Successful treatment of invasive aspergillosis in chronic granulomatous disease by bone marrow transplantation, granulocyte colony-stimulating factor-mobilized granulocytes, and liposomal amphotericin-B
| Autor: | Ozsahin, H., Planta, M., Müller, I., Steinen, H. C., Nadal, D., Roger Lauener, Tuchschmid, P., Willi, U. V., Ozsahin, M., Crompton, N. E. A., Seger, R. A. |
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| Jazyk: | angličtina |
| Rok vydání: | 1998 |
| Předmět: |
Granulomatous Disease
Chronic/complications/therapy Male Antifungal Agents Immunology Amphotericin B/administration & dosage/therapeutic use Apoptosis Granulomatous Disease Chronic Graft Survival/drug effects Biochemistry Aspergillus nidulans Leukocyte Count Granulocyte Colony-Stimulating Factor/therapeutic use Amphotericin B Granulocyte Colony-Stimulating Factor Aspergillosis Humans Child Lung Diseases Fungal/drug therapy Bone Marrow Transplantation Drug Carriers ddc:618 Lung Diseases Fungal Granulocytes/physiology Graft Survival Antifungal Agents/administration & dosage/therapeutic use Cell Biology Hematology Combined Modality Therapy Leukocyte Transfusion Treatment Outcome Aspergillosis/drug therapy/prevention & control/radionuclide imaging/therapy Liposomes Itraconazole/therapeutic use Itraconazole Granulocytes Tomography Emission-Computed |
| Zdroj: | Blood, Vol. 92, No 8 (1998) pp. 2719-24 Scopus-Elsevier |
| ISSN: | 0006-4971 |
| Popis: | 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 γ-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.© 1998 by The American Society of Hematology. |
| Databáze: | OpenAIRE |
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