Ocular flutter as the presenting manifestation of autoimmune glial fibrillary acidic protein astrocytopathy
A 39-year-old man exhibited ocular flutter and cerebellar ataxia following a subacute disturbance of consciousness and partial seizure. He was diagnosed with autoimmune glial fibrillary acidic protein (GFAP) astrocytopathy by tissue- and cell-based antibody assays. Brain single-photon emission compu...
Gespeichert in:
| Veröffentlicht in: | Clinical neurology and neurosurgery 2022-08, Vol.219, p.107307-107307, Article 107307 |
|---|---|
| Hauptverfasser: | , , , , , , , , , , , |
| Format: | Artikel |
| Sprache: | eng |
| Schlagworte: | |
| Online-Zugang: | Volltext |
| Tags: |
Tag hinzufügen
Keine Tags, Fügen Sie den ersten Tag hinzu!
|
| container_end_page | 107307 |
|---|---|
| container_issue | |
| container_start_page | 107307 |
| container_title | Clinical neurology and neurosurgery |
| container_volume | 219 |
| creator | Wada, Taishi Higashiyama, Yuichi Kunii, Misako Jono, Takashi Kobayashi, Takuo Kubota, Shun Tada, Mikiko Hara, Makoto Kimura, Akio Doi, Hiroshi Takeuchi, Hideyuki Tanaka, Fumiaki |
| description | A 39-year-old man exhibited ocular flutter and cerebellar ataxia following a subacute disturbance of consciousness and partial seizure. He was diagnosed with autoimmune glial fibrillary acidic protein (GFAP) astrocytopathy by tissue- and cell-based antibody assays. Brain single-photon emission computed tomography detected a significant increase in blood flow in the fastigial nucleus, a critical region for eye saccade control. Immunotherapies diminished the ocular flutter and reduced hyperperfusion in the fastigial nucleus. This case suggests that autoimmune GFAP astrocytopathy can cause ocular flutter and provides strong imaging evidence supporting the hypothesis that ocular flutter is caused by hyperactivity or disinhibition of the fastigial nucleus.
•Ocular flutter is a novel manifestation of autoimmune GFAP astrocytopathy.•Ocular flutter may be caused by dysfunction of the fastigial nucleus. |
| doi_str_mv | 10.1016/j.clineuro.2022.107307 |
| format | Article |
| fullrecord | <record><control><sourceid>proquest_cross</sourceid><recordid>TN_cdi_proquest_miscellaneous_2673598364</recordid><sourceformat>XML</sourceformat><sourcesystem>PC</sourcesystem><els_id>S0303846722001883</els_id><sourcerecordid>2697340224</sourcerecordid><originalsourceid>FETCH-LOGICAL-c554t-741e10deeeaaabec7936ab9290872704ff989d9e4862702919b0df51133a0a933</originalsourceid><addsrcrecordid>eNqFkU9PHSEUxUljU5-2X8GQuOlmnjDMwLCzMVpNTNzYNWGYi_KcgSd_mrxvLy9Pu-jGFYH87uHccxA6o2RNCeUXm7WZnYcSw7olbVsfBSPiC1rRQbQNl3w4QivCCGuGjotjdJLShhDCGB--oWPWc96Svl-hlwdTZh2xnUvOELFOOD8D3kZI4LPzT3jR3llIWWcXPA4W65KDW5biAT_NTs_YujG6uarssDZucqaOhwzOV7Ucg9nlsNX5efcdfbV6TvDj_TxFf26uH69um_uH33dXv-4b0_ddbkRHgZIJALTWIxghGdejbCWpqwnSWSsHOUnoBl6vraRyJJPtKWVMEy0ZO0U_D7rVxmup1tXikoHq0EMoSbVcsF4OjHcVPf8P3YQSfXVXKSlYV7PdU_xAmRhSimDVNrql7qsoUfs61EZ91KH2dahDHXXw7F2-jAtM_8Y-8q_A5QGAmsdfB1El48AbmFwEk9UU3Gd_vAEspKCQ</addsrcrecordid><sourcetype>Aggregation Database</sourcetype><iscdi>true</iscdi><recordtype>article</recordtype><pqid>2697340224</pqid></control><display><type>article</type><title>Ocular flutter as the presenting manifestation of autoimmune glial fibrillary acidic protein astrocytopathy</title><source>Elsevier ScienceDirect Journals</source><source>ProQuest Central UK/Ireland</source><creator>Wada, Taishi ; Higashiyama, Yuichi ; Kunii, Misako ; Jono, Takashi ; Kobayashi, Takuo ; Kubota, Shun ; Tada, Mikiko ; Hara, Makoto ; Kimura, Akio ; Doi, Hiroshi ; Takeuchi, Hideyuki ; Tanaka, Fumiaki</creator><creatorcontrib>Wada, Taishi ; Higashiyama, Yuichi ; Kunii, Misako ; Jono, Takashi ; Kobayashi, Takuo ; Kubota, Shun ; Tada, Mikiko ; Hara, Makoto ; Kimura, Akio ; Doi, Hiroshi ; Takeuchi, Hideyuki ; Tanaka, Fumiaki</creatorcontrib><description>A 39-year-old man exhibited ocular flutter and cerebellar ataxia following a