Clinical characteristics and predictors for recurrence in chronic nonbacterial osteomyelitis: a retrospective multicenter analysis

Authors: KADİR ULU, RANA İŞGÜDER, ŞERİFE GÜL KARADAĞ, ESRA BAĞLAN, GÜLŞAH KAVRUL KAYAALP, GÜLÇİN OTAR YENER, KÜBRA ÖZTÜRK, HAFİZE EMİNE SÖNMEZ, SEMANUR ÖZDEL, FERHAT DEMİR, BALAHAN MAKAY, ŞEVKET ERBİL ÜNSAL, BETÜL SÖZERİ, NURAY AKTAY AYAZ, MUSTAFA ÇAKAN

Abstract: Background/aim: Chronic nonbacterial osteomyelitis (CNO) is a rare disease of unknown etiology and most commonly occurs during childhood or adolescence. The purpose of this study is to collect data on the clinical features, outcomes, and management of the disease and to identify the factors affecting recurrence. Materials and methods: This is a retrospective multicenter cross-sectional study of pediatric patients diagnosed with CNO. A total of 87 patients with a diagnosis of CNO followed for at least 6 months in 8 pediatric rheumatology centers across the country between January 2010 and December 2021 were included in this study. Results: The study included 87 patients (38 girls, 49 boys; median age: 12.5 years). The median follow-up time was 20 months (IQR: 8.5?40). The median time of diagnostic delay was 9.9 months (IQR: 3?24). Arthralgia and bone pain were the most common presenting symptoms. Multifocal involvement was detected in 86.2% of the cases and a recurrent course was reported in one-third of those included in the study. The most commonly involved bones were the femur and tibia. Vertebrae and clavicles were affected in 19.5% and 20.6% of cases, respectively. The erythrocyte sedimentation rate (ESR) values of 60.9% of the patients were above 20 mm/h and the C-reactive protein values of 44.8% were above 5 mg/L. The remission rate was 13.3% in patients using nonsteroidal antiinflammatory drugs and 75.0% in those using biological drugs. Vertebral and mandibular involvement and high ESR values at the time of diagnosis were associated with recurrence. Conclusion: In this multicenter study, CNO with vertebral and mandibular involvement and high ESR at diagnosis were associated with recurrence.

Keywords: Chronic nonbacterial osteomyelitis, chronic recurrent multifocal osteomyelitis, autoinflammation, children

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