A Review of Screeners to Identify Risk of Developmental Language Disorder.

PURPOSE: This review aims to comprehensively summarize, compare, and evaluate screeners used to identify risk for developmental language disorder (DLD), a common learning disability that is underidentified. Screening for DLD is a cost-effective way to identify children in need of further assessment and, in turn, provides much needed supports. METHOD: We identified 15 commercially available English language DLD screeners in North America. We then characterized each screener on 27 aspects in three domains, including (a) accessibility information (acronym, subtest, website, cost, materials included, publish year, examiner qualification, age range, administration time, and administration format), (b) usability features (dialect compatibility, progress monitoring function, actionable follow-up instruction, group assessment capability, and online administration availability), and (c) technical standards (the availability of a technical manual, conceptual definition, the sample size used in classification accuracy calculation, sample distribution, year of sample collection, outcome measure, sample base rate, cutoff score, sensitivity, specificity, positive predictive value, and negative predictive value). RESULTS: We obtained sufficient accessibility information from 14 out of 15 (93%) screeners. In contrast, none of the screeners (0%) included comprehensive usability features. Ten screeners (67%) included a range of classification accuracy (70%-100% sensitivity and 68%-90% specificity). We provided areas of strength and weakness for each screener as a quick reference for users and generated screener recommendations for five practical scenarios. CONCLUSIONS: Our findings presented some DLD screeners that meet most standards and highlight numerous areas for improvement, including improving classification accuracy and clarifying follow-up instructions for children who are identified with DLD risk. Screening for DLD is critical to provide timely early identification, intervention, and classroom support, which in turn facilitates student outcomes.

Phenome risk classification enables phenotypic imputation and gene discovery in developmental stuttering.

Developmental stuttering is a speech disorder characterized by disruption in the forward movement of speech. This disruption includes part-word and single-syllable repetitions, prolongations, and involuntary tension that blocks syllables and words, and the disorder has a life-time prevalence of 6-12%. Within Vanderbilt's electronic health record (EHR)-linked biorepository (BioVU), only 142 individuals out of 92,762 participants (0.15%) are identified with diagnostic ICD9/10 codes, suggesting a large portion of people who stutter do not have a record of diagnosis within the EHR. To identify individuals affected by stuttering within our EHR, we built a PheCode-driven Gini impurity-based classification and regression tree model, PheML, by using comorbidities enriched in individuals affected by stuttering as predicting features and imputing stuttering status as the outcome variable. Applying PheML in BioVU identified 9,239 genotyped affected individuals (a clinical prevalence of ∼10%) for downstream genetic analysis. Ancestry-stratified GWAS of PheML-imputed affected individuals and matched control individuals identified rs12613255, a variant near CYRIA on chromosome 2 (B = 0.323; p value = 1.31 × 10-8) in European-ancestry analysis and rs7837758 (B = 0.518; p value = 5.07 × 10-8), an intronic variant found within the ZMAT4 gene on chromosome 8, in African-ancestry analysis. Polygenic-risk prediction and concordance analysis in an independent clinically ascertained sample of developmental stuttering cases validate our GWAS findings in PheML-imputed affected and control individuals and demonstrate the clinical relevance of our population-based analysis for stuttering risk.

Skill Profiles of College Students With a History of Developmental Language Disorder and Developmental Dyslexia.

Developmental language disorder (DLD) and developmental dyslexia (DD) are two prevalent subtypes of Specific Learning Disabilities (SLDs; Diagnostic and Statistical Manual of Mental Disorders [5th ed.; DSM-5]). Yet, little information is available regarding the distinct challenges faced by adults with DLD and/or DD in college. The purpose of the present report is to characterize the relative strengths and challenges of college students with a history of DLD and/or DD, as this information is critical for providing appropriate institutional support. We examined the cognitive skill profiles of 352 college students (ages 18-35 years), using standardized and research-validated measures of reading, spoken language, nonverbal cognition, and self-reported childhood diagnostic history. We classified college students as having DLD (n = 50), and/or DD (n = 40), or as typically developed adults (n = 132) according to procedures described for adults with DLD and DD. A structural equation model determined the cognitive, language, and reading measures predicted by the classification group. Adults with DLD demonstrated poor verbal working memory and speeded sentence-level reading. Adults with DD primarily demonstrated deficits in phonology-based skills. These results indicate that adults with DLD and/or DD continue to face similar challenges as they did during childhood, and thus may benefit from differentially targeted accommodations in college.

