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Adenoid cystic carcinoma of the Bartholin gland is not HPV-related: A case report and review of literature
Institution:1. Department of Gynaecologic Oncology, Erasmus MC, University Medical Centre Rotterdam, Rotterdam, The Netherlands;2. Department of Pathology, Erasmus MC, University Medical Centre Rotterdam, Rotterdam, The Netherlands;3. Department of Pathology, University Clinics Saint-Luc, Brussels, Belgium;1. Post-Graduate School of Pediatrics, University of Florence, Florence, Italy;2. Pediatric Unit, Meyer Children''s University Hospital, Florence, Italy;3. Pediatric Gynecology Unit, Meyer Children''s University Hospital, Florence, Italy;4. Department of Minimally Invasive Gynecological Surgery, Centre Tuscany USL, Italy;5. Radiology Unit, Anna Meyer Children''s University Hospital, Florence, Italy;6. Department of NEUROFARBA, Meyer Children''s University Hospital, University of Florence, Florence, Italy;1. Department of Urology, Oyokyo Kidney Research Institute, Hirosaki, Japan;2. Department of Urology, Hirosaki University Graduate School of Medicine, Hirosaki, Japan;3. Department of Cardiology and Nephrology, Hirosaki University Graduate School of Medicine, Hirosaki, Japan;4. Department of Advanced Transplant and Regenerative Medicine, Hirosaki University Graduate School of Medicine, Hirosaki, Japan;5. Department of Transplant Surgery, Nagoya Daini Red Cross Hospital, Nagoya, Japan;1. Department of Radiology, Royal Melbourne Hospital, Melbourne, Australia;2. Department of Anatomical Pathology, Royal Melbourne Hospital, Melbourne, Australia;3. Faculty of Medicine, Dentistry, and Health Sciences at the University of Melbourne, Parkville, Australia
Abstract:Adenoid cystic carcinoma (ACC) of the Bartholin gland is a rare gynaecological entity. Despite its slow growth and inconspicuous presentation, vulvar ACC has a propensity for perineural invasion and is therefore associated with high local recurrence rates.We report a case of vulvar ACC in a 61-year-old woman with a prolonged swelling of the Bartholin gland. This patient presented with pulmonary metastases at the moment of histological diagnosis. The vulvar and the pulmonary lesions showed identical histology. Despite a history of human papilloma virus (HPV)-related usual type vulvar intra-epithelial neoplasia and cervical squamous cell carcinoma, the vulvar ACC was negative for both p16 immunohistochemistry and HPV-DNA.We conclude that HPV is not involved in the pathogenesis of pure ACC of the Bartholin gland. Additionally, we advocate a low threshold for performing biopsies of vulvar swellings in women aged >40 years, to rule out malignancy and to prevent diagnostic delays.
Keywords:Vulva  Bartholin gland  Adenoid cystic carcinoma  Human papilloma virus  Lung metastasis  p16
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