Literature DB >> 8529684

Arthrogryposis, renal dysfunction and cholestasis syndrome: report of five patients from three Italian families.

M Di Rocco1, F Callea, B Pollice, M Faraci, F Campiani, C Borrone.   

Abstract

We report on five patients from three families with neurogenic arthrogryposis, cholestasis and tubular renal dysfunction. Despite a similar clinical picture the liver histology showed a broad pathological spectrum, ranging from pigment storage to parenchymal giant cell transformation and ductopenia. The findings are compared with those of other cases from the literature in search of a correct nosology of the syndrome characterized by arthrogryposis, renal and liver disease. CONCLUSION. We propose to consider the picture of arthrogryposis, renal tubular dysfunction and cholestasis as a single syndrome.

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Year:  1995        PMID: 8529684     DOI: 10.1007/bf01959793

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


  7 in total

1.  Arthrogryposis multiplex congenita with renal and hepatic abnormalities in a female infant.

Authors:  J M Saraiva; C Lemos; I Gonçalves; F Carneiro; H C Mota
Journal:  J Pediatr       Date:  1990-11       Impact factor: 4.406

2.  Defective biliary excretion of epinephrine metabolites in mutant (TR-) rats: relation to the pathogenesis of black liver in the Dubin-Johnson syndrome and Corriedale sheep with an analogous excretory defect.

Authors:  T Kitamura; J Alroy; Z Gatmaitan; M Inoue; T Mikami; P Jansen; I M Arias
Journal:  Hepatology       Date:  1992-06       Impact factor: 17.425

3.  Diagnostic value of serum gamma-glutamyl transpeptidase activity in liver diseases in children.

Authors:  G Maggiore; O Bernard; M Hadchouel; A Lemonnier; D Alagille
Journal:  J Pediatr Gastroenterol Nutr       Date:  1991-01       Impact factor: 2.839

Review 4.  Liver histology in the arthrogryposis multiplex congenita, renal dysfunction, and cholestasis (ARC) syndrome: report of three new cases and review.

Authors:  S P Horslen; O W Quarrell; M S Tanner
Journal:  J Med Genet       Date:  1994-01       Impact factor: 6.318

5.  A lethal familial syndrome associating arthrogryposis multiplex congenita, renal dysfunction, and a cholestatic and pigmentary liver disease.

Authors:  C Nezelof; M C Dupart; F Jaubert; E Eliachar
Journal:  J Pediatr       Date:  1979-02       Impact factor: 4.406

6.  Arthrogryposis, cholestatic pigmentary liver disease and renal dysfunction: report of a second family.

Authors:  M Di Rocco; E Reboa; A Barabino; A Larnaout; M Canepa; C Savioli; M Cremonte; C Borrone
Journal:  Am J Med Genet       Date:  1990-10

7.  Renal tubular insufficiency, cholestatic jaundice, and multiple congenital anomalies--a new multisystem syndrome.

Authors:  M A Mikati; A Y Barakat; H B Sulh; V M Der Kaloustian
Journal:  Helv Paediatr Acta       Date:  1984-12
  7 in total
  8 in total

1.  Two novel VPS33B mutations in a patient with arthrogryposis, renal dysfunction and cholestasis syndrome in mainland China.

Authors:  Li-Ting Li; Jing Zhao; Rui Chen; Jian-She Wang
Journal:  World J Gastroenterol       Date:  2014-01-07       Impact factor: 5.742

2.  ARC syndrome with high GGT cholestasis caused by VPS33B mutations.

Authors:  Jian-She Wang; Jing Zhao; Li-Ting Li
Journal:  World J Gastroenterol       Date:  2014-04-28       Impact factor: 5.742

3.  Lethal contractural syndrome type 3 (LCCS3) is caused by a mutation in PIP5K1C, which encodes PIPKI gamma of the phophatidylinsitol pathway.

Authors:  Ginat Narkis; Rivka Ofir; Daniella Landau; Esther Manor; Micha Volokita; Relly Hershkowitz; Khalil Elbedour; Ohad S Birk
Journal:  Am J Hum Genet       Date:  2007-07-24       Impact factor: 11.025

Review 4.  Arc syndrome without arthrogryposis, with hip dislocation and renal glomerulocystic appearance: a case report.

Authors:  Ebru Arhan; Arzu Meltem Yusufoğlu; Tülin Revide Sayli
Journal:  Eur J Pediatr       Date:  2008-10-30       Impact factor: 3.183

5.  The Role of Platelets and ε-Aminocaproic Acid in Arthrogryposis, Renal Dysfunction, and Cholestasis (ARC) Syndrome Associated Hemorrhage.

Authors:  Angela C Weyand; Rebecca M Lombel; Steven W Pipe; Jordan A Shavit
Journal:  Pediatr Blood Cancer       Date:  2015-10-27       Impact factor: 3.167

6.  Clinical and molecular genetic features of ARC syndrome.

Authors:  Paul Gissen; Louise Tee; Colin A Johnson; Emmanuelle Genin; Almuth Caliebe; David Chitayat; Carol Clericuzio; Jonas Denecke; Maja Di Rocco; Björn Fischler; David FitzPatrick; Angeles García-Cazorla; Delphine Guyot; Sebastien Jacquemont; Sibylle Koletzko; Bruno Leheup; Hanna Mandel; Maria Teresa Vieira Sanseverino; Roderick H J Houwen; Patrick J McKiernan; Deirdre A Kelly; Eamonn R Maher
Journal:  Hum Genet       Date:  2006-08-01       Impact factor: 4.132

7.  A Novel VPS33B Variant Identified by Exome Sequencing in a Patient with Arthrogryposis-Renal Dysfunction-Cholestasis Syndrome.

Authors:  Min Ju Lee; Chae Ri Suh; Jeong Hee Shin; Jee Hyun Lee; Yoon Lee; Baik-Lin Eun; Kee Hwan Yoo; Jung Ok Shim
Journal:  Pediatr Gastroenterol Hepatol Nutr       Date:  2019-11-11

8.  Novel gene mutations in three Japanese patients with ARC syndrome associated mild phenotypes: a case series.

Authors:  Yoshinori Satomura; Kazuhiko Bessho; Nobutoshi Nawa; Hidehito Kondo; Shogo Ito; Takao Togawa; Masanao Yano; Yuki Yamano; Taisuke Inoue; Miho Fukui; Shinsuke Onuma; Tomoya Fukuoka; Kie Yasuda; Takeshi Kimura; Makiko Tachibana; Taichi Kitaoka; Shin Nabatame; Keiichi Ozono
Journal:  J Med Case Rep       Date:  2022-02-13
  8 in total

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