Dramatic Response to Pramipexole in Delayed-Onset Parkinsonism from Osmotic Demyelinating Syndrome

Tremor Other Hyperkinet Mov (N Y). 2020 Jun 16:10:9. doi: 10.5334/tohm.66.

Abstract

Background: Delayed parkinsonism and dystonia are recognized phenomena in osmotic demyelinating syndrome (ODS). Dopamine receptor agonists and levodopa have been reported to benefit select patients.

Case report: We report a patient with ODS with severe pseudobulbar deficits, parkinsonism and dystonia, poorly responsive to levodopa, who experienced a remarkable improvement with pramipexole.

Discussion: A marked response to pramipexole with lack of response to levodopa suggests a pre-synaptic source for his deficits coupled with injuries to non-nigral compensatory structures.

Highlights: This case highlights a dramatic response of osmotic demyelination-induced parkinsonism/dystonia to pramipexole. A lack of response to levodopa suggests deficits in the pre-synaptic nigral as well as non-nigral compensatory structures.

Keywords: Parkinsonism; extrapyramidal symptoms; osmotic demyelinating syndrome.

Publication types

  • Case Reports
  • Video-Audio Media

MeSH terms

  • Adult
  • Antiparkinson Agents / therapeutic use*
  • Deamino Arginine Vasopressin / adverse effects
  • Demyelinating Diseases / drug therapy
  • Demyelinating Diseases / etiology
  • Demyelinating Diseases / physiopathology
  • Dystonia / drug therapy*
  • Dystonia / physiopathology
  • Epistaxis / drug therapy
  • Hemostatics / adverse effects
  • Humans
  • Hyponatremia / chemically induced
  • Hyponatremia / therapy*
  • Levodopa / therapeutic use
  • Locked-In Syndrome / physiopathology
  • Male
  • Myelinolysis, Central Pontine / drug therapy*
  • Myelinolysis, Central Pontine / etiology
  • Myelinolysis, Central Pontine / physiopathology
  • Osmotic Pressure
  • Parkinsonian Disorders / drug therapy*
  • Parkinsonian Disorders / physiopathology
  • Postoperative Hemorrhage / drug therapy
  • Pramipexole / therapeutic use*
  • Pseudobulbar Palsy / drug therapy*
  • Pseudobulbar Palsy / physiopathology
  • Rhinoplasty
  • Tetrahydronaphthalenes / therapeutic use
  • Thiophenes / therapeutic use
  • Treatment Failure
  • Treatment Outcome
  • von Willebrand Disease, Type 1 / complications

Substances

  • Antiparkinson Agents
  • Hemostatics
  • Tetrahydronaphthalenes
  • Thiophenes
  • Levodopa
  • Pramipexole
  • rotigotine
  • Deamino Arginine Vasopressin