Manuela Pardeo1,2, Denise Pires Marafon3,4, Virginia Messia3,4, Maria Carmen Garganese3,4, Fabrizio De Benedetti3,4, Antonella Insalaco1,2. 1. From the Division of Rheumatology, and the Nuclear Medicine Unit, Ospedale Pediatrico Bambino Gesù IRCCS, Rome, Italy. manuela.pardeo@opbg.net antonella.insalaco@opbg.net. 2. M. Pardeo, MD, Division of Rheumatology, Ospedale Pediatrico Bambino Gesù IRCCS; D. Pires Marafon, MD, Division of Rheumatology, Ospedale Pediatrico Bambino Gesù IRCCS; V. Messia, MD, Division of Rheumatology, Ospedale Pediatrico Bambino Gesù IRCCS; M.C. Garganese, MD, Nuclear Medicine Unit, Ospedale Pediatrico Bambino Gesù IRCCS; F. De Benedetti, MD, PhD, Division of Rheumatology, Ospedale Pediatrico Bambino Gesù IRCCS; A. Insalaco, MD, Division of Rheumatology, Ospedale Pediatrico Bambino Gesù IRCCS. manuela.pardeo@opbg.net antonella.insalaco@opbg.net. 3. From the Division of Rheumatology, and the Nuclear Medicine Unit, Ospedale Pediatrico Bambino Gesù IRCCS, Rome, Italy. 4. M. Pardeo, MD, Division of Rheumatology, Ospedale Pediatrico Bambino Gesù IRCCS; D. Pires Marafon, MD, Division of Rheumatology, Ospedale Pediatrico Bambino Gesù IRCCS; V. Messia, MD, Division of Rheumatology, Ospedale Pediatrico Bambino Gesù IRCCS; M.C. Garganese, MD, Nuclear Medicine Unit, Ospedale Pediatrico Bambino Gesù IRCCS; F. De Benedetti, MD, PhD, Division of Rheumatology, Ospedale Pediatrico Bambino Gesù IRCCS; A. Insalaco, MD, Division of Rheumatology, Ospedale Pediatrico Bambino Gesù IRCCS.
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.
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.
Entities:
Keywords:
ANAKINRA; CHRONIC NONBACTERIAL OSTEOMYELITIS; RESPONSE TO TREATMENT