Murine Models of Caytaxin Deficiency

Research output: Chapter in Book/Report/Conference proceedingChapter

Abstract

Although classically ascribed to dysfunction of the basal ganglia, work with the dystonic (dt) rat has shown that dystonia can arise from abnormal cerebellar output. The dt rat manifests generalized dystonia caused by deficiency of the neuronally restricted protein, caytaxin. Electrophysiological and biochemical studies provided evidence that defects at the climbing fiber-Purkinje cell synapse in the dt rat lead to abnormal bursting firing patterns in the cerebellar nuclei, which increase linearly with postnatal age. Mouse models of caytaxin deficiency (jittery, sidewinder, and hesitant) show variable degrees of dystonia and ataxia.

Original languageEnglish (US)
Title of host publicationMovement Disorders
Subtitle of host publicationGenetics and Models: Second Edition
PublisherElsevier Inc.
Pages439-452
Number of pages14
ISBN (Print)9780124051959
DOIs
StatePublished - Jan 1 2015

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Dystonia
Cerebellar Nuclei
Purkinje Cells
Ataxia
Basal Ganglia
Synapses
Proteins

All Science Journal Classification (ASJC) codes

  • Medicine(all)

Cite this

Ledoux, M. (2015). Murine Models of Caytaxin Deficiency. In Movement Disorders: Genetics and Models: Second Edition (pp. 439-452). Elsevier Inc.. https://doi.org/10.1016/B978-0-12-405195-9.00025-1

Murine Models of Caytaxin Deficiency. / Ledoux, Mark.

Movement Disorders: Genetics and Models: Second Edition. Elsevier Inc., 2015. p. 439-452.

Research output: Chapter in Book/Report/Conference proceedingChapter

Ledoux, M 2015, Murine Models of Caytaxin Deficiency. in Movement Disorders: Genetics and Models: Second Edition. Elsevier Inc., pp. 439-452. https://doi.org/10.1016/B978-0-12-405195-9.00025-1
Ledoux M. Murine Models of Caytaxin Deficiency. In Movement Disorders: Genetics and Models: Second Edition. Elsevier Inc. 2015. p. 439-452 https://doi.org/10.1016/B978-0-12-405195-9.00025-1
Ledoux, Mark. / Murine Models of Caytaxin Deficiency. Movement Disorders: Genetics and Models: Second Edition. Elsevier Inc., 2015. pp. 439-452
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