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SOFT syndrome caused by compound heterozygous mutations of POC1A and its skeletal manifestation.
Ko, Jung Min; Jung, Soyoon; Seo, Jieun; Shin, Choong Ho; Cheong, Hae Il; Choi, Murim; Kim, Ok-Hwa; Cho, Tae-Joon.
Afiliação
  • Ko JM; Department of Pediatrics, Seoul National University College of Medicine, Seoul, Korea.
  • Jung S; Research Coordination Center for Rare Diseases, Seoul National University College of Medicine, Seoul, Korea.
  • Seo J; Department of Pediatrics, Seoul National University College of Medicine, Seoul, Korea.
  • Shin CH; Department of Biomedical Sciences, Seoul National University College of Medicine, Seoul, Korea.
  • Cheong HI; Department of Pediatrics, Seoul National University College of Medicine, Seoul, Korea.
  • Choi M; Department of Pediatrics, Seoul National University College of Medicine, Seoul, Korea.
  • Kim OH; Research Coordination Center for Rare Diseases, Seoul National University College of Medicine, Seoul, Korea.
  • Cho TJ; Department of Biomedical Sciences, Seoul National University College of Medicine, Seoul, Korea.
J Hum Genet ; 61(6): 561-4, 2016 Jun.
Article em En | MEDLINE | ID: mdl-26791357
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.
Assuntos

Texto completo: 1 Base de dados: MEDLINE Assunto principal: Fenótipo / Esqueleto / Anormalidades Múltiplas / Proteínas / Heterozigoto / Mutação Limite: Humans / Infant / Male Idioma: En Revista: J Hum Genet Assunto da revista: GENETICA MEDICA Ano de publicação: 2016 Tipo de documento: Article

Texto completo: 1 Base de dados: MEDLINE Assunto principal: Fenótipo / Esqueleto / Anormalidades Múltiplas / Proteínas / Heterozigoto / Mutação Limite: Humans / Infant / Male Idioma: En Revista: J Hum Genet Assunto da revista: GENETICA MEDICA Ano de publicação: 2016 Tipo de documento: Article