Definitions of Nonverbal and Minimally Verbal in Research for Autism: A Systematic Review of the Literature.

This systematic review examined definitions of "nonverbal" or "minimally verbal" and assessment measures used to evaluate communication in intervention studies focusing on improving expressive verbal communication in children with autism spectrum disorder (ASD). We reviewed sample size, number of participants, participant age, and male/female representation. Our analysis yielded relatively few studies with non/minimally verbal children with ASD focusing on verbal expressive communication. Further, we found large inconsistencies in measures used, definitions of "nonverbal" and "minimally verbal", and ages targeted. Guidelines are suggested to create a more uniform assessment protocol with systematic descriptions of early communication learners as a foundational step for understanding the heterogeneity in this group and replicating research findings for this subgroup of children with ASD.

Construct Validity of the Viking Speech Scale.

OBJECTIVE: The Viking Speech Scale (VSS) reliably classifies the speech performance of children with cerebral palsy. This paper aims to establish the construct validity of the VSS by testing the extent to which percentage intelligibility in single word speech and connected speech predicts VSS rating. PATIENTS AND METHODS: This is a secondary analysis of two sets of anonymised data collected for previous research. The full data set comprised 79 children with cerebral palsy from the US (n = 43) and the UK (n = 36): (43 boys, 36 girls); mean age 7.2 years (SD 3.3). Single word intelligibility was measured using the TOCS+ words for US children and Children's Speech Intelligibility Measure for the UK children. Connected speech intelligibility was measured from a subset of repeated sentences in TOCS+ for US children and picture description for the UK children. We used ordinal logistic regression to examine prediction of VSS rating by percentage single word and connected speech intelligibility scores in both samples. RESULTS: Percentage single word intelligibility and connected speech intelligibility predicted VSS rating in univariate and multivariate regression models for both the US and UK samples. CONCLUSION: Intelligibility predicts VSS for both single words and connected speech, establishing the construct validity of VSS.

Developmental language disorder: Maternal stress level and behavioural difficulties of children with expressive and mixed receptive-expressive DLD.

PURPOSE: The current study aimed to compare bullying and victimisation behaviour, levels of emotional and behavioural problems and maternal stress in children with expressive and mixed receptive-expressive language disorders. METHODS: We measured the bullying and victimisation behaviour, the emotional and behavioural problems as well as the maternal stress in a sample of 4-9-year-old children with language disorders (N = 65). RESULTS: Our main findings were that although bullying and victimisation were common in both groups; the children with expressive language disorders tended to be perpetrators (teacher-reported) while the children with mixed language disorders experienced more bullying and tended to be victims (self-reported); the latters' mothers perceived significantly more stress compared to mothers of children with expressive language disorders. Furthermore, the results of the study indicate that, although the number of children within the family was identified as the strongest predictor explaining maternal stress, maternal stress is a multifactorial concept influenced by many factors. CONCLUSION: For a successful intervention in children with developmental language disorders, it seems to be very important to identify parental stressors beyond the treatment of the child's problems.

Reconceptualizing developmental language disorder as a spectrum disorder: issues and evidence.

