Skip to main page content
U.S. flag

An official website of the United States government

Dot gov

The .gov means it’s official.
Federal government websites often end in .gov or .mil. Before sharing sensitive information, make sure you’re on a federal government site.

Https

The site is secure.
The https:// ensures that you are connecting to the official website and that any information you provide is encrypted and transmitted securely.

Access keys NCBI Homepage MyNCBI Homepage Main Content Main Navigation
Case Reports
. 2022 Sep 1;61(17):2671-2675.
doi: 10.2169/internalmedicine.8473-21. Epub 2022 Feb 8.

Synovitis-acne-pustulosis-hyperostosis-osteitis Syndrome with Bilateral Pleural Effusion

Affiliations
Case Reports

Synovitis-acne-pustulosis-hyperostosis-osteitis Syndrome with Bilateral Pleural Effusion

Maho Adachi-Katayama et al. Intern Med. .

Abstract

Pleural effusion is a rare manifestation in synovitis-acne-pustulosis-hyperostosis-osteitis (SAPHO) syndrome, which is characterized by the presence of osteoarticular lesions and dermatological involvement. We herein report a 71-year-old man with pleural effusion resulting from SAPHO syndrome. He was successfully treated using corticosteroids and has experienced no recurrence for one year. We should consider SAPHO syndrome when encountering cases of anterior chest pain and pleural fluid.

Keywords: SAPHO syndrome; anterior chest pain; hypoxia; pleural effusion.

PubMed Disclaimer

Conflict of interest statement

The authors state that they have no Conflict of Interest (COI).

Figures

Figure 1.
Figure 1.
Chest X-ray (A), pelvic X-ray (B), and lumbar spine X-ray (C). Chest X-ray (A) showed bilateral pleural effusion. Pelvic X-ray (B) revealed joint space narrowing, and supine X-ray (C) showed bamboo spine.
Figure 2.
Figure 2.
FDG PET/CT images: bilateral pleural fluid and pleural thickness with the uptake of FDG (A) and sternoclavicular joints with the slight uptake of FDG (B).
Figure 3.
Figure 3.
Contrast MRI of the thorax showed enlarged bilateral sternoclavicular joints.
Figure 4.
Figure 4.
Thoracoscopic pleural biopsy specimen (Hematoxylin and Eosin staining ×400).
Figure 5.
Figure 5.
Chest X-ray (A) after treatment and chest CT before and after treatment (B). Chest X-ray (A) demonstrated the improvement of the bilateral pleural effusion. Chest CT (B) also showed a reduction in the bilateral effusion and pleural thickening compared to before treatment.

References

    1. Chamot AM, Benhamou CL, Kahn MF, Beraneck L, Kaplan G, Prost A. [Acne-pustulosis-hyperostosis-osteitis syndrome. Results of a national survey. 85 cases]. Rev Rhum Mal Osteoartic 54: 187-196, 1987(in French). - PubMed
    1. Furer V, Kishimoto M, Shigeyoshi T, et al. . The diagnosis and treatment of adult patients with SAPHO syndrome: controversies revealed in a multidisciplinary international survey of physicians. Rheumatol Ther 7: 883-891, 2020. - PMC - PubMed
    1. Benhamou CL, Chamot AM, Kahn MF. Synovitis-acne-pustulosis hyperostosis-osteomyelitis syndrome (SAPHO). A new syndrome among the spondyloarthropathies? Clin Exp Rheumatol 6: 109-112, 1988. - PubMed
    1. Magrey M, Khan MA. New insights into synovitis, acne, pustulosis, hyperostosis, and osteitis (SAPHO) syndrome. Curr Rheumatol Rep 11: 329-333, 2009. - PubMed
    1. Dumolard A, Gaudin P, Juvin R, et al. . SAPHO syndrome or psoriatic arthritis? A familial case study. Rheumatology 38: 463-467, 1999. - PubMed

Publication types

MeSH terms