Literature DB >> 15884633

Clinical and pathological aspects of ARC (arthrogryposis, renal dysfunction and cholestasis) syndrome in two siblings.

Neslihan Tekin1, Sultan Durmuş-Aydoğdu, Ener Cağri Dinleyici, Ozcan Bör, Kismet Bildirici, Arif Akşit.   

Abstract

We describe the first family report of ARC syndrome (arthrogryposis multiplex congenita, renal dysfunction, and cholestasis) diagnosed in Turkey. ARC syndrome is a rare cause of cholestatic jaundice and skeletal abnormalities in the neonatal period. Fanconi-like renal tubular dysfunction completed the clinical picture. Consanguinity and affected membership are the other typical components of this rare disorder, and possibility of autosomal recessive transmission was considered. A broad spectrum of histopathological abnormalities have been described in the liver and kidney. In this report, we describe two male siblings with ARC syndrome who had cholestatic jaundice, arthrogryposis multiplex congenital-like joint contractures and renal involvement with additional clinical features. Clinical and pathological aspects of the syndrome are discussed and compared with the other cases in the literature.

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Year:  2005        PMID: 15884633

Source DB:  PubMed          Journal:  Turk J Pediatr        ISSN: 0041-4301            Impact factor:   0.552


  5 in total

1.  Metazoan cell biology of the HOPS tethering complex.

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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

Review 3.  Arc syndrome without arthrogryposis, with hip dislocation and renal glomerulocystic appearance: a case report.

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Journal:  Eur J Pediatr       Date:  2008-10-30       Impact factor: 3.183

4.  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

Review 5.  Arthrogryposis-renal dysfunction-cholestasis (ARC) syndrome: from molecular genetics to clinical features.

Authors:  Yaoyao Zhou; Junfeng Zhang
Journal:  Ital J Pediatr       Date:  2014-09-20       Impact factor: 2.638

  5 in total

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