A unique case of isolated vulvar endometriosis

Endometriosis is a gynecologic disease that involves endometrial glands and stroma occurring outside the uterine cavity, with most common sites including the ovaries, anterior and posterior cul-de-sac, posterior broad ligaments, uterosacral ligaments, uterus, fallopian tubes, sigmoid colon, appendix, and round ligaments [1, 2]. Endometriosis of the vulva is extremely rare, often presenting as vulvodynia or dyspareunia. Very few cases have been reported in the literature, and in most of these case reports either concomitant pelvic disease is present or it is undetermined whether more extensive disease is present as laparoscopy was not performed [3–6]. In one systematic review, it was noted that 95.3% of patients presenting with vulvo-perineal endometriosis have undergone either episiotomy, perineal trauma, vaginal injury, or vaginal surgery [7]. In most cases, patients presented with cyclical vulvar pain, and were initially thought to have a Bartholin gland cyst. In three reported cases, during marsupialization of what was thought to be a Bartholin cyst, chocolate colored fluid drained from the mass, and the diagnosis was confirmed by pathology [3, 6, 8]. In all reported cases, surgical excision was the management of choice. In one report, the patient received six months of treatment with a luteinizing hormone releasing hormone analogue with a good evolution [8]. As there are few such cases reported in literature, there is no good data to comment on complication or recurrence rates.

Our patient presents with a unique, spontaneous case of isolated vulvar endometriosis unrelated to prior trauma or surgery, with intra-abdominal sites of disease ruled out laparoscopically.

Endometriosis, Vulvar endometriosis, Vul - var mass How to cite this article Ali AA, Musselman K, Smithgall M, Pepin K. A unique case of isolated vulvar endometriosis. J Case Rep Images Obstet Gynecol 2022;8(2):4–6. Article ID: 100124Z08AA2022 ********* doi: 10.5348/100124Z08AA2022CI

1. Gustofson RL, Kim N, Liu S, Stratton P. Endometriosis and the appendix: A case series and comprehensive review of the literature. Fertil Steril 2006;86(2):298– 303. 2. Jenkins S, Olive DL, Haney AF. Endometriosis: Pathogenetic implications of the anatomic distribution. Obstet Gynecol 1986;67(3):335–8. 3. Gocmen A, Inaloz HS, Sari I, Inaloz SS. Endometriosis in the Bartholin gland. Eur J Obstet Gynecol Reprod Biol 2004;114(1):110–1. 4. Brug P, Gueye NA, Bachmann G. Vulvar endometriosis presenting with dyspareunia: A case report. J Reprod Med 2012;57(3–4):175–7. 5. Eyvazzadeh AD, Smith YR, Lieberman R, Quint EH. A rare case of vulvar endometriosis in an adolescent girl. Fertil Steril 2009;91(3):929.e9–11. 6. Heijink T, Bogers H, Steensma A. Endometriosis of the Bartholin gland: A case report and review of the literature. J Med Case Rep 2020;14(1):85. 7. Maillard C, Cherif Alami Z, Squifflet JL, et al. Diagnosis and treatment of vulvo-perineal endometriosis: A systematic review. Front Surg 2021;8:637180. 8. Hakimi I, Benabdejlil Y, Kouach J, Moussaoui D, Dehayn M. Endometriosis in the Bartholin gland: A case report. J Obstet Gynaecol 2014;2(5):75–6. ********* Acknowledgments The authors would like to thank Dr. Evelyn Cantillo, MD, MPH, Gynecologic Oncologist and Assistant Professor at New York Presbyterian Hospital/Weill Cornell Medical Center for her diligent care of this patient. Author Contributions Ayesha Aziz Ali – Design of the work, Acquisition of data, Drafting the work, Revising the work critically for important intellectual content, Final approval of the version to be published, Agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved Kelsey Musselman – Design of the work, Acquisition of data, Drafting the work, Revising the work critically for important intellectual content, Final approval of the version to be published, Agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved Marie Smithgall – Analysis of data, Interpretation of data, Revising the work critically for important intellectual content, Final approval of the version to be published, Agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved Journal of Case Reports and Images in Obstetrics and Gynecology, Volume 8, Issue 2, 2022; Pages 4–6. ISSN: 2582-0249 J Case Rep Images Obstet Gynecol 2022;8(2):4–6. www.ijcriog.com Ali et al. 6 Kristen Pepin – Conception of the work, Revising the work critically for important intellectual content, Final approval of the version to be published, Agree to be accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved Guarantor of Submission The corresponding author is the guarantor of submission. Source of Support None. Consent Statement Written informed consent was obtained from the patient for publication of this article. Conflict of Interest The authors declare that they have no conflicts of interest and nothing to disclose. These data have not been published or presented elsewhere and the manuscript is not under review at any other journal. Data Availability All relevant data are within the paper and its Supporting Information files. Copyright © 2022 Ayesha Aziz Ali et al. This article is distributed under the terms of Creative Commons Attribution License which permits unrestricted use, distribution and reproduction in any medium provided the original author(s) and original publisher are properly credited. Please see the copyright policy on the journal website for more information. Access full text article on other devices Access PDF of article on other devices Submit your manuscripts at www.edoriumjournals.com