{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,8]],"date-time":"2026-10-08T16:05:27Z","timestamp":1791475527649,"version":"4.3.4"},"reference-count":18,"publisher":"Springer Science and Business Media LLC","issue":"6","license":[{"start":{"date-parts":[[2024,3,25]],"date-time":"2024-03-25T00:00:00Z","timestamp":1711324800000},"content-version":"tdm","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"},{"start":{"date-parts":[[2024,3,25]],"date-time":"2024-03-25T00:00:00Z","timestamp":1711324800000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/creativecommons.org\/licenses\/by\/4.0"}],"content-domain":{"domain":["link.springer.com"],"crossmark-restriction":false},"short-container-title":["CEN Case Rep"],"abstract":"<jats:title>Abstract<\/jats:title><jats:p>Recombinant human erythropoietin (rHuEPO) is commonly used to treat anemia associated with chronic kidney disease (CKD). EPO-induced Pure Red Cell Aplasia (PRCA) is a rare condition of profound anemia with EPO treatment. Upon finding the development of EPO-induced PRCA, the treatment requires immediate withdrawal of EPO therapy and initiate new treatments with immunosuppression or renal transplantation. Anti-EPO antibody assay is not always positive in EPO-induced PRCA. Here, we report a case on the sudden development of PRCA in a hemodialysis patient receiving EPO and how we treated the condition successfully with cyclosporine and subsequently maintained the hemoglobin with Roxadustat, a hypoxia-inducible factor prolyl hydroxylase inhibitor (HIF-PHI). Even though the anti-EPO antibody was negative by Enzyme Linked Immunosorbent Assay (ELISA) in our case, the clinical course, the markedly reduced reticulocyte count\u2009&lt;\u200910,000\/\u03bcL, the bone marrow (BM) biopsy revealing reduced erythroblasts, and its subsequent response to cyclosporine, were similar to EPO-induced PRCA. The clinical picture of EPO-induced PRCA, the limitation of the EPO-neutralizing antibody (Ab) assay, and treatment strategies were discussed.<\/jats:p>","DOI":"10.1007\/s13730-024-00865-3","type":"journal-article","created":{"date-parts":[[2024,3,25]],"date-time":"2024-03-25T18:02:15Z","timestamp":1711389735000},"page":"445-449","update-policy":"https:\/\/doi.org\/10.1007\/springer_crossmark_policy","source":"Crossref","is-referenced-by-count":6,"title":["Positive response of a hemodialysis patient with pure red cell aplasia on recombinant human erythropoietin therapy to cyclosporine and Roxadustat"],"prefix":"10.1007","volume":"13","author":[{"given":"Xuejuan","family":"Ma","sequence":"first","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Pearl","family":"Pai","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Wenjuan","family":"Zhu","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Xiaowei","family":"Chen","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Liwen","family":"Cui","sequence":"additional","affiliation":[],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"297","published-online":{"date-parts":[[2024,3,25]]},"reference":[{"issue":"8","key":"865_CR1","doi-asserted-by":"publisher","first-page":"1754","DOI":"10.1111\/j.1537-2995.2008.01749.x","volume":"48","author":"JM McKoy","year":"2008","unstructured":"McKoy JM, Stonecash RE, Cournoyer D, Rossert J, Nissenson AR, Raisch DW, Casadevall N, Bennett CL. Epoetin-associated pure red cell aplasia: past, present, and future considerations. Transfusion. 