speech and language disorders prevalence

Language and Speech Disorders in Children

Helping children learn language, what to do if there are concerns.

• Detecting problems

Children are born ready to learn a language, but they need to learn the language or languages that their family and environment use. Learning a language takes time, and children vary in how quickly they master milestones in language and speech development. Typically developing children may have trouble with some sounds, words, and sentences while they are learning. However, most children can use language easily around 5 years of age.

Parents and caregivers are the most important teachers during a child’s early years. Children learn language by listening to others speak and by practicing. Even young babies notice when others repeat and respond to the noises and sounds they make. Children’s language and brain skills get stronger if they hear many different words. Parents can help their child learn in many different ways, such as

• Responding to the first sounds, gurgles, and gestures a baby makes.
• Repeating what the child says and adding to it.
• Talking about the things that a child sees.
• Asking questions and listening to the answers.
• Looking at or reading books.
• Telling stories.
• Singing songs and sharing rhymes.

This can happen both during playtime and during daily routines.

Parents can also observe the following:

• How their child hears and talks and compare it with typical milestones for communication skills external icon .
• How their child reacts to sounds and have their hearing tested if they have concerns .

Learn more about language milestones .  Watch milestones in action.

  Top of Page

Some languages are visual rather than spoken. American Sign Language uses visual signals, including gestures, facial expressions, and body movement to communicate.

Some children struggle with understanding and speaking and they need help. They may not master the language milestones at the same time as other children, and it may be a sign of a language or speech delay or disorder.

Language development has different parts, and children might have problems with one or more of the following:

• Not hearing the words (hearing loss).
• Not understanding the meaning of the words.
• Not knowing the words to use.
• Not knowing how to put words together.
• Knowing the words to use but not being able to express them.

Language and speech disorders can exist together or by themselves. Examples of problems with language and speech development include the following:

• Difficulty with forming specific words or sounds correctly.
• Difficulty with making words or sentences flow smoothly, like stuttering or stammering.
• Language delay – the ability to understand and speak develops more slowly than is typical
• Aphasia (difficulty understanding or speaking parts of language due to a brain injury or how the brain works).
• Auditory processing disorder (difficulty understanding the meaning of the sounds that the ear sends to the brain)

Learn more about language disorders external icon .

Language or speech disorders can occur with other learning disorders that affect reading and writing. Children with language disorders may feel frustrated that they cannot understand others or make themselves understood, and they may act out, act helpless, or withdraw. Language or speech disorders can also be present with emotional or behavioral disorders, such as attention-deficit/hyperactivity disorder (ADHD) or anxiety . Children with developmental disabilities including autism spectrum disorder may also have difficulties with speech and language. The combination of challenges can make it particularly hard for a child to succeed in school. Properly diagnosing a child’s disorder is crucial so that each child can get the right kind of help.

Detecting problems with language or speech

If a child has a problem with language or speech development, talk to a healthcare provider about an evaluation. An important first step is to find out if the child may have a hearing loss. Hearing loss may be difficult to notice particularly if a child has hearing loss only in one ear or has partial hearing loss, which means they can hear some sounds but not others. Learn more about hearing loss, screening, evaluation, and treatment .

A language development specialist like a speech-language pathologist external icon will conduct a careful assessment to determine what type of problem with language or speech the child may have.

Overall, learning more than one language does not cause language disorders, but children may not follow exactly the same developmental milestones as those who learn only one language. Developing the ability to understand and speak in two languages depends on how much practice the child has using both languages, and the kind of practice. If a child who is learning more than one language has difficulty with language development, careful assessment by a specialist who understands development of skills in more than one language may be needed.

Treatment for language or speech disorders and delays

Children with language problems often need extra help and special instruction. Speech-language pathologists can work directly with children and their parents, caregivers, and teachers.

Having a language or speech delay or disorder can qualify a child for early intervention external icon (for children up to 3 years of age) and special education services (for children aged 3 years and older). Schools can do their own testing for language or speech disorders to see if a child needs intervention. An evaluation by a healthcare professional is needed if there are other concerns about the child’s hearing, behavior, or emotions. Parents, healthcare providers, and the school can work together to find the right referrals and treatment.

What every parent should know

Children with specific learning disabilities, including language or speech disorders, are eligible for special education services or accommodations at school under the Individuals with Disabilities in Education Act (IDEA) external icon and Section 504 external icon , an anti-discrimination law.

Get help from your state’s Parent Training and Information Center external icon

The role of healthcare providers

Healthcare providers can play an important part in collaborating with schools to help a child with speech or language disorders and delay or other disabilities get the special services they need. The American Academy of Pediatrics has created a report that describes the roles that healthcare providers can have in helping children with disabilities external icon , including language or speech disorders.

More information

CDC Information on Hearing Loss

National Institute on Deafness and Other Communication Disorders external icon

Birth to 5: Watch me thrive external icon

The American Speech-Language-Hearing Association external icon

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Am Fam Physician. 2024;109(4):361-362

As published by the USPSTF.

The full recommendation statement is available at https://www.uspreventiveservicestaskforce.org/uspstf/recommendation/speech-and-language-delay-and-disorders-in-children-age-5-and-younger-screening .

The USPSTF recommendations are independent of the U.S. government. They do not represent the views of the Agency for Healthcare Research and Quality, the U.S. Department of Health and Human Services, or the U.S. Public Health Service.

This series is coordinated by Joanna Drowos, DO, contributing editor.

A collection of USPSTF recommendation statements published in AFP is available at https://www.aafp.org/afp/uspstf .

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Recommendations

Public comments and nominations, about the uspstf.

• Recommendation
• Evidence Summary: Speech and Language Delay and Disorders in Children: Screening

Evidence Summary

Speech and language delay and disorders in children: screening, january 23, 2024.

Recommendations made by the USPSTF are independent of the U.S. government. They should not be construed as an official position of the Agency for Healthcare Research and Quality or the U.S. Department of Health and Human Services.

By Cynthia Feltner, MD, MPH; Ina F. Wallace, PhD; Sallie W. Nowell, PhD, CCC-SLP; Colin J. Orr, MD, MPH; Brittany Raffa, MD; Jennifer Cook Middleton, PhD; Jessica Vaughan, MPH; Claire Baker; Roger Chou, MD; Leila Kahwati, MD, MPH

The information in this article is intended to help clinicians, employers, policymakers, and others make informed decisions about the provision of health care services. This article is intended as a reference and not as a substitute for clinical judgment.

This article may be used, in whole or in part, as the basis for the development of clinical practice guidelines and other quality enhancement tools, or as a basis for reimbursement and coverage policies. AHRQ or U.S. Department of Health and Human Services endorsement of such derivative products may not be stated or implied.

This article was published online in JAMA on January 23, 2023 ( JAMA . 2024;331(4):335-351. doi:10.1001/jama.2023.24647).

Importance: Children with speech and language difficulties are at risk for learning and behavioral problems.

Objective: To review the evidence on screening for speech and language delay or disorders in children 5 years or younger to inform the US Preventive Services Task Force.

Data Sources: PubMed/MEDLINE, Cochrane Library, PsycInfo, ERIC, Linguistic and Language Behavior Abstracts (ProQuest), and trial registries through January 17, 2023; surveillance through November 24, 2023.

Study Selection: English-language studies of screening test accuracy, trials or cohort studies comparing screening vs no screening; randomized clinical trials (RCTs) of interventions.

Data Extraction and Synthesis: Dual review of abstracts, full-text articles, study quality, and data extraction; results were narratively summarized.

Main Outcomes and Measures: Screening test accuracy, speech and language outcomes, school performance, function, quality of life, and harms.

Results: Thirty-eight studies in 41 articles were included (N = 9006). No study evaluated the direct benefits of screening vs no screening. Twenty-one studies (n = 7489) assessed the accuracy of 23 different screening tools that varied with regard to whether they were designed to be completed by parents vs trained examiners, and to screen for global (any) language problems vs specific skills (eg, expressive language). Three studies assessing parent-reported tools for expressive language skills found consistently high sensitivity (range, 88%-93%) and specificity (range, 88%-85%). The accuracy of other screening tools varied widely. Seventeen RCTs (n = 1517) evaluated interventions for speech and language delay or disorders, although none enrolled children identified by routine screening in primary care. Two RCTs evaluating relatively intensive parental group training interventions (11 sessions) found benefit for different measures of expressive language skills, and 1 evaluating a less intensive intervention (6 sessions) found no difference between groups for any outcome. Two RCTs (n = 76) evaluating the Lidcombe Program of Early Stuttering Intervention delivered by speech-language pathologists featuring parent training found a 2.3% to 3.0% lower proportion of syllables stuttered at 9 months compared with the control group when delivered in clinic and via telehealth, respectively. Evidence on other interventions was limited. No RCTs reported on the harms of interventions.

Conclusions and Relevance: No studies directly assessed the benefits and harms of screening. Some parent-reported screening tools for expressive language skills had reasonable accuracy for detecting expressive language delay. Group parent training programs for speech delay that provided at least 11 parental training sessions improved expressive language skills, and a stuttering intervention delivered by speech-language pathologists reduced stuttering frequency.

An estimated 8% of US children aged 3 to 17 years have a communication disorder. 1 Boys are almost twice as likely to be affected than girls (9.6% vs 5.7%,) and higher rates are observed among Black children (10%) compared with Hispanic (6.9%) or White (7.8%) children. 1 These data and other nationally representative prevalence estimates are limited in terms of distinguishing children who have a delay vs specific speech and/or language disorder.

A “delay” refers to development of speech and language in the correct sequence but at a slower rate than expected, whereas a “disorder” refers to development of speech and/or language ability that is qualitatively different from typical development. Speech disorders are characterized by difficulty with forming specific sounds or words correctly (articulation or phonological disorders) or making words or sentences flow smoothly (fluency disorders), and language disorders are characterized by difficulty understanding (receptive language) or speaking (expressive language) relative to their peers. 2 The focus of this review is routine screening for developmental (or “primary”) speech or language delay and disorders that are not caused by an injury or another condition (acquired or “secondary” disorders) such as hearing loss (eg, secondary to infection or genetic syndrome) or autism. Evaluation of children with known conditions that affect speech or language development would be part of disease management rather than screening; however, in the context of routine screening, some children who screen positive may go on to receive a primary diagnosis for a disorder such as hearing loss following a diagnostic evaluation.

Many children identified with speech or language delay go on to recover without an intervention. 3 However, observational evidence suggests that school-aged children with speech or language delay may be at increased risk of learning and literacy disabilities. 4-6 and social and behavioral problems, 7 some of which may persist through adulthood. 8 , 9 Screening for speech and language delay is distinct from overall developmental screening recommended by the American Academy of Pediatrics at 18 and 30 months. 10 Children who screen positive require referral for a diagnostic evaluation to confirm the suspected delay or disorder. Once a diagnosis is confirmed, treatment is variable and individualized to the needs of the child based on how the disorder impairs their function in different settings.

In 2015, the US Preventive Services Task Force (USPSTF) concluded that the evidence was insufficient to assess the balance of benefits and harms of screening for speech and language delay and disorders in children 5 years or younger (I statement). 11 The purpose of the current systematic review was to update the previous evidence review on the benefits and harms of screening for speech and language delay and disorders in children to inform the USPSTF in updating its recommendation.

Scope of Review

Figure 1 shows the analytic framework and key questions (KQs) that guided the review. Detailed methods are available in the full evidence review. 12 In addition to the KQs, this review looked for evidence related to 3 contextual questions that focused on disparities in the prevalence, detection, and provision and utilization of treatment for speech and language delay or disorders among specific populations of children (eContextual Questions in the JAMA Supplement).

Data Sources and Searches

PubMed/MEDLINE, the Cochrane Library, APA PsycInfo, ERIC, and Linguistic and Language Behavior Abstracts (ProQuest) were searched for English-language articles published through January 17, 2023 (eMethods in the  JAMA  Supplement). ClinicalTrials.gov was searched for unpublished studies. The searches were supplemented by reviewing reference lists of pertinent articles, studies suggested by peer reviewers, and comments received during public commenting periods. From January 17, 2023, through November 24, 2023, ongoing surveillance was conducted through article alerts and targeted searches of journals to identify major studies published in the interim that may affect the conclusions or understanding of the evidence and the related USPSTF recommendation.

Study Selection

Two investigators independently reviewed titles, abstracts, and full-text articles using prespecified eligibility criteria (eTable 4 in the JAMA  Supplement). Disagreements were resolved by discussion and consensus. For all KQs, English-language studies enrolling unselected children 5 years or younger from primary care or primary care–relevant settings (including childcare, schools, and other education settings) who communicate using any language were eligible. In addition, only studies set in countries categorized as “very high” on the Human Development Index 13 and rated as fair or good quality were included. For studies assessing the benefits and harms of interventions (KQ4, KQ5, and KQ6), those enrolling children referred for treatment or identified by educators or parents as having a possible speech or language problem, and those enrolling children up to age 6 years were also eligible.

For KQ2, studies assessing the accuracy of a screening instrument against a diagnosis reference standard (diagnostic interview, diagnostic questionnaire, or both) were included. Eligible screening instruments had to be feasible for use in primary care and included short questionnaires that could be delivered and interpreted in 10 minutes or less in clinical settings and longer questionnaires completed by parents or teachers outside of a scheduled visit. Studies focusing on the accuracy of general developmental screening tools that did not include a separate component for speech and language skills were excluded.

Randomized clinical trials (RCTs), nonrandomized clinical trials, and controlled cohort studies were eligible for KQ1 and KQ3 (benefit and harms of screening compared with no screening) and KQ6 (harms of interventions compared with an inactive control). For studies reporting on the benefit of interventions to improve speech and language outcomes (KQ4) or academic skills, behavior, function, or quality of life (KQ5), RCTs comparing an intervention with an inactive control were eligible. For KQ4, KQ5, and KQ6, eligible interventions included any treatment designed to improve speech and/or language delay or disorders among eligible populations, regardless of format (eg, individual or group settings, face-to-face, or via telehealth) or delivery personnel (eg, speech-language pathologists [SLPs] or other clinicians, parents, or teachers).

Data Extraction and Quality Assessment

For each included study, 1 investigator extracted pertinent information about the methods, populations, interventions, comparators, outcomes, timing, settings, and study designs. All data extractions were checked by a second investigator for completeness and accuracy. For newly identified studies, 2 reviewers independently assessed each study’s methodological quality using predefined criteria developed by the USPSTF (eMethods in the JAMA  Supplement) and informed by tools designed for various study designs (Cochrane Risk of Bias 2.0 tool for RCTs; 14 Quality Assessment of Diagnostic Accuracy Studies 2 for screening test accuracy). 15 For eligible studies included in the previous update for this topic, quality ratings were spot-checked and carried forward. Disagreements were resolved by discussion.

