Treatment with rhG-CSF for osteomyelitis in a patient with p47-phox-deficient chronic granulomatous disease

A 24-year-old woman with osteomyelitis was diagnosed as having p47-phox-deficient chronic granulomatous disease (CGD). The patient showed a marked deficiency of p47-phox, which is very rare in Japan. As the clinical response to various antibiotics including sulfamethoxazole-trimethoprim was not sati...

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Bibliographic Details
Published in:Annals of hematology 1997-11, Vol.75 (5-6), p.243-246
Main Authors: ISOTANI, H, FUKUMOTO, Y, KAWAMURA, H, SASADA, M, HATTORI, K, FUJIWARA, T, KOBAYASHI, Y
Format: Article
Language:English
Subjects:
Adult
Biological and medical sciences
Female
Granulocyte Colony-Stimulating Factor - therapeutic use
Granulomatous Disease, Chronic - complications
Granulomatous Disease, Chronic - genetics
Granulomatous Disease, Chronic - metabolism
Hematologic and hematopoietic diseases
Humans
Medical sciences
NADPH Oxidases
Osteomyelitis - complications
Osteomyelitis - drug therapy
Other diseases. Hematologic involvement in other diseases
Phosphoproteins - deficiency
Phosphoproteins - genetics
Recombinant Proteins - therapeutic use
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Summary:A 24-year-old woman with osteomyelitis was diagnosed as having p47-phox-deficient chronic granulomatous disease (CGD). The patient showed a marked deficiency of p47-phox, which is very rare in Japan. As the clinical response to various antibiotics including sulfamethoxazole-trimethoprim was not satisfactory, we added recombinant human granulocyte colony-stimulating factor (rhG-CSF) to the treatment protocol. We report the beneficial clinical course of the patient, together with the effect of rhG-CSF on the granulocyte function, and the present report indicates that rhG-CSF is useful for the treatment of antibiotic-resistant infection in the variant type of p47-phox-defective CGD.
ISSN:0939-5555
1432-0584
DOI:10.1007/s002770050351