Characteristics, Treatment Options, and Outcomes of Chronic Non-bacterial Osteomyelitis in Children
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Pediatric Rheumatology (G Martini, Section Editor)
Characteristics, Treatment Options, and Outcomes of Chronic Non-bacterial Osteomyelitis in Children Raphae¨l Kraus, MD* Ronald M. Laxer, MD Address * Hospital for Sick Children, University of Toronto, Toronto, Canada Email: [email protected]
* Springer Nature Switzerland AG 2020
This article is part of the Topical Collection on Pediatric Rheumatology Keywords Chronic non-bacterial osteomyelitis I Chronic recurrent multifocal osteomyelitis I Autoinflammation I Pediatrics
Abstract Purpose of review Chronic non-bacterial osteomyelitis (CNO) is an autoinflammatory bone disease most prevalent in children. In this review, we seek to highlight the most recent advances in the understanding of the pathophysiology, treatment, and outcomes of CNO. Recent findings Recent observations support a pathophysiologic framework involving imbalanced expression of pro-inflammatory versus anti-inflammatory cytokines, thereby inducing increased differentiation and activation of osteoclasts. Although no medication nor protocol is yet specifically indicated in the treatment of CNO, there exists an expanding body of evidence for agents exerting their effects via interference with these pro-inflammatory pathways (namely non-steroidal anti-inflammatory drugs, diseasemodifying anti-rheumatic drugs, bisphosphonates, and biologics), highlighted herein. Summary With an incidence rivaling that of bacterial osteomyelitis, childhood CNO represents an important and growing field of study. Once thought to be benign and self-limiting, CNO is now recognized to follow a relapsing and remitting course in the majority of children, and to carry substantial risk of irreversible growth and bone sequelae. Perhaps of greatest importance to the study of CNO treatment is the recent development of the Childhood Arthritis and Rheumatology Research Alliance (CARRA) consensus treatment plans, the implementation of which will allow for prospective comparative effectiveness studies and, ultimately, the development of standardized treatment protocols.
Pediatric Rheumatology (G Martini, Section Editor)
Introduction First described in 1972 by Gideon et al. as “subacute and chronic symmetrical osteomyelitis,” chronic nonbacterial osteomyelitis (CNO) is an autoinflammatory bone disease most prevalent in children [1]. While terminology remains inconsistent across the literature, we apply CNO to describe a spectrum of sterile bone inflammation irrespective of chronicity and lesional distribution, with chronic recurrent multifocal osteomyelitis (CRMO; implying chronicity and/or recurrence and multiple sites of involvement) as its most severe form [2•, 3••]. Primarily affecting the metaphyses of long bones, clavicles, mandible, pelvic bones, and (of particular clinical importance) vertebral bodies, CNO generally presents with localized bone pain and imaging findings of osteolysis, sclerosis, and hyperostosis on plain radiographs and bone marrow edema on magnetic resonance imaging (MRI) [2•, 4, 5]. One recent study
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