Marked phenotypic variable expression among brothers with duplication of Xq27.1 involving the SOX3 gene.


Journal

Journal of pediatric endocrinology & metabolism : JPEM
ISSN: 2191-0251
Titre abrégé: J Pediatr Endocrinol Metab
Pays: Germany
ID NLM: 9508900

Informations de publication

Date de publication:
26 Mar 2020
Historique:
received: 25 03 2015
accepted: 17 07 2015
pubmed: 10 9 2015
medline: 15 12 2020
entrez: 10 9 2015
Statut: ppublish

Résumé

We describe four phenotypically different brothers who share the same microduplication of Xq27.1, which contains the SOX3 gene. SOX3 mutations have been associated with growth hormone deficiency, variable degrees of additional pituitary hormone deficiencies, and mental retardation. SOX3 also appears to play an important role in pharyngeal arch segmentation that gives rise to craniofacial structures. While these four brothers have inherited the same mutation, they manifest a spectrum of phenotypes, ranging from complete, multiple pituitary hormone deficiencies to no apparent pituitary hormone deficiency with or without craniopharyngeal/facial dysmorphisms. We look to the literature to provide putative explanations for the variable expression of the brothers' shared SOX3 mutation.

Identifiants

pubmed: 26352083
doi: 10.1515/jpem-2015-0131
pii: /j/jpem.ahead-of-print/jpem-2015-0131/jpem-2015-0131.xml
doi:
pii:

Substances chimiques

SOX3 protein, human 0
SOXB1 Transcription Factors 0

Types de publication

Case Reports Journal Article

Langues

eng

Sous-ensembles de citation

IM

Pagination

443-447

Auteurs

Elizabeth T Rosolowsky (ET)

Division of Endocrinology, Department of Pediatrics, University of Alberta, 4-509 11405-87th Ave, Edmonton, AB Canada T6G1C9, Canada, Phone: +780-248-5483, Fax: +888-775-8879.

Robert Stein (R)

Division of Pediatric Endocrinology, Schulich School of Medicine, Western University, London, Ontario, Canada.

Seth D Marks (SD)

Section of Pediatric Endocrinology and Metabolism, Department of Pediatrics and Child Health, University of Manitoba, Winnipeg, Manitoba, Canada.

Norma Leonard (N)

Department of Medical Genetics, University of Alberta, Edmonton, Alberta, Canada.

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Classifications MeSH