Association between early and current gastro-intestinal symptoms and co-morbidities in children and adolescents with Angelman syndrome.
Angelman syndrome
Global Angelman Syndrome Registry
co-morbidity
gastrointestinal symptoms
Journal
Journal of intellectual disability research : JIDR
ISSN: 1365-2788
Titre abrégé: J Intellect Disabil Res
Pays: England
ID NLM: 9206090
Informations de publication
Date de publication:
11 2022
11 2022
Historique:
revised:
22
07
2022
received:
02
04
2022
accepted:
09
08
2022
pubmed:
3
9
2022
medline:
14
10
2022
entrez:
2
9
2022
Statut:
ppublish
Résumé
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. 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. 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. Future research needs to investigate the association between AS and GI co-morbidity in adults with AS.
Sections du résumé
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.
Identifiants
pubmed: 36052644
doi: 10.1111/jir.12975
pmc: PMC9826167
doi:
Types de publication
Journal Article
Research Support, Non-U.S. Gov't
Langues
eng
Sous-ensembles de citation
IM
Pagination
865-879Informations de copyright
© 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.
Références
Alpha Psychiatry. 2021 Jan 19;22(1):43-48
pubmed: 36426208
Clin Genet. 2004 Dec;66(6):530-6
pubmed: 15521981
Orphanet J Rare Dis. 2017 Aug 1;12(1):134
pubmed: 28764722
Contemp Clin Trials. 2015 Jan;40:212-7
pubmed: 25533730
Autism Res. 2012 Apr;5(2):101-8
pubmed: 22511450
J Intellect Disabil Res. 2011 Feb;55(2):109-20
pubmed: 20977515
Am J Med Genet A. 2015 Feb;167A(2):331-44
pubmed: 25428759
Am J Med Genet A. 2018 Jun;176(6):1327-1334
pubmed: 29696750
Am J Speech Lang Pathol. 2017 May 17;26(2):369-382
pubmed: 28384804
Am J Med Genet A. 2020 Jan;182(1):53-63
pubmed: 31729827
Am J Med Genet A. 2006 Mar 1;140(5):413-8
pubmed: 16470747
Sleep Med. 2008 May;9(4):434-41
pubmed: 17765640
Res Dev Disabil. 2014 Nov;35(11):2681-90
pubmed: 25064682
Am J Med Genet A. 2019 Jun;179(6):983-992
pubmed: 30942555
Neurotherapeutics. 2015 Jul;12(3):641-50
pubmed: 26040994
Res Dev Disabil. 2009 Sep-Oct;30(5):1095-106
pubmed: 19361955
Lancet Psychiatry. 2015 Oct;2(10):909-16
pubmed: 26341300
J Appl Res Intellect Disabil. 2018 Jan;31(1):e49-e58
pubmed: 27990716
Am J Ment Retard. 2000 Jan;105(1):25-31
pubmed: 10683706
PLoS One. 2015 May 20;10(5):e0126982
pubmed: 25992621
Pediatr Neurol. 2013 Apr;48(4):271-9
pubmed: 23498559
Res Dev Disabil. 2007 Jul-Sep;28(4):341-52
pubmed: 16765022
Eur J Paediatr Neurol. 2013 Jul;17(4):374-82
pubmed: 23352289
Am J Med Genet. 1995 Mar 27;56(2):176-83
pubmed: 7625442
Orphanet J Rare Dis. 2017 Oct 16;12(1):164
pubmed: 29037196
Neurogenetics. 2007 Aug;8(3):169-78
pubmed: 17415598
Wien Med Wochenschr. 2017 Jun;167(9-10):205-218
pubmed: 26758979
J Intellect Disabil Res. 2018 May;62(5):431-443
pubmed: 29633452
Res Dev Disabil. 2009 Jul-Aug;30(4):799-807
pubmed: 19162439
Appl Clin Genet. 2014 May 16;7:93-104
pubmed: 24876791
J Sleep Res. 1996 Dec;5(4):251-61
pubmed: 9065877
J Intellect Disabil Res. 2006 Jan;50(Pt 1):33-53
pubmed: 16316429
J Dev Behav Pediatr. 2010 Sep;31(7):592-601
pubmed: 20729760
Am J Med Genet A. 2017 Oct;173(10):2703-2709
pubmed: 28816003
Ci Ji Yi Xue Za Zhi. 2019 Oct 31;32(2):137-144
pubmed: 32269945
J Intellect Disabil Res. 2022 Nov;66(11):865-879
pubmed: 36052644
Sleep Med. 2009 Apr;10(4):457-63
pubmed: 18706856
Augment Altern Commun. 2013 Jun;29(2):146-58
pubmed: 23621361
Dev Neurorehabil. 2014 Aug;17(4):243-50
pubmed: 23957894
J Autism Dev Disord. 2007 May;37(5):940-7
pubmed: 17019625
Nat Rev Neurol. 2016 Oct;12(10):584-93
pubmed: 27615419
J Neurodev Disord. 2018 Mar 1;10(1):9
pubmed: 29490614
J Clin Anesth. 2014 Feb;26(1):75-9
pubmed: 24440034
Sleep Med Rev. 2018 Feb;37:69-84
pubmed: 28784434
Eur J Pediatr. 2017 Feb;176(2):225-232
pubmed: 28000035
Am J Med Genet A. 2011 Jan;155A(1):81-90
pubmed: 21204213
Can Fam Physician. 2020 Dec;66(12):901-903
pubmed: 33334958
Mol Autism. 2021 Feb 25;12(1):18
pubmed: 33632309