Data Synthesis and Analysis

Findings for each KQ were summarized in tabular and narrative format. The overall strength of the evidence for each KQ was assessed as high, moderate, low, or insufficient based on the overall quality of the studies, consistency of results between studies, precision of findings, risk of reporting bias, and limitations of the body of evidence using methods developed for the USPSTF (and the Evidence-based Practice Center program). 16 , 17 Additionally, the applicability of the findings to US primary care populations and settings was assessed. Discrepancies were resolved through consensus discussion.

For studies included for KQ2 (accuracy of screening tools), sensitivity, specificity, likelihood ratios, and predictive values were calculated based on data reported by articles, when sufficient, to compare consistency across similar measures. To determine whether meta-analyses were appropriate, the clinical heterogeneity and methodological heterogeneity of the studies were assessed following established guidance. 18 Due to heterogeneity in populations, outcome measures and other factors, as well as few studies assessing the same screening tool or interventions, meta-analysis was not appropriate.

A total of 38 studies (reported in 41 articles) were included ( Figure 2 ) in the review. Individual study quality ratings are reported in eTables 5 through 10 in the  JAMA  Supplement.

Benefits of Screening

Key Question 1. Does screening for speech and language delay or disorders in children age 5 years or younger improve speech and language outcomes, school performance, function, or quality-of-life outcomes?

No eligible study addressed this question.

Accuracy of Screening

Key Question 2. What is the accuracy of screening tools to detect speech and language delay or disorders in children age 5 years or younger?

Twenty-one studies (reported in 23 articles) assessed the accuracy of 23 screening instruments for detecting speech and language delay and disorders in young children against a reference standard (n = 7489) ( Table 1 ). 19-41 Seven studies were new to this update. 24 , 27 , 30-32 , 39 , 41 Of the 23 instruments,13 19-23 , 28-32 , 35 , 37 , 38 were designed to be administered to children by a trained examiner, and 10 23-27 , 33-36 , 39-41 were parent reports of children’s speech or language skills ( Table 2 ).

Some screening tools, termed global screening tools, screen for any language problems, while others provide scores for specific aspects of language (eg, expressive communication, receptive language, vocabulary). Twelve global screening tools were evaluated in the studies included the Ages and Stages Questionnaire (ASQ), 23 , 41 the Davis Observation Checklist for Texas, 19 the Developmental Nurse Screen, 35 the Early Language Scale, 39 the Fluharty Preschool Screening Test (FPST), 20 the General Language Screen, 36 the Hackney Early Language Screening Test/Structured Screening Test (HELST/SST), 28 , 29 the Infant-Toddler Checklist, 40 the Nurse Screening, 30 , 31 the Parent Questionnaire, 35 the Screening Kit of Language Development (SKOLD)/Screening Kit of Language Development Black English (SKOLDBE), 21 and the language component of the Sentence Repetition Screening Test (SRST). 38

Nine other tools provided scores for specific aspects of language, including the Brigance Preschool Screen, 23 the Early Screening Profiles, 23 the Battelle the Elternfragebogen für die Fruberkennung von Riskokindern (ELFRA-2), 33 , 34 the Sprachentwicklungsscreening (SPES-3) instrument, 24 the Language Development Survey (LDS), 25 , 26 the Quick Interactive Language Screener (QUILS), 32 the Sure Start Language Measure (SSLM), 41 the Northwestern Syntax Screening Test, 20 and the Battelle Developmental Inventory Screening Test–Communication. 23 Three of the trained examiner tools specifically screened for articulation skills—the Denver Articulation Screening Exam 22 and the articulation portion of both the Fluharty Preschool Speech and Language Screening Test (FPSLST) 37 and the SRST 38 —and 1 parent-administered instrument measured articulation. 27 The articulation instruments were considered separately from specific language instruments. All but 3 instruments (ie, ASQ, 23 , 41 HELST/SST, 28 , 29 and Nurse Screening 30 , 31 ) were examined in only 1 study each. In addition, 2 studies examined the FPST 20 and a later version with a language component, the FPSLST. 37

Excluding 2 studies 33 , 40 that enrolled all children who screened positive and a random sample of children who screened negative, the prevalence of speech and language disorders based on reference standards ranged from 4% to 33% ( Table 3 ).

Accuracy of Instruments

As shown in Table 3 , the sensitivity of instruments for detecting speech and language disorders and delay ranged from 17% and 100% (median, 86%), and specificity ranged between 32% and 98% (median, 87%). To further examine accuracy, the source of the information (parent report vs trained examiner) and whether the instrument was designed as a global index of speech or language, a specific language skill (eg, word knowledge), or a measure of articulation were considered.

Parent Reported

Sensitivity and specificity of 14 parent-reported tools varied widely ( Table 3 ). Sensitivity ranged from 55% to 93% (median, 84%) and specificity ranged from 32% to 96% (median, 84%).

Global Language vs Specific Language vs Articulation . Limiting analysis to global language instruments based on parent reports, median sensitivity was 74%, ranging between 55% and 89%. Specificity was less variable, ranging between 73% and 95% (median, 79%). In contrast, both sensitivity and specificity of the 3 parent-reported instruments of specific skills (all emerging expressive language skills) were fairly consistent and high (median sensitivity, 91% [range, 83%-93%]; median specificity, 88% [range, 81%-96%]). The 1 parent-rated measure of articulation had a reasonably high sensitivity (86%) but low specificity (32%).

Trained Examiners

The median sensitivity of the 13 screening tools that trained examiners administered to children was 87% (range, 17%-100%), and the median specificity was 88% (range, 58% to 98%). Similar to parent-reported instruments, there is substantial variability in the accuracy of examiner-administered tools.

Global Language vs Specific Language vs Articulation . Restricting the accuracy summary to trained examiner screenings of global language resulted in median sensitivity of 88% (range, 17%-100%) and median specificity of 89% (69%-98%). The median sensitivity of trained examiner instruments for specific language skills was 86% (range, 56%-94%) and median specificity was 70% (range, 58%-90%). Across the 3 trained examiner tools for assessing articulation, the median sensitivity was only 66% (range, 43%-92%); however, median specificity was 96% (range, 93%-97%).

Harms of Screening

Key Question 3. What are the harms of screening for speech and language delay or disorders in children age 5 years or younger?

Benefits of Treatment

Key Question 4. Do interventions for speech and language delay or disorders in children age 6 years or younger improve speech and language outcomes?

Seventeen RCTs (18 articles) compared an intervention for speech and language delay or disorders with an inactive control (no treatment or wait-list control/delayed treatment). 42-59 Study characteristics are shown in eTable 11 in the  JAMA  Supplement. No studies enrolled children identified by routine screening in primary care. Most recruited participants from referrals to speech and language treatment centers (6 studies), 42 , 47 , 49 , 50 , 53 , 54 schools or early childhood education centers (4 studies), 43 , 46 , 48 , 56 or via advertisements or a mix of advertisements and outreach to schools, clinical settings, or community-based programs. 44 , 45 , 55 , 57 The mean age of enrolled populations ranged from 18.1 months to 67.8 months, with most (10 studies) enrolling a sample with a mean age of 48 months or older. The proportion of participants who were female ranged from 10% to 49%. Few studies reported on race or ethnicity; in 3 studies set in the US, populations were described as 100% Latino, 45 100% White, 57 and 1 was inclusive of different groups (2% American Indian, 3% Asian, 2% Black, 26% Hispanic, 12% multiracial, 54% White). 48 Interventions evaluated were heterogeneous and varied in terms of the range of disorders targeted, delivery personnel, intensity/duration, settings, and other factors (eTable 11 in the  JAMA  Supplement).

Eight RCTs assessed interventions specific to children with delayed expressive language (“late talkers”) and no obvious fluency or speech-sound impairment. 44 , 45 , 50-52 , 56-59 Of these, 3 RCTs evaluated parent-group training interventions focused on strategies to promote their child’s language development; training approaches and specific content varied, but all focused on naturalistic strategies (eg, expanding on child utterances, following the child’s interests, repeating what the child says, setting up the environment to encourage communication). Of these, 2 RCTs assessed modifications of the Hanen Program for Parents curriculum (featuring a combination of group training sessions composed of a small group of parents and a trained SLP or other trained facilitator, and individual consultations with the SLP while the child is present), 51 , 58 and 1 evaluated a similar group training program focused on improving child linguistic complexity. 50 Results varied by duration of the intervention and mean age of enrolled populations. In 2 RCTs in which the intervention was delivered to children with a mean age of 27 to 30 months over a longer duration (11 bimonthly 60- to 75-minute sessions in one of the trials 50 and 11 weekly 2.5-hour sessions plus 3 weekly home visits in the other trial 51 ), there was consistent benefit across different measures of expressive language outcomes (eTable 12 in the  JAMA  Supplement). The RCT delivering the parent group training to children with a mean age of 18 months over a shorter duration (6 weekly 2-hour sessions) found no significant difference between groups on any measure of receptive or expressive language outcomes. 58

Five other RCTs assessed different interventions for children with language delay and varied in terms of setting, delivery personnel, and other factors. 44 , 45 , 56 , 57 , 59 In general, results were inconsistent, with some studies showing improvement on some measures of receptive or expressive language but others not. Results are further summarized in the eResults and eTable 12 in the  JAMA  Supplement.

Two RCTs assessed fluency treatment for young children. Both focused on the Lidcombe Program of Early Stuttering Intervention. 54 , 55 This intervention is led by an SLP who trains parents to provide verbal contingencies for stutter-free speech (eg, “that was smooth talking”) and stuttering (eg, “that was a bit bumpy”) and requests for self-evaluation and self-correction (eg, “can you say that again”). In one of these RCTs, the intervention was delivered in a face-to-face format in a clinical setting 54 and in the other it was delivered via telehealth. 55 Results were consistent in showing a statistically significant improvement in stuttering fluency associated with the intervention. In the face-to-face intervention, children in the intervention group had a 2.3% (95% CI, 0.8-3.9) lower proportion of syllables stuttered than children in the control group at 9 months. Per the authors, this is above the minimum clinically important difference of 1.0% of syllables stuttered (the minimum difference that a listener would be able to distinguish). 54 However, no reference or clear rationale was provided to support this threshold. In the RCT using telehealth delivery of the intervention, the difference between the intervention and control group in change from baseline mean number of syllables stuttered was −3.0% ( P = .02) at 9 months. 55

Evidence on other intervention types targeting specific speech or language problems was limited and is further described in the eResults in the  JAMA  Supplement.

Key Question 5. Do interventions for speech and language delay or disorders in children age 6 years or younger improve school performance, function, or quality-of-life outcomes?

Eight RCTs reported on 1 or more outcomes specific to school performance, function, or quality of life using heterogeneous measures. 42 , 43 , 47 , 48 , 53 , 57-59 Characteristics are described above in KQ4 and detailed results are shown in eTable 15 in the  JAMA  Supplement. No RCTs assessing a similar intervention type reported on the same outcome domain, and most studies reporting on similar domains (eg, early literacy) used different outcome measures. In 4 RCTs reporting on a measure of early or emergent literacy skills, 3 found no significant difference between groups. 42 , 43 , 48 In contrast, 1 RCT assessing a home-based language delay intervention delivered by trained assistants found benefit for improving letter knowledge associated with the intervention. 59 Two RCTs reported on 1 or more measures of functional communication 42 , 47 and quality of life/wellbeing in children 43 , 53 and found no difference between groups, while 1 RCT evaluating an individual intervention for language delay found significant improvement favoring the intervention for improving child socialization skills and parental stress levels. 57

Harms of Treatment

Key Question 6. What are the harms of interventions for speech and language delay or disorders?

This systematic review synthesized evidence relevant to screening for speech and language delay or disorders in children 5 years or younger. Table 4 summarizes the main findings of the evidence review. There was no direct evidence on the benefits and harms of screening (KQ1). Potential harms of screening (KQ3) include false-positive results that can lead to unnecessary referrals (and the associated time and economic burden), labeling or stigma, parent anxiety, and other psychosocial harms. Other harms of screening are likely to be minimal because screening is noninvasive.

The studies of screening test accuracy (KQ2) included in this review assessed 23 different tools that varied in terms of whether they were completed by parents vs trained examiners and whether they were designed to detect global speech or language problems vs problems related to specific language skills or articulation. Some screening tools usable in clinical practice may identify children who have a speech or language disorder with reasonable sensitivity and specificity. However, overall evidence was mixed and few screening tools were assessed by more than 1 study each, limiting the ability to make stronger conclusions about the accuracy of specific tools. Parent-reported screening instruments designed to assess expressive language skills displayed consistently high sensitivity and specificity, although precision varied by instrument. In contrast, the accuracy of the parent-reported instruments for global language skill assessment was inconsistent, and precision varied across instruments. The accuracy of examiner-administered screening instruments varied, particularly for instruments designed to assess specific language skills.

Few studies of interventions for speech and language delay or disorder enrolled similar populations and evaluated similar types of interventions (KQ4). Although 2 RCTs of treatment enrolled children newly referred from primary care, it is not clear whether the children were identified via routine screening vs case finding. Other included studies enrolled children referred or recruited via advertisements, and most focused on a specific type of speech delay or disorder. Given these factors, the body of evidence on treatment available for inclusion in this review may not be applicable to the type and severity of disorders that would be detected via routine screening in primary care settings.

Studies of children referred for language delay without obvious speech-sound or fluency disorder suggested that group training interventions offering at least 11 parent training sessions improved expressive language outcomes. For children identified with stuttering, the Lidcombe Program of Early Stuttering Intervention delivered by SLPs improved stuttering fluency at 9 months when delivered either in person or via telehealth. Although 8 RCTs reported on 1 or more outcomes specific to school performance or early literacy, health-related quality of life, function, behavior, or socialization (KQ5), the interventions and populations evaluated were heterogeneous, which limited the ability to assess consistency; most studies found no difference between groups for measures of early literacy, function, and quality of life. However, most trials may not have followed up children for a long enough duration to detect an improvement in quality of life or function that could result from early treatment of a speech and language delay or disorder. No RCTs reported on the harms of interventions; however, given the nature of the interventions, serious harms are unlikely.

Trials are needed that enroll asymptomatic or unselected populations from general primary care settings and directly assess the benefit of screening specifically for speech and language problems. The control groups in these trials could receive either no screening or routine screening for general developmental delay, with no separate score for speech and language problems. Studies are also needed on the potential harms of screening, such as labeling, and harms from false-positive results, such as burden on parents due to unnecessary referrals. Such studies would also inform the potential for overdiagnosis associated with routine screening, given that many children who have a speech delay may recover without intervention. 3

Similarly, studies assessing the accuracy of screening tools among unselected populations, who are ideally recruited through primary care settings, are needed because the prevalence of speech and language problems may vary compared with populations enrolled via advertisements or specialty settings. Specifically, studies that assess the accuracy of existing tools, compared with similar reference standards, would help determine the consistency of findings; because few included studies evaluated the same instrument, our ability to make a strong conclusion about accuracy was limited. Trials of treatment enrolling populations recruited from US primary care settings would help inform the potential benefit of screening because the range of severity and conditions is likely different compared with trials that enroll referred populations. Last, studies that followup children for a sufficiently long duration to detect improvement in academic performance, function, and quality of life would help in the understanding of whether immediate changes in speech and language outcomes (eg, short-term expansion of vocabulary words) translate into benefit for health and social outcomes.

