{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,5]],"date-time":"2026-10-05T15:40:29Z","timestamp":1791214829728,"version":"4.1.0"},"reference-count":24,"publisher":"Wiley","issue":"1","license":[{"start":{"date-parts":[[2024,4,18]],"date-time":"2024-04-18T00:00:00Z","timestamp":1713398400000},"content-version":"vor","delay-in-days":0,"URL":"http:\/\/onlinelibrary.wiley.com\/termsAndConditions#vor"}],"funder":[{"DOI":"10.13039\/100000002","name":"National Institutes of Health","doi-asserted-by":"publisher","award":["RO1NS126227"],"award-info":[{"award-number":["RO1NS126227"]}],"id":[{"id":"10.13039\/100000002","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/100010269","name":"Wellcome Trust","doi-asserted-by":"publisher","award":["203930\/B\/16\/Z"],"award-info":[{"award-number":["203930\/B\/16\/Z"]}],"id":[{"id":"10.13039\/100010269","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":["onlinelibrary.wiley.com"],"crossmark-restriction":true},"short-container-title":["Annals of Neurology"],"published-print":{"date-parts":[[2024,7]]},"abstract":"<jats:sec><jats:title>Objectives<\/jats:title><jats:p>To report an autoimmune paraneoplastic encephalitis characterized by immunoglobulin G (IgG) antibody targeting synaptic protein calmodulin kinase\u2010like vesicle\u2010associated (CAMKV).<\/jats:p><\/jats:sec><jats:sec><jats:title>Methods<\/jats:title><jats:p>Serum and cerebrospinal fluid (CSF) samples harboring unclassified antibodies on murine brain\u2010based indirect immunofluorescence assay (IFA) were screened by human protein microarray. In 5 patients with identical cerebral IFA staining, CAMKV was identified as top\u2010ranking candidate antigen. Western blots, confocal microscopy, immune\u2010absorption, and mass spectrometry were performed to substantiate CAMKV specificity. Recombinant CAMKV\u2010specific assays (cell\u2010based [fixed and live] and Western blot) provided additional confirmation.<\/jats:p><\/jats:sec><jats:sec><jats:title>Results<\/jats:title><jats:p>Of 5 CAMKV\u2010IgG positive patients, 3 were women (median symptom\u2010onset age was 59\u2009years; range, 53\u201374). Encephalitis\u2010onset was subacute (4) or acute (1) and manifested with: altered mental status (all), seizures (4), hyperkinetic movements (4), psychiatric features (3), memory loss (2), and insomnia (2). Paraclinical testing revealed CSF lymphocytic pleocytosis (all 4 tested), electrographic seizures (3 of 4 tested), and striking MRI abnormalities in all (mesial temporal lobe T2 hyperintensities [all patients], caudate head T2 hyperintensities [3], and cortical diffusion weighted hyperintensities [2]). None had post\u2010gadolinium enhancement. Cancers were uterine adenocarcinoma (3 patients: poorly differentiated or neuroendocrine\u2010differentiated in 2, both demonstrated CAMKV immunoreactivity), bladder urothelial carcinoma (1), and non\u2010Hodgkin lymphoma (1). Two patients developed encephalitis following immune checkpoint inhibitor cancer therapy (atezolizumab [1], pembrolizumab [1]). All treated patients (4) demonstrated an initial response to immunotherapy (corticosteroids [4], IVIG [2]), though 3 died from cancer.<\/jats:p><\/jats:sec><jats:sec><jats:title>Interpretation<\/jats:title><jats:p>CAMKV\u2010IgG is a biomarker of immunotherapy\u2010responsive paraneoplastic encephalitis with temporal and extratemporal features and uterine cancer as a prominent oncologic association. ANN NEUROL 2024;96:21\u201333<\/jats:p><\/jats:sec>","DOI":"10.1002\/ana.26943","type":"journal-article","created":{"date-parts":[[2024,4,18]],"date-time":"2024-04-18T11:30:26Z","timestamp":1713439826000},"page":"21-33","update-policy":"https:\/\/doi.org\/10.1002\/crossmark_policy","source":"Crossref","is-referenced-by-count":13,"title":["Paraneoplastic Calmodulin Kinase\u2010Like