{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2025,12,17]],"date-time":"2025-12-17T17:59:56Z","timestamp":1765994396755,"version":"3.44.0"},"reference-count":19,"publisher":"BMJ","content-domain":{"domain":["bmj.com"],"crossmark-restriction":true},"short-container-title":["BMJ Case Reports"],"accepted":{"date-parts":[[2017,4,12]]},"abstract":"<jats:p>We present the case of a 21-month-old girl with two rare and life-threatening conditions, atypical haemolytic uraemic syndrome (aHUS) and haemophagocytic lymphohistiocytosis (HLH), triggered by a cytomegalovirus (CMV) infection. Soon after admission, the girl became anuric and required continuous venovenous haemodiafiltration.<\/jats:p>\n               <jats:p>Initial treatments included methylprednisolone, fibrinogen and plasma infusion (for HLH), plasmapheresis (for thrombotic microangiopathy), immunoglobulins (for inflammation), ganciclovir (for CMV infection) and the antibiotic cefotaxime. On day 5, eculizumab (600\u2009mg) was given for aHUS, with rapid improvement in haematological and nephrological parameters. Despite a subsequent isolated episode of right heart thrombosis that resolved with heparin treatment, the patient showed a favourable response to eculizumab (300\u2009mg\/15 days), with improved renal function, normal haematological values, and no treatment complications. In conclusion, eculizumab effectively treated aHUS in this case despite a comorbid immunological disease.<\/jats:p>","DOI":"10.1136\/bcr-2016-219065","type":"journal-article","created":{"date-parts":[[2017,4,26]],"date-time":"2017-04-26T08:15:35Z","timestamp":1493194535000},"page":"bcr-2016-219065","update-policy":"https:\/\/doi.org\/10.1136\/crossmarkpolicy","source":"Crossref","is-referenced-by-count":7,"title":["Eculizumab in a child with atypical haemolytic uraemic syndrome and haemophagocytic lymphohistiocytosis triggered by cytomegalovirus infection"],"prefix":"10.1136","volume":"2017","author":[{"given":"Gloria M","family":"Fraga-Rodriguez","sequence":"first","affiliation":[{"name":"Hospital de la Santa Creu i Sant Pau, Universitat Aut\u00f2noma de Barcelona, Barcelona, Catalunya, Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Sonia","family":"Bri\u00f3-Sanagustin","sequence":"additional","affiliation":[{"name":"Hospital de la Santa Creu i Sant Pau, Universitat Aut\u00f2noma de Barcelona, Barcelona, Catalunya, Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Eulalia","family":"Tur\u00f3n-Vi\u00f1as","sequence":"additional","affiliation":[{"name":"Hospital de la Santa Creu i Sant Pau, Universitat Aut\u00f2noma de Barcelona, Barcelona, Catalunya, Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Bradley P","family":"Dixon","sequence":"additional","affiliation":[{"name":"Cincinnati Children's Hospital Medical Center, Cincinnati, Ohio, USA"}],"role":[{"role":"author","vocabulary":"crossref"}]},{"given":"Eduardo","family":"Carreras-Gonz\u00e1lez","sequence":"additional","affiliation":[{"name":"Hospital de la Santa Creu i Sant Pau, Universitat Aut\u00f2noma de Barcelona, Barcelona, Catalunya, Spain"}],"role":[{"role":"author","vocabulary":"crossref"}]}],"member":"239","published-online":{"date-parts":[[2017,4,26]]},"reference":[{"key":"2025082803083544000_2017.apr26_1.bcr-2016-219065.1","doi-asserted-by":"publisher","DOI":"10.1007\/s00467-008-0964-1"},{"key":"2025082803083544000_2017.apr26_1.bcr-2016-219065.2","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMra0902814"},{"key":"2025082803083544000_2017.apr26_1.bcr-2016-219065.3","doi-asserted-by":"publisher","DOI":"10.2215\/CJN.04760512"},{"key":"2025082803083544000_2017.apr26_1.bcr-2016-219065.4","doi-asserted-by":"publisher","DOI":"10.1186\/1750-1172-6-60"},{"key":"2025082803083544000_2017.apr26_1.bcr-2016-219065.5","doi-asserted-by":"publisher","DOI":"10.1016\/j.jpeds.2013.06.053"},{"key":"2025082803083544000_2017.apr26_1.bcr-2016-219065.6","doi-asserted-by":"publisher","DOI":"10.1002\/pbc.21039"},{"key":"2025082803083544000_2017.apr26_1.bcr-2016-219065.7","first-page":"362","article-title":"Thrombotic microangiopathy in hemophagocytic syndrome: a case report","volume":"101","author":"Chiang","year":"2002","journal-title":"J Formos Med Assoc"},{"key":"2025082803083544000_2017.apr26_1.bcr-2016-219065.8","doi-asserted-by":"crossref","first-page":"224","DOI":"10.5262\/tndt.2013.1002.18","article-title":"An unusual patient with atypical haemolytic uraemic syndrome who developed haemophagocytic lymphohistiocytosis","volume":"22","author":"Aky\u00fcz","year":"2013","journal-title":"Turk Nephrol Dial Transplant"},{"key":"2025082803083544000_2017.apr26_1.bcr-2016-219065.9","doi-asserted-by":"publisher","DOI":"10.1038\/nrneph.2012.214"},{"key":"2025082803083544000_2017.apr26_1.bcr-2016-219065.10","doi-asserted-by":"publisher","DOI":"10.1056\/NEJMoa1208981"},{"key":"2025082803083544000_2017.apr26_1.bcr-2016-219065.11","doi-asserted-by":"publisher","DOI":"10.1681\/ASN.2006080811"},{"key":"2025082803083544000_2017.apr26_1.bcr-2016-219065.12","doi-asserted-by":"publisher","DOI":"10.2215\/CJN.02210310"},{"key":"2025082803083544000_2017.apr26_1.bcr-2016-219065.13","doi-asserted-by":"publisher","DOI":"10.1007\/s00467-008-0872-4"},{"key":"2025082803083544000_2017.apr26_1.bcr-2016-219065.14","doi-asserted-by":"publisher","DOI":"10.1016\/S1473-3099(07)70290-6"},{"key":"2025082803083544000_2017.apr26_1.bcr-2016-219065.15","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM199811263392202"},{"key":"2025082803083544000_2017.apr26_1.bcr-2016-219065.16","doi-asserted-by":"publisher","DOI":"10.1056\/NEJM199811263392203"},{"key":"2025082803083544000_2017.apr26_1.bcr-2016-219065.17","doi-asserted-by":"publisher","DOI":"10.1038\/ki.2014.423"},{"key":"2025082803083544000_2017.apr26_1.bcr-2016-219065.18","doi-asserted-by":"crossref","unstructured":"Macia M , de Alvaro Moreno F , Dutt T , et al . Current evidence on the discontinuation of eculizumab in patients with atypical haemolytic uraemic syndrome. 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