Tsuji K, Narahara K, Kikkawa K, Murakami M, Yokoyama Y, Ninomiya S, Seino Y
Department of Pediatrics, Okayama University Medical School, Japan.
Am J Med Genet. 1994 Jan 1;49(1):98-102. doi: 10.1002/ajmg.1320490119.
Craniosynostosis (CRS) is frequently seen in the del(7p) syndrome, and the gene for this cranial anomaly (CRS1) has been assigned to 7p21. We present a 3-year-old boy with CRS involving the sagittal and coronal sutures, who had a de novo and apparently balanced translocation, t(6;7)(q16.2;p15.3). Southern blot analysis of several loci on 7p14-->pter showed that the patient was heterozygous for HOX1I and IL6, possibly homozygous for D7S149, but hemizygous for D7S135 with a loss of the paternal allele. These findings suggest the localization of a candidate gene for CRS1 to be on 7p15.3 in the close proximity to the D7S135 locus.
颅缝早闭(CRS)在7号染色体短臂缺失(del(7p))综合征中较为常见,这种颅骨异常的基因(CRS1)已被定位于7p21。我们报告一名3岁男孩,患有累及矢状缝和冠状缝的颅缝早闭,其存在一个新发且明显平衡的易位,即t(6;7)(q16.2;p15.3)。对7p14至末端(pter)多个位点的Southern印迹分析显示,该患者HOX1I和IL6为杂合子,D7S149可能为纯合子,但D7S135为半合子,父本等位基因缺失。这些发现提示CRS1候选基因定位于7p15.3,紧邻D7S135位点。