000153279 001__ 153279 000153279 005__ 20240420115847.0 000153279 0247_ $$2pmc$$apmc:PMC7346383 000153279 0247_ $$2doi$$a10.1186/s13063-020-04516-7 000153279 0247_ $$2ISSN$$a1468-6694 000153279 0247_ $$2ISSN$$a1468-6708 000153279 0247_ $$2ISSN$$a1745-6215 000153279 0247_ $$2altmetric$$aaltmetric:85620150 000153279 0247_ $$2pmid$$apmid:32641101 000153279 037__ $$aDZNE-2020-01276 000153279 041__ $$aEnglish 000153279 082__ $$a610 000153279 1001_ $$aWickel, Jonathan$$b0 000153279 245__ $$aGenerate-Boost: study protocol for a prospective, multicenter, randomized controlled, double-blinded phase II trial to evaluate efficacy and safety of bortezomib in patients with severe autoimmune encephalitis 000153279 260__ $$aLondon$$bBioMed Central$$c2020 000153279 3367_ $$2DRIVER$$aarticle 000153279 3367_ $$2DataCite$$aOutput Types/Journal article 000153279 3367_ $$0PUB:(DE-HGF)16$$2PUB:(DE-HGF)$$aJournal Article$$bjournal$$mjournal$$s1713534493_11034 000153279 3367_ $$2BibTeX$$aARTICLE 000153279 3367_ $$2ORCID$$aJOURNAL_ARTICLE 000153279 3367_ $$00$$2EndNote$$aJournal Article 000153279 520__ $$aBackgroundAutoimmune encephalitis is a new spectrum of autoimmune disorders of the central nervous system (CNS), which are characterized by pathogenic autoantibodies against neuronal surface antigens. Clinical presentations range from acute to subacute encephalopathy with neurological and psychiatric symptoms, and life-threatening autonomic dysfunction in severe cases. There exist no approved therapies nor is data available from controlled clinical trials. Patients are usually treated with diverse combinations of immunotherapy. However, effect of immunotherapy on antibody-producing cells and thus on levels of pathogenic autoantibodies is insufficient. Therefore, therapeutic response is sometimes prolonged with necessity of long-time intensive care treatment and also irreversible deficits occur in severe cases. This trial will investigate the efficacy and safety of bortezomib, a proteasome inhibitor known to selectively deplete plasma cells, in patients with severe autoimmune encephalitis who have been treated with rituximab with insufficient response.MethodsGenerate-Boost is an investigator-initiated, multicenter, double-blinded, randomized controlled phase II trial which will be conducted in specialized neurological hospitals within the GENERATE (GErman NEtwork for Research on AuToimmune Encephalitis) network in Germany. Adult patients with severe autoimmune encephalitis (modified Rankin scale, mRS ≥ 3), autoantibodies against neuronal surface antigens, and pretreatment with rituximab are eligible for study participation. Fifty patients will be randomized 1:1 and undergo up to 3 cycles (each 21 days with 4 s. c. applications) of bortezomib or placebo. All patients will receive concomitant medication with dexamethasone, acyclovir and co-trimoxazole. The primary efficacy endpoint is the mRS score 17 weeks after first treatment application. Secondary endpoints are neurocognitive function, antibody titers, markers of neuronal cell damage, length of ICU/hospital stay, and mRS and Glasgow coma scale scores throughout the trial up to week 17. General and bortezomib-specific adverse events are monitored continuously.DiscussionThe expected outcome of the study is to obtain first reliable data on a hypothesis-driven therapeutic option in severe and difficult-to-treat autoimmune encephalitis. If treatment with bortezomib is beneficial in these cases, this will be the basis for implementation in the current guidelines. 