Key Laboratory for Experimental Teratology of the Ministry of Education and Department of Medical Genetics, Shandong University School of Medicine, Jinan 250012, PR China.
Gene. 2012 May 10;499(1):48-51. doi: 10.1016/j.gene.2012.02.046. Epub 2012 Mar 3.
Human synpolydactyly (SPD), belonging to syndactyly (SD) II, is an inherited autosomal-dominant limb malformation characterized by SD of finger 3 or 4 or toe 4 or 5, usually with digit duplication. Previous studies have demonstrated that homeobox protein D13 (HOXD13) is responsible for this Mendelian disorder. In this paper, we report on a family with SPD - 7 members show typical SPD malformations. We used PCR and Sanger sequencing of DNA from peripheral blood samples and found an 8-Ala expansion in exon 1 of HOXD13 by mutation detection; this variant was absent in unaffected members and in 50 unaffected non-related subjects. This study further confirmed the correlation between SPD and alanine expansion in HOXD13.
人类并指畸形(SPD)属于并指畸形(SD)II 型,是一种遗传性常染色体显性肢体畸形,表现为手指 3 或 4 或脚趾 4 或 5 的并指,通常伴有数字重复。先前的研究表明,同源盒蛋白 D13(HOXD13)负责这种孟德尔疾病。在本文中,我们报道了一个具有 SPD 的家族 - 7 名成员表现出典型的 SPD 畸形。我们使用来自外周血样本的 PCR 和 Sanger 测序进行 DNA 检测,发现 HOXD13 外显子 1 中的 8-丙氨酸扩展突变;该变体在未受影响的成员和 50 名未受影响的非相关受试者中不存在。这项研究进一步证实了 SPD 与 HOXD13 中丙氨酸扩展之间的相关性。