Erratum to "Comorbidity and Severity in Childhood Apraxia of Speech: A Retrospective Chart Review".
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PROVIDER: S-EPMC10715840 | biostudies-literature | 2023 Nov
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Erratum to "Comorbidity and Severity in Childhood Apraxia of Speech: A Retrospective Chart Review".
Journal of speech, language, and hearing research : JSLHR 20231023 11
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Project description:PurposeThe purpose of this study was to investigate comorbidity prevalence and patterns in childhood apraxia of speech (CAS) and their relationship to severity.MethodIn this retroactive cross-sectional study, medical records for 375 children with CAS (M age = 4;9 [years;months], SD = 2;9) were examined for comorbid conditions. The total number of comorbid conditions and the number of communication-related comorbidities were regressed on CAS severity as rated by speech-language pathologists during diagnosis. The relationship between CAS severity and the presence of four common comorbid conditions was also examined using ordinal or multinomial regressions.ResultsOverall, 83 children were classified with mild CAS; 35, with moderate CAS; and 257, with severe CAS. Only one child had no comorbidities. The average number of comorbid conditions was 8.4 (SD = 3.4), and the average number of communication-related comorbidities was 5.6 (SD = 2.2). Over 95% of children had comorbid expressive language impairment. Children with comorbid intellectual disability (78.1%), receptive language impairment (72.5%), and nonspeech apraxia (37.3%; including limb, nonspeech oromotor, and oculomotor apraxia) were significantly more likely to have severe CAS than children without these comorbidities. However, children with comorbid autism spectrum disorder (33.6%) were no more likely to have severe CAS than children without autism.ConclusionsComorbidity appears to be the rule, rather than the exception, for children with CAS. Comorbid intellectual disability, receptive language impairment, and nonspeech apraxia confer additional risk for more severe forms of CAS. Findings are limited by being from a convenience sample of participants but inform future models of comorbidity.Supplemental materialhttps://doi.org/10.23641/asha.22096622.
| S-EPMC10205100 | biostudies-literature
Project description:PurposeThe purpose of this study was to investigate the association between perceived single-word speech severity and intelligibility in children with childhood apraxia of speech (CAS), with and without comorbid language impairment (LI), and to investigate the contribution of different CAS signs to perceived single-word speech severity and single-word intelligibility.MethodThirty children with CAS, 18 with comorbid LI, completed the Goldman-Fristoe Test of Articulation-Second Edition (GFTA-2). Trained judges coded children's responses for signs of CAS and percent phonemes correct. Nine listeners, blind to diagnoses, rated speech severity using a visual analog scale. Intelligibility was assessed by comparing listeners' orthographic transcriptions of children's responses to target responses.ResultsMeasures of speech severity (GFTA-2 standard score, number of unique CAS signs, total CAS signs, and mean severity rating) were significantly correlated with measures of intelligibility (GFTA-2 raw score, percent phonemes correct, and mean intelligibility score). Speech severity and intelligibility did not differ significantly between children with and without LI. Only consonant errors contributed significant variability to speech severity. Consonant errors and stress errors contributed significant variability to intelligibility.ConclusionsFindings suggest that visual analog scale ratings are a valid and convenient measure of single-word speech severity and that GFTA-2 raw score is an equally convenient measure of single-word intelligibility. The result that consonant errors were by far the major contributor to single-word speech severity and intelligibility in children with CAS, with stress errors also making a small contribution to intelligibility, suggests that consonant accuracy and appropriate lexical stress should be prime therapeutic targets for these children in the context of treatment addressing motor planning/programming, self-monitoring, and self-correcting.Supplemental materialhttps://doi.org/10.23641/asha.19119350.
| S-EPMC9150686 | biostudies-literature
Project description:Event-related potentials (ERPs) were recorded during a picture naming task of simple and complex words in children with typical speech and with childhood apraxia of speech (CAS). Results reveal reduced amplitude prior to speaking complex (multisyllabic) words relative to simple (monosyllabic) words for the CAS group over the right hemisphere during a time window thought to reflect phonological encoding of word forms. Group differences were also observed prior to production of spoken tokens regardless of word complexity during a time window just prior to speech onset (thought to reflect motor planning/programming). Results suggest differences in pre-speech neurolinguistic processes.
