Literature DB >> 8775219

Paucity of intrahepatic bile ducts, solitary kidney and atrophic pancreas with diabetes mellitus: atypical Alagille syndrome?

K Devriendt1, L Dooms, W Proesmans, F de Zegher, V Desmet, E Eggermont.   

Abstract

UNLABELLED: A child with the tentative diagnosis of Alagille syndrome is reported. Additional renal abnormalities are unilateral kidney agenesis and a kidney with subcortical cysts with decreased function. At the age of 5 years, insulin-dependent diabetes mellitus developed, with the pancreas being atrophic and negative pancreatic islet cell antibodies.
CONCLUSION: This observation extends the picture of Alagille syndrome and suggests an overlap with renal-hepatic-pancreatic dysplasia (Ivemark syndrome).

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Year:  1996        PMID: 8775219     DOI: 10.1007/bf02075756

Source DB:  PubMed          Journal:  Eur J Pediatr        ISSN: 0340-6199            Impact factor:   3.183


  15 in total

Review 1.  Alagille syndrome and deletion of 20p.

Authors:  F Anad; J Burn; D Matthews; I Cross; B C Davison; R Mueller; M Sands; D M Lillington; E Eastham
Journal:  J Med Genet       Date:  1990-12       Impact factor: 6.318

2.  del(20p) with manifestations of arteriohepatic dysplasia.

Authors:  J L Byrne; M J Harrod; J M Friedman; P N Howard-Peebles
Journal:  Am J Med Genet       Date:  1986-08

3.  Segregation analysis of Alagille syndrome.

Authors:  S Dhorne-Pollet; J F Deleuze; M Hadchouel; C Bonaïti-Pellié
Journal:  J Med Genet       Date:  1994-06       Impact factor: 6.318

4.  Renal abnormalities in arteriohepatic dysplasia and nonsyndromic intrahepatic biliary hypoplasia.

Authors:  A E Oestreich; R J Sokol; F J Suchy; J E Heubi
Journal:  Ann Radiol (Paris)       Date:  1983 Feb-Mar

Review 5.  The Alagille syndrome (arteriohepatic dysplasia).

Authors:  R F Mueller
Journal:  J Med Genet       Date:  1987-10       Impact factor: 6.318

6.  Investigation of serum bile acids; seven patients with Alagille syndrome.

Authors:  K Obinata; N Nakatsu; T Watanabe; S Niijima; O Arisaka; H Sasaki; H Nittono; K Yabuta; T Miyano
Journal:  Eur J Pediatr       Date:  1985-09       Impact factor: 3.183

7.  Four generations of arteriohepatic dysplasia.

Authors:  D R LaBrecque; F A Mitros; R J Nathan; K G Romanchuk; G F Judisch; G H El-Khoury
Journal:  Hepatology       Date:  1982 Jul-Aug       Impact factor: 17.425

8.  Syndromic paucity of interlobular bile ducts (Alagille syndrome or arteriohepatic dysplasia): review of 80 cases.

Authors:  D Alagille; A Estrada; M Hadchouel; M Gautier; M Odièvre; J P Dommergues
Journal:  J Pediatr       Date:  1987-02       Impact factor: 4.406

9.  Exocrine pancreatic insufficiency in syndromic paucity of interlobular bile ducts.

Authors:  S K Chong; J Lindridge; C Moniz; A P Mowat
Journal:  J Pediatr Gastroenterol Nutr       Date:  1989-11       Impact factor: 2.839

10.  Renal-hepatic-pancreatic dysplasia and its variants.

Authors:  I W Lurie; I A Kirillova; I V Novikova; I V Burakovski
Journal:  Genet Couns       Date:  1991
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  3 in total

1.  Renal anomalies in Alagille syndrome: a disease-defining feature.

Authors:  Binita M Kamath; Gisele Podkameni; Anne L Hutchinson; Laura D Leonard; Jennifer Gerfen; Ian D Krantz; David A Piccoli; Nancy B Spinner; Kathleen M Loomes; Kevin Meyers
Journal:  Am J Med Genet A       Date:  2011-11-21       Impact factor: 2.802

2.  A classification of ductal plate malformations based on distinct pathogenic mechanisms of biliary dysmorphogenesis.

Authors:  Peggy Raynaud; Joshua Tate; Céline Callens; Sabine Cordi; Patrick Vandersmissen; Rodolphe Carpentier; Christine Sempoux; Olivier Devuyst; Christophe E Pierreux; Pierre Courtoy; Karin Dahan; Katty Delbecque; Sébastien Lepreux; Marco Pontoglio; Lisa M Guay-Woodford; Frédéric P Lemaigre
Journal:  Hepatology       Date:  2011-05-02       Impact factor: 17.425

Review 3.  Renal involvement and the role of Notch signalling in Alagille syndrome.

Authors:  Binita M Kamath; Nancy B Spinner; Norman D Rosenblum
Journal:  Nat Rev Nephrol       Date:  2013-06-11       Impact factor: 28.314

  3 in total

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