Fatal deterioration of neurological disease after orthotopic liver transplantation for valproic acid-induced liver damage

Pediatr Transplant. 2000 Aug;4(3):211-4. doi: 10.1034/j.1399-3046.2000.00115.x.

Abstract

We describe a 12-year-old girl with an early onset neurologic disease of slow progressiveness and electro-encephalography showing epileptic activity. The girl developed fulminant liver failure 5 months after the start of valproic acid treatment. Repeated mitochondrial assays failed to prove a mitochondrial disorder, but muscle biopsies were slightly pathological. Liver histology indicated acute-on-chronic liver disease. Six weeks after a successful orthotopic liver transplantation her neurological condition deteriorated rapidly, soon leading to generalized cortical disease and death. Post-mortem brain examination showed advanced central nervous destruction. We suggest that this is a late-onset Huttenlocher variant of Alpers' syndrome, where fulminant liver failure can be triggered by valproic acid, and orthotopic liver transplantation can subsequently trigger a fatal neurologic deterioration. Our case illustrates that when a referral center receives a previously unknown patient with hepatocellular insufficiency, it might be impossible to differentiate between fulminant vs. acute-on-chronic liver failure, and the decision whether to perform a liver transplantation or not would become difficult.

Publication types

  • Case Reports

MeSH terms

  • Adolescent
  • Anticonvulsants / adverse effects*
  • Diffuse Cerebral Sclerosis of Schilder / complications*
  • Diffuse Cerebral Sclerosis of Schilder / pathology*
  • Electroencephalography
  • Epilepsy / complications
  • Epilepsy / drug therapy
  • Fatal Outcome
  • Female
  • Humans
  • Liver Failure, Acute / chemically induced*
  • Liver Failure, Acute / surgery*
  • Liver Transplantation*
  • Risk Factors
  • Valproic Acid / adverse effects*

Substances

  • Anticonvulsants
  • Valproic Acid