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Title

Successful treatment of invasive aspergillosis in chronic granulomatous disease by bone marrow transplantation, granulocyte colony-stimulating factor-mobilized granulocytes, and liposomal amphotericin-B.

Authors
Müller, I
Steinert, H C
Nadal, D
Lauener, R
Tuchschmid, P
Willi, U V
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Published in Blood. 1998, vol. 92, no. 8, p. 2719-24
Abstract X-linked chronic granulomatous disease (X-CGD) is a primary immunodeficiency with complete absence or malfunction of the nicotinamide adenine dinucleotide phosphate (NADPH) oxidase in the phagocytic cells. Life-threatening infections especially with aspergillus are common despite optimal antimicrobial therapy. Bone marrow transplantation (BMT) is contraindicated during invasive aspergillosis in any disease setting. We report an 8-year-old patient with CGD who underwent HLA-genoidentical BMT during invasive multifocal aspergillus nidulans infection, nonresponsive to treatment with amphotericin-B and gamma-interferon. During the first 10 days post-BMT, the patient received granulocyte colony-stimulating factor (G-CSF)-mobilized, 25 Gy irradiated granulocytes from healthy volunteers plus G-CSF beginning on day 3 to prolong the viability of the transfused granulocytes. This was confirmed in vitro by apoptosis assays and in vivo by finding nitroblue tetrazolium (NBT)-positive granulocytes in peripheral blood 12 and 36 hours after the transfusions. Clinical and biological signs of infection began to disappear on day 7 post-BMT. Positron emission tomography with F18-fluorodeoxyglucose (FDG-PET) and computed tomography (CT) scans at 3 months post-BMT showed complete disappearance of infectious foci. At 2 years post-BMT, the patient is well with full immune reconstitution and no sign of aspergillus infection. Our results show that HLA-identical BMT may be successful during invasive, noncontrollable aspergillus infection, provided that supportive therapy is optimal.
Keywords Amphotericin B/administration & dosage/therapeutic useAntifungal Agents/administration & dosage/therapeutic useApoptosisAspergillosis/drug therapy/prevention & control/radionuclide imaging/therapyAspergillus nidulansBone Marrow TransplantationChildCombined Modality TherapyDrug CarriersGraft Survival/drug effectsGranulocyte Colony-Stimulating Factor/therapeutic useGranulocytes/physiologyGranulomatous Disease, Chronic/complications/therapyHumansItraconazole/therapeutic useLeukocyte CountLeukocyte TransfusionLiposomesLung Diseases, Fungal/drug therapyMaleTomography, Emission-ComputedTreatment Outcome
Identifiers
PMID: 9763555
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OZSAHIN, Ayse Hulya et al. Successful treatment of invasive aspergillosis in chronic granulomatous disease by bone marrow transplantation, granulocyte colony-stimulating factor-mobilized granulocytes, and liposomal amphotericin-B. In: Blood, 1998, vol. 92, n° 8, p. 2719-24. https://archive-ouverte.unige.ch/unige:74571

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Deposited on : 2015-08-10

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