Neural Changes Induced by a Speech Motor Treatment in Childhood Apraxia of Speech: A Case Series.
Ontology highlight
ABSTRACT:
SUBMITTER: Fiori S
PROVIDER: S-EPMC8461047 | biostudies-literature |
REPOSITORIES: biostudies-literature
Similar Datasets
Project description:ObjectiveThe cerebral substrates of apraxia of speech (AOS) recovery remain unclear. Resting state fMRI post stroke can inform on altered functional connectivity (FC) within cortical language networks. Some initial studies report reduced FC between bilateral premotor cortices in patients with AOS, with lowest FC in patients with the most severe AOS. However, longitudinal FC studies in stroke are lacking. The aims of the present longitudinal study in early post stroke patients with AOS were (i) to compare connectivity strength in AOS patients to that in left hemisphere (LH) lesioned stroke patients without a speech-language impairment, (ii) to investigate the relation between FC and severity of AOS, aphasia and non-verbal oral apraxia (NVOA) and (iii) to investigate longitudinal changes in FC, from the subacute phase to the chronic phase to identify predictors of AOS recovery.MethodsFunctional connectivity measures and comprehensive speech-language assessments were obtained at 4 weeks and 6 months after stroke in nine patients with AOS after a LH stroke and in six LH lesioned stroke patients without speech-language impairment. Functional connectivity was investigated in a network for speech production: inferior frontal gyrus (IFG), anterior insula (aINS), and ventral premotor cortex (vPMC), all bilaterally to investigate signs of adaptive or maladaptive changes in both hemispheres.ResultsInterhemispheric vPMC connectivity was significantly reduced in patients with AOS compared to LH lesioned patients without speech-language impairment. At 6 months, the AOS severity was associated with interhemispheric aINS and vPMC connectivity. Longitudinal changes in FC were found in individuals, whereas no significant longitudinal change in FC was found at the group level. Degree of longitudinal AOS recovery was strongly associated with interhemispheric IFG connectivity strength at 4 weeks.ConclusionEarly interhemispheric IFG connectivity may be a strong predictor of AOS recovery. The results support the importance of interhemispheric vPMC connection in speech motor planning and severity of AOS and suggest that also bilateral aINS connectivity may have an impact on AOS severity. These findings need to be validated in larger cohorts.
| S-EPMC9748434 | biostudies-literature
Project description:The purpose of this study was to evaluate the role of practice variability, through prosodic variation during speech sound training, in biofeedback treatment for children with childhood apraxia of speech. It was hypothesized that variable practice would facilitate speech sound learning.Six children ages 8-16 years with persisting speech sound errors due to childhood apraxia of speech participated in a single-subject experimental design. For each participant, 2 speech sound targets were treated with ultrasound visual feedback training: one with prosodic variation (i.e., practicing sound targets in words and phrases spoken fast, slow, loud, as a question, command, and declarative), and one without prosodic variation. Each target was treated for half of the 1-hr session for 14 treatment sessions.As measured by standardized effect sizes, all participants showed greater change on generalization probes for sound targets treated under the prosodic variation condition with mean effect sizes (d2) of 14.5 for targets treated with prosodic variation and 8.3 for targets treated without prosodic variation. The average increase in generalization scores was 38% in the prosodic variation condition compared to 31% without.Ultrasound visual feedback may facilitate speech sound learning and learning may be enhanced by treating speech sounds with explicit prosodic variation.https://doi.org/10.23641/asha.5150119.
| S-EPMC5829796 | 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:ObjectiveThis study describes the phenotype in a large family with a strong, multigenerational history of severe speech sound disorder (SSD) persisting into adolescence and adulthood in approximately half the cases. Aims were to determine whether a core phenotype, broader than speech, separated persistent from resolved SSD cases; and to ascertain the uniqueness of the phenotype relative to published cases.MethodEleven members of the PM family (9-55 years) were assessed across cognitive, language, literacy, speech, phonological processing, numeracy, and motor domains. Between group comparisons were made using the Mann-Whitney U-test (p < 0.01). Participant performances were compared to normative data using standardized tests and to the limited published data on persistent SSD phenotypes.ResultsSignificant group differences were evident on multiple speech, language, literacy, phonological processing, and verbal intellect measures without any overlapping scores. Persistent cases performed within the impaired range on multiple measures. Phonological memory impairment and subtle literacy weakness were present in resolved SSD cases.ConclusionA core phenotype distinguished persistent from resolved SSD cases that was characterized by a multiple verbal trait disorder, including Childhood Apraxia of Speech. Several phenotypic differences differentiated the persistent SSD phenotype in the PM family from the few previously reported studies of large families with SSD, including the absence of comorbid dysarthria and marked orofacial apraxia. This study highlights how comprehensive phenotyping can advance the behavioral study of disorders, in addition to forming a solid basis for future genetic and neural studies.
| S-EPMC7427608 | biostudies-literature
Project description:PurposeThis study sought to determine if children with childhood apraxia of speech (CAS) plus another major diagnosis (CAS+) are equivalent in communication and motor profiles to those with a primary diagnosis of CAS and no indication or report of any other diagnosis (CAS-Primary).MethodThis retrospective case-control study included a chart review of 143 children who were suspected of having CAS at Children's Hospital-Wisconsin between 1998 and 2004. Participants were between 30 and 127 months old and included 107 males. Participants were assigned to the suspected CAS-Primary group (n = 114) if they had characteristics of CAS but no other major diagnosis (e.g., galactosemia) and to the CAS+ group (n = 29) if a comorbid diagnosis was present. Groups were compared across demographic, communication, and motor characteristics.ResultsChildren with CAS+ evidenced more severe motor profiles than those with CAS-Primary, χ2 = (1, n = 122) = 4.952, p = .026, and a small-to-medium effect size (Φ = .201). On average, communication profiles also tended to be more severe among those with CAS+ wherein receptive language was poorer and phonemic inventories were smaller than those with CAS-Primary.ConclusionsThese retrospective data suggest that comorbid diagnosis may play an important role in communication and motor development in children with suspected CAS. These exploratory findings should motivate future prospective studies that consider the role of concomitant diagnoses in symptom profile and response to treatment in children with CAS.
| S-EPMC10019349 | 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: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: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