Neuropathological changes in essential tremor: 33 cases compared with 21 controls
Identifieur interne : 002508 ( Main/Exploration ); précédent : 002507; suivant : 002509Neuropathological changes in essential tremor: 33 cases compared with 21 controls
Auteurs : Elan D. Louis [États-Unis] ; Phyllis L. Faust [États-Unis] ; Jean-Paul G. Vonsattel [États-Unis] ; Lawrence S. Honig [États-Unis] ; Alex Rajput [États-Unis] ; Christopher A. Robinson [États-Unis] ; Ali Rajput [États-Unis] ; Rajesh Pahwa [États-Unis] ; Kelly E. Lyons [États-Unis] ; G. Webster Ross [États-Unis] ; Sarah Borden [États-Unis] ; Carol B. Moskowitz [États-Unis] ; Arlene Lawton [États-Unis] ; Nora Hernandez [États-Unis]Source :
- Brain [ 0006-8950 ] ; 2007-12.
Abstract
Despite its being one of the most commonly observed neurological disorders, neuropathological studies of essential tremor (ET) are rare. There have been surprisingly few autopsy studies and even fewer case-control comparisons. The primary objective was to describe and quantify the pathological changes in 33 ET and 21 control brains. A secondary objective was to correlate clinical and pathological features. We examined autopsy tissue from the Essential Tremor Centralized Brain Repository. Eight (24.2%) of the 33 ET brains had Lewy bodies in the brainstem, mainly in the locus ceruleus. However, the majority of ET brains (25/33, 75.8%) had no Lewy bodies, but had pathological changes in the cerebellum. The mean number of Purkinje cells per 100× field was reduced in ET cases without Lewy bodies (6.6 ± 2.4 versus 9.6 ± 3.4, P < 0.01), and there were ∼7× more Purkinje cell torpedoes per section (12.6 ± 7.9 versus 1.7 ± 1.4, P < 0.001) compared to controls. ET cases without Lewy bodies also had degeneration of the dentate nucleus (two cases). Other findings in ET cases were Purkinje cell heterotopias and dendrite swellings. Lewy body ET cases were older than ET cases without Lewy bodies. Several trends were observed in ET cases without Lewy bodies, including a younger age of onset of tremor and higher proportions with gait difficulty and family history of ET. The pathological changes of ET seem to be heterogeneous and degenerative. The majority have cerebellar changes without Lewy bodies; a smaller proportion has brainstem Lewy bodies. The clinical differences between cases with versus without Lewy bodies require additional study.
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DOI: 10.1093/brain/awm266
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Louis</name><affiliation wicri:level="4"><country xml:lang="fr">États-Unis</country><wicri:regionArea>GH Sergievsky Center, Department of Neurology, Taub Institute for Research on Alzheimer's Disease and the Aging Brain, Department of Pathology, College of Physicians and Surgeons, Columbia University, New York, Department of Epidemiology, Mailman School of Public Health, Columbia University, New York, NY, USA, Royal University Hospital, Saskatoon, Saskatchewan, Canada, Department of Neurology, University of Kansas Medical Center, Kansas City, KS, USA, Veterans Affairs Pacific Islands Health Care System, Departments of Medicine and Geriatrics, University of Hawaii John A. Burns School of Medicine, Pacific Health Research Institute and Kuakini Medical Center/Honolulu-Asia Aging Study, Honolulu, HI</wicri:regionArea><placeName><region type="state">Hawaï</region><settlement type="city">New York</settlement></placeName><orgName type="university">Université Columbia</orgName></affiliation><affiliation wicri:level="1"><country wicri:rule="url">États-Unis</country></affiliation></author><author><name sortKey="Faust, Phyllis L" sort="Faust, Phyllis L" uniqKey="Faust P" first="Phyllis L." last="Faust">Phyllis L. Faust</name><affiliation wicri:level="4"><country xml:lang="fr">États-Unis</country><wicri:regionArea>GH