subacute disturbance of consciousness and partial seizure. He was diagnosed with autoimmune glial fibrillary acidic protein (GFAP) astrocytopathy by tissue- and cell-based antibody assays. Brain single-photon emission computed tomography detected a significant increase in blood flow in the fastigial nucleus, a critical region for eye saccade control. Immunotherapies diminished the ocular flutter and reduced hyperperfusion in the fastigial nucleus. This case suggests that autoimmune GFAP astrocytopathy can cause ocular flutter and provides strong imaging evidence supporting the hypothesis that ocular flutter is caused by hyperactivity or disinhibition of the fastigial nucleus.
•Ocular flutter is a novel manifestation of autoimmune GFAP astrocytopathy.•Ocular flutter may be caused by dysfunction of the fastigial nucleus.</description><identifier>ISSN: 0303-8467</identifier><identifier>EISSN: 1872-6968</identifier><identifier>DOI: 10.1016/j.clineuro.2022.107307</identifier><identifier>PMID: 35662055</identifier><language>eng</language><publisher>Netherlands: Elsevier B.V</publisher><subject>Astrocytopathy ; Ataxia ; Autoantibody ; Blood flow ; Brain research ; Cerebellar ataxia ; Cerebellum ; Computed tomography ; Consciousness ; Editing ; Eye movements ; Glial fibrillary acidic protein ; Headaches ; Hyperactivity ; Immunotherapy ; Magnetic resonance imaging ; Neuroimaging ; Neurology ; Ocular flutter ; Opsoclonus ; Pathogenesis ; Proteins ; Saccadic eye movements ; Seizures ; Supervision ; Tomography ; Writing</subject><ispartof>Clinical neurology and neurosurgery, 2022-08, Vol.219, p.107307-107307, Article 107307</ispartof><rights>2022 Elsevier B.V.</rights><rights>Copyright © 2022 Elsevier B.V. All rights reserved.</rights><rights>2022. Elsevier B.V.</rights><lds50>peer_reviewed</lds50><oa>free_for_read</oa><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c554t-741e10deeeaaabec7936ab9290872704ff989d9e4862702919b0df51133a0a933</citedby><cites>FETCH-LOGICAL-c554t-741e10deeeaaabec7936ab9290872704ff989d9e4862702919b0df51133a0a933</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktohtml>$$Uhttps://www.proquest.com/docview/2697340224?pq-origsite=primo$$EHTML$$P50$$Gproquest$$H</linktohtml><link.rule.ids>314,776,780,3536,27903,27904,45974,64362,64364,64366,72216</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/35662055$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Wada, Taishi</creatorcontrib><creatorcontrib>Higashiyama, Yuichi</creatorcontrib><creatorcontrib>Kunii, Misako</creatorcontrib><creatorcontrib>Jono, Takashi</creatorcontrib><creatorcontrib>Kobayashi, Takuo</creatorcontrib><creatorcontrib>Kubota, Shun</creatorcontrib><creatorcontrib>Tada, Mikiko</creatorcontrib><creatorcontrib>Hara, Makoto</creatorcontrib><creatorcontrib>Kimura, Akio</creatorcontrib><creatorcontrib>Doi, Hiroshi</creatorcontrib><creatorcontrib>Takeuchi, Hideyuki</creatorcontrib><creatorcontrib>Tanaka, Fumiaki</creatorcontrib><title>Ocular flutter as the presenting manifestation of autoimmune glial fibrillary acidic protein astrocytopathy</title><title>Clinical neurology and neurosurgery</title><addtitle>Clin Neurol Neurosurg</addtitle><description>A 39-year-old man exhibited ocular flutter and cerebellar ataxia following a subacute disturbance of consciousness and partial seizure. He was diagnosed with autoimmune glial fibrillary acidic protein (GFAP) astrocytopathy by tissue- and cell-based antibody assays. Brain single-photon emission computed tomography detected a significant increase in blood flow in the fastigial nucleus, a critical region for eye saccade control. Immunotherapies diminished the ocular flutter and reduced hyperperfusion in the fastigial nucleus. This case suggests that autoimmune GFAP astrocytopathy can cause ocular flutter and provides strong imaging evidence supporting the hypothesis that ocular flutter is caused by hyperactivity or disinhibition of the fastigial nucleus.
