Mitchell J J, Vekemans M, Luscombe S, Hayden M, Weber B, Richter A, Sparkes R, Kojis T, Watters G, Der Kaloustian V M
Divisions of Medical Genetics, Montreal Children's Hospital, McGill Centre for Human Genetics, Quebec, Canada.
Am J Med Genet. 1994 Feb 15;49(4):384-7. doi: 10.1002/ajmg.1320490406.
A mentally retarded male with dysmorphic features was found to have a de novo 46,XY,inv dup(8) (p.23.1-->12). Confirmation of the segments duplicated in the rearrangement was achieved by biochemical analysis of glutathione reductase, which maps to 8p21.1, and DNA studies using the chromosome specific probe y-19-1D (D85131), which maps to 8p21. Assay of cathepsin B, which has been localised to 8p22, did not differ from controls with normal chromosomal constitution. DNA studies using the Defensin 1 gene probe, which maps to 8p23, showed a previously undetected deletion of that segment. We propose that the inverted tandem duplication/deletion arose as a single U-type exchange within an inversion loop.
一名具有畸形特征的智力发育迟缓男性被发现存在一个新发的46,XY,inv dup(8) (p.23.1-->12)。通过对定位于8p21.1的谷胱甘肽还原酶进行生化分析以及使用定位于8p21的染色体特异性探针y-19-1D (D85131)进行DNA研究,证实了重排中重复的片段。对定位于8p22的组织蛋白酶B的检测结果与染色体组成正常的对照无差异。使用定位于8p23的防御素1基因探针进行的DNA研究显示该片段存在先前未检测到的缺失。我们推测反向串联重复/缺失是在倒位环内作为单个U型交换产生的。