{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,26]],"date-time":"2026-10-26T22:44:21Z","timestamp":1793054661004,"version":"4.5.0"},"reference-count":27,"publisher":"Oxford University Press (OUP)","issue":"2","license":[{"start":{"date-parts":[[2020,10,30]],"date-time":"2020-10-30T00:00:00Z","timestamp":1604016000000},"content-version":"vor","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/journals\/pages\/open_access\/funder_policies\/chorus\/standard_publication_model"}],"funder":[{"name":"Sanofi-Aventis France"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"published-print":{"date-parts":[[2021,1,19]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:sec>\n                  <jats:title>Background<\/jats:title>\n                  <jats:p>Intestinal microsporidiosis due to Enterocytozoon bieneusi is a cause of chronic diarrhoea in immunocompromised patients. Fumagillin has been approved in France for its treatment.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Objectives<\/jats:title>\n                  <jats:p>To investigate the efficacy and safety of fumagillin in a real-life setting.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Methods<\/jats:title>\n                  <jats:p>As required by the French Medicine Agency, all patients receiving fumagillin were enrolled in a prospective study to evaluate its efficacy and safety. Stool examination with identification of E. bieneusi by PCR was performed at baseline, end of treatment and monthly thereafter for 6\u2009months. Safety was monitored up to 6\u2009months and full blood counts were monitored up to 42\u2009days after treatment initiation. The primary endpoint was safety. Parasite clearance and relapses were secondary endpoints.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Results<\/jats:title>\n                  <jats:p>From 2007 to 2018, 166 patients received fumagillin, including 6 children. Patients were transplant recipients (84%), HIV-infected patients (13%) or had another cause of immunosuppression (5%). Serious adverse events were reported in 41 patients (25%), mainly thrombocytopenia (15%) and neutropenia (5%), with two haemorrhagic events leading to one death. Severe thrombocytopenia (&amp;lt;50\u2009G\/L) developed in 50 patients (29.6%), neutropenia (&amp;lt;1\u2009G\/L) in 20 patients (11.8%) and severe anaemia (&amp;lt;8\u2009g\/dL) in 21 patients (12.4%). At the end of treatment, 94% of patients with available stool examination (n\u2009=\u2009132) had no spores detected. Among 99 patients with available follow-up after the end of treatment, three parasite relapses were documented.<\/jats:p>\n               <\/jats:sec>\n               <jats:sec>\n                  <jats:title>Conclusions<\/jats:title>\n                  <jats:p>E. bieneusi microsporidiosis was mainly diagnosed in transplant recipients. Fumagillin was associated with haematological toxicity but showed high efficacy with a low relapse rate.<\/jats:p>\n               <\/jats:sec>","DOI":"10.1093\/jac\/dkaa438","type":"journal-article","created":{"date-parts":[[2020,10,6]],"date-time":"2020-10-06T19:15:48Z","timestamp":1602011748000},"page":"487-494","source":"Crossref","is-referenced-by-count":26,"title":["Safety and efficacy of fumagillin for the treatment of intestinal microsporidiosis. A French prospective cohort study"],"prefix":"10.1093","volume":"76","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-7165-4103","authenticated-orcid":false,"given":"Alexis","family":"Maillard","sequence":"first","affiliation":[{"name":"Department of Infectious Diseases, St-Louis Hospital, APHP and University of Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Anne","family":"Scemla","sequence":"additional","affiliation":[{"name":"Kidney Transplant Unit, H\u00f4pital Necker Enfants Malades, APHP and University of Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Benjamin","family":"Laffy","sequence":"additional","affiliation":[{"name":"Kidney Transplant Unit, H\u00f4pital Saint-Louis, APHP and University of Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Nadir","family":"Mahloul","sequence":"additional","affiliation":[{"name":"Medical Department, Sanofi-Aventis France, Gentilly, France"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jean-Michel","family":"Molina","sequence":"additional","affiliation":[{"name":"Department of Infectious Diseases, St-Louis Hospital, APHP and University of Paris, France"}],"role":[{"vocabulary":"crossref","role":"author"}]}],"member":"286","published-online":{"date-parts":[[2020,10,30]]},"reference":[{"key":"2021012006030883100_dkaa438-B1","doi-asserted-by":"crossref","first-page":"485","DOI":"10.1097\/01.qco.0000244055.46382.23","article-title":"Microsporidiosis: current