| Literature DB >> 26791357 |
Jung Min Ko1,2, Soyoon Jung1, Jieun Seo3, Choong Ho Shin1, Hae Il Cheong1,2, Murim Choi3, Ok-Hwa Kim4, Tae-Joon Cho5.
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.Entities:
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Year: 2016 PMID: 26791357 DOI: 10.1038/jhg.2015.174
Source DB: PubMed Journal: J Hum Genet ISSN: 1434-5161 Impact factor: 3.172