Minelli A, Floridia G, Rossi E, Clementi M, Tenconi R, Camurri L, Bernardi F, Hoeller H, Previde Re C, Maraschio P
Università di Pavia, Italy.
Hum Genet. 1993 Oct;92(4):391-6. doi: 10.1007/BF01247342.
Ten patients with inverted duplication of 8p (inv dup 8p) were studied with cytogenetic, biochemical and molecular techniques. The duplication for the region 8p12-p22 was always associated with a deletion of the locus D8S7 (mapped in 8p23.1) as demonstrated with the probe pSW50 by both in situ hybridization and Southern blot. Restriction fragment length polymorphisms detected by probes pSW50 (1 case) and by pG2LPL35 (locus LPL) (two cases) were informative as to a maternal origin of the anomaly. The activity of glutathione reductase, whose gene maps in the duplicated region at 8p21.1, was increased in all patients. The recognizable phenotype of inv dup 8p includes neonatal hypotonia, prominent forehead, large mouth with everted lower lip, abnormally shaped large ears, brain malformations and severe mental retardation. Our findings indicate that the chromosome rearrangement is homogeneous at least for the presence of the deletion and support the hypothesis of a common mechanism of origin.
我们运用细胞遗传学、生化和分子技术对10例8号染色体短臂倒位重复(inv dup 8p)患者进行了研究。通过原位杂交和Southern印迹法使用探针pSW50证实,8p12 - p22区域的重复总是与位于8p23.1的D8S7位点缺失相关。探针pSW50(1例)和pG2LPL35(LPL位点)(2例)检测到的限制性片段长度多态性提示该异常起源于母体。谷胱甘肽还原酶的活性在所有患者中均升高,其基因定位于重复区域8p21.1。inv dup 8p可识别的表型包括新生儿肌张力减退、前额突出、大嘴伴下唇外翻、形状异常的大耳朵、脑畸形和严重智力发育迟缓。我们的研究结果表明,至少就缺失的存在而言,染色体重排是一致的,并支持共同起源机制的假说。