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Dental Journals | Dentistry Journals | Oral Health | High Impact Articles | Scient Open Access
Journal of Dental and Oral Health


Case Report

Pediatric Treatment-Resistant Nonbacterial Osteomyelitis of the Mandible Associated with SAPHO Syndrome

Yu Kamata, Tomohiro Yamada*, Tomoki Sumida, Hiroyuki Nakano, Goro Sugiyama, Azusa Nakashima and Yoshihide Mori

Correspondence Address :

Tomohiro Yamada
Section of Oral and Maxillofacial Surgery
Faculty of Dental Science
Kyushu University
Tel: +81-92-642-6452, Fax: +81-92-642-6392

Received on: July 03, 2017, Accepted on: July 10, 2017, Published on: July 17, 2017

Citation: Yu Kamata, Tomohiro Yamada, Tomoki Sumida, et al. (2017). Pediatric Treatment-Resistant Nonbacterial Osteomyelitis of the Mandible Associated with SAPHO Syndrome

Copyright: 2017 Tomohiro Yamada, et al. This is an open-access article distributed under the terms of the Creative Commons Attribution License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.

  • Abstract

SAPHO syndrome is a chronic disease of unknown etiology, which is characterized by synovitis, acne, pustulosis, hyperostosis, and osteitis, however, pediatric osteomyelitis of the jawbone is rarely reported. We present a report of pediatric nonbacterial osteomyelitis of the mandible associated with SAPHO syndrome.
A seven-year-old girl presented complaining of pain in, and swelling of, the right retromolar region. Antibiotics had no effect and her past history included linea alba, strabismus, Palmoplantar Pustulosis (PPP) and old fractures of the Thoracic Vertebrae (Th7-9). Together with histological findings, she was diagnosed as suffering from SAPHO syndrome with mandibular osteomyelitis.
NSAIDs (Naproxen), Corticosteroids (Dexamethasone), and Methotrexate (MTX) were effective for several months but the effects were transient. As the third line therapy, an anti- TNFα agent (infliximab) was administered in addition to MTX. The mandibular symptoms have subsequently been under control for over a year.
For treatment of mandible osteomyelitis with SAPHO syndrome, systemic immunosuppressive therapy should be considered, beside continuous oral management.