Limitations

This review excluded studies in children who had a condition known to cause a speech or language problem (eg, hearing loss, autism) to improve the applicability of evidence to populations likely to be detected by routine screening. Studies evaluating primary prevention strategies to promote speech and language development (eg, interventions among groups considered “at risk” or school-based curricula emphasizing language development among children with no developmental delay or disorder) were also excluded. The aim was to limit the review to interventions that are relevant to children with screen-detected speech and language problems and that are appropriate to deliver in primary care settings or refer to from primary care.

This review found no eligible studies that reported on direct benefits or harms of screening compared with usual care or no screening. Parent-reported screening tools for expressive language delay had reasonable accuracy. In contrast, parent-reported screening tools for global language delay had inconsistent accuracy. The accuracy of examiner-administered instruments was also variable, especially for examiner-administered instruments of specific language skills. Existing evidence on treatment of speech and language delay is available from referral populations but not from screen-detected populations. This evidence indicates the benefit from group parent-training programs for speech delay that provide at least 11 parental training sessions for improving expressive language skills, as well as the Lidcombe Program of Early Stuttering Intervention delivered by SLPs for reducing stuttering frequency. Few studies reported on outcomes specific to school performance, function, quality of life, or behavior, and none reported on the harms of interventions.

Source: This article was published online in JAMA on January 23, 2023 ( JAMA . 2024;331(4):335-351. doi:10.1001/jama.2023.24647).

Conflict of Interest Disclosures: None reported.

Funding/Support: None reported.

Role of the Funder/Sponsor: Investigators worked with USPSTF members and AHRQ staff to develop the scope, analytic framework, and key questions for this review. AHRQ had no role in study selection, quality assessment, or synthesis. AHRQ staff provided project oversight, reviewed the evidence review to ensure that the analysis met methodological standards, and distributed the draft for peer review. Otherwise, AHRQ had no role in the conduct of the study; collection, management, analysis, and interpretation of the data; and preparation, review, or approval of the manuscript findings. The opinions expressed in this document are those of the authors and do not reflect the official position of AHRQ or the US Department of Health and Human Services.

Additional Contributions: We gratefully acknowledge the following individuals for their contributions to this project, including AHRQ staff (Justin Mills, MD, MPH; Tracy Wolff, MD, MPH), Scientific Resource Center for the AHRQ Evidence-based Practice Center Program staff (Robin A. Paynter, MLIS), Pacific Northwest Evidence-based Practice Center staff (Christina Bougatsos, MPH), and RTI International–University of North Carolina–Chapel Hill Evidence-based Practice Center staff (Manny Schwimmer, MPH; Christiane E. Voisin, MSLS; Roberta Wines, MPH; Mary Gendron; Sharon Barrell, MA; Alexander Cone; Teyonna Downing; Michelle Bogus). The USPSTF members, expert reviewers, and federal partner reviewers did not receive financial compensation for their contributions. Evidence-based Practice Center personnel received compensation for their roles in this project.

Additional Information: A draft version of the full evidence review underwent external peer review from 3 content experts (Abigail D. Delehanty, PhD, CCC-SLP, Duquesne University; Virginia Moyer, MD, MPH, University of North Carolina at Chapel Hill; Thelma E. Uzonyi, PhD, CCC-SLP, IMH-E, Kennedy Krieger Institute) and 3 federal partner reviewers (Centers for Disease Control and Prevention; Eunice Kennedy Shriver National Institute of Child Health and Human Development; and National Institute on Deafness and Other Communication Disorders). Comments from reviewers were presented to the USPSTF during its deliberation of the evidence and were considered in preparing the final evidence review. USPSTF members and peer reviewers did not receive financial compensation for their contributions.

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Evidence reviews for the US Preventive Services Task Force (USPSTF) use an analytic framework to visually display the key questions that the review will address to allow the USPSTF to evaluate the effectiveness and safety of a preventive service. The questions are depicted by linkages that relate interventions and outcomes. A dashed line depicts a health outcome that follows an intermediate outcome. For additional information, see the USPSTF Procedure Manual. 16 , 17

ERIC indicates Education Resources Information Center; KQ, key question; LLBA, Linguistics and Language Behavior Abstracts; USPSTF, US Preventive Services Task Force; and WHO ICTRP,World Health Organization International Clinical Trials Registry Platform. a The sum of the number of studies per KQ exceeds the total number of studies because some studies were applicable to multiple KQs.

Abbreviations: ASQ, Ages and Stages Questionnaire; ASQ-CD, ASQ–Communication Domain; BDIST-CD, Battelle Developmental Inventory Screening Test–Communication Domain; BPS, Brigance Preschool Screen; CDI, MacArthur-Bates Communicative Development Inventory; CSBS, Communication and Symbolic Behavior Scales; DASE, Denver Articulation Screening Exam; DNS, Developmental Nurse Screen; DOCT, Davis Observation Checklist for Texas; ELFRA-2, Elternfragebogen für die Fruberkennung von Riskokindern; ELS, Early Language Scale; ESP, Early Screening Profiles; FPSLST, Fluharty Preschool Speech and Language Screening Test; FPST, Fluharty Preschool Screening Test; GLS, General Language Screen; HELST, Hackney Early Language Screening Test; ICS-TC, Intelligibility in Context Scale–Traditional Chinese; ITC, Infant-Toddler Checklist; KQ, key question; LDS, Language Development Survey; NR, not reported; NSST, Northwestern Syntax Screening Test; QUILS, Quick Interactive Language Screening; SKOLD, Screening Kit of Language Development; SKOLDBE, Screening Kit of Language Development Black English; SPES-3, Sprachentwicklungsscreening; SRST, Sentence Repetition Screening Test; SSLM, Sure Start Language Measure; SST, Structured Screening Test; WIC, Women, Infants, and Children. a Full sample size, based on multiple imputation. b Includes 11 children (10.5%) who did not cooperate during screening and were considered screen positive. c Includes 11 children who were noncooperative during screening. For Model 4, parents of 10 children did not complete parental information. d Based on full sample.

Abbreviations: CDI, MacArthur-Bates Communicative Development Inventory; CSBS, Communication and Symbolic Behavior Scales; ELFRA-2, Elternfragebogen für die Fruberkennung von Riskokindern; KQ, key question; NR, not reported; SPES-3, Sprachentwicklungsscreening; WH questions, who, when, where, why, what, and how. a Only the Battelle Developmental Inventory Test Receptive Language Scale is included in accuracy analyses. b Although the SPES-3 was designed as both a parent-reported and trained examiner instrument, the authors recommended that only the parent-reported subscales be included as a screen for language delay; therefore, the SPES-3 was classified as a parent-reported instrument.

Abbreviations: AAPS-R, Arizona Articulation Proficiency Scale–Revised; ASQ-CD, Ages and Stages Questionnaire–Communication Domain; AWST-R, AktiverWortschatztest für 3-bis 5-jährige Kinder; BDIST-CD, Battelle Developmental Inventory Screening Test–Communication Domain; BLST, Bankson Language Screening Test; BPS, Brigance Preschool Screen; CCC-2, Children’s Communication Checklist, 2nd Edition–Netherlands; CDI, MacArthur-Bates Communicative Development Inventory; CSBS, Communication and Symbolic Behavior Scales; DASE, Denver Articulation Screening Exam; DNS, Developmental Nurse Screen; DOCT, Davis Observational Checklist for Texas; DP-II, Developmental Profile II; ELFRA-2, Elternfragebogen für die Fruberkennung von Riskokindern; ELS, Early Language Scale; ESP, Early Screening Profiles; FPSLST, Fluharty Preschool Speech and Language Screening Test; FPST, Fluharty Preschool Screening Test; GFTA, Goldman-Fristoe Test of Articulation; GLS, General Language Screen; HAT, Henja Articulation Test; HELST, Hackney Early Language Screening Test; HKCAT, Hong Kong Cantonese Articulation Test; ICS-TC, Intelligibility in Context Scale–Traditional Chinese; ITC, Infant-Toddler Checklist; ITPA, Illinois Test of Psycholinguistic Abilities; LDS, Language Development Survey; LLC, Lexilist Comprehension; LLP, Lexilist Production; LR+, positive likelihood ratio; LR–, negative likelihood ratio; LS, Language Standard; MLU, mean length of utterance; MSCA, McCarthy Scales of Children’s Abilities; MSEL, Mullen Scales of Early Learning; NPV, negative predictive value; NR, not reported; NSST, Northwestern Syntax Screening Test; PLS-4-C, Preschool Language Scale, Fourth Edition–Comprehension, PLS-4-E, Preschool Language Scale, Fourth Edition–Expression; PLS-5, Preschool Language Scale, Fifth Edition; PPV, positive predictive value; QUILS, Quick Interactive Language Screener; RDLS, Reynell Developmental Language Scales; ROC, receiver operating characteristic; SETK-2, Sprachentwicklungstest für zweijahrige Kinder; SETK-3, Sprachentwicklungstest für zweijahrige Kinder; SICD, Sequenced Inventory of Communication Development; SKOLD, Screening Kit of Language Development; SKOLDBE, Screening Kit of Language Development Black English; SLC, Schlichting Tests for Language Comprehension; SLP, speech-language pathologist; SPES-3, Sprachentwicklungsscreening; SSP, Schlichting Tests for Sentence Production; SRST, Sentence Repetition Screening Test; SSLM, Sure Start Language Measure; SST, Structured Screening Test; SWP, Schlichting Tests for Word Production; TACL-R, Test for Auditory Comprehension of Language–Revised; TD, Templin-Darley Tests of Articulation Consonant Singles Subtest; TOLD-P, Test of Language Development Primary. a Calculated by the Evidence-based Practice Center. b Optimal cut point using Youden index. c Prevalence not reported for this subsample. Median for sensitivity/specificity includes full sample only and not the English-speaking subsample. d Prevalence for screen failures more than 1.5 SD below the mean is 18%; study calculated accuracy using this value as well as prevalence using cut point of more than 2 SDs below the mean, which was 6%. Data were included for only the former prevalence. e Sample size and prevalence based on imputed sample, which corrected for oversampling of children with positive screening results. f Prevalence data provided by study authors. g Includes 11 children who were noncooperative during screening. h The study investigators weighted the ns based on a stratified sample of 69. i Only the BDIST-CD Receptive Scale is included in accuracy analyses.

Abbreviations: ANOVA, analysis of variance; ASQ, Ages and Stages Questionnaire; DOCT, Davis Observational Checklist for Texas; ELFRA-2, Elternfragebogen für die Fruberkennung von Riskokindern; ELS, Early Language Scale; HELST, Hackney Early Language Screening Test; KQ, key question; LDS, Language Development Survey; LR–, negative likelihood ratio; LR+, positive likelihood ratio; NA, not applicable; QOL, quality of life; RCT, randomized clinical trial; SKOLD, Screening Kit of Language Development; SLP, speech-language pathologist; SPES-3, Sprachentwicklungsscreening; SRST, Sentence Repetition Screening Test; SST, Structured Screening Test. a Frisk et al 23 examined 3 instruments and included separate accuracy calculations for the expressive and receptive PLS-4 reference measure. Accuracy outcomes were omitted for the Battelle Developmental Inventory Screening Test with the PLS-4 Expressive Communication Scale due to a possible reporting error in the study.

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Statistics and Epidemiology

Hearing, ear infections, and deafness.

• Quick Statistics About Hearing  
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• What the Numbers Mean: An Epidemiological Perspective on Hearing

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Epidemiology

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• Introduction
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Evidence reviews for the US Preventive Services Task Force (USPSTF) use an analytic framework to visually display the key questions that the review will address to allow the USPSTF to evaluate the effectiveness and safety of a preventive service. The questions are depicted by linkages that relate interventions and outcomes. A dashed line depicts a health outcome that follows an intermediate outcome. For additional information, see the USPSTF Procedure Manual. 16,17

ERIC indicates Education Resources Information Center; KQ, key question; LLBA, Linguistics and Language Behavior Abstracts; USPSTF, US Preventive Services Task Force; and WHO ICTRP, World Health Organization International Clinical Trials Registry Platform.

a The sum of the number of studies per KQ exceeds the total number of studies because some studies were applicable to multiple KQs.

eContextual Questions

eReferences

• USPSTF Recommendation: Screening for Speech and Language Delay and Disorders JAMA US Preventive Services Task Force January 23, 2024 This 2023 Recommendation Statement from the US Preventive Services Task Force concludes that the current evidence is insufficient to assess the balance of benefits and harms of screening for speech and language delay and disorders in children 5 years or younger without signs or symptoms (I statement). US Preventive Services Task Force; Michael J. Barry, MD; Wanda K. Nicholson, MD, MPH, MBA; Michael Silverstein, MD, MPH; David Chelmow, MD; Tumaini Rucker Coker, MD, MBA; Esa M. Davis, MD, MPH; Katrina E. Donahue, MD, MPH; Carlos Roberto Jaén, MD, PhD, MS; Li Li, MD, PhD, MPH; Carol M. Mangione, MD, MSPH; Gbenga Ogedegbe, MD, MPH; Goutham Rao, MD; John M. Ruiz, PhD; James Stevermer, MD, MSPH; Joel Tsevat, MD, MPH; Sandra Millon Underwood, PhD, RN; John B. Wong, MD
• Recommendations for Speech and Language Screenings JAMA Editorial January 23, 2024 Marisha L. Speights, PhD, CCC-SLP; Maranda K. Jones, BA; Megan Y. Roberts, PhD, CCC-SLP
• Patient Information: Screening for Speech and Language Problems in Young Children JAMA JAMA Patient Page January 23, 2024 This JAMA Patient Page describes the pros and cons of screening for speech and language problems in children aged 5 years or younger. Jill Jin, MD, MPH
• Early Language Screening and Improved Developmental Outcomes JAMA Network Open Editorial January 23, 2024 Ann P. Kaiser, PhD; Jason C. Chow, PhD; Jennifer E. Baumingham, PhD

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Feltner C , Wallace IF , Nowell SW, et al. Screening for Speech and Language Delay and Disorders in Children 5 Years or Younger : Evidence Report and Systematic Review for the US Preventive Services Task Force . JAMA. 2024;331(4):335–351. doi:10.1001/jama.2023.24647

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Screening for Speech and Language Delay and Disorders in Children 5 Years or Younger : Evidence Report and Systematic Review for the US Preventive Services Task Force

• 1 RTI International–University of North Carolina at Chapel Hill Evidence-based Practice Center
• 2 Department of Medicine, University of North Carolina at Chapel Hill
• 3 Cecil G. Sheps Center for Health Services Research, University of North Carolina at Chapel Hill
• 4 RTI International, Research Triangle Park, North Carolina
• 5 Frank Porter Graham Child Development Institute, University of North Carolina at Chapel Hill
• 6 Department of Pediatrics, University of North Carolina at Chapel Hill
• 7 The Pacific Northwest Evidence-Based Practice Center, Department of Medical Informatics and Clinical Epidemiology, Oregon Health & Science University
• 8 School of Medicine, Division of General Internal Medicine, Oregon Health & Science University
• Editorial Recommendations for Speech and Language Screenings Marisha L. Speights, PhD, CCC-SLP; Maranda K. Jones, BA; Megan Y. Roberts, PhD, CCC-SLP JAMA
• Editorial Early Language Screening and Improved Developmental Outcomes Ann P. Kaiser, PhD; Jason C. Chow, PhD; Jennifer E. Baumingham, PhD JAMA Network Open
• US Preventive Services Task Force USPSTF Recommendation: Screening for Speech and Language Delay and Disorders US Preventive Services Task Force; Michael J. Barry, MD; Wanda K. Nicholson, MD, MPH, MBA; Michael Silverstein, MD, MPH; David Chelmow, MD; Tumaini Rucker Coker, MD, MBA; Esa M. Davis, MD, MPH; Katrina E. Donahue, MD, MPH; Carlos Roberto Jaén, MD, PhD, MS; Li Li, MD, PhD, MPH; Carol M. Mangione, MD, MSPH; Gbenga Ogedegbe, MD, MPH; Goutham Rao, MD; John M. Ruiz, PhD; James Stevermer, MD, MSPH; Joel Tsevat, MD, MPH; Sandra Millon Underwood, PhD, RN; John B. Wong, MD JAMA
• JAMA Patient Page Patient Information: Screening for Speech and Language Problems in Young Children Jill Jin, MD, MPH JAMA

Importance   Children with speech and language difficulties are at risk for learning and behavioral problems.

