{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,8,19]],"date-time":"2026-08-19T19:36:36Z","timestamp":1787168196249,"version":"build-2736575974"},"reference-count":14,"publisher":"Springer Science and Business Media LLC","issue":"1","license":[{"start":{"date-parts":[[2023,2,3]],"date-time":"2023-02-03T00:00:00Z","timestamp":1675382400000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2023,2,3]],"date-time":"2023-02-03T00:00:00Z","timestamp":1675382400000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"funder":[{"DOI":"10.13039\/501100000646","name":"Japan Society for the Promotion of Science London","doi-asserted-by":"publisher","award":["21H04805"],"award-info":[{"award-number":["21H04805"]}],"id":[{"id":"10.13039\/501100000646","id-type":"DOI","asserted-by":"publisher"}]},{"DOI":"10.13039\/501100004721","name":"The University of Tokyo","doi-asserted-by":"crossref","id":[{"id":"10.13039\/501100004721","id-type":"DOI","asserted-by":"crossref"}]}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["Int J Hematol"],"published-print":{"date-parts":[[2023,7]]},"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:p>Plasma cell leukemia is a rare yet aggressive form of multiple myeloma characterized by high levels of plasma cells circulating in the peripheral blood. We recently experienced a case of plasma cell leukemia that had been in stringent complete remission for nine years after autologous stem cell transplantations with subsequent courses of lenalidomide maintenance therapy, and then relapsed as an extramedullary plasmacytoma in the central nervous system. Assessment of the bone marrow did not prove proliferation of plasma cells at relapse, but imbalanced elevation of serum levels of free light chains was observed without changes in other clinical biomarkers including immunoglobulin levels. Salvage chemotherapy with isatuximab, pomalidomide, and dexamethasone (IsaPD) was promptly initiated. After two courses of IsaPD, significant remission was achieved and the neuronal symptoms completely resolved. When excessive serum levels of clonotypic free light chains are noted, their significance should be carefully assessed even when plasma cell propagation in the bone marrow is not observed. In such cases, hematologists should search for extramedullary proliferation of plasma cells, including in the immune-privileged central nervous system.<\/jats:p>","DOI":"10.1007\/s12185-023-03545-7","type":"journal-article","created":{"date-parts":[[2023,2,3]],"date-time":"2023-02-03T07:03:03Z","timestamp":1675407783000},"page":"135-140","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":4,"title":["Isolated relapse of plasma cell leukemia in the central nervous systems: a case report and literature review"],"prefix":"10.1007","volume":"118","author":[{"given":"Takafumi","family":"Obo","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ken","family":"Morita","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yutaro","family":"Sumida","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kumi","family":"Nakazaki-Watadani","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Masako","family":"Ikemura","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Koichiro","family":"Yasaka","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Osamu","family":"Abe","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hirokazu","family":"Takami","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shunsaku","family":"Takayanagi","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Shota","family":"Tanaka","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Hiroaki","family":"Maki","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Yosuke","family":"Masamoto","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Akiyoshi","family":"Miwa","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-4034-2422","authenticated-orcid":false,"given":"Mineo","family":"Kurokawa","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2023,2,3]]},"reference":[{"issue":"12","key":"3545_CR1","doi-asserted-by":"publisher","first-page":"192","DOI":"10.1038\/s41408-021-00587-0","volume":"11","author":"C