{"status":"ok","message-type":"work","message-version":"1.0.0","message":{"indexed":{"date-parts":[[2026,7,26]],"date-time":"2026-07-26T02:23:17Z","timestamp":1785032597450,"version":"3.55.0"},"reference-count":25,"publisher":"American Society of Hematology","issue":"24","content-domain":{"domain":["ashpublications.org"],"crossmark-restriction":true},"short-container-title":[],"published-print":{"date-parts":[[2023,12,14]]},"abstract":"<jats:title>Abstract<\/jats:title>\n               <jats:p>Chronic granulomatous disease (CGD) is a primary immunodeficiency characterized by life-threatening infections and inflammatory conditions. Hematopoietic cell transplantation (HCT) is the definitive treatment for CGD, but questions remain regarding patient selection and impact of active disease on transplant outcomes. We performed a multi-institutional retrospective and prospective study of 391 patients with CGD treated either conventionally (non-HCT) enrolled from 2004 to 2018 or with HCT from 1996 to 2018. Median follow-up after HCT was 3.7 years with a 3-year overall survival of 82% and event-free survival of 69%. In a multivariate analysis, a Lansky\/Karnofsky score &amp;lt;90 and use of HLA-mismatched donors negatively affected survival. Age, genotype, and oxidase status did not affect outcomes. Before HCT, patients had higher infection density, higher frequency of noninfectious lung and liver diseases, and more steroid use than conventionally treated patients; however, these issues did not adversely affect HCT survival. Presence of pre-HCT inflammatory conditions was associated with chronic graft-versus-host disease. Graft failure or receipt of a second HCT occurred in 17.6% of the patients and was associated with melphalan-based conditioning and\/or early mixed chimerism. At 3 to 5 years after HCT, patients had improved growth and nutrition, resolved infections and inflammatory disease, and lower rates of antimicrobial prophylaxis or corticosteroid use compared with both their baseline and those of conventionally treated patients. HCT leads to durable resolution of CGD symptoms and lowers the burden of the disease. Patients with active infection or inflammation are candidates for transplants; HCT should be considered before the development of comorbidities that could affect performance status. This trial was registered at www.clinicaltrials.gov as #NCT02082353.<\/jats:p>","DOI":"10.1182\/blood.2022019586","type":"journal-article","created":{"date-parts":[[2023,8,10]],"date-time":"2023-08-10T20:15:52Z","timestamp":1691698552000},"page":"2105-2118","update-policy":"https:\/\/doi.org\/10.1182\/blood.2019cm0000","source":"Crossref","is-referenced-by-count":27,"title":["Genotype, oxidase status, and preceding infection or autoinflammation do not affect allogeneic HCT outcomes for CGD"],"prefix":"10.1182","volume":"142","author":[{"ORCID":"https:\/\/orcid.org\/0000-0002-7573-9390","authenticated-orcid":false,"given":"Jennifer W.","family":"Leiding","sequence":"first","affiliation":[{"name":"1Division of Allergy and Immunology, Department of Pediatrics, Johns Hopkins University, Baltimore, MD"},{"name":"2Institute for Clinical and Translational Research, Johns Hopkins All Children\u2019s Hospital, St. Petersburg, FL"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7501-806X","authenticated-orcid":false,"given":"Danielle E.","family":"Arnold","sequence":"additional","affiliation":[{"name":"3National Cancer Institute, National Institutes of Health, Bethesda, MD"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-6066-9852","authenticated-orcid":false,"given":"Suhag","family":"Parikh","sequence":"additional","affiliation":[{"name":"4Aflac Cancer and Blood Disorders Center, Emory University and Children\u2019s Healthcare of Atlanta, Atlanta, GA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Brent","family":"Logan","sequence":"additional","affiliation":[{"name":"5Division of Biostatistics, Medical College of Wisconsin, Milwaukee, WI"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Rebecca A.","family":"Marsh","sequence":"additional","affiliation":[{"name":"6Division of Bone Marrow Transplantation and Immune Deficiency, Department of Pediatrics, Cincinnati Children\u2019s Hospital Medical Center, University of Cincinnati, Cincinnati, OH"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Linda M.","family":"Griffith","sequence":"additional","affiliation":[{"name":"7Division of Allergy, Immunology, and Transplantation, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Ruizhe","family":"Wu","sequence":"additional","affiliation":[{"name":"5Division of Biostatistics, Medical College of Wisconsin, Milwaukee, WI"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Sharon","family":"Kidd","sequence":"additional","affiliation":[{"name":"8Pediatric Allergy, Immunology, and Blood and Marrow Transplant Division, UCSF Benioff Children\u2019s Hospital, San Francisco, CA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Kanwaldeep","family":"Mallhi","sequence":"additional","affiliation":[{"name":"9Fred