Beneficial effect of long term intravenous bisphosphonate treatment of osteogenesis imperfecta

被引:177
作者
Åström, E
Söderhäll, S
机构
[1] Astrid Lindgren Childrens Hosp, Dept Woman & Child Hlth, Div Paediat Neurol, S-17176 Stockholm, Sweden
[2] Astrid Lindgren Childrens Hosp, Childhood Canc Res Unit, S-17176 Stockholm, Sweden
[3] Soder Sjukhuset, S-10064 Stockholm, Sweden
关键词
D O I
10.1136/adc.86.5.356
中图分类号
R72 [儿科学];
学科分类号
100202 ;
摘要
Aim: To find an effective symptomatic treatment for osteogenesis imperfecta (OI). Methods: In a prospective observational study disodium pamidronate (APD) was given as monthly intravenous infusions to 28 children and adolescents (aged 0.6-18 years) with severe OI or a milder form of the disease, but with spinal compression fractures. Results: During treatment for 2-9 years, dual energy x ray absorptiometry measurements of the total body and of the lumbar spine showed a gradual increase in bone density. All bone metabolism variables in serum (alkaline phosphatase, osteocalcin, Procollagen 1 C-terminal Peptide, collagen 1 teleopeptide) and urine (deoxypyridinoline) indicated that there was a decrease in bone turnover. All patients experienced beneficial effects and the younger patients reported a major improvement in wellbeing, pain, and mobility without significant side effects. Vertebral remodelling was also seen. Conclusions: APD seems to be an efficient symptomatic treatment for children and adolescents with OI.
引用
收藏
页码:356 / 364
页数:9
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