Dataset Information

Assessment of Dysphonia in Children with Pompe Disease Using Auditory-Perceptual and Acoustic/Physiologic Methods.

ABSTRACT: Bulbar and respiratory weakness occur commonly in children with Pompe disease and frequently lead to dysarthria. However, changes in vocal quality associated with this motor speech disorder are poorly described. The goal of this study was to characterize the vocal function of children with Pompe disease using auditory-perceptual and physiologic/acoustic methods. High-quality voice recordings were collected from 21 children with Pompe disease. The Grade, Roughness, Breathiness, Asthenia, and Strain (GRBAS) scale was used to assess voice quality and ratings were compared to physiologic/acoustic measurements collected during sustained phonation tasks, reading of a standard passage, and repetition of a short phrase at maximal volume. Based on ratings of grade, dysphonia was present in 90% of participants and was most commonly rated as mild or moderate in severity. Duration of sustained phonation tasks was reduced and shimmer was increased in comparison to published reference values for children without dysphonia. Specific measures of loudness were found to have statistically significant relationships with perceptual ratings of grade, breathiness, asthenia, and strain. Our data suggest that dysphonia is common in children with Pompe disease and primarily reflects impairments in respiratory and laryngeal function; however, the primary cause of dysphonia remains unclear. Future studies should seek to quantify the relative contribution of deficits in individual speech subsystems on voice quality and motor speech performance more broadly.

SUBMITTER: Crisp KD

PROVIDER: S-EPMC8396833 | biostudies-literature | 2021 Aug

REPOSITORIES: biostudies-literature

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Assessment of Dysphonia in Children with Pompe Disease Using Auditory-Perceptual and Acoustic/Physiologic Methods.

Crisp Kelly D KD Neel Amy T AT Amarasekara Sathya S Marcus Jill J Nichting Gretchen G Korlimarla Aditi A Kishnani Priya S PS Jones Harrison N HN

Journal of clinical medicine 20210816 16

Bulbar and respiratory weakness occur commonly in children with Pompe disease and frequently lead to dysarthria. However, changes in vocal quality associated with this motor speech disorder are poorly described. The goal of this study was to characterize the vocal function of children with Pompe disease using auditory-perceptual and physiologic/acoustic methods. High-quality voice recordings were collected from 21 children with Pompe disease. The Grade, Roughness, Breathiness, Asthenia, and Stra ...[more]

PMID: 34441913

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Project description:Objectives/hypothesisThe objectives of this study were to (1) identify optimal clusters of 15 standard acoustic and aerodynamic voice metrics recommended by the American Speech-Language-Hearing Association (ASHA) to improve characterization of patients with primary muscle tension dysphonia (pMTD) and (2) identify combinations of these 15 metrics that could differentiate pMTD from other types of voice disorders.Study designRetrospective multiparametric METHODS: Random forest modeling, independent t-tests, logistic regression, and affinity propagation clustering were implemented on a retrospective dataset of 15 acoustic and aerodynamic metrics.ResultsTen percent of patients seen at the New York University (NYU) Voice Center over two years met the study criteria for pMTD (92 out of 983 patients), with 65 patients with pMTD and 701 of non-pMTD patients with complete data across all 15 acoustic and aerodynamic voice metrics. PCA plots and affinity propagation clustering demonstrated substantial overlap between the two groups on these parameters. The highest ranked parameters by level of importance with random forest models-(1) mean airflow during voicing (L/sec), (2) mean SPL during voicing (dB), (3) mean peak air pressure (cmH2O), (4) highest F0 (Hz), and (5) CPP mean vowel (dB)-accounted for only 65% of variance. T-tests showed three of these parameters-(1) CPP mean vowel (dB), (2) highest F0 (Hz), and (3) mean peak air pressure (cmH2O)-were statistically significant; however, the log2-fold change for each parameter was minimal.ConclusionComputational models and multivariate statistical testing on 15 acoustic and aerodynamic voice metrics were unable to adequately characterize pMTD and determine differences between the two groups (pMTD and non-pMTD). Further validation of these metrics is needed with voice elicitation tasks that target physiological challenges to the vocal system from baseline vocal acoustic and aerodynamic ouput. Future work should also place greater focus on validating metrics of physiological correlates (eg, neuromuscular processes, laryngeal-respiratory kinematics) across the vocal subsystems over traditional vocal output measures (eg, acoustics, aerodynamics) for patients with pMTD.Level of evidenceII.