2008;48(8):1754\u201362. https:\/\/doi.org\/10.1111\/j.1537-2995.2008.01749.x.","journal-title":"Transfusion"},{"issue":"10","key":"865_CR2","doi-asserted-by":"publisher","first-page":"641","DOI":"10.1111\/j.1537-2995.2008.01749.x","volume":"15","author":"J Sch\u00f6del","year":"2019","unstructured":"Sch\u00f6del J, Ratcliffe PJ. Mechanisms of hypoxia signalling: new implications for nephrology. Nat Rev Nephrol. 2019;15(10):641\u201359. https:\/\/doi.org\/10.1111\/j.1537-2995.2008.01749.x.","journal-title":"Nat Rev Nephrol"},{"issue":"3","key":"865_CR3","doi-asserted-by":"publisher","first-page":"S67","DOI":"10.1111\/j.1537-2995.2008.01749.x","volume":"16","author":"N Casadevall","year":"2005","unstructured":"Casadevall N, Eckardt K-U, Rossert JAACA. Epoetin-induced autoimmune pure red cell aplasia. J Am Soc Nephrol. 2005;16(3):S67\u20139. https:\/\/doi.org\/10.1111\/j.1537-2995.2008.01749.x.","journal-title":"J Am Soc Nephrol"},{"issue":"Suppl 4","key":"865_CR4","doi-asserted-by":"publisher","first-page":"iv3","DOI":"10.1111\/j.1537-2995.2008.01749.x","volume":"20","author":"N Casadevall","year":"2005","unstructured":"Casadevall N. What is antibody-mediated pure red cell aplasia (PRCA)? Nephrol Dial Transplant. 2005;20(Suppl 4):iv3-8. https:\/\/doi.org\/10.1111\/j.1537-2995.2008.01749.x.","journal-title":"Nephrol Dial Transplant"},{"issue":"23","key":"865_CR5","doi-asserted-by":"publisher","first-page":"6042","DOI":"10.12998\/wjcc.v8.i23.6042","volume":"8","author":"W-H Yu","year":"2020","unstructured":"Yu W-H, Li X-J, Yuan F. Roxadustat for treatment of erythropoietin-hyporesponsive anemia in a hemodialysis patient: a case report. World J Clin Cases. 2020;8(23):6042\u20139.","journal-title":"World J Clin Cases"},{"issue":"2","key":"865_CR6","doi-asserted-by":"publisher","first-page":"398","DOI":"10.1111\/j.1537-2995.2008.01749.x","volume":"15","author":"J Rossert","year":"2004","unstructured":"Rossert J, Casadevall N, Eckardt K-U. Anti-erythropoietin antibodies and pure red cell aplasia. J Am Soc Nephrol. 2004;15(2):398\u2013406. https:\/\/doi.org\/10.1111\/j.1537-2995.2008.01749.x.","journal-title":"J Am Soc Nephrol"},{"issue":"1","key":"865_CR7","doi-asserted-by":"publisher","first-page":"78","DOI":"10.1007\/s13730-015-0196-8","volume":"5","author":"K Hirai","year":"2016","unstructured":"Hirai K, Ookawara S, Miyazawa H, Ito K, Ueda Y, Kaku Y, Hoshino T, Kimura S, Yoshida I, Kakuta S, Morishita Y, Tabei K. Successful treatment of a hemodialyzed patient with pure red cell aplasia associated with epoetin beta pegol therapy with cyclosporine. CEN Case Rep. 2016;5(1):78\u201382. https:\/\/doi.org\/10.1007\/s13730-015-0196-8.","journal-title":"CEN Case Rep"},{"issue":"10","key":"865_CR8","doi-asserted-by":"publisher","first-page":"550","DOI":"10.1046\/j.1442-2042.2003.00672.x","volume":"10","author":"Y Tanuma","year":"2003","unstructured":"Tanuma Y, Yokoo A, Takashima H. Acquired pure red cell aplasia in a hemodialyzed patient. Int J Urol. 2003;10(10):550\u20133. https:\/\/doi.org\/10.1046\/j.1442-2042.2003.00672.x.","journal-title":"Int J Urol"},{"issue":"suppl4","key":"865_CR9","doi-asserted-by":"publisher","first-page":"16","DOI":"10.1093\/ndt\/gfh1086","volume":"20","author":"R Thorpe","year":"2005","unstructured":"Thorpe R, Swanson SJ. Assays for detecting and diagnosing antibody-mediated pure red cell aplasia (PRCA): an assessment of available procedures. Nephrol Dial Transplant. 