Vesicle\u2010Associated Protein (<scp>CAMKV<\/scp>) Autoimmune Encephalitis"],"prefix":"10.1002","volume":"96","author":[{"given":"Michael","family":"Gilligan","sequence":"first","affiliation":[{"name":"Department of Laboratory Medicine and Pathology Mayo Clinic  Rochester MN USA"},{"name":"Department of Neurology University College Dublin, St Vincent's Hospital Elm Park  Dublin Ireland"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Connie E.","family":"Lesnick","sequence":"additional","affiliation":[{"name":"Department of Laboratory Medicine and Pathology Mayo Clinic  Rochester MN USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yong","family":"Guo","sequence":"additional","affiliation":[{"name":"Department of Neurology Mayo Clinic  Rochester MN USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-7133-4679","authenticated-orcid":false,"given":"Michael J.","family":"Bradshaw","sequence":"additional","affiliation":[{"name":"Department of Neurology University of Washington and Billings Clinic  Billings MT USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shafeeq S.","family":"Ladha","sequence":"additional","affiliation":[{"name":"Department of Neurology Barrow Neurological Institute  Phoenix AZ USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mihaela","family":"Nowak","sequence":"additional","affiliation":[{"name":"Department of Neurology Jefferson Hospital  Jefferson Hills PA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Maulik P.","family":"Shah","sequence":"additional","affiliation":[{"name":"Department of Neurology University of California  San Francisco CA USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"John R.","family":"Wittenborn","sequence":"additional","affiliation":[{"name":"Department of Neurology Robert Wood Johnson University Hospital  New Brunswick NJ USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Eati","family":"Basal","sequence":"additional","affiliation":[{"name":"Department of Laboratory Medicine and Pathology Mayo Clinic  Rochester MN USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shannon","family":"Hinson","sequence":"additional","affiliation":[{"name":"Department of Laboratory Medicine and Pathology Mayo Clinic  Rochester MN USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Binxia","family":"Yang","sequence":"additional","affiliation":[{"name":"Department of Laboratory Medicine and Pathology Mayo Clinic  Rochester MN USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4302-4075","authenticated-orcid":false,"given":"Divyanshu","family":"Dubey","sequence":"additional","affiliation":[{"name":"Department of Laboratory Medicine and Pathology Mayo Clinic  Rochester MN USA"},{"name":"Department of Neurology Mayo Clinic  Rochester MN USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"John R.","family":"Mills","sequence":"additional","affiliation":[{"name":"Department of Laboratory Medicine and Pathology Mayo Clinic  Rochester MN USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6140-5584","authenticated-orcid":false,"given":"Sean J.","family":"Pittock","sequence":"additional","affiliation":[{"name":"Department of Laboratory Medicine and Pathology Mayo Clinic  Rochester MN USA"},{"name":"Department of Neurology Mayo Clinic  Rochester MN USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-1960-9065","authenticated-orcid":false,"given":"Anastasia","family":"Zekeridou","sequence":"additional","affiliation":[{"name":"Department of Laboratory Medicine and Pathology Mayo Clinic  Rochester MN USA"},{"name":"Department of Neurology Mayo Clinic  Rochester MN USA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6856-8143","authenticated-orcid":false,"given":"Andrew","family":"McKeon","sequence":"additional","affiliation":[{"name":"Department of Laboratory Medicine and Pathology