Clinicopathological review of pallidonigroluysian atrophy
Identifieur interne : 001018 ( Main/Exploration ); précédent : 001017; suivant : 001019Clinicopathological review of pallidonigroluysian atrophy
Auteurs : Janice C. Wong [Canada] ; Melissa J. Armstrong [Canada] ; Anthony E. Lang [Canada] ; Lili-Naz Hazrati [Canada]Source :
- Movement Disorders [ 0885-3185 ] ; 2013-03.
Abstract
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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DOI: 10.1002/mds.25232
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Wong</name><affiliation wicri:level="4"><country xml:lang="fr">Canada</country><wicri:regionArea>Tanz Centre for Research in Neurodegenerative Diseases, University of Toronto, Ontario, Toronto</wicri:regionArea><orgName type="university">Université de Toronto</orgName><placeName><settlement type="city">Toronto</settlement><region type="state">Ontario</region></placeName></affiliation></author><author><name sortKey="Armstrong, Melissa J" sort="Armstrong, Melissa J" uniqKey="Armstrong M" first="Melissa J." last="Armstrong">Melissa J. Armstrong</name><affiliation wicri:level="4"><country xml:lang="fr">Canada</country><wicri:regionArea>Department of Medicine (Neurology), Morton and Gloria Shulman Movement Disorders Clinic and the Edmond J. 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Safra Program in Parkinson's Disease, University of Toronto, Toronto Western Hospital, Ontario, Toronto</wicri:regionArea><orgName type="university">Université de Toronto</orgName><placeName><settlement type="city">Toronto</settlement><region type="state">Ontario</region></placeName></affiliation></author><author><name sortKey="Hazrati, Lili Az" sort="Hazrati, Lili Az" uniqKey="Hazrati L" first="Lili-Naz" last="Hazrati">Lili-Naz Hazrati</name><affiliation wicri:level="4"><country xml:lang="fr">Canada</country><wicri:regionArea>Tanz Centre for Research in Neurodegenerative Diseases, University of Toronto, Ontario, Toronto</wicri:regionArea><orgName type="university">Université de Toronto</orgName><placeName><settlement type="city">Toronto</settlement><region type="state">Ontario</region></placeName></affiliation><affiliation wicri:level="1"><country wicri:rule="url">Canada</country></affiliation></author></analytic><monogr></monogr><series><title level="j">Movement Disorders</title><title level="j" type="abbrev">Mov Disord</title><idno type="ISSN">0885-3185</idno><idno type="eISSN">1531-8257</idno><imprint><publisher>Blackwell Publishing Ltd</publisher><date type="published" when="2013-03">2013-03</date><biblScope unit="volume">28</biblScope><biblScope unit="issue">3</biblScope><biblScope unit="page" from="274">274</biblScope><biblScope unit="page" to="281">281</biblScope></imprint><idno type="ISSN">0885-3185</idno></series><idno type="istex">F6FEA17A8D4A4CD869C437407922AF388C9A3B54</idno><idno type="DOI">10.1002/mds.25232</idno><idno type="ArticleID">MDS25232</idno></biblStruct></sourceDesc><seriesStmt><idno type="ISSN">0885-3185</idno></seriesStmt></fileDesc><profileDesc><textClass></textClass><langUsage><language ident="en">en</language></langUsage></profileDesc></teiHeader><front><div type="abstract">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</div></front></TEI><affiliations><list><country><li>Canada</li></country><region><li>Ontario</li></region><settlement><li>Toronto</li></settlement><orgName><li>Université de Toronto</li></orgName></list><tree><country name="Canada"><region name="Ontario"><name sortKey="Wong, Janice C" sort="Wong, Janice C" uniqKey="Wong J" first="Janice C." last="Wong">Janice C. Wong</name></region><name sortKey="Armstrong, Melissa J" sort="Armstrong, Melissa J" uniqKey="Armstrong M" first="Melissa J." last="Armstrong">Melissa J. Armstrong</name><name sortKey="Hazrati, Lili Az" sort="Hazrati, Lili Az" uniqKey="Hazrati L" first="Lili-Naz" last="Hazrati">Lili-Naz Hazrati</name><name sortKey="Hazrati, Lili Az" sort="Hazrati, Lili Az" uniqKey="Hazrati L" first="Lili-Naz" last="Hazrati">Lili-Naz Hazrati</name><name sortKey="Lang, Anthony E" sort="Lang, Anthony E" uniqKey="Lang A" first="Anthony E." last="Lang">Anthony E. Lang</name></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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