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Association between early and current gastro‐intestinal symptoms and co‐morbidities in children and adolescents with Angelman syndrome

BACKGROUND: Angelman syndrome (AS) is a neurogenetic disorder that causes severe intellectual disability, expressive language deficits, motor impairment, ataxia, sleep problems, epileptic seizures and a happy disposition. People with AS frequently experience gastrointestinal (GI) symptoms. METHOD: T...

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Autores principales: Leader, G., Whelan, S., Chonaill, N. N., Coyne, R., Tones, M., Heussler, H., Bellgard, M., Mannion, A.
Formato: Online Artículo Texto
Lenguaje:English
Publicado: John Wiley and Sons Inc. 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9826167/
https://www.ncbi.nlm.nih.gov/pubmed/36052644
http://dx.doi.org/10.1111/jir.12975
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author Leader, G.
Whelan, S.
Chonaill, N. N.
Coyne, R.
Tones, M.
Heussler, H.
Bellgard, M.
Mannion, A.
author_facet Leader, G.
Whelan, S.
Chonaill, N. N.
Coyne, R.
Tones, M.
Heussler, H.
Bellgard, M.
Mannion, A.
author_sort Leader, G.
collection PubMed
description BACKGROUND: Angelman syndrome (AS) is a neurogenetic disorder that causes severe intellectual disability, expressive language deficits, motor impairment, ataxia, sleep problems, epileptic seizures and a happy disposition. People with AS frequently experience gastrointestinal (GI) symptoms. METHOD: This study used data from the Global Angelman Syndrome Registry to explore the relationship between early and current GI symptoms and co‐morbidity in children and adolescents with AS (n = 173). Two groups that experienced a high (n = 91) and a low (n = 82) frequency of GI symptoms were examined in relation to feeding and GI history in infancy, sleep and toileting problems, levels of language and communication and challenging behaviours. Predictors of GI symptoms were then investigated using a series of logistic regressions. RESULTS: This analysis found that constipation and gastroesophageal reflux affected 84% and 64%, of the sample, respectively. The high frequency of GI symptoms were significantly associated with: ‘refusal to nurse’, ‘vomiting’, ‘arching’, ‘difficulty gaining weight’, gastroesophageal reflux, ‘solid food transition’, frequency of night‐time urinary continence and sleep hyperhidrosis during infancy. GI symptoms were not significantly associated with sleep, toileting, language or challenging behaviours. Significant predictors of high frequency GI symptoms were gastroesophageal reflux and sleep hyperhidrosis. CONCLUSIONS: Future research needs to investigate the association between AS and GI co‐morbidity in adults with AS.
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spelling pubmed-98261672023-01-09 Association between early and current gastro‐intestinal symptoms and co‐morbidities in children and adolescents with Angelman syndrome Leader, G. Whelan, S. Chonaill, N. N. Coyne, R. Tones, M. Heussler, H. Bellgard, M. Mannion, A. J Intellect Disabil Res Original Articles BACKGROUND: Angelman syndrome (AS) is a neurogenetic disorder that causes severe intellectual disability, expressive language deficits, motor impairment, ataxia, sleep problems, epileptic seizures and a happy disposition. People with AS frequently experience gastrointestinal (GI) symptoms. METHOD: This study used data from the Global Angelman Syndrome Registry to explore the relationship between early and current GI symptoms and co‐morbidity in children and adolescents with AS (n = 173). Two groups that experienced a high (n = 91) and a low (n = 82) frequency of GI symptoms were examined in relation to feeding and GI history in infancy, sleep and toileting problems, levels of language and communication and challenging behaviours. Predictors of GI symptoms were then investigated using a series of logistic regressions. RESULTS: This analysis found that constipation and gastroesophageal reflux affected 84% and 64%, of the sample, respectively. The high frequency of GI symptoms were significantly associated with: ‘refusal to nurse’, ‘vomiting’, ‘arching’, ‘difficulty gaining weight’, gastroesophageal reflux, ‘solid food transition’, frequency of night‐time urinary continence and sleep hyperhidrosis during infancy. GI symptoms were not significantly associated with sleep, toileting, language or challenging behaviours. Significant predictors of high frequency GI symptoms were gastroesophageal reflux and sleep hyperhidrosis. CONCLUSIONS: Future research needs to investigate the association between AS and GI co‐morbidity in adults with AS. John Wiley and Sons Inc. 2022-09-02 2022-11 /pmc/articles/PMC9826167/ /pubmed/36052644 http://dx.doi.org/10.1111/jir.12975 Text en © 2022 The Authors. Journal of Intellectual Disability Research published by MENCAP and International Association of the Scientific Study of Intellectual and Developmental Disabilities and John Wiley & Sons Ltd. https://creativecommons.org/licenses/by-nc/4.0/This is an open access article under the terms of the http://creativecommons.org/licenses/by-nc/4.0/ (https://creativecommons.org/licenses/by-nc/4.0/) License, which permits use, distribution and reproduction in any medium, provided the original work is properly cited and is not used for commercial purposes.
spellingShingle Original Articles
Leader, G.
Whelan, S.
Chonaill, N. N.
Coyne, R.
Tones, M.
Heussler, H.
Bellgard, M.
Mannion, A.
Association between early and current gastro‐intestinal symptoms and co‐morbidities in children and adolescents with Angelman syndrome
title Association between early and current gastro‐intestinal symptoms and co‐morbidities in children and adolescents with Angelman syndrome
title_full Association between early and current gastro‐intestinal symptoms and co‐morbidities in children and adolescents with Angelman syndrome
title_fullStr Association between early and current gastro‐intestinal symptoms and co‐morbidities in children and adolescents with Angelman syndrome
title_full_unstemmed Association between early and current gastro‐intestinal symptoms and co‐morbidities in children and adolescents with Angelman syndrome
title_short Association between early and current gastro‐intestinal symptoms and co‐morbidities in children and adolescents with Angelman syndrome
title_sort association between early and current gastro‐intestinal symptoms and co‐morbidities in children and adolescents with angelman syndrome
topic Original Articles
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9826167/
https://www.ncbi.nlm.nih.gov/pubmed/36052644
http://dx.doi.org/10.1111/jir.12975
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