complete mystery. 

"They're all of what we describe as the unknown type of preservation. That is, they're the only soft tissue amongst otherwise skeletonized remains," Morton-Hayward said. "So just a brain rattling around in a skull."

The oldest, she says, dates back 12,000 years. And they come from all over.

"They are found in just the most intriguing sites," she said. "We have your regular cemetery burials, but we also have   Korean tombs, we have sunken shipwrecks, folks dressed in otter skins and buried in tree-log coffins. We have burial in clay in Japan, swamps in Florida — just all sorts of different environments."

While the origins are diverse, the brains themselves share a lot of similarities. For one, they tend to be stained bright red, orange or yellow.

"They literally look rusty,"  Morton-Hayward said.

They're all remarkably well-preserved, she said — just shrunken.

"They fit really neatly in the palm of your hand, kind of like a large walnut, if you will."

Studying death to learn about life

So what keeps these brains intact when other organs are long gone?

"It is very strange, but the fact that they do preserve in the absence of any other soft tissue suggests a mechanism of preservation that's unique to the nervous system," Morton-Hayward said.

"And we need to be thinking about what is unique about the nervous system in its biochemical makeup in life that could be dictating how it behaves in decay after death."

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The nervous system, she says, has a "really unusual ratio" of lipids, which are fatty compounds, and proteins, which are complex molecules in the cells. 

"Those proteins and those lipids are very specific types that occur more often in the brain than any other organ. And over time, those proteins and lipids can fuse together, particularly in the presence of metal like iron, which naturally accumulate in the brain during the process of aging," she said.

That process, she says, could potentially create molecules that allow the brain tissue to survive long-term. 

That mechanism, she says, is similar to what happens in brains with dementia. 

"I never thought when I set out to study ancient brains that it could have real world medical applications, but maybe, just maybe, it can help us understand brain aging and life," she said. 

"It can help us to shed light on past issues as well — things like paleo-pathology, the study of ancient diseases, the study of health status, of diet. All sorts of different things could be answered by recovering these ancient biomolecules, these proteins and lipids, from these brains."

'Untapped potential' 

Howard Williams, an archaeologist at the U.K.'s University of Chester who was not involved in the study, agreed. 

"It's showing the untapped potential for further analysis of human brains from archaeological deposits," he told CBC.

It also, he says, raises ethical questions, and could further complicate already tricky discussions about, for example, the repatriation and reburial of remains from different Indigenous peoples. 

He also finds himself wondering how many preserved brains have already been either overlooked or discarded at archeological dig sites over the decades.

"I have a horrible feeling that the thousands of brains examined are probably only a fraction of what we could know if people had realized earlier the potential these soft tissue human remains have for us," he said.

Interview with Alexandra Morton-Hayward produced by Leila Ahouman

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