Marked amelioration of alcohol-responsive posthypoxic myoclonus by γ-hydroxybutyric acid (Xyrem)

Steven J. Frucht; Yvette Bordelon; William H. Houghton

doi:10.1002/mds.20452

Marked amelioration of alcohol-responsive posthypoxic myoclonus by γ-hydroxybutyric acid (Xyrem)

Steven J. Frucht, Yvette Bordelon, William H. Houghton

Research output: Contribution to journal › Article › peer-review

23 Scopus citations

Abstract

We conducted an open-label, dose-finding, blinded-rating trial of γ-hydroxybutyric acid (Xyrem) in a single patient with severe alcohol-responsive posthypoxic myoclonus refractory to treatment with standard antimyoclonic agents. Xyrem was given in divided doses during the day and was well tolerated. Intensity of myoclonus was measured using the Unified Myoclonus Rating Scale, and blinded videotape review demonstrated complete resolution of myoclonus at rest and stimulus-sensitive myoclonus. Action myoclonus and functional performance also improved in ways that were practically meaningful, allowing her to feed herself, to accomplish daily hygiene tasks, and to walk with assistance. The possible mechanisms of action and potential uses of this agent in other alcohol-responsive movement disorders are discussed.

Original language	English
Pages (from-to)	745-751
Number of pages	7
Journal	Movement Disorders
Volume	20
Issue number	6
DOIs	https://doi.org/10.1002/mds.20452
State	Published - Jun 2005
Externally published	Yes

Keywords

Gamma-hydroxybutyric acid
Hypoxia
Myclonus
Rating scale
Xyrem

Access to Document

10.1002/mds.20452

Cite this

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abstract = "We conducted an open-label, dose-finding, blinded-rating trial of γ-hydroxybutyric acid (Xyrem) in a single patient with severe alcohol-responsive posthypoxic myoclonus refractory to treatment with standard antimyoclonic agents. Xyrem was given in divided doses during the day and was well tolerated. Intensity of myoclonus was measured using the Unified Myoclonus Rating Scale, and blinded videotape review demonstrated complete resolution of myoclonus at rest and stimulus-sensitive myoclonus. Action myoclonus and functional performance also improved in ways that were practically meaningful, allowing her to feed herself, to accomplish daily hygiene tasks, and to walk with assistance. The possible mechanisms of action and potential uses of this agent in other alcohol-responsive movement disorders are discussed.",

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AU - Bordelon, Yvette

AU - Houghton, William H.

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AB - We conducted an open-label, dose-finding, blinded-rating trial of γ-hydroxybutyric acid (Xyrem) in a single patient with severe alcohol-responsive posthypoxic myoclonus refractory to treatment with standard antimyoclonic agents. Xyrem was given in divided doses during the day and was well tolerated. Intensity of myoclonus was measured using the Unified Myoclonus Rating Scale, and blinded videotape review demonstrated complete resolution of myoclonus at rest and stimulus-sensitive myoclonus. Action myoclonus and functional performance also improved in ways that were practically meaningful, allowing her to feed herself, to accomplish daily hygiene tasks, and to walk with assistance. The possible mechanisms of action and potential uses of this agent in other alcohol-responsive movement disorders are discussed.

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KW - Hypoxia

KW - Myclonus

KW - Rating scale

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Marked amelioration of alcohol-responsive posthypoxic myoclonus by γ-hydroxybutyric acid (Xyrem)

Abstract

Keywords

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