{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,10,4]],"date-time":"2025-10-04T00:38:45Z","timestamp":1759538325296,"version":"build-2065373602"},"reference-count":10,"publisher":"SMC Media","issue":"10","content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"abstract":"<jats:p>Introduction:\u00a0Paroxysmal nocturnal haemoglobinuria (PNH) is a rare bone marrow failure disorder that results in complement-mediated haemolysis (mostly intravascular), thrombosis and bone marrow suppression. Terminal complement inhibitors, such as eculizumab, have improved outcomes in pregnant and non-pregnant individuals. However, the optimal management of PNH during pregnancy has not been fully established. Case description: We describe the case of a 30-year-old Caucasian woman with classical PNH who became pregnant while on eculizumab therapy. Despite complete terminal complement blockade, as confirmed by appropriately suppressed functional C5 levels and elevated C5 antigen levels, the patient experienced recurrent breakthrough haemolysis and transfusion dependence during the first trimester. Her eculizumab dose was escalated beyond the standard recommended doses, which improved haemoglobin levels and reduced transfusion requirements, although lactate dehydrogenase (LDH) levels remained elevated. The patient delivered a healthy infant via elective caesarean section at term without complications. Postpartum, she transitioned to a combination of eculizumab and pegcetacoplan to address ongoing haemolysis, likely due to C3b mediated opsonisation. Conclusion:\u00a0The case illustrates the complexity of managing PNH during pregnancy and highlights the importance of individualised therapy and close monitoring to achieve optimal foetomaternal outcomes. Our experience suggests that despite appropriately suppressed complement levels, pregnant patients with continued transfusion dependence and elevated LDH levels may benefit from higher than standard doses of eculizumab. Given the paucity of data, detailed reporting of such cases is essential to inform treatment strategies for pregnant patients.<\/jats:p>","DOI":"10.12890\/2025_005721","type":"journal-article","created":{"date-parts":[[2025,9,1]],"date-time":"2025-09-01T11:05:02Z","timestamp":1756724702000},"source":"Crossref","is-referenced-by-count":0,"title":["Higher doses of eculizumab may be beneficial in paroxysmal nocturnal haemoglobinuria in pregnancy"],"prefix":"10.12890","volume":"12","author":[{"given":"Harshank Vishnubhai","family":"Patel","sequence":"first","affiliation":[{"name":"Western Michigan University, Homer Stryker M.D. School of Medicine, Kalamazoo, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Steve","family":"Stone","sequence":"first","affiliation":[{"name":"Western Michigan University, Homer Stryker M.D. School of Medicine, Kalamazoo, USA; Bronson Methodist Hospital, Kalamazoo, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Madison C.","family":"Laird","sequence":"first","affiliation":[{"name":"Western Michigan University, Homer Stryker M.D. School of Medicine, Kalamazoo, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Vanita","family":"Motiani","sequence":"first","affiliation":[{"name":"Western Michigan University, Homer Stryker M.D. School of Medicine, Kalamazoo, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Ayema","family":"Haque","sequence":"first","affiliation":[{"name":"Western Michigan University, Homer Stryker M.D. School of Medicine, Kalamazoo, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Hemasri","family":"Tokala","sequence":"first","affiliation":[{"name":"Western Michigan University, Homer Stryker M.D. School of Medicine, Kalamazoo, USA; Ascension Borgess Hospital, Kalamazoo, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Akshay","family":"Amaraneni","sequence":"first","affiliation":[{"name":"Banner Health, University Medical Center, Tucson, USA; University of Arizona College of Medicine, Tucson, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"10397","published-online":{"date-parts":[[2025,9,1]]},"reference":[{"key":"10.12890\/2025_005721_ref1","doi-asserted-by":"crossref","unstructured":"Brodsky RA. How I treat paroxysmal nocturnal hemoglobinuria. Blood 2021;137:1304\u20131309. doi:10.1182\/blood.2019003812","DOI":"10.1182\/blood.2019003812"},{"key":"10.12890\/2025_005721_ref2","doi-asserted-by":"crossref","unstructured":"Hillmen P, Szer J, Weitz I, R\u00f6th A, H\u00f6chsmann B, Panse J, et