The impact of enhanced Milieu teaching with phonological emphasis (EMT + PE) on the speech and language outcomes for toddlers with cleft palate in Brazil and the United States of America.

PURPOSE: The purpose of this study was to compare the speech and language outcomes of children with cleft palate with or without cleft lip (CP+/-L) in the USA to children with CP+/-L in Brazil who underwent intervention with enhanced Milieu teaching with phonological emphasis (EMT + PE), as there are few cross-country intervention comparisons for children with CP+/-L. METHOD: This is a retrospective analysis of 29 participants from the USA and 24 participants from Brazil who were matched on age. The US participants were between the ages of 13-35 months (M = 23.76), spoke Standard American English in the home, and were recruited from East Tennessee State University and Vanderbilt University. The Brazilian participants were between the ages of 20-34 months (M = 25.04), spoke Brazilian Portuguese in the home, and were recruited from the Hospital de Reabilitação de Anomalias Craniofaciais-Universidade de São Paulo. All treatment participants received EMT + PE from trained speech-language pathologists in hospital-university clinics. RESULT: The treatment groups demonstrated greater gains than comparison groups in percent consonants correct, number of different words, and expressive/receptive vocabulary. There was no main effect nor interaction by country. CONCLUSION: The application of EMT + PE in a second culture and language is a viable early intervention option for participants with CP+/-L.

Go Beyond Compliance: Use Individualized Education Programs to Answer Strategic Questions and Improve Programs.

PURPOSE: The most significant document to ensure effective and compliant design, implementation, monitoring, and enforcement of a program of special education services in the United States is the Individualized Education Program (IEP). Although IEPs have been used to document procedural compliance with the Individuals with Disabilities Education Act (IDEA) for individual students, IEPs also provide extensive data that can and should be used by a variety of stakeholders including speech-language pathologists (SLPs), school administrators, and state education agencies to design targeted professional development and collectively improve programs, processes, and outcomes in special education. METHOD: We summarize existing literature on the use of IEP data and describe opportunities to use IEP data to analyze individual student service patterns and SLP practice patterns. Aggregated IEP data also provide a robust view of district-wide and state trends in eligibility rates and least restrictive environment settings. Information on current and potential IEP data uses, reflection questions for substantive compliance, and lessons learned from a large-scale analysis of IEP data are provided. These lessons include potential software adjustments to enhance usability as a data source for substantive compliance; program improvement; and monitoring individual, school-wide, and district-wide outcomes. CONCLUSIONS: IEP data are a rich data source of information that may be used to (a) identify trends; (b) assist schools, districts, and states with ensuring substantive compliance with IDEA; (c) examine service equity and efficacy; (d) identify professional development needs; and (e) identify promising practices and provide opportunities to use real-time data to improve models and address public policy.

The Effect of Phonological Overlap on English and Spanish Expressive Vocabulary.

PURPOSE: This study examined the effect of phonological overlap on English and Spanish expressive vocabulary accuracy as measured by the bilingual Expressive One-Word Picture Vocabulary Test-Fourth Edition (EOWPVT-IV). We hypothesized that if languages interact during an expressive vocabulary task, then higher phonological overlap will predict higher expressive vocabulary accuracy, especially in the nondominant language. METHOD: Twenty English-dominant bilingual children (English-Spanish) completed the bilingual EOWPVT-IV. We calculated phonological overlap between 117 English and Spanish words included in the bilingual EOWPVT using the Crosslinguistic Overlap Scale for Phonology. RESULTS: Generalized logistic mixed-effect models revealed that phonological overlap and word frequency predicted vocabulary accuracy in Spanish, in addition to item difficulty. Age and item difficulty were the only predictors of English accuracy. CONCLUSIONS: Phonological overlap was a significant predictor of Spanish vocabulary accuracy (the least dominant language of the children in our sample), suggesting a transfer between vocabulary in the dominant language into vocabulary in the least dominant language. Future studies should investigate how languages interact in bilingual children to provide us with information about how to create and administer vocabulary tests that represent vocabulary in each language and use vocabulary teaching strategies that promote dual language development. SUPPLEMENTAL MATERIAL: https://doi.org/10.23641/asha.21554838.

Reading Development in Children With Nonsyndromic Cleft Palate With or Without Cleft Lip: Meta-analysis and Systematic Review.