000153279 536__ $$0G:(DE-HGF)POF3-342$$a342 - Disease Mechanisms and Model Systems (POF3-342)$$cPOF3-342$$fPOF III$$x0 000153279 588__ $$aDataset connected to CrossRef 000153279 650_2 $$2MeSH$$aAcyclovir 000153279 650_2 $$2MeSH$$aAdult 000153279 650_2 $$2MeSH$$aAutoantibodies: blood 000153279 650_2 $$2MeSH$$aBortezomib: adverse effects 000153279 650_2 $$2MeSH$$aBortezomib: therapeutic use 000153279 650_2 $$2MeSH$$aClinical Trials, Phase II as Topic 000153279 650_2 $$2MeSH$$aDexamethasone 000153279 650_2 $$2MeSH$$aDouble-Blind Method 000153279 650_2 $$2MeSH$$aDrug Therapy, Combination 000153279 650_2 $$2MeSH$$aEncephalitis: drug therapy 000153279 650_2 $$2MeSH$$aEncephalitis: immunology 000153279 650_2 $$2MeSH$$aGermany 000153279 650_2 $$2MeSH$$aGlasgow Coma Scale 000153279 650_2 $$2MeSH$$aHashimoto Disease: drug therapy 000153279 650_2 $$2MeSH$$aHashimoto Disease: immunology 000153279 650_2 $$2MeSH$$aHumans 000153279 650_2 $$2MeSH$$aImmunotherapy 000153279 650_2 $$2MeSH$$aMulticenter Studies as Topic 000153279 650_2 $$2MeSH$$aProspective Studies 000153279 650_2 $$2MeSH$$aProteasome Inhibitors: adverse effects 000153279 650_2 $$2MeSH$$aProteasome Inhibitors: therapeutic use 000153279 650_2 $$2MeSH$$aRandomized Controlled Trials as Topic 000153279 650_2 $$2MeSH$$aTime Factors 000153279 650_2 $$2MeSH$$aTreatment Outcome 000153279 650_2 $$2MeSH$$aTrimethoprim, Sulfamethoxazole Drug Combination 000153279 7001_ $$aChung, Ha-Yeun$$b1 000153279 7001_ $$aPlatzer, Stephanie$$b2 000153279 7001_ $$aLehmann, Thomas$$b3 000153279 7001_ $$0P:(DE-2719)2810931$$aPrüss, Harald$$b4$$udzne 000153279 7001_ $$aLeypoldt, Frank$$b5 000153279 7001_ $$aGünther, Albrecht$$b6 000153279 7001_ $$aScherag, André$$b7 000153279 7001_ $$00000-0002-9859-581X$$aGeis, Christian$$b8$$eCorresponding author 000153279 773__ $$0PERI:(DE-600)2040523-6$$a10.1186/s13063-020-04516-7$$gVol. 21, no. 1, p. 625$$n1$$p625$$tTrials$$v21$$x1745-6215$$y2020 000153279 8564_ $$uhttps://link.springer.com/article/10.1186/s13063-020-04516-7 000153279 8564_ $$uhttps://pub.dzne.de/record/153279/files/DZNE-2020-01276.pdf$$yOpenAccess 000153279 8564_ $$uhttps://pub.dzne.de/record/153279/files/DZNE-2020-01276.pdf?subformat=pdfa$$xpdfa$$yOpenAccess 000153279 909CO $$ooai:pub.dzne.de:153279$$popenaire$$popen_access$$pVDB$$pdriver$$pdnbdelivery 000153279 9101_ $$0I:(DE-588)1065079516$$6P:(DE-2719)2810931$$aDeutsches Zentrum für Neurodegenerative Erkrankungen$$b4$$kDZNE 000153279 9131_ $$0G:(DE-HGF)POF3-342$$1G:(DE-HGF)POF3-340$$2G:(DE-HGF)POF3-300$$3G:(DE-HGF)POF3$$4G:(DE-HGF)POF$$aDE-HGF$$bGesundheit$$lErkrankungen des Nervensystems$$vDisease Mechanisms and Model Systems$$x0 000153279 9141_ $$y2020 000153279 915__ $$0StatID:(DE-HGF)0200$$2StatID$$aDBCoverage$$bSCOPUS$$d2022-11-25 000153279 915__ $$0LIC:(DE-HGF)CCBYNV$$2V:(DE-HGF)$$aCreative Commons Attribution CC BY (No Version)$$bDOAJ$$d2020-08-29 000153279 915__ $$0StatID:(DE-HGF)0600$$2StatID$$aDBCoverage$$bEbsco Academic Search$$d2022-11-25 000153279 915__ $$0StatID:(DE-HGF)0100$$2StatID$$aJCR$$bTRIALS : 2021$$d2022-11-25 000153279 915__ $$0StatID:(DE-HGF)0501$$2StatID$$aDBCoverage$$bDOAJ Seal$$d2018-09-24T10:24:34Z 000153279 915__ $$0StatID:(DE-HGF)0113$$2StatID$$aWoS$$bScience Citation Index Expanded$$d2020-08-29 000153279 915__ $$0StatID:(DE-HGF)0500$$2StatID$$aDBCoverage$$bDOAJ$$d2018-09-24T10:24:34Z 000153279 915__ $$0StatID:(DE-HGF)0300$$2StatID$$aDBCoverage$$bMedline$$d2022-11-25 000153279 915__ $$0StatID:(DE-HGF)0150$$2StatID$$aDBCoverage$$bWeb of Science Core Collection$$d2022-11-25 000153279 915__ $$0StatID:(DE-HGF)9900$$2StatID$$aIF < 5$$d2022-11-25 000153279 915__ $$0StatID:(DE-HGF)0510$$2StatID$$aOpenAccess 000153279 915__ $$0StatID:(DE-HGF)0030$$2StatID$$aPeer Review$$bASC$$d2022-11-25 000153279 915__ $$0StatID:(DE-HGF)0561$$2StatID$$aArticle Processing Charges$$d2020-08-29 000153279 915__ $$0StatID:(DE-HGF)0160$$2StatID$$aDBCoverage$$bEssential Science Indicators$$d2020-08-29 000153279 915__ $$0StatID:(DE-HGF)1110$$2StatID$$aDBCoverage$$bCurrent Contents - Clinical Medicine$$d2022-11-25 000153279 915__ $$0StatID:(DE-HGF)0199$$2StatID$$aDBCoverage$$bClarivate Analytics Master Journal List$$d2022-11-25 000153279 915__ $$0StatID:(DE-HGF)0700$$2StatID$$aFees$$d2020-08-29 000153279 9201_ $$0I:(DE-2719)1810003$$kAG Prüß$$lAutoimmune Encephalopathies$$x0 000153279 980__ $$ajournal 000153279 980__ $$aVDB 000153279 980__ $$aUNRESTRICTED 000153279 980__ $$aI:(DE-2719)1810003 000153279 9801_ $$aFullTexts