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.
Copyright: © 2020 The Author(s).
Publication types
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Case Reports
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Video-Audio Media
MeSH terms
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Adult
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Antiparkinson Agents / therapeutic use*
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Deamino Arginine Vasopressin / adverse effects
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Demyelinating Diseases / drug therapy
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Demyelinating Diseases / etiology
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Demyelinating Diseases / physiopathology
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Dystonia / drug therapy*
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Dystonia / physiopathology
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Epistaxis / drug therapy
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Hemostatics / adverse effects
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Humans
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Hyponatremia / chemically induced
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Hyponatremia / therapy*
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Levodopa / therapeutic use
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Locked-In Syndrome / physiopathology
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Male
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Myelinolysis, Central Pontine / drug therapy*
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Myelinolysis, Central Pontine / etiology
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Myelinolysis, Central Pontine / physiopathology
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Osmotic Pressure
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Parkinsonian Disorders / drug therapy*
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Parkinsonian Disorders / physiopathology
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Postoperative Hemorrhage / drug therapy
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Pramipexole / therapeutic use*
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Pseudobulbar Palsy / drug therapy*
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Pseudobulbar Palsy / physiopathology
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Rhinoplasty
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Tetrahydronaphthalenes / therapeutic use
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Thiophenes / therapeutic use
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Treatment Failure
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Treatment Outcome
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von Willebrand Disease, Type 1 / complications
Substances
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Antiparkinson Agents
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Hemostatics
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Tetrahydronaphthalenes
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Thiophenes
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Levodopa
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Pramipexole
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rotigotine
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Deamino Arginine Vasopressin