Sergievsky Center, Department of Neurology, Taub Institute for Research on Alzheimer's Disease and the Aging Brain, Department of Pathology, College of Physicians and Surgeons, Columbia University, New York, Department of Epidemiology, Mailman School of Public Health, Columbia University, New York, NY, USA, Royal University Hospital, Saskatoon, Saskatchewan, Canada, Department of Neurology, University of Kansas Medical Center, Kansas City, KS, USA, Veterans Affairs Pacific Islands Health Care System, Departments of Medicine and Geriatrics, University of Hawaii John A. Burns School of Medicine, Pacific Health Research Institute and Kuakini Medical Center/Honolulu-Asia Aging Study, Honolulu, HI</wicri:regionArea><placeName><region type="state">Hawaï</region><settlement type="city">New York</settlement></placeName><orgName type="university">Université Columbia</orgName></affiliation></author><author><name sortKey="Vonsattel, Jean Paul G" sort="Vonsattel, Jean Paul G" uniqKey="Vonsattel J" first="Jean-Paul G." last="Vonsattel">Jean-Paul G. Vonsattel</name><affiliation wicri:level="4"><country xml:lang="fr">États-Unis</country><wicri:regionArea>GH Sergievsky Center, Department of Neurology, Taub Institute for Research on Alzheimer's Disease and the Aging Brain, Department of Pathology, College of Physicians and Surgeons, Columbia University, New York, Department of Epidemiology, Mailman School of Public Health, Columbia University, New York, NY, USA, Royal University Hospital, Saskatoon, Saskatchewan, Canada, Department of Neurology, University of Kansas Medical Center, Kansas City, KS, USA, Veterans Affairs Pacific Islands Health Care System, Departments of Medicine and Geriatrics, University of Hawaii John A. Burns School of Medicine, Pacific Health Research Institute and Kuakini Medical Center/Honolulu-Asia Aging Study, Honolulu, HI</wicri:regionArea><placeName><region type="state">Hawaï</region><settlement type="city">New York</settlement></placeName><orgName type="university">Université Columbia</orgName></affiliation></author><author><name sortKey="Honig, Lawrence S" sort="Honig, Lawrence S" uniqKey="Honig L" first="Lawrence S." last="Honig">Lawrence S. Honig</name><affiliation wicri:level="4"><country xml:lang="fr">États-Unis</country><wicri:regionArea>GH Sergievsky Center, Department of Neurology, Taub Institute for Research on Alzheimer's Disease and the Aging Brain, Department of Pathology, College of Physicians and Surgeons, Columbia University, New York, Department of Epidemiology, Mailman School of Public Health, Columbia University, New York, NY, USA, Royal University Hospital, Saskatoon, Saskatchewan, Canada, Department of Neurology, University of Kansas Medical Center, Kansas City, KS, USA, Veterans Affairs Pacific Islands Health Care System, Departments of Medicine and Geriatrics, University of Hawaii John A. Burns School of Medicine, Pacific Health Research Institute and Kuakini Medical Center/Honolulu-Asia Aging Study, Honolulu, HI</wicri:regionArea><placeName><region type="state">Hawaï</region><settlement type="city">New York</settlement></placeName><orgName type="university">Université Columbia</orgName></affiliation></author><author><name sortKey="Rajput, Alex" sort="Rajput, Alex" uniqKey="Rajput A" first="Alex" last="Rajput">Alex Rajput</name><affiliation wicri:level="4"><country xml:lang="fr">États-Unis</country><wicri:regionArea>GH Sergievsky Center, Department of Neurology, Taub Institute for Research on Alzheimer's Disease and the Aging Brain, Department of Pathology, College of Physicians and Surgeons, Columbia University, New York, Department of Epidemiology, Mailman School of Public Health, Columbia University, New York, NY, USA, Royal University Hospital, Saskatoon, Saskatchewan, Canada, Department of