Gleeson Helena, Wiltshire Esko, Briody Julie, Hall Jill, Chaitow Jeff, Sillence David, Cowell Christopher, Munns Craig
Institute of Endocrinology and Diabetes, Children's Hospital at Westmead, Sydney, Australia.
J Rheumatol. 2008 Apr;35(4):707-12. Epub 2008 Mar 15.
Chronic relapsing multifocal osteomyelitis (CRMO) results in significant morbidity, especially in those with vertebral collapse. Symptomatic benefit with intravenous pamidronate (PAM) has been shown; however, few studies have demonstrated radiological benefit. We describe clinical and radiological data on 7 pediatric cases of CRMO treated with PAM.
Retrospective chart review on all children with CRMO treated with PAM. Response to PAM was measured by subjective reports and radiology including vertebral morphometry.
Seven patients (1 male) presented with bone pain at a median age of 8 years (range 5-14). Symptoms had been present for a median of 18 months (range 11-51) before PAM therapy. All patients had involvement of multiple nonspinal sites, 5 children had spinal involvement with vertebral fractures, and 5 had joint involvement. Six cases had symptomatic improvement within 6 months of starting PAM, which was sustained during PAM therapy (median 26 mo, range 6-41) and persisted in the 4 cases who had ceased treatment for the duration of followup (27 mo, range 18-51). The least benefit was seen in the 3 cases with synovial joint involvement. The 3 cases with spinal radiological followup showed modeling of vertebral fractures and in one patient improvement in kyphosis. No radiological improvement in nonspinal lesions was seen.
PAM therapy was associated with symptomatic improvement and vertebral modeling in children with CRMO. We suggest that children with bone pain and/or spinal involvement be considered for PAM therapy early after diagnosis.
慢性复发性多灶性骨髓炎(CRMO)会导致严重的发病率,尤其是在那些发生椎体塌陷的患者中。已显示静脉注射帕米膦酸盐(PAM)有症状改善作用;然而,很少有研究证明其有放射学改善作用。我们描述了7例接受PAM治疗的儿童CRMO患者的临床和放射学数据。
对所有接受PAM治疗的CRMO儿童进行回顾性病历审查。通过主观报告和包括椎体形态测量在内的放射学检查来评估对PAM的反应。
7例患者(1例男性),中位年龄8岁(范围5 - 14岁),表现为骨痛。在接受PAM治疗前,症状出现的中位时间为18个月(范围11 - 51个月)。所有患者均有多个非脊柱部位受累,5例儿童有脊柱受累伴椎体骨折,5例有关节受累。6例患者在开始PAM治疗后6个月内症状改善,并在PAM治疗期间持续改善(中位时间26个月,范围6 - 41个月),且在4例停止治疗的患者随访期间(27个月,范围18 - 51个月)仍持续改善。滑膜关节受累的3例患者获益最少。3例接受脊柱放射学随访的患者显示椎体骨折塑形,1例患者脊柱后凸改善。非脊柱病变未见放射学改善。
PAM治疗与CRMO儿童的症状改善和椎体塑形有关。我们建议,对于诊断后早期出现骨痛和/或脊柱受累的儿童应考虑PAM治疗。