Squamous cell carcinoma of the Bartholm's glands is a very rare form of vulvar neoplasm. This case presents a 46-year-old female diagnosed with advanced primary squamous cell cancer of the Bartholin glands that has proven positive for human papillomavirus (HPV). The patient was treated with a wide excision of the tumor, an ipsilateral lymphadenectomy, and an adjuvant chemotherapy and irradiation. After two years of follow-up the patient remains in remission.