2005;20(suppl4):16\u201322. https:\/\/doi.org\/10.1093\/ndt\/gfh1086.","journal-title":"Nephrol Dial Transplant"},{"issue":"7","key":"865_CR10","doi-asserted-by":"publisher","first-page":"469","DOI":"10.1056\/NEJMoa011931","volume":"346","author":"N Casadevall","year":"2002","unstructured":"Casadevall N, Nataf J, Viron B, Kolta A, Kiladjian J-J, Martin-Dupont P, Michaud P, Papo T, Ugo V, Teyssandier I, Varet B, Mayeux P. Pure red-cell aplasia and antierythropoietin antibodies in patients treated with recombinant erythropoietin. N Engl J Med. 2002;346(7):469\u201375. https:\/\/doi.org\/10.1056\/NEJMoa011931.","journal-title":"N Engl J Med"},{"issue":"2","key":"865_CR11","doi-asserted-by":"publisher","first-page":"591","DOI":"10.1007\/s00277-020-04269-y","volume":"100","author":"K Wan","year":"2021","unstructured":"Wan K, Yin Y, Luo Z, Cheng J. Remarkable response to Roxadustat in a case of anti-erythropoietin antibody-mediated pure red cell aplasia. Ann Hematol. 2021;100(2):591\u20133. https:\/\/doi.org\/10.1007\/s00277-020-04269-y.","journal-title":"Ann Hematol"},{"issue":"21","key":"865_CR12","doi-asserted-by":"publisher","first-page":"2504","DOI":"10.1182\/blood-2016-05-717140","volume":"128","author":"RT Means","year":"2016","unstructured":"Means RT. Pure red cell aplasia. Blood. 2016;128(21):2504\u20139. https:\/\/doi.org\/10.1182\/blood-2016-05-717140.","journal-title":"Blood"},{"issue":"9423","key":"865_CR13","doi-asserted-by":"publisher","first-page":"1768","DOI":"10.1016\/S0140-6736(04)16302-2","volume":"363","author":"D Verhelst","year":"2004","unstructured":"Verhelst D, Rossert J, Casadevall N, Kr\u00fcger A, Eckardt K-U, Macdougall IC. Treatment of erythropoietin-induced pure red cell aplasia: a retrospective study. The Lancet. 2004;363(9423):1768\u201371. https:\/\/doi.org\/10.1016\/S0140-6736(04)16302-2.","journal-title":"The Lancet"},{"issue":"11","key":"865_CR14","doi-asserted-by":"publisher","first-page":"1001","DOI":"10.1056\/NEJMoa1813599","volume":"381","author":"N Chen","year":"2019","unstructured":"Chen N, Hao C, Peng X, Lin H, Yin A, Hao L, Tao Y, Liang X, Liu Z, Xing C, Chen J, Luo L, Zuo L, Liao Y, Liu B-C, Leong R, Wang C, Liu C, Neff T, Szczech L, Yu K-HP. Roxadustat for anemia in patients with kidney disease not receiving dialysis. N Engl J Med. 2019;381(11):1001\u201310. https:\/\/doi.org\/10.1056\/NEJMoa1813599.","journal-title":"N Engl J Med"},{"issue":"11","key":"865_CR15","doi-asserted-by":"publisher","first-page":"1011","DOI":"10.1056\/NEJMoa1901713","volume":"381","author":"N Chen","year":"2019","unstructured":"Chen N, Hao C, Liu B-C, Lin H, Wang C, Xing C, Liang X, Jiang G, Liu Z, Li X, Zuo L, Luo L, Wang J, Zhao M-H, Liu Z, Cai G-Y, Hao L, Leong R, Wang C, Liu C, Neff T, Szczech L, Yu K-HP. Roxadustat treatment for anemia in patients undergoing long-term dialysis. N Engl J Med. 2019;381(11):1011\u201322. https:\/\/doi.org\/10.1056\/NEJMoa1901713.","journal-title":"N Engl J Med"},{"key":"865_CR16","doi-asserted-by":"publisher","DOI":"10.1111\/ijlh.13325","author":"Y Wu","year":"2020","unstructured":"Wu Y, Cai X, Ni J, Lin X. Resolution of epoetin-induced pure red cell aplasia, successful re-challenge with Roxadustat. Int J Lab Hematology. 