Hageman A T, Gabreëls F J, Liem K D, Renkawek K, Boon J M
Department of Neurology, University Hospital Nijmegen, The Netherlands.
J Neurol Sci. 1993 Mar;115(1):95-101. doi: 10.1016/0022-510x(93)90072-7.
The congenital variant of myotonic dystrophy (CMD) is a severe disease with a high mortality. CMD is only seen in the offspring of mothers who themselves have myotonic dystrophy (MD). We present 13 patients with clinical symptoms of CMD and neuropathological findings of five of them. The most characteristic symptoms during pregnancy are reduced fetal movements and polyhydramnios. In the neonatal period generalized hypotonia, facial weakness, hyporeflexia, feeding and respiratory difficulties are present. Most of the children have a characteristic tented upper lip. The symptoms greatly diminish after a few weeks. All the children who survive the neonatal period are psychomotor retarded. On pathological examination no specific features were found in muscle tissue or in the brain. The pathogenesis and the cause of the maternal inheritance of CMD is not clear. A review of the literature is provided.
先天性肌强直性营养不良(CMD)是一种严重疾病,死亡率很高。CMD仅见于患有肌强直性营养不良(MD)的母亲的后代。我们报告了13例有CMD临床症状的患者及其5例的神经病理学发现。孕期最具特征性的症状是胎动减少和羊水过多。新生儿期出现全身肌张力减退、面部无力、反射减弱、喂养和呼吸困难。大多数患儿有特征性的上唇呈帐篷状。数周后症状会大为减轻。所有度过新生儿期存活下来的患儿均有精神运动发育迟缓。病理检查在肌肉组织或大脑中未发现特异性特征。CMD母系遗传的发病机制和病因尚不清楚。本文提供了文献综述。