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Blood, Vol. 92 No. 8 (October 15), 1998:
pp. 2719-2724
Successful Treatment of Invasive Aspergillosis in Chronic
Granulomatous Disease by Bone Marrow Transplantation, Granulocyte
Colony-Stimulating Factor-Mobilized Granulocytes, and Liposomal
Amphotericin-B
Hülya Ozsahin,
Maya von Planta,
Irene Müller,
Hans C. Steinert,
David Nadal,
Roger Lauener,
Peter Tuchschmid,
Ulrich
V. Willi,
Mahmut Ozsahin,
Nigel E.A. Crompton, and
Reinhard A. Seger
From the Divisions of Immunology/Hematology and Radiology, University
Children's Hospital of Zurich; the Division of Nuclear Medicine, the
University Hospital of Zurich, Zurich, Switzerland; and the Department
of Life Sciences, Paul Scherrer Institute, Villigen, Switzerland.
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  -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.
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