status","volume":"19","author":"Didier","year":"2006","journal-title":"Curr Opin Infect Dis"},{"key":"2021012006030883100_dkaa438-B2","doi-asserted-by":"crossref","first-page":"217","DOI":"10.1093\/infdis\/167.1.217","article-title":"Intestinal microsporidiosis in human immunodeficiency virus-infected patients with chronic unexplained diarrhea: prevalence and clinical and biologic features","volume":"167","author":"Molina","year":"1993","journal-title":"J Infect Dis"},{"key":"2021012006030883100_dkaa438-B3","doi-asserted-by":"crossref","first-page":"475","DOI":"10.1016\/0046-8177(90)90003-N","article-title":"Intestinal microsporidiosis as a cause of diarrhea in human immunodeficiency virus-infected patients: a report of 20 cases","volume":"21","author":"Orenstein","year":"1990","journal-title":"Hum Pathol"},{"key":"2021012006030883100_dkaa438-B4","doi-asserted-by":"crossref","first-page":"1039","DOI":"10.3201\/eid2306.161825","article-title":"Enterocytozoon bieneusi microsporidiosis in stem cell transplant recipients treated with fumagillin","volume":"23","author":"Bukreyeva","year":"2017","journal-title":"Emerg Infect Dis"},{"key":"2021012006030883100_dkaa438-B5","doi-asserted-by":"crossref","first-page":"1925","DOI":"10.1111\/j.1600-6143.2010.03166.x","article-title":"Fumagillin for treatment of intestinal microsporidiosis in renal transplant recipients","volume":"10","author":"Champion","year":"2010","journal-title":"Am J Transplant"},{"key":"2021012006030883100_dkaa438-B6","doi-asserted-by":"crossref","first-page":"83","DOI":"10.1111\/j.1399-3062.2008.00347.x","article-title":"Microsporidiosis in solid organ transplant recipients: two Enterocytozoon bieneusi cases and review","volume":"11","author":"Lanternier","year":"2009","journal-title":"Transpl Infect Dis"},{"key":"2021012006030883100_dkaa438-B7","doi-asserted-by":"crossref","first-page":"445","DOI":"10.1016\/j.cll.2015.02.011","article-title":"Intestinal microsporidiosis","volume":"35","author":"Field","year":"2015","journal-title":"Clin Lab Med"},{"key":"2021012006030883100_dkaa438-B8","doi-asserted-by":"crossref","first-page":"874","DOI":"10.1093\/infdis\/174.4.874","article-title":"Detection and species identification of intestinal microsporidia by polymerase chain reaction in duodenal biopsies from human immunodeficiency virus-infected patients","volume":"174","author":"David","year":"1996","journal-title":"J Infect Dis"},{"key":"2021012006030883100_dkaa438-B9","doi-asserted-by":"crossref","first-page":"723","DOI":"10.1097\/00002030-199706000-00004","article-title":"Diagnosis of infections caused by Enterocytozoon bieneusi and Encephalitozoon intestinalis using polymerase chain reaction in stool specimens","volume":"11","author":"Liguory","year":"1997","journal-title":"AIDS"},{"key":"2021012006030883100_dkaa438-B10","doi-asserted-by":"crossref","first-page":"248","DOI":"10.1016\/j.diagmicrobio.2013.07.004","article-title":"Comparison of staining techniques and multiplex nested PCR for diagnosis of intestinal microsporidiosis","volume":"77","author":"Saigal","year":"2013","journal-title":"Diagn Microbiol Infect Dis"},{"key":"2021012006030883100_dkaa438-B11","doi-asserted-by":"crossref","first-page":"256","DOI":"10.1016\/S0140-6736(97)07529-6","article-title":"Treatment of HIV-1-associated microsporidiosis and cryptosporidiosis with combination antiretroviral therapy","volume":"351","author":"Carr","year":"1998","journal-title":"Lancet"},{"key":"2021012006030883100_dkaa438-B12","doi-asserted-by":"crossref","first-page":"527","DOI":"10.1128\/JB.58.4.527-529.1949","article-title":"An antiphage agent isolated from Aspergillus sp","volume":"58","author":"Hanson","year":"1949","journal-title":"J Bacteriol"},{"key":"2021012006030883100_dkaa438-B13","doi-asserted-by":"crossref","first-page":"70","DOI":"10.1126\/science.115.2977.70","article-title":"Control of nosema disease of honeybees with fumagillin","volume":"115","author":"Katznelson","year":"1952","journal-title":"Science"},{"key":"2021012006030883100_dkaa438-B14","doi-asserted-by":"crossref","first-page":"1963","DOI":"10.1056\/NEJMoa012924","article-title":"Fumagillin treatment of intestinal microsporidiosis","volume":"346","author":"Molina","year":"2002","journal-title":"N Engl J Med"},{"key":"2021012006030883100_dkaa438-B15","doi-asserted-by":"crossref","first-page":"1603","DOI":"10.1097\/00002030-199713000-00009","article-title":"Potential efficacy of fumagillin in intestinal microsporidiosis due to Enterocytozoon bieneusi in patients with HIV infection: results of a drug screening study","volume":"11","author":"Molina","year":"1997","journal-title":"AIDS"},{"key":"2021012006030883100_dkaa438-B16","doi-asserted-by":"crossref","first-page":"1341","DOI":"10.1097\/00002030-200007070-00006","article-title":"Trial