Shen J J, Williams B J, Zipursky A, Doyle J, Sherman S L, Jacobs P A, Shugar A L, Soukup S W, Hassold T J
Department of Human Genetics, Case Western Reserve University School of Medicine, Cleveland, OH 44106, USA.
Am J Hum Genet. 1995 Apr;56(4):915-25.
There is an increased risk of leukemia in Down syndrome (DS) patients, with estimates ranging from 14 to 30 times the incidence rate observed for chromosomally normal children. Furthermore, one type of leukemia, called "transient leukemia" (TL), occurs almost exclusively in DS infants. The basis of the association between DS and leukemia is unknown, but we and others have hypothesized that it may be influenced by the mechanism of origin of the extra chromosome. Therefore, we initiated a cytogenetic and molecular study of nondisjunction in leukemia DS individuals. To date, we have obtained blood and/or tissue samples from 55 individuals consisting of 17 cases with TL, 7 cases of acute nonlymphocytic leukemia subtype M7 (ANLL-M7, or acute megakaryoblastic leukemia, postulated to be related to TL), and 31 cases of other forms of leukemia. Analysis of these cases suggests differences between DS children with TL and those with other types of leukemia or DS individuals with no history of leukemia. Specifically, the TL and ANLL-M7 cases have a highly significant increase in the frequency of "atypical" constitutional karyotypes (i.e., mosaic trisomies, rings, and/or isochromosomes) and are almost always male. Additionally, genetic mapping studies suggest an increase in the frequency of disomic homozygosity, especially in proximal 21q, in DS individuals with TL and ANLL-M7.
唐氏综合征(DS)患者患白血病的风险增加,据估计其发病率是染色体正常儿童的14至30倍。此外,有一种白血病,称为“暂时性白血病”(TL),几乎仅发生于DS婴儿。DS与白血病之间关联的基础尚不清楚,但我们和其他人推测,这可能受额外染色体起源机制的影响。因此,我们启动了一项针对DS白血病个体不分离现象的细胞遗传学和分子研究。迄今为止,我们已从55名个体中获取了血液和/或组织样本,其中包括17例TL患者、7例急性非淋巴细胞白血病M7亚型(ANLL-M7,即急性巨核细胞白血病,推测与TL有关)患者以及31例其他形式白血病患者。对这些病例的分析表明,患有TL的DS儿童与患有其他类型白血病的DS儿童或无白血病病史的DS个体之间存在差异。具体而言,TL和ANLL-M7病例中“非典型”染色体核型(即嵌合三体、环状染色体和/或等臂染色体)的频率显著增加,且几乎均为男性。此外,基因定位研究表明,患有TL和ANLL-M7的DS个体中,二体纯合性频率增加,尤其是在21号染色体长臂近端。