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Longitudinal Growth in Intelligibility of Connected Speech From 2 to 8 Years in Children With Cerebral Palsy: A Novel Bayesian Approach.

ABSTRACT: Aim The aim of the study was to examine longitudinal growth in intelligibility in connected speech from 2 to 8 years of age in children with cerebral palsy. Method Sixty-five children with cerebral palsy participated in the longitudinal study. Children were classified into speech-language profile groups using age-4 data: no speech motor impairment (SMI), SMI with typical language comprehension, and SMI with impaired language comprehension. We fit a Bayesian nonlinear mixed-effects model of intelligibility growth at the child and group levels. We compared groups by age of steepest growth, maximum growth rate, and predicted intelligibility at 8 years of age. Results The no SMI group showed earlier and steeper intelligibility growth and higher average outcomes compared to the SMI groups. The SMI groups had more variable growth trajectories, but the SMI with typical language comprehension group had higher age-8 outcomes and steeper rates of maximum growth than the SMI with impaired language comprehension group. Language comprehension impairment at age of 4 years predicted lower intelligibility outcomes at age of 8 years, compared to typical language at age of 4 years. Interpretation Children with SMI at age of 4 years show highly variable intelligibility growth trajectories, and comorbid language comprehension impairment predicts lower intelligibility outcomes. Supplemental Material https://doi.org/10.23641/asha.12777659.

SUBMITTER: Mahr TJ

PROVIDER: S-EPMC7890228 | biostudies-literature | 2020 Sep

REPOSITORIES: biostudies-literature

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Longitudinal Growth in Intelligibility of Connected Speech From 2 to 8 Years in Children With Cerebral Palsy: A Novel Bayesian Approach.

Mahr Tristan J TJ Rathouz Paul J PJ Hustad Katherine C KC

Journal of speech, language, and hearing research : JSLHR 20200812 9

Aim The aim of the study was to examine longitudinal growth in intelligibility in connected speech from 2 to 8 years of age in children with cerebral palsy. Method Sixty-five children with cerebral palsy participated in the longitudinal study. Children were classified into speech-language profile groups using age-4 data: no speech motor impairment (SMI), SMI with typical language comprehension, and SMI with impaired language comprehension. We fit a Bayesian nonlinear mixed-effects model of intel ...[more]

PMID: 32783783

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Project description:Purpose We examined whether there were differences among speech-language profile groups of children with cerebral palsy (CP) in age of crossing 25%, 50%, and 75% intelligibility thresholds; age of greatest intelligibility growth; rate of intelligibility growth; maximum attained intelligibility at 8 years; and how well intelligibility at 36 months predicts intelligibility at 96 months when group membership is accounted for. Profile groups were children with no speech motor impairment (NSMI), those with speech motor impairment and language comprehension that is typically developing (SMI-LCT), and those with speech motor impairment and language comprehension impairment (SMI-LCI). Method Sixty-eight children with CP were followed longitudinally between 24 and 96 months of age. A total of 564 time points were examined across children (M = 8.3 time points per child, SD = 2.6). We fitted a nonlinear random effects model for longitudinal observations, allowing for differences between profile groups. We used the fitted model trajectories to generate descriptive analyses of intelligibility growth by group and to generate simulations to analyze how well 36-month intelligibility data predicted 96-month data accounting for profile groups. Results Children with CP who have NSMI have different growth and better intelligibility outcomes than those with speech motor impairment. Children with SMI-LCT tend to have better outcomes but similar intelligibility growth as children with SMI-LCI. There may be a subset of children that cut across SMI-LCI and SMI-LCT groups who have severe speech motor involvement and show limited growth in intelligibility. Conclusions Intelligibility outcomes for children with CP are affected by profile group membership. Intelligibility growth tends to be delayed in children with speech motor impairment. Intelligibility at 3 years is highly predictive of later outcomes regardless of profile group. Intervention decision making should include consideration of early intelligibility, and treatment directions should include consideration of augmentative and alternative communication.

Project description:Cerebral palsy is primarily an upper motor neuron disease that results in a spectrum of progressive movement disorders. Secondary to the neurological lesion, muscles from patients with cerebral palsy are often spastic and form debilitating contractures that limit range of motion and joint function. With no genetic component, the pathology of skeletal muscle in cerebral palsy is a response to aberrant neurological input in ways that are not fully understood. This study was designed to gain further understanding of the skeletal muscle response to cerebral palsy using microarrays and correlating the transcriptional data with functional measures. Hamstring biopsies from gracilis and semitendinosus muscles were obtained from a cohort of patients with cerebral palsy (n=10) and typically developing patients (n=10) undergoing surgery. Affymetrix HG-U133A 2.0 chips (n=40) were used and expression data was verified for 6 transcripts using quantitative real-time PCR, as well as for two genes not on the microarray. Chips were clustered based on their expression and those from patients with cerebral palsy clustered separately. Significant genes were determined conservatively based on the overlap of three summarization algorithms (n=1,398). Significantly altered genes were analyzed for over-representation among gene ontologies, transcription factors, pathways, microRNA and muscle specific networks. These results centered on an increase in extracellular matrix expression in cerebral palsy as well as a decrease in metabolism and ubiquitin ligase activity. The increase in extracellular matrix products was correlated with mechanical measures demonstrating the importance in disability. These data lay a framework for further studies and novel therapies. Skeletal muscle biopsies from both the gracilis and semitendinosus were obtained during surgery for 20 pediatric subjects for affymetrix microarray analysis. We obtained a group of 10 patients undergoing medial hamstring lengthening in the cerebral palsy group and 10 patients undergoing ACL reconstruction with hamstring autograft in the control group. This provided 40 microarrays in 4 groups to analyze the effect of cerebral palsy and also differences between muscles.

