Milunsky J M, Genest D R, Milunsky A
Center for Human Genetics, Boston University School of Medicine, MA 02118, USA.
Pediatr Nephrol. 1997 Aug;11(4):494-6. doi: 10.1007/s004670050325.
Renal tubular dysgenesis, a rare, lethal, autosomal recessive disorder, is characterized by short and poorly differentiated proximal convoluted tubules associated with oligohydramnios, Potter sequence, and neonatal death due to respiratory failure. Abnormalities of the skull may occur in some cases. We report an infant born of a consanguineous union who also had microcephaly, among other features. A history of oligohydramnios with or without skull abnormalities and a lethal outcome without obvious explanation should occasion renal histological study aimed at precise diagnosis and genetic counselling.
肾小管发育不全是一种罕见的致死性常染色体隐性疾病,其特征为近端曲管短且分化不良,伴有羊水过少、波特序列征以及因呼吸衰竭导致的新生儿死亡。部分病例可能出现颅骨异常。我们报告了一例近亲结婚所生的婴儿,该婴儿除其他特征外还患有小头畸形。有羊水过少病史,无论有无颅骨异常,且出现不明原因的致死结局,均应进行肾脏组织学检查,以明确诊断并提供遗传咨询。