OBJECTIVE: We conducted a meta-analysis and systematic review of literature comparing pre-reading and general reading in school-age children with nonsyndromic cleft palate with or without cleft lip (NSCP/L) to their peers without NSCP/L. METHODS: Our literature search identified 1238 possible records. After screening we identified 11 samples for inclusion for systematic review and eight for meta-analysis. We compared 292 children with NSCP/L to 311 peers for 23 pre-reading effect sizes and 17 general reading effect sizes (EFg). We conducted a random-effects metaregression using robust variance estimation. RESULTS: On average school-age children with NSCP/L scored lower on pre-reading (EFg = -0.36) and general reading measures (EFg = -0.38) compared to their peers. We conducted post-hoc analyses on phonological awareness and word decoding effect sizes; children with NSCP/L performed lower on phonological awareness (EFg = -0.22) and word decoding (EFg = -0.39) compared to their peers. There was weak evidence that hearing status and/or speech-language functioning might moderate reading development. There was limited evidence that age or socioeconomic status moderated reading development. However, samples did not consistently report several characteristics that were coded for this project. CONCLUSIONS: Our findings suggest that school-age children with NSCP/L have persistent reading problems. Further research is needed to explore reading development in children with NSCP/L, as well as the relationships among hearing, speech, language, and reading development.

Selective visual attention skills differentially predict decoding and reading comprehension performance across reading ability profiles.

Background: The purpose of this study was to examine the relationship between selective visual attention (SVA), reading decoding, listening comprehension and reading comprehension in children with and without a reading disorder. Methods: We used longitudinal data from the Avon Longitudinal Study of Parents and Children. We split children into four groups: Typical Readers, Dyslexics, Poor Comprehenders and Comorbid Reading Disorder. We included measures of single word reading, nonword reading, spelling, phonological processing, vocabulary, receptive language, nonverbal intelligence, selective attention and reading comprehension. We used analysis of variance, correlations and structural equation modelling to examine the relationship between SVA and reading. We fit two possible models: SVA Indirect and SVA Direct. The difference between these models was the inclusion of a direct path from SVA to reading comprehension. Results: We examined an indirect model, where SVA predicted reading comprehension through word decoding and listening comprehension, and a direct model, which included a pathway from SVA to reading comprehension. Based on our analysis of variance and correlation results, we collapsed the Dyslexic, Poor Comprehenders and Comorbid Reading Disorder groups for the structural equation modelling. We found evidence that for Typical Readers, an indirect model was the best fit, whereas the direct model was the best model for children with a reading disorder. Conclusions: Selective visual attention is related to reading comprehension. This relationship differs for children with and without a reading disorder.

Identifying interactive biological pathways associated with reading disability.

INTRODUCTION: Past research has suggested that reading disability is a complex disorder involving genetic and environment contributions, as well as gene-gene and gene-environment interaction, but to date little is known about the underlying mechanisms. METHOD: Using the Avon Longitudinal Study of Parents and Children, we assessed the contributions of genetic, demographic, and environmental variables on case-control status using machine learning. We investigated the functional interactions between genes using pathway and network analysis. RESULTS: Our results support a systems approach to studying the etiology of reading disability with many genes (e.g., RAPGEF2, KIAA0319, DLC1) and biological pathways (e.g., neuron migration, positive regulation of dendrite regulation, nervous system development) interacting with each other. We found that single nucleotide variants within genes often had opposite effects and that enriched biological pathways were mediated by neuron migration. We also identified behavioral (i.e., receptive language, nonverbal intelligence, and vocabulary), demographic (i.e., mother's highest education), and environmental (i.e., birthweight) factors that influenced case-control status when accounting for genetic information. DISCUSSION: The behavioral and demographic factors were suggested to be protective against reading disability status, while birthweight conveyed risk. We provided supporting evidence that reading disability has a complex biological and environmental etiology and that there may be a shared genetic and neurobiological architecture for reading (dis)ability.

The Structure of Word Learning in Young School-Age Children.

Purpose We investigated four theoretically based latent variable models of word learning in young school-age children. Method One hundred sixty-seven English-speaking second graders with typical development from three U.S. states participated. They completed five different tasks designed to assess children's creation, storage, retrieval, and production of the phonological and semantic representations of novel words and their ability to link those representations. The tasks encompassed the triggering and configuration stages of word learning. Results Results showed that a latent variable model with separate phonological and semantic factors and linking indicators constrained to load on the phonological factor best fit the data. Discussion The structure of word learning during triggering and configuration reflects separate but related phonological and semantic factors. We did not find evidence for a unidimensional latent variable model of word learning or for separate receptive and expressive word learning factors. In future studies, it will be interesting to determine whether the structure of word learning differs during the engagement stage of word learning when phonological and semantic representations, as well as the links between them, are sufficiently strong to affect other words in the lexicon.

