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