Roume J, Genin E, Cormier-Daire V, Ma H W, Mehaye B, Attie T, Razavi-Encha F, Fallet-Bianco C, Buenerd A, Clerget-Darpoux F, Munnich A, Le Merrer M
Unité de Recherches sur les Handicaps Génétiques de lEnfant, INSERMU. 393, France.
Am J Hum Genet. 1998 Oct;63(4):1095-101. doi: 10.1086/302062.
Meckel syndrome (MKS) is a rare autosomal recessive lethal condition of unknown origin, characterized by (i) an occipital meningo-encephalocele with (ii) enlarged kidneys, with multicystic dysplasia and fibrotic changes in the portal area of the liver and with ductal proliferation, and (iii) postaxial polydactyly. A gene responsible for MKS in Finland has been mapped to chromosome 17q21-q24. Studying a subset of Middle Eastern and northern African MKS families, we have recently excluded the chromosome 17 region and have suggested a genetic heterogeneity. In the present study, we report on the mapping of a second MKS locus (MKS2) to chromosome 11q13, by homozygosity mapping in seven families that do not show linkage to chromosome 17q21-q24 (maximum LOD score 4.41 at recombination fraction .01). Most interestingly, the affected fetuses of southern Tunisian ancestry shared a particular haplotype at loci D11S911 and D11S906, suggesting that a founder effect is involved. Our observation gives support to the clinical and genetic heterogeneity of MKS.
梅克尔综合征(MKS)是一种罕见的常染色体隐性致死性疾病,病因不明,其特征为:(i)枕部脑膜脑膨出;(ii)肾脏增大,伴有多囊性发育异常以及肝脏门区的纤维化改变和导管增生;(iii)轴后多指畸形。芬兰一个与MKS相关的基因已被定位于17号染色体q21 - q24区域。在对中东和北非地区一部分MKS家系进行研究时,我们最近排除了17号染色体区域,并提出存在遗传异质性。在本研究中,我们通过对7个与17号染色体q21 - q24区域无连锁关系的家系进行纯合性定位(重组率为0.01时最大对数优势分数为4.41),报告了第二个MKS基因座(MKS2)定位于11号染色体q13区域。最有意思的是,突尼斯南部血统的患病胎儿在D11S911和D11S906基因座共享一种特定单倍型,提示存在奠基者效应。我们的观察结果支持了MKS在临床和遗传方面的异质性。