Effects of a naturalistic intervention on the speech outcomes of young children with cleft palate.

Purpose: The purpose of this study was to investigate the extent to which a naturalistic communication intervention, Enhanced Milieu Teaching with Phonological Emphasis (EMT + PE) improved the speech outcomes of toddlers with cleft palate with or without cleft lip (CP ± L)Method: This study was a stratified randomised controlled trial.Setting: Treatment was delivered in a university clinic by a trained speech-language pathologist.Thirty children aged between 15 and 36 months (M = 25) with nonsyndromic CP ± CL and typical cognitive development were randomly assigned to a treatment (EMT + PE) or business as usual comparison condition.Participants in the EMT + PE treatment group received 48, 30-min sessions, over a 6-month period. Fidelity of treatment was high across participants.The primary outcome measures were percent consonants correct (PCC), consonant inventory, compensatory articulation errors, and nasal emission.Result: Regression analyses controlling for pre-intervention child characteristics were conducted for PCC and consonant inventory. Intervention was not a significant predictor of post-intervention outcome. Words per minute differentiated the children who benefitted from the intervention from those who did not. Reduction in compensatory errors and nasal emission occurred in both groups but to a greater degree in the EMT + PE group.Conclusion: EMT + PE is a promising early speech intervention for young children with CP ± L, especially for children with higher rates of word use.

Early Speech and Language Development in Children With Nonsyndromic Cleft Lip and/or Palate: A Meta-Analysis.

Objective The aim of the study was to conduct a meta-analysis of research examining the early speech and language functioning of young children, birth to age 8;11 (years;months), with nonsyndromic cleft lip and/or palate (NSCL/P) compared to their peers without NSCL/P. Method We conducted a random-effects metaregression using 241 effect sizes from 31 studies comparing 955 young children with NSCL/P to 938 typically developing peers on measures of speech and language functioning. Moderators were sample characteristics (i.e., age, cleft type, publication year, and study location) and measurement characteristics (i.e., speech sample material, language modality and domain, and assessment type). Results Young children with NSCL/P scored significantly lower on measures of speech and language compared to children without NSCL/P. Children with NSCL/P had smaller consonant inventories (standardized mean difference effect size [ESg] = -1.24), less accurate articulation (ESg = -1.13), and more speech errors (ESg = 0.93) than their peers. Additionally, children with NSCL/P had poorer expressive (ESg = -0.57) and receptive (ESg = -0.59) language skills than their peers. Age and assessment type moderated effect sizes for expressive language. As children with NSCL/P aged, their expressive language performance became more similar to their peers. Expressive language effect sizes from parent reports and observational language measures (estimated effect size = -0.74) were significantly lower than those from standardized norm-referenced tests (estimated effect size = -0.45). Conclusions These findings suggest that young children with NSCL/P experience delays relative to their peers across multiple speech and language constructs. Differences between children with NSCL/P and their typically developing peers appear to decrease with age. Supplemental Material https://doi.org/10.23641/asha.11356904.

Exome Sequencing of Two Siblings with Sporadic Autism Spectrum Disorder and Severe Speech Sound Disorder Suggests Pleiotropic and Complex Effects.

Recent studies of autism spectrum disorder (ASD) and childhood apraxia of speech (CAS) have resulted in conflicting conclusions regarding the comorbidity of these disorders on phenotypic grounds. In a nuclear family with two dually affected and one unaffected offspring, whole-exome sequences were evaluated for single nucleotide and indel variants and CNVs. The affected siblings but not the unaffected sibling share a rare deleterious compound heterozygous mutation in WWOX, implicated both in ASD and motor control. In addition, one of the affected children carries a rare deleterious de novo mutation in the ASD candidate gene RIMS1. The two affected children but not their unaffected sibling inherited deleterious variants with relevance for ASD and/or CAS. WWOX, RIMS1, and several of the genes harboring the inherited variants are expressed in the brain during prenatal and early postnatal development. Results suggest compound heterozygosity as a cause of ASD and CAS, pleiotropic gene effects, and potentially additional, complex genetic effects.

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.