Objective   To review the evidence on screening for speech and language delay or disorders in children 5 years or younger to inform the US Preventive Services Task Force.

Data Sources   PubMed/MEDLINE, Cochrane Library, PsycInfo, ERIC, Linguistic and Language Behavior Abstracts (ProQuest), and trial registries through January 17, 2023; surveillance through November 24, 2023.

Study Selection   English-language studies of screening test accuracy, trials or cohort studies comparing screening vs no screening; randomized clinical trials (RCTs) of interventions.

Data Extraction and Synthesis   Dual review of abstracts, full-text articles, study quality, and data extraction; results were narratively summarized.

Main Outcomes and Measures   Screening test accuracy, speech and language outcomes, school performance, function, quality of life, and harms.

Results   Thirty-eight studies in 41 articles were included (N = 9006). No study evaluated the direct benefits of screening vs no screening. Twenty-one studies (n = 7489) assessed the accuracy of 23 different screening tools that varied with regard to whether they were designed to be completed by parents vs trained examiners, and to screen for global (any) language problems vs specific skills (eg, expressive language). Three studies assessing parent-reported tools for expressive language skills found consistently high sensitivity (range, 88%-93%) and specificity (range, 88%-85%). The accuracy of other screening tools varied widely. Seventeen RCTs (n = 1517) evaluated interventions for speech and language delay or disorders, although none enrolled children identified by routine screening in primary care. Two RCTs evaluating relatively intensive parental group training interventions (11 sessions) found benefit for different measures of expressive language skills, and 1 evaluating a less intensive intervention (6 sessions) found no difference between groups for any outcome. Two RCTs (n = 76) evaluating the Lidcombe Program of Early Stuttering Intervention delivered by speech-language pathologists featuring parent training found a 2.3% to 3.0% lower proportion of syllables stuttered at 9 months compared with the control group when delivered in clinic and via telehealth, respectively. Evidence on other interventions was limited. No RCTs reported on the harms of interventions.

Conclusions and Relevance   No studies directly assessed the benefits and harms of screening. Some parent-reported screening tools for expressive language skills had reasonable accuracy for detecting expressive language delay. Group parent training programs for speech delay that provided at least 11 parental training sessions improved expressive language skills, and a stuttering intervention delivered by speech-language pathologists reduced stuttering frequency.

An estimated 8% of US children aged 3 to 17 years have a communication disorder. 1 Boys are almost twice as likely to be affected than girls (9.6% vs 5.7%,) and higher rates are observed among Black children (10%) compared with Hispanic (6.9%) or White (7.8%) children. 1 These data and other nationally representative prevalence estimates are limited in terms of distinguishing children who have a delay vs specific speech and/or language disorder.

A “delay” refers to development of speech and language in the correct sequence but at a slower rate than expected, whereas a “disorder” refers to development of speech and/or language ability that is qualitatively different from typical development. Speech disorders are characterized by difficulty with forming specific sounds or words correctly (articulation or phonological disorders) or making words or sentences flow smoothly (fluency disorders), and language disorders are characterized by difficulty understanding (receptive language) or speaking (expressive language) relative to their peers. 2 The focus of this review is routine screening for developmental (or “primary”) speech or language delay and disorders that are not caused by an injury or another condition (acquired or “secondary” disorders) such as hearing loss (eg, secondary to infection or genetic syndrome) or autism. Evaluation of children with known conditions that affect speech or language development would be part of disease management rather than screening; however, in the context of routine screening, some children who screen positive may go on to receive a primary diagnosis for a disorder such as hearing loss following a diagnostic evaluation.

Many children identified with speech or language delay go on to recover without an intervention. 3 However, observational evidence suggests that school-aged children with speech or language delay may be at increased risk of learning and literacy disabilities. 4 - 6 and social and behavioral problems, 7 some of which may persist through adulthood. 8 , 9 Screening for speech and language delay is distinct from overall developmental screening recommended by the American Academy of Pediatrics at 18 and 30 months. 10 Children who screen positive require referral for a diagnostic evaluation to confirm the suspected delay or disorder. Once a diagnosis is confirmed, treatment is variable and individualized to the needs of the child based on how the disorder impairs their function in different settings.

In 2015, the US Preventive Services Task Force (USPSTF) concluded that the evidence was insufficient to assess the balance of benefits and harms of screening for speech and language delay and disorders in children 5 years or younger (I statement). 11 The purpose of the current systematic review was to update the previous evidence review on the benefits and harms of screening for speech and language delay and disorders in children to inform the USPSTF in updating its recommendation.

Figure 1 shows the analytic framework and key questions (KQs) that guided the review. Detailed methods are available in the full evidence review. 12 In addition to the KQs, this review looked for evidence related to 3 contextual questions that focused on disparities in the prevalence, detection, and provision and utilization of treatment for speech and language delay or disorders among specific populations of children (eContextual Questions in the Supplement ).

PubMed/MEDLINE, the Cochrane Library, APA PsycInfo, ERIC, and Linguistic and Language Behavior Abstracts (ProQuest) were searched for English-language articles published through January 17, 2023 (eMethods in the Supplement ). ClinicalTrials.gov was searched for unpublished studies. The searches were supplemented by reviewing reference lists of pertinent articles, studies suggested by peer reviewers, and comments received during public commenting periods. From January 17, 2023, through November 24, 2023, ongoing surveillance was conducted through article alerts and targeted searches of journals to identify major studies published in the interim that may affect the conclusions or understanding of the evidence and the related USPSTF recommendation.

Two investigators independently reviewed titles, abstracts, and full-text articles using prespecified eligibility criteria (eTable 4 in Supplement ). Disagreements were resolved by discussion and consensus. For all KQs, English-language studies enrolling unselected children 5 years or younger from primary care or primary care–relevant settings (including childcare, schools, and other education settings) who communicate using any language were eligible. In addition, only studies set in countries categorized as “very high” on the Human Development Index 13 and rated as fair or good quality were included. For studies assessing the benefits and harms of interventions (KQ4, KQ5, and KQ6), those enrolling children referred for treatment or identified by educators or parents as having a possible speech or language problem, and those enrolling children up to age 6 years were also eligible.

For KQ2, studies assessing the accuracy of a screening instrument against a diagnosis reference standard (diagnostic interview, diagnostic questionnaire, or both) were included. Eligible screening instruments had to be feasible for use in primary care and included short questionnaires that could be delivered and interpreted in 10 minutes or less in clinical settings and longer questionnaires completed by parents or teachers outside of a scheduled visit. Studies focusing on the accuracy of general developmental screening tools that did not include a separate component for speech and language skills were excluded.

Randomized clinical trials (RCTs), nonrandomized clinical trials, and controlled cohort studies were eligible for KQ1 and KQ3 (benefit and harms of screening compared with no screening) and KQ6 (harms of interventions compared with an inactive control). For studies reporting on the benefit of interventions to improve speech and language outcomes (KQ4) or academic skills, behavior, function, or quality of life (KQ5), RCTs comparing an intervention with an inactive control were eligible. For KQ4, KQ5, and KQ6, eligible interventions included any treatment designed to improve speech and/or language delay or disorders among eligible populations, regardless of format (eg, individual or group settings, face-to-face, or via telehealth) or delivery personnel (eg, speech-language pathologists [SLPs] or other clinicians, parents, or teachers).

For each included study, 1 investigator extracted pertinent information about the methods, populations, interventions, comparators, outcomes, timing, settings, and study designs. All data extractions were checked by a second investigator for completeness and accuracy. For newly identified studies, 2 reviewers independently assessed each study’s methodological quality using predefined criteria developed by the USPSTF (eMethods in Supplement ) and informed by tools designed for various study designs (Cochrane Risk of Bias 2.0 tool for RCTs 14 ; Quality Assessment of Diagnostic Accuracy Studies 2 for screening test accuracy). 15 For eligible studies included in the previous update for this topic, quality ratings were spot-checked and carried forward. Disagreements were resolved by discussion.

Findings for each KQ were summarized in tabular and narrative format. The overall strength of the evidence for each KQ was assessed as high, moderate, low, or insufficient based on the overall quality of the studies, consistency of results between studies, precision of findings, risk of reporting bias, and limitations of the body of evidence using methods developed for the USPSTF (and the Evidence-based Practice Center program). 16 , 17 Additionally, the applicability of the findings to US primary care populations and settings was assessed. Discrepancies were resolved through consensus discussion.

For studies included for KQ2 (accuracy of screening tools), sensitivity, specificity, likelihood ratios, and predictive values were calculated based on data reported by articles, when sufficient, to compare consistency across similar measures. To determine whether meta-analyses were appropriate, the clinical heterogeneity and methodological heterogeneity of the studies were assessed following established guidance. 18 Due to heterogeneity in populations, outcome measures and other factors, as well as few studies assessing the same screening tool or interventions, meta-analysis was not appropriate.

A total of 38 studies (reported in 41 articles) were included ( Figure 2 ) in the review. Individual study quality ratings are reported in eTables 5 through 10 in the Supplement .

Key Question 1. Does screening for speech and language delay or disorders in children age 5 years or younger improve speech and language outcomes, school performance, function, or quality-of-life outcomes?

No eligible study addressed this question.

Key Question 2. What is the accuracy of screening tools to detect speech and language delay or disorders in children age 5 years or younger?

Twenty-one studies (reported in 23 articles) assessed the accuracy of 23 screening instruments for detecting speech and language delay and disorders in young children against a reference standard (n = 7489) ( Table 1 ). 19 - 41 Seven studies were new to this update. 24 , 27 , 30 - 32 , 39 , 41 Of the 23 instruments, 13 19 - 23 , 28 - 32 , 35 , 37 , 38 were designed to be administered to children by a trained examiner, and 10 23 - 27 , 33 - 36 , 39 - 41 were parent reports of children’s speech or language skills ( Table 2 ).

Some screening tools, termed global screening tools, screen for any language problems, while others provide scores for specific aspects of language (eg, expressive communication, receptive language, vocabulary). Twelve global screening tools were evaluated in the studies included the Ages and Stages Questionnaire (ASQ), 23 , 41 the Davis Observation Checklist for Texas, 19 the Developmental Nurse Screen, 35 the Early Language Scale, 39 the Fluharty Preschool Screening Test (FPST), 20 the General Language Screen, 36 the Hackney Early Language Screening Test/Structured Screening Test (HELST/SST), 28 , 29 the Infant-Toddler Checklist, 40 the Nurse Screening, 30 , 31 the Parent Questionnaire, 35 the Screening Kit of Language Development (SKOLD)/Screening Kit of Language Development Black English (SKOLDBE), 21 and the language component of the Sentence Repetition Screening Test (SRST). 38

Nine other tools provided scores for specific aspects of language, including the Brigance Preschool Screen, 23 the Early Screening Profiles, 23 the Battelle the Elternfragebogen für die Fruberkennung von Riskokindern (ELFRA-2), 33 , 34 the Sprachentwicklungsscreening (SPES-3) instrument, 24 the Language Development Survey (LDS), 25 , 26 the Quick Interactive Language Screener (QUILS), 32 the Sure Start Language Measure (SSLM), 41 the Northwestern Syntax Screening Test, 20 and the Battelle Developmental Inventory Screening Test–Communication. 23 Three of the trained examiner tools specifically screened for articulation skills—the Denver Articulation Screening Exam 22 and the articulation portion of both the Fluharty Preschool Speech and Language Screening Test (FPSLST) 37 and the SRST 38 —and 1 parent-administered instrument measured articulation. 27 The articulation instruments were considered separately from specific language instruments. All but 3 instruments (ie, ASQ, 23 , 41 HELST/SST, 28 , 29 and Nurse Screening 30 , 31 ) were examined in only 1 study each. In addition, 2 studies examined the FPST 20 and a later version with a language component, the FPSLST. 37

Excluding 2 studies 33 , 40 that enrolled all children who screened positive and a random sample of children who screened negative, the prevalence of speech and language disorders based on reference standards ranged from 4% to 33% ( Table 3 ).

As shown in Table 3 , the sensitivity of instruments for detecting speech and language disorders and delay ranged from 17% and 100% (median, 86%), and specificity ranged between 32% and 98% (median, 87%). To further examine accuracy, the source of the information (parent report vs trained examiner) and whether the instrument was designed as a global index of speech or language, a specific language skill (eg, word knowledge), or a measure of articulation were considered.

Sensitivity and specificity of 14 parent-reported tools varied widely ( Table 3 ). Sensitivity ranged from 55% to 93% (median, 84%) and specificity ranged from 32% to 96% (median, 84%).

