Treatment of chronic recurrent multifocal osteomyelitis with bisphosphonates in children

Joint Bone Spine. 2019 Nov;86(6):783-788. doi: 10.1016/j.jbspin.2019.06.005. Epub 2019 Jun 16.

Abstract

Objectives: Assessment of bisphosphonates efficiency in the therapy of NSAIDs-refractory Chronic Recurrent Multifocal Osteomyelitis.

Methods: Retrospective analysis of records of patients treated for Chronic Recurrent Multifocal Osteomyelitis between 2012 and 2018.

Results: Between 2012 and 2018, 76 children and adolescents were diagnosed with Chronic Recurrent Multifocal Osteomyelitis in our department. All patients underwent an initial course of NSAIDs therapy that provided a remission in 46% of cases. Of 41 NSAIDs-resistant cases, 7 patients were male and 34 were female. Disease started meanly in the age of 10. Most frequently pain localised in foot, clavicle and hip. In presented group, pamidronate was administered intravenously in the dose of 1mg/kg/day for 3 days. Patients received 6 series (1-17 series) on average with mean interval of 10 weeks (4-14 weeks). Our observations demonstrated rapid decrease of symptoms intensity after first dose of pamidronate with relapse of pain after 3-4 weeks. The frequency of pamidronate dosage was dependent of patient's symptoms. No serious adverse effects were reported. We finished the therapy after complete remission of symptoms and complete bone remodelling in imaging. Of 41 patients, 32 achieved remission and 9 continue their therapy. In remission group patients received 7 series of pamidronate on average and their treatment lasted meanly 20 months.

Conclusions: Pamidronate is a safe and efficient method of CRMO therapy, particularly in cases refractory to NSAIDs treatment. Treatment with pamidronate provides both symptomatic relief as well as normalisation of bone morphology.

Keywords: Bisphosphonates; Chronic non-bacterial osteitis; Chronic recurrent multifocal osteomyelitis; Paediatric bone disorders; Pamidronate.

MeSH terms

  • Adolescent
  • Anti-Inflammatory Agents, Non-Steroidal / administration & dosage
  • Child
  • Cohort Studies
  • Diphosphonates / administration & dosage*
  • Dose-Response Relationship, Drug
  • Drug Administration Schedule
  • Female
  • Follow-Up Studies
  • Humans
  • Male
  • Osteomyelitis / diagnostic imaging*
  • Osteomyelitis / drug therapy*
  • Pamidronate / administration & dosage*
  • Radiography / methods
  • Retrospective Studies
  • Risk Assessment
  • Severity of Illness Index
  • Time Factors
  • Treatment Outcome

Substances

  • Anti-Inflammatory Agents, Non-Steroidal
  • Diphosphonates
  • Pamidronate

Supplementary concepts

  • Chronic recurrent multifocal osteomyelitis