•Ocular flutter is a novel manifestation of autoimmune GFAP astrocytopathy.•Ocular flutter may be caused by dysfunction of the fastigial nucleus.</description><subject>Astrocytopathy</subject><subject>Ataxia</subject><subject>Autoantibody</subject><subject>Blood flow</subject><subject>Brain research</subject><subject>Cerebellar ataxia</subject><subject>Cerebellum</subject><subject>Computed tomography</subject><subject>Consciousness</subject><subject>Editing</subject><subject>Eye movements</subject><subject>Glial fibrillary acidic protein</subject><subject>Headaches</subject><subject>Hyperactivity</subject><subject>Immunotherapy</subject><subject>Magnetic resonance imaging</subject><subject>Neuroimaging</subject><subject>Neurology</subject><subject>Ocular flutter</subject><subject>Opsoclonus</subject><subject>Pathogenesis</subject><subject>Proteins</subject><subject>Saccadic eye movements</subject><subject>Seizures</subject><subject>Supervision</subject><subject>Tomography</subject><subject>Writing</subject><issn>0303-8467</issn><issn>1872-6968</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2022</creationdate><recordtype>article</recordtype><sourceid>8G5</sourceid><sourceid>ABUWG</sourceid><sourceid>AFKRA</sourceid><sourceid>AZQEC</sourceid><sourceid>BENPR</sourceid><sourceid>CCPQU</sourceid><sourceid>DWQXO</sourceid><sourceid>GNUQQ</sourceid><sourceid>GUQSH</sourceid><sourceid>M2O</sourceid><recordid>eNqFkU9PHSEUxUljU5-2X8GQuOlmnjDMwLCzMVpNTNzYNWGYi_KcgSd_mrxvLy9Pu-jGFYH87uHccxA6o2RNCeUXm7WZnYcSw7olbVsfBSPiC1rRQbQNl3w4QivCCGuGjotjdJLShhDCGB--oWPWc96Svl-hlwdTZh2xnUvOELFOOD8D3kZI4LPzT3jR3llIWWcXPA4W65KDW5biAT_NTs_YujG6uarssDZucqaOhwzOV7Ucg9nlsNX5efcdfbV6TvDj_TxFf26uH69um_uH33dXv-4b0_ddbkRHgZIJALTWIxghGdejbCWpqwnSWSsHOUnoBl6vraRyJJPtKWVMEy0ZO0U_D7rVxmup1tXikoHq0EMoSbVcsF4OjHcVPf8P3YQSfXVXKSlYV7PdU_xAmRhSimDVNrql7qsoUfs61EZ91KH2dahDHXXw7F2-jAtM_8Y-8q_A5QGAmsdfB1El48AbmFwEk9UU3Gd_vAEspKCQ</recordid><startdate>20220801</startdate><enddate>20220801</enddate><creator>Wada, Taishi</creator><creator>Higashiyama, Yuichi</creator><creator>Kunii, Misako</creator><creator>Jono, Takashi</creator><creator>Kobayashi, Takuo</creator><creator>Kubota, Shun</creator><creator>Tada, Mikiko</creator><creator>Hara, Makoto</creator><creator>Kimura, Akio</creator><creator>Doi, Hiroshi</creator><creator>Takeuchi, Hideyuki</creator><creator>Tanaka, Fumiaki</creator><general>Elsevier B.V</general><general>Elsevier Limited</general><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>3V.</scope><scope>7TK</scope><scope>7X7</scope><scope>7XB</scope><scope>88E</scope><scope>88G</scope><scope>8FI</scope><scope>8FJ</scope><scope>8FK</scope><scope>8G5</scope><scope>ABUWG</scope><scope>AFKRA</scope><scope>AZQEC</scope><scope>BENPR</scope><scope>CCPQU</scope><scope>DWQXO</scope><scope>FYUFA</scope><scope>GHDGH</scope><scope>GNUQQ</scope><scope>GUQSH</scope><scope>K9.</scope><scope>M0S</scope><scope>M1P</scope><scope>M2M</scope><scope>M2O</scope><scope>MBDVC</scope><scope>PQEST</scope><scope>PQQKQ</scope><scope>PQUKI</scope><scope>PRINS</scope><scope>PSYQQ</scope><scope>Q9U</scope><scope>7X8</scope></search><sort><creationdate>20220801</creationdate><title>Ocular flutter as the presenting manifestation of autoimmune glial fibrillary acidic protein astrocytopathy</title><author>Wada, Taishi ; Higashiyama, Yuichi ; Kunii, Misako ; Jono, Takashi ; Kobayashi, Takuo ; Kubota, Shun ; Tada, Mikiko ; Hara, Makoto ; Kimura, Akio ; Doi, Hiroshi ; Takeuchi, Hideyuki ; Tanaka, Fumiaki</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c554t-741e10deeeaaabec7936ab9290872704ff989d9e4862702919b0df51133a0a933</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2022</creationdate><topic>Astrocytopathy</topic><topic>Ataxia</topic><topic>Autoantibody</topic><topic>Blood flow</topic><topic>Brain research</topic><topic>Cerebellar ataxia</topic><topic>Cerebellum</topic><topic>Computed tomography</topic><topic>Consciousness</topic><topic>Editing</topic><topic>Eye movements</topic><topic>Glial fibrillary acidic protein</topic><topic>Headaches</topic><topic>Hyperactivity</topic><topic>Immunotherapy</topic><topic>Magnetic resonance imaging</topic><topic>Neuroimaging</topic><topic>Neurology</topic><topic>Ocular flutter</topic><topic>Opsoclonus</topic><topic>Pathogenesis</topic><topic>Proteins</topic><topic>Saccadic eye movements</topic><topic>Seizures</topic><topic>Supervision</topic><topic>Tomography</topic><topic>Writing</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Wada, Taishi</creatorcontrib><creatorcontrib>Higashiyama, Yuichi</creatorcontrib><creatorcontrib>Kunii, Misako</creatorcontrib><creatorcontrib>Jono, Takashi</creatorcontrib><creatorcontrib>Kobayashi, Takuo</creatorcontrib><creatorcontrib>Kubota, Shun</creatorcontrib><creatorcontrib>Tada, Mikiko</creatorcontrib><creatorcontrib>Hara, Makoto</creatorcontrib><creatorcontrib>Kimura, Akio</creatorcontrib><creatorcontrib>Doi, Hiroshi</creatorcontrib><creatorcontrib>Takeuchi, Hideyuki</creatorcontrib><creatorcontrib>Tanaka, Fumiaki</creatorcontrib><collection>PubMed</collection><collection>CrossRef</collection><collection>ProQuest Central (Corporate)</collection><collection>Neurosciences Abstracts</collection><collection>Health & Medical Collection</collection><collection>ProQuest Central (purchase pre-March 2016)</collection><collection>Medical Database (Alumni Edition)</collection><collection>Psychology Database (Alumni)</collection><collection>Hospital Premium Collection</collection><collection>Hospital Premium Collection (Alumni Edition)</collection><collection>ProQuest Central (Alumni) (purchase pre-March 2016)</collection><collection>Research Library (Alumni Edition)</collection><collection>ProQuest Central (Alumni Edition)</collection><collection>ProQuest Central UK/Ireland</collection><collection>ProQuest Central Essentials</collection><collection>ProQuest Central</collection><collection>ProQuest One Community College</collection><collection>ProQuest Central Korea</collection><collection>Health Research Premium Collection</collection><collection>Health Research Premium Collection (Alumni)</collection><collection>ProQuest Central Student</collection><collection>Research Library Prep</collection><collection>ProQuest Health & Medical Complete (Alumni)</collection><collection>Health & Medical Collection (Alumni Edition)</collection><collection>Medical Database</collection><collection>Psychology Database</collection><collection>Research Library</collection><collection>Research Library (Corporate)</collection><collection>ProQuest One Academic Eastern Edition (DO NOT USE)</collection><collection>ProQuest One Academic</collection><collection>ProQuest One Academic UKI Edition</collection><collection>ProQuest Central China</collection><collection>ProQuest One Psychology</collection><collection>ProQuest Central Basic</collection><collection>MEDLINE - Academic</collection><jtitle>Clinical neurology and