Autism spectrum disorders in boys at a major UK hemophilia center: prevalence and risk factors.

autism spectrum disorder child communication executive function hemophilia A

Journal

Research and practice in thrombosis and haemostasis
ISSN: 2475-0379
Titre abrégé: Res Pract Thromb Haemost
Pays: United States
ID NLM: 101703775

Informations de publication

Date de publication:
Jan 2023
Historique:
received: 02 12 2021
revised: 07 10 2022
accepted: 22 10 2022
entrez: 9 3 2023
pubmed: 10 3 2023
medline: 10 3 2023
Statut: epublish

Résumé

Autism spectrum disorders (ASDs) are diagnosed by social communication difficulties strong, narrow interests, and repetitive stereotyped behavior. An apparently-elevated prevalence of ASD at a major UK hemophilia center warranted investigation. To screen boys with hemophilia for difficulties in social communication and executive function and identify the prevalence and risk factors for ASD. Parents of boys with hemophilia aged 5 to 16 years completed the Social Communication Questionnaire, Children's Communication Checklist, and the Behavior Rating Inventory of executive function. Prevalence and potential risk factors for ASD were evaluated. Boys with an existing diagnosis of ASD did not complete questionnaires, but were included in the prevalence analysis. Negative scores on all 3 questionnaires were observed for 60 of 79 boys. Positive scores on 1, 2, and 3 questionnaires were seen in 12 of 79, 3 of 79, and 4 of 79 boys, respectively. In addition to the 11 of 214 boys with a prior ASD diagnosis, 3 further boys were diagnosed with ASD, yielding a prevalence of 14 (6.5%) of 214, greater than that of boys in the UK general population. Premature birth was linked to having ASD, but did not fully explain the increased prevalence with more boys born <37 weeks scoring positively on the Social Communications Questionnaire and Children's Communication Checklist compared with those born at term. This study identified an increased prevalence of ASD at 1 UK hemophilia center. Prematurity was identified as a risk factor but did not fully explain the higher prevalence of ASD. Further investigation in the wider national/global hemophilia communities is warranted to determine whether this is an isolated finding.

Sections du résumé

Background UNASSIGNED
Autism spectrum disorders (ASDs) are diagnosed by social communication difficulties strong, narrow interests, and repetitive stereotyped behavior. An apparently-elevated prevalence of ASD at a major UK hemophilia center warranted investigation.
Objectives UNASSIGNED
To screen boys with hemophilia for difficulties in social communication and executive function and identify the prevalence and risk factors for ASD.
Methods UNASSIGNED
Parents of boys with hemophilia aged 5 to 16 years completed the Social Communication Questionnaire, Children's Communication Checklist, and the Behavior Rating Inventory of executive function. Prevalence and potential risk factors for ASD were evaluated. Boys with an existing diagnosis of ASD did not complete questionnaires, but were included in the prevalence analysis.
Results UNASSIGNED
Negative scores on all 3 questionnaires were observed for 60 of 79 boys. Positive scores on 1, 2, and 3 questionnaires were seen in 12 of 79, 3 of 79, and 4 of 79 boys, respectively. In addition to the 11 of 214 boys with a prior ASD diagnosis, 3 further boys were diagnosed with ASD, yielding a prevalence of 14 (6.5%) of 214, greater than that of boys in the UK general population. Premature birth was linked to having ASD, but did not fully explain the increased prevalence with more boys born <37 weeks scoring positively on the Social Communications Questionnaire and Children's Communication Checklist compared with those born at term.
Conclusion UNASSIGNED
This study identified an increased prevalence of ASD at 1 UK hemophilia center. Prematurity was identified as a risk factor but did not fully explain the higher prevalence of ASD. Further investigation in the wider national/global hemophilia communities is warranted to determine whether this is an isolated finding.

Identifiants

pubmed: 36891525
doi: 10.1016/j.rpth.2022.100013
pii: S2475-0379(22)02198-7
pmc: PMC9986098
doi:

Types de publication

Journal Article

Langues

eng

Pagination

100013

Informations de copyright

Crown Copyright © 2022 Published by Elsevier Inc. on behalf of International Society on Thrombosis and Haemostasis.

Références

Sleep Med. 2019 Mar;55:33-39
pubmed: 30743208
J Pediatr. 2014 Jan;164(1):20-5
pubmed: 24161222
Child Neuropsychol. 2002 Dec;8(4):249-57
pubmed: 12759822
Haemophilia. 2012 Mar;18(2):229-34
pubmed: 21910786
J Am Acad Child Adolesc Psychiatry. 2017 Jun;56(6):466-474
pubmed: 28545751
Autism Res. 2020 Nov;13(11):1856-1866
pubmed: 33460309
BMJ Open. 2013 Oct 16;3(10):e003219
pubmed: 24131525
Nat Rev Dis Primers. 2021 Jun 24;7(1):45
pubmed: 34168126
Dev Med Child Neurol. 2001 Dec;43(12):809-18
pubmed: 11769267
MMWR Surveill Summ. 2020 Mar 27;69(4):1-12
pubmed: 32214087
Pediatr Blood Cancer. 2020 Jan;67(1):e28004
pubmed: 31595670
J Am Acad Child Adolesc Psychiatry. 2007 Oct;46(10):1324-1332
pubmed: 17885574
JAMA Pediatr. 2021 Jun 1;175(6):e210054
pubmed: 33779707
J Am Acad Child Adolesc Psychiatry. 1996 Aug;35(8):1078-85
pubmed: 8755805
J Autism Dev Disord. 2014 Jan;44(1):31-40
pubmed: 23719853
Curr Opin Neurol. 2013 Apr;26(2):146-53
pubmed: 23406909
Front Neurosci. 2013 Oct 17;7:180
pubmed: 24146637
J Child Psychol Psychiatry. 1998 Sep;39(6):879-91
pubmed: 9758196
Pediatrics. 2018 Sep;142(3):
pubmed: 30076190
Br J Psychiatry. 2009 Jun;194(6):500-9
pubmed: 19478287
Lancet. 2022 Jan 15;399(10321):271-334
pubmed: 34883054
Clin Pediatr (Phila). 2013 Oct;52(10):950-9
pubmed: 23872342
J Autism Dev Disord. 1994 Oct;24(5):659-85
pubmed: 7814313
Mol Psychiatry. 2018 May;23(5):1198-1204
pubmed: 28439105
Pediatrics. 2007 Sep;120(3):584-93
pubmed: 17766532
Br J Psychiatry. 1999 Nov;175:444-51
pubmed: 10789276
Front Med (Lausanne). 2022 Feb 02;9:794456
pubmed: 35186990
Haemophilia. 2009 Jan;15(1):184-92
pubmed: 18702617
Lancet. 2006 Jul 15;368(9531):210-5
pubmed: 16844490

Auteurs

Melanie Bladen (M)

Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK.
UCL Great Ormond Street Institute of Child Health, London, UK.

Nicola Thorpe (N)

Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK.

Deborah Ridout (D)

UCL Great Ormond Street Institute of Child Health, London, UK.

Alpha Barrie (A)

Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK.

Emma McGibbon (E)

Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK.

Abigail Mance (A)

Great Ormond Street Hospital for Children NHS Foundation Trust, London, UK.

Lucy Watson (L)

University of Surrey, London, UK.

Eleanor Main (E)

UCL Great Ormond Street Institute of Child Health, London, UK.

Classifications MeSH