Introduction: Hidradenitis suppurativa (HS) is a rare painful, chronic inflammation of the apocrine glands. It is characterized by painful, inflamed cutaneous lesions causing nodules, sinus tracts and abcesses. This condition typically manifests in intertriginous areas such as axillae, groin, perineum and submammary regions. However, sacrococcygeal HS is an atypical localization and scarcely reported in the literature. Case presentation: We report a clinical case involving a 34-year-old young North-African male with no prior medical or surgical history or known predisposing factors. He presented an unusual anatomical presentation of hidradenitis suppurativa evolving in the last year prior to day clinic consultation. The patient self-took non documented oral antibiotics with no results before consultation. The lesion presented lower back pain and malodorant discharge. The evolution of the lesion although antibiotics motivated consultation. Our patient benefited a large surgical resection of the lesion with good evolution from week 7 post-surgery. Conclusion: Sacrococcygeal localization of HS is atypical but should be considered. This case illustrates the problems raised by a seemingly trivial disease. Surgery with adjuvant therapy may be the option in extensive lesion.