Neurology, University of Kansas Medical Center, Kansas City, KS, USA, Veterans Affairs Pacific Islands Health Care System, Departments of Medicine and Geriatrics, University of Hawaii John A. Burns School of Medicine, Pacific Health Research Institute and Kuakini Medical Center/Honolulu-Asia Aging Study, Honolulu, HI</wicri:regionArea><placeName><region type="state">Hawaï</region><settlement type="city">New York</settlement></placeName><orgName type="university">Université Columbia</orgName></affiliation></author><author><name sortKey="Robinson, Christopher A" sort="Robinson, Christopher A" uniqKey="Robinson C" first="Christopher A." last="Robinson">Christopher A. Robinson</name><affiliation wicri:level="4"><country xml:lang="fr">États-Unis</country><wicri:regionArea>GH Sergievsky Center, Department of Neurology, Taub Institute for Research on Alzheimer's Disease and the Aging Brain, Department of Pathology, College of Physicians and Surgeons, Columbia University, New York, Department of Epidemiology, Mailman School of Public Health, Columbia University, New York, NY, USA, Royal University Hospital, Saskatoon, Saskatchewan, Canada, Department of Neurology, University of Kansas Medical Center, Kansas City, KS, USA, Veterans Affairs Pacific Islands Health Care System, Departments of Medicine and Geriatrics, University of Hawaii John A. Burns School of Medicine, Pacific Health Research Institute and Kuakini Medical Center/Honolulu-Asia Aging Study, Honolulu, HI</wicri:regionArea><placeName><region type="state">Hawaï</region><settlement type="city">New York</settlement></placeName><orgName type="university">Université Columbia</orgName></affiliation></author><author><name sortKey="Rajput, Ali" sort="Rajput, Ali" uniqKey="Rajput A" first="Ali" last="Rajput">Ali Rajput</name><affiliation wicri:level="4"><country xml:lang="fr">États-Unis</country><wicri:regionArea>GH Sergievsky Center, Department of Neurology, Taub Institute for Research on Alzheimer's Disease and the Aging Brain, Department of Pathology, College of Physicians and Surgeons, Columbia University, New York, Department of Epidemiology, Mailman School of Public Health, Columbia University, New York, NY, USA, Royal University Hospital, Saskatoon, Saskatchewan, Canada, Department of Neurology, University of Kansas Medical Center, Kansas City, KS, USA, Veterans Affairs Pacific Islands Health Care System, Departments of Medicine and Geriatrics, University of Hawaii John A. Burns School of Medicine, Pacific Health Research Institute and Kuakini Medical Center/Honolulu-Asia Aging Study, Honolulu, HI</wicri:regionArea><placeName><region type="state">Hawaï</region><settlement type="city">New York</settlement></placeName><orgName type="university">Université Columbia</orgName></affiliation></author><author><name sortKey="Pahwa, Rajesh" sort="Pahwa, Rajesh" uniqKey="Pahwa R" first="Rajesh" last="Pahwa">Rajesh Pahwa</name><affiliation wicri:level="4"><country xml:lang="fr">États-Unis</country><wicri:regionArea>GH Sergievsky Center, Department of Neurology, Taub Institute for Research on Alzheimer's Disease and the Aging Brain, Department of Pathology, College of Physicians and Surgeons, Columbia University, New York, Department of Epidemiology, Mailman School of Public Health, Columbia University, New York, NY, USA, Royal University Hospital, Saskatoon, Saskatchewan, Canada, Department of Neurology, University of Kansas Medical Center, Kansas City, KS, USA, Veterans Affairs Pacific Islands Health Care System, Departments of Medicine and Geriatrics, University of Hawaii John A. Burns School of Medicine, Pacific Health Research Institute and Kuakini Medical Center/Honolulu-Asia Aging Study, Honolulu, HI</wicri:regionArea><placeName><region type="state">Hawaï</region><settlement type="city">New York</settlement></placeName><orgName