Clayton-Smith J, Webb T, Cheng X J, Pembrey M E, Malcolm S
Department of Medical Genetics, St Mary's Hospital, Whitfield Park, Manchester, UK.
J Med Genet. 1993 Jun;30(6):529-31. doi: 10.1136/jmg.30.6.529.
Duplications of the proximal long arm of chromosome 15 have been seen in the Prader-Willi syndrome (PWS), and in subjects without the Prader-Willi phenotype but with other clinical features including short stature, diabetes, anal and jejunal atresia, and acanthosis nigricans. The non-PWS subjects all had different phenotypes despite the identical findings on cytogenetic analysis. A normal phenotype has also been observed in patients with similar duplications. We report a further patient with a duplication of 15q11-13 which was detected cytogenetically and confirmed on molecular genetic analysis. She has developmental delay, particularly concerning the acquisition of speech, and an ataxic gait. These are interesting clinical features in view of the association of Angelman syndrome with abnormalities of 15q11-13.
15号染色体近端长臂重复在普拉德-威利综合征(PWS)患者中已被发现,在无普拉德-威利综合征表型但有其他临床特征(包括身材矮小、糖尿病、肛门和空肠闭锁以及黑棘皮病)的个体中也有发现。尽管细胞遗传学分析结果相同,但非普拉德-威利综合征患者均有不同的表型。在有类似重复的患者中也观察到了正常表型。我们报告了另一例15q11 - 13重复的患者,该重复通过细胞遗传学检测发现,并经分子遗传学分析证实。她有发育迟缓,尤其是在语言习得方面,还有共济失调步态。鉴于天使综合征与15q11 - 13异常有关,这些都是有趣的临床特征。