Literature DB >> 8733053

Renal-hepatic-pancreatic dysplasia: an autosomal recessive malformation.

R Torra1, L Alós, J Ramos, X Estivill.   

Abstract

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.

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Year:  1996        PMID: 8733053      PMCID: PMC1050612          DOI: 10.1136/jmg.33.5.409

Source DB:  PubMed          Journal:  J Med Genet        ISSN: 0022-2593            Impact factor:   6.318


  27 in total

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Authors:  M D Crawfurd
Journal:  Clin Genet       Date:  1978-12       Impact factor: 4.438

7.  Dysplasia of the kidneys, liver, and pancreas: report of a variant of Ivemark's syndrome.

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Journal:  Hum Pathol       Date:  1979-03       Impact factor: 3.466

8.  Genetics of the Meckel syndrome (dysencephalia splanchnocystica).

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Journal:  Pediatrics       Date:  1971-08       Impact factor: 7.124

9.  A highly polymorphic DNA marker linked to adult polycystic kidney disease on chromosome 16.

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Journal:  Nature       Date:  1985 Oct 10-16       Impact factor: 49.962

10.  Cystic dysplastic kidneys associated with Dandy-Walker malformation and congenital hepatic fibrosis. Report of two cases.

Authors:  M Kudo; K Tamura; Y Fuse
Journal:  Am J Clin Pathol       Date:  1985-10       Impact factor: 2.493

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  4 in total

1.  Failure to Thrive, Jaundice, and Polyuria in Early Infancy: Common Presentation with an Uncommon Lethal Etiology.

Authors:  Sonia Sharma
Journal:  J Pediatr Genet       Date:  2019-11-11

Review 2.  Liver and kidney disease in ciliopathies.

Authors:  Meral Gunay-Aygun
Journal:  Am J Med Genet C Semin Med Genet       Date:  2009-11-15       Impact factor: 3.908

Review 3.  Renal-hepatic-pancreatic dysplasia-1 with a novel NPHP3 genotype: a case report and review of the literature.

Authors:  Hui Zhu; Zhi-Hui Zhao; Shu-Yao Zhu; Fu Xiong; Li-Hong He; Yong Zhang; Jin Wang
Journal:  BMC Pediatr       Date:  2022-10-18       Impact factor: 2.567

4.  Immunohistochemical study of the phenotypic change of the mesenchymal cells during portal tract maturation in normal and fibrous (ductal plate malformation) fetal liver.

Authors:  Julien Villeneuve; Fanny Pelluard-Nehme; Chantal Combe; Dominique Carles; Christine Chaponnier; Jean Ripoche; Charles Balabaud; Paulette Bioulac-Sage; Sébastien Lepreux
Journal:  Comp Hepatol       Date:  2009-07-14
  4 in total

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