Polymeropoulos M H, Ide S E, Wright M, Goodship J, Weissenbach J, Pyeritz R E, Da Silva E O, Ortiz De Luna R I, Francomano C A
Gene Mapping Unit, Laboratory of Genetic Disease Research, National Center for Human Genome Research, National Institutes of Health, Building 49, Bethesda, Maryland, 20892, USA.
Genomics. 1996 Jul 1;35(1):1-5. doi: 10.1006/geno.1996.0315.
Ellis-van Creveld syndrome (EVC) is an autosomal recessive disorder characterized by disproportionate dwarfism, polydactyly, and congenital heart disease. This rare disorder is found with increased frequency among the Old Order Amish community in Lancaster County, Pennsylvania. We have used linkage analysis to localize the gene responsible for the EVC phenotype in nine interrelated Amish pedigrees and three unrelated families from Mexico, Ecuador, and Brazil. We now report the linkage for the Ellis-van Creveld syndrome gene to markers on the distal short arm of human chromosome 4, with Zmax = 6.91 at theta = 0.02 for marker HOX7, in a region proximal to the FGFR3 gene responsible for the achondroplasia phenotype.
埃利斯-范克里维尔德综合征(EVC)是一种常染色体隐性疾病,其特征为不成比例的侏儒症、多指(趾)畸形和先天性心脏病。这种罕见疾病在宾夕法尼亚州兰开斯特县的旧秩序阿米什人群体中出现的频率较高。我们利用连锁分析,在9个有亲缘关系的阿米什家系以及来自墨西哥、厄瓜多尔和巴西的3个无亲缘关系的家庭中,对导致EVC表型的基因进行定位。我们现在报告,埃利斯-范克里维尔德综合征基因与人类4号染色体短臂远端的标记物连锁,对于标记物HOX7,在θ = 0.02时Zmax = 6.91,位于导致软骨发育不全表型的FGFR3基因近端的一个区域。