Paediatrics (RCH) - Theses
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ItemDevelopment and validation of the short language measure (SLaM): a brief measure of general language ability for children in their first year at school( 2018)The identification of children with language difficulties relies on the referral of children to speech-language services. Yet, the referrers of young school-aged children cannot always accurately discriminate those with and without language difficulties. Consequently, a high proportion of children with language difficulties are never referred for services and many children referred for suspected language difficulties have age-appropriate communication skills. The administration of a short language measure by speech-language therapists or paraprofessionals to identify children who require further language evaluation could improve the accuracy of language service referrals. However, existing short language measures for young school-aged children lack sufficient discrimination capacity. This may be due to the inadequate development of available measures, which have omitted crucial test development steps. The aim of this project was to create a sufficiently accurate, valid and reliable short language measure for children in their first year at school by applying stringent test development procedures.
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ItemMild and moderate congenital hearing loss in childhood: trends and associations with language outcomes( 2018)Background: Children born with mild and moderate bilateral hearing loss experience poorer language outcomes than peers without hearing loss. Following implementation of universal newborn hearing screening (UNHS) in the early 2000s, most children are now fitted with hearing aids very early, even without firm evidence of efficacy. Filling this knowledge gap would support clinicians and families to make appropriate decisions for affected children. Aims: In population-based samples of children born with mild and moderate bilateral hearing loss in two age groups, I aimed to describe the: 1) language and child wellbeing outcomes at 5-8 years of age examined both by system of hearing loss detection and age at detection; 2) relationship between measures of hearing ability (aided and unaided) and speech/language outcomes at 5-7 years of age; and 3) expressive vocabulary of children at age 1-3 years, with comparison to same-aged children without hearing loss. Methods: All participants were drawn from population-based studies or databanks with comparable measures and demographic composition in Victoria and New South Wales. In Aim 1, 21 directly assessed children (5-7 years) were analysed with 125 children (5-8 years) from historical hearing loss cohorts and 1217 children (7 years) without known hearing loss. For Aim 2, hearing aid data were obtained for 19 directly assessed children. In Aim 3, questionnaire data were compared between 20 children (1-3 years) with and 1711 children (2 years) without known hearing loss. Language was assessed or parent-reported, with additional data collected by parent questionnaire. Aim 1 was analysed using unadjusted and adjusted regression models, with potential confounders identified a priori. For Aims 2 and 3, analyses were descriptive and exploratory. Results: Outcomes for 5-7 year olds with moderate loss improved across detection systems spanning opportunistic through to UNHS (mean expressive language p for trend .05, receptive vocabulary p for trend .06) and were better when diagnosed before age 6 months. Children with mild loss did not show similar benefit, despite trends toward earlier diagnosis and hearing aid fitting. Children with mild-moderate loss had poorer expressive language than children without loss (adjusted mean difference -8.9 points, 95% CI -14.7 to -3.1). Audibility measures correlated with unaided hearing (r=-.60 to -.79, p=.02 to <.001) and speech recognition ability (r=-.63 to -.73, p=.04 to .01). Unaided and aided hearing measures did not show associations with speech and language. On average 1-3 year olds with, versus without, hearing loss had lower expressive vocabulary raw scores even though older at assessment. Language of children with mild loss was on average 9 months behind expectations (SD 5.6 months), moderate loss on average 6.6 months behind (SD 6.7 months). Conclusions: Children with mild and moderate hearing loss show early language delays that persist into primary school. Children with moderate loss showed some benefit from earlier detection and fitting with hearing aids. Children with mild loss did not, which is concerning given the current practice of earlier hearing aid fitting. A population-based trial of hearing aids for mild losses would clarify the benefits versus costs of this approach.