Limiting analysis to global language instruments based on parent reports, median sensitivity was 74%, ranging between 55% and 89%. Specificity was less variable, ranging between 73% and 95% (median, 79%). In contrast, both sensitivity and specificity of the 3 parent-reported instruments of specific skills (all emerging expressive language skills) were fairly consistent and high (median sensitivity, 91% [range, 83%-93%]; median specificity, 88% [range, 81%-96%]). The 1 parent-rated measure of articulation had a reasonably high sensitivity (86%) but low specificity (32%).

The median sensitivity of the 13 screening tools that trained examiners administered to children was 87% (range, 17%-100%), and the median specificity was 88% (range, 58% to 98%). Similar to parent-reported instruments, there is substantial variability in the accuracy of examiner-administered tools.

Restricting the accuracy summary to trained examiner screenings of global language resulted in median sensitivity of 88% (range, 17%-100%) and median specificity of 89% (69%-98%). The median sensitivity of trained examiner instruments for specific language skills was 86% (range, 56%-94%) and median specificity was 70% (range, 58%-90%). Across the 3 trained examiner tools for assessing articulation, the median sensitivity was only 66% (range, 43%-92%); however, median specificity was 96% (range, 93%-97%).

Key Question 3. What are the harms of screening for speech and language delay or disorders in children age 5 years or younger?

Key Question 4. Do interventions for speech and language delay or disorders in children age 6 years or younger improve speech and language outcomes?

Seventeen RCTs (18 articles) compared an intervention for speech and language delay or disorders with an inactive control (no treatment or wait-list control/delayed treatment). 42 - 59 Study characteristics are shown in eTable 11 in the Supplement . No studies enrolled children identified by routine screening in primary care. Most recruited participants from referrals to speech and language treatment centers (6 studies), 42 , 47 , 49 , 50 , 53 , 54 schools or early childhood education centers (4 studies), 43 , 46 , 48 , 56 or via advertisements or a mix of advertisements and outreach to schools, clinical settings, or community-based programs. 44 , 45 , 55 , 57 The mean age of enrolled populations ranged from 18.1 months to 67.8 months, with most (10 studies) enrolling a sample with a mean age of 48 months or older. The proportion of participants who were female ranged from 10% to 49%. Few studies reported on race or ethnicity; in 3 studies set in the US, populations were described as 100% Latino, 45 100% White, 57 and 1 was inclusive of different groups (2% American Indian, 3% Asian, 2% Black, 26% Hispanic, 12% multiracial, 54% White). 48 Interventions evaluated were heterogeneous and varied in terms of the range of disorders targeted, delivery personnel, intensity/duration, settings, and other factors (eTable 11 in Supplement ).

Eight RCTs assessed interventions specific to children with delayed expressive language (“late talkers”) and no obvious fluency or speech-sound impairment. 44 , 45 , 50 - 52 , 56 - 59 Of these, 3 RCTs evaluated parent-group training interventions focused on strategies to promote their child’s language development; training approaches and specific content varied, but all focused on naturalistic strategies (eg, expanding on child utterances, following the child’s interests, repeating what the child says, setting up the environment to encourage communication). Of these, 2 RCTs assessed modifications of the Hanen Program for Parents curriculum (featuring a combination of group training sessions composed of a small group of parents and a trained SLP or other trained facilitator, and individual consultations with the SLP while the child is present), 51 , 58 and 1 evaluated a similar group training program focused on improving child linguistic complexity. 50 Results varied by duration of the intervention and mean age of enrolled populations. In 2 RCTs in which the intervention was delivered to children with a mean age of 27 to 30 months over a longer duration (11 bimonthly 60- to 75-minute sessions in one of the trials 50 and 11 weekly 2.5-hour sessions plus 3 weekly home visits in the other trial 51 ), there was consistent benefit across different measures of expressive language outcomes (eTable 12 in the Supplement ). The RCT delivering the parent group training to children with a mean age of 18 months over a shorter duration (6 weekly 2-hour sessions) found no significant difference between groups on any measure of receptive or expressive language outcomes. 58

Five other RCTs assessed different interventions for children with language delay and varied in terms of setting, delivery personnel, and other factors. 44 , 45 , 56 , 57 , 59 In general, results were inconsistent, with some studies showing improvement on some measures of receptive or expressive language but others not. Results are further summarized in the eResults and eTable 12 in the Supplement .

Two RCTs assessed fluency treatment for young children. Both focused on the Lidcombe Program of Early Stuttering Intervention. 54 , 55 This intervention is led by an SLP who trains parents to provide verbal contingencies for stutter-free speech (eg, “that was smooth talking”) and stuttering (eg, “that was a bit bumpy”) and requests for self-evaluation and self-correction (eg, “can you say that again”). In one of these RCTs, the intervention was delivered in a face-to-face format in a clinical setting 54 and in the other it was delivered via telehealth. 55 Results were consistent in showing a statistically significant improvement in stuttering fluency associated with the intervention. In the face-to-face intervention, children in the intervention group had a 2.3% (95% CI, 0.8-3.9) lower proportion of syllables stuttered than children in the control group at 9 months. Per the authors, this is above the minimum clinically important difference of 1.0% of syllables stuttered (the minimum difference that a listener would be able to distinguish). 54 However, no reference or clear rationale was provided to support this threshold. In the RCT using telehealth delivery of the intervention, the difference between the intervention and control group in change from baseline mean number of syllables stuttered was −3.0% ( P  = .02) at 9 months. 55

Evidence on other intervention types targeting specific speech or language problems was limited and is further described in the eResults in the Supplement .

Key Question 5. Do interventions for speech and language delay or disorders in children age 6 years or younger improve school performance, function, or quality-of-life outcomes?

Eight RCTs reported on 1 or more outcomes specific to school performance, function, or quality of life using heterogeneous measures. 42 , 43 , 47 , 48 , 53 , 57 - 59 Characteristics are described above in KQ4 and detailed results are shown in eTable 15 in the Supplement . No RCTs assessing a similar intervention type reported on the same outcome domain, and most studies reporting on similar domains (eg, early literacy) used different outcome measures. In 4 RCTs reporting on a measure of early or emergent literacy skills, 3 found no significant difference between groups. 42 , 43 , 48 In contrast, 1 RCT assessing a home-based language delay intervention delivered by trained assistants found benefit for improving letter knowledge associated with the intervention. 59 Two RCTs reported on 1 or more measures of functional communication 42 , 47 and quality of life/well-being in children 43 , 53 and found no difference between groups, while 1 RCT evaluating an individual intervention for language delay found significant improvement favoring the intervention for improving child socialization skills and parental stress levels. 57

Key Question 6. What are the harms of interventions for speech and language delay or disorders?

This systematic review synthesized evidence relevant to screening for speech and language delay or disorders in children 5 years or younger. Table 4 summarizes the main findings of the evidence review. There was no direct evidence on the benefits and harms of screening (KQ1). Potential harms of screening (KQ3) include false-positive results that can lead to unnecessary referrals (and the associated time and economic burden), labeling or stigma, parent anxiety, and other psychosocial harms. Other harms of screening are likely to be minimal because screening is noninvasive.

The studies of screening test accuracy (KQ2) included in this review assessed 23 different tools that varied in terms of whether they were completed by parents vs trained examiners and whether they were designed to detect global speech or language problems vs problems related to specific language skills or articulation. Some screening tools usable in clinical practice may identify children who have a speech or language disorder with reasonable sensitivity and specificity. However, overall evidence was mixed and few screening tools were assessed by more than 1 study each, limiting the ability to make stronger conclusions about the accuracy of specific tools. Parent-reported screening instruments designed to assess expressive language skills displayed consistently high sensitivity and specificity, although precision varied by instrument. In contrast, the accuracy of the parent-reported instruments for global language skill assessment was inconsistent, and precision varied across instruments. The accuracy of examiner-administered screening instruments varied, particularly for instruments designed to assess specific language skills.

Few studies of interventions for speech and language delay or disorder enrolled similar populations and evaluated similar types of interventions (KQ4). Although 2 RCTs of treatment enrolled children newly referred from primary care, it is not clear whether the children were identified via routine screening vs case finding. Other included studies enrolled children referred or recruited via advertisements, and most focused on a specific type of speech delay or disorder. Given these factors, the body of evidence on treatment available for inclusion in this review may not be applicable to the type and severity of disorders that would be detected via routine screening in primary care settings.

Studies of children referred for language delay without obvious speech-sound or fluency disorder suggested that group training interventions offering at least 11 parent training sessions improved expressive language outcomes. For children identified with stuttering, the Lidcombe Program of Early Stuttering Intervention delivered by SLPs improved stuttering fluency at 9 months when delivered either in person or via telehealth. Although 8 RCTs reported on 1 or more outcomes specific to school performance or early literacy, health-related quality of life, function, behavior, or socialization (KQ5), the interventions and populations evaluated were heterogeneous, which limited the ability to assess consistency; most studies found no difference between groups for measures of early literacy, function, and quality of life. However, most trials may not have followed up children for a long enough duration to detect an improvement in quality of life or function that could result from early treatment of a speech and language delay or disorder. No RCTs reported on the harms of interventions; however, given the nature of the interventions, serious harms are unlikely.

Trials are needed that enroll asymptomatic or unselected populations from general primary care settings and directly assess the benefit of screening specifically for speech and language problems. The control groups in these trials could receive either no screening or routine screening for general developmental delay, with no separate score for speech and language problems. Studies are also needed on the potential harms of screening, such as labeling, and harms from false-positive results, such as burden on parents due to unnecessary referrals. Such studies would also inform the potential for overdiagnosis associated with routine screening, given that many children who have a speech delay may recover without intervention. 3

Similarly, studies assessing the accuracy of screening tools among unselected populations, who are ideally recruited through primary care settings, are needed because the prevalence of speech and language problems may vary compared with populations enrolled via advertisements or specialty settings. Specifically, studies that assess the accuracy of existing tools, compared with similar reference standards, would help determine the consistency of findings; because few included studies evaluated the same instrument, our ability to make a strong conclusion about accuracy was limited. Trials of treatment enrolling populations recruited from US primary care settings would help inform the potential benefit of screening because the range of severity and conditions is likely different compared with trials that enroll referred populations. Last, studies that follow up children for a sufficiently long duration to detect improvement in academic performance, function, and quality of life would help in the understanding of whether immediate changes in speech and language outcomes (eg, short-term expansion of vocabulary words) translate into benefit for health and social outcomes.

This review excluded studies in children who had a condition known to cause a speech or language problem (eg, hearing loss, autism) to improve the applicability of evidence to populations likely to be detected by routine screening. Studies evaluating primary prevention strategies to promote speech and language development (eg, interventions among groups considered “at risk” or school-based curricula emphasizing language development among children with no developmental delay or disorder) were also excluded. The aim was to limit the review to interventions that are relevant to children with screen-detected speech and language problems and that are appropriate to deliver in primary care settings or refer to from primary care.

This review found no eligible studies that reported on direct benefits or harms of screening compared with usual care or no screening. Parent-reported screening tools for expressive language delay had reasonable accuracy. In contrast, parent-reported screening tools for global language delay had inconsistent accuracy. The accuracy of examiner-administered instruments was also variable, especially for examiner-administered instruments of specific language skills. Existing evidence on treatment of speech and language delay is available from referral populations but not from screen-detected populations. This evidence indicates the benefit from group parent-training programs for speech delay that provide at least 11 parental training sessions for improving expressive language skills, as well as the Lidcombe Program of Early Stuttering Intervention delivered by SLPs for reducing stuttering frequency. Few studies reported on outcomes specific to school performance, function, quality of life, or behavior, and none reported on the harms of interventions.

Accepted for Publication: December 2, 2023.

Corresponding Author: Cynthia Feltner, MD, MPH, Cecil G. Sheps Center for Health Services Research, University of North Carolina at Chapel Hill, 725 Martin Luther King Jr Blvd, CB#7295, Chapel Hill, NC 27599 ( [email protected] ).

Author Contributions: Dr. Feltner had full access to all of the data in the study and takes responsibility for the integrity of the data and the accuracy of the data analysis.

Concept and design: Feltner, Wallace, Nowell, Raffa, Kahwati.

Acquisition, analysis, or interpretation of data: All authors.

Drafting of the manuscript: Feltner, Wallace, Nowell, Raffa, Middleton, Vaughan, Baker, Kahwati.

Critical review of the manuscript for important intellectual content: Feltner, Wallace, Nowell, Orr, Chou, Kahwati.

Statistical analysis: Feltner, Orr.

Obtained funding: Feltner, Chou, Kahwati.

Administrative, technical, or material support: Orr, Middleton, Vaughan, Baker.

Supervision: Feltner.

Conflict of Interest Disclosures: None reported.

Funding/Support: This research was funded under contract 75Q80120D00006, Task Order 75Q80121F32009, from the Agency for Healthcare Research and Quality (AHRQ), US Department of Health and Human Services, under a contract to support the US Preventive Services Task Force (USPSTF).

Role of the Funder/Sponsor: Investigators worked with USPSTF members and AHRQ staff to develop the scope, analytic framework, and key questions for this review. AHRQ had no role in study selection, quality assessment, or synthesis. AHRQ staff provided project oversight, reviewed the evidence review to ensure that the analysis met methodological standards, and distributed the draft for peer review. Otherwise, AHRQ had no role in the conduct of the study; collection, management, analysis, and interpretation of the data; and preparation, review, or approval of the manuscript findings. The opinions expressed in this document are those of the authors and do not reflect the official position of AHRQ or the US Department of Health and Human Services.

Additional Contributions: We gratefully acknowledge the following individuals for their contributions to this project, including AHRQ staff (Justin Mills, MD, MPH; Tracy Wolff, MD, MPH), Scientific Resource Center for the AHRQ Evidence-based Practice Center Program staff (Robin A. Paynter, MLIS), Pacific Northwest Evidence-based Practice Center staff (Christina Bougatsos, MPH), and RTI International–University of North Carolina–Chapel Hill Evidence-based Practice Center staff (Manny Schwimmer, MPH; Christiane E. Voisin, MSLS; Roberta Wines, MPH; Mary Gendron; Sharon Barrell, MA; Alexander Cone; Teyonna Downing; Michelle Bogus). The USPSTF members, expert reviewers, and federal partner reviewers did not receive financial compensation for their contributions. Evidence-based Practice Center personnel received compensation for their roles in this project.

Additional Information: A draft version of the full evidence review underwent external peer review from 3 content experts (Abigail D. Delehanty, PhD, CCC-SLP, Duquesne University; Virginia Moyer, MD, MPH, University of North Carolina at Chapel Hill; Thelma E. Uzonyi, PhD, CCC-SLP, IMH-E, Kennedy Krieger Institute) and 3 federal partner reviewers (Centers for Disease Control and Prevention; Eunice Kennedy Shriver National Institute of Child Health and Human Development; and National Institute on Deafness and Other Communication Disorders). Comments from reviewers were presented to the USPSTF during its deliberation of the evidence and were considered in preparing the final evidence review. USPSTF members and peer reviewers did not receive financial compensation for their contributions.