Project description:ObjectivesTo test the feasibility of recruitment, retention, outcome measures and internet delivery of dysarthria therapy for young people with cerebral palsy in a randomised controlled trial.DesignMixed methods. Single blind pilot randomised controlled trial, with control offered Skype therapy at end of study. Qualitative study of the acceptability of therapy delivery via Skype.SettingNine speech and language therapy departments in northern England recruited participants to the study. Skype therapy was provided in a university setting.ParticipantsTwenty-two children (14 M, 8 F) with dysarthria and cerebral palsy (mean age 8.8 years (SD 3.2)) agreed to take part. Participants were randomised to dysarthria therapy via Skype (n=11) or treatment as usual (n=11).InterventionsChildren received either usual speech therapy from their local therapist for 6?weeks or dysarthria therapy via Skype from a research therapist. Usual therapy sessions varied in frequency, duration and content. Skype dysarthria therapy focused on breath control and phonation to produce clear speech at a steady rate, and comprised three 40?min sessions per week for 6?weeks.Primary and secondary outcome measuresFeasibility and acceptability of the trial design, intervention and outcome measures.ResultsDepartments recruited two to three participants. All participants agreed to random allocation. None withdrew from the study. Recordings of children's speech were made at all time points and rated by listeners. Families allocated to Skype dysarthria therapy judged internet delivery of the therapy to be acceptable. All families reported that the study design was acceptable. Treatment integrity checks suggested that the phrases practised in one therapy exercise should be reduced in length.ConclusionsA delayed treatment design, in which dysarthria therapy is offered at the end of the study to families allocated to treatment as usual, is acceptable. A randomised controlled trial of internet delivered dysarthria therapy is feasible.

Project description:BackgroundChildren with cerebral palsy (CP) often have communication impairments, including speech altered intelligibility. Multiple levels of disrupted speech have been reported in CP, which negatively impact on participation and quality of life, with increase of care needs. Augmentative Alternative Communication (AAC) is an option, with debated benefits and limitations, in particular for its functional use. This is supported by a substantial lack of defined evidences in favor of direct speech articulation intervention in CP. Motor learning-based interventions are effective in CP and are the basis of speech motor interventions such as PROMPT (Prompts for Restructuring Oral Muscular Phonetic Targets). The PROMPT speech motor treatment provides tactile-kinesthetic inputs to facilitate articulatory movements by dynamic modelling, resulting in more efficient motor patterns that can be integrated into speech and communication. In CP, exploratory evidences support the feasibility and preliminarily advantages on intelligibility of motor speech treatments, such as PROMPT, with increased speech motor control, also documented by kinematic analyses.MethodsA randomized waitlist-control trial will be conducted in children aged between 3- and 10-years having CP and dysarthria (estimated sample size = 60 children). Children will be allocated in the immediate intervention or in the waitlist control group. The intervention consists of an intensive 3 weeks period of twice-a-day administration of PROMPT. Standard care will be administered in the control (waitlist) group. After repeated baseline assessments (T0), the PROMPT treated group will undergo the experimental 3-week intervention period, with T1 assessment at the end. A further T2 assessment will be provided at medium term (3 months after the end of the intervention) for evaluating the stability of intervention. Primary and secondary speech clinical and kinematics outcome measures will be collected at T0, T1 and T2.DiscussionThis paper describes the study protocol consisting of a RCT with two main objectives: (1) to evaluate the or short-term benefits of an intensive speech motor intervention on speech and intelligibility in children with CP and the stability of the intervention at medium term; (2) to describe the kinematic correlates of speech motor control modifications.Trial registrationTrial registration date 06/12/2019; ClinicalTrials.gov Identifier: NCT04189159 .

Dataset Information

Longitudinal Growth in Intelligibility of Connected Speech From 2 to 8 Years in Children With Cerebral Palsy: A Novel Bayesian Approach.

Publications

Longitudinal Growth in Intelligibility of Connected Speech From 2 to 8 Years in Children With Cerebral Palsy: A Novel Bayesian Approach.

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