SOFT syndrome caused by compound heterozygous mutations of POC1A and its skeletal manifestation.
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.
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Base de dados:
MEDLINE
Assunto principal:
Fenótipo
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Esqueleto
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Anormalidades Múltiplas
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Proteínas
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Heterozigoto
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Mutação
Limite:
Humans
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Infant
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Male
Idioma:
En
Revista:
J Hum Genet
Assunto da revista:
GENETICA MEDICA
Ano de publicação:
2016
Tipo de documento:
Article