Pettigrew A L, Gollin S M, Greenberg F, Riccardi V M, Ledbetter D H
Institute for Molecular Genetics, Baylor College of Medicine, Houston, Texas 77030.
Am J Med Genet. 1987 Dec;28(4):791-802. doi: 10.1002/ajmg.1320280403.
We describe an apparent duplication of proximal 15q, i.e., 15q11q12 or 15q12q13 in two patients. Prometaphase chromosome analysis, C-banding and distamycin A/DAPI staining were used to exclude a translocation between the abnormal 15 homolog and another chromosome. The 2 patients have many manifestations of the Prader-Willi syndrome (PWS) including at least 5 of the following: obesity, compulsive eating, mental retardation, short stature, central hypotonia, hypogonadism, small hands and feet, hypopigmentation, and feeding problems in infancy. Results of high resolution chromosome analysis of the parents of both patients were normal. A comparison between these patients and 2 subjects from previous reports demonstrates phenotypic heterogeneity among the duplication 15q PWS patients. Two patients have the hypopigmentation seen in chromosomally normal and deletion PWS patients. These cases add to the variety of chromosome 15 aberrations which are associated with PWS.
我们描述了两例患者近端15q即15q11q12或15q12q13的明显重复。使用前中期染色体分析、C带染色和放线菌素A/ 4,6-二脒基-2-苯基吲哚(DAPI)染色来排除异常的15号同源染色体与另一染色体之间的易位。这两名患者有许多普拉德-威利综合征(PWS)的表现,包括以下至少5种:肥胖、强迫性进食、智力发育迟缓、身材矮小、中枢性肌张力减退、性腺功能减退、手脚小、色素减退以及婴儿期喂养问题。两名患者父母的高分辨率染色体分析结果均正常。将这些患者与之前报道的2名受试者进行比较,显示15q重复的PWS患者之间存在表型异质性。两名患者出现了染色体正常和缺失型PWS患者中所见的色素减退。这些病例增加了与PWS相关的15号染色体畸变的种类。