Liver hepatoblastoma and multiple OXPHOS deficiency in the follow-up of a patient with methylmalonic aciduria

Mol Genet Metab. 2008 Sep-Oct;95(1-2):107-9. doi: 10.1016/j.ymgme.2008.06.007. Epub 2008 Aug 3.

Abstract

A boy who was diagnosed with methylmalonic aciduria (MMA) at the age of 10 days developed persistent hepatomegaly and raised transaminases from the age of 4 years. He was subsequently diagnosed with Leigh syndrome and required a kidney transplantation for end-stage renal failure. A massive hepatoblastoma led to his death by the age of 11 years. Methylmalonyl-CoA mutase activity was undetectable on both cultured skin fibroblasts and kidney biopsy and multiple respiratory chain deficiency was demonstrated in the kidney. Mitochondrial dysfunction and/or post-transplant immunosuppressive therapy should be considered as a possible cause of liver cancer in this patient.

Publication types

  • Case Reports
  • Research Support, Non-U.S. Gov't

MeSH terms

  • Cells, Cultured
  • Child
  • Electron Transport
  • Fatal Outcome
  • Fibroblasts / enzymology
  • Follow-Up Studies
  • Hepatoblastoma / enzymology*
  • Hepatoblastoma / etiology
  • Hepatoblastoma / genetics
  • Hepatoblastoma / therapy
  • Humans
  • Immunosuppressive Agents / adverse effects
  • Kidney / enzymology
  • Kidney / metabolism
  • Kidney Transplantation / adverse effects
  • Lipid Metabolism, Inborn Errors / complications*
  • Lipid Metabolism, Inborn Errors / enzymology*
  • Lipid Metabolism, Inborn Errors / genetics
  • Lipid Metabolism, Inborn Errors / therapy
  • Male
  • Methylmalonic Acid / metabolism
  • Methylmalonyl-CoA Mutase / genetics
  • Methylmalonyl-CoA Mutase / metabolism*
  • Mutation

Substances

  • Immunosuppressive Agents
  • Methylmalonic Acid
  • Methylmalonyl-CoA Mutase