Ortmann M, Querfeld U, Stollorz M, Schröder R
Institut für Pathologie der Universität zu Köln.
Pathologe. 1995 Mar;16(2):143-7. doi: 10.1007/s002920050087.
Renal tubular dysgenesis is a rarely recognized condition characterized by oligohydramnios, Potter's sequence and congenital anuria leading to stillbirth or neonatal death from respiratory failure. It is thought to be inherited in an autosomal recessive manner. Definitive diagnosis is based on renal histology, revealing the lack of proximal tubule differentiation. Two additional cases of affected sibs in a family with parental consanguinity are reported. Lectin and immunohistochemical studies confirmed structural and functional immaturity of the proximal tubule. Further findings include bilateral renal vein thrombosis. The clinical and morphological parameters defining this disorder and the possible mechanisms of pathogenesis are discussed.
肾小管发育不全是一种罕见的疾病,其特征为羊水过少、波特序列征以及先天性无尿,可导致死产或因呼吸衰竭而新生儿死亡。该病被认为是以常染色体隐性方式遗传。确诊基于肾脏组织学检查,显示近端肾小管缺乏分化。本文报告了一个有近亲婚配的家庭中另外两例患病同胞的情况。凝集素和免疫组化研究证实了近端肾小管在结构和功能上的不成熟。进一步的发现包括双侧肾静脉血栓形成。本文讨论了定义该疾病的临床和形态学参数以及可能的发病机制。