Korean J Pediatr.
2007 Nov;50(11):1129-1133. 10.3345/kjp.2007.50.11.1129.
Long-term recombinant interferon-gamma treatment in 2 cases of osteopetrosis
- Affiliations
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- 1Department of Pediatrics, Chonnam National University Hwasun Hospital, Chonnam National University Medical School Chonnam, Korea. hoonkook@chonnam.ac.kr
- 2Department of Orthopedic Surgery, Chonnam National University Hwasun Hospital, Chonnam National University Medical School Chonnam, Korea.
- KMID: 2279374
- DOI: http://doi.org/10.3345/kjp.2007.50.11.1129
Abstract
- Osteopetrosis, a rare osteosclerotic bone disease characterized by a defect in osteoclast function and the reduced generation of superoxide by leukocytes, can be classified into several types based on their mode of inheritance, age of onset, severity, and associated clinical symptoms. Stem cell transplantation is the only curative therapy for the infantile malignant type, although alternative treatments, such as corticosteroids, calcitriol, and interferon (IFN)-gamma have been attempted in patients with milder clinical types. In addition, IFN-gamma therapy has been reported to increase bone resorption and hematopoiesis and to improve leukocyte function. Here, we present the cases of two patients with osteopetrosis who benefited from either 3 or 6 years of INF-gamma therapy that resulted in improved blood counts and no further pathological fractures.
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