neurosurgery</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Wada, Taishi</au><au>Higashiyama, Yuichi</au><au>Kunii, Misako</au><au>Jono, Takashi</au><au>Kobayashi, Takuo</au><au>Kubota, Shun</au><au>Tada, Mikiko</au><au>Hara, Makoto</au><au>Kimura, Akio</au><au>Doi, Hiroshi</au><au>Takeuchi, Hideyuki</au><au>Tanaka, Fumiaki</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Ocular flutter as the presenting manifestation of autoimmune glial fibrillary acidic protein astrocytopathy</atitle><jtitle>Clinical neurology and neurosurgery</jtitle><addtitle>Clin Neurol Neurosurg</addtitle><date>2022-08-01</date><risdate>2022</risdate><volume>219</volume><spage>107307</spage><epage>107307</epage><pages>107307-107307</pages><artnum>107307</artnum><issn>0303-8467</issn><eissn>1872-6968</eissn><abstract>A 39-year-old man exhibited ocular flutter and cerebellar ataxia following a subacute disturbance of consciousness and partial seizure. He was diagnosed with autoimmune glial fibrillary acidic protein (GFAP) astrocytopathy by tissue- and cell-based antibody assays. Brain single-photon emission computed tomography detected a significant increase in blood flow in the fastigial nucleus, a critical region for eye saccade control. Immunotherapies diminished the ocular flutter and reduced hyperperfusion in the fastigial nucleus. This case suggests that autoimmune GFAP astrocytopathy can cause ocular flutter and provides strong imaging evidence supporting the hypothesis that ocular flutter is caused by hyperactivity or disinhibition of the fastigial nucleus.
•Ocular flutter is a novel manifestation of autoimmune GFAP astrocytopathy.•Ocular flutter may be caused by dysfunction of the fastigial nucleus.</abstract><cop>Netherlands</cop><pub>Elsevier B.V</pub><pmid>35662055</pmid><doi>10.1016/j.clineuro.2022.107307</doi><tpages>1</tpages><oa>free_for_read</oa></addata></record> |
| fulltext | fulltext |
| identifier | ISSN: 0303-8467 |
| ispartof | Clinical neurology and neurosurgery, 2022-08, Vol.219, p.107307-107307, Article 107307 |
| issn | 0303-8467 1872-6968 |
| language | eng |
| recordid | cdi_proquest_miscellaneous_2673598364 |
| source | Elsevier ScienceDirect Journals; ProQuest Central UK/Ireland |
| subjects | Astrocytopathy Ataxia Autoantibody Blood flow Brain research Cerebellar ataxia Cerebellum Computed tomography Consciousness Editing Eye movements Glial fibrillary acidic protein Headaches Hyperactivity Immunotherapy Magnetic resonance imaging Neuroimaging Neurology Ocular flutter Opsoclonus Pathogenesis Proteins Saccadic eye movements Seizures Supervision Tomography Writing |
| title | Ocular flutter as the presenting manifestation of autoimmune glial fibrillary acidic protein astrocytopathy |
| url | https://sfx.bib-bvb.de/sfx_tum?ctx_ver=Z39.88-2004&ctx_enc=info:ofi/enc:UTF-8&ctx_tim=2025-01-22T08%3A34%3A47IST&url_ver=Z39.88-2004&url_ctx_fmt=infofi/fmt:kev:mtx:ctx&rfr_id=info:sid/primo.exlibrisgroup.com:primo3-Article-proquest_cross&rft_val_fmt=info:ofi/fmt:kev:mtx:journal&rft.genre=article&rft.atitle=Ocular%20flutter%20as%20the%20presenting%20manifestation%20of%20autoimmune%20glial%20fibrillary%20acidic%20protein%20astrocytopathy&rft.jtitle=Clinical%20neurology%20and%20neurosurgery&rft.au=Wada,%20Taishi&rft.date=2022-08-01&rft.volume=219&rft.spage=107307&rft.epage=107307&rft.pages=107307-107307&rft.artnum=107307&rft.issn=0303-8467&rft.eissn=1872-6968&rft_id=info:doi/10.1016/j.clineuro.2022.107307&rft_dat=%3Cproquest_cross%3E2697340224%3C/proquest_cross%3E%3Curl%3E%3C/url%3E&disable_directlink=true&sfx.directlink=off&sfx.report_link=0&rft_id=info:oai/&rft_pqid=2697340224&rft_id=info:pmid/35662055&rft_els_id=S0303846722001883&rfr_iscdi=true |