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Am J Med Genet ; 37(2): 213-23, 1990 Oct.
Artigo em Inglês | MEDLINE | ID: mdl-2248288

RESUMO

GAPO syndrome was described in 12 patients from 7 families. Constant manifestations include dwarfism, alopecia, pseudoanodontia, and a peculiar, "geriatric" facial appearance. We describe the autopsy findings and all available clinical data on one deceased patient and his living affected sister, previously reported as short abstracts (Epps et al.: Cienc Cult 29(Suppl):740, 1977; Wajntal et al.: Cienc Cult 34(Suppl):705, 1982). Both had the characteristic anomalies of this syndrome but optic atrophy was absent; instead, they had glaucoma and keratoconus; hypogonadism was present in both patients. Biopsy and autopsy findings show that the GAPO syndrome is a dyshistogenetic sequence due to accumulation of extracellular material and thus should be called GAPO dysplasia. We suggest that the basic defect in this autosomal recessive disorder is possibly related to a lack of breakdown of the extracellular components, perhaps due to an enzyme deficiency involved in the metabolism of extracellular matrix.


Assuntos
Anormalidades Múltiplas/genética , Alopecia/genética , Anodontia/genética , Doenças do Tecido Conjuntivo/genética , Nanismo/genética , Atrofia Óptica/genética , Adulto , Criança , Face/anormalidades , Feminino , Seguimentos , Genes Recessivos , Humanos , Masculino , Linhagem , Síndrome
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