BACKGROUND: There is considerable variability in the presentation of developmental language disorder (DLD). Disagreement amongst professionals about how to characterize and interpret the variability complicates both the research on understanding the nature of DLD and the best clinical framework for diagnosing and treating children with DLD. We describe and statistically examine three primary possible models for characterizing the variability in presentation in DLD: predictable subtypes; individual differences; and continuum/spectrum. AIMS: To test these three models of DLD in a population-based sample using two distinct types of cluster analyses. METHODS & PROCEDURES: This study included children with DLD (n = 505) from the US Epidemiological Study of Language Impairment database. All available language and cognitive measures were included. Two cluster methods were used: Ward's method and K-means. Optimal cluster sizes were selected using Bayesian information criteria (BIC). Bootstrapping and permutation methods were used to evaluate randomness of clustering. OUTCOMES & RESULTS: Both clustering analyses yielded more than 10 clusters, and the clusters did not have spatial distinction: many of these clusters were not clinically interpretable. However, tests of random clustering revealed that the cluster solutions obtained did not arise from random aggregation. CONCLUSIONS & IMPLICATIONS: Non-random clustering coupled with a large number of non-interpretable subtypes provides empirical support for the continuum/spectrum and individual differences models. Although there was substantial support for the continuum/spectrum model and weaker support for the individual differences model, additional research testing these models should be completed. Based on these results, clinicians working with children with DLD should focus on creating treatment plans that address the severity of functioning rather than seeking to identify and treat distinct subtypes. Additional consideration should be given to reconceptualizing DLD as a spectrum condition.

Discriminating autism and language impairment and specific language impairment through acuity of musical imagery.

Deficits in auditory short-term memory have been widely reported in children with Specific Language Impairment (SLI), and recent evidence suggests that children with Autism Spectrum Disorder and co-morbid language impairment (ALI) experience similar difficulties. Music, like language relies on auditory memory and the aim of the study was to extend work investigating the impact of auditory short-term memory impairments to musical perception in children with neurodevelopmental disorders. Groups of children with SLI and ALI were matched on chronological age (CA), receptive vocabulary, non-verbal intelligence and digit span, and compared with CA matched typically developing (TD) controls, on tests of pitch and temporal acuity within a voluntary musical imagery paradigm. The SLI participants performed at significantly lower levels than the ALI and TD groups on both conditions of the task and their musical imagery and digit span scores were positively correlated. In contrast ALI participants performed as well as TD controls on the tempo condition and better than TD controls on the pitch condition of the task. Whilst auditory short-term memory and receptive vocabulary impairments were similar across ALI and SLI groups, these were not associated with a deficit in voluntary musical imagery performance in the ALI group.

Differential profiles of four groups of children referred to an autism diagnostic service in Belgium: Autism-specific hallmarks.

OBJECTIVE:: This article aims at exploring distinctive hallmarks of autistic disorders compared to other groups presenting mimicking and/or overlapping conditions. METHOD:: The exploratory study involved 196 children with autism, intellectual disability, language impairment and psycho-affective disorders previously referred to an autism diagnostic service. The autism discriminative tool (ADT), a behavioural checklist, was used to compile and analyse the children's profiles based on the clinical information gathered during diagnostic assessments. All samples were compared and analysed separately according to the checklist's four domains. RESULTS:: Children with autism showed greater frequency and severity of impairments on the ADT's social and communication categories. These children also differed from other groups in terms of specific rigid behaviours and high frequency of atypical sensory responses. CONCLUSIONS:: Results illustrate the psychopathology of autism spectrum disorder suspected children referred to a specialized autism diagnostic service, doing so by providing us with specific clinical profiles.

Subgroups in language trajectories from 4 to 11 years: the nature and predictors of stable, improving and decreasing language trajectory groups.

