{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,2,26]],"date-time":"2026-02-26T15:21:53Z","timestamp":1772119313715,"version":"3.50.1"},"reference-count":10,"publisher":"Springer Science and Business Media LLC","issue":"7","license":[{"start":{"date-parts":[[2024,5,24]],"date-time":"2024-05-24T00:00:00Z","timestamp":1716508800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2024,5,24]],"date-time":"2024-05-24T00:00:00Z","timestamp":1716508800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/100017804","name":"City of Hope","doi-asserted-by":"crossref","id":[{"id":"10.13039\/100017804","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Ann Hematol"],"published-print":{"date-parts":[[2024,7]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Blinatumomab as a single agent has demonstrated superiority over salvage chemotherapy in patients with relapsed and refractory B-cell acute lymphoblastic leukemia (B-ALL), with manageable safety and efficacy. Though known to have anticipated drug toxicities including cytokine release syndrome (CRS) and neurotoxicity, there is only one prior report of macrophage activating syndrome (MAS) due to blinatumomab. Case Presentation: We report the first case of blinatumomab-induced MAS in an adult. The patient presented with fever, cough, and weakness on the second cycle of blinatumomab. Complete blood count was notable for severe leukopenia, with comprehensive metabolic panel notable for elevated alkaline phosphatase, AST, ALT, LDH, and hyperferritinemia consistent with MAS. The patient was already in MRD-negative remission at presentation with MAS. She responded rapidly to withholding the drug and administration of both tocilizumab and dexamethasone. She was able to restart therapy with blinatumomab dosed at 9 mcg\/day with no recurrence of symptoms. Though MAS is not an expected association with blinatumomab, the risk for CRS is. Secondary MAS in this case likely shares a mechanism with other hyperinflammatory conditions. Management includes holding the offending agent, like blinatumomab, and administering tocilizumab and dexamethasone. Future research will be needed to predict which patients are at highest risk to develop MAS after similar T-cell therapies.<\/jats:p>","DOI":"10.1007\/s00277-024-05795-9","type":"journal-article","created":{"date-parts":[[2024,5,24]],"date-time":"2024-05-24T15:01:26Z","timestamp":1716562886000},"page":"2541-2543","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":1,"title":["Blinatumomab-induced macrophage activating syndrome (MAS) in adult with B-cell acute lymphoblastic leukemia (B-ALL)"],"prefix":"10.1007","volume":"103","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-5176-070X","authenticated-orcid":false,"given":"Adam","family":"Braun","sequence":"first","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Salman","family":"Otoukesh","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Jose","family":"Tinajero","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Guido","family":"Marcucci","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ibrahim","family":"Aldoss","sequence":"additional","affiliation":[],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"297","published-online":{"date-parts":[[2024,5,24]]},"reference":[{"issue":"9","key":"5795_CR1","doi-asserted-by":"publisher","first-page":"836","DOI":"10.1056\/NEJMoa1609783","volume":"376","author":"H Kantarjian","year":"2017","unstructured":"Kantarjian H, Stein A, G\u00f6kbuget N, Fielding AK, Schuh AC, Ribera JM et al (2017) Blinatumomab versus Chemotherapy for Advanced Acute Lymphoblastic Leukemia. N Engl J Med 376(9):836\u2013847. https:\/\/doi.org\/10.1056\/NEJMoa1609783. PMID: 28249141; PMCID: PMC5881572","journal-title":"N Engl J Med"},{"key":"5795_CR2","doi-asserted-by":"publisher","unstructured":"Shah BD, Ghobadi A, Oluwole OO, Logan AC, Boissel N, Cassaday RD et al (2021) KTE-X19 for relapsed or refractory adult B-cell acute lymphoblastic leukaemia: phase 2 results of the single-arm, open-label, multicentre ZUMA-3 study. Lancet. 