Trientine-induced neurological deterioration in a patient with Wilson's disease

J Clin Neurosci. 2013 Apr;20(4):606-8. doi: 10.1016/j.jocn.2012.02.041. Epub 2012 Dec 27.

Abstract

Trientine (triethylenetetramine dihydrochloride) is a copper-chelating agent used to treat patients with Wilson's disease (WD). It has been considered safe, rarely causing neurological deterioration during initial treatment. We describe a patient diagnosed with WD who became neurologically disabled after treatment with trientine. On a fluid attenuated inversion recovery sequence, brain MRI showed increased areas of high signal intensity compared with initial brain MRI. The patient's neurological signs partially resolved after cessation of trientine treatment. Our findings suggest that treatment with trientine is associated with a risk of neurological deterioration in patients with WD.

Publication types

  • Case Reports

MeSH terms

  • Brain / pathology
  • Chelating Agents / adverse effects*
  • Chelating Agents / therapeutic use
  • Copper / metabolism
  • Dystonia / etiology
  • Gait Disorders, Neurologic / etiology
  • Gait Disorders, Neurologic / pathology
  • Hepatolenticular Degeneration / complications*
  • Hepatolenticular Degeneration / drug therapy
  • Hepatolenticular Degeneration / pathology
  • Humans
  • Magnetic Resonance Imaging
  • Male
  • Nervous System Diseases / chemically induced*
  • Nervous System Diseases / complications
  • Nervous System Diseases / pathology
  • Trientine / adverse effects*
  • Trientine / therapeutic use
  • Young Adult
  • Zinc / therapeutic use

Substances

  • Chelating Agents
  • Copper
  • Zinc
  • Trientine