Muñoz Rojas María Verónica, Gonçalves Luís Flávio
Clínica Materno-Fetal, Florianópolis, Brazil.
Am J Med Genet. 2002 Nov 22;113(2):193-9. doi: 10.1002/ajmg.10682.
Grebe-Quelce-Salgado chondrodystrophy is an autosomal recessive non-lethal skeletal dysplasia. Affected individuals have normal head, neck, and trunk skeleton, relatively normal humeri and femora, short and deformed radii, ulnae, tibiae, and fibulae, and severe abnormalities of hands and feet. Polydactyly is frequent. Digits present as globular appendages and are characteristic of the condition. The disease is caused by a missense mutation in the gene encoding cartilage-derived morphogenetic protein-1 (CDMP-1). Most cases described in the literature are from Brazil and, among these, all had ancestors from a particular region in the state of Bahia. We describe two cases of Grebe-Quelce-Salgado chondrodystrophy visualized by prenatal ultrasound. The patients presented in this report do not descend from the population of Bahia and, to our knowledge, case two is the only case with prenatal clinical diagnosis in a family with no previously affected children.
格雷贝-凯尔塞-萨尔加多软骨发育不良是一种常染色体隐性非致死性骨骼发育不良症。患病个体的头部、颈部和躯干骨骼正常,肱骨和股骨相对正常,桡骨、尺骨、胫骨和腓骨短且畸形,手足严重异常。多指畸形很常见。手指呈球状附属物,是该病的特征。这种疾病是由编码软骨衍生形态发生蛋白-1(CDMP-1)的基因中的错义突变引起的。文献中描述的大多数病例来自巴西,其中所有病例的祖先都来自巴伊亚州的一个特定地区。我们描述了两例经产前超声检查确诊的格雷贝-凯尔塞-萨尔加多软骨发育不良病例。本报告中的患者并非来自巴伊亚州人群,据我们所知,病例二是一个此前没有患病子女的家庭中唯一一例产前临床诊断病例。