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Pediatric Rheumatology

, 15:56

First Online: 11 July 2017Received: 13 March 2017Accepted: 03 July 2017


BackgroundChronic nonbacterial osteomyelitis CNO is a multifocal autoinflammatory disease that often impairs daily life in children. This study aimed to investigate the bone metabolic and inflammatory characteristics of patients with CNO, and to assess the differences between responders and nonresponders to conservative treatment.

MethodsWe investigated the clinical symptoms; laboratory data including inflammatory and bone metabolic biomarkers; and imaging findings from plain radiography, magnetic resonance imaging MRI, fluorodeoxyglucose-positron emission tomography FDG-PET, and dual-energy x-ray absorption DEXA in 14 patients with CNO. All patients underwent first-line treatment comprising systemic nonsteroidal anti-inflammatory drugs with or without bisphosphonate. According to the response to the first-line treatment, the patients were divided into the clinical remission-partial response group and the no response group. The differences in bone metabolic and inflammatory characteristics between the two groups were assessed.

ResultsAll patients had low bone mineral density assessed with DEXA. The bone metabolic biomarkers bone-specific alkaline phosphatase and tartrate-resistant acid phosphatase 5b were increased in boys of all ages and in young girls. Multiple inflammatory regions were detected in all patients by using FDG-PET including asymptomatic regions. The no response group had higher immunoglobulin G IgG and a greater number of bone inflammatory lesions detected on MRI than the clinical remission-partial response group.

ConclusionOur data indicate the involvement of abnormal bone turnover, necessity of whole-body scanning, and association of higher serum IgG levels and greater numbers of inflammatory lesions with prolonged disease activity in patients with CNO.

KeywordsChronic nonbacterial osteomyelitis Bone metabolism Magnetic resonance imaging Fluorodeoxyglucose-PET Inflammatory biomarker AbbreviationsBAPBone alkaline phosphatase

BMDBone mineral density

CNOChronic nonbacterial osteomyelitis

CRMOChronic recurrent multifocal osteomyelitis

CRPC-reactive protein

ESRErythrocyte sedimentation rate

FDG-PETFluorodeoxyglucose-positron emission tomography

IgGImmunoglobulin G

MRIMagnetic resonance imaging

SAASerum amyloid A

SUVStandardized uptake value

T2-STIRT2-signal intensity in short tau inversion recovery

TRAP5bTartrate-resistant acid phosphatase serum band 5


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Autor: Yurika Ata - Yutaka Inaba - Hyonmin Choe - Naomi Kobayashi - Jiro Machida - Naoyuki Nakamura - Tomoyuki Saito


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