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 (99m)Tc-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 (99m)Tc-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.

Keywords: 14-membered ring macrolide antibiotics; SAPHO syndrome; bisphosphonates; diffuse sclerosing osteomyelitis; new quinolone antibiotics.

Figure 1

Panoramic radiograph at first visit, showing a ground-glass appearance (yellow arrows) from the left premolar to the mandibular ramus region and enlarged canals of the left mandible (red arrow).

Figure 2

Plain computed tomography at first visit revealed increased density of cancellous bone.

Figure 3

Magnetic resonance imaging at first visit. Note: The left side of the ascending ramus of the mandible shows heterogeneous intermediate-to-high signal intensity on fat-suppressed T₂-weighted images.

Figure 4

Bone scintigram (^99mTc-labeled methylene diphosphonate) at first visit, showing extremely intense tracer uptake in the left side of the mandible, the sternum, and the sternocostal and sternoclavicular joints.

Figure 5

CT and MRI at 4-month follow-up after the first visit: (A) plain CT showing diffuse cortical bone resorption of the left condyle of the mandible; (B) MRI, with the left condyle of the mandible in part showing low-high signal intensity on a T₁-weighted image; (C) MRI, with soft tissue around the left side of the ascending ramus of the mandible showing heterogeneous high-signal intensity on a fat-suppressed T₂-weighted image. Abbreviations: CT, computed tomography; MRI, magnetic resonance imaging.

Figure 6

Bone scintigram 6 months after administration of antibiotics. Note: Radioisotope uptake in the left mandible is unchanged, while radioisotope uptake in the sternum and in the sternocostal and sternoclavicular joints is increased, compared with the images of the first visit.