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SOFT syndrome caused by compound heterozygous mutations of POC1A and its skeletal manifestation.

Authors :
Ko JM
Jung S
Seo J
Shin CH
Cheong HI
Choi M
Kim OH
Cho TJ
Source :
Journal of human genetics [J Hum Genet] 2016 Jun; Vol. 61 (6), pp. 561-4. Date of Electronic Publication: 2016 Jan 21.
Publication Year :
2016

Abstract

SOFT syndrome (MIM614813) is an extremely rare primordial dwarfism characterized by short stature, onychodysplasia, facial dysmorphism and hypotrichosis, which is caused by biallelic mutations in the POC1A gene. Only 19 patients with mutation-confirmed SOFT syndrome have been reported to date, all of whom carried homozygous variants that were strongly associated with consanguineous marriages. We report an 8.5-year-old boy with SOFT syndrome showing primordial dwarfism, no effect of growth-hormone therapy and skeletal dysplasia. This is the first report of compound heterozygous variants in POC1A, one previously reported and the other novel. A characteristic skeletal manifestation is reported.

Details

Language :
English
ISSN :
1435-232X
Volume :
61
Issue :
6
Database :
MEDLINE
Journal :
Journal of human genetics
Publication Type :
Academic Journal
Accession number :
26791357
Full Text :
https://doi.org/10.1038/jhg.2015.174