398(10299):491\u2013502. https:\/\/doi.org\/10.1016\/S0140-6736(21)01222-8. Epub 2021 Jun 4. PMID: 34097852","DOI":"10.1016\/S0140-6736(21)01222-8"},{"key":"5795_CR3","doi-asserted-by":"publisher","first-page":"1080","DOI":"10.1038\/s41417-021-00418-1","volume":"29","author":"M Sheykhhasan","year":"2022","unstructured":"Sheykhhasan M, Manoochehri H, Dama P (2022) Use of CAR T-cell for acute lymphoblastic leukemia (ALL) treatment: a review study. Cancer Gene Ther 29:1080\u20131096. https:\/\/doi.org\/10.1038\/s41417-021-00418-1","journal-title":"Cancer Gene Ther"},{"issue":"26","key":"5795_CR4","doi-asserted-by":"publisher","first-page":"5154","DOI":"10.1182\/blood-2013-02-485623","volume":"121","author":"DT Teachey","year":"2013","unstructured":"Teachey DT, Rheingold SR, Maude SL, Zugmaier G, Barrett DM, Seif AE et al (2013) Cytokine release syndrome after blinatumomab treatment related to abnormal macrophage activation and ameliorated with cytokine-directed therapy. Blood 121(26):5154\u20135157. https:\/\/doi.org\/10.1182\/blood-2013-02-485623. Epub 2013 May 15. PMID: 23678006; PMCID: PMC4123427","journal-title":"Blood"},{"issue":"Suppl","key":"5795_CR5","doi-asserted-by":"publisher","first-page":"e68516d0","DOI":"10.1097\/01.HS9.0000967352.68516.d0","volume":"7","author":"E Jabbour","year":"2023","unstructured":"Jabbour E, Kantarjian H, Aldoss I, Montesinos P, Leonard J, Gomez D et al (2023) S110: PHALLCON: A PHASE 3 STUDY COMPARING PONATINIB VERSUS IMATINIB IN NEWLY DIAGNOSED PH\u2009+\u2009ALL. Hemasphere 7(Suppl):e68516d0. https:\/\/doi.org\/10.1097\/01.HS9.0000967352.68516.d0. PMCID: PMC10428451","journal-title":"Hemasphere"},{"issue":"1","key":"5795_CR6","doi-asserted-by":"publisher","first-page":"e24","DOI":"10.1016\/S2352-3026(22)00319-2","volume":"10","author":"E Jabbour","year":"2023","unstructured":"Jabbour E, Short NJ, Jain N, Huang X, Montalban-Bravo G, Banerjee P et al (2023) Ponatinib and Blinatumomab for Philadelphia chromosome-positive acute lymphoblastic leukaemia: a US, single-centre, single-arm, phase 2 trial. Lancet Haematol 10(1):e24\u2013e34. https:\/\/doi.org\/10.1016\/S2352-3026(22)00319-2. Epub 2022 Nov 16. PMID: 36402146","journal-title":"Lancet Haematol"},{"issue":"10","key":"5795_CR7","doi-asserted-by":"publisher","first-page":"1271","DOI":"10.1136\/ard-2023-224123","volume":"82","author":"B Shakoory","year":"2023","unstructured":"Shakoory B, Geerlinks A, Wilejto M, Kernan K, Hines M, Romano M et al (2023) HLH\/MAS task force. The 2022 EULAR\/ACR points to consider at the early stages of diagnosis and management of suspected haemophagocytic lymphohistiocytosis\/macrophage activation syndrome (HLH\/MAS). Ann Rheum Dis 82(10):1271\u20131285. https:\/\/doi.org\/10.1136\/ard-2023-224123. Epub 2023 Jul 24. PMID: 37487610","journal-title":"Ann Rheum Dis"},{"key":"5795_CR8","doi-asserted-by":"publisher","unstructured":"Fardet L, Galicier L, Lambotte O, Marzac C, Aumont C, Chahwan D et al (2014) Development and validation of the HScore, a score for the diagnosis of reactive hemophagocytic syndrome. Arthritis Rheumatol. ;66(9):2613-20. https:\/\/doi.org\/10.1002\/art.38690. PMID: 24782338","DOI":"10.1002\/art.38690"},{"key":"5795_CR9","doi-asserted-by":"publisher","first-page":"745320","DOI":"10.3389\/fimmu.2021.745320","volume":"12","author":"PF Caimi","year":"2021","unstructured":"Caimi PF, Pacheco Sanchez G, Sharma A, Otegbeye F, Ahmed N, Rojas P et al (2021) Prophylactic Tocilizumab Prior to Anti-CD19 CAR-T cell therapy for Non-hodgkin Lymphoma. 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It does not meet the Federal Policy for the Protection of Human Subjects definition of Research. This is for educational purposes.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethical approval"}},{"value":"The patient verbally consented to deidentified material being shared in the form of this case report.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent to publish"}},{"value":"The authors report there are no competing interests to declare.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Competing of interest"}},{"value":"No material is being reproduced.","order":5,"name":"Ethics","group":{"name":"EthicsHeading","label":"Permission to reproduce material from other sources"}},{"value":"Not applicable.","order":6,"name":"Ethics","group":{"name":"EthicsHeading","label":"Clinical trial registration"}}]}}