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ItemLanguage development in autism spectrum disorder: longitudinal comparison with a community cohort of children with language impairment and typical development( 2016)Background: Autism spectrum disorder (ASD) is a lifelong developmental disability that affects more than 1 in 50 children in Australia. Language difficulties are common in ASD with up to 30% of children unable to communicate using words. Despite a substantial number of studies examining language outcomes in individuals with ASD, it is difficult for families and clinicians to interpret study findings. This is because many studies use; selected clinical samples that may not generalise to the broader population, heterogeneous methodology, and the quality of studies varies. Rarely do studies use standardised language-specific tools. In addition, few studies have compared development in children with ASD to children without ASD, which means it is difficult to place differences in children with ASD within a developmental context. As a consequence, we do not yet fully understand how language develops in children with ASD and we are not able to accurately predict language outcomes. Parents and clinicians need evidence about language trajectories in ASD to inform decision-making and understand prognosis. Service providers and policy makers also require information for appropriate resource allocation for current and future needs. Aims: The overall aim of this thesis was to examine trajectories of language development in individuals diagnosed with ASD. To achieve this aim, three main studies were conducted. Study 1 systematically reviewed and synthesised the extant literature on language outcomes in individuals with ASD. Studies 2 and 3 examined individual and mean trajectories of language development in children with ASD from 1 to 2 years and 4 to 7 years, respectively, and compared these trajectories to large samples of children with language impairment (LI) and typical language development (TD). Study 3 described language trajectories for children with ASD and investigated predictors of language outcomes from 4 to 7 years in children with and without ASD. Method: Children in studies 2 and 3 were recruited from a prospective longitudinal community-based study of 1910 children in Victoria (the Early Language in Victoria Study; ELVS). Individual and mean trajectories were mapped from 1 to 2 years (ASD; n=41, LI: n=119, TD: n=861) and 4 to 7 years (ASD; n=27, LI n=110, TD: n=831). In Study 2, individual and mean communication trajectories were mapped using scores from two parent checklists, namely the Communication Symbolic Behaviour Scales Infant Toddler Checklist and the MacArthur Bates Communicative Development Inventories. We compared the proportion of children who lost specific communication skills between the three groups (ASD, LI, TD) and the spread of loss across different communication domains. In Study 3 we used the Clinical Evaluation of Language Fundamentals data collected in ELVS (preschool second edition and fourth edition) to map individual and mean language trajectories from 4 to 7 years. The proportion of children who had declined, remained stable or accelerated in language skills was compared across groups. Putative predictors of language outcomes were also investigated. Results: Language ability was heterogeneous, however, mean scores for children with ASD and LI were lower than scores for children with TD and reference norms, in all studies included in the systematic review and Studies 2 and 3 in the community sample. From 1 to 2 years, the gap between children with ASD and TD/LI grew larger in all communication domains except in the areas of speech and expressive vocabulary, which was similar for children with LI and ASD. From 4-7 years, despite having lower language ability on average compared with the typically developing group, most children with ASD were developing language at the same pace as the LI and TD children. A diagnosis of ASD did not predict a greater gap between receptive and expressive language ability. The child’s early language ability and IQ were most important in predicting language ability at a later age. Conclusion: Systematic review and synthesis of existing studies showed that children with ASD in all studies (with one exception) had lower scores at baseline when compared with reference norms but children tracked in parallel to reference norms. The limited amount of data available from studies investigating children over 9 years made it difficult to draw accurate conclusions on trajectories beyond this age, however the few studies that had presented data suggest rate of language progress may slow from around 10 years. In Studies 2 and 3 that utilised the ELVS, children with ASD demonstrated communication abilities that were not significantly different to other children at12 months of age in most areas. However, on average their language and social communication progressed at a slower rate than other children from around 12 months to 2 years in most areas of communication. Findings from children in the ELVS aged 4 to 7 years were consistent with the systematic review findings. In Study 3 children with ASD who were verbal and had IQ in the normal range demonstrated lower language scores at baseline and follow up on average, but tracked in parallel to reference norms over time. The knowledge gained from this thesis will help guide prognostic information to be provided to parents. It will also assist with planning for future support needs of individuals with ASD.
