Literature DB >> 28435998

A 6-year-old boy with Wilson disease-A diagnostic dilemma.

Ramaswamy Ganesh1, N Suresh2, T Vasanthi2, Malathi Sathiyasekaran2, R Thulasiraman2.   

Abstract

A 6-year-old boy presented with 2 months history of progressive abdominal distension and jaundice. He was deeply icteric with ascites, hepatosplenomegaly, hyperbilirubinemia, raised transaminases, and coagulopathy. Viral markers and slit lamp examination for Kayser-Fleischer ring were negative. Serum ceruloplasmin and 24-h urinary copper post-D-pencillamine challenge were normal. Anti-smooth muscle antibody was positive 1:20, and liver biopsy showed micronodular cirrhosis with abundant Mallory hyaline and stainable copper deposits. The liver histology was indicative of Indian childhood cirrhosis, whereas the presence of autoantibodies, elevated transaminases, and increased globulin was suggestive of autoimmune hepatitis. Gene studies identified p.R969Q mutation in ATP7B gene, which solved the dilemma and confirmed the diagnosis of Wilson disease (WD). We report a clinicopathological conference of this boy to highlight the challenges faced by pediatricians in the diagnosis of Wilson disease. ᅟ.

Entities:  

Keywords:  Autoimmune hepatitis; Indian childhood cirrhosis; Wilson disease; p.R969Q mutation

Mesh:

Substances:

Year:  2017        PMID: 28435998     DOI: 10.1007/s12664-017-0746-4

Source DB:  PubMed          Journal:  Indian J Gastroenterol        ISSN: 0254-8860


  22 in total

1.  Wilson's disease--a diagnostic dilemma?

Authors:  Ashish Bavdekar
Journal:  Indian J Gastroenterol       Date:  2003 Jan-Feb

2.  Diagnosis and treatment of Wilson disease: an update.

Authors:  Eve A Roberts; Michael L Schilsky
Journal:  Hepatology       Date:  2008-06       Impact factor: 17.425

3.  Wilson's disease in children: 37-year experience and revised King's score for liver transplantation.

Authors:  Anil Dhawan; Rachel M Taylor; Paul Cheeseman; Pamela De Silva; Leah Katsiyiannakis; Giorgina Mieli-Vergani
Journal:  Liver Transpl       Date:  2005-04       Impact factor: 5.799

Review 4.  Wilson's disease.

Authors:  Anand Pandit; Ashish Bavdekar; Sheila Bhave
Journal:  Indian J Pediatr       Date:  2002-09       Impact factor: 1.967

Review 5.  Wilson's disease with superimposed autoimmune features: report of two cases and review.

Authors:  P Milkiewicz; S Saksena; S G Hubscher; E Elias
Journal:  J Gastroenterol Hepatol       Date:  2000-05       Impact factor: 4.029

6.  Screening for Wilson's disease in patients with liver diseases by serum ceruloplasmin.

Authors:  E Cauza; T Maier-Dobersberger; C Polli; K Kaserer; L Kramer; P Ferenci
Journal:  J Hepatol       Date:  1997-08       Impact factor: 25.083

7.  Genotype-phenotype correlations for a wide spectrum of mutations in the Wilson disease gene (ATP7B).

Authors:  Eleni Panagiotakaki; Maria Tzetis; Nina Manolaki; Giorgos Loudianos; Athanasios Papatheodorou; Emmanuel Manesis; Sanda Nousia-Arvanitakis; V Syriopoulou; Emmanuel Kanavakis
Journal:  Am J Med Genet A       Date:  2004-12-01       Impact factor: 2.802

8.  Wilson's disease--early onset and lessons from a pediatric cohort in India.

Authors:  V Kalra; D Khurana; R Mittal
Journal:  Indian Pediatr       Date:  2000-06       Impact factor: 1.411

9.  Atypical copper cirrhosis in Indian children.

Authors:  B Ramakrishna; A Date; C Kirubakaran; P Raghupathy
Journal:  Ann Trop Paediatr       Date:  1995-09

Review 10.  Diagnosis and phenotypic classification of Wilson disease.

Authors:  Peter Ferenci; Karel Caca; Georgios Loudianos; Georgina Mieli-Vergani; Stuart Tanner; Irmin Sternlieb; Michael Schilsky; Diane Cox; Frieder Berr
Journal:  Liver Int       Date:  2003-06       Impact factor: 5.828

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