Igarashi M, Tsukahara M, Sugio Y, Katayama K, Kajii T
Ann Genet. 1985;28(4):241-4.
A 10-year-old girl with short stature and other features of Turner's syndrome was found to be a mosaic consisting of 46,X,i(Xq) and 47,XX,+13 cell lines, a hitherto undescribed situation. She had none of the clinical features of trisomy 13 syndrome, with a possible exception of postaxial polydactyly of the left foot. Her PHA-stimulated blood lymphocytes and EB virus-transformed B lymphocytes both revealed the Xi(Xq)/XX,+13 mosaicism, while her skin fibroblasts showed an exclusively 46,X,i(Xq) karyotype. Studies using Q-and R-banding heteromorphisms as markers indicated that the patient started as a 13 trisomic zygote resulting from a maternal meiotic error, followed by the loss of chromosome 13 at an early mitotic division. C-banding analysis revealed two C banding blocks in the iso X chromosome, an indication that the chromosome was dicentric. BrdU-Hoechst-Giemsa analysis revealed that the iso X chromosome was late-replicating with both its arms either synchronously or asynchronously replicating. The iso X chromosome was thus designated as idic (Xq)(p11:p11). In view of the presence of the XX cell line, it was concluded that the patient started as an XX,+13 zygote, followed by two mitotic events, the loss of a chromosome 13 and the formation of the iso X chromosome, occurring either simultaneously or in succession.
一名患有特纳综合征矮小身材及其他特征的10岁女孩被发现是由46,X,i(Xq)和47,XX,+13细胞系组成的嵌合体,这是一种此前未被描述过的情况。她没有13三体综合征的任何临床特征,可能的例外是左脚轴后多指。她的PHA刺激的血液淋巴细胞和EB病毒转化的B淋巴细胞均显示出Xi(Xq)/XX,+13嵌合现象,而她的皮肤成纤维细胞显示出仅为46,X,i(Xq)的核型。使用Q带和R带异态性作为标记的研究表明,该患者最初是由于母亲减数分裂错误产生的13三体合子,随后在早期有丝分裂中丢失了13号染色体。C带分析显示等臂X染色体中有两个C带块,这表明该染色体是双着丝粒的。BrdU- Hoechst-吉姆萨分析显示等臂X染色体复制较晚,其双臂同步或异步复制。因此,等臂X染色体被指定为idic(Xq)(p11:p11)。鉴于存在XX细胞系,得出的结论是,该患者最初是XX,+13合子,随后发生了两个有丝分裂事件,即丢失一条13号染色体和形成等臂X染色体,这两个事件可能同时发生或相继发生。