Sequential processing deficit as a shared persisting biomarker in dyslexia and childhood apraxia of speech.

The purpose of this study was to investigate the hypothesis that individuals with dyslexia and individuals with childhood apraxia of speech share an underlying persisting deficit in processing sequential information. Levels of impairment (sensory encoding, memory, retrieval, and motor planning/programming) were also investigated. Participants were 22 adults with dyslexia, 10 adults with a probable history of childhood apraxia of speech (phCAS), and 22 typical controls. All participants completed nonword repetition, multisyllabic real word repetition, and nonword decoding tasks. Using phonological process analysis, errors were classified as sequence or substitution errors. Adults with dyslexia and adults with phCAS showed evidence of persisting nonword repetition deficits. In all three tasks, the adults in the two disorder groups produced more errors of both classes than the controls, but disproportionally more sequencing than substitution errors during the nonword repetition task. During the real word repetition task, the phCAS produced the most sequencing errors, whereas during the nonword decoding task, the dyslexia group produced the most sequencing errors. Performance during multisyllabic motor speech tasks, relative to monosyllabic conditions, was correlated with the sequencing error component during nonword repetition. The results provide evidence for a shared persisting sequential processing deficit in the dyslexia and phCAS groups during linguistic and motor speech tasks. Evidence for impairments in sensory encoding, short-term memory, and motor planning/programming was found in both disorder groups. Future studies should investigate clinical applications regarding preventative and targeted interventions towards cross-modal treatment effects.

Subjective Fatigue in Children With Hearing Loss Assessed Using Self- and Parent-Proxy Report.

PURPOSE: The primary purposes of this study were to examine the effects of hearing loss and respondent type (self- vs. parent-proxy report) on subjective fatigue in children. We also examined associations between child-specific factors and fatigue ratings. METHOD: Subjective fatigue was assessed using the Pediatric Quality of Life Inventory Multidimensional Fatigue Scale (PedsQL-MFS; Varni, Burwinkle, Katz, Meeske, & Dickinson, 2002). We compared self- and parent-proxy ratings from 60 children with hearing loss (CHL) and 43 children with normal hearing (CNH). The children ranged in age from 6 to 12 years. RESULTS: School-age CHL experienced more overall and cognitive fatigue than CNH, although the differences were smaller than previously reported. Parent-proxy report was not strongly associated with child self-report, and parents tended to underestimate their child's fatigue, particularly sleep/rest fatigue. Language ability was also associated with subjective fatigue. For CHL and CNH, as language abilities increased, cognitive fatigue decreased. CONCLUSIONS: School-age CHL experience more subjective fatigue than CNH. The poor association between parent-proxy and child reports suggests that the parent-proxy version of the PedsQL-MFS should not be used in isolation when assessing fatigue in school-age children. Future research should examine how language abilities may modulate fatigue and its potential academic consequences in CHL.

Two unrelated children with overlapping 6q25.3 deletions, motor speech disorders, and language delays.

Interstitial and terminal 6q25 deletions are associated with developmental delays, hypotonia, eye pathologies, craniofacial dysmorphologies, and structural brain anomalies. In most cases, speech and language deficits are not described in detail. We report on a case (Patient 1, age 7 years) with a de novo 6q25.3-qter deletion, 11.1 Mb long and encompassing 108 genes, and a case (Patient 2, age 5 years) with an inherited interstitial 6q25.3 deletion, located within Patient 1's deletion region and 403 kb long, the smallest 6q25 deletion reported to date. Both children have hypotonia, motor speech disorders, and expressive language delays. Patient 1's speech was characterized by childhood apraxia of speech (CAS) and dysarthria. Other findings include developmental delay, ataxic cerebral palsy, optic nerve dysplagia, and atypical brain morphologies regarding the corpus callosum and gyration patterns, a clinical profile that closely matches a previously reported case with a nearly identical deletion. Patient 2 had speech characterized by CAS and typical nonverbal processing abilities. His father, a carrier, had typical speech and language but showed difficulties with complex motor speech and hand motor tasks, similar to other adults with residual signs of CAS. The small deletion in this family contains the IGF2R-AIRN-SLC22A2-SLC22A3 gene cluster, which is associated with imprinting and maternal-specific expression of Igf2R, Slc22a2, and Slc22a3 in mice, whereas imprinting in humans is a polymorphic trait. The shared phenotypes in the two patients might be associated with the deletion of the gene cluster.