Pediatric Chronic Nonbacterial Osteomyelitis of the Jaw: Clinical, Radiographic, and Histopathologic Features

Publication date: Available online 26 May 2016
Source:Journal of Oral and Maxillofacial Surgery
Author(s): Bonnie L. Padwa, Kelley Dentino, Caroline D. Robson, Sook Bin Woo, Kyle Kurek, Cory M. Resnick
PurposeChronic nonbacterial osteomyelitis (CNO) is a focal sterile inflammatory osteitis in children that most commonly develops in the long bones but can occur in any bone. The disease course is variable, ranging from acute and self-resolving isolated lesions to chronic recurrent multifocal osteomyelitis (CRMO), which is frequently associated with extraosseous inflammatory disease. The purpose of this study was to present our clinical experience with CNO of the mandible in children. The specific aims were to (1) document the clinical characteristics, radiographic findings and histological features of CNO and (2) determine the percentage of our sample with multifocal disease (CRMO).Materials and MethodsThis is retrospective case series of patients with mandibular CNO. To be included, patients had to have a mandibular lesion radiographically consistent with osteomyelitis without infection, onset before 18 years-of-age and complete records. Medical records were reviewed for history, clinical features, imaging, and pathology. Descriptive data were summarized.ResultsThe sample included 22 subjects (13 females and 9 males) with disease onset at a mean age of 9.05 + 2.4 years. On presentation, all subjects reported mandibular pain and swelling, and 45% had trismus. All had clinical and/or radiographic findings of multifocal intraosseous disease and/or extraosseous inflammatory lesions. Twelve (54%) had a documented family history of auto-immune or auto-inflammatory disease and 15 (68%) had elevated erythrocyte sedimentation rates during a flare. CT scans typically revealed expansion of the affected mandible with sclerosis of the medullary space, small foci of poorly-defined lytic destruction with a lamellated periosteal reaction, and swollen muscles of mastication. Four distinct histological features were noted including parallel and interconnected osteoid seams, atypical osteoid, areas of woven bone and hypocellular fibroblastic stroma resembling fibrous dysplasia, and patchy nodular fibrosis.ConclusionPediatric CNO of the mandible has characteristic radiographic and pathologic features and is usually found as one of multiple disease foci in chronic recurrent multifocal osteomyelitis (CRMO) rather than as an isolated lesion.



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