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ItemSpeech and language abilities of children with cerebral palsy: a population-based study( 2014)Although speech and language development are known to be vulnerable in children with cerebral palsy (CP), little is known about the prevalence and features of impairments in these areas during early childhood. Speech and language are known to influence the activity and participation of children with CP, yet no population-based study has examined the contribution of speech versus language. Activity and participation are recognised as key health outcomes, however, at the time of this research, no tool measuring these functional areas existed specifically for children with CP with communication impairment. This study consisted of three parts: (1) a qualitative study exploring the activity and participation of children with CP to provide the basis of a questionnaire measuring these constructs; (2) development and piloting of the questionnaire; and (3) a population-based study examining the speech and language outcomes of children with CP aged 5 and 6 years at an impairment and activity/participation level. In Part 1, activity and participation were explored by interviewing 13 parents of children with CP. Parents’ responses were thematically analysed to reveal key areas to include in a structured questionnaire of activity and participation. Children’s reported activities and participation were related to: (1) leisure, early learning and educational activities (e.g., reading, classroom activities); (2) communication (e.g., asking questions, expressing needs); (3) independence within the home (e.g., activities of daily living, household chores); and (4) engagement within the community (e.g., shopping, visiting family). Barriers affecting children’s activity and participation included aspects of parents’ own interactions with their child (e.g., not offering choices), unfamiliar people and settings, the negative attitudes of others, and the child’s frustration. Facilitators included the support received from the child’s family and school, being amongst children, having a familiar routine, and the child’s positive disposition (e.g., being determined). These qualitative findings informed the development of an activity and participation questionnaire (Part 2). In Part 2, questionnaire items were devised based on the codes identified during the thematic analysis of the qualitative data. The developed items focused on children’s activity and participation within the home, school and community, and the environmental and personal factors affecting these areas of functioning. Face validity of the questionnaire was determined by verifying that the items adequately described and measured the most relevant aspects of the underlying construct. Content validity was established through expert opinion of the questionnaire’s items. The questionnaire was piloted on seven parents of children with CP and was modified accordingly prior to its use in the population-based study. In Part 3, 84 children with CP born between August 2005 and August 2007 were recruited through the Victorian Cerebral Palsy Register. This represented 37% of the known living population of this age range. Participants were representative of non-participants across key variables (e.g., CP type and distribution, Gross Motor Function Classification level, cognitive status). Children underwent a comprehensive face-to-face assessment. The primary outcome measures were the Diagnostic Evaluation of Articulation and Phonology, the Preschool Language Scale-4, and the developed activity and participation questionnaire. The internal consistency of the questionnaire was examined during this population-based study and was found to be good-to-excellent for items relating to activity and participation. Findings from the population-based study revealed that 80% (66/83) of the sample had some form of speech and/or language difficulty (24%, 20/84, were non-verbal). Speech delay/disorder and language impairment were identified in 66% (40/61) and 62% (51/82) of children, respectively. The subgroups of language impairment identified were isolated receptive (7%, 6/82) or expressive (5%, 4/82), and mixed receptive-expressive (45%, 37/82). Speech delay/disorder consisted of the following subgroups: articulation delay (26%, 16/61), articulation disorder (28%, 17/61), phonological delay (17%, 10/60), consistent phonological disorder (7%, 4/58), and inconsistent phonological disorder (19%, 11/58). Children at most risk for speech delay/disorder and/or language impairment were those with mixed or non-spastic CP motor types, quadriplegia, severe fine motor limitations, epilepsy, hearing, cognitive or vision impairment, and those born with an extremely low birth weight. Non-verbal children and those with language impairment were at the greatest risk for restrictions in activity and participation compared to children with age appropriate speech and language and those with speech delay/disorder in the absence of language impairment. Activity and participation were best facilitated by positive child attributes and the support received from others, whereas barriers included communication partners (e.g., their negative attitudes or not understanding the child) and the difficulty parents experienced in supporting their child. Study findings highlight that the majority of children with CP experience some form of speech and/or language difficulty at 5 and 6 years of age, underscoring the need to routinely assess and monitor these areas. The subtypes of speech delay/disorder and language impairment identified provide suggestions for how to characterise speech and language in children with CP. Findings indicate that activity and participation-based therapy may not be warranted for children with speech delay/disorder who have age appropriate language skills. To confirm this, further research is needed comparing the activity and participation outcomes of this subgroup against typically developing children. The environmental and personal factors that were found to have the strongest impact on children’s activity and participation may provide suggestions for how to optimise activity and participation through intervention. For instance, the effectiveness of reinforcing positive child attributes and counselling parents on early communicative strategies that encourage children to indicate their needs may be trialled in future research to determine their clinical utility in enhancing activity and participation outcomes.