A pilot study shows the positive effects of continuous airway pressure for treating hypernasal speech in children with infantile-onset Pompe disease

Abstract Children with infantile-onset Pompe disease (IOPD) demonstrate hypernasality. This study aimed to evaluate whether continuous positive airway pressure (CPAP) training may reduce hypernasality in children with IOPD. Five children with IOPD were enrolled in a single-subject experimental desig...

Descripción completa

Guardado en:

Detalles Bibliográficos
Autores principales:	Yin-Ting Zeng, Wen-Yu Liu, Pao-Chuan Torng, Wuh-Liang Hwu, Ni-Chung Lee, Chun-Yi Lin, Yin-Hsiu Chien
Formato:	article
Lenguaje:	EN
Publicado:	Nature Portfolio 2021
Materias:	Medicine R Science Q
Acceso en línea:	https://doaj.org/article/01d6fa4a01ae4e3ea1ad5cb577d18c41
Etiquetas:	Agregar Etiqueta Sin Etiquetas, Sea el primero en etiquetar este registro!

id	oai:doaj.org-article:01d6fa4a01ae4e3ea1ad5cb577d18c41
record_format	dspace
spelling	oai:doaj.org-article:01d6fa4a01ae4e3ea1ad5cb577d18c412021-12-02T18:13:53ZA pilot study shows the positive effects of continuous airway pressure for treating hypernasal speech in children with infantile-onset Pompe disease10.1038/s41598-021-97877-12045-2322https://doaj.org/article/01d6fa4a01ae4e3ea1ad5cb577d18c412021-09-01T00:00:00Zhttps://doi.org/10.1038/s41598-021-97877-1https://doaj.org/toc/2045-2322Abstract Children with infantile-onset Pompe disease (IOPD) demonstrate hypernasality. This study aimed to evaluate whether continuous positive airway pressure (CPAP) training may reduce hypernasality in children with IOPD. Five children with IOPD were enrolled in a single-subject experimental design of type A-B-A′. The intervention comprised an 8-week, 6-day-per-week regimen of CPAP training at home. Participants continued traditional speech therapy once per week throughout the 24-week study duration. The outcome measurements included the degree of hypernasality (DH), the percentage of consonants correct (PCC), and the speech intelligibility score (SIS). C-statistic analysis with an α of 0.05 was used along with visual analysis to assess speech changes. Three patients completed the study. During the CPAP training phase, the DH, PCC, and SIS were significantly improved compared with the baseline (p < 0.05). At the follow-up phase, both DH and SIS were improved compared with the baseline (p < 0.05), but the PCC had returned to the baseline level. CPAP training demonstrated effectiveness in reducing nasal sounds in IOPD patients. Further studies training younger children with normal hearing may help elucidate the persistence of the effects in children with IOPD.Yin-Ting ZengWen-Yu LiuPao-Chuan TorngWuh-Liang HwuNi-Chung LeeChun-Yi LinYin-Hsiu ChienNature PortfolioarticleMedicineRScienceQENScientific Reports, Vol 11, Iss 1, Pp 1-8 (2021)
institution	DOAJ
collection	DOAJ
language	EN
topic	Medicine R Science Q
spellingShingle	Medicine R Science Q Yin-Ting Zeng Wen-Yu Liu Pao-Chuan Torng Wuh-Liang Hwu Ni-Chung Lee Chun-Yi Lin Yin-Hsiu Chien A pilot study shows the positive effects of continuous airway pressure for treating hypernasal speech in children with infantile-onset Pompe disease
description	Abstract Children with infantile-onset Pompe disease (IOPD) demonstrate hypernasality. This study aimed to evaluate whether continuous positive airway pressure (CPAP) training may reduce hypernasality in children with IOPD. Five children with IOPD were enrolled in a single-subject experimental design of type A-B-A′. The intervention comprised an 8-week, 6-day-per-week regimen of CPAP training at home. Participants continued traditional speech therapy once per week throughout the 24-week study duration. The outcome measurements included the degree of hypernasality (DH), the percentage of consonants correct (PCC), and the speech intelligibility score (SIS). C-statistic analysis with an α of 0.05 was used along with visual analysis to assess speech changes. Three patients completed the study. During the CPAP training phase, the DH, PCC, and SIS were significantly improved compared with the baseline (p < 0.05). At the follow-up phase, both DH and SIS were improved compared with the baseline (p < 0.05), but the PCC had returned to the baseline level. CPAP training demonstrated effectiveness in reducing nasal sounds in IOPD patients. Further studies training younger children with normal hearing may help elucidate the persistence of the effects in children with IOPD.
