The American journal of medicine · 2011
Interferon alpha and pamidronate in osteoporosis with fracture secondary to mastocytosis.
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Laroche Michel, Livideanu Christina, Paul Carle, Cantagrel Alain
Аннотация
BACKGROUND: The mechanism of osteoporosis with fracture secondary to mastocytosis is little known, and its treatment is poorly codified.
METHODS: Ten patients with a mean age of 52.5 years with systemic mastocytosis and osteoporotic fractures were treated with interferon alpha 1.5 million U 3 times per week, combined with monthly pamidronate infusions (1 mg/kg) for 2 years, followed by pamidronate infusions every 3 months.
RESULTS: Before treatment, the mean number of vertebral fractures was 3.5, spinal T-score was -3±1, hip T-score was -1.9±0.7, serum C-terminal telopeptide was 357±258 pg/mL (N=80-800), bone alkaline phosphatase was 20±3.2 IU (N=8-25), and tryptase was 49±36 μg/mL (N<10). Interferon alpha was discontinued in 2 patients because of poor tolerance. Mean follow-up was 60 months. No patient developed a fracture under treatment. In the 8 patients treated with interferon alpha and pamidronate, the mean annual increase in spinal bone mineral density was 12.6%±5.6% and 1.93% in hip bone mineral density. Serum C-terminal telopeptide decreased by 66%, bone alkaline phosphatase decreased by 25%, and tryptase decreased by 34%. In the 2 patients treated with pamidronate alone, mean annual bone mineral density increase was 2.4%±0.1% at the spine and 0%±01% at the hip.
CONCLUSION: Osteoporosis secondary to mastocytosis mainly affects trabecular bone, and markers of bone remodeling are normal. Combined treatment with low doses of interferon and pamidronate markedly increased bone density.
METHODS: Ten patients with a mean age of 52.5 years with systemic mastocytosis and osteoporotic fractures were treated with interferon alpha 1.5 million U 3 times per week, combined with monthly pamidronate infusions (1 mg/kg) for 2 years, followed by pamidronate infusions every 3 months.
RESULTS: Before treatment, the mean number of vertebral fractures was 3.5, spinal T-score was -3±1, hip T-score was -1.9±0.7, serum C-terminal telopeptide was 357±258 pg/mL (N=80-800), bone alkaline phosphatase was 20±3.2 IU (N=8-25), and tryptase was 49±36 μg/mL (N<10). Interferon alpha was discontinued in 2 patients because of poor tolerance. Mean follow-up was 60 months. No patient developed a fracture under treatment. In the 8 patients treated with interferon alpha and pamidronate, the mean annual increase in spinal bone mineral density was 12.6%±5.6% and 1.93% in hip bone mineral density. Serum C-terminal telopeptide decreased by 66%, bone alkaline phosphatase decreased by 25%, and tryptase decreased by 34%. In the 2 patients treated with pamidronate alone, mean annual bone mineral density increase was 2.4%±0.1% at the spine and 0%±01% at the hip.
CONCLUSION: Osteoporosis secondary to mastocytosis mainly affects trabecular bone, and markers of bone remodeling are normal. Combined treatment with low doses of interferon and pamidronate markedly increased bone density.