Copyright
©The Author(s) 2025. Published by Baishideng Publishing Group Inc. All rights reserved.
Synovitis, acne, pustulosis, hyperostosis, and osteitis syndrome as a cause of pneumothorax: A case report
Zi-Xiang Zheng, Meng-Jiao Gu, Tian-Lun Kang, Yu-Ru Zhang, Yu-Nuo Wang, Chen Li, Yuan-Hao Wu
Zi-Xiang Zheng, Meng-Jiao Gu, Yu-Ru Zhang, Yu-Nuo Wang, Yuan-Hao Wu, Department of Rheumatology and Immunology, The First Teaching Hospital of Tianjin University of Traditional Chinese Medicine, Tianjin 300193, China
Zi-Xiang Zheng, Meng-Jiao Gu, Yu-Nuo Wang, Graduate School, Tianjin University of Traditional Chinese Medicine, Tianjin 300193, China
Zi-Xiang Zheng, Meng-Jiao Gu, Yu-Ru Zhang, Yu-Nuo Wang, Yuan-Hao Wu, Department of Rheumatology and Immunology, National Clinical Research Center for Chinese Medicine Acupuncture and Moxibustion, Tianjin 300193, China
Tian-Lun Kang, Department of Rheumatism, Dongfang Hospital, Beijing University of Chinese Medicine, Beijing 100000, China
Chen Li, Department of Chinese Medicine, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing 100730, China
Co-first authors: Zi-Xiang Zheng and Meng-Jiao Gu.
Co-corresponding authors: Chen Li and Yuan-Hao Wu.
Author contributions: Zheng ZX and Gu MJ collected the case content and wrote the manuscript; Kang TL and Zhang YR provided guidance on manuscript writing; Wang YN was responsible for finding relevant materials; Li C and Wu YH jointly proposed the overall direction of the topic. All authors have read and approved the final manuscript.
Supported by The National Natural Science Foundation of China, No. 82074246.
Informed consent statement: All study participants, or their legal guardian, provided informed written consent prior to study enrollment.
Conflict-of-interest statement: The authors declare no competing interests.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See:
https://creativecommons.org/Licenses/by-nc/4.0/ Corresponding author: Chen Li, Doctor, MD, Chief Doctor, Staff Physician, Department of Chinese Medicine, Peking Union Medical College Hospital, Chinese Academy of Medical Sciences and Peking Union Medical College, No. 9 Dongdansanlu, Dongcheng District, Beijing 100730, China.
casio1981@163.com
Received: September 9, 2024
Revised: December 4, 2024
Accepted: December 30, 2024
Published online: January 7, 2025
Processing time: 118 Days and 14.6 Hours
BACKGROUND
Synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome, a rare inflammatory disease, was described in 1987 by Chamot et al, who identified its common characteristics and transitional forms through case reports. The incidence rate is now 1 in 10000 among Caucasians and 0.00144 in 10000 among Japanese. Pneumothorax occurs when gas enters the pleural cavity, typically due to lung disease or chest trauma, and is characterized as either spontaneous or traumatic. Spontaneous pneumothorax is commonly linked to congenital lung tissue abnormalities or lung conditions, such as ruptured pulmonary bullae, emphysema, and bronchial asthma. Respiratory involvement in SAPHO syndrome has historically been noted.
CASE SUMMARY
We present the case of a 44-year-old male patient who had previously been diagnosed with SAPHO syndrome. Following a two-year cessation of methotrexate therapy, he experienced recurrent anterior chest wall and joint pain accompanied by dyspnea. We conducted a comprehensive examination for the patient, which included physical examination, laboratory tests, and imaging studies. The clinical presentation and treatment outcomes suggest that his pneumothorax was related to an episode of SAPHO syndrome. We performed closed thoracic drainage surgery for the patient and treated his symptoms with etoricoxib (60 mg daily). Upon discharge, methotrexate was prescribed again. In subsequent follow-ups, there was no recurrence of joint and respiratory symptoms observed in the patient.
CONCLUSION
SAPHO syndrome has been proposed as a transitional stage between ankylosing spondylitis and psoriatic arthritis. One study reported a 0.29% incidence of spontaneous pneumothorax in patients with ankylosing spondylitis, higher than in the general population. Additionally, 30% of patients with SAPHO syndrome test positive for HLA-B27, an antigen linked to ankylosing spondylitis. Symptomatically, some patients with SAPHO syndrome meet diagnostic criteria for ankylosing spondylitis, with sternoclavicular joint involvement and paravertebral ossifications resembling syndesmophytes in ankylosing spondylitis. These features suggest a possible linked between SAPHO syndrome and ankylosing spondylitis.
Core Tip: This case report introduces a novel association between synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome and pneumothorax. The 44-year-old patient’s diagnosis and treatment underscore the importance of recognizing this rare but serious condition, broadening our understanding of SAPHO syndrome’s clinical manifestations.
