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Reumatol Clin. 2017 May 19. pii: S1699-258X(17)30090-6. doi: 10.1016/j.reuma.2017.04.002. [Epub ahead of print]

Recurrent multifocal osteomyelitis in children: Experience in a tertiary care center.

[Article in English, Spanish]

Author information

1
UGC Pediatría, Hospital Materno-Infantil de Málaga, Málaga, España. Electronic address: micodemas@hotmail.com.
2
Unidad de Reumatología Pediátrica, Hospital Materno-Infantil de Málaga, Málaga, España.
3
UGC Pediatría, Hospital Materno-Infantil de Málaga, Málaga, España.
4
UGC Reumatología, Hospital Regional Universitario de Málaga, Málaga, España.

Abstract

INTRODUCTION:

Chronic recurrent multifocal osteomyelitis is a rare aseptic bone inflammation that affects pediatric patients. Its management and treatment have not yet been standardized.

METHODS:

Retrospective, descriptive study of patients under 14 years of age diagnosed with chronic nonbacterial osteomyelitis (CNBO) in a tertiary hospital. We included patients diagnosed over the last 6 years (2010-2015) who met the Jansson criteria. The clinical and radiological characteristics of CNBO were analyzed, as was the outcome after different therapeutic options.

RESULTS:

We report 12 patients, with a mean age of 11 years (±1.6 standard deviation [SD]) and female predominance (10:2). The mean number of foci was 3.5 (±2.2 SD). The most common locations were ankle (58%), clavicle (50%), sternum (33%) and hip (25%). The mean disease duration was 10.5 months (±10.3 SD), and the median time to diagnosis was 2.38 months (range 0.17-16). Bone scintigraphy detected asymptomatic foci in 33% and we detected lytic lesions in 50% through magnetic resonance imaging. Biopsy was performed in 60%; 2/12 (16%) were associated with inflammatory disease and 1/12 (8.3%) later developed lymphoma. In all, 58% received antibiotic therapy with little response, 100% anti-inflammatory agents, 50% systemic corticosteroids, 41.6% methotrexate/pamidronate and 16% anti-tumor necrosis factor (TNF) α. The mean duration of treatment was 14.8 months (±12.4 SD) and 66% had recurrences. Currently, 83% are in clinical remission without treatment.

CONCLUSIONS:

When CNBO is refractory to treatment with anti-inflammatory drugs, intravenous pamidronate can be an alternative. Anti-TNF drugs can be considered in patients who fail with pamidronate, as can agents associated with other autoimmune conditions.

KEYWORDS:

Children; Chronic recurrent multifocal osteomyelitis; Niños; Nonbacterial osteomyelitis; Osteomielitis multifocal crónica recurrente; Osteítis no bacteriana; Pamidronate; Pamidronato; Tratamiento antifactor de necrosis tumoral alfa; Tumor necrosis factor α inhibitor therapy

PMID:
28528870
DOI:
10.1016/j.reuma.2017.04.002
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