Abstract
ObjectiveThe aim of this study was to evaluate demographic, clinical, laboratory, imaging, histopathology characteristics, and treatment responses of children with Chronic nonbacterial osteomyelitis (CNO).MethodsRetrospective multi-center case series study of pediatric patients diagnosed with CNO treated at five tertiary centers in south China.ResultsTotally there were 18 patients diagnosed as CNO between 2014 and 2020. The median age of onset was 9.2 years (range 3.7–13.1) and 55.6% were female. Median delay in diagnosis was 10.9 months (range 1.0–72.0). The most frequent presenting symptoms were bone pain (100%) and fever (44.4%). Most patients had more than one lesion (median of 5, range 1–7). Most frequently affected bones were tibiofibula (88.9%) and femur (77.8%). The MRI characteristics mainly presented as bone edema and hyperintensity in bone marrow. Bone biopsy was conducted in 11 patients (61.1%) with inflammatory cells infiltration manifested as chronic osteomyelitis, and none showed bacterial infection or tumor. In treatment, non-steroid anti-inflamatory drugs (NSAIDs) is used as the first-line drug followed by steriods, methotexate (MTX), salazosulfadimidine (SASP), Bisphosphonates and TNF-α inhibitor. Two refractory cases received combination therapy with Bisphosphonates and TNF-α inhibitor, and achieved good therapeutic effect.ConclusionsThe present study described a multicenter series of CNO from south China and highlighted the clinical features, laboratory tests, imaging characteristics and treatment outcomes. Increasing awareness of this disease is important to decrease time to diagnosis, improve access to treatment, and reduce complications.
Highlights
Chronic nonbacterial osteomyelitis (CNO) is a benign and noninfectious autoinflammatory bone disease that mostly affects children and adolescents [1,2,3]
The term SAPHO syndrome is commonly used when referring to manifestations of the disease, which include synovitis, acne, pustulosis, hyperostosis, and osteitis [10]
CNO is usually diagnosed by exclusion with magnetic resonance imaging (MRI) and/or through bone biopsy, which reveals chronic inflammation without infectious and oncological agents
Summary
Chronic nonbacterial osteomyelitis (CNO) is a benign and noninfectious autoinflammatory bone disease that mostly affects children and adolescents [1,2,3]. Whether CNO and SAPHO syndrome are different manifestations of the same disease at different ages of onset or different outcomes of different clinical manifestations of the same disease remains unresolved [11]. A study conducted in Germany reported an incidence of 0.4 per 100,000 children [13]. The true incidence of CNO has likely been underestimated in past studies [14]. CNO is usually diagnosed by exclusion with magnetic resonance imaging (MRI) and/or through bone biopsy, which reveals chronic inflammation without infectious and oncological agents. The treatment protocol for CNO includes NSAIDs, steroids, MTX, SASP, TNF-α inhibitors, and bisphosphonates
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