Mandibular involvement in SAPHO syndrome: a retrospective study
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RESEARCH
Mandibular involvement in SAPHO syndrome: a retrospective study Mu Wang1†, Yueting Li3†, Yihan Cao4, Xinyu Lu5, Yuchen Liu5, Jizhi Zhao1*, Wen Zhang3* and Chen Li2*
Abstract Background: Mandible osteomyelitis can occur in synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome, a rare chronic inflammatory disease; however, few studies have explored its characteristics and management. Methods: We reviewed the medical records of consecutive SAPHO patients with mandible involvement diagnosed in Peking Union Medical College Hospital from September 2014 to July 2019. Demographic, clinical, laboratory, and imaging data were collected at baseline. Prescription data and follow-up magnetic resonance imaging (MRI) and cone beam computed tomography (CBCT) images were collected from the hospital information system. An electronic questionnaire was distributed to all patients to obtain their latest symptoms. Results: A total of 26 SAPHO patients with mandibular involvement were involved, all of whom responded to the questionnaire (38.5% male; median age, 28 years; median follow-up duration, 2.1 years). Ten patients (38.5%) had undergone an oral procedure 1 month before the onset of mandibular symptoms. All 14 of the patients who underwent a surgical intervention relapsed within a median duration of 2 months (range 0.25–4.0 months), and 24 patients (92.3%) achieved improvement with conservative treatment. Following bisphosphonate treatment, remission of bone marrow oedema and osteolysis was observed on MRI and CBCT, and 5 patients receiving bisphosphonates with follow-up CBCT after remission did not relapse in 5.4 months (mean 6.0, range 3.2–9.9 months). Conclusion: Mandibular involvement of SAPHO syndrome predominantly occurs in young women. Dental procedures are a possible risk factor. Conservative treatment, especially intravenous bisphosphonates, can lead to oral improvement. Keywords: Mandible, Osteitis, Cone-beam computed tomography, Magnetic resonance imaging, Diphosphonates, Etanercept
*Correspondence: [email protected]; [email protected]; [email protected] † Mu Wang and Yueting Li contributed equally to this manuscript 1 Department of Stomatology, Peking Union Medical College Hospital, Peking Union Medical College, Chinese Academy of Medical Sciences, Beijing 100730, China 2 Department of Traditional Chinese Medicine, Peking Union Medical College Hospital, Peking Union Medical College, Chinese Academy of Medical Sciences, Beijing 100730, China 3 Department of Rheumatology and Clinical Immunology, Peking Union Medical College Hospital, Clinical Immunology Center, Peking Union Medical College, Chinese Academy of Medical Sciences, Beijing 100730, China Full list of author information is available at the end of the article
Introduction Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome is a chronic inflammatory disease characterized by osteoarticular involvement with or without palmoplantar pustulosis (PPP) or severe acne (SA) [1]. This rare disease has
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