{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,9,30]],"date-time":"2026-09-30T17:03:11Z","timestamp":1790787791658,"version":"4.1.0"},"reference-count":35,"publisher":"Oxford University Press (OUP)","issue":"4","license":[{"start":{"date-parts":[[2023,11,8]],"date-time":"2023-11-08T00:00:00Z","timestamp":1699401600000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0\/"}],"funder":[{"DOI":"10.13039\/501100010724","name":"Health Systems Research Institute","doi-asserted-by":"publisher","award":["61-049"],"award-info":[{"award-number":["61-049"]}],"id":[{"id":"10.13039\/501100010724","id-type":"DOI","asserted-by":"publisher"}]}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2024,4,10]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec>\n                  <jats:title>Background<\/jats:title>\n                  <jats:p>Currently, there is no effective treatment for adult-onset immunodeficiency (AOID) syndrome with anti-interferon-gamma autoantibodies (anti-IFN-\u03b3-auto-Abs). This study aimed to investigate the effectiveness of bortezomib (BTZ) for decreasing anti-IFN-\u03b3-auto-Abs.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Methods<\/jats:title>\n                  <jats:p>A pre- and post-intervention study was conducted from February 2017 through June 2019 at Siriraj Hospital (Bangkok, Thailand). Five patients were invited to receive once-weekly BTZ (1.3\u2005mg\/m2 body surface area) subcutaneously for 8 weeks followed by oral cyclophosphamide (1\u2005mg\/kg\/d) for 4 months. The primary outcomes were the difference in antibody level at 8 and 48 weeks compared with baseline and the incidence of serious adverse events (AEs). The secondary outcome was the occurrence of opportunistic infections (OIs) during the 72 weeks after starting BTZ.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Results<\/jats:title>\n                  <jats:p>The median patient age was 46 years (range, 34\u201353). All patients had 3\u20135 OIs prior to enrollment. All patients were receiving antimycobacterial agents for treatment of nontuberculous mycobacterial infection at enrollment. There was no significant difference in the mean optical density of auto-Abs at 8 weeks (3.73 \u00b1 0.72) or 48 weeks (3.74 \u00b1 0.53) compared with baseline (3.84 \u00b1 0.49; P = .336 and P = .555, respectively). However, after serum dilution, the antibody titer nonsignificantly decreased 8\u201316 weeks after BTZ initiation (P = .345). Ten OIs were observed 24\u201372 weeks after BTZ initiation.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Conclusions<\/jats:title>\n                  <jats:p>Treatment with BTZ followed by cyclophosphamide yielded no significant decrease in antibody titer levels, and 10 OIs were observed during 24\u201372 weeks of BTZ treatment. No serious AEs were observed. Combining rituximab with BTZ is likely necessary to prevent generation of new autoantibody-producing plasma cells.<\/jats:p>\n                  <jats:p>Clinical Trials Registration. NCT03103555.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1093\/cid\/ciad676","type":"journal-article","created":{"date-parts":[[2023,11,10]],"date-time":"2023-11-10T11:33:40Z","timestamp":1699616020000},"page":"1033-1042","source":"Crossref","is-referenced-by-count":15,"title":["Efficacy of Bortezomib for Treating Anti-Interferon-Gamma Autoantibody-Associated Adult-Onset Immunodeficiency Syndrome"],"prefix":"10.1093","volume":"78","author":[{"ORCID":"https:\/\/orcid.org\/0000-0001-5723-4877","authenticated-orcid":false,"given":"Nasikarn","family":"Angkasekwinai","sequence":"first","affiliation":[{"name":"Division of Infectious Diseases and Tropical Medicine, Department of Medicine, Faculty of Medicine Siriraj Hospital, Mahidol University , Bangkok , Thailand"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yupin","family":"Suputtamongkol","sequence":"additional","affiliation":[{"name":"Division of Infectious Diseases and Tropical Medicine, Department of Medicine, 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