type="university">Université Columbia</orgName></affiliation></author><author><name sortKey="Lyons, Kelly E" sort="Lyons, Kelly E" uniqKey="Lyons K" first="Kelly E." last="Lyons">Kelly E. Lyons</name><affiliation wicri:level="4"><country xml:lang="fr">États-Unis</country><wicri:regionArea>GH Sergievsky Center, Department of Neurology, Taub Institute for Research on Alzheimer's Disease and the Aging Brain, Department of Pathology, College of Physicians and Surgeons, Columbia University, New York, Department of Epidemiology, Mailman School of Public Health, Columbia University, New York, NY, USA, Royal University Hospital, Saskatoon, Saskatchewan, Canada, Department of Neurology, University of Kansas Medical Center, Kansas City, KS, USA, Veterans Affairs Pacific Islands Health Care System, Departments of Medicine and Geriatrics, University of Hawaii John A. Burns School of Medicine, Pacific Health Research Institute and Kuakini Medical Center/Honolulu-Asia Aging Study, Honolulu, HI</wicri:regionArea><placeName><region type="state">Hawaï</region><settlement type="city">New York</settlement></placeName><orgName type="university">Université Columbia</orgName></affiliation></author><author><name sortKey="Ross, G Webster" sort="Ross, G Webster" uniqKey="Ross G" first="G. Webster" last="Ross">G. Webster Ross</name><affiliation wicri:level="4"><country xml:lang="fr">États-Unis</country><wicri:regionArea>GH Sergievsky Center, Department of Neurology, Taub Institute for Research on Alzheimer's Disease and the Aging Brain, Department of Pathology, College of Physicians and Surgeons, Columbia University, New York, Department of Epidemiology, Mailman School of Public Health, Columbia University, New York, NY, USA, Royal University Hospital, Saskatoon, Saskatchewan, Canada, Department of Neurology, University of Kansas Medical Center, Kansas City, KS, USA, Veterans Affairs Pacific Islands Health Care System, Departments of Medicine and Geriatrics, University of Hawaii John A. Burns School of Medicine, Pacific Health Research Institute and Kuakini Medical Center/Honolulu-Asia Aging Study, Honolulu, HI</wicri:regionArea><placeName><region type="state">Hawaï</region><settlement type="city">New York</settlement></placeName><orgName type="university">Université Columbia</orgName></affiliation></author><author><name sortKey="Borden, Sarah" sort="Borden, Sarah" uniqKey="Borden S" first="Sarah" last="Borden">Sarah Borden</name><affiliation wicri:level="4"><country xml:lang="fr">États-Unis</country><wicri:regionArea>GH Sergievsky Center, Department of Neurology, Taub Institute for Research on Alzheimer's Disease and the Aging Brain, Department of Pathology, College of Physicians and Surgeons, Columbia University, New York, Department of Epidemiology, Mailman School of Public Health, Columbia University, New York, NY, USA, Royal University Hospital, Saskatoon, Saskatchewan, Canada, Department of Neurology, University of Kansas Medical Center, Kansas City, KS, USA, Veterans Affairs Pacific Islands Health Care System, Departments of Medicine and Geriatrics, University of Hawaii John A. Burns School of Medicine, Pacific Health Research Institute and Kuakini Medical Center/Honolulu-Asia Aging Study, Honolulu, HI</wicri:regionArea><placeName><region type="state">Hawaï</region><settlement type="city">New York</settlement></placeName><orgName type="university">Université Columbia</orgName></affiliation></author><author><name sortKey="Moskowitz, Carol B" sort="Moskowitz, Carol B" uniqKey="Moskowitz C" first="Carol B." last="Moskowitz">Carol B. Moskowitz</name><affiliation wicri:level="4"><country xml:lang="fr">États-Unis</country><wicri:regionArea>GH Sergievsky Center, Department of Neurology, Taub Institute for Research on Alzheimer's Disease and the Aging Brain, Department of