Mayo Clinic  Rochester MN USA"},{"name":"Department of Neurology Mayo Clinic  Rochester MN USA"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"311","published-online":{"date-parts":[[2024,4,18]]},"reference":[{"key":"e_1_2_12_2_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1474-4422(15)00401-9"},{"key":"e_1_2_12_3_1","doi-asserted-by":"publisher","DOI":"10.1212\/CPJ.0000000000200151"},{"key":"e_1_2_12_4_1","doi-asserted-by":"publisher","DOI":"10.1212\/NXI.0000000000001014"},{"key":"e_1_2_12_5_1","doi-asserted-by":"publisher","DOI":"10.1212\/NXI.0000000000200145"},{"key":"e_1_2_12_6_1","doi-asserted-by":"publisher","DOI":"10.1038\/ncomms13282"},{"key":"e_1_2_12_7_1","doi-asserted-by":"publisher","DOI":"10.3389\/fnsyn.2022.926570"},{"key":"e_1_2_12_8_1","doi-asserted-by":"publisher","DOI":"10.1523\/JNEUROSCI.14-01-00001.1994"},{"key":"e_1_2_12_9_1","first-page":"197","article-title":"A novel Druggable dual\u2010specificity tYrosine\u2010regulated Kinase3\/Calmodulin kinase\u2010like vesicle\u2010associated signaling module with therapeutic implications in Neuroblastoma","volume":"12","author":"Rozen EJ","year":"2024","journal-title":"Biomedicine"},{"key":"e_1_2_12_10_1","doi-asserted-by":"publisher","DOI":"10.1111\/ejn.14958"},{"key":"e_1_2_12_11_1","doi-asserted-by":"publisher","DOI":"10.3389\/fonc.2020.00302"},{"key":"e_1_2_12_12_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.prp.2019.152570"},{"key":"e_1_2_12_13_1","doi-asserted-by":"publisher","DOI":"10.1186\/s12967-019-02148-3"},{"key":"e_1_2_12_14_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0000000000007971"},{"key":"e_1_2_12_15_1","doi-asserted-by":"publisher","DOI":"10.1007\/s00415-018-9143-x"},{"key":"e_1_2_12_16_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1474-4422(23)00369-1"},{"key":"e_1_2_12_17_1","doi-asserted-by":"publisher","DOI":"10.1111\/ene.15835"},{"key":"e_1_2_12_18_1","doi-asserted-by":"publisher","DOI":"10.1136\/jnnp-2021-327302"},{"key":"e_1_2_12_19_1","doi-asserted-by":"publisher","DOI":"10.1016\/S1474-4422(12)70310-1"},{"key":"e_1_2_12_20_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jaut.2012.07.008"},{"key":"e_1_2_12_21_1","doi-asserted-by":"publisher","DOI":"10.1212\/01.WNL.0000289761.64862.ce"},{"key":"e_1_2_12_22_1","doi-asserted-by":"publisher","DOI":"10.1002\/ana.24881"},{"key":"e_1_2_12_23_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jneuroim.2022.577861"},{"key":"e_1_2_12_24_1","doi-asserted-by":"publisher","DOI":"10.1016\/j.jneuroim.2007.03.015"},{"key":"e_1_2_12_25_1","doi-asserted-by":"publisher","DOI":"10.1212\/WNL.0b013e3182364a44"}],"container-title":["Annals of Neurology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/pdf\/10.1002\/ana.26943","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,1,30]],"date-time":"2025-01-30T05:23:21Z","timestamp":1738214601000},"score":1,"resource":{"primary":{"URL":"https:\/\/onlinelibrary.wiley.com\/doi\/10.1002\/ana.26943"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,4,18]]},"references-count":24,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2024,7]]}},"alternative-id":["10.1002\/ana.26943"],"URL":"https:\/\/doi.org\/10.1002\/ana.26943","archive":["Portico"],"relation":{},"ISSN":["0364-5134","1531-8249"],"issn-type":[{"value":"0364-5134","type":"print"},{"value":"1531-8249","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,4,18]]},"assertion":[{"value":"2024-01-24","order":0,"name":"received","label":"Received","group":{"name":"publication_history","label":"Publication History"}},{"value":"2024-04-03","order":2,"name":"accepted","label":"Accepted","group":{"name":"publication_history","label":"Publication History"}},{"value":"2024-04-18","order":3,"name":"published","label":"Published","group":{"name":"publication_history","label":"Publication History"}}]}}