Neurologic improvement in a type 3 Gaucher disease patient treated with imiglucerase/miglustat combination

Epilepsia. 2007 Jul;48(7):1406-8. doi: 10.1111/j.1528-1167.2007.01074.x. Epub 2007 Apr 13.

Abstract

Purpose: Gaucher disease (GD) is an autosomal recessive lysosomal disorder caused by a deficiency of glucocerebrosidase. The neurologic manifestations of GD patients have to date been refractory to any treatment approach. We present a report of a neuronopathic GD patient whose myoclonic epilepsy improved after combination therapy with imiglucerase and miglustat.

Methods: In an adult type 3 GD patient who, despite good visceral and analytic response to ERT, developed progressive neurologic deterioration with marked myoclonic epilepsy and dystonia, we added miglustat to the enzyme-replacement therapy.

Results: After 2 years of combined miglustat (200 mg, 3 t.i.d.) and imiglucerase (60 IU/kg every 2 weeks), generalized tonic-clonic seizures decreased, speech improved, and the general neurologic clinical picture improved markedly. The EEG showed a reduction in focal and generalized paroxysmal discharges. No significant adverse effects were observed.

Conclusions: Combined imiglucerase and miglustat therapy may be beneficial for some neuronopathic forms of GD.

Publication types

  • Case Reports

MeSH terms

  • 1-Deoxynojirimycin / analogs & derivatives*
  • 1-Deoxynojirimycin / therapeutic use
  • Adult
  • Comorbidity
  • Disease Progression
  • Drug Administration Schedule
  • Drug Therapy, Combination
  • Dystonia / drug therapy
  • Dystonia / epidemiology
  • Electroencephalography / drug effects
  • Electroencephalography / statistics & numerical data
  • Enzyme Inhibitors / therapeutic use*
  • Epilepsies, Myoclonic / drug therapy
  • Epilepsies, Myoclonic / epidemiology
  • Gaucher Disease / diagnosis
  • Gaucher Disease / drug therapy*
  • Gaucher Disease / epidemiology
  • Glucosylceramidase / therapeutic use*
  • Humans
  • Male
  • Nervous System Diseases / diagnosis
  • Nervous System Diseases / drug therapy*
  • Nervous System Diseases / epidemiology
  • Recombinant Proteins / therapeutic use
  • Syndrome
  • Treatment Outcome

Substances

  • Enzyme Inhibitors
  • Recombinant Proteins
  • 1-Deoxynojirimycin
  • miglustat
  • Glucosylceramidase
  • imiglucerase