BACKGROUND: Little is known about the nature, range and prevalence of different subgroups in language trajectories extant in a population from 4 to 11 years. This hinders strategic targeting and design of interventions, particularly targeting those whose difficulties will likely persist. METHODS: Children's language abilities from 4 to 11 years were investigated in a specialist language longitudinal community cohort (N = 1,910). Longitudinal trajectory latent class modelling was used to characterise trajectories and identify subgroups. Multinomial logistic regression was used to identify predictors associated with the language trajectories children followed. RESULTS: Three language trajectory groups were identified: 'stable' (94% of participants), 'low-decreasing' (4%) and 'low-improving' (2%). A range of child and family factors were identified that were associated with following either the low-improving or low-increasing language trajectory; many of them shared. The low-improving group was associated with mostly environmental risks: non-English-speaking background, social disadvantage and few children's books in the home. The low-decreasing group was associated with mainly biological risks: low birth weight, socioemotional problems, lower family literacy and learning disability. CONCLUSIONS: By 4 years, services can be confident that most children with low language will remain low to 11 years. Using rigid cut-points in language ability to target interventions is not recommended due to continued individual variability in language development. Service delivery models should incorporate monitoring over time, targeting according to language abilities and associated risks and delivery of a continuum of interventions across the continuum of need.

Why is it so hard to reach agreement on terminology? The case of developmental language disorder (DLD).

A recent project entitled CATALISE used the Delphi method to reach a consensus on terminology for unexplained language problems in children. 'Developmental language disorder' (DLD) was the term agreed by a panel of 57 experts. Here I reflect on points of difficulty that arose when attempting to reach a consensus, using qualitative information from comments made by panel members to illustrate the kinds of argument used. One issue of debate was the use of labels, in particular the term 'disorder', which was seen as having both pros and cons. The potential for labels to stigmatize or create low expectations was a particular concern. However, labels could also ensure language problems were not trivialized and could help avoid stigma by providing an explanation for behaviours that might otherwise meet with disapproval. Further debate surrounded issues of how best to identify cases of disorder. Although it was agreed there should be a focus on cases with a poor prognosis, it was recognized that our knowledge of factors related to prognosis was still incomplete. Furthermore, there was a tension between use of standardized tests, which allow for a relatively objective and reliable assessment of language, and more qualitative observations, which can capture functional aspects of communication that are not always picked up on formal assessment. Debate also surrounded the issue of the relationship between DLD and other conditions. Some favoured drawing a distinction between DLD and language disorders associated with other conditions, and others regarded such distinctions as unnecessary. We concluded that it was misleading to assume co-occurring conditions were causes of language disorder, but it was helpful to distinguish DLD from cases of language disorder associated with 'differentiating conditions' that had a known or likely biomedical origin, including brain injury, sensorineural hearing loss, genetic syndromes, intellectual disability and autism spectrum disorder. Furthermore, DLD could co-occur with milder neurodevelopmental disorders that did not have a clear biomedical aetiology. Normal-range non-verbal IQ has traditionally been incorporated in the diagnosis of DLD, but this was rejected as unsupported by evidence. DLD is a category that has utility in identifying children who would benefit from speech-language therapy services, but it should not be thought of as a well-defined condition. DLD has a multifactorial aetiology, is heterogeneous in terms of language features and overlaps with other neurodevelopmental disorders. Our notions of DLD are likely to be refined by further research into aetiology, associated characteristics and intervention effectiveness.

A Diagnostic Marker to Discriminate Childhood Apraxia of Speech From Speech Delay: IV. The Pause Marker Index.

Purpose: Three previous articles provided rationale, methods, and several forms of validity support for a diagnostic marker of childhood apraxia of speech (CAS), termed the pause marker (PM). Goals of the present article were to assess the validity and stability of the PM Index (PMI) to scale CAS severity. Method: PM scores and speech, prosody, and voice precision-stability data were obtained for participants with CAS in idiopathic, neurogenetic, and complex neurodevelopmental disorders; adult-onset apraxia of speech consequent to stroke and primary progressive apraxia; and idiopathic speech delay. Three studies were completed including criterion and concurrent validity studies of the PMI and a temporal stability study of the PMI using retrospective case studies. Results: PM scores were significantly correlated with other signs of CAS precision and stability. The best fit of the distribution of PM scores to index CAS severity was obtained by dividing scores into 4 ordinal severity classifications: mild, mild-moderate, moderate-severe, and severe. Severity findings for the 4 classifications and retrospective longitudinal findings from 8 participants with CAS supported the validity and stability of the PMI. Conclusion: Findings support research and clinical use of the PMI to scale the severity of CAS.