Editorial Disclaimer: This evidence review is presented as a document in support of the accompanying USPSTF Recommendation Statement. It did not undergo additional peer review after submission to JAMA .

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Tasman’s Psychiatry pp 1–27 Cite as

Neurodevelopmental Disorders: Speech and Language Disorders

• Michelle L. Palumbo 10 ,
• Maria Mody 11 ,
• William M. Klykylo 12 ,
• Kirrie J. Ballard 13 ,
• Christopher J. McDougle 10 &
• Frank H. Guenther 14  
• Living reference work entry
• First Online: 11 August 2023

46 Accesses

Psychiatric practice is founded upon communication, and knowledge of communication disorders is thus crucial to the psychiatric team. This is especially true for the care of children, since communication impairments are deeply interwoven in all aspects of normal development, psychopathology, and the functions of daily life. Neurodevelopmental communication disorders are classified according to the affected system (speech, language) as well as behavioral manifestations. Language disorder is characterized by lasting difficulties in language acquisition and use as a result of deficits in the production or understanding of language. Speech sound disorder (SSD) is characterized by failure to use speech sounds in a manner appropriate for one’s developmental level. Childhood-onset fluency disorder (COFD), commonly known as stuttering, is one of the most widely recognized disorders of speech and is characterized by interruptions in the normal flow of speech, including blocks, prolongations, and repetitions of words or part-words. Social (pragmatic) communication disorder (SCD) involves impaired pragmatic or social aspects of language, such as inferring humor or sarcasm during conversations or interpreting body language. All of these disorders must develop during childhood and must not be fully explainable by another medical, neurological, anatomical, psychiatric, or sensory condition. The communication disorders as a whole have a male predominance and are often familial. The evaluation and management of these disorders requires a multidisciplinary team, including a speech-language pathologist working with the psychiatrist and other team members.

This chapter is an update from the 4th edition. Previous edition authors were Michelle L. Palumbo, Maria Mody, William M. Klykylo, Christopher J. McDougle and Frank H. Guenther

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Departments of Pediatrics and Psychiatry, The Lurie Center for Autism, Massachusetts General Hospital for Children, Lexington, MA, USA

Michelle L. Palumbo & Christopher J. McDougle

Department of Radiology, MGH/MIT/HMS Athinoula A. Martinos Center for Biomedical Imaging, Charlestown, MA, USA

Department of Psychiatry, Wright State University Boonshoft School of Medicine, Dayton, OH, USA

William M. Klykylo

Sydney School of Health Sciences in the Faculty of Medicine and Health and the Brain and Mind Centre, The University of Sydney, Camperdown, NSW, Australia

Kirrie J. Ballard

Departments of Speech, Language, & Hearing Sciences and Biomedical Engineering, Boston University, Boston, MA, USA

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Renato D. Alarcón

Columbia University Medical Center, New York, USA

César A. Alfonso

Kyushu University, Fukuoka, Japan

Shigenobu Kanba

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Malaysian Research Inst on Ageing, Universiti Putra Malaysia, Selangor, Malaysia

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Palumbo, M.L., Mody, M., Klykylo, W.M., Ballard, K.J., McDougle, C.J., Guenther, F.H. (2023). Neurodevelopmental Disorders: Speech and Language Disorders. In: Tasman, A., et al. Tasman’s Psychiatry. Springer, Cham. https://doi.org/10.1007/978-3-030-42825-9_86-1

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Estimates of the prevalence of speech and motor speech disorders in persons with complex neurodevelopmental disorders

Lawrence d. shriberg.

a Intellectual and Developmental Disabilities Research Center, Waisman Center, University of Wisconsin-Madison, Madison, WI, USA

Edythe A. Strand

b Department of Neurology, Mayo Clinic, Rochester, MN, USA

Kathy J. Jakielski

c Department of Communication Sciences and Disorders, Augustana College, Rock Island, IL, USA

Heather L. Mabie

Associated data.

Estimates of the prevalence of speech and motor speech disorders in persons with complex neurodevelopmental disorders (CND) can inform research in the biobehavioural origins and treatment of CND. The goal of this research was to use measures and analytics in a diagnostic classification system to estimate the prevalence of speech and motor speech disorders in convenience samples of speakers with one of eight types of CND. Audio-recorded conversational speech samples from 346 participants with one of eight types of CND were obtained from a database of participants recruited for genetic and behavioural studies of speech sound disorders (i.e., excluding dysfluency) during the past three decades. Data reduction methods for the speech samples included narrow phonetic transcription, prosody-voice coding, and acoustic analyses. Standardized measures were used to cross-classify participants’ speech and motor speech status. Compared to the 17.8% prevalence of four types of motor speech disorders reported in a study of 415 participants with idiopathic Speech Delay (SD), 47.7% of the present participants with CND met criteria for one of four motor speech disorders, including Speech Motor Delay (25.1%), Childhood Dysarthria (13.3%), Childhood Apraxia of Speech (4.3%), and concurrent Childhood Dysarthria and Childhood Apraxia of Speech (4.9%). Findings are interpreted to indicate a substantial prevalence of speech disorders, and notably, a substantial prevalence of motor speech disorders in persons with some types of CND. We suggest that diagnostic classification information from standardized motor speech assessment protocols can contribute to research in the pathobiologies of CND.

Speech is one of five heritable verbal traits (speech, language, reading, writing, spelling) in which some children have developmental delays or disorders ( Shriberg, Lohmeier, Strand, & Jakielski, 2012 ; Truong et al., 2016 ). As with the other verbal traits, speech delays and disorders occur in both idiopathic contexts, and in the context of complex neurodevelopmental disorders (CND). The term complex in the latter classification is used to include environmental contributions to neurodevelopmental disorders. The goal of the present study was to estimate the prevalence of speech disorders and the prevalence of motor speech disorders in a database of audio-recorded speech samples from speakers with one of eight types of CND. The following is an overview of classification terms and concepts used in this paper.

Classification terms and concepts in childhood speech sound disorders

Although there is international consensus on Speech Sound Disorders ( SSD ) as the cover term for childhood ( paediatric in medical contexts) speech and motor speech disorders (excluding stuttering), there is currently no consensus on standardized measures and a classification system to identify and quantify the severity of types of SSD ( Bernthal, Bankson, & Flipsen, 2017 ; Bowen, 2015 ; McLeod & Baker, 2017 ; Rvachew, 2015 ; Rvachew & Brosseau-Lapré, 2012 ; Waring & Knight, 2013 ). A comparative analysis of measures and classification systems in SSD is beyond the focus of the present report. It is useful, however, to describe four dichotomies that are addressed in classification proposals for SSD that are central to discussions of the prevalence estimates based on findings described in the present study.

Idiopathic SSD and SSD in CND

As shown in Figure 1 , the primary dichotomy in classification systems for childhood SSD is the division introduced – SSD in children with no known developmental involvements and SSD in children with disorders affecting cognitive, structural, sensory, motor, and/or affective development. Issues in and alternative perspectives to this categorical rather than dimensional classification of SSD in Figure 1 have been described for speech pathology (e.g., Morgan & Liégeois, 2010 ; Weismer, 2006 ) and in other literatures (e.g., Beglinger & Smith, 2001 ). In introductory textbooks in SSD, the primary focus is on idiopathic SSD, with disorders that have SSD in the context of CND typically sampled in chapters on ‘special populations’ (e.g., children with hearing disorders, children with craniofacial disorders, children with autism spectrum disorders). Unlike the idiopathic SSD literature, many CND in which speakers are at increased risk for speech disorder have their own research and clinical journals, professional associations, clinical specialists, and advocacy groups. This research, educational, and clinical separation of the two contexts for SSD is a scientific constraint on research addressing common biobehavioural questions.

Four classification dichotomies in Speech Sound Disorders (SSD).

Speech errors and speech delay

A second dichotomy shown in Figure 1 is the division of speakers with SSD in each context into two classifications of speech disorders. Some speakers’ speech errors consist solely of common clinical distortions of speech sounds in challenging phonetic feature classes, such as in American English, distortions of the sibilant consonant in ‘ s ee’ (sometimes referred to as a lisp ) or distortions of the rhotic consonant in ‘ r ay.’ Other speakers, in addition to distorting some speech sounds, have age-inappropriate deletions of speech sounds and/or substitutions of speech sounds for one another. Unlike speakers with only speech sound distortions, speakers with age-inappropriate speech sound deletions and/or substitutions are at risk for delays and disorders in the four other verbal traits listed previously: language, reading, writing and spelling ( Peterson, Pennington, Shriberg, & Boada, 2009 ). Conventional classification terms for this dichotomy, respectively, are articulation disorder and phonological disorder ( Bowen, 2015 ). As defined presently, the classification system in this paper uses the terms Speech Errors (SE) for articulation disorder (only distortions), and Speech Delay (SD) for phonological disorder (deletions/substitutions and optionally distortions). Point-prevalence estimates using convenience and population-based samples of adults average 1.5–2% SE ( Flipsen, 2015 ); population-based estimates of the prevalence of SD at 4–8 years in three countries average 3.6% ( Eadie et al., 2015 ; Shriberg, Tomblin, & McSweeny, 1999 ; Wren, Miller, Peters, Emond, & Roulstone, 2016 ). As indicated previously, estimates of the prevalence of speech and motor speech disorders in CND are fractionated, with no research to date using the same or comparable methods, measures, and classification system with a representative sample of speakers with CND ( Shriberg et al., 2010a , 2010b ).

Normalization and persistence of SE and SD

As shown in Figure 1 , a third SSD classification dichotomy differentiates speech disorders that normalize with or without treatment during the speech acquisition period from those that persist beyond the well-documented stages of speech development. The classification system to be described classifies speakers with SE (i.e., only speech sound distortions) past 9 years of age at assessment as having Persistent Speech Errors (PSE) and speakers with SD (i.e., speech sound deletions and/or substitutions and optionally distortions) past 9 years of age at assessment as having Persistent Speech Delay (PSD). As shown in Figure 1 and used in the present research, it is useful for many purposes to aggregate speakers younger and older than 9 years at assessment within the same research group or cohort (i.e., SE/PSE and SD/PSD).

Speech disorder and motor speech disorder

A fourth classification dichotomy in SSD, the primary focus of the present paper, addresses the hypothesis that SD and particularly PSD in some speakers may be associated with delays in neuromotor development. Processing deficits in neurocognitive domains are widely studied in idiopathic SD and other verbal trait disorders, whereas research in processing deficits in neuromotor domains in children with idiopathic speech-language deficits is less well-developed. In addition to the possibility of Childhood Apraxia of Speech (CAS; a neuromotor deficit in speech planning/programming) or Childhood Dysarthria (CD; a neuromotor deficit in speech execution), there is continuing research on the hypothesis of a third motor speech classification characterized by a delay in the development of precise and stable articulation that does not meet criteria for dysarthria or apraxia of speech (e.g., Bishop, 2002 ; Bradford, Murdoch, Thompson, & Stokes, 1997 ; Gaines & Missiuna, 2007 ; Goffman, 1999 ; Hill, 2001 ; Newmeyer et al., 2007 ; Rechetnikov & Maitra, 2009 ; Redle et al., 2015 ; Shriberg et al., 2010a ; Vick et al., 2014 ; Visscher, Houwen, Scherder, Moolenaar, & Hartman, 2007 ; Zwicker, Missiuna, & Boyd, 2009 ). As shown in Figure 1 and described presently, Speech Motor Delay (SMD) has recently been proposed as a classification term for children proposed to have this third type of developmental deficit in speech motor processes ( Shriberg, 2017 ; Shriberg, Kwiatkowski, & Mabie, 2019 ).

Neurocognitive and neuromotor processes in speech and motor speech disorders

The classification entities in Figure 2 are part of a research framework termed the Speech Disorders Classification System (SDCS; Shriberg, 2010a , 2010b ; Shriberg, Kwiatkowski, & Mabie, 2019 ). As shown in the top section of Figure 2 , distal substrates of speech and motor speech disorders include genomic, neurodevelopmental, and environmental risk and protective factors. Proximal substrates in the second section are divided into three speech processing domains – Representation, Transcoding, and Execution – each of which are mediated by feedforward and feedback processes. This generic sketch is based on many contemporary speech processing perspectives (e.g., Friederici, 2012 ; Guenther & Vladusich, 2012 ; Hickok& Poeppel, 2004 ; Nijland, Maassen, & van der Meulen, 2003 ; Terband, Maassen, Guenther, & Brumberg, 2014 ; van der Merwe, 2009 ; Ziegler & Ackermann, 2013 ; Ziegler, Aichert, & Staiger, 2012 ). The two speech disorders in the third section and reviewed previously, SD and SE, are presumed to be due to delays in auditory and somatosensory representational processes (e.g., Perkell, 2012 ; Terband et al., 2014 ). A deficit in Transcoding, a cover term for planning and/or programming speech movements, is generally proposed as the speech processing deficit in CAS ( Shriberg et al., 2017 ; van der Merwe, 2009 ). Execution deficits, including deficits in the spatiotemporal movements in speech, prosody, and voice, are proposed to underlie CD as well as the recently proposed SMD. The term ‘delay’ in the latter classification is supported by findings indicating high early normalization rates in children with concurrent idiopathic SD ( Shriberg, Campbell, Mabie, & McGlothlin, 2019 ). The fourth section in Figure 2 includes the behavioural markers currently used to identify the subtypes of SE and Motor Speech Disorder (MSD) shown above each sign or measure. The Method section and a Supplement for each of the papers in this research series include additional information on classification methods and measures.

The Speech Disorders Classification System (SDCS).

Statement of purpose

Estimates of the prevalence of speech disorders and motor speech disorders in speakers with different CND are not presently available in research using the same methods, measures, and classification system. The goal of the present research was to obtain initial estimates of the prevalence of speech disorders and motor speech disorders in samples of persons with CND in a database of audio-recorded conversational speech samples from 346 speakers with one of eight types of CND. The hypothesis is that using similar methods, measures, and cross-classification system, estimates of the prevalence of motor speech disorders in persons with some types of CND will be significantly higher than recent estimates of the prevalence of motor speech disorders in children with idiopathic SD.

Participants

Table 1 includes summary assessment, demographic, and cognitive-language information for participants in convenience samples of eight types of CND. Participants were recruited during the past three decades in research with investigators in several USA cities. All participants were assented and/or consented using procedures and forms approved by institutional review boards at the University of Wisconsin-Madison, the Mayo Clinic-Rochester, Minnesota, and research and clinical institutions in multiple cities where the participants were recruited and assessed. Appendix 1 includes the inclusionary criteria for participation in each of the eight study samples and a brief description of participants. As described presently, each of the 346 audio-recorded conversational samples that comprise the database for the present study were obtained using the same or comparable recording instrumentation, recording procedures, conversational speech sampling protocol, data reduction protocol and data reduction software. Data reduction for all speech samples from all groups was completed by the same group of research specialists.