2020. https:\/\/doi.org\/10.1111\/ijlh.13325.","journal-title":"Int J Lab Hematology"},{"issue":"11","key":"865_CR17","doi-asserted-by":"publisher","first-page":"2444","DOI":"10.1093\/ckj\/sfab134","volume":"14","author":"B Xu","year":"2021","unstructured":"Xu B, Liu S, Li Y, Zhao L, Song X, Chen T. Roxadustat in the treatment of a hemodialysis patient with anti-erythropoietin antibody-mediated pure red cell aplasia. Clin Kidney J. 2021;14(11):2444\u20135. https:\/\/doi.org\/10.1093\/ckj\/sfab134.","journal-title":"Clin Kidney J"},{"issue":"11","key":"865_CR18","doi-asserted-by":"publisher","first-page":"2548","DOI":"10.1093\/ndt\/gfi018","volume":"20","author":"J Andrade","year":"2005","unstructured":"Andrade J, Taylor PA, Love JM, Levin A. Successful reintroduction of a different erythropoiesis-stimulating agent after pure red cell aplasia: relapse after successful therapy with prednisone. Nephrol Dial Transplant. 2005;20(11):2548\u201351. https:\/\/doi.org\/10.1093\/ndt\/gfi018.","journal-title":"Nephrol Dial Transplant"}],"container-title":["CEN Case Reports"],"original-title":[],"language":"en","link":[{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s13730-024-00865-3.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/article\/10.1007\/s13730-024-00865-3\/fulltext.html","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/link.springer.com\/content\/pdf\/10.1007\/s13730-024-00865-3.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2024,12,1]],"date-time":"2024-12-01T02:04:54Z","timestamp":1733018694000},"score":1,"resource":{"primary":{"URL":"https:\/\/link.springer.com\/10.1007\/s13730-024-00865-3"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2024,3,25]]},"references-count":18,"journal-issue":{"issue":"6","published-online":{"date-parts":[[2024,12]]}},"alternative-id":["865"],"URL":"https:\/\/doi.org\/10.1007\/s13730-024-00865-3","relation":{},"ISSN":["2192-4449"],"issn-type":[{"value":"2192-4449","type":"electronic"}],"subject":[],"published":{"date-parts":[[2024,3,25]]},"assertion":[{"value":"15 December 2023","order":1,"name":"received","label":"Received","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"28 February 2024","order":2,"name":"accepted","label":"Accepted","group":{"name":"ArticleHistory","label":"Article History"}},{"value":"25 March 2024","order":3,"name":"first_online","label":"First Online","group":{"name":"ArticleHistory","label":"Article History"}},{"order":1,"name":"Ethics","group":{"name":"EthicsHeading","label":"Declarations"}},{"value":"The authors declared no conflict of interest.","order":2,"name":"Ethics","group":{"name":"EthicsHeading","label":"Conflict of interest"}},{"value":"The Medical Ethics Committee of Hong Kong University-Shenzhen Hospital has approved the publication of this case report (IRB approval number \u4f26[2023]271) which meets the 1964 Helsinki Declaration and its later amendments or comparable ethical standards.","order":3,"name":"Ethics","group":{"name":"EthicsHeading","label":"Ethical approval"}},{"value":"The patient has given consent to the publication of this case report.","order":4,"name":"Ethics","group":{"name":"EthicsHeading","label":"Consent for publication"}}]}}