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 :
Section of Oral and Maxillofacial Surgery
Faculty of Dental Science
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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.
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.