of oral fumagillin for the treatment of intestinal microsporidiosis in patients with HIV infection. ANRS 054 Study Group. Agence Nationale de Recherche sur le SIDA","volume":"14","author":"Molina","year":"2000","journal-title":"AIDS"},{"key":"2021012006030883100_dkaa438-B17","doi-asserted-by":"crossref","first-page":"136","DOI":"10.1097\/01.tp.0000189947.55624.74","article-title":"Interaction between tacrolimus and fumagillin in two kidney transplant recipients","volume":"81","author":"Arzouk","year":"2006","journal-title":"Transplantation"},{"key":"2021012006030883100_dkaa438-B18","doi-asserted-by":"crossref","first-page":"e13618","DOI":"10.1111\/ctr.13618","article-title":"Intestinal parasites including Cryptosporidium, Cyclospora, Giardia, and Microsporidia, Entamoeba histolytica, Strongyloides, Schistosomiasis, and Echinococcus: guidelines from the American Society of Transplantation Infectious Diseases Community of Practice","volume":"33","author":"La Hoz","year":"2019","journal-title":"Clin Transplant"},{"key":"2021012006030883100_dkaa438-B19","doi-asserted-by":"crossref","first-page":"694","DOI":"10.1136\/jcp.46.8.694","article-title":"Diagnosis of intestinal and disseminated microsporidial infections in patients with HIV by a new rapid fluorescence technique","volume":"46","author":"van Gool","year":"1993","journal-title":"J Clin Pathol"},{"key":"2021012006030883100_dkaa438-B20","doi-asserted-by":"crossref","first-page":"805","DOI":"10.1128\/JCM.33.4.805-810.1995","article-title":"Diagnosis of intestinal microsporidiosis by examination of stool and duodenal aspirate with Weber\u2019s modified trichrome and Uvitex 2B strains","volume":"33","author":"DeGirolami","year":"1995","journal-title":"J Clin Microbiol"},{"key":"2021012006030883100_dkaa438-B21"},{"key":"2021012006030883100_dkaa438-B22","doi-asserted-by":"crossref","first-page":"E147","DOI":"10.1111\/tid.12010","article-title":"Fumagillin-induced aseptic meningoencephalitis in a kidney transplant recipient with microsporidiosis","volume":"14","author":"Audemard","year":"2012","journal-title":"Transpl Infect Dis"},{"key":"2021012006030883100_dkaa438-B23","doi-asserted-by":"crossref","first-page":"E250","DOI":"10.1111\/tid.12158","article-title":"Successful treatment with fumagillin of the first pediatric case of digestive microsporidiosis in a liver-kidney transplant","volume":"15","author":"Desoubeaux","year":"2013","journal-title":"Transpl Infect Dis"},{"key":"2021012006030883100_dkaa438-B24","doi-asserted-by":"crossref","first-page":"e66-67","DOI":"10.1097\/TP.0b013e3182a902e7","article-title":"Intestinal microsporidiosis due to Enterocytozoon bieneusi in a pediatric kidney transplant recipient successfully treated with fumagillin","volume":"96","author":"Godron","year":"2013","journal-title":"Transplantation"},{"key":"2021012006030883100_dkaa438-B25","doi-asserted-by":"crossref","first-page":"7","DOI":"10.3390\/toxins12010007","article-title":"Fumagillin, a mycotoxin of Aspergillus fumigatus: biosynthesis, biological activities, detection, and applications","volume":"12","author":"Guruceaga","year":"2019","journal-title":"Toxins (Basel)"},{"key":"2021012006030883100_dkaa438-B26","doi-asserted-by":"crossref","first-page":"63306","DOI":"10.18632\/oncotarget.11216","article-title":"MetAP1 and MetAP2 drive cell selectivity for a potent anti-cancer agent in synergy, by controlling glutathione redox state","volume":"7","author":"Frottin","year":"2016","journal-title":"Oncotarget"},{"key":"2021012006030883100_dkaa438-B27"}],"container-title":["Journal of Antimicrobial Chemotherapy"],"original-title":[],"language":"en","link":[{"URL":"http:\/\/academic.oup.com\/jac\/article-pdf\/76\/2\/487\/35925239\/dkaa438.pdf","content-type":"application\/pdf","content-version":"vor","intended-application":"syndication"},{"URL":"http:\/\/academic.oup.com\/jac\/article-pdf\/76\/2\/487\/35925239\/dkaa438.pdf","content-type":"unspecified","content-version":"vor","intended-application":"similarity-checking"}],"deposited":{"date-parts":[[2021,1,20]],"date-time":"2021-01-20T14:08:15Z","timestamp":1611151695000},"score":1,"resource":{"primary":{"URL":"https:\/\/academic.oup.com\/jac\/article\/76\/2\/487\/5944181"}},"subtitle":[],"short-title":[],"issued":{"date-parts":[[2020,10,30]]},"references-count":27,"journal-issue":{"issue":"2","published-online":{"date-parts":[[2020,10,30]]},"published-print":{"date-parts":[[2021,1,19]]}},"URL":"https:\/\/doi.org\/10.1093\/jac\/dkaa438","relation":{},"ISSN":["0305-7453","1460-2091"],"issn-type":[{"value":"0305-7453","type":"print"},{"value":"1460-2091","type":"electronic"}],"subject":[],"published-other":{"date-parts":[[2021,2]]},"published":{"date-parts":[[2020,10,30]]}}}