Corticobasal syndrome with novel argyrophilic glial inclusions.
Identifieur interne : 003139 ( PubMed/Curation ); précédent : 003138; suivant : 003140Corticobasal syndrome with novel argyrophilic glial inclusions.
Auteurs : Gregory A. Rippon [États-Unis] ; S M Staugaitis ; Steven S M. Chin ; James E. Goldman ; K. MarderSource :
- Movement disorders : official journal of the Movement Disorder Society [ 0885-3185 ] ; 2005.
English descriptors
- KwdEn :
- Adult, Apraxia, Ideomotor (complications), Apraxia, Ideomotor (metabolism), Apraxia, Ideomotor (pathology), Atrophy (pathology), Basal Ganglia (metabolism), Basal Ganglia (pathology), Dysarthria (complications), Fatal Outcome, Female, Frontal Lobe (pathology), Glial Fibrillary Acidic Protein (metabolism), Humans, Muscle Rigidity (complications), Muscle Rigidity (metabolism), Muscle Rigidity (pathology), Nerve Degeneration (complications), Nerve Degeneration (metabolism), Nerve Degeneration (pathology), Syndrome, Ubiquitin (immunology), Ubiquitin (metabolism), alpha-Crystallins (immunology), alpha-Crystallins (metabolism).
- MESH :
- chemical , immunology : Ubiquitin, alpha-Crystallins.
- chemical , metabolism : Glial Fibrillary Acidic Protein, Ubiquitin, alpha-Crystallins.
- complications : Apraxia, Ideomotor, Dysarthria, Muscle Rigidity, Nerve Degeneration.
- metabolism : Apraxia, Ideomotor, Basal Ganglia, Muscle Rigidity, Nerve Degeneration.
- pathology : Apraxia, Ideomotor, Atrophy, Basal Ganglia, Frontal Lobe, Muscle Rigidity, Nerve Degeneration.
- Adult, Fatal Outcome, Female, Humans, Syndrome.
Abstract
A 42-year-old, left-handed woman first noted impaired dexterity of the dominant hand, soon followed by dysarthria and cognitive decline. Over a 4-year period, she developed severe left-sided apraxia with eventual neglect of the left arm and progressive extrapyramidal signs. Cognitive testing showed progressive executive, visuospatial, fluency, and naming impairment with relative preservation of memory. Single-photon emission computed tomography demonstrated asymmetric right posterior frontal and superior parietal hypoperfusion. The clinical impression was corticobasal degeneration. At autopsy, severe atrophy was seen in the perirolandic and frontal regions. There was marked neuronal loss and gliosis in the posterior frontal and precentral regions and less severe pathology in prefrontal, temporal, and parietal areas. Mild to moderate gliosis and neuronal loss were also seen in the putamen, globus pallidus, subthalamic, and dentate nuclei. Gallyas silver stain revealed numerous inclusions adjacent to oligodendrocyte nuclei in white and gray matter of affected cortical and subcortical regions. The gracile inclusions were wavy, slender, and stained positively with antibodies to ubiquitin and alphaB-crystallin but not to microtubule-associated proteins (tau, MAP1B, MAP2), tubulin, neurofilaments, glial fibrillary acidic protein, or alpha-synuclein. The argyrophilic inclusions identified in this case are distinct from those previously described in neurodegenerative diseases.