format	article
author	Yin-Ting Zeng Wen-Yu Liu Pao-Chuan Torng Wuh-Liang Hwu Ni-Chung Lee Chun-Yi Lin Yin-Hsiu Chien
author_facet	Yin-Ting Zeng Wen-Yu Liu Pao-Chuan Torng Wuh-Liang Hwu Ni-Chung Lee Chun-Yi Lin Yin-Hsiu Chien
author_sort	Yin-Ting Zeng
title	A pilot study shows the positive effects of continuous airway pressure for treating hypernasal speech in children with infantile-onset Pompe disease
title_short	A pilot study shows the positive effects of continuous airway pressure for treating hypernasal speech in children with infantile-onset Pompe disease
title_full	A pilot study shows the positive effects of continuous airway pressure for treating hypernasal speech in children with infantile-onset Pompe disease
title_fullStr	A pilot study shows the positive effects of continuous airway pressure for treating hypernasal speech in children with infantile-onset Pompe disease
title_full_unstemmed	A pilot study shows the positive effects of continuous airway pressure for treating hypernasal speech in children with infantile-onset Pompe disease
title_sort	pilot study shows the positive effects of continuous airway pressure for treating hypernasal speech in children with infantile-onset pompe disease
publisher	Nature Portfolio
publishDate	2021
url	https://doaj.org/article/01d6fa4a01ae4e3ea1ad5cb577d18c41
work_keys_str_mv	AT yintingzeng apilotstudyshowsthepositiveeffectsofcontinuousairwaypressurefortreatinghypernasalspeechinchildrenwithinfantileonsetpompedisease AT wenyuliu apilotstudyshowsthepositiveeffectsofcontinuousairwaypressurefortreatinghypernasalspeechinchildrenwithinfantileonsetpompedisease AT paochuantorng apilotstudyshowsthepositiveeffectsofcontinuousairwaypressurefortreatinghypernasalspeechinchildrenwithinfantileonsetpompedisease AT wuhlianghwu apilotstudyshowsthepositiveeffectsofcontinuousairwaypressurefortreatinghypernasalspeechinchildrenwithinfantileonsetpompedisease AT nichunglee apilotstudyshowsthepositiveeffectsofcontinuousairwaypressurefortreatinghypernasalspeechinchildrenwithinfantileonsetpompedisease AT chunyilin apilotstudyshowsthepositiveeffectsofcontinuousairwaypressurefortreatinghypernasalspeechinchildrenwithinfantileonsetpompedisease AT yinhsiuchien apilotstudyshowsthepositiveeffectsofcontinuousairwaypressurefortreatinghypernasalspeechinchildrenwithinfantileonsetpompedisease AT yintingzeng pilotstudyshowsthepositiveeffectsofcontinuousairwaypressurefortreatinghypernasalspeechinchildrenwithinfantileonsetpompedisease AT wenyuliu pilotstudyshowsthepositiveeffectsofcontinuousairwaypressurefortreatinghypernasalspeechinchildrenwithinfantileonsetpompedisease AT paochuantorng pilotstudyshowsthepositiveeffectsofcontinuousairwaypressurefortreatinghypernasalspeechinchildrenwithinfantileonsetpompedisease AT wuhlianghwu pilotstudyshowsthepositiveeffectsofcontinuousairwaypressurefortreatinghypernasalspeechinchildrenwithinfantileonsetpompedisease AT nichunglee pilotstudyshowsthepositiveeffectsofcontinuousairwaypressurefortreatinghypernasalspeechinchildrenwithinfantileonsetpompedisease AT chunyilin pilotstudyshowsthepositiveeffectsofcontinuousairwaypressurefortreatinghypernasalspeechinchildrenwithinfantileonsetpompedisease AT yinhsiuchien pilotstudyshowsthepositiveeffectsofcontinuousairwaypressurefortreatinghypernasalspeechinchildrenwithinfantileonsetpompedisease
_version_	1718378432489848832

A pilot study shows the positive effects of continuous airway pressure for treating hypernasal speech in children with infantile-onset Pompe disease

Ejemplares similares