Intellectual and Behavioral Phenotypes of Smith-Magenis Syndrome: Comparisons between Individuals with a 17p11.2 Deletion and Pathogenic

17p11.2 deletion Smith–Magenis syndrome behavioral problems intellectual disability pathogenic RAI1 variant rare disorders

Journal

Genes
ISSN: 2073-4425
Titre abrégé: Genes (Basel)
Pays: Switzerland
ID NLM: 101551097

Informations de publication

Date de publication:
25 07 2023
Historique:
received: 29 06 2023
revised: 22 07 2023
accepted: 23 07 2023
medline: 28 8 2023
pubmed: 26 8 2023
entrez: 26 8 2023
Statut: epublish

Résumé

Smith-Magenis syndrome (SMS) is a rare genetic neurodevelopmental disorder caused by a 17p11.2 deletion or pathogenic variant in the We reviewed available clinical records from individuals (aged 0-45 years) with SMS, ascertained through a Dutch multidisciplinary SMS specialty clinic. We included a total of 66 individuals ( The results of this study suggest that 17p11.2 deletions are associated with a lower level of intellectual functioning and less internalizing of problems compared to pathogenic

Identifiants

pubmed: 37628566
pii: genes14081514
doi: 10.3390/genes14081514
pmc: PMC10453904
pii:
doi:

Types de publication

Review Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

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Auteurs

Cathelijne C Linders (CC)

Advisium, 's Heeren Loo, 3818 LA Amersfoort, The Netherlands.
Department of Genetics, University Medical Centre Utrecht, 3584 CX Utrecht, The Netherlands.

Agnies M van Eeghen (AM)

Advisium, 's Heeren Loo, 3818 LA Amersfoort, The Netherlands.
Department of Pediatrics, Emma Children's Hospital, Amsterdam, University Medical Center, 1105 AZ Amsterdam, The Netherlands.

Janneke R Zinkstok (JR)

Department of Psychiatry, Radboud University Medical Centre, 6500 HB Nijmegen, The Netherlands.
Karakter Child and Adolescent Psychiatry, 6501 BB Nijmegen, The Netherlands.
Department of Psychiatry and Brain Center, University Medical Center Utrecht, 3584 CG Utrecht, The Netherlands.

Marie-José van den Boogaard (MJ)

Department of Genetics, University Medical Centre Utrecht, 3584 CX Utrecht, The Netherlands.

Erik Boot (E)

Advisium, 's Heeren Loo, 3818 LA Amersfoort, The Netherlands.
The Dalglish Family 22q Clinic, University Health Network, Toronto, ON M5G 2C4, Canada.
Department of Psychiatry & Neuropsychology, Maastricht University, 6200 AB Maastricht, The Netherlands.

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Classifications MeSH