McGhee E M, Qu Y, Wohlferd M M, Goldberg J D, Norton M E, Cotter P D
Department of Pediatrics - Medical Genetics, University of California San Francisco, San Francisco, CA 94143, USA.
Clin Genet. 2001 Apr;59(4):274-8. doi: 10.1034/j.1399-0004.2001.590410.x.
Monosomy for the short arm of chromosome 18 is one of the most frequent autosomal deletions observed. While most cases result from terminal deletion of 18p, 16% of cases reported were as a result of an unbalanced whole arm translocation resulting in monosomy 18p. The origin and structure of these derivative chromosomes were reported in only a few cases. We report the prenatal diagnosis and characterization of a new case of monosomy 18p as a result of an unbalanced whole arm translocation. Amniocentesis was performed at 15 weeks of gestation on a 34-year-old woman initially referred for advanced maternal age. Holoprosencephaly was identified by ultrasound at the time of amniocentesis. Karyotype analysis showed an unbalanced whole arm translocation between the long arm of one chromosome 18 and the long arm of one chromosome 22, 45,XX,der(18;22)(q10;q10), in all metaphases. In effect, the fetus had monosomy for 18p. Parental karyotypes were normal, suggesting a de novo origin for the der(18;22). Fluorescence in situ hybridization (FISH) analysis was performed with alpha-satellite probes D18Z1 and D14Z1/D22Z1 to identify the origin of the centromere on the der(18;22). Signal was observed with both probes, indicating that the centromere was composed of alpha-satellite DNA from both constituent chromosomes. Genotyping of the fetus and her parents with chromosome 18p STS marker D18S391 showed only the paternal 187 bp allele was present in the fetus, indicating that it was the maternal chromosome 18 involved in the der(18;22). This case and previous reports show that de novo unbalanced whole arm translocations are more likely to retain alpha-satellite sequences from the two chromosomes involved.
18号染色体短臂单体是最常见的常染色体缺失之一。虽然大多数病例是由于18p的末端缺失所致,但报告的病例中有16%是由于不平衡的整条臂易位导致18p单体。这些衍生染色体的起源和结构仅在少数病例中有报道。我们报告了一例因不平衡的整条臂易位导致18p单体的产前诊断和特征。一名34岁的女性因高龄初产最初前来就诊,在妊娠15周时进行了羊水穿刺。羊水穿刺时超声检查发现全前脑畸形。核型分析显示,在所有中期相中,一条18号染色体的长臂与一条22号染色体的长臂之间发生了不平衡的整条臂易位,核型为45,XX,der(18;22)(q10;q10)。实际上,胎儿为18p单体。父母的核型正常,提示der(18;22)为新发。使用α-卫星探针D18Z1和D14Z1/D22Z1进行荧光原位杂交(FISH)分析,以确定der(18;22)上着丝粒的起源。两个探针均观察到信号,表明着丝粒由来自两条组成染色体的α-卫星DNA组成。使用18号染色体p臂STS标记D18S391对胎儿及其父母进行基因分型,结果显示胎儿中仅存在父源的187 bp等位基因,表明参与der(18;22)的是母源18号染色体。该病例及既往报告表明,新发的不平衡整条臂易位更有可能保留来自两条相关染色体的α-卫星序列。