A Diagnostic Marker to Discriminate Childhood Apraxia of Speech From Speech Delay: I. Development and Description of the Pause Marker.

Purpose: The goal of this article (PM I) is to describe the rationale for and development of the Pause Marker (PM), a single-sign diagnostic marker proposed to discriminate early or persistent childhood apraxia of speech from speech delay. Method: The authors describe and prioritize 7 criteria with which to evaluate the research and clinical utility of a diagnostic marker for childhood apraxia of speech, including evaluation of the present proposal. An overview is given of the Speech Disorders Classification System, including extensions completed in the same approximately 3-year period in which the PM was developed. Results: The finalized Speech Disorders Classification System includes a nosology and cross-classification procedures for childhood and persistent speech disorders and motor speech disorders (Shriberg, Strand, & Mabie, 2017). A PM is developed that provides procedural and scoring information, and citations to papers and technical reports that include audio exemplars of the PM and reference data used to standardize PM scores are provided. Conclusions: The PM described here is an acoustic-aided perceptual sign that quantifies one aspect of speech precision in the linguistic domain of phrasing. This diagnostic marker can be used to discriminate early or persistent childhood apraxia of speech from speech delay.

A Diagnostic Marker to Discriminate Childhood Apraxia of Speech From Speech Delay: II. Validity Studies of the Pause Marker.

Purpose: The purpose of this 2nd article in this supplement is to report validity support findings for the Pause Marker (PM), a proposed single-sign diagnostic marker of childhood apraxia of speech (CAS). Method: PM scores and additional perceptual and acoustic measures were obtained from 296 participants in cohorts with idiopathic and neurogenetic CAS, adult-onset apraxia of speech and primary progressive apraxia of speech, and idiopathic speech delay. Results: Adjusted for questionable specificity disagreements with a pediatric Mayo Clinic diagnostic standard, the estimated sensitivity and specificity, respectively, of the PM were 86.8% and 100% for the CAS cohort, yielding positive and negative likelihood ratios of 56.45 (95% confidence interval [CI]: [1.15, 2763.31]) and 0.13 (95% CI [0.06, 0.30]). Specificity of the PM for 4 cohorts totaling 205 participants with speech delay was 98.5%. Conclusion: These findings are interpreted as providing support for the PM as a near-conclusive diagnostic marker of CAS.

Phase 2 of CATALISE: a multinational and multidisciplinary Delphi consensus study of problems with language development: Terminology.

BACKGROUND: Lack of agreement about criteria and terminology for children's language problems affects access to services as well as hindering research and practice. We report the second phase of a study using an online Delphi method to address these issues. In the first phase, we focused on criteria for language disorder. Here we consider terminology. METHODS: The Delphi method is an iterative process in which an initial set of statements is rated by a panel of experts, who then have the opportunity to view anonymised ratings from other panel members. On this basis they can either revise their views or make a case for their position. The statements are then revised based on panel feedback, and again rated by and commented on by the panel. In this study, feedback from a second round was used to prepare a final set of statements in narrative form. The panel included 57 individuals representing a range of professions and nationalities. RESULTS: We achieved at least 78% agreement for 19 of 21 statements within two rounds of ratings. These were collapsed into 12 statements for the final consensus reported here. The term 'Language Disorder' is recommended to refer to a profile of difficulties that causes functional impairment in everyday life and is associated with poor prognosis. The term, 'Developmental Language Disorder' (DLD) was endorsed for use when the language disorder was not associated with a known biomedical aetiology. It was also agreed that (a) presence of risk factors (neurobiological or environmental) does not preclude a diagnosis of DLD, (b) DLD can co-occur with other neurodevelopmental disorders (e.g. ADHD) and (c) DLD does not require a mismatch between verbal and nonverbal ability. CONCLUSIONS: This Delphi exercise highlights reasons for disagreements about terminology for language disorders and proposes standard definitions and nomenclature.