Pathology, College of Physicians and Surgeons, Columbia University, New York, Department of Epidemiology, Mailman School of Public Health, Columbia University, New York, NY, USA, Royal University Hospital, Saskatoon, Saskatchewan, Canada, Department of Neurology, University of Kansas Medical Center, Kansas City, KS, USA, Veterans Affairs Pacific Islands Health Care System, Departments of Medicine and Geriatrics, University of Hawaii John A. Burns School of Medicine, Pacific Health Research Institute and Kuakini Medical Center/Honolulu-Asia Aging Study, Honolulu, HI</wicri:regionArea><placeName><region type="state">Hawaï</region><settlement type="city">New York</settlement></placeName><orgName type="university">Université Columbia</orgName></affiliation></author><author><name sortKey="Lawton, Arlene" sort="Lawton, Arlene" uniqKey="Lawton A" first="Arlene" last="Lawton">Arlene Lawton</name><affiliation wicri:level="4"><country xml:lang="fr">États-Unis</country><wicri:regionArea>GH Sergievsky Center, Department of Neurology, Taub Institute for Research on Alzheimer's Disease and the Aging Brain, Department of Pathology, College of Physicians and Surgeons, Columbia University, New York, Department of Epidemiology, Mailman School of Public Health, Columbia University, New York, NY, USA, Royal University Hospital, Saskatoon, Saskatchewan, Canada, Department of Neurology, University of Kansas Medical Center, Kansas City, KS, USA, Veterans Affairs Pacific Islands Health Care System, Departments of Medicine and Geriatrics, University of Hawaii John A. Burns School of Medicine, Pacific Health Research Institute and Kuakini Medical Center/Honolulu-Asia Aging Study, Honolulu, HI</wicri:regionArea><placeName><region type="state">Hawaï</region><settlement type="city">New York</settlement></placeName><orgName type="university">Université Columbia</orgName></affiliation></author><author><name sortKey="Hernandez, Nora" sort="Hernandez, Nora" uniqKey="Hernandez N" first="Nora" last="Hernandez">Nora Hernandez</name><affiliation wicri:level="4"><country xml:lang="fr">États-Unis</country><wicri:regionArea>GH Sergievsky Center, Department of Neurology, Taub Institute for Research on Alzheimer's Disease and the Aging Brain, Department of Pathology, College of Physicians and Surgeons, Columbia University, New York, Department of Epidemiology, Mailman School of Public Health, Columbia University, New York, NY, USA, Royal University Hospital, Saskatoon, Saskatchewan, Canada, Department of Neurology, University of Kansas Medical Center, Kansas City, KS, USA, Veterans Affairs Pacific Islands Health Care System, Departments of Medicine and Geriatrics, University of Hawaii John A. Burns School of Medicine, Pacific Health Research Institute and Kuakini Medical Center/Honolulu-Asia Aging Study, Honolulu, HI</wicri:regionArea><placeName><region type="state">Hawaï</region><settlement type="city">New York</settlement></placeName><orgName type="university">Université Columbia</orgName></affiliation></author></analytic><monogr></monogr><series><title level="j">Brain</title><idno type="ISSN">0006-8950</idno><idno type="eISSN">1460-2156</idno><imprint><publisher>Oxford University Press</publisher><date type="published" when="2007-12">2007-12</date><biblScope unit="volume">130</biblScope><biblScope unit="issue">12</biblScope><biblScope unit="page" from="3297">3297</biblScope><biblScope unit="page" to="3307">3307</biblScope></imprint><idno type="ISSN">0006-8950</idno></series></biblStruct></sourceDesc><seriesStmt><idno type="ISSN">0006-8950</idno></seriesStmt></fileDesc><profileDesc><textClass></textClass></profileDesc></teiHeader><front><div type="abstract">Despite its being one of the most commonly observed neurological disorders, neuropathological studies of essential tremor (ET) are rare. There