Goodship J, Curtis A, Cross I, Brown J, Emslie J, Wolstenholme J, Bhattacharya S, Burn J
Department of Human Genetics, Newcastle upon Tyne.
J Med Genet. 1992 Jul;29(7):451-4.
A 2 year old girl presented with developmental delay and subtle dysmorphic features suggestive of Wolf-Hirschhorn syndrome (WHS). High resolution chromosome analysis was normal in the child and both parents. Molecular analysis indicated that the child had not inherited a maternal allele of probes from 4p16, confirming the clinical diagnosis. Prenatal diagnosis in the next pregnancy showed that again the fetus had no maternal allele for probes mapping to 4p16. Fluorescent in situ hybridisation in the mother showed a submicroscopic translocation, t(4;10). A normal karyotype in a child with clinical features of WHS is an indication for further investigation.
一名2岁女童出现发育迟缓及轻微畸形特征,提示可能患有沃尔夫-赫希霍恩综合征(WHS)。该患儿及其父母的高分辨率染色体分析结果均正常。分子分析表明,患儿未从母亲处继承4p16区域的等位基因,从而证实了临床诊断。下一胎的产前诊断显示,胎儿同样未继承位于4p16区域的母亲等位基因。对母亲进行的荧光原位杂交显示存在亚显微易位,即t(4;10)。具有WHS临床特征但染色体核型正常的患儿需要进一步检查。