DOI: 10.1002/mds.20396
PubMed: 15726537
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Rippon</name><affiliation wicri:level="1"><nlm:affiliation>Department of Neurology, Columbia University College of Physicians and Surgeons, Columbia University, New York, New York 10032, USA. grippon@sergievsky.cpmc.columbia.edu</nlm:affiliation><country xml:lang="fr">États-Unis</country><wicri:regionArea>Department of Neurology, Columbia University College of Physicians and Surgeons, Columbia University, New York, New York 10032</wicri:regionArea></affiliation></author><author><name sortKey="Staugaitis, S M" sort="Staugaitis, S M" uniqKey="Staugaitis S" first="S M" last="Staugaitis">S M Staugaitis</name></author><author><name sortKey="Chin, Steven S M" sort="Chin, Steven S M" uniqKey="Chin S" first="Steven S M" last="Chin">Steven S M. Chin</name></author><author><name sortKey="Goldman, James E" sort="Goldman, James E" uniqKey="Goldman J" first="James E" last="Goldman">James E. Goldman</name></author><author><name sortKey="Marder, K" sort="Marder, K" uniqKey="Marder K" first="K" last="Marder">K. Marder</name></author></titleStmt><publicationStmt><idno type="wicri:source">PubMed</idno><date when="2005">2005</date><idno type="doi">10.1002/mds.20396</idno><idno type="RBID">pubmed:15726537</idno><idno type="pmid">15726537</idno><idno type="wicri:Area/PubMed/Corpus">003139</idno><idno type="wicri:Area/PubMed/Curation">003139</idno></publicationStmt><sourceDesc><biblStruct><analytic><title xml:lang="en">Corticobasal syndrome with novel argyrophilic glial inclusions.</title><author><name sortKey="Rippon, Gregory A" sort="Rippon, Gregory A" uniqKey="Rippon G" first="Gregory A" last="Rippon">Gregory A. Rippon</name><affiliation wicri:level="1"><nlm:affiliation>Department of Neurology, Columbia University College of Physicians and Surgeons, Columbia University, New York, New York 10032, USA. grippon@sergievsky.cpmc.columbia.edu</nlm:affiliation><country xml:lang="fr">États-Unis</country><wicri:regionArea>Department of Neurology, Columbia University College of Physicians and Surgeons, Columbia University, New York, New York 10032</wicri:regionArea></affiliation></author><author><name sortKey="Staugaitis, S M" sort="Staugaitis, S M" uniqKey="Staugaitis S" first="S M" last="Staugaitis">S M Staugaitis</name></author><author><name sortKey="Chin, Steven S M" sort="Chin, Steven S M" uniqKey="Chin S" first="Steven S M" last="Chin">Steven S M. Chin</name></author><author><name sortKey="Goldman, James E" sort="Goldman, James E" uniqKey="Goldman J" first="James E" last="Goldman">James E. Goldman</name></author><author><name sortKey="Marder, K" sort="Marder, K" uniqKey="Marder K" first="K" last="Marder">K. Marder</name></author></analytic><series><title level="j">Movement disorders : official journal of the Movement Disorder Society</title><idno type="ISSN">0885-3185</idno><imprint><date when="2005" type="published">2005</date></imprint></series></biblStruct></sourceDesc></fileDesc><profileDesc><textClass><keywords scheme="KwdEn" xml:lang="en"><term>Adult</term><term>Apraxia, Ideomotor (complications)</term><term>Apraxia, Ideomotor (metabolism)</term><term>Apraxia, Ideomotor (pathology)</term><term>Atrophy (pathology)</term><term>Basal Ganglia (metabolism)</term><term>Basal Ganglia (pathology)</term><term>Dysarthria (complications)</term><term>Fatal Outcome</term><term>Female</term><term>Frontal Lobe (pathology)</term><term>Glial Fibrillary Acidic Protein (metabolism)</term><term>Humans</term><term>Muscle Rigidity (complications)</term><term>Muscle Rigidity (metabolism)</term><term>Muscle Rigidity (pathology)</term><term>Nerve Degeneration (complications)</term><term>Nerve Degeneration (metabolism)</term><term>Nerve Degeneration (pathology)</term><term>Syndrome</term><term>Ubiquitin (immunology)</term><term>Ubiquitin (metabolism)</term><term>alpha-Crystallins (immunology)</term><term>alpha-Crystallins (metabolism)</term></keywords><keywords scheme="MESH" type="chemical" qualifier="immunology" xml:lang="en"><term>Ubiquitin</term><term>alpha-Crystallins</term></keywords><keywords scheme="MESH" type="chemical" qualifier="metabolism" xml:lang="en"><term>Glial Fibrillary Acidic Protein</term><term>Ubiquitin</term><term>alpha-Crystallins</term></keywords><keywords scheme="MESH" qualifier="complications" xml:lang="en"><term>Apraxia, Ideomotor</term><term>Dysarthria</term><term>Muscle Rigidity</term><term>Nerve Degeneration</term></keywords><keywords