Kousseff B G, Diamond T, Essig Y, Miller K, Tedesco T
Division of Medical Genetics, University of South Florida, Tampa 33612-4799.
Am J Med Genet. 1987 Dec;28(4):803-11. doi: 10.1002/ajmg.1320280404.
A 16-year-old boy with Prader-Labhart-Willi syndrome (PLWS) had hypotonia, feeding difficulties, failure to thrive, strabismus and bilateral inguinal hernias with cryptorchidism during infancy followed by hyperphagia, marked early-onset obesity with insulin-dependent diabetes mellitus and necrobiosis lipoidica diabeticorum, short stature, hypogonadotropic hypogonadism and some of the facial characteristics of the individuals with the PLWS. IQ is estimated around 90. Cytogenetic studies showed mosaicism: 45,X, t(Y;15) with partial deletion 15 (15pter----15q12); 46,X, t(Y;15), dic (15)(15pter----15q12::15q12----15pter) and 47, X, t(Y;15), dic(15), dic(15). The dic(15) was bisatellited, NOR-positive on both arms and represented inv dup(15). Thus, the 2 lines with the dic(15) showed partial trisomy 15 (15pter----15q12) and partial pentasomy 15 (15pter----15q12), respectively. The cell line ratios were different in lymphocyte and fibroblast cultures. The unique cytogenetic findings in this patient, the reports of a variety of chromosome 15 aberrations in PLWS, as well as aberrations of other chromosomes, suggest that the condition is a contiguous gene syndrome rather than an aneuploidy syndrome.
一名患有普拉德-威利综合征(PLWS)的16岁男孩,婴儿期出现肌张力减退、喂养困难、生长发育迟缓、斜视以及双侧腹股沟疝合并隐睾,随后出现食欲亢进、显著的早发性肥胖伴胰岛素依赖型糖尿病和糖尿病性类脂质渐进性坏死、身材矮小、低促性腺激素性性腺功能减退以及一些PLWS患者的面部特征。智商估计约为90。细胞遗传学研究显示为嵌合体:45,X,t(Y;15)伴15号染色体部分缺失(15pter----15q12);46,X,t(Y;15),dic(15)(15pter----15q12::15q12----15pter)以及47,X,t(Y;15),dic(15),dic(15)。dic(15)有双随体,双臂均为核仁组织区阳性,代表倒位重复(15)。因此,两条含有dic(15)的细胞系分别显示15号染色体部分三体(15pter----15q12)和部分五体(15pter----15q12)。淋巴细胞和成纤维细胞培养中的细胞系比例不同。该患者独特的细胞遗传学发现、PLWS中多种15号染色体畸变的报告以及其他染色体的畸变表明,该病症是一种邻接基因综合征而非非整倍体综合征。