Tuberculosis of the extra-oral region is uncommon and is rarely primary. Extra-oral involvement of the cheek in the absence of tuberculosis elsewhere in the body is rare. To the best of our knowledge, we report here the first case of primary tuberculosis of the cheek in a 31-year-old male presenting as a nodular swelling of the cheek. Previous reported cases of extra-oral involvement of the cheek involved either fistula or sinus of the cheek. Excisional biopsy for tissue diagnosis and bacterial examination with culture should be performed for an early diagnosis as a delay in treatment can lead to devastating consequences.

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