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Speech Disorders in Children With Pompe Disease: Articulation, Resonance, and Voice Measures.

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Title: Speech Disorders in Children With Pompe Disease: Articulation, Resonance, and Voice Measures.
Authors: Neel, Amy T.1 atneel@unm.edu, Crisp, Kelly D.2, Kishnani, Priya S.3, Jones, Harrison N.4
Source: American Journal of Speech-Language Pathology. Jul2023, Vol. 32 Issue 4, p1501-1516. 16p.
Subject Terms: *Speech evaluation, *Verbal behavior testing, *Data analysis, *Articulation (Speech), Statistics, Physiological aspects of speech, Multiple regression analysis, Speech disorders in children, Visual analog scale, Mann Whitney U Test, Pearson correlation (Statistics), Inborn errors of carbohydrate metabolism, Research funding, Data analysis software
Abstract: Purpose: Children with Pompe disease, a rare genetic metabolic myopathy, often have speech impairments. In this study, we provide a comprehensive description of articulation, resonance, and voice in children with Pompe disease. Method: Fifteen children with Pompe disease (11 with infantile-onset Pompe disease [IOPD], four with late-onset Pompe disease [LOPD]) ranging from 6 to 18 years of age participated in standard speech assessments. Measures included maximum tongue pressure; nasalance; cepstral peak prominence (CPP); low/high ratio (L/H ratio); diadochokinetic (DDK) rates; percent consonants correct (PCC); and visual analog scale (VAS) ratings of articulation, resonance, voice quality, and overall speech severity. Maximum tongue pressures, nasalance, CPP, L/H ratio, DDK rates, and PCC were compared to normative data from typically developing (TD) children. Correlation analyses and multiple regression models of speech measure predictors were conducted. Results: Children with IOPD had greater speech impairment than those with LOPD. The IOPD group had lower maximum tongue pressures, slower articulation rates, lower PCC scores, higher nasalance, and higher L/H voice ratios than TD children. VAS ratings confirmed the presence of impaired articulatory precision, hypernasality, and dysphonia for most of the children with IOPD, with severity of impairment ratings ranging from mild to severe. The LOPD group had mildly elevated nasalance and L/H ratio values relative to TD children, and auditory-perceptual ratings suggested mild to no speech impairment. Conclusions: Speech disorders involving articulatory precision, resonance balance, and voice quality are common in children with Pompe disease, especially in those with IOPD. With improvements in the detection and treatment of Pompe disease, clinicians should be aware of the associated speech deficits. [ABSTRACT FROM AUTHOR]
Copyright of American Journal of Speech-Language Pathology is the property of American Speech-Language-Hearing Association and its content may not be copied or emailed to multiple sites without the copyright holder's express written permission. Additionally, content may not be used with any artificial intelligence tools or machine learning technologies. However, users may print, download, or email articles for individual use. This abstract may be abridged. No warranty is given about the accuracy of the copy. Users should refer to the original published version of the material for the full abstract. (Copyright applies to all Abstracts.)
Database: Education Research Complete
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  Data: Speech Disorders in Children With Pompe Disease: Articulation, Resonance, and Voice Measures.
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  Data: <searchLink fieldCode="AR" term="%22Neel%2C+Amy+T%2E%22">Neel, Amy T.</searchLink><relatesTo>1</relatesTo><i> atneel@unm.edu</i><br /><searchLink fieldCode="AR" term="%22Crisp%2C+Kelly+D%2E%22">Crisp, Kelly D.</searchLink><relatesTo>2</relatesTo><br /><searchLink fieldCode="AR" term="%22Kishnani%2C+Priya+S%2E%22">Kishnani, Priya S.</searchLink><relatesTo>3</relatesTo><br /><searchLink fieldCode="AR" term="%22Jones%2C+Harrison+N%2E%22">Jones, Harrison N.</searchLink><relatesTo>4</relatesTo>
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  Data: <searchLink fieldCode="JN" term="%22American+Journal+of+Speech-Language+Pathology%22">American Journal of Speech-Language Pathology</searchLink>. Jul2023, Vol. 32 Issue 4, p1501-1516. 16p.
