Delobel B, Delannoy V, Pini G, Zapella M, Tardieu M, Vallée L, Croquette M F
Centre de Génétique Chromosomique, Hôpital Saint Antoine, Lille, France.
Am J Med Genet. 1998 Nov 16;80(3):273-80.
We report on an interstitial duplication of the long arm of chromosome 11 [46XX,dup(11) (q23.3)] in a girl with atypical Rett syndrome (RS). This case was discovered during a systematic cytogenetic study of RS. Fluorescent in situ hybridization including total chromosome painting and use of regional specific YAC, cosmid and plasmid probes, was used to confirm the chromosome 11q involvement and to identify the landmarks of the smallest 11q duplication reported to date. The findings are compared to cases of trisomy 11q reported previously, all of which have a larger duplication and different clinical manifestations. Surprisingly, mental retardation and behavior disorders are less severe in these cases.
我们报告了一名患有非典型瑞特综合征(RS)的女孩存在11号染色体长臂的间质重复[46XX,dup(11)(q23.3)]。该病例是在对RS进行系统细胞遗传学研究期间发现的。荧光原位杂交,包括全染色体涂染以及使用区域特异性酵母人工染色体(YAC)、黏粒和质粒探针,用于确认11号染色体长臂的受累情况,并确定迄今报道的最小11号染色体长臂重复的界标。将这些发现与先前报道的11号染色体长臂三体病例进行了比较,所有这些病例的重复片段更大且临床表现不同。令人惊讶的是,这些病例中的智力发育迟缓及行为障碍没那么严重。