Anakinra in a Cohort of Children with Chronic Nonbacterial Osteomyelitis

J Rheumatol. 2017 Aug;44(8):1231-1238. doi: 10.3899/jrheum.160690. Epub 2017 Jun 15.

Abstract

Objective: To report efficacy and safety in patients with chronic nonbacterial osteomyelitis (CNO) unresponsive to nonsteroidal antiinflammatory drugs (NSAID) and bisphosphonates and/or glucocorticoids treated with anakinra.

Methods: Nine patients (6 females) with refractory CNO were treated with anakinra for at least 6 months. We recorded, at baseline and after 6 months of treatment, clinical and laboratory features, and number and distribution of bone lesions detected by 99mTc-MDP bone scintigraphy. Disease activity was evaluated using a physician's global assessment (PGA).

Results: At baseline, 9/9 patients had mild to severe PGA. After 6 months of treatment, in 5 patients the PGA score was graded from none to minimal. At baseline, erythrocyte sedimentation rate (ESR) and C-reactive protein (CRP) were elevated in 8 out of 9 patients. After 6 months, 5/9 patients had normalized CRP and ESR and in all except 1, CRP and ESR decreased. Before starting anakinra, a total of 77 bone lesions were detected by bone scintigraphy. After 6 months of treatment of the 77 lesions, 42 had resolved and 35 were stable. In 7/9 patients, 20 new lesions appeared during treatment; 2 of these 7 patients were symptomatic. At the last followup visit (median 1.7 yrs, range 0.8-2.8), 6/9 patients maintained a PGA graded as none to minimal.

Conclusion: Anakinra is a possible therapeutic alternative in patients with refractory CNO. The practical significance of clinically silent bone lesions detected by bone scintigraphy remains to be established.

Keywords: ANAKINRA; CHRONIC NONBACTERIAL OSTEOMYELITIS; RESPONSE TO TREATMENT.

MeSH terms

  • Adolescent
  • Anti-Inflammatory Agents, Non-Steroidal / therapeutic use*
  • Child
  • Female
  • Humans
  • Interleukin 1 Receptor Antagonist Protein / therapeutic use*
  • Male
  • Osteomyelitis / drug therapy*
  • Retreatment
  • Retrospective Studies
  • Severity of Illness Index
  • Treatment Outcome

Substances

  • Anti-Inflammatory Agents, Non-Steroidal
  • Interleukin 1 Receptor Antagonist Protein