Clinicopathological review of pallidonigroluysian atrophy.
Identifieur interne : 000A62 ( Main/Exploration ); précédent : 000A61; suivant : 000A63Clinicopathological review of pallidonigroluysian atrophy.
Auteurs : Janice C. Wong [Canada] ; Melissa J. Armstrong ; Anthony E. Lang ; Lili-Naz HazratiSource :
- Movement disorders : official journal of the Movement Disorder Society [ 1531-8257 ] ; 2013.
English descriptors
- KwdEn :
- Adult, Aged, Atrophy (complications), Databases, Factual (statistics & numerical data), Female, Frontotemporal Dementia (pathology), Globus Pallidus (pathology), Humans, Male, Middle Aged, Substantia Nigra (pathology), Subthalamic Nucleus (pathology), Tauopathies (complications), Tauopathies (pathology).
- MESH :
- complications : Atrophy, Tauopathies.
- pathology : Frontotemporal Dementia, Globus Pallidus, Substantia Nigra, Subthalamic Nucleus, Tauopathies.
- statistics & numerical data : Databases, Factual.
- Adult, Aged, Female, Humans, Male, Middle Aged.
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.
DOI: 10.1002/mds.25232
PubMed: 23114877
Affiliations:
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<record><TEI><teiHeader><fileDesc><titleStmt><title xml:lang="en">Clinicopathological review of pallidonigroluysian atrophy.</title><author><name sortKey="Wong, Janice C" sort="Wong, Janice C" uniqKey="Wong J" first="Janice C" last="Wong">Janice C. Wong</name><affiliation wicri:level="4"><nlm:affiliation>Tanz Centre for Research in Neurodegenerative Diseases, University of Toronto, Toronto, Ontario, Canada.</nlm:affiliation><country xml:lang="fr">Canada</country><wicri:regionArea>Tanz Centre for Research in Neurodegenerative Diseases, University of Toronto, Toronto, Ontario</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></author><author><name sortKey="Lang, Anthony E" sort="Lang, Anthony E" uniqKey="Lang A" first="Anthony E" last="Lang">Anthony E. Lang</name></author><author><name sortKey="Hazrati, Lili Naz" sort="Hazrati, Lili Naz" uniqKey="Hazrati L" first="Lili-Naz" last="Hazrati">Lili-Naz Hazrati</name></author></titleStmt><publicationStmt><idno type="wicri:source">PubMed</idno><date when="2013">2013</date><idno type="doi">10.1002/mds.25232</idno><idno type="RBID">pubmed:23114877</idno><idno type="pmid">23114877</idno><idno type="wicri:Area/PubMed/Corpus">000B99</idno><idno type="wicri:Area/PubMed/Curation">000B99</idno><idno type="wicri:Area/PubMed/Checkpoint">000A72</idno><idno type="wicri:Area/Ncbi/Merge">003863</idno><idno type="wicri:Area/Ncbi/Curation">003863</idno><idno type="wicri:Area/Ncbi/Checkpoint">003863</idno><idno type="wicri:Area/Main/Merge">000A62</idno><idno type="wicri:Area/Main/Curation">000A62</idno><idno type="wicri:Area/Main/Exploration">000A62</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en">Clinicopathological review of pallidonigroluysian atrophy.</title><author><name sortKey="Wong, Janice C" sort="Wong, Janice C" uniqKey="Wong J" first="Janice C" last="Wong">Janice C. Wong</name><affiliation wicri:level="4"><nlm:affiliation>Tanz Centre for Research in Neurodegenerative Diseases, University of Toronto, Toronto, Ontario, Canada.</nlm:affiliation><country xml:lang="fr">Canada</country><wicri:regionArea>Tanz Centre for Research in Neurodegenerative Diseases, University of Toronto, Toronto, Ontario</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></author><author><name sortKey="Lang, Anthony E" sort="Lang, Anthony E" uniqKey="Lang A" first="Anthony E" last="Lang">Anthony E. Lang</name></author><author><name sortKey="Hazrati, Lili Naz" sort="Hazrati, Lili Naz" uniqKey="Hazrati L" first="Lili-Naz" last="Hazrati">Lili-Naz Hazrati</name></author></analytic><series><title level="j">Movement disorders : official journal of the Movement Disorder Society</title><idno type="eISSN">1531-8257</idno><imprint><date when="2013" type="published">2013</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Adult</term><term>Aged</term><term>Atrophy (complications)</term><term>Databases, Factual (statistics & numerical data)</term><term>Female</term><term>Frontotemporal Dementia (pathology)</term><term>Globus Pallidus (pathology)</term><term>Humans</term><term>Male</term><term>Middle Aged</term><term>Substantia Nigra (pathology)</term><term>Subthalamic Nucleus (pathology)</term><term>Tauopathies (complications)</term><term>Tauopathies (pathology)</term></keywords><keywords scheme="MESH" qualifier="complications" xml:lang="en"><term>Atrophy</term><term>Tauopathies</term></keywords><keywords scheme="MESH" qualifier="pathology" xml:lang="en"><term>Frontotemporal Dementia</term><term>Globus Pallidus</term><term>Substantia Nigra</term><term>Subthalamic Nucleus</term><term>Tauopathies</term></keywords><keywords scheme="MESH" qualifier="statistics & numerical data" xml:lang="en"><term>Databases, Factual</term></keywords><keywords scheme="MESH" xml:lang="en"><term>Adult</term><term>Aged</term><term>Female</term><term>Humans</term><term>Male</term><term>Middle Aged</term></keywords></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">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.</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><noCountry><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 Naz" sort="Hazrati, Lili Naz" 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></noCountry><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></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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