Project description:Learning is considered to consist of two distinct phases-acquisition and consolidation. Acquisition can be disrupted when short periods of training on more than one task are interleaved, whereas consolidation can be disrupted when a second task is trained after the first has been initiated. Here we investigated the conditions governing the disruption to acquisition and consolidation during mixed-training regimens in which primary and secondary amplitude modulation tasks were either interleaved or presented consecutively. The secondary task differed from the primary task in either task-irrelevant (carrier frequency) or task-relevant (modulation rate) stimulus features while requiring the same perceptual judgment (amplitude modulation depth discrimination), or shared both irrelevant and relevant features but required a different judgment (amplitude modulation rate discrimination). Based on previous literature we predicted that acquisition would be disrupted by varying the task-relevant stimulus feature during training (stimulus interference), and that consolidation would be disrupted by varying the perceptual judgment required (task interference). We found that varying the task-relevant or -irrelevant stimulus features failed to disrupt acquisition but did disrupt consolidation, whereas mixing two tasks requiring a different perceptual judgment but sharing the same stimulus features disrupted both acquisition and consolidation. Thus, a distinction between acquisition and consolidation phases of perceptual learning cannot simply be attributed to (task-relevant) stimulus versus task interference. We propose instead that disruption occurs during acquisition when mixing two tasks requiring a perceptual judgment based on different cues, whereas consolidation is always disrupted regardless of whether different stimulus features or tasks are mixed. The current study not only provides a novel insight into the underlying mechanisms of perceptual learning, but also has practical implications for the optimal design and delivery of training programs that aim to remediate perceptual difficulties.

Project description:ObjectiveThe primary objective of the present systematic review is to: (1) identify the current vocal tasks being used for acoustic and/or auditory perceptual analysis to differentiate between individuals with and without voice disorders. The secondary objectives are to: (2) evaluate the evidence of the sensitivity, specificity and accuracy of those vocal tasks for acoustic and/or auditory perceptual analysis in discriminating the individuals with voice disorders from those without; and (3) compare the values between the vocal tasks in discriminating individuals with voice disorders from those without.Method and analysisWe search the following electronic databases: MEDLINE, EMBASE, CINAHL, Scopus, Web of Science Core Collection, PubMed Central and Google Scholar. Grey literature searches will include ProQuest Dissertations and Theses, ClinicalTrials.gov and the Cochrane Register of Controlled Trials. Websites of professional organisations and textbooks will be hand searched for relevant information related to the research question. Study screening, selection and data extraction will be conducted independently by two reviewers. Any disagreements will be resolved by discussion or by involving a third reviewer.The methodological quality of the included studies will be appraised using the relevant Critical Appraisal Tools by JBI. The clinical guidelines and recommendations for voice assessment by professional bodies will be appraised using the Reporting Items for practice Guidelines in HealThcare (RIGHT) checklist. The findings will be presented in the form of an information matrix with the tasks identified tabulated against the nature of the task, dimensions being tested, and their accuracy, sensitivity, and specificity in identifying individuals with voice problems.Ethics and disseminationFormal ethics approval is not required. The findings will be presented at national and international conferences and published in a peer-reviewed journal.Prospero registration numberCRD42023431634.

Dataset Information

Assessment of Dysphonia in Children with Pompe Disease Using Auditory-Perceptual and Acoustic/Physiologic Methods.

Publications

Assessment of Dysphonia in Children with Pompe Disease Using Auditory-Perceptual and Acoustic/Physiologic Methods.

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