al. Pegcetacoplan versus eculizumab in paroxysmal nocturnal hemoglobinuria. N Engl J Med 2021;384:1028\u20131037. doi:10.1056\/NEJMoa240003","DOI":"10.1056\/NEJMoa2029073"},{"key":"10.12890\/2025_005721_ref3","doi-asserted-by":"crossref","unstructured":"Manning JE, Ciantar E, Griffin M, Kelly RJ. Paroxysmal nocturnal haemoglobinuria in pregnancy \u2013 a systematic review with meta analysis. Ann Hematol 2025;104:2517\u20132525. doi:10.1007\/s00277-025-06353-7","DOI":"10.1007\/s00277-025-06353-7"},{"key":"10.12890\/2025_005721_ref4","doi-asserted-by":"crossref","unstructured":"Kelly RJ, H\u00f6chsmann B, Szer J, Kulasekararaj A, de Guibert S, R\u00f6th A, et al. Eculizumab in pregnant patients with paroxysmal nocturnal hemoglobinuria. N Engl J Med 2015;373:1032\u20131039. doi:10.1056\/NEJmoa1502950","DOI":"10.1056\/NEJMoa1502950"},{"key":"10.12890\/2025_005721_ref5","doi-asserted-by":"crossref","unstructured":"Alashkar F, Saner FH, Vance C, Schm\u00fccker U, Herich-Terh\u00fcrne D, D\u00fchrsen U, et al. Pregnancy in classical paroxysmal nocturnal hemoglobinuria and aplastic anemia\u2013paroxysmal nocturnal hemoglobinuria: a high-risk constellation. Front Med (Lausanne) 2020;7:543372. doi:10.3389\/fmed.2020.543372","DOI":"10.3389\/fmed.2020.543372"},{"key":"10.12890\/2025_005721_ref6","doi-asserted-by":"crossref","unstructured":"H\u00f6chsmann B, Leopold W, Schneider A, Koerper S, Kalhammer U, Schrezenmeier H. Ravulizumab in pregnant women with paroxysmal nocturnal hemoglobinuria (PNH) \u2013 favourable experience from a retrospective case series. Blood 2024;144:5251. doi:10.1182\/blood-2024-209857","DOI":"10.1182\/blood-2024-209857"},{"key":"10.12890\/2025_005721_ref7","doi-asserted-by":"crossref","unstructured":"McNamara LA, Topaz N, Wang X, Hariri S, Fox L, MacNeil JR. High risk for invasive meningococcal disease among patients receiving eculizumab (Soliris) despite receipt of meningococcal vaccine. MMWR Morb Mortal Wkly Rep 2017;66:734\u2013737. doi:10.15585\/mmwr.mm6627e1","DOI":"10.15585\/mmwr.mm6627e1"},{"key":"10.12890\/2025_005721_ref8","doi-asserted-by":"crossref","unstructured":"Kelly RJ, Nishimori H, Horneff R, Hillmen P, Al-Adhami M, Lallier S, et al. Thrombosis and meningococcal infection rates in pegcetacoplan-treated patients with paroxysmal nocturnal hemoglobinuria in the clinical trial and postmarketing settings. Res Pract Thromb Haemost 2024;8:102416. doi:10.1016\/j.rpth.2024.102416","DOI":"10.1016\/j.rpth.2024.102416"},{"key":"10.12890\/2025_005721_ref9","doi-asserted-by":"crossref","unstructured":"Du W, Mei L. A case report of pegcetacoplan use for a pregnant woman with paroxysmal nocturnal hemoglobinuria. Res Pract Thromb Haemost 2024;8:102435. doi:10.1016\/j.rpth.2024.102435","DOI":"10.1016\/j.rpth.2024.102435"},{"key":"10.12890\/2025_005721_ref10","doi-asserted-by":"crossref","unstructured":"Kim JS, Lee JW, Kim BK, Lee JH, Chung J. The use of the complement inhibitor eculizumab (Soliris\u00ae) for treating Korean patients with paroxysmal nocturnal hemoglobinuria. Korean J Hematol 2010;45:269\u2013274. doi:10.5045\/kjh.2010.45.4.269","DOI":"10.5045\/kjh.2010.45.4.269"}],"container-title":["European Journal of Case Reports in Internal Medicine"],"original-title":[],"link":[{"URL":"https:\/\/www.ejcrim.com\/index.php\/EJCRIM\/article\/download\/5721\/4834","content-type":"text\/html","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/www.ejcrim.com\/index.php\/EJCRIM\/article\/download\/5721\/4829","content-type":"application\/pdf","content-version":"vor","intended-application":"text-mining"},{"URL":"https:\/\/www.ejcrim.com\/index.php\/EJCRIM\/article\/download\/5721\/4829","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2025,10,3]],"date-time":"2025-10-03T13:03:22Z","timestamp":1759496602000},"score":1,"resource":{"primary":{"URL":"https:\/\/www.ejcrim.com\/index.php\/EJCRIM\/article\/view\/5721"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2025,9,1]]},"references-count":10,"journal-issue":{"issue":"10","published-online":{"date-parts":[[2025]]}},"URL":"https:\/\/doi.org\/10.12890\/2025_005721","relation":{"has-manifestation":[{"id-type":"other","id":"1-130-5721-g4834","asserted-by":"subject"},{"id-type":"other","id":"1-130-5721-g4829","asserted-by":"subject"}],"is-part-of":[{"id-type":"other","id":"1-130","asserted-by":"subject"}]},"ISSN":["2284-2594"],"issn-type":[{"type":"electronic","value":"2284-2594"}],"subject":[],"published":{"date-parts":[[2025,9,1]]}}}