Depraetere M, Dehauwere R, Mariën P, Fryns J P
Department of Paediatrics, University of Leuven, Belgium.
Genet Couns. 1995;6(4):303-7.
Severe axial mesodermal dysplasia spectrum in an infant of a diabetic mother: We report a 6 weeks old girl with manifestations of the caudal regression sequence. At birth she presented severe contractures of both lower extremities, bilateral talipes equinovarus, absence of the spinal vertebral bodies below the 12th thoracic vertebra, agenesis of sacrum and coccyx, a transversal twelfth rib, bilateral small kidneys, a neurogenic bladder, hypoplastic external genitalia, anal sfincter insufficiency and an atrial septum defect. Pregnancy was complicated by severe diabetes. This spectrum of anomalies is probably due to a generalized alteration in mesodermal cell migration during the primitive streak period. This sequence is strongly associated with maternal diabetes which seems to play a major teratogenic role.
我们报告一名6周大的女孩,有尾部退化序列的表现。出生时,她出现双下肢严重挛缩、双侧马蹄内翻足、第12胸椎以下椎体缺如、骶骨和尾骨发育不全、第十二肋横向生长、双侧小肾脏、神经源性膀胱、外生殖器发育不全、肛门括约肌功能不全和房间隔缺损。妊娠合并严重糖尿病。这种异常谱可能是由于原条期期间中胚层细胞迁移的普遍改变所致。这一序列与母亲糖尿病密切相关,母亲糖尿病似乎起主要致畸作用。