{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,10,29]],"date-time":"2026-10-29T01:17:04Z","timestamp":1793236624639,"version":"4.5.0"},"reference-count":50,"publisher":"Oxford University Press (OUP)","license":[{"start":{"date-parts":[[2026,6,30]],"date-time":"2026-06-30T00:00:00Z","timestamp":1782777600000},"content-version":"am","delay-in-days":0,"URL":"https:\/\/academic.oup.com\/pages\/standard-publication-reuse-rights"}],"content-domain":{"domain":[],"crossmark-restriction":false},"short-container-title":[],"abstract":"<jats:title>Abstract<\/jats:title>\n                  <jats:sec>\n                    <jats:title>Background<\/jats:title>\n                    <jats:p>Inborn errors of immunity (IEI) display increased susceptibility to infections among which cryptosporidiosis is an opportunistic infection poorly characterized in this population. We report cases of Cryptosporidium spp. infections in French patients with IEI based on data from the registry of the French National Reference Center for IEI (CEREDIH).<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Methods<\/jats:title>\n                    <jats:p>Patients diagnosed with an IEI between 1986 and 2025 and who experienced proven cryptosporidiosis were included.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Results<\/jats:title>\n                    <jats:p>Of the 10 004 patients in the CEREDIH registry, 149 were identified with at least one episode of parasitic infection (\u22481.4%). Forty-one patients (27.5%) had cryptosporidiosis (0.41% of the cohort) and had the following diagnoses: combined immunodeficiency (CID, n = 25 including 8 hyper IgM [HIGM] syndromes, 9 other CID, 4 HLA class II deficiency), hyper IgE syndrome (n = 2), diaphanous related formin 1 mutation (n = 1), signal transducer and activator of transcription 1 (STAT1) gain-of-function (GOF) (n = 1), activated PI3K-delta syndrome (n = 6), common variable immunodeficiency disorders (CVID, n = 3), and 7 miscellaneous. The male-to-female ratio was 1.92, and the mean age at onset of cryptosporidiosis was 16.6 \u00b1 17.5 years. Twenty (50%) patients had cholangitis and 23 (63%) had an indication for hematopoietic stem cell transplantation, which was performed in 15 (38%). Seventeen (41.5%) patients died.<\/jats:p>\n                  <\/jats:sec>\n                  <jats:sec>\n                    <jats:title>Conclusions<\/jats:title>\n                    <jats:p>Cryptosporidiosis is a serious comorbidity in patients with IEI with a high mortality rate reaching more than one third of the patients. Combined immunodeficiency including HIGM syndrome and other CID were the most represented underlying condition in this study with the highest morbidity and mortality (cholangitis and its complications).<\/jats:p>\n                  <\/jats:sec>","DOI":"10.1093\/infdis\/jiag339","type":"journal-article","created":{"date-parts":[[2026,6,30]],"date-time":"2026-06-30T23:50:00Z","timestamp":1782863400000},"source":"Crossref","is-referenced-by-count":1,"title":["Cryptosporidiosis in Patients With Inborn Errors of Immunity: Retrospective Cohort Study of the French National Reference Center (CEREDIH)"],"prefix":"10.1093","author":[{"given":"Deborah To","family":"Puzenat","sequence":"first","affiliation":[{"name":"Infectious Diseases and Tropical Medicine Department, Necker-Enfants Malades University Hospital, Assistance Publique-H\u00f4pitaux de Paris , Paris ,","place":["France"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Morgane","family":"Cheminant","sequence":"additional","affiliation":[{"name":"Paris Cit\u00e9 University , Paris ,","place":["France"]},{"name":"Haematology Department, Necker-Enfants Malades University Hospital, Assistance Publique-H\u00f4pitaux de Paris , Paris ,","place":["France"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2817-3830","authenticated-orcid":false,"given":"Eric","family":"Dannaoui","sequence":"additional","affiliation":[{"name":"Paris Cit\u00e9 University , Paris ,","place":["France"]},{"name":"Parasitology-Mycology Unit, Microbiology Department, Necker-Enfants Malades University Hospital, Assistance Publique-H\u00f4pitaux de Paris , Paris ,","place":["France"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Emilie","family":"Sitterl\u00e9","sequence":"additional","affiliation":[{"name":"Parasitology-Mycology Unit, 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,","place":["France"]},{"name":"Department of Pathology, Assistance Publique-H\u00f4pitaux de Paris, Necker and Robert Debr\u00e9 Hospital , Paris ,","place":["France"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Despina","family":"Moshous","sequence":"additional","affiliation":[{"name":"Paris Cit\u00e9 University , Paris ,","place":["France"]},{"name":"French National Reference Center for Primary Immunodeficiencies (CEREDIH), Necker-Enfants Malades University Hospital, Assistance Publique-H\u00f4pitaux de Paris , Paris ,","place":["France"]},{"name":"Pediatric Immunology, Hematology and Rheumatology Unit, Necker-Enfants Malades University Hospital, Assistance Publique-H\u00f4pitaux de Paris , Paris ,","place":["France"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jacinta","family":"Bustamante","sequence":"additional","affiliation":[{"name":"Laboratory of Human Genetics of Infectious Diseases, Necker Branch INSERM U1163, Necker-Enfants Malades University Hospital , Paris ,","place":["France"]},{"name":"Paris Cit\u00e9 University, Imagine Institute , Paris ,","place":["France"]},{"name":"Laboratory of Lymphocyte Activation and Susceptibility to EBV Infection, Inserm UMR 1163, Institut Imagine , Paris ,","place":["France"]},{"name":"St.Giles Laboratory of Human Genetics of Infectious Diseases, Rockefeller University , New York, New York ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Capucine","family":"Picard","sequence":"additional","affiliation":[{"name":"Paris Cit\u00e9 University , Paris ,","place":["France"]},{"name":"French National Reference Center for Primary Immunodeficiencies (CEREDIH), Necker-Enfants Malades University Hospital, Assistance Publique-H\u00f4pitaux de Paris , Paris ,","place":["France"]},{"name":"Pediatric Immunology, Hematology and Rheumatology Unit, Necker-Enfants Malades University Hospital, Assistance Publique-H\u00f4pitaux de Paris , Paris ,","place":["France"]},{"name":"Laboratory of Human Genetics of Infectious Diseases, Necker Branch INSERM U1163, Necker-Enfants Malades University Hospital , Paris ,","place":["France"]},{"name":"Paris Cit\u00e9 University, Imagine Institute , Paris ,","place":["France"]},{"name":"Laboratory of Lymphocyte Activation and Susceptibility to EBV Infection, Inserm UMR 1163, Institut Imagine , Paris ,","place":["France"]},{"name":"St.Giles Laboratory of Human Genetics of Infectious Diseases, Rockefeller University , New York, New York ,","place":["USA"]}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-2822-161X","authenticated-orcid":false,"given":"J\u00e9r\u00e9my","family":"Rosain","sequence":"additional","affiliation":[{"name":"Laboratory of Human Genetics of Infectious Diseases, Necker Branch INSERM U1163, Necker-Enfants Malades University Hospital , Paris ,","place":["France"]},{"name":"Paris Cit\u00e9 University, Imagine Institute , Paris 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