Sarah Elitzur1,2, Salvador Fischer1,2, Nira Arad-Cohen3,4, Assaf Barg2,5, Miriam Ben-Harosh6, Dana Danino7, Ronit Elhasid2,8, Aharon Gefen3,4, Gil Gilad1,2, Itzhak Levy2,9, Yael Shachor-Meyouhas4,10, Sigal Weinreb11, Shai Izraeli1,2, Shlomit Barzilai-Birenboim1,2. 1. The Rina Zaizov Division of Pediatric Hematology-Oncology, Schneider Children's Medical Center, Petah Tikva 4920235, Israel. 2. Sackler Faculty of Medicine, Tel Aviv University, Tel Aviv 6997801, Israel. 3. Pediatric Hematology-Oncology Department, Ruth Rappaport Children's Hospital, Rambam Health Care Campus, Haifa 3109601, Israel. 4. Rappaport Faculty of Medicine, Technion-Israel Institute of Technology, Haifa 3200003, Israel. 5. Division of Pediatric Hematology, Oncology and BMT, The Edmond and Lily Safra Children's Hospital, Sheba Medical Center, Ramat Gan 5262161, Israel. 6. Department of Pediatric Hematology-Oncology, Soroka Medical Center, Ben Gurion University, Beer Sheva 8489501, Israel. 7. Pediatric Infectious Disease Unit, Soroka Medical Center, Ben Gurion University, Beer Sheva 8489501, Israel. 8. Department of Pediatric Hemato-Oncology, Sourasky Medical Center, Tel Aviv 6423906, Israel. 9. Pediatric Infectious Disease Unit; Schneider Children's Medical Center, Petah Tikva 4920235, Israel. 10. Pediatric Infectious Disease Unit, Ruth Rappaport Children's Hospital, Rambam Health Care Campus, Haifa 3109601, Israel. 11. Pediatric Hematology-Oncology, Hadassah Hebrew University Medical Center, Jerusalem 9112000, Israel.
Abstract
BACKGROUND: Mucormycosis is a life-threatening infection with a tendency for angioinvasion that may lead to progressive dissemination. Disseminated mucormycosis, defined as the involvement of two or more non-contiguous sites, is rare in children, and data concerning its management and outcome are scarce. The aim of this study was to assess the contemporary management strategies and outcomes of disseminated mucormycosis in the pediatric population. METHODS: We conducted a retrospective search in six large tertiary medical centers for all cases of disseminated mucormycosis that occurred between 2009-2020 in patients aged 1-20 years. RESULTS: Twelve cases were identified. Underlying conditions included hematological malignancies (n = 10), solid tumor (post-autologous hematopoietic stem cell transplantations; n = 1), and solid organ (liver) transplantation (n = 1). In all cases, amphotericin B formulations were administered as first-line therapy; in eight cases, they were also administered in combination with an echinocandin or triazole. Seven patients underwent surgical debridement procedures. The six-week mortality was 58%. Among the patients diagnosed between 2009-2015, one of the six survived, and of those diagnosed between 2016-2020, four of the six were salvaged. CONCLUSIONS: Disseminated mucormycosis is a life-threatening and often fatal disease, and improved diagnostic and therapeutic strategies are needed. Nevertheless, in this population-based study, five patients (42%) were salvaged through combined liposomal amphotericin/triazole treatment and extensive surgical interventions.
BACKGROUND: Mucormycosis is a life-threatening infection with a tendency for angioinvasion that may lead to progressive dissemination. Disseminated mucormycosis, defined as the involvement of two or more non-contiguous sites, is rare in children, and data concerning its management and outcome are scarce. The aim of this study was to assess the contemporary management strategies and outcomes of disseminated mucormycosis in the pediatric population. METHODS: We conducted a retrospective search in six large tertiary medical centers for all cases of disseminated mucormycosis that occurred between 2009-2020 in patients aged 1-20 years. RESULTS: Twelve cases were identified. Underlying conditions included hematological malignancies (n = 10), solid tumor (post-autologous hematopoietic stem cell transplantations; n = 1), and solid organ (liver) transplantation (n = 1). In all cases, amphotericin B formulations were administered as first-line therapy; in eight cases, they were also administered in combination with an echinocandin or triazole. Seven patients underwent surgical debridement procedures. The six-week mortality was 58%. Among the patients diagnosed between 2009-2015, one of the six survived, and of those diagnosed between 2016-2020, four of the six were salvaged. CONCLUSIONS:Disseminated mucormycosis is a life-threatening and often fatal disease, and improved diagnostic and therapeutic strategies are needed. Nevertheless, in this population-based study, five patients (42%) were salvaged through combined liposomal amphotericin/triazole treatment and extensive surgical interventions.
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