scheme="MESH" qualifier="metabolism" xml:lang="en"><term>Apraxia, Ideomotor</term><term>Basal Ganglia</term><term>Muscle Rigidity</term><term>Nerve Degeneration</term></keywords><keywords scheme="MESH" qualifier="pathology" xml:lang="en"><term>Apraxia, Ideomotor</term><term>Atrophy</term><term>Basal Ganglia</term><term>Frontal Lobe</term><term>Muscle Rigidity</term><term>Nerve Degeneration</term></keywords><keywords scheme="MESH" xml:lang="en"><term>Adult</term><term>Fatal Outcome</term><term>Female</term><term>Humans</term><term>Syndrome</term></keywords></textClass></profileDesc></teiHeader><front><div type="abstract" xml:lang="en">A 42-year-old, left-handed woman first noted impaired dexterity of the dominant hand, soon followed by dysarthria and cognitive decline. Over a 4-year period, she developed severe left-sided apraxia with eventual neglect of the left arm and progressive extrapyramidal signs. Cognitive testing showed progressive executive, visuospatial, fluency, and naming impairment with relative preservation of memory. Single-photon emission computed tomography demonstrated asymmetric right posterior frontal and superior parietal hypoperfusion. The clinical impression was corticobasal degeneration. At autopsy, severe atrophy was seen in the perirolandic and frontal regions. There was marked neuronal loss and gliosis in the posterior frontal and precentral regions and less severe pathology in prefrontal, temporal, and parietal areas. Mild to moderate gliosis and neuronal loss were also seen in the putamen, globus pallidus, subthalamic, and dentate nuclei. Gallyas silver stain revealed numerous inclusions adjacent to oligodendrocyte nuclei in white and gray matter of affected cortical and subcortical regions. The gracile inclusions were wavy, slender, and stained positively with antibodies to ubiquitin and alphaB-crystallin but not to microtubule-associated proteins (tau, MAP1B, MAP2), tubulin, neurofilaments, glial fibrillary acidic protein, or alpha-synuclein. The argyrophilic inclusions identified in this case are distinct from those previously described in neurodegenerative diseases.</div></front></TEI><pubmed><MedlineCitation Owner="NLM" Status="MEDLINE"><PMID Version="1">15726537</PMID><DateCreated><Year>2005</Year><Month>05</Month><Day>05</Day></DateCreated><DateCompleted><Year>2005</Year><Month>09</Month><Day>01</Day></DateCompleted><Article PubModel="Print"><Journal><ISSN IssnType="Print">0885-3185</ISSN><JournalIssue CitedMedium="Print"><Volume>20</Volume><Issue>5</Issue><PubDate><Year>2005</Year><Month>May</Month></PubDate></JournalIssue><Title>Movement disorders : official journal of the Movement Disorder Society</Title><ISOAbbreviation>Mov. Disord.</ISOAbbreviation></Journal><ArticleTitle>Corticobasal syndrome with novel argyrophilic glial inclusions.</ArticleTitle><Pagination><MedlinePgn>598-602</MedlinePgn></Pagination><Abstract><AbstractText>A 42-year-old, left-handed woman first noted impaired dexterity of the dominant hand, soon followed by dysarthria and cognitive decline. Over a 4-year period, she developed severe left-sided apraxia with eventual neglect of the left arm and progressive extrapyramidal signs. Cognitive testing showed progressive executive, visuospatial, fluency, and naming impairment with relative preservation of memory. Single-photon emission computed tomography demonstrated asymmetric right posterior frontal and superior parietal hypoperfusion. The clinical impression was corticobasal degeneration. At autopsy, severe atrophy was seen in the perirolandic and frontal regions. There was marked neuronal loss and gliosis in the posterior frontal and precentral regions and less severe pathology in prefrontal, temporal, and parietal areas. Mild to moderate gliosis and neuronal loss were also seen in the putamen, globus pallidus, subthalamic, and dentate nuclei. Gallyas silver stain revealed numerous inclusions adjacent to oligodendrocyte nuclei in white and gray matter of affected cortical and subcortical regions. The gracile inclusions were wavy, slender, and stained positively with antibodies to ubiquitin and alphaB-crystallin but not to microtubule-associated proteins (tau, MAP1B, MAP2), tubulin, neurofilaments, glial fibrillary acidic protein, or alpha-synuclein. The argyrophilic inclusions identified in this case are distinct from those previously described in neurodegenerative diseases.</AbstractText><CopyrightInformation>Copyright 2005 Movement Disorder Society.