Bone and Mineral Medicine, Department of Endocrinology and Diabetes, Children's Hospital at Westmead, Hawkesbury Rd, Westmead NSW 2145, Australia.
Bone. 2010 Apr;46(4):885-8. doi: 10.1016/j.bone.2009.11.034. Epub 2009 Dec 5.
Complex regional pain syndrome (CRPS) is a disorder that can cause significant functional morbidity. While it usually presents in adulthood, it has also been reported in children. Multiple treatment modalities have been reported with mixed success. Bisphosphonate therapy has been shown to be effective in adult patients, but there are limited data in children. We report the successful use of intravenous pamidronate therapy in diminishing pain, improving function, and restoring bone mass in an 11-year-old girl with CRPS of her left lower limb following a tibial fracture. Previous treatment with intense physiotherapy and regional sympathetic blockade had not improved her symptoms. Pain improved within weeks of the first pamidronate infusion, with subsequent improvement in function. The benefit in pain reduction and function was sustained during the 2-year treatment regime. Improvement in bone mass and density was demonstrated by dual-energy X-ray absorptiometry (DXA) and peripheral quantitative computerised tomography (pQCT). pQCT scans showed marked improvement in bone size and geometry and muscle bulk on the affected side. No adverse affects were reported. We conclude that intravenous pamidronate was associated with reduced pain, a return of function, and recovery of bone and muscle parameters in a child with CRPS. Before definitive conclusions can be drawn, a randomised controlled trial similar to those undertaken in adults previously is required to fully validate this approach.
复杂性区域疼痛综合征 (CRPS) 是一种可导致显著功能障碍的疾病。尽管它通常发生在成年期,但也有儿童发病的报道。已有多种治疗方法被报道,疗效不一。双膦酸盐治疗已被证明对成年患者有效,但在儿童中数据有限。我们报告了一例成功使用静脉帕米膦酸盐治疗的病例,一名 11 岁女孩左下肢胫骨骨折后发生 CRPS,静脉帕米膦酸盐治疗减轻了疼痛、改善了功能并恢复了骨量。此前强烈的物理治疗和区域交感神经阻滞治疗并未改善她的症状。在第一次帕米膦酸盐输注后的数周内,疼痛得到改善,随后功能得到改善。在 2 年的治疗期间,疼痛减轻和功能改善的效果得以维持。双能 X 线吸收法 (DXA) 和外周定量计算机断层扫描 (pQCT) 显示骨量和密度有所改善。pQCT 扫描显示,受影响侧的骨大小和几何形状以及肌肉体积有明显改善。未报告不良反应。我们得出结论,静脉帕米膦酸盐治疗与儿童 CRPS 疼痛减轻、功能恢复以及骨和肌肉参数恢复有关。在得出明确结论之前,需要进行类似先前在成人中进行的随机对照试验,以充分验证这种方法。