Information for participants with one of eight types of Complex Neurodevelopmental Disorders (ordered alphabetically). a

As shown in the first four columns in Table 1 , only findings for participants who met SDCS criteria for classification of their motor speech status were included in the present study. A later section describes criteria for classifiable samples. The group-wise percentage of classifiable participants in the eight CND samples ranged from 88.5% to 100%, averaging 95.6%. The chronological ages of participants averaged 13.3 years and the male:female ratios across groups averaged 1.8:1. Participants in the five groups with available data had average standardized scores that were significantly lower than their typically-developing same-sexed age-matched peers on measures of cognition ( M = 70.0; SD = 11.0) and language ( M = 69.4; SD = 10.6).

Speech and motor speech classification

Cross-classification.

Cross-classification of the speech and motor speech status of each participant was completed using an analytic termed the Speech Disorders Classification System Summary (SDCSS; Mabie & Shriberg, 2017 ). Figure 3 includes sample SDCSS outputs from the computer software termed Programs to Examine Phonetic and Phonologic Evaluation Records ( PEPPER, 2019 ).

Sample outputs from the Speech Disorders Classification System Summary (SDCSS).

The SDCSS cross-classifies an individual speaker’s (upper panel) or a group of speakers’ (lower panel) speech and motor speech status using speech, prosody, and voice data obtained from a conversational speech sample and standardization reference data from 200 typically-developing speakers ( Potter et al., 2012 ; Scheer-Cohen et al., 2013 ). SDCSS findings provided the primary prevalence information for the present research. It is efficient first to describe the principal elements of the SDCSS, followed by description of the measures used to identify each of the speech and motor speech classifications in Figure 3 .

The rows in the SDCSS include the speech disorder classifications discussed previously, arranged vertically in order of increasing severity of involvement. The software classifies a speaker as having Normal (or Normalized) Speech Acquisition (NSA) if the speaker does not have in their conversational speech at assessment, any speech sound deletions, substitutions, or distortions (including additions) that are inappropriate for their age. SE, or PSE if the speaker is older than 9 years, is the classification assigned to speakers with age-inappropriate speech sound distortions. SD, or Persistent PSD if the speaker is older than 9 years, is the classification assigned to speakers with age-inappropriate speech sound deletions or substitutions. As described previously ( Figure 1 ), a slash convention is used to aggregate speakers younger and older than 9 years of age with the same class of speech disorder (i.e., SE/PSE and SD/PSD).

The columns in Figure 3 include five motor speech classifications arranged left to right in presumed order of increasing severity of involvement: No Motor Speech Disorder (No MSD), SMD, CD, CAS and concurrent CD & CAS. The purpose of the concurrent classification, CD & CAS, is to acknowledge the genomic, neuropathological, and clinical correlates of deficits in both neuromotor planning/programming (apraxia) and neuromotor execution (dysarthria) phases of speech production. Examples and discussion of the neurogenetic implications of concurrent apraxia and dysarthria compared to apraxia alone are increasingly emergent in the FOXP2 and other speech-genetics literatures (e.g., Carrigg, Parry, Baker, Shriberg, & Ballard, 2016 ; Chilosi et al., 2015 ; Eising et al., 2018 ; Morgan, Fisher, Scheffer, & Hildebrand, 2016 ; Rice et al., 2012 ; Shriberg, Strand, & Mabie, 2016 ; Turner, Morgan, Perez, & Scheffer, 2015 ; Worthey et al., 2013 ). The ‘X’ in the upper sample SDCSS in Figure 3 cross-classifies a speaker’s speech and motor speech status – in the present example, the 18-year-old daughter (“T”) of a woman (“B”) in a case study of a family with persistent CAS associated with a mutation in FOXP2 ( Shriberg et al., 2006 ). As shown, this participant is cross-classified as having PSD (speech axis) and CD & CAS (motor speech axis). In the lower sample SDCSS in Figure 3 , the percentages in the cells and marginal totals are group-wise findings from 28 children with CAS, classified by consensus using two different diagnostic procedures for CAS ( Shriberg & Strand, 2018 ). As shown, concurrent CD & CAS was approximately as prevalent as CAS alone in this sample of children recruited for CAS.

Classification conventions

The SDCSS is a phenotype analytic in which the five classifications within speech disorders and the five classifications within motor speech disorders are each mutually exclusive. Therefore, as shown in the example of grouped SDCSS data in Figure 3 , the marginal values for each axis total 100%. The five mutually exclusive classifications are accomplished using two conventions.

First, in addition to identifying speakers with no speech errors, the NSA classification identifies speakers with age-appropriate deletions, substitutions, and/or distortions (speakers with such behaviours in conversational speech are coded NSA-in the PEPPER software; Shriberg, 1993 , Appendix). Thus, NSA classifications include speakers with no speech errors, and is also the default classification for participants whose number and/or type of speech errors do not meet criteria for SE (or PSE) or for SD (or PSD).

Second, the SDCSS software classifies speakers who meet criteria for more than one of the speech disorders or the motor speech disorders as having the more severe disorder. Thus, a speaker who meets criteria for both SE and SD (or PSE and PSD if older than 9 years) is classified as SD or PSD (i.e., the more severe of the two types of speech disorders). Similarly, for speakers who meet criteria for both SMD and any of the other three motor speech disorders classifications (CD, CAS, or CD & CAS), the program classifies the speaker as having the other presumably more severe motor speech disorder.

Measures and classification procedures

The assessment protocols varied somewhat for each of the eight participant groups in Table 1 , with participants in some of the groups receiving more extensive original assessments. As described, each of the eight study samples included a conversational sample using comparable interactional questions and responses to encourage participants to talk about their daily activities ( Shriberg & Kwiatkowski, 1985 ). Appendix 2 includes descriptions of the five speech and motor speech classifications, and for participants meeting classification criteria for CD, the five dysarthria subtypes. SDCS classifications are completed by software that provides standardized speech, prosody, and voice measurement (z-scores) using two reference databases of typical speakers 3 to 80 years of age ( Potter et al., 2012 ; Scheer-Cohen et al., 2013 ). The speech classifications were made using a program that has been used for previous classification research in speech disorders ( Shriberg, 1993 , Appendix Table A; Shriberg, Austin et al., 1997 , Appendix A). The motor speech classifications in the present Appendix 2 were developed in research to identify genomic and phenotypic substrates of childhood speech sound disorders of known and unknown origin. The perceptual and acoustic signs of dysarthria and dysarthria subtypes were based on operationalized adaptations of Duffy’s (2013) diagnostic signs of neuro-genic motor speech disorders. A Supplement for this research series includes detailed information on classification methods [ Supplementary Data ]. Several reports provide information on the development and validation of the measures and normative reference data ( Mabie & Shriberg, 2017 ; Shriberg, 2017 ; Shriberg et al., 2009 ; Shriberg & Mabie, 2017 ; Shriberg et al., 2017 ; Tilkens et al., 2017 ).

Data from some of the original participants had to be excluded from the present research due to missing information on one of the measures needed to classify their motor speech status. As shown in Table 1 , the motor speech status of 4.4% of the original CND participants could not be classified (100% – 95.6% classifiable). The primary reason motor speech status could not be classified was because the conversational speech sample did not include the minimum of 40 pause opportunities needed to compute a score on the measure used to identify CAS (termed the Pause Marker; see Supplement ). In clinical practice, such children are typically resampled on the same or another day to obtain a sufficient number of pause opportunities, but such information was not available for participants in the database. The other reason some samples could not be classified was that a participant had an indeterminate Pause Marker score that could not be resolved using the Supplementary Pause Marker Index (see Supplement ). Resolution of indeterminate Pause Marker scores requires information from a nonword repetition task ( Shriberg et al., 2009 ) that was not yet available for research at the time some of the participants in the eight CND groups were assessed.

Data reduction and reliability estimates

Four research specialists completed transcription, prosody-voice coding, and acoustic analyses of the conversational speech samples and transcription of the nonword repetition task. For estimates of intrajudge reliability, each specialist completed a second analysis of the samples she had originally reduced. For estimates of interjudge reliability, each specialist completed an approximately equal number of samples completed by one of the other specialists.

Estimates of the interjudge and intrajudge reliability for all data reduction tasks were based on approximately 20% samples of participants in the four CND groups with the highest prevalence of motor speech disorder (see Figure 6 ). A total of 34 randomly selected conversational speech samples included 10 samples from participants with Down syndrome, 4 samples from participants with 22q11.2 deletion syndrome, 10 samples from participants with Idiopathic Intellectual Disability, and 10 samples from participants with fragile X syndrome.

Percentage of participants in eight Complex Neurodevelopmental Disorders classified into one of five motor speech classifications.

Table 2 is a summary of point-to-point interjudge and intrajudge agreement percentages for the three types of data reduction. The findings for the four CND groups were group-averaged (i.e., the averaged percentage of agreement findings for each of the four groups were divided by four). As shown in the reliability estimates in Table 2 , interjudge and intrajudge percentages of agreement were similar to the approximately mid-70% to mid-90% ranges reported in reviews of reliability findings in the speech sound disorders literature ( McSweeny & Shriberg, 1995 ; Shriberg et al., 2010b ; Shriberg & Lof, 1991 ). The average reliabilities in the mid-80% for variables assessed using transcription, prosody-voice coding, and acoustics systems within each of the four CND groups with significant motor speech disorders is viewed as particularly positive given the diverse perceptual and acoustic signs of deficits in speech, prosody, and voice assessed in the measures shown in the Supplement .

Reliability estimates for phonetic transcription, prosody-voice coding, and acoustic analyses.

Statistical analyses

Statistical findings are primarily descriptive, with some inferential statistics completed to guide discussion and interpretation of findings. To minimize Type II errors in these initial prevalence comparisons, many based on relatively small cell sizes, the number of inferential statistical tests were minimized and treated family-wise (see Feise, 2002 ; Nakagawa, 2004 ; Perneger, 1998 ).

Results and discussion

Figure 4 includes the cross-classification findings for the prevalence of speech and motor speech disorders in the 346 participants in the eight CND groups. The summary cross-classification finding derived from the data in Figure 4 warrants comment before examining the individual data for each of the eight CND. As shown in the upper left data cell, 37.3% (129/346) of the participants were cross-classified as NSA and No MSD at assessment. Thus, by subtraction, a total of 62.7% (217/346) of participants in the eight CND – over 60% of participants – had a speech and/or a motor speech disorder at assessment. The following sections report prevalence findings and discuss implications of findings for each type of speech and motor speech classification ( Figure 2 ) for participants in each of the eight CND.

Speech Disorders Classification System Summary (SDCSS) findings for participants in eight Complex Neurodevelopmental Disorders.

Speech disorders in eight CND

High and low prevalence groups.

Figure 5 includes the prevalence findings for speech classifications within each of the CND groups. Groups are ordered vertically in the two columns by the highest to the lowest total prevalence of the two classes of speech disorders, SE and SD. Using an arbitrary 50% criterion, at least half of the participants in the first five of the eight CND (Down syndrome [DS], Idiopathic Intellectual Disability [IID], Galactosemia [GAL], fragile X syndrome [FXS], and 22q11.2 Deletion syndrome [22q]) were classified as either SE/PSE or SD/PSD. Fewer than 50% of the participants in the other three CND (16p11.2 [16p]; Severe Traumatic Brain Injury [TBI], and Autism Spectrum Disorder [ASD]) met classification criteria for either of the two speech disorders classifications. The large range in the prevalence of the two classes of speech disorders across the eight CND in Figure 5 , particularly for SD/PSD (16.7%−93.3%), is consistent with the heterogeneous neurocognitive and neuromotor deficits posited to underlie speech sound deletions, substitutions, and distortions, with implications for genetic and genomic correlates and clinical management.

Percentage of participants in eight Complex Neurodevelopmental Disorders classified into one of three speech classifications.

Speech errors/persistent speech errors

The prevalence of SE or PSE as the only speech disorder in participants in the eight groups of speakers with CND averaged 11.4%, with prevalences in Figure 5 ranging from 0% (22q, ASD) to 43.5% (IID). Thus, SE/PSE as the only speech disorder was relatively infrequent in the present samples of speakers with CND.

The high percentage of PSE (43.5%) in the IID group ( M : 36.4 years; SD : 7 years) is of interest. Unlike participants in the other CND groups, the intellectual deficit in persons in this group was idiopathic, rather than associated with syndromic and other neurodevelopmental disorders with well-described phenotypic deficits in sensorimotor domains. A research question that cannot be addressed with the current database is whether PSE in persons with IID was a residual of earlier SE or whether it was a residual of earlier SD. That is, did these speakers always have SE/PSE, or were their common and/or uncommon speech sound distortions ( Shriberg, 1993 ; Appendix) at assessment in their third decade of life the residual of prior SD/PSD with distortions? Tracking such longitudinal speech and motor speech phenotypes in appropriately selected and controlled CND groups with different types and severity of intellectual deficits could be informative for speech-genetics research. A number of instrumental methods (e.g., palatography, ultrasound, kinematics, motion capture tracking) are becoming increasingly available for detailed phenotypic description of SE/PSE (cf. Ludlow, Kent, & Gray, 2018 ). Such fine-grained data on SE/PSE in persons with selected CND should inform accounts of the pathobiological correlates of misarticulations, in turn leading to treatment targeting for the distortions that maximally contribute to speakers’ intelligibility, comprehensibility, and acceptability (e.g., McAllister Byun & Preston, 2015 ; Torrington Eaton, 2015 ; Yoder, Camarata, & Woynaroski, 2016 ).

Speech delay/persistent speech delay

The percentage of participants with SD/PSD in the eight CND, as shown in Figure 5 , ranged from 16.7% (ASD) to 93.3% (DS), averaging 40.0%.

A research implication of this wide prevalence range for SD/PSD across CND is the potential for comparative study of speech processing deficits in SD ( Figure 2 ). As reviewed previously, early and persistent SD is posited to reflect deficits in representational processes, rather than in speech production deficits in transcoding (planning/programming) or in speech execution. Research that compares the deletion and substitution errors of SD/PSD in different CND to those in children with idiopathic SD could inform questions about the single and multiple neural and psycholinguistic loci of speech processing deficits in SD ( Pennington, 2006 ).