Fernandez de Larrea","year":"2021","unstructured":"Fernandez de Larrea C, Kyle R, Rosinol L, Paiva B, Engelhardt M, Usmani S, et al. Primary plasma cell leukemia: consensus definition by the International Myeloma Working Group according to peripheral blood plasma cell percentage. Blood Cancer J. 2021;11(12):192. https:\/\/doi.org\/10.1038\/s41408-021-00587-0.","journal-title":"Blood Cancer J"},{"key":"3545_CR2","doi-asserted-by":"publisher","first-page":"106687","DOI":"10.1016\/j.leukres.2021.106687","volume":"111","author":"K Gowin","year":"2021","unstructured":"Gowin K, Skerget S, Keats JJ, Mikhael J, Cowan AJ. Plasma cell leukemia: A review of the molecular classification, diagnosis, and evidenced-based treatment. Leuk Res. 2021;111:106687. https:\/\/doi.org\/10.1016\/j.leukres.2021.106687.","journal-title":"Leuk Res"},{"issue":"2","key":"3545_CR3","doi-asserted-by":"publisher","first-page":"23","DOI":"10.1038\/s41408-021-00414-6","volume":"11","author":"SA Tuazon","year":"2021","unstructured":"Tuazon SA, Holmberg LA, Nadeem O, Richardson PG. A clinical perspective on plasma cell leukemia; current status and future directions. Blood Cancer J. 2021;11(2):23. https:\/\/doi.org\/10.1038\/s41408-021-00414-6.","journal-title":"Blood Cancer J"},{"issue":"17","key":"3545_CR4","doi-asserted-by":"publisher","first-page":"2666","DOI":"10.1182\/blood.2021014968","volume":"139","author":"T Cazaubiel","year":"2022","unstructured":"Cazaubiel T, Leleu X, Perrot A, Manier S, Buisson L, Maheo S, et al. Primary plasma cell leukemias displaying t(11;14) have specific genomic, transcriptional, and clinical features. Blood. 2022;139(17):2666\u201372. https:\/\/doi.org\/10.1182\/blood.2021014968.","journal-title":"Blood"},{"issue":"3","key":"3545_CR5","doi-asserted-by":"publisher","first-page":"224","DOI":"10.1007\/s12149-014-0931-9","volume":"29","author":"M Okasaki","year":"2015","unstructured":"Okasaki M, Kubota K, Minamimoto R, Miyata Y, Morooka M, Ito K, et al. Comparison of (11)C-4\u2019-thiothymidine, (11)C-methionine, and (18)F-FDG PET\/CT for the detection of active lesions of multiple myeloma. Ann Nucl Med. 2015;29(3):224\u201332. https:\/\/doi.org\/10.1007\/s12149-014-0931-9.","journal-title":"Ann Nucl Med"},{"issue":"1","key":"3545_CR6","doi-asserted-by":"publisher","first-page":"13","DOI":"10.1046\/j.1365-2141.2001.02962.x","volume":"115","author":"RA Kyle","year":"2001","unstructured":"Kyle RA. Henry Bence Jones\u2013physician, chemist, scientist and biographer: a man for all seasons. Br J Haematol. 2001;115(1):13\u20138. https:\/\/doi.org\/10.1046\/j.1365-2141.2001.02962.x.","journal-title":"Br J Haematol"},{"issue":"5","key":"3545_CR7","doi-asserted-by":"publisher","first-page":"464","DOI":"10.1001\/jama.2022.0003","volume":"327","author":"AJ Cowan","year":"2022","unstructured":"Cowan AJ, Green DJ, Kwok M, Lee S, Coffey DG, Holmberg LA, et al. Diagnosis and management of multiple myeloma: a review. JAMA. 2022;327(5):464\u201377. https:\/\/doi.org\/10.1001\/jama.2022.0003.","journal-title":"JAMA"},{"issue":"8","key":"3545_CR8","doi-asserted-by":"publisher","first-page":"e71754","DOI":"10.1371\/journal.pone.0071754","volume":"8","author":"Z Li","year":"2013","unstructured":"Li Z, Qiu Y, Personett D, Huang P, Edenfield B, Katz J, et al. Pomalidomide shows significant therapeutic activity against CNS lymphoma with a major impact on the tumor microenvironment in murine models. PLoS ONE. 2013;8(8):e71754. https:\/\/doi.org\/10.1371\/journal.pone.0071754.","journal-title":"PLoS ONE"},{"issue":"9","key":"3545_CR9","doi-asserted-by":"publisher","first-page":"1411","DOI":"10.1182\/blood-2014-11-612069","volume":"125","author":"X Leleu","year":"2015","unstructured":"Leleu X, Karlin L, Macro M, Hulin C, Garderet L, Roussel M, et al. Pomalidomide plus low-dose dexamethasone in multiple myeloma with deletion 17p and\/or translocation (4;14): IFM 2010\u201302 trial results. Blood. 