Dynamic Assessment of Narratives: Efficient, Accurate Identification of Language Impairment in Bilingual Students.

Purpose: This study investigated the classification accuracy of a concentrated English narrative dynamic assessment (DA) for identifying language impairment (LI). Method: Forty-two Spanish-English bilingual kindergarten to third-grade children (10 LI and 32 with no LI) were administered two 25-min DA test-teach-test sessions. Pre- and posttest narrative retells were scored in real time. Using a structured intervention approach, examiners taught children missing story grammar elements and subordination. A posttest was administered using a parallel story. Results: Four classification predictors were analyzed: posttest scores, gain scores, modifiability ratings, and teaching duration. Discriminant function analysis indicated that an overall modifiability rating was the best classifier, with 100% sensitivity and 88% specificity after 1 DA session and 100% sensitivity and specificity after 2 sessions. Any 2 combinations of posttest scores, modifiability ratings, and teaching duration for just 1 session resulted in sensitivity and specificity rates over 90%. Receiver operating characteristic analyses were used to identify clinically usable cutoff points. Post hoc exploration indicated that similar results could be obtained after only one 5-10-min teaching cycle, potentially further abbreviating the DA process. Conclusion: Concentrated English narrative DA results in high classification accuracy for bilingual children with and without LI. This efficient version of DA is amenable to clinical use.

Validity of the Communication Function Classification System for use with preschool children with communication disorders.

AIM: To evaluate construct and predictive validity of the Communication Function Classification System (CFCS) for use with preschool children with a range of speech and language disorders. METHOD: Seventy-seven preschool children with speech and language disorders (50 males, 27 females; mean 2y 7mo, standard deviation [SD] 1y) participated in this cohort study. Preschool children had speech and language, language-only, or speech-only disorders. Together with parent input, speech-language pathologists (SLPs) completed the CFCS at time 1. Parents and SLPs then independently completed a validated change-detecting functional communication outcome measure, the Focus on the outcomes of Communication Under Six (FOCUS), three times: at assessment (time 1), at the start of treatment (time 2), and at the end of treatment (time 3). RESULTS: There was a significant negative correlation between CFCS classifications and FOCUS scores at all three measurement points for the ratings by both parents and SLPs (correlations ranged from -0.60 to -0.76). As expected, no correlations between CFCS classifications and FOCUS change scores were statistically significant. INTERPRETATION: This study provides evidence of construct and predictive validity of the CFCS, demonstrating its value as a discriminative tool for use with preschool children with a range of speech and language disorders.

Atypical speech and language development: a consensus study on clinical signs in the Netherlands.

BACKGROUND: Atypical speech and language development is one of the most common developmental difficulties in young children. However, which clinical signs characterize atypical speech-language development at what age is not clear. AIM: To achieve a national and valid consensus on clinical signs and red flags (i.e. most urgent clinical signs) for atypical speech-language development in children from 1 to 6 years of age. METHODS & PROCEDURES: A two-round Delphi study in the Netherlands with a national expert panel (n = 24) of speech and language therapists was conducted. The panel members responded to web-based questionnaires addressing clinical signs. Consensus was defined as ≥ 70% of the experts agreeing on an issue. OUTCOMES & RESULTS: The first round resulted in a list of 161 characteristics of atypical speech and language development. The second round led to agreement on 124 clinical signs and 34 red flags. CONCLUSIONS & IMPLICATIONS: Dutch national consensus concerns 17-23 clinical signs per age year for the description of an atypical speech-language development in young children and three to 10 characteristics per age year being red flags for atypical speech-language development. This consensus contributes to early identification and diagnosis of children with atypical speech-language development, awareness and research.