A Rare Coexistence of SAPHO Syndrome and Hidradenitis Suppurativa: Case Report and Clinical Insights

Background Synovitis, acne, pustulosis, hyperostosis, osteitis (SAPHO) syndrome is a rare disease which seems to have a multifactorial origin with genetic, environmental, immunologic, and infectious components.[1] Hidradenitis suppurativa (HS) is a chronic autoinflammatory keratinization disease that affects sweat glands.[2] Both SAPHO syndrome and HS are chronic, relapsing diseases that pose a severe impairment to a patient’s quality of life. While the coexistence of these 2 conditions is rare, this case presents a 28-year-old male with a co-presentation of SAPHO syndrome and HS. Case A 28-year-old male from an Indian Ocean’s Island with a two-year history of multiple skin lesions including acne, pustules, and anorectal abscess and a 4-month history of recurrent musculoskeletal pain, presented to hospital with fever and an acute pain in the left elbow and in the right wrist increasing in recent days (Table 1). During examination, 2 synovitis were found and 10 cm by 10 cm ulcerations were observed in the axillary regions with serous discharge, granulation and sinus tracts which were later found to be HS. Anamnesis revealed that these skin lesions had been evolving for 2 years, and that the patient was reluctant to seek medical assessment. During hospitalization, blood work revealed leukocytosis, thrombocytosis, eosinophilia, and c-reactive protein reaching 137 mg/L. A bone scan revealed bilateral uptake on acromioclavicular and sternoclavicular bones and the first costochondral junctions, which was consistent with the diagnosis of SAPHO syndrome. After an infusion of pamidronate 60 mg, bone pain was considerably improved, and skin lesions abated with local care only. Table 1- Characteristics of patients with an association of SAPHO syndrome and hidradenitis suppurativa (HS) Conclusion Although the association of SAPHO syndrome and HS is unclear, this case demonstrates a rare example of the coexistence of these 2 diseases. This case supports previous literature which showed that HS in SAPHO syndrome was more common among African descents compared to other ethnicities.[2,3] It is crucial to look for signs of HS during physical examinations when there is a suspicion of SAPHO syndrome, as the co-presentation of these 2 conditions is often associated with poor response to medical therapy and more comorbidities.[2,3] Patients with HS may be hesitant to seek medical attention or reveal their skin lesions due to discomfort or embarrassment, leading to a progression of the disease. Identifying HS early during physical examination can help clinicians provide timely intervention and potentially preventing further complications, particularly in the more prevalent subgroup of African descents with HS and SAPHO syndrome. [1.] Girschick H. SAPHO syndrome [Internet]. Orphanet; 2019. Available from: https://www.orpha.net/en/disease/detail/793 [2.] Crowley EL. SAGE Open Med Case Rep 2018;6:2050313X18778723. [3.] Steinhoff J. Journal of Clinical Rheumatology 2002;8:13-22.

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