Wilson disease: histopathological correlations with treatment on follow-up liver biopsies

World J Gastroenterol. 2010 Mar 28;16(12):1487-94. doi: 10.3748/wjg.v16.i12.1487.

Abstract

Aim: To investigate the progression of hepatic histopathology in serial liver biopsies from Wilson disease (WD) patients.

Methods: We report a group of 12 WD patients treated with zinc and/or penicillamine who underwent multiple follow-up liver biopsies. Demographic, clinical and laboratory data were gathered and all patients underwent an initial biopsy and at least one repeat biopsy.

Results: Time to repeat biopsy ranged from 2 to 12 years. Six patients (non-progressors) showed stable hepatic histology or improvement. In one case, we observed improvement of fibrosis from stage 2 to 0. Six patients (progressors) had worsening of fibrosis. There was no significant correlation between the histological findings and serum aminotransferases or copper metabolism parameters. The hepatic copper concentration reached normal levels in only two patients: one from the non-progressors and one from the progressors group. The estimated rate of progression of hepatic fibrosis in the entire group was 0 units per year in the time frame between the first and the second liver biopsy (4 years), and 0.25 between the second and the third (3 years). In the progressors group, the rate of progression of liver fibrosis was estimated at 0.11 fibrosis units per year between the first and second biopsy and, 0.6 fibrosis units between the second and third biopsy.

Conclusion: The inability of clinical tools to detect fibrosis progression in WD suggests that a liver biopsy with hepatic copper quantification every 3 years should be considered.

MeSH terms

  • Adolescent
  • Adult
  • Alanine Transaminase / blood
  • Aspartate Aminotransferases / blood
  • Biomarkers / blood
  • Biopsy
  • Chelating Agents / therapeutic use*
  • Child
  • Copper / metabolism*
  • Copper / urine
  • Female
  • Hepatolenticular Degeneration / complications
  • Hepatolenticular Degeneration / drug therapy*
  • Hepatolenticular Degeneration / metabolism
  • Hepatolenticular Degeneration / pathology
  • Humans
  • Liver / drug effects*
  • Liver / metabolism
  • Liver / pathology
  • Liver Cirrhosis / drug therapy*
  • Liver Cirrhosis / etiology
  • Liver Cirrhosis / metabolism
  • Liver Cirrhosis / pathology
  • Male
  • Penicillamine / therapeutic use*
  • Time Factors
  • Treatment Outcome
  • Young Adult
  • Zinc Sulfate / therapeutic use*

Substances

  • Biomarkers
  • Chelating Agents
  • Zinc Sulfate
  • Copper
  • Aspartate Aminotransferases
  • Alanine Transaminase
  • Penicillamine