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  Data: *<searchLink fieldCode="DE" term="%22Speech+evaluation%22">Speech evaluation</searchLink><br />*<searchLink fieldCode="DE" term="%22Verbal+behavior+testing%22">Verbal behavior testing</searchLink><br />*<searchLink fieldCode="DE" term="%22Data+analysis%22">Data analysis</searchLink><br />*<searchLink fieldCode="DE" term="%22Articulation+%28Speech%29%22">Articulation (Speech)</searchLink><br /><searchLink fieldCode="DE" term="%22Statistics%22">Statistics</searchLink><br /><searchLink fieldCode="DE" term="%22Physiological+aspects+of+speech%22">Physiological aspects of speech</searchLink><br /><searchLink fieldCode="DE" term="%22Multiple+regression+analysis%22">Multiple regression analysis</searchLink><br /><searchLink fieldCode="DE" term="%22Speech+disorders+in+children%22">Speech disorders in children</searchLink><br /><searchLink fieldCode="DE" term="%22Visual+analog+scale%22">Visual analog scale</searchLink><br /><searchLink fieldCode="DE" term="%22Mann+Whitney+U+Test%22">Mann Whitney U Test</searchLink><br /><searchLink fieldCode="DE" term="%22Pearson+correlation+%28Statistics%29%22">Pearson correlation (Statistics)</searchLink><br /><searchLink fieldCode="DE" term="%22Inborn+errors+of+carbohydrate+metabolism%22">Inborn errors of carbohydrate metabolism</searchLink><br /><searchLink fieldCode="DE" term="%22Research+funding%22">Research funding</searchLink><br /><searchLink fieldCode="DE" term="%22Data+analysis+software%22">Data analysis software</searchLink>
– Name: Abstract
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  Data: Purpose: Children with Pompe disease, a rare genetic metabolic myopathy, often have speech impairments. In this study, we provide a comprehensive description of articulation, resonance, and voice in children with Pompe disease. Method: Fifteen children with Pompe disease (11 with infantile-onset Pompe disease [IOPD], four with late-onset Pompe disease [LOPD]) ranging from 6 to 18 years of age participated in standard speech assessments. Measures included maximum tongue pressure; nasalance; cepstral peak prominence (CPP); low/high ratio (L/H ratio); diadochokinetic (DDK) rates; percent consonants correct (PCC); and visual analog scale (VAS) ratings of articulation, resonance, voice quality, and overall speech severity. Maximum tongue pressures, nasalance, CPP, L/H ratio, DDK rates, and PCC were compared to normative data from typically developing (TD) children. Correlation analyses and multiple regression models of speech measure predictors were conducted. Results: Children with IOPD had greater speech impairment than those with LOPD. The IOPD group had lower maximum tongue pressures, slower articulation rates, lower PCC scores, higher nasalance, and higher L/H voice ratios than TD children. VAS ratings confirmed the presence of impaired articulatory precision, hypernasality, and dysphonia for most of the children with IOPD, with severity of impairment ratings ranging from mild to severe. The LOPD group had mildly elevated nasalance and L/H ratio values relative to TD children, and auditory-perceptual ratings suggested mild to no speech impairment. Conclusions: Speech disorders involving articulatory precision, resonance balance, and voice quality are common in children with Pompe disease, especially in those with IOPD. With improvements in the detection and treatment of Pompe disease, clinicians should be aware of the associated speech deficits. [ABSTRACT FROM AUTHOR]
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  Data: <i>Copyright of American Journal of Speech-Language Pathology is the property of American Speech-Language-Hearing Association and its content may not be copied or emailed to multiple sites without the copyright holder's express written permission. Additionally, content may not be used with any artificial intelligence tools or machine learning technologies. However, users may print, download, or email articles for individual use. This abstract may be abridged. No warranty is given about the accuracy of the copy. Users should refer to the original published version of the material for the full abstract.</i> (Copyright applies to all Abstracts.)
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        Value: 10.1044/2023_AJSLP-22-00382
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      – SubjectFull: Speech evaluation
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      – TitleFull: Speech Disorders in Children With Pompe Disease: Articulation, Resonance, and Voice Measures.
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              Text: Jul2023
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