Paediatrics (RCH) - Research Publications
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ItemNo Preview AvailableArticulation or phonology? Evidence from longitudinal error data(TAYLOR & FRANCIS INC, 2018)Children's speech difficulties can be motor (phone misarticulation) or linguistic (impaired knowledge of phonological contrasts and constraints). These two difficulties sometimes co-occur. This paper reports longitudinal data from the Early Language in Victoria Study (ELVS) at 4 and 7 years of age. Of 1494 participants, 93 made non-age appropriate speech errors on standardised assessments at 4 years, and were able to be reassessed at 7 years. At 4 years, 85% of these children only made phonological errors, 14% made both articulation and phonological errors and one child only made articulation errors (a lateral lisp). In total, 8 of 13 children making both articulation and phonological errors at 4 years had resolved by 7 years. Unexpectedly, eight children who had demonstrated articulation of fricatives at 4 years, acquired distorted production of ≥ 50% of occurrences of/s, z/ by 7 years. In total, then, 22 children (24% of children with speech difficulties) made articulatory errors at one or both assessments. Case data for all children are presented. Theoretical and clinical implications are considered.
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ItemAtypical Callosal Morphology in Children with Speech Sound Disorder(PERGAMON-ELSEVIER SCIENCE LTD, 2017-12-26)Speech sound disorder (SSD) is common, yet its neurobiology is poorly understood. Recent studies indicate atypical structural and functional anomalies either in one hemisphere or both hemispheres, which might be accompanied by alterations in inter-hemispheric connectivity. Indeed, abnormalities of the corpus callosum - the main fiber tract connecting the two hemispheres - have been linked to speech and language deficits in associated disorders, such as stuttering, dyslexia, aphasia, etc. However, there is a dearth of studies examining the corpus callosum in SSD. Here, we investigated whether a sample of 18 children with SSD differed in callosal morphology from 18 typically developing children carefully matched for age. Significantly reduced dimensions of the corpus callosum, particularly in the callosal anterior third, were observed in children with SSD. These findings indicating pronounced callosal aberrations in SSD make an important contribution to an understudied field of research and may suggest that SSD is accompanied by atypical lateralization of speech and language function.
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ItemExploring the speech and language of individuals with non-syndromic submucous cleft palate: a preliminary report(WILEY, 2019-09)BACKGROUND: Submucous cleft palate (SMCP) has a heterogeneous presentation and is often identified late or misdiagnosed. Diagnosis is prompted by speech, resonance or feeding symptoms associated with velopharyngeal insufficiency. However, the broader impacts of SMCP on communication have rarely been examined and therefore are poorly understood. AIM: To describe the communicative profile of individuals with non-syndromic SMCP by examining speech, language and pragmatics (social language). METHODS & PROCEDURES: Fifteen participants with SMCP aged 5;1-12;8, without a genetic diagnosis, participated in the study. Participants completed standardized assessments examining language, resonance, speech and non-verbal intellect. Parents also completed the Children's Communication Checklist (CCC-2), which provided a measure of overall communicative ability, including pragmatic skills. Formal language outcomes were compared with two cohorts: 36 individuals with overt non-syndromic clefts and 129 individuals with no history of clefting. OUTCOMES & RESULTS: Speech intelligibility was reduced secondary to hypernasality, disordered articulation and/or impaired phonology (n = 7) in children with SMCP. Poorer overall language outcomes were observed for children with SMCP compared with both those with overt clefts and no history of clefting (p < 0.001). Language scores for children with SMCP ranged from impaired (n = 6) to above the standardized mean (n = 4). Receptive and expressive language performance were independently correlated with non-verbal IQ (p < 0.01). Those with severe language impairment (n = 4) also had borderline or impaired non-verbal IQ. Parents reported that speech and semantics were the most affected sub-domains of communication, while scores were the highest for the initiation domain. Speech and language skills were correlated strongly with pragmatics (r = 0.877, p < 0.01). CONCLUSIONS & IMPLICATIONS: Overall, performance was variable within the SMCP group across speech, language and pragmatic assessments. In addition to well-documented speech difficulties, children with SMCP may have language or pragmatic impairments, suggesting that further neurodevelopmental influences may be at play. As such, for individuals with SMCP, additional clinical screening of language and pragmatic abilities may be required to ensure accurate diagnosis and guide both cleft and non-cleft related therapy programmes.
