RT Journal Article
SR Electronic
T1 Anakinra in a Cohort of Children with Chronic Nonbacterial Osteomyelitis
JF The Journal of Rheumatology
JO J Rheumatol
FD The Journal of Rheumatology
SP 1231
OP 1238
DO 10.3899/jrheum.160690
VO 44
IS 8
A1 Manuela Pardeo
A1 Denise Pires Marafon
A1 Virginia Messia
A1 Maria Carmen Garganese
A1 Fabrizio De Benedetti
A1 Antonella Insalaco
YR 2017
UL http://www.jrheum.org/content/44/8/1231.abstract
AB 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.