The present cross-classification findings for SD/PSD in CND underscore an important clinical question. Findings in Figure 4 indicate that whereas 27.6% (35/127) of the participants with SD/PSD had No MSD, the remaining 72.4% (92/127) also had one of the four types of motor speech disorders discussed in the next section. A two-group test of proportions indicated that the latter group was significantly more prevalent in the present database (Fisher’s exact test; p -value = 0.000). For the transcoding deficits in CAS and the execution delays/deficits in SMD and CD ( Figure 2 ), respectively, current trends are to base speech treatment wholly or substantially on principles of motor learning (e.g., Maas, Gildersleeve-Neumann, Jakielski, & Stoeckel, 2014 ; Maas et al., 2008 ). If speech sound deletions and substitutions in persons with idiopathic SD or PSD are proposed to reflect cognitive rather than motor speech neurodevelopmental constraints ( Figure 2 ), a clinical question is whether treatment based wholly or substantially on the principles of motor-learning is appropriate for the nearly 3/4ths (72.4%) of the present speakers with CND, SD/PSD, and concurrent motor speech disorder (i.e., SMD, CD, CAS, or CD & CAS). Rather, treatment would seem to be more appropriately based on each individual speaker’s cross-classification findings, which include the type and severity of involvement in both cognitive and motor domains ( Nijland, Terband, & Maassen, 2015 ; Shriberg et al., 2012 ).

Motor speech disorders in eight CND

Figure 6 includes findings for the prevalence of the five classifications of MSD (including No MSD) in the eight samples of participants with CND. Although not in the same order, the five CND with the highest prevalence of SD in Figure 5 (DS, IID, GAL, FXS, 22q) also had the highest prevalence of one of the four types of MSD in Figure 6 (DS, 22q, IID, FXS, GAL). The following sections review prevalence findings for each of the four MSD.

Speech motor delay

As shown in Figure 4 , 25.1% of the participants in the eight groups of participants with CND met SDCS requirements for SMD. The prevalence of SMD in the adults with IID (47.8%; Figure 6 ) was nearly double the group-averaged mean of the prevalence of SMD in the other seven groups (23.5%), which ranged from 14.3% to 29.4%. In comparison, the per-participant prevalence of SMD in a sample of 415, 3-to 16-year-old participants with idiopathic SD from six cities in the U.S. was 12% ( Shriberg, Kwiatkowski, & Mabie, 2019 ).

The present high prevalence of SMD in participants with CND, together with the previous prevalence findings for SMD in children with SD ( Shriberg, Kwiatkowski, & Mabie, 2019 ), supports SMD as a classification entity for speakers with imprecise and/or unstable speech, prosody, and/or voice that does not meet criteria for CD and/or CAS. In the present data, of the 61 participants who did meet criteria for CD and/or CAS, 52 (85.2%) also met criteria for SMD. As expected, because both SMD and CD are disorders of speech execution ( Figure 2 ), 89.1% of the participants with CD also met criteria for SMD, whereas 73.3% of the participants with CAS also met criteria for SMD; a Fisher’s exact test was non-significant ( p -value = 0.204).

In addition to the high prevalence of SMD in seven of the CND groups (total group average = 23.5%), the substantial prevalence of SMD in participants with IID (47.8%) discussed previously is of particular research and clinical interest. As described previously, nearly 80% of the adult participants with IID met criteria for either NSA (34.8%) or SE/PSE (43.5%), the latter of which was the highest percentage obtained among the eight groups of CND. Only approximately 20% of these participants had the persistent speech sound deletions or substitutions that define PSD. As described in the original study of these participants ( Shriberg & Widder, 1990 ) and in item level-detail on their performance on the Precision-Stability Index ( Shriberg & Mabie, 2017 ), their most prevalent perceptual and acoustic signs of SMD were in the domains of prosody and voice (i.e., not in the domain of speech production). Thus, SMD may be an especially appropriate phenotypic classification for speakers with intellectual disability whose speech perceptually suggests a motor component that does not meet criteria for the processing deficits in transcoding that define CAS or the processing deficits in execution that define CD ( Figure 2 ).

Last, SMD may have an important role in epidemiological and other studies of the prevalence and phenotype of motor speech disorders in CND. Subsequent discussion speculates on the possibility first proposed in preliminary research in SMD ( Shriberg, 2017 ) that SMD is the true-positive classification for the false-positive classifications of CAS in research and clinical speech pathology ( American Speech-Language-Hearing Association, 2007 ; Royal College of Speech and Language Therapists, 2011 ).

Childhood dysarthria

The prevalence of CD in the eight samples of CND, as shown in Figure 4 , was 13.3%, with an additional 4.9% of participants meeting criteria for concurrent CAS (CD & CAS) at assessment. Thus, a total of 18.2% of the present participants met criteria for either CD alone or concurrent with CAS, in comparison to the total percentage of participants with SMD (25.1%). The percentages of participants with CD or CD & CAS in the first five CND in Figure 6 , (DS [60%], 22q [41.2%], IID [17.4%], FXS [32.1%], and GAL [29%]) were considerably higher than the percentages in the remaining three CND. Notably for neurogenetic research questions, the prevalence of CD alone was the same or higher than the prevalence of CD & CAS in each of the eight CND. In comparison to the above values, 3.4% of 415 children with idiopathic SD ( Shriberg, Kwiatkowski, & Mabie, 2019 ) met criteria for CD and none met criteria for concurrent CD & CAS.

Subtypes of childhood dysarthria

Table 3 includes prevalence estimates for the five subtypes of dysarthria listed in the last five rows of Appendix 2 . The dysarthria subtype indices are each comprised of 12–19 of the 34 signs in the Dysarthria Index. As shown in the Supplement , the SDCS signs of subtypes of dysarthria were operationalized and standardized using the Mayo Clinic classification system definitions and subscale item weightings ( Duffy, 2013 ). Subtype signs are not mutually exclusive, with some signs proposed to be diagnostic of more than 1 of the 5 subtypes of dysarthria ( Duffy, 2013 ). Details on how the percentile values in Table 3 were derived from a database of 442 participants at risk for childhood motor speech disorders are described in a technical report ( Mabie & Shriberg, 2017 ; pp. 203–204). Values ≤ 10 th percentile on each dysarthria subtype index were classified as positive for that subtype.

Percentage of participants with Childhood Dysarthria (CD) and Childhood Dysarthria & Childhood Apraxia of Speech (CD & CAS) with scores ≤ 10th percentile on subtypes of CD. a

As shown in Table 3 , the subtypes for which at least 50% of the participants in each of the eight CND were positive are bolded. For example, in the first data row in Table 3 , 77.8% of the participants with DS who met criteria for CD alone or CD & CAS were positive for Ataxia. As is consistent with the childhood and adult literatures in developmental and acquired dysarthria, participants may be positive for one subtype of dysarthria ( pure ) or more than one subtype ( mixed ). Mixed dysarthrias may be in part due to the high collinearity among subtypes with some of the same clinical signs (e.g., slow rate is common to several subtypes of dysarthria).

With the exception of the findings for DS discussed next, the estimates in Table 3 are preliminary, due to the low number of participants with CD (63) that comprise the denominators for the percentage estimates across the 120 cells in Table 3 (i.e., 8 CND x 5 dysarthria subtypes x 3 classifications of CD [CD, CD & CAS, total]). Specifically, other than the total of 27 participants with DS ( Table 3 , fourth data column), the percentage of participants in each CND that met percentile criteria for dysarthria subtypes are based on from 2 to 9 participants with CD. Therefore, with the exception of the following discussion of findings for participants with DS, the dysarthria subtype findings for the remaining CND in Table 3 are provided only for their possible value to generate additional questions for CND-speech research in dysarthria.

Findings for participants with DS in Table 3 for the 60% (27/45) who met criteria for CD or CD & CAS are interpreted as strong support for Ataxia as the prevalent subtype of their CD. Ataxic dysarthria was prevalent in participants with DS with both CD alone (82%) and CD & CAS (70%). As shown in Table 3 , the only other CD subtype meeting the 50% criteria for these participants was Hyperkinetic, which met the criteria of 50% of signs ≤ 10 th percentile for 5 of the 10 participants with CD & CAS. As indicated in Table 3 , ataxic dysarthria is associated with deficits in cerebellar processes (e.g., Kent & Vorperian, 2013 ; Nadel, 2003 ), with implications for genomic and speech treatment research for persons with DS (cf. Wilson, Abbeduto, Camarata, & Shriberg, 2019a , 2019b ).

Childhood apraxia of speech

The previous findings ( Figure 4 ) indicated that CD alone occurred somewhat more frequently (13.3%) than CAS alone (4.3%). As shown in Figure 6 , CD was more prevalent than CAS in 6 of the eight CND, with the prevalence of participants with CAS alone ranging from 0% (ASD) to 11.8% (22q). In comparison to these values, 2.4% of 415 children with idiopathic SD ( Shriberg, Kwiatkowski, & Mabie, 2019 ) met criteria for CAS and none met criteria for concurrent CD & CAS.

The prevalence finding of 4.3% for CAS in CND (and an additional 4.9% CAS concurrent with CD [ Figure 4 ] to be discussed), supports the efficiency of studying CAS in the context of CND. A recent population-based, point-prevalence estimate of CAS in children with idiopathic SD is 1 per 1,000 children at 4 to 8 years of age ( Shriberg, Kwiatkowski, & Mabie, 2019 ). The diverse and well-studied neurogenomic substrates of many CND provide additional rationale for studying CAS in CND that have high rates of motor speech disorders ( Shriberg, 2010b ).

The present prevalence findings for CAS in the context of CND also have implications for continuing research and clinical findings indicating that CAS is overdiagnosed ( Shriberg & McSweeny, 2002 ). As noted previously, reviews of clinical studies in several countries indicate false positive CAS rates ranging from approximately 50% to approximately 90% ( American Speech-Language-Hearing Association, 2007 ; Royal College of Speech and Language Therapists, 2011 ), with SMD possibly accounting for a substantial percentage of the false positives ( Shriberg, 2017 ). Item analyses of the speech, prosody, and voice signs most associated with false positives for CAS need to be completed to test the validity of this speculation ( Shriberg, Campbell, et al., 2019 ).

A second question raised by the prevalence findings for CAS in the context of CND is the low prevalence of CAS in three of the eight CND compared to the other five CND ( Figure 6 ). Because the measurement and classification procedures were similar for all groups, the implication is that participants in the latter groups do not have the neurogenetic substrates of CAS present in participants in the first five groups. Reviews of the neurogenetic literatures in each of the eight CND are beyond the scope of the present prevalence study, but associated questions could possibly be resolved by results from meta-analyses of genetic, genomic, neurologic, and behavioural findings in the respective literatures.

Childhood dysarthria & childhood apraxia of speech

The prevalence of concurrent CD & CAS (4.9%), as shown in Figure 4 , was marginally higher than the prevalence of CAS alone (4.3%) within the 9.2% of the present speakers with CND meeting criteria for CAS. As indicated for the five CND with the highest prevalence of motor speech disorders in Figure 6 , the percentage of participants with CD & CAS was higher than CAS alone in two CND groups (DS, GAL), the same as CAS alone in one group (22q) and lower than CAS alone in two groups (IID, FXS).

The present findings for the prevalence of CD & CAS compared to CAS alone in CND support trends in the genetic and other literatures in CAS. Although the earliest phenotype of the British family with a disruption in FOXP2 described a disorder consistent with CAS ( Vargha-Khadem et al., 1998 ), later descriptions of the family and increasingly other studies of CAS associated with FOXP2 and other genes have broadened the phenotype to include CD (e.g., Liégeois & Morgan, 2012 ; Liégeois, Morgan, Connelly, & Vargha-Khadem, 2011 ; Morgan & Liégeois, 2010 ; Peter et al., 2017 ; Rice et al., 2012 ; Shriberg et al., 2006 ; Shriberg, Jakielski, & El-Shanti, 2008 ; Turner et al., 2013 ; Vernes et al., 2011 ).

Methodological considerations

Three methodological considerations warrant comment. First, the three behavioural measures of SMD, CD, and CAS used in this research have only recently become available. Each measure warrants additional research by other research groups using additional and alternative measurement modalities (e.g., neurologic, physiologic, kinematic) to cross-validate the diagnostic classifications and provide more finely-grained phenotypic detail. Second, generalizations from the present findings are limited to participants with the eight types of CND that were available in an audio-recorded database of conversational speech samples. Moreover, generalizations are limited to persons with the cognitive, linguistic, and affective abilities and dispositions to complete a continuous speech task and other SDCS supplementary tasks. Future estimates of the prevalence of speech and motor speech disorders in CND should include CND selected specifically for their genomic, neurodevelopmental, and behavioural similarities and differences. Last, the descriptive and inferential statistical findings from the present participants and methods were limited by the available cell sizes within each CND group, and consequently, within each of the four types of motor speech disorders.

Pending cross-validation, the primary findings of this research support the hypothesis that speech disorders, and notably motor speech disorders are substantially prevalent in persons with some types of complex neurodevelopmental disorders. A corollary conclusion is that comparative study of motor speech disorders in the context of complex neurodevelopmental disorders has the potential to inform programmatic research in biobehavioural causal pathways, treatment efficacy, and in primary, secondary, and tertiary forms of prevention.

Supplementary Material

Acknowledgments.

We thank each of the following colleagues for her or his contribution to this research: Leonard Abbeduto, Nancy Alarcon, Adriane Baylis, Raphael Bernier, Lois Black, Richard Boada, Roger Brown, Stephen Camarata, Thomas Campbell, Joseph Duffy, Marios Fourakis, Lisa Freebairn, Jordan Green, Sheryl Hall, Katherina Hauner, Heather Karlsson, Joan Kwiatkowski, Barbara Lewis, Jane McSweeny, Jennifer McGlothlin, Christopher Moore, Rhea Paul, Bruce Pennington, Nancy Potter, Heather Rusiewicz, Carmen Rasmussen, Alison Scheer-Cohen, Kristie Spencer, Christie Tilkens, Jan van Santen, Jennell Vick, Emily White, Carol Widder, David Wilson, and Erin Wilson.

This work was supported by grants from the National Institute on Deafness and Other Communication Disorders [DC000496] and a core grant to the Waisman Center from the National Institute of Child Health and Human Development [U54 HD090256].

Abbreviations:

Appendix 1. participants and inclusionary criteria.

Description and inclusionary criteria for participants in each of the eight Complex Neurodevelopmental Disorders groups (see text, Table 1 ). A technical report ( Shriberg & Mabie, 2017 ) includes speech, prosody, and voice assessment data for participants in each of the eight groups.

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Appendix 2. Speech, motor speech, and dysarthria subtype classifications in the Speech Disorders Classification System (SDCS)

The five Speech classifications and five Motor Speech classifications in the SDCS are each mutually exclusive. The five dysarthria subtype classifications are not mutually exclusive. That is, a speaker can meet percentile criteria for more than one of the five listed dysarthria subtype classifications (i.e., mixed dysarthria). See Supplement for the procedures and measures used to classify each motor speech disorder.

Notes : PSI = Precision-Stability Index; DI = Dysarthria Index; DSI = Dysarthria Subtype Index; PM = Pause Marker.

Disclosure Statement

The authors report no declarations of interest.

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