Background: Tuberculosis has existed for millennia and remains a major global health problem. Soft tissue tuberculosis is one of the rare forms of extrapulmonary tuberculosis, and isolated muscular localization is uncommon.

Case Presentation: We report the extremely rare case of a 56-year-old patient without any medical history who presented a pseudo-tumoral gluteal mass of tuberculous origin without other localizations of the disease. He underwent a one-piece excision of the mass measuring 18 cm. Histopathological examination was in favor of a remodeled tuberculoma and the diagnosis was confirmed with polymerase chain reaction (PCR) examination and the positive result of the quantiferon test. The patient completed his antituberculous therapy successfully and the 3-year follow-up revealed no recurrence or complications.

Conclusion: Although isolated muscle tuberculosis is a rare entity, it must always be taken into consideration as a differential diagnosis for isolated muscle swelling, especially in a country with a high TB endemic. The diagnosis is mainly bacteriological and/or pathological. Treatment uses anti-bacillary chemotherapy alone or in combination with surgery. The prognosis is often favorable when the management is early.