Chronic nonbacterial osteomyelitis associated with pediatric inflammatory bowel disease: A multicenter retrospective study from the Paediatric inflammatory bowel disease Porto Group of ESPGHAN.
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OBJECTIVES: Chronic nonbacterial osteomyelitis (CNO) is a rare, sterile autoinflammatory bone disorder that can develop in patients with inflammatory bowel disease (IBD). We aimed to identify the clinical features and natural history of patients with a dual diagnosis of CNO and IBD. METHODS: Medical records of patients with a dual diagnosis of IBD and CNO were reviewed in centers from the Paediatric IBD Porto Group and IBD Interest Group of European Society for Pediatric Gastroenterology, Hepatology, and Nutrition (ESPGHAN). Collected data included demographic characteristics, disease features, laboratory studies, bone imaging findings, and clinical outcomes. RESULTS: Forty-five patients (24 [53%] males), were included. Median age at the time of dual diagnosis was 10 (interquartile range [IQR]: 12-13) years. Thiry-two (71%) patients were diagnosed with Crohn's disease, and 14 (44%) of them exhibited perianal disease. CNO presented in 15 patients (33%) within 3 months of IBD diagnosis, and in additional 20 (44%) patients after IBD diagnosis. In most patients, CNO manifested while IBD was clinically active, but not necessarily. However, in 10 (22%) patients CNO preceded the diagnosis of IBD with a median time 46 (25-248) weeks. Upon diagnosis of CNO, most patients were treated with anti-tumor necrosis factor. CNO remission was achieved in all patients at some point during follow-up; However, complications occurred in six patients and included vertebral collapse, bone fracture, and bone deformity. CONCLUSIONS: CNO can be associated with active or quiescent intestinal inflammation, manifesting before, during, or after the diagnosis of IBD. While CNO remission was achieved in all patients, some developed significant bone complications.