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

Ozsahin, Ayse Hulya; Von Planta, Maya; Müller, I; Steinert, H C; Nadal, D; Lauener, R; Tuchschmid, P; Willi, U V; Ozsahin, M; Crompton, N E; Seger, R A

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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		Ozsahin, Ayse Hulya Von Planta, Maya Müller, I Steinert, H C Nadal, D Lauener, R Tuchschmid, P Willi, U V Ozsahin, M Crompton, N E Seger, R A show all authors [1 - 11]
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 use — Antifungal Agents/administration & dosage/therapeutic use — Apoptosis — Aspergillosis/drug therapy/prevention & control/radionuclide imaging/therapy — Aspergillus nidulans — Bone Marrow Transplantation — Child — Combined Modality Therapy — Drug Carriers — Graft Survival/drug effects — Granulocyte Colony-Stimulating Factor/therapeutic use — Granulocytes/physiology — Granulomatous Disease, Chronic/complications/therapy — Humans — Itraconazole/therapeutic use — Leukocyte Count — Leukocyte Transfusion — Liposomes — Lung Diseases, Fungal/drug therapy — Male — Tomography, Emission-Computed — Treatment Outcome
Identifiers		PMID: 9763555
Full text		Article (Published version) (189 Kb) - Limited access to UNIGE Other version: http://www.bloodjournal.org/content/92/8/2719.long?sso-checked=true
Structures		Faculté de médecine / Section de médecine clinique / Département de pédiatrie
Citation (ISO format)		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