Cools Martine, Rooman Raoul P A, Wauters Jan, Jacqemyn Yves, Du Caju Marc V L
Department of Pediatrics, Antwerp University Hospital, Antwerp, Belgium.
Fertil Steril. 2004 Oct;82(4):923-5. doi: 10.1016/j.fertnstert.2004.02.129.
To describe a woman with a nonmosaic (45,X) form of Turner's syndrome who gave birth to a girl with 45,X Turner syndrome.
Patient report.
Outpatient clinic of a university hospital.
PATIENT(S): A woman with typical phenotypic features of Turner syndrome and a 45,X karyotype and her daughter with the same karyotype.
INTERVENTION(S): None.
MAIN OUTCOME MEASURE(S): Routine karyotype analysis on 200 white blood cells on two different occasions, on skin fibroblasts (1,000 mitoses) and on ovarian fibroblasts. Translocation of X-chromosome material was investigated by a complete X paint and fluorescent in situ hybridization analysis.
RESULT(S): The patient had a spontaneous puberty and became pregnant on three occasions. Her first daughter has a normal karyotype, the second pregnancy ended in spontaneous abortion, and after the third pregnancy, a girl was born with a 45,X karyotype. Karyotype analysis of a large number of mitoses in three different cell types failed to demonstrate any mosaicism. Translocation of X-chromosome material was ruled out by fluorescent in situ hybridization analysis with an X paint.
CONCLUSION(S): This is a rare case of pregnancy in a nonmosaic Turner syndrome patient and, to our knowledge, is the only one that resulted in a live-born baby with the same karyotype. Cryptic mosaicism could not be found despite thorough investigations. Some hypotheses are presented that may explain this unique event.
描述一名患有非嵌合型(45,X)特纳综合征的女性,她生下了一名患有45,X特纳综合征的女孩。
病例报告。
大学医院门诊。
一名具有特纳综合征典型表型特征且核型为45,X的女性及其核型相同的女儿。
无。
在两个不同时间对200个白细胞、皮肤成纤维细胞(1000个有丝分裂)和卵巢成纤维细胞进行常规核型分析。通过完整的X染色体涂染和荧光原位杂交分析研究X染色体物质的易位情况。
该患者自然青春期发育,并三次怀孕。她的第一个女儿核型正常,第二次怀孕以自然流产告终,第三次怀孕后,一名核型为45,X的女孩出生。对三种不同细胞类型中的大量有丝分裂进行核型分析,未发现任何嵌合现象。用X染色体涂染进行荧光原位杂交分析排除了X染色体物质的易位。
这是一例非嵌合型特纳综合征患者怀孕并分娩出核型相同的活产婴儿的罕见病例。据我们所知,这是唯一的一例。尽管进行了全面调查,但未发现隐匿性嵌合现象。本文提出了一些可能解释这一独特事件的假说。