Case Report

A Bull’s Head Sign in SAPHO

Abstract

A syndrome of synovitis, acne, pustulosis, hyperostosis and osteitis (SAPHO) is a rare autoimmune disease. It occurs in genetically predisposed individuals by immune activation triggered by possible infectious agents. A 60-year-old man was presented with musculoskeletal and cutaneous manifestations. After exclusion of differential diagnoses, he was diagnosed with SAPHO and successfully treated with adalimumab. He had a typical image defined as a bull’s head sign in bone scintigraphy with 99mTc- methylene diphosphonate. Positron emission tomography also revealed increased inflammatory activity in related anterior chest joints and soft tissue. We want to emphasize diagnostic radiological images in patients with SAPHO to increase the awareness of clinicians.

1. Firinu D, Garcia-Larsen V, Manconi PE, Del Giacco SR. SAPHO Syndrome: Current Developments and Approaches to Clinical treatment. Curr Rheumatol Rep. 2016;16(6):35. https://doi. org/10.1007/s11926-016-0583-y
2. Carneiro S, Sampaio-Barros PD. SAPHO syndrome. Rheum Dis Clin North Am. 2013;39(2):401-18. https://doi.org/10.1016/j. rdc.2013.02.009
3. Nguyen MT, Borchers A, Selmi C, Naguwa SM, Cheema G, Gershwin ME. The SAPHO syndrome. Semin Arthritis Rheum. 2012;42(3): 254-65. https://doi.org/10.1016/j.semarthrit.2012.05.006
4. Okuno H, Watanuki M, Kuwahara Y, Sekiguchi A, Mori Y, Hitachi S, et al. Clinical features and radiological findings of 67 patients with SAPHO syndrome. Mod Rheumatol. 2018;28(4):703-708. https://doi.org/10.1080/14397595.2017.1372874
5. Berenguer Frances MA, Lafaurie Acevedo A, Tormo Ferrero V, et al. SAPHO syndrome in the differential diagnosis of me- tastasis. Rheumatol Clin. 2016;12(5):288-291. https://doi. org/10.1016/j.reumae.2015.08.004
6. Himuro H, Kurata S, Nagat S, Sumi A, Tsubaki F, Matsuda A, et al. Imaging features in patients with SAPHO/CRMO: a pic- torial review. Jpn J Radiol. 2020;38(7):622-629. https://doi. org/10.1007/s11604-020-00953-1
7. Jurik AG, Klicman RF, Simoni P, Robinson P, Teh J. SAPHO and CRMO: The Value of Imaging. Semin Musculoskelet Radiol. 2018;22(2):207-224. https://doi.org/10.1055/s-0038-1639469
8. Fu Z, Liu M, Li Z, Fan Y, Zhang J, Zhang X, et al. Is the bullhead sign on bone scintigraphy really common in the patient with SAPHO syndrome? A single-center study of a 16-year experi- ence. Nucl Med Commun. 2016;37(4):387-92. https://doi. org/10.1097/MNM.0000000000000451
9. Quirico Rodríguez M, Casáns Tormo I, Redal Peña MC, López Castillo V. The importance of bone scintigraphy in the diagno- sis of SAPHO syndrome. Rev Esp Med Nucl. 2010;29(3):127-30. https://doi.org/10.1016/j.remn.2010.01.001
10. Sun X, Li C, Cao Y, Shi X, Li L, Zhang W, et al. F-18 FDG PET/CT in 26 patients with SAPHO syndrome: a new vision of clinical and bone scintigraphy correlation. J Orthop Surg Res. 2018(22);13(1):120. https://doi.org/10.1186/s13018-018-0795-0
11. Cao Y, Liu Y, Li C. Severe deformity in long-term SAPHO syn- drome. Rheumatology (Oxford). 2021(1);60(2):982-983. https://doi.org/10.1093/rheumatology/keaa307
12. Cianci F, Zoli A, Gremese E, Ferraccioli G. Clinical heterogeneity of SAPHO syndrome: challenging diagnose and treatment. Clin Rheumatol. 2017;36(9):2151-2158. https://doi.org/10.1007/ s10067-017-3751-1
Files
IssueVol 7 No 5 (2022): September-October QRcode
SectionCase Report(s)
DOI https://doi.org/10.18502/crcp.v7i5.11860
Keywords
Synovitis Acne Pustulosis Hyperostosis Osteitis
Rights and permissions
Creative Commons License This work is licensed under a Creative Commons Attribution-NonCommercial 4.0 International License.
How to Cite
1.
Ulutaş F, Ufuk F, Bozdemir A, Çobankara V. A Bull’s Head Sign in SAPHO. CRCP. 2023;7(5):227-230.