Torra R, Alós L, Ramos J, Estivill X
Centre for Medical Genetics, Hospital Clinic, Barcelona, Catalunya, Spain.
J Med Genet. 1996 May;33(5):409-12. doi: 10.1136/jmg.33.5.409.
We report two brothers with a cystic malformation of the kidneys, liver, and pancreas. In both cases the malformation was fatal and the children died shortly after birth. The pathological findings, consisting of multicystic dysplastic kidneys, dilated and dysgenetic bile ducts, dilated pancreatic ducts, and polysplenia, correspond to those reported by Ivemark as renal-hepatic-pancreatic dysplasia. Many polymalformation syndromes include cystic affectation of these three organs, so this syndrome could be an isolated entity or a final common pathway of response of these organs to a variety of developmental disturbances, which could also include splenic abnormalities. We propose an autosomal recessive pattern of inheritance for renal-hepatic-pancreatic dysplasia.
我们报告了两例患有肾、肝和胰腺囊性畸形的兄弟。在这两个病例中,畸形都是致命的,孩子出生后不久就死亡了。病理检查结果包括多囊性发育不良肾、扩张和发育异常的胆管、扩张的胰管以及多脾,与Ivemark报道的肾-肝-胰发育异常相符。许多多畸形综合征都包括这三个器官的囊性病变,因此这种综合征可能是一个独立的实体,或者是这些器官对各种发育障碍反应的最终共同途径,其中也可能包括脾脏异常。我们提出肾-肝-胰发育异常为常染色体隐性遗传模式。