| Clinicopathological review of pallidonigroluysian atrophy. | |
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MedLine Citation:
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PMID: 23114877 Owner: NLM Status: Publisher |
Abstract/OtherAbstract:
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Pallidonigroluysian atrophy is a rare neurodegenerative disease characterized by degeneration of the globus pallidus, substantia nigra, and subthalamic nucleus. Few studies have comprehensively documented the clinical and pathological features of pallidonigroluysian atrophy. A systematic review of all published cases of pallidonigroluysian atrophy in English since 1970 was performed. We also report a new case of pallidonigroluysian atrophy. Twenty-five cases of pathologically proven pallidonigroluysian atrophy were reviewed, 24 from the literature and 1 of our own. Average age of onset was 54.3 ± 14.3 years, and average duration of disease was 7.9 ± 5.8 years. The most common first symptom was gait or balance disturbance. Patients had a diversity of movement disorders, including chorea in 5 cases (20%). Nine cases (36%) had coexistent motor neuron disease. Almost all cases had gliosis, and many cases had iron-positive pigments in the pallidonigroluysian system. Tauopathy was absent to rare in this region. Widespread tau-negative, p62-positive glial inclusions, described in 1 previous case, were also present in our patient. As pallidonigroluysian atrophy has a diversity of clinical presentations, it is best defined neuropathologically. The relative lack of tauopathy and the presence of p62-positive glial inclusions or iron-positive pigments in the pallidonigroluysian region may help to distinguish pallidonigroluysian atrophy from similar disease entities. © 2012 Movement Disorder Society. |
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Authors:
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Janice C Wong; Melissa J Armstrong; Anthony E Lang; Lili-Naz Hazrati |
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Publication Detail:
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Type: JOURNAL ARTICLE Date: 2012-10-31 |
Journal Detail:
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Title: Movement disorders : official journal of the Movement Disorder Society Volume: - ISSN: 1531-8257 ISO Abbreviation: Mov. Disord. Publication Date: 2012 Oct |
Date Detail:
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Created Date: 2012-11-1 Completed Date: - Revised Date: - |
Medline Journal Info:
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Nlm Unique ID: 8610688 Medline TA: Mov Disord Country: - |
Other Details:
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Languages: ENG Pagination: - Citation Subset: - |
Copyright Information:
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Copyright © 2012 Movement Disorder Society. |
Affiliation:
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Tanz Centre for Research in Neurodegenerative Diseases, University of Toronto, Toronto, Ontario, Canada. |
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From MEDLINE®/PubMed®, a database of the U.S. National Library of Medicine
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