Successful treatment of invasive aspergillosis in chronic granulomatous disease by bone marrow transplantation, granulocyte colony-stimulating factor-mobilized granulocytes, and liposomal amphotericin-B
Details
Serval ID
serval:BIB_1C6E6BE7B9BD
Type
Article: article from journal or magazin.
Publication sub-type
Case report (case report): feedback on an observation with a short commentary.
Collection
Publications
Institution
Title
Successful treatment of invasive aspergillosis in chronic granulomatous disease by bone marrow transplantation, granulocyte colony-stimulating factor-mobilized granulocytes, and liposomal amphotericin-B
Journal
Blood
ISSN
0006-4971
Publication state
Published
Issued date
10/1998
Peer-reviewed
Oui
Volume
92
Number
8
Pages
2719-24
Notes
Case Reports
Journal Article --- Old month value: Oct 15
Journal Article --- Old month value: Oct 15
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
Pubmed
Web of science
Create date
24/01/2008 17:16
Last modification date
20/08/2019 12:52