Joosten A M, De Vos S, Van Opstal D, Brandenburg H, Gaillard J L, Vermeij-Keers C
Department of Anatomy, Erasmus University/University Hospital Rotterdam, The Netherlands.
Prenat Diagn. 1997 Mar;17(3):271-5. doi: 10.1002/(sici)1097-0223(199703)17:3<271::aid-pd51>3.0.co;2-p.
We describe a case of full monosomy 21 which was prenatally diagnosed in chorionic villi by fluorescent in situ hybridization (FISH). Because of intrauterine fetal death, a curettage was performed and cytogenetic analysis of skin fibroblasts confirmed the presence of monosomy 21 in fetal cells. DNA investigations showed a paternal origin of the single chromosome 21. Inspection and autopsy of the fetus revealed several congenital malformations. Some of them have been reported in earlier studies of monosomy 21; others concern new observations. Regarding the eye, the following abnormalities were microscopically observed: absence of the anterior and posterior eye chambers, aniridy, a hypoplastic ciliary body, Peter's anomaly, and a double retina with secondary dysplasia. In addition, malformations of the extremities were seen: partial, proximal syndactyly of digits 3 and 4 of the right hand; pes varus position of the right foot; and transverse reduction defect at the tarsals of the left foot. To our knowledge, this is the first case in which full monosomy 21 has been proven.
我们描述了一例通过荧光原位杂交(FISH)在绒毛膜绒毛中产前诊断出的21号染色体完全单体病例。由于宫内胎儿死亡,进行了刮宫术,皮肤成纤维细胞的细胞遗传学分析证实胎儿细胞中存在21号染色体单体。DNA研究表明单条21号染色体源自父亲。对胎儿进行检查和尸检发现了几种先天性畸形。其中一些在先前关于21号染色体单体的研究中已有报道;其他则是新的观察结果。关于眼睛,显微镜下观察到以下异常:无前房和后房、无虹膜、睫状体发育不全、彼得异常以及伴有继发性发育异常的双层视网膜。此外,还发现了四肢畸形:右手第3和4指部分近端并指;右脚内翻;左脚跗骨横向发育不全。据我们所知,这是首例经证实的21号染色体完全单体病例。