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Mandibular involvement in SAPHO syndrome: a retrospective study
Mu Wang1, Yueting Li2, Yihan Cao3
1Department of Stomatology, Peking Union Medical College Hospital, Peking Union Medical College, Chinese Academy of Medical Sciences, Beijing, 100730, China.
Orphanet Journal of Rare Diseases
|November 6, 2020
Summary
Mandible osteomyelitis in Synovitis, Acne, Pustulosis, Hyperostosis, and Osteitis (SAPHO) syndrome primarily affects young women. Conservative treatments, particularly bisphosphonates, show promise for managing this rare condition.
Area of Science:
- Rheumatology
- Oral and Maxillofacial Surgery
- Dermatology
Background:
- Synovitis, Acne, Pustulosis, Hyperostosis, and Osteitis (SAPHO) syndrome is a rare chronic inflammatory disorder.
- Mandibular osteomyelitis is an uncommon manifestation of SAPHO syndrome, with limited research on its characteristics and management.
Purpose of the Study:
- To investigate the clinical characteristics, risk factors, and treatment outcomes of mandibular involvement in SAPHO syndrome.
- To evaluate the efficacy of conservative management, including bisphosphonate therapy, for SAPHO-related mandibular osteomyelitis.
Main Methods:
- Retrospective review of 26 SAPHO patients with mandibular involvement diagnosed between September 2014 and July 2019.
- Collection of demographic, clinical, laboratory, and imaging data; analysis of prescription and follow-up MRI/CBCT data.
- Utilized an electronic questionnaire to assess current symptoms and treatment response.
Main Results:
- The study included 26 patients (38.5% male, median age 28 years) with a median follow-up of 2.1 years.
- Oral procedures preceded mandibular symptoms in 38.5% of patients; surgical intervention led to relapse in all 14 patients.
- Conservative treatment improved symptoms in 92.3% of patients, with bisphosphonates achieving remission of bone marrow edema and osteolysis on imaging.
Conclusions:
- Mandibular SAPHO syndrome predominantly affects young women, with dental procedures as a potential risk factor.
- Conservative treatment, especially intravenous bisphosphonates, is effective in managing mandibular osteomyelitis in SAPHO syndrome.
- Bisphosphonate therapy demonstrated sustained remission in patients with follow-up imaging, suggesting its therapeutic potential.