</CopyrightInformation></Abstract><AuthorList CompleteYN="Y"><Author ValidYN="Y"><LastName>Rippon</LastName><ForeName>Gregory A</ForeName><Initials>GA</Initials><AffiliationInfo><Affiliation>Department of Neurology, Columbia University College of Physicians and Surgeons, Columbia University, New York, New York 10032, USA. grippon@sergievsky.cpmc.columbia.edu</Affiliation></AffiliationInfo></Author><Author ValidYN="Y"><LastName>Staugaitis</LastName><ForeName>S M</ForeName><Initials>SM</Initials></Author><Author ValidYN="Y"><LastName>Chin</LastName><ForeName>Steven S M</ForeName><Initials>SS</Initials></Author><Author ValidYN="Y"><LastName>Goldman</LastName><ForeName>James E</ForeName><Initials>JE</Initials></Author><Author ValidYN="Y"><LastName>Marder</LastName><ForeName>K</ForeName><Initials>K</Initials></Author></AuthorList><Language>eng</Language><PublicationTypeList><PublicationType UI="D002363">Case Reports</PublicationType><PublicationType UI="D016428">Journal Article</PublicationType></PublicationTypeList></Article><MedlineJournalInfo><Country>United States</Country><MedlineTA>Mov Disord</MedlineTA><NlmUniqueID>8610688</NlmUniqueID><ISSNLinking>0885-3185</ISSNLinking></MedlineJournalInfo><ChemicalList><Chemical><RegistryNumber>0</RegistryNumber><NameOfSubstance UI="D005904">Glial Fibrillary Acidic Protein</NameOfSubstance></Chemical><Chemical><RegistryNumber>0</RegistryNumber><NameOfSubstance UI="D025801">Ubiquitin</NameOfSubstance></Chemical><Chemical><RegistryNumber>0</RegistryNumber><NameOfSubstance UI="D038201">alpha-Crystallins</NameOfSubstance></Chemical></ChemicalList><CitationSubset>IM</CitationSubset><MeshHeadingList><MeshHeading><DescriptorName MajorTopicYN="N" UI="D000328">Adult</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D020240">Apraxia, Ideomotor</DescriptorName><QualifierName MajorTopicYN="N" UI="Q000150">complications</QualifierName><QualifierName MajorTopicYN="Y" UI="Q000378">metabolism</QualifierName><QualifierName MajorTopicYN="N" UI="Q000473">pathology</QualifierName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D001284">Atrophy</DescriptorName><QualifierName MajorTopicYN="N" UI="Q000473">pathology</QualifierName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D001479">Basal Ganglia</DescriptorName><QualifierName MajorTopicYN="Y" UI="Q000378">metabolism</QualifierName><QualifierName MajorTopicYN="Y" UI="Q000473">pathology</QualifierName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D004401">Dysarthria</DescriptorName><QualifierName MajorTopicYN="N" UI="Q000150">complications</QualifierName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D017809">Fatal Outcome</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D005260">Female</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D005625">Frontal Lobe</DescriptorName><QualifierName MajorTopicYN="N" UI="Q000473">pathology</QualifierName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D005904">Glial Fibrillary Acidic Protein</DescriptorName><QualifierName MajorTopicYN="Y" UI="Q000378">metabolism</QualifierName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D006801">Humans</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D009127">Muscle Rigidity</DescriptorName><QualifierName MajorTopicYN="N" UI="Q000150">complications</QualifierName><QualifierName MajorTopicYN="Y" UI="Q000378">metabolism</QualifierName><QualifierName MajorTopicYN="N" UI="Q000473">pathology</QualifierName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D009410">Nerve Degeneration</DescriptorName><QualifierName MajorTopicYN="N" UI="Q000150">complications</QualifierName><QualifierName MajorTopicYN="Y" UI="Q000378">metabolism</QualifierName><QualifierName MajorTopicYN="N" UI="Q000473">pathology</QualifierName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D013577">Syndrome</DescriptorName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D025801">Ubiquitin</DescriptorName><QualifierName MajorTopicYN="N" UI="Q000276">immunology</QualifierName><QualifierName MajorTopicYN="N" UI="Q000378">metabolism</QualifierName></MeshHeading><MeshHeading><DescriptorName MajorTopicYN="N" UI="D038201">alpha-Crystallins</DescriptorName><QualifierName MajorTopicYN="N" UI="Q000276">immunology</QualifierName><QualifierName MajorTopicYN="N" UI="Q000378">metabolism</QualifierName></MeshHeading></MeshHeadingList></MedlineCitation><PubmedData><History><PubMedPubDate 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