have been surprisingly few autopsy studies and even fewer case-control comparisons. The primary objective was to describe and quantify the pathological changes in 33 ET and 21 control brains. A secondary objective was to correlate clinical and pathological features. We examined autopsy tissue from the Essential Tremor Centralized Brain Repository. Eight (24.2%) of the 33 ET brains had Lewy bodies in the brainstem, mainly in the locus ceruleus. However, the majority of ET brains (25/33, 75.8%) had no Lewy bodies, but had pathological changes in the cerebellum. The mean number of Purkinje cells per 100× field was reduced in ET cases without Lewy bodies (6.6 ± 2.4 versus 9.6 ± 3.4, P < 0.01), and there were ∼7× more Purkinje cell torpedoes per section (12.6 ± 7.9 versus 1.7 ± 1.4, P < 0.001) compared to controls. ET cases without Lewy bodies also had degeneration of the dentate nucleus (two cases). Other findings in ET cases were Purkinje cell heterotopias and dendrite swellings. Lewy body ET cases were older than ET cases without Lewy bodies. Several trends were observed in ET cases without Lewy bodies, including a younger age of onset of tremor and higher proportions with gait difficulty and family history of ET. The pathological changes of ET seem to be heterogeneous and degenerative. The majority have cerebellar changes without Lewy bodies; a smaller proportion has brainstem Lewy bodies. The clinical differences between cases with versus without Lewy bodies require additional study.</div></front></TEI><affiliations><list><country><li>États-Unis</li></country><region><li>Hawaï</li></region><settlement><li>New York</li></settlement><orgName><li>Université Columbia</li></orgName></list><tree><country name="États-Unis"><region name="Hawaï"><name sortKey="Louis, Elan D" sort="Louis, Elan D" uniqKey="Louis E" first="Elan D." last="Louis">Elan D. Louis</name></region><name sortKey="Borden, Sarah" sort="Borden, Sarah" uniqKey="Borden S" first="Sarah" last="Borden">Sarah Borden</name><name sortKey="Faust, Phyllis L" sort="Faust, Phyllis L" uniqKey="Faust P" first="Phyllis L." last="Faust">Phyllis L. Faust</name><name sortKey="Hernandez, Nora" sort="Hernandez, Nora" uniqKey="Hernandez N" first="Nora" last="Hernandez">Nora Hernandez</name><name sortKey="Honig, Lawrence S" sort="Honig, Lawrence S" uniqKey="Honig L" first="Lawrence S." last="Honig">Lawrence S. Honig</name><name sortKey="Lawton, Arlene" sort="Lawton, Arlene" uniqKey="Lawton A" first="Arlene" last="Lawton">Arlene Lawton</name><name sortKey="Louis, Elan D" sort="Louis, Elan D" uniqKey="Louis E" first="Elan D." last="Louis">Elan D. Louis</name><name sortKey="Lyons, Kelly E" sort="Lyons, Kelly E" uniqKey="Lyons K" first="Kelly E." last="Lyons">Kelly E. Lyons</name><name sortKey="Moskowitz, Carol B" sort="Moskowitz, Carol B" uniqKey="Moskowitz C" first="Carol B." last="Moskowitz">Carol B. Moskowitz</name><name sortKey="Pahwa, Rajesh" sort="Pahwa, Rajesh" uniqKey="Pahwa R" first="Rajesh" last="Pahwa">Rajesh Pahwa</name><name sortKey="Rajput, Alex" sort="Rajput, Alex" uniqKey="Rajput A" first="Alex" last="Rajput">Alex Rajput</name><name sortKey="Rajput, Ali" sort="Rajput, Ali" uniqKey="Rajput A" first="Ali" last="Rajput">Ali Rajput</name><name sortKey="Robinson, Christopher A" sort="Robinson, Christopher A" uniqKey="Robinson C" first="Christopher A." last="Robinson">Christopher A. Robinson</name><name sortKey="Ross, G Webster" sort="Ross, G Webster" uniqKey="Ross G" first="G. Webster" last="Ross">G. Webster Ross</name><name sortKey="Vonsattel, Jean Paul G" sort="Vonsattel, Jean Paul G" uniqKey="Vonsattel J" first="Jean-Paul G." last="Vonsattel">Jean-Paul G. Vonsattel</name></country></tree></affiliations></record>Pour manipuler ce document sous Unix (Dilib)
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