| S-EPMC4130155 | biostudies-literature
Project description:PurposeTo investigate the latent factors underlying signs of childhood apraxia of speech (CAS) in a group of 57 children with CAS.MethodThe speech of 57 children with CAS (aged 3;5-17;0) was coded for signs of CAS. All participants showed at least five signs of CAS and were judged to have CAS by speech pathologists experienced in pediatric speech disorders. Participants were selected to represent a range of severity of CAS: 30 children were verbal and 27 were minimally verbal with comorbid autism. Participants' scores for each sign (the number of times that sign appeared during a child's speech sample) were converted to z-scores, then entered as variables into an exploratory factor analysis. Models were compared using the Akaike Information Criterion (AIC).ResultsThe three-factor model had the lowest AIC and best fit the data. After oblique rotation, syllable segmentation, slow rate, and stress errors loaded most highly on Factor 1. Groping, addition of phonemes other than schwa, and difficulty with coarticulation loaded most highly on Factor 2. Variable errors loaded most highly on Factor 3. Thus, factors were interpreted as being associated with (1) prosody, (2) coarticulation, and (3) inconsistency.ConclusionsResults are consistent with the three consensus criteria for CAS from the American Speech-Language-Hearing Association: Inappropriate prosody, disrupted coarticulatory transitions, and inconsistent errors on repeated tokens. High loading of the syllable segmentation sign on the inappropriate prosody factor also supports the use of a pause-related biomarker for CAS.
| S-EPMC7494519 | biostudies-literature
Project description:Childhood apraxia of speech (CAS) is a paediatric speech sound disorder in which precision and consistency of speech movements are impaired. Most children with idiopathic CAS have normal structural brain MRI. We hypothesize that children with CAS have altered structural connectivity in speech/language networks compared to controls and that these altered connections are related to functional speech/language measures. Whole brain probabilistic tractography, using constrained spherical deconvolution, was performed for connectome generation in 17 children with CAS and 10 age-matched controls. Fractional anisotropy (FA) was used as a measure of connectivity and the connections with altered FA between CAS and controls were identified. Further, the relationship between altered FA and speech/language scores was determined. Three intra-hemispheric/interhemispheric subnetworks showed reduction of FA in CAS compared to controls, including left inferior (opercular part) and superior (dorsolateral, medial and orbital part) frontal gyrus, left superior and middle temporal gyrus and left post-central gyrus (subnetwork 1); right supplementary motor area, left middle and inferior (orbital part) frontal gyrus, left precuneus and cuneus, right superior occipital gyrus and right cerebellum (subnetwork 2); right angular gyrus, right superior temporal gyrus and right inferior occipital gyrus (subnetwork 3). Reduced FA of some connections correlated with diadochokinesis, oromotor skills, expressive grammar and poor lexical production in CAS. These findings provide evidence of structural connectivity anomalies in children with CAS across specific brain regions involved in speech/language function. We propose altered connectivity as a possible epiphenomenon of complex pathogenic mechanisms in CAS which need further investigation.
| S-EPMC5114583 | biostudies-literature
Project description:Childhood apraxia of speech (CAS) is a rare, severe, persistent pediatric motor speech disorder with associated deficits in sensorimotor, cognitive, language, learning and affective processes. Among other neurogenetic origins, CAS is the disorder segregating with a mutation in FOXP2 in a widely studied, multigenerational London family. We report the first whole-exome sequencing (WES) findings from a cohort of 10 unrelated participants, ages 3 to 19 years, with well-characterized CAS.As part of a larger study of children and youth with motor speech sound disorders, 32 participants were classified as positive for CAS on the basis of a behavioral classification marker using auditory-perceptual and acoustic methods that quantify the competence, precision and stability of a speaker's speech, prosody and voice. WES of 10 randomly selected participants was completed using the Illumina Genome Analyzer IIx Sequencing System. Image analysis, base calling, demultiplexing, read mapping, and variant calling were performed using Illumina software. Software developed in-house was used for variant annotation, prioritization and interpretation to identify those variants likely to be deleterious to neurodevelopmental substrates of speech-language development.Among potentially deleterious variants, clinically reportable findings of interest occurred on a total of five chromosomes (Chr3, Chr6, Chr7, Chr9 and Chr17), which included six genes either strongly associated with CAS (FOXP1 and CNTNAP2) or associated with disorders with phenotypes overlapping CAS (ATP13A4, CNTNAP1, KIAA0319 and SETX). A total of 8 (80%) of the 10 participants had clinically reportable variants in one or two of the six genes, with variants in ATP13A4, KIAA0319 and CNTNAP2 being the most prevalent.Similar to the results reported in emerging WES studies of other complex neurodevelopmental disorders, our findings from this first WES study of CAS are interpreted as support for heterogeneous genetic origins of this pediatric motor speech disorder with multiple genes, pathways and complex interactions. We also submit that our findings illustrate the potential use of WES for both gene identification and case-by-case clinical diagnostics in pediatric motor speech disorders.
