Chronic mandibular osteomyelitis with suspected underlying synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome: a case report

Abstract

Chronic mandibular osteomyelitis is an intractable disease. In recent years, some case reports have related this disease process to synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome, which is chronic with frequent remissions and exacerbations. This report describes a case of chronic mandibular osteomyelitis suspected to be SAPHO syndrome. A 68-year-old woman presented with pain on the left side of the mandible. On the basis of clinical and radiological findings, chronic mandibular diffuse sclerosing osteomyelitis was initially diagnosed. We administrated oral clarithromycin (400 mg daily) and levofloxacin (500 mg daily), and her pain subsequently resolved. On 99mTc-labeled methylene diphosphonate scintigraphy, tracer uptake in the asymptomatic mandible was unchanged, but there was increasing tracer uptake in the sternocostal and sternoclavicular joints, compared with 99mTc-labeled methylene diphosphonate scintigraphic findings of the first visit. We diagnosed SAPHO syndrome and administrated oral sodium risedronate hydrate (2.5 mg daily). Although there has been no pain or swelling in the area of the left mandibular lesion, we have followed up on other skin and osteoarticular manifestations in conjunction with other medical departments.

Strontium ranelate improves delayed healing of osteolytic lesions of the jaw in a man with chronic osteomyelitis. Case report

“…In cases due to bacterial infection, antibiotics alone or in combination with various surgical procedures are considered the treatment of choice (1). When SAPHO syndrome is suspected, notably in cases of sclerosing osteomyelitis, bisphosphonates as well as corticosteroids have been rarely reported as additional therapy (20). It is also well known that teriparatide has been used in several cases of bisphosphonate-related osteonecrosis of the jaw with positive results (18,19).…”

Implication of bisphosphonate use in the treatment of SAPHO syndrome: Case report and discussion of current literature

“…To date its etiology has remained unknown [2][3][4][5] SAPHO syndrome is a rheumatic disease belonging to the group of spondyloarthropathies and combining osteomyelitis, osteitis, arthritis and skin disease [1,[5][6][7][8][9]. Its diagnosis can be made with the aid of three diagnostic criteria classified by Kahn et al [10] ( Table 1).…”

SAPHO Syndrome as a Potential Cause of Chronic Mandibular Bone Inflammation and Temporomandibular Joint Dysfunction – Case Report

“…The incidence of not more than 1/10,000 is reported in literature, but this data seems underestimated [4][5][6][7][8].…”

“…Most often it affects children (CRMO), young adults, and middle-aged people [4][5][6][7][8][9]. It rarely appears later than in the 6th decade [5][6][7]9].…”

“…They are often accompanied with painful edema of the surrounding soft tissues [9]. A change affecting the mandibular bone can be isolated or coexist with osseous manifestations in other parts of the skeleton [4,7,11,16,20].…”