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Tytuł:
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Pulmonary mucormycosis following autologous hematopoietic stem cell transplantation for rapidly progressive diffuse cutaneous systemic sclerosis: A case report.
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Autorzy:
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Boumaza X; Department of Internal Medicine.
Lelièvre L; Department of infectious and tropical diseases, Toulouse University Hospital.
Guenounou S; Hematology Department.
Borel C; Hematology Department.
Huynh A; Hematology Department.
Beziat G; Hematology Department.
Delavigne K; Department of Internal Medicine, Institut Universitaire du Cancer de Toulouse - Oncopole.
Guinault D; Department of Nephrology and Organ Transplantation.
Garric M; Department of Internal Medicine.
Piel-Julian M; Department of Internal Medicine.
Paricaud K; Department of Internal Medicine.
Moulis G; Department of Internal Medicine.; Clinical Investigation Center, Toulouse University Hospital.; UMR 1027 INSERM, University of Toulouse, Toulouse.
Astudillo L; Department of Internal Medicine.
Sailler L; Department of Internal Medicine.; Clinical Investigation Center, Toulouse University Hospital.; UMR 1027 INSERM, University of Toulouse, Toulouse.
Farge D; Unité de Médecine Interne: Maladies Auto-immunes et Pathologie Vasculaire (UF 04), Hôpital St-Louis, AP-HP, 1 Avenue Claude Vellefaux.; Centre de Référence des Maladies auto-immunes Systémiques Rares d'Ile-de-France.; EA 3518, Université Denis Diderot, Paris, France.; Department of Internal Medicine, McGill University, Montréal, Canada.
Pugnet G; Department of Internal Medicine.; Clinical Investigation Center, Toulouse University Hospital.; UMR 1027 INSERM, University of Toulouse, Toulouse.
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Źródło:
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Medicine [Medicine (Baltimore)] 2020 Jul 31; Vol. 99 (31), pp. e21431.
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Typ publikacji:
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Case Reports; Journal Article
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Język:
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English
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Imprint Name(s):
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Original Publication: Hagerstown, Md : Lippincott Williams & Wilkins
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MeSH Terms:
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Hematopoietic Stem Cell Transplantation/*adverse effects
Mucormycosis/*etiology
Scleroderma, Diffuse/*etiology
Scleroderma, Systemic/*therapy
Transplantation, Autologous/*adverse effects
Acute Disease ; Administration, Intravenous ; Aftercare ; Amphotericin B/administration & dosage ; Amphotericin B/therapeutic use ; Antifungal Agents/administration & dosage ; Antifungal Agents/therapeutic use ; Female ; Hematopoietic Stem Cell Transplantation/methods ; Humans ; Lung Diseases, Fungal/diagnostic imaging ; Lung Diseases, Fungal/microbiology ; Lung Diseases, Fungal/pathology ; Mucorales/genetics ; Respiratory Distress Syndrome/etiology ; Scleroderma, Diffuse/pathology ; Transplantation, Autologous/methods ; Treatment Outcome ; Young Adult
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References:
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Substance Nomenclature:
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0 (Antifungal Agents)
7XU7A7DROE (Amphotericin B)
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Entry Date(s):
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Date Created: 20200807 Date Completed: 20200817 Latest Revision: 20221005
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Update Code:
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20240105
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PubMed Central ID:
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PMC7402716
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DOI:
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10.1097/MD.0000000000021431
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PMID:
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32756151
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Rationale: The use of autologous hematopoietic stem cell transplantation (AHSCT) for autoimmune diseases has become the first indication for transplant in nonmalignant disease. Mucormycosis is a rare invasive infection with increasing incidence in patients treated with AHSCT. We report the first case of pulmonary mucormycosis following AHSCT for systemic sclerosis (SSc).
Patient Concerns: A 24-year-old woman with rapidly progressive diffuse cutaneous SSc presented with an acute respiratory distress syndrome 6 days after AHSCT.
Diagnoses: The results of clinical and computed tomography scan were consistent with pulmonary mucormycosis and the diagnosis was confirmed by a positive Mucorales Polymerase Chain Reaction on a peripheral blood sample.
Interventions and Outcomes: Early antifungal therapy by intravenous amphotericin B provided rapid improvement within 4 days and sustained recovery after 2 years of follow-up.
Lessons: With the progressively increasing use of AHSCT and other stem cell therapy for treatment of severe SSc and other autoimmune diseases, the potential onset of rare post-transplant fungal infections, such as mucormycosis, requires careful patient monitoring and better awareness of early initiation of adequate therapy.