Full text article. Also available at https://doi.org/10.1093/jscr/rjaa312The purpose of this paper is to report unusual, rarest and interesting case of a patient with nodular hyperplasia of Bartholin’s gland with dysplastic changes. The case of a 30-year old female with right-sided Bartholin’s gland hyperplasia with dysplastic changes, which was confirmed histopathologically, is presented in this paper. The patient reported increased swelling of the right major labium when she became sexually aroused with intermittent dyspareunia during intercourse. Surgical excision of the swelling was done under general anaesthesia. Healing of the surgical scar was complete and she reported no any discomfort or dyspareunia during sexual intercourse. Bartholin’s gland swellings with firm consistency require complete excision due to possibility of being neoplastic rather than just inflammation, cyst or obstructed gland particularly in postmenopausal women. Diagnosis of nodular hyperplasia of Bartholin’s gland with areas of dysplastic changes in the present case is of great interest and requires further investigation.