| S-EPMC3851280 | biostudies-literature
Project description:Purpose:Childhood apraxia of speech (CAS) is hypothesized to arise from deficits in speech motor planning and programming, but the influence of abnormal speech perception in CAS on these processes is debated. This study examined speech perception abilities among children with CAS with and without language impairment compared to those with language impairment, speech delay, and typically developing peers. Method:Speech perception was measured by discrimination of synthesized speech syllable continua that varied in frequency (/d?/-/??/). Groups were classified by performance on speech and language assessments and compared on syllable discrimination thresholds. Within-group variability was also evaluated. Results:Children with CAS without language impairment did not significantly differ in syllable discrimination compared to typically developing peers. In contrast, those with CAS and language impairment showed significantly poorer syllable discrimination abilities compared to children with CAS only and typically developing peers. Children with speech delay and language impairment also showed significantly poorer discrimination abilities, with appreciable within-group variability. Conclusions:These findings suggest that speech perception deficits are not a core feature of CAS but rather occur with co-occurring language impairment in a subset of children with CAS. This study establishes the significance of accounting for language ability in children with CAS. Supplemental Materials:https://doi.org/10.23641/asha.5848056.
| S-EPMC6195067 | biostudies-literature
Project description:Childhood apraxia of speech (CAS) is a severe and rare form of speech sound disorder (SSD). CAS is typically sporadic, but may segregate in families with broader speech and language deficits. We hypothesize that genetic changes may be involved in the etiology of CAS. We conduct whole-genome sequencing in 27 families with CAS, 101 individuals in all. We identify 17 genomic regions including 19 unique copy number variants (CNVs). Three variants are shared across families, but the rest are unique; three events are de novo. In four families, siblings with milder phenotypes co-inherited the same CNVs, demonstrating variable expressivity. We independently validate eight CNVs using microarray technology and find many of these CNVs are present in children with milder forms of SSD. Bioinformatic investigation reveal four CNVs with substantial functional consequences (cytobands 2q24.3, 6p12.3-6p12.2, 11q23.2-11q23.3, and 16p11.2). These discoveries show that CNVs are a heterogeneous, but prevalent, cause of CAS.
| S-EPMC11455877 | biostudies-literature
Project description:ObjectiveWhile it is known that connected speech has different features to single-word speech, there are currently few recommendations regarding connected speech transcription. This research therefore aimed to develop a clinically feasible protocol for connected speech transcription. The protocol was then used to assist with description of the connected speech of children with childhood apraxia of speech (CAS), as little is known about their connected speech characteristics.Participants and methodsFollowing a literature review, the Connected Speech Transcription Protocol (CoST-P) was iteratively developed and trialled. The CoST-P was then used to transcribe 50 connected utterances produced by 12 children (aged 6-13 years) with CAS. The characteristics of participants' connected speech were analysed to capture independent and relational analyses.ResultsThe CoST-P was developed, trialled, and determined to have adequate reliability and fidelity. The frequency of inter-word segregation (mean = 29) was higher than intra-word segregation (mean = 4). Juncture accuracy was correlated with intelligibility metrics such as percentage of consonants correct.ConclusionConnected speech transcription is challenging. The CoST-P may be a useful resource for speech-language pathologists and clinical researchers. Use of the CoST-P assisted in displaying CAS speech characteristics unique to connected speech (e.g., inter-word segregation and juncture).
| S-EPMC6940559 | biostudies-literature
Project description:We report a case series of children with childhood apraxia of speech, by describing behavioral and white matter microstructural changes following 2 different treatment approaches.Five children with childhood apraxia of speech were assigned to a motor speech treatment (PROMPT) and 5 to a language, nonspeech oral motor treatment. Speech assessment and brain MRI were performed pre- and post-treatment. The ventral (tongue/larynx) and dorsal (lips) corticobulbar tracts were reconstructed in each subject. Mean fractional anisotropy and mean diffusivity were extracted. The hand corticospinal tract was assessed as a control pathway. In both groups speech improvements paralleled changes in the left ventral corticobulbar tract fractional anisotropy. The PROMPT treated group also showed fractional anisotropy increase and mean diffusivity decrease in the left dorsal corticobulbar tract. No changes were detected in the hand tract. Our results may provide preliminary support to the possible neurobiologic effect of a multimodal speech motor treatment in childhood apraxia of speech.
| S-EPMC8461047 | biostudies-literature