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ItemSpeech and language in bilateral perisylvian polymicrogyria: a systematic review(WILEY, 2019-10)AIM: We aimed to systematically review the speech production, language, and oral function phenotype of bilateral perisylvian polymicrogyria (BPP), and examine the correlation between the topography of polymicrogyria and the severity of speech, language, and oral functional impairment. METHOD: A systematic search of MEDLINE, Embase, and PubMed databases was completed on 26th October 2017 using Medical Subject Heading terms synonymous with BPP and speech, language, or oral motor impairment. In total, 2411 papers were identified and 48 met inclusion criteria. RESULTS: Expressive and receptive language impairment and oral structural and functional deficits are frequent in BPP. Expressive deficits are frequently more severe than receptive. Only one study used formal assessments to demonstrate the presence of speech disorder, namely dysarthria. Seven studies reported an association between diffuse BPP and more severe language impairment. INTERPRETATION: Findings confirmed that language deficits are common in BPP, though assessment of the specific speech phenotype is limited. The paucity of high quality studies detailing the specific communication phenotype of BPP highlights the need for further investigation. Improving understanding of this phenotype will inform the development of targeted therapies and lead to better long-term outcomes. WHAT THIS PAPER ADDS: Speech, language, and oral functional impairments are common in individuals with bilateral perisylvian polymicrogyria. Posterior polymicrogyria is associated with a less severe language impairment than anterior polymicrogyria. Deeper investigation of speech is needed to understand implicated networks in this malformation.
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ItemCharacterization of speech and language phenotype in children with NRXN1 deletions(WILEY, 2018-12)Neurexin 1 gene (NRXN1) deletions are associated with several neurodevelopmental disorders. Communication difficulties have been reported, yet no study has examined specific speech and language features of individuals with NRXN1 deletions. Here, we characterized speech and language phenotypes in 21 children (14 families), aged 1.8-17 years, with NRXN1 deletions. Deletions ranged from 74 to 702 kb and consisted mostly of either exons 1-3 or 1-5. Speech sound disorders were frequent (69%), although few were severe. The majority (57%) of children had difficulty with receptive and/or expressive language, although no homogeneous profiles of deficit were seen across semantic, morphological, or grammatical systems. Social language difficulties were seen in over half the sample (53%). All but two individuals with language difficulties also had intellectual disability/developmental delay. Overall, while speech and language difficulties were common, there was substantial heterogeneity in the severity and type of difficulties observed and no striking communication phenotype was seen. Rather, the speech and language deficits are likely part of broader concomitant neurodevelopmental profiles (e.g., intellectual disability, social skill deficits). Nevertheless, given the high rate of affectedness, it is important speech/language development is assessed so interventions can be applied during childhood in a targeted and timely manner.
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ItemSpeech and language characteristics in individuals with nonsyndromic submucous cleft palate-A systematic review(WILEY, 2018-11)BACKGROUND: Up to 80% of individuals with unrepaired submucous cleft palate (SMCP) experience speech difficulties secondary to velopharyngeal insufficiency. Language delays are reported in the broader cleft lip and/or palate population, suggesting that individuals with SMCP may also be at risk. However, contemporary understanding of this population remains limited as there has been no systematic examination of the literature. This review aims to systematically review and document the speech and language characteristics of individuals with nonsyndromic SMCP and, in addition, to identify factors reported to impact speech and language outcomes. METHOD: This review followed the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines. Five databases were comprehensively searched using keywords and indexed headings. Included studies had to report speech or language outcomes of individuals with nonsyndromic SMCP. Risk of bias and methodological design quality were examined using tools from the Scottish Intercollegiate Guidelines Network. Relevant data were extracted for analysis. RESULTS: Eighteen studies met inclusion criteria, yielding 598 participants. Study results showed that individuals with unrepaired nonsyndromic SMCP may have speech difficulties secondary to velopharyngeal insufficiency including increased nasal resonance and palatalized or glottal articulation. Lower age at primary surgical repair led to better postsurgical speech outcomes. There is a paucity of literature outlining motor or phonological aspects of speech and receptive or expressive language abilities of this population. CONCLUSION: Individuals with nonsyndromic SMCP present with speech difficulties similar to those experienced by individuals with overt cleft palate. Health care professionals should be aware of possible presenting symptoms and consider early SMCP diagnoses where appropriate. Further research is needed to specify the broader communication profile in this population.
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ItemValidation of Dodd's Model for Differential Diagnosis of childhood speech sound disorders: a longitudinal community cohort study(WILEY, 2019-06)AIM: Dodd's Model for Differential Diagnosis is one of the available clinical diagnostic classification systems of childhood speech sound disorders. Yet we do not understand the validity of this system beyond clinical samples, precluding its application in epidemiological or population-based research. This study aimed to determine the prevalence of subgroups of speech sound disorders in a community sample, relative to past clinical samples, in children speaking standard Australian English. METHOD: We examined speech development in a community-ascertained sample of children at 4 years (n=1607). Inclusion for speech sound disorder was a score of less than or equal to 1 standard deviation on a standardized speech test, and/or research assistant concern, and/or three or more speech errors on sounds typically acquired by 4 years. Dodd's model was then applied to 126 children. RESULTS: Data revealed proportions of children across Dodd's diagnostic subgroups as follows: suspected atypical speech motor control (10%); inconsistent phonological disorder (15%); consistent atypical phonological disorder (20%); phonological delay (55%); and articulation disorder alone (0%). The findings are in line with known prevalence of these subgroups in clinical populations. INTERPRETATION: Our findings provide additional support for speech-language pathologists to use this system in clinical practice for differential diagnosis and targeted intervention of speech sound disorders in children. WHAT THIS PAPER ADDS: Dodd's Model for Differential Diagnosis is the first classification system of speech sound disorders to be applicable to both clinical and community cohorts.
