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Vânia Sousa1, João Farela Neves1, Ana Fernandes2, Luís Varandas1, Rosário Malheiro2, Marta Conde3.

1 - Unidade de Infecciologia Pediátrica, Hospital Dona Estefânia, Centro Hospitalar Lisboa Central, E.P.E.;
2 - Serviço de Estomatologia Pediátrica, Hospital Dona Estefânia, Centro Hospitalar Lisboa Central, E.P.E.;
3 - Unidade de Reumatologia Pediátrica, Hospital Dona Estefânia, Centro Hospitalar Lisboa Central, E.P.E.

 - The 30th Annual Meeting of the European Society for Paediatrics Infectious Diseases (ESPID). Thessaloniki, Greece, May 2012.

Introduction: Chronic non-bacterial osteomyelitis is a rare auto-inflammatory disorder which typically occurs during childhood. Most frequently has multifocal bone involvement. Cases with unifocal involvement have been reported making it necessary to exclude malignancy and infection. It affects mainly the metaphysis of the long bones and mandibular involvement is uncommon.

Case report: A 12-year-old boy, previously diagnosed with Dandy-Walker Syndrome presented with pain and swelling in his left mandible starting two months before without dental foci. Despite two antibiotic courses (oral and intravenous) bony enlargement showed progressive worsening. Erythrocyte sedimentation rate was 39mm/h, CT scan revealed lytic and sclerotic lesions of the cortical bone and tc-99 bone scintigraphy increase of bone turnover both in the left side of mandible without additional bone foci suggesting neoplastic or infectious disease. Histopathologic findings of the involved bone were consistent with chronic osteomyielitis. Extensive microbial investigation (blood and biopsy tissue) was negative. Antituberculous treatment was started without improvement and stopped after negative bone PCR for Mycobacterium tuberculosis and Lowenstein culture. With exclusion of infection and malignancy the patient was started on indometacin resulting in impressive clinical and laboratory improvement.

Discussion: At initial presentation, chronic non-bacterial osteomyelitis may mimic acute hematogenous osteomyelitis or malignancy. The definite diagnosis is made after excluding these conditions especially if it presents in an unusual bone and with a single lesion. Prompt diagnosis of chronic non-bacterial osteomyelitis will allow patients to avoid the risks associated with lengthy courses of antibiotic therapy and repeated bone biopsies, which can delay the appropriate therapy.

Key-words: chronic non-bacterial osteomyelitis, mandible, unifocal.