Hutchinson Cancer Research Center, Department of Pediatrics, University of Washington, and Seattle Children\u2019s Hospital, Seattle, WA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0003-3518-9790","authenticated-orcid":false,"given":"Deepak","family":"Chellapandian","sequence":"additional","affiliation":[{"name":"10Center for Cell and Gene Therapy for Non-Malignant Conditions, Johns Hopkins All Children\u2019s Hospital, St Petersburg, FL"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-5983-581X","authenticated-orcid":false,"given":"Stephanie J.","family":"Si Lim","sequence":"additional","affiliation":[{"name":"11Department of Pediatrics, John A. Burns School of Medicine, University of Hawai'i Cancer Center, University of Hawai'i at M\u0101noa, Honolulu, HI"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Eyal","family":"Grunebaum","sequence":"additional","affiliation":[{"name":"12Division of Immunology and Allergy, Department of Pediatrics, The Hospital for Sick Children, Toronto, ON, Canada"},{"name":"13Faculty of Medicine, University of Toronto, Toronto, ON, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"E. Liana","family":"Falcone","sequence":"additional","affiliation":[{"name":"14Center for Inflammation, Immunity and Infectious Diseases, Montreal Clinical Research Institute, Montreal, QC, Canada"},{"name":"15Laboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-6893-3366","authenticated-orcid":false,"given":"Luis","family":"Murguia-Favela","sequence":"additional","affiliation":[{"name":"16Section of Hematology\/Immunology, Alberta Children's Hospital, University of Calgary, Calgary, AB, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Debbi","family":"Grossman","sequence":"additional","affiliation":[{"name":"15Laboratory of Clinical Immunology and Microbiology, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-8151-8212","authenticated-orcid":false,"given":"Vinod K.","family":"Prasad","sequence":"additional","affiliation":[{"name":"17Division of Pediatric Transplant and Cellular Therapy, Department of Pediatrics, Duke University Medical Center, Durham, NC"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jennifer R.","family":"Heimall","sequence":"additional","affiliation":[{"name":"18Division of Allergy and Immunology, Department of Pediatrics, Children\u2019s Hospital of Philadelphia, Perelman School of Medicine at University of Pennsylvania, Philadelphia, PA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0002-0889-4905","authenticated-orcid":false,"given":"Fabien","family":"Touzot","sequence":"additional","affiliation":[{"name":"19Department of Pediatrics, Centre Hospitalier Universitaire Sainte-Justine, University of Montreal, Montreal, QC, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Lauri M.","family":"Burroughs","sequence":"additional","affiliation":[{"name":"9Fred Hutchinson Cancer Research Center, Department of Pediatrics, University of Washington, and Seattle Children\u2019s Hospital, Seattle, WA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jack","family":"Bleesing","sequence":"additional","affiliation":[{"name":"6Division of Bone Marrow Transplantation and Immune Deficiency, Department of Pediatrics, Cincinnati Children\u2019s Hospital Medical Center, University of Cincinnati, Cincinnati, OH"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Neena","family":"Kapoor","sequence":"additional","affiliation":[{"name":"20Division of Hematology, Oncology and Blood and Marrow Transplant, Children\u2019s Hospital, Los Angeles, CA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Jasmeen","family":"Dara","sequence":"additional","affiliation":[{"name":"8Pediatric Allergy, Immunology, and Blood and Marrow Transplant Division, UCSF Benioff Children\u2019s Hospital, San Francisco, CA"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Olatundun","family":"Williams","sequence":"additional","affiliation":[{"name":"21Division of Hematology, Oncology and Stem Cell Transplantation, Ann & Robert H. 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E.","family":"Cuvelier","sequence":"additional","affiliation":[{"name":"27Manitoba Blood and Marrow Transplant Program, CancerCare Manitoba, University of Manitoba, Winnipeg, MB, Canada"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-9402-1934","authenticated-orcid":false,"given":"Lisa R.","family":"Forbes Satter","sequence":"additional","affiliation":[{"name":"28Immunology, Allergy and Retrovirology, Baylor College of Medicine, Texas Children\u2019s Hospital, Houston, TX"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"ORCID":"https:\/\/orcid.org\/0000-0001-7141-2457","authenticated-orcid":false,"given":"Caridad","family":"Martinez","sequence":"additional","affiliation":[{"name":"29Department of Pediatrics, Baylor College of Medicine, and Texas Children's Hospital Center for Gene and Cell Therapy, Houston, TX"}],"role":[{"vocabulary":"crossref","role":"author"}]},{"given":"Mark T.","family":"Vander 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