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ItemAltered gray matter volumes in language-associated regions in children with developmental language disorder and speech sound disorder(WILEY, 2018-11)Developmental language disorder (DLD) and speech sound disorder (SSD) are common, and although scientific evidence for structural and functional alterations in DLD/SSD is accumulating, current neuroimaging studies provide an incongruent picture. Here, we hypothesized that children affected by DLD and SSD present with gray matter (or gray matter asymmetry) aberrations in brain areas associated with language processing compared to typically developing (TD) children. To assess this hypothesis, we enhanced MRI-based information with microscopically defined cytoarchitectonic probabilities of Broca's area (BA 45, BA 44) as well as an auditory area (TE 3.0). We detected a larger rightward gray matter asymmetry in BA 45 in children with DLD (n = 13) and with SSD (n = 18) compared to TD children (n = 18), albeit only on a trend level. Interestingly though, we observed significantly larger gray matter volumes in right BA 45 in DLD compared to SSD children (and also compared to TD children).
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ItemReceptive and expressive language characteristics of school-aged children with non-syndromic cleft lip and/or palate(WILEY, 2018-09)BACKGROUND: Research investigating language skills in school-aged children with non-syndromic cleft lip and/or palate is sparse. Past studies focus on younger populations, lack key comparisons to demographically matched control cohorts or explore language as a component of broader academic skills. Trends of existing studies suggest that affected children may perform at a lower level compared with typically developing peers. AIMS: To examine the receptive and expressive language skills of middle-school-aged children with non-syndromic cleft lip and palate (CLP) and cleft palate only (CP). Additionally, to explore the language skills of children with clefts compared with a non-cleft control group. METHODS & PROCEDURES: Thirty-seven participants with orofacial clefts (aged 7;1-14;1 years) participated in the study: 19 with CLP (10 males; 9 females) and 18 with CP (8 males; 10 females). A non-cleft comparison group consisted of 129 individuals matched on age, sex and maternal education level. Participants completed formal language (Clinical Evaluation of Language Fundamentals, Fourth Edition) and non-verbal intellectual measurements (Wechsler Abbreviated Scale of Intelligence-WASI). Demographic and developmental information was obtained via parental interview. Further clinical details (e.g., surgery; hearing status) were extracted from patient medical files. Cleft and non-cleft language and non-verbal IQ outcomes were reported separately. Language outcomes were then compared between groups. OUTCOMES & RESULTS: Participants with clefts achieved core (mean = 103.31, standard deviation (SD) = 10.31), receptive (mean = 102.51, SD = 11.60) and expressive (mean = 102.89, SD = 12.17) language index scores within the normative average range. A total of 14.1% and 17.8% of the cleft and non-cleft groups respectively had impairment (i.e., ≥ 1.25 SD below the mean) in one or more language domains. No significant differences were found in the three language index scores between cleft and non-cleft groups. CONCLUSIONS & IMPLICATIONS: This study is the first formally to examine language skills alongside non-verbal IQ in school-aged children with clefts compared with a large matched non-cleft population. Results suggest that health professionals should evaluate each child as they present and not assume that a child with non-syndromic CLP or CP will also have co-occurring language difficulties. Where language falls in the average range, these skills can be harnessed to support areas of difficulty often associated with orofacial clefting, such as speech.
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ItemWhite matter microstructure is associated with language in children born very preterm(ELSEVIER SCI LTD, 2018)Very preterm birth is associated with altered white matter microstructure and language difficulties, which may compromise communication, social function and academic achievement, but the relationship between these two factors is unclear. The aim of this study was to explore associations between white matter microstructure and language domains of semantics, grammar and phonological awareness at 7-years of age on a whole-brain level and within the arcuate fasciculus, an important language pathway, in very preterm and term-born children. Language was assessed in 145 very preterm-born (<30 weeks' gestation and/or <1250 g birth weight) and 33 term-born children aged 7 years. Fractional anisotropy (FA), axial diffusivity (AD), radial diffusivity (RD), mean diffusivity (MD), axon orientation dispersion and axon density were estimated from diffusion magnetic resonance images also obtained at 7 years. The correlation between diffusion values and language was assessed using Tract-Based Spatial Statistics (TBSS). The arcuate fasciculus was delineated using constrained spherical deconvolution tractography and diffusion parameters from this tract were related to language measures using linear regression. While there was evidence for widespread associations between white matter microstructure and language, there was little evidence of differences in these associations between very preterm and term-born groups. TBSS analyses revealed that higher FA and lower AD, RD, and MD in major fibre tracts, including those subserving language, were associated with better semantic, grammar and phonological awareness performance. Higher axon density in widespread fibre tracts was also associated with better semantic performance. The tractography analyses of the arcuate fasciculus showed some evidence for associations between white matter microstructure and language outcomes. White matter microstructural organisation in widespread fibre tracts, including language-relevant pathways, was associated with language performance in whole-brain and tract-based analyses. The associations were similar for very preterm and term-born groups, despite very preterm children performing more poorly across language domains.