A case of SAPHO syndrome initially diagnosed as diffuse sclerosing osteomyelitis of the mandible and effectively treated by bisphosphonate

Abstract

Etiology of diffuse sclerosing osteomyelitis (DSO) of the mandible is still controversy. Whereas symptoms of DSO tend to recur after administration of antibiotics or surgery, an alternative hypothesis that DSO is a metabolic disorder due to osteoclasts has been proposed. We report a case of DSO effectively treated with pamidronate, a Bisphosphonate (BP), and finally diagnosed as SAPHO (synovitis, acne, pustulosis, hyperostosis, osteitis) syndrome after 10 years of follow-up period.
The patient is a 32-year-old male with DSO of the mandible showing resistance to long-term administration of antibiotics and hyperbaric oxygen therapy (HBO). After intravenous administration of pamidronate, pain disappeared completely, and the remarkable improvement of the bone structure of the mandible was confirmed by CT examination 5 months after the BP administration.
After the 10 years of follow-up period, bone scintigram showed high-uptake at sternoclavicular joint as well as sacroiliac joint, and the patient was diagnosed as SAPHO syndrome. It is necessary to take that DSO is a part symptom of the SAPHO syndrome into consideration.