2015;125(9):1411\u20137. https:\/\/doi.org\/10.1182\/blood-2014-11-612069.","journal-title":"Blood"},{"issue":"4","key":"3545_CR10","doi-asserted-by":"publisher","first-page":"864","DOI":"10.3109\/10428194.2012.718343","volume":"54","author":"A Mussetti","year":"2013","unstructured":"Mussetti A, Dalto S, Montefusco V. Effective treatment of pomalidomide in central nervous system myelomatosis. Leuk Lymphoma. 2013;54(4):864\u20136. https:\/\/doi.org\/10.3109\/10428194.2012.718343.","journal-title":"Leuk Lymphoma"},{"key":"3545_CR11","doi-asserted-by":"publisher","DOI":"10.3390\/pharmaceutics12010062","author":"M Cavaco","year":"2020","unstructured":"Cavaco M, Gaspar D, Arb Castanho M, Neves V. Antibodies for the treatment of brain metastases, a dream or a reality? Pharmaceutics. 2020. https:\/\/doi.org\/10.3390\/pharmaceutics12010062.","journal-title":"Pharmaceutics"},{"key":"3545_CR12","doi-asserted-by":"publisher","first-page":"273565","DOI":"10.1155\/2015\/273565","volume":"2015","author":"CW Eskelund","year":"2015","unstructured":"Eskelund CW, Andersen NF. Localized relapse of primary plasma cell leukaemia in the central nervous system. Case Rep Hematol. 2015;2015:273565. https:\/\/doi.org\/10.1155\/2015\/273565.","journal-title":"Case Rep Hematol"},{"issue":"2","key":"3545_CR13","doi-asserted-by":"publisher","first-page":"307","DOI":"10.1038\/sj.leu.2401309","volume":"13","author":"X Leleu","year":"1999","unstructured":"Leleu X, Jouet JP, Plantier I, Zandecki M, Lai JL, Mucha D, et al. Isolated neurological relapse following stem cell transplantation in plasma cell leukemia: a report of two cases. Leukemia. 1999;13(2):307\u20139. https:\/\/doi.org\/10.1038\/sj.leu.2401309.","journal-title":"Leukemia"},{"issue":"4","key":"3545_CR14","doi-asserted-by":"publisher","first-page":"893","DOI":"10.1080\/10428190290017060","volume":"43","author":"J Wellwood","year":"2002","unstructured":"Wellwood J, Taylor K, Wright S, Bentley M, Hill G, Phan H, et al. Relapsed plasma cell leukaemia presenting with aggressive extramedullary plasmacytomas in the meninges and testis\u2013a missed opportunity for induction CNS prophylaxis. Leuk Lymphoma. 2002;43(4):893\u20136. https:\/\/doi.org\/10.1080\/10428190290017060.","journal-title":"Leuk Lymphoma"}],"container-title":["International Journal of Hematology"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s12185-023-03545-7.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1007\/s12185-023-03545-7\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s12185-023-03545-7.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2023,6,21]],"date-time":"2023-06-21T13:20:38Z","timestamp":1687353638000},"score":1,"resource":{"primary":{"URL":"https:\/\/link.springer.com\/10.1007\/s12185-023-03545-7"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2023,2,3]]},"references-count":14,"journal-issue":{"issue":"1","published-print":{"date-parts":[[2023,7]]}},"alternative-id":["3545"],"URL":"https:\/\/doi.org\/10.1007\/s12185-023-03545-7","relation":{},"ISSN":["0925-5710","1865-3774"],"issn-type":[{"value":"0925-5710","type":"print"},{"value":"1865-3774","type":"electronic"}],"subject":[],"published":{"date-parts":[[2023,2,3]]},"assertion":[{"value":"5 September 2022","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"17 January 2023","order":2,"name":"revised","label":"Revised","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"17 January 2023","order":3,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"3 February 2023","order":4,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"The authors have no conflicting financial interests.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Conflict of interest"}}]}}