{"paper_id":"3bf2fb9f-a859-4491-b220-479625edda48","body_text":"Journal of Case Reports and Images in Obstetrics and Gynecology, Volume 8, Issue 2, 2022; Pages 4–6. ISSN: 2582-0249\nJ Case Rep Images Obstet Gynecol 2022;8(2):4–6. \nwww.ijcriog.com\nAli et al. 4\nCASE REPORT OPEN ACCESS \nA unique case of isolated vulvar endometriosis\nAyesha Aziz Ali, Kelsey Musselman, Marie Smithgall, Kristen Pepin\nCASE REPORT\nA 40-year-old nulligravida woman with longstanding \ndysmenorrhea and heavy menstrual bleeding underwent \ncomputed tomography (CT) and magnetic resonance \nimaging (MRI), and alongside findings of uterine fibroids \nwas incidentally found to have a 4.0 ×1.8×1.6 cm right \nlabial soft tissue structure anterior to the right pubic \nbone (Figure 1A–C). The patient noted that the mass \nenlarged during her menses, and her exam revealed a \npalpable, firm, semi-mobile right vulvar mass slightly \nadherent to the pubic symphysis with no overlying skin \nabnormalities or lymphadenopathy. Notably, the patient \nhad an inguinal hernia repair and diagnostic gynecologic \nlaparoscopy performed five years prior during which no \nendometriosis or fibroids were identified.\nThe patient underwent laparoscopic myomectomy, \nhysteroscopic endometrial polypectomy, and vulvar \nmass resection. To excise the vulvar mass, an incision \nwas made with a scalpel, and extended to the level of \nthe mass with electrosurgery. The mass was grasped \nwith allis clamps, and was carefully separated from \nsurrounding tissue using electrosurgery. The wound was \ncopiously irrigated and then closed in three layers using \nvicryl suture. The specimen was sent for permanent \nAyesha Aziz Ali1, Kelsey Musselman2, Marie Smithgall3, Kris-\nten Pepin4\nAffiliations: 1MD, Medical Resident, Department of Obstet -\nrics and Gynecology, New York Presbyterian Hospital/Weill \nCornell Medical Center, New York, NY, USA; 2MD, Assistant \nClinical Professor, Department of Obstetrics and Gynecol -\nogy, New York Presbyterian Hospital/Weill Cornell Medical \nCenter, New York, NY, USA; 3Fellow in Pathology and Labo-\nratory Medicine, Division of Pathology and Laboratory Medi-\ncine, New York Presbyterian Hospital/Weill Cornell Medical \nCenter, New York, NY, USA; 4Assistant Attending and Assis-\ntant Professor, Department of Obstetrics and Gynecology, \nNew York Presbyterian Hospital/Weill Cornell Medical Cent-\ner, New York, NY, USA.\nCorresponding Author: Ayesha Aziz Ali, MD, 525 E 68th St, \nSuite M7, New York, NY 10065, USA; Email: aaa9049@nyp.\norg\nReceived: 26 May 2022\nAccepted: 12 August 2022\nPublished: 06 September 2022\npathologic evaluation. Microscopically, the specimen \nshowed fibroadipose tissue containing endometrial \nstroma and glands with associated chronic inflammation \nconsistent with extensive endometriosis (Figure 2A and \nB). No other lesions suspicious for endometriosis were \nidentified on laparoscopy (Figure 3A–C). On the patient’s \npost-operative visit, her vulvar incision was noted to be \nwell healed, with no recurrent mass or swelling palpable.\nCLINICAL IMAGE PEER REVIEWED | OPEN ACCESS \nFigure 1: CT and MRI findings. (A) Axial CT with 2 cm soft tissue \nmass (yellow arrow) anterior to right pubic bone. No enlarged \ninguinal lymph nodes were identified. (B) T2-weighted axial \nMRI with ill-defined but mass-like enhancing soft tissue (yellow \narrow) in the subcutaneous tissues of the right anterior–superior \nlabia majora and mons pubis measuring 4.0 cm×1.8 cm×1.6 cm. \n(C) T2-weighted sagittal MRI with labial mass (yellow arrow) \nand fibroid uterus (blue arrow).\nFigure 2: Histologic findings confirming endometriosis. (A) \nSection of the vulvar mass at low power (2×) shows fibroadipose \ntissue with extensive endometriosis. (B) High power (10×) \nshows endometrial stroma and glands within the tissue with \nassociated chronic inflammation.\nFigure 3: Laparoscopic findings. (A) No endometriosis identified \nin posterior cul de sac. (B) Uterus with multiple anterior \nsubserosal and intramural fibroids. No anterior cul de sac \nendometriosis identified. (C) Uterus status post laparoscopic \nmyomectomy.\n\nJournal of Case Reports and Images in Obstetrics and Gynecology, Volume 8, Issue 2, 2022; Pages 4–6. ISSN: 2582-0249\nJ Case Rep Images Obstet Gynecol 2022;8(2):4–6. \nwww.ijcriog.com\nAli et al. 5\nDISCUSSION\nEndometriosis is a gynecologic disease that involves \nendometrial glands and stroma occurring outside the \nuterine cavity, with most common sites including the \novaries, anterior and posterior cul-de-sac, posterior \nbroad ligaments, uterosacral ligaments, uterus, fallopian \ntubes, sigmoid colon, appendix, and round ligaments [1, \n2]. Endometriosis of the vulva is extremely rare, often \npresenting as vulvodynia or dyspareunia. Very few cases \nhave been reported in the literature, and in most of these \ncase reports either concomitant pelvic disease is present \nor it is undetermined whether more extensive disease is \npresent as laparoscopy was not performed [3–6]. In one \nsystematic review, it was noted that 95.3% of patients \npresenting with vulvo-perineal endometriosis have \nundergone either episiotomy, perineal trauma, vaginal \ninjury, or vaginal surgery [7]. In most cases, patients \npresented with cyclical vulvar pain, and were initially \nthought to have a Bartholin gland cyst. In three reported \ncases, during marsupialization of what was thought to \nbe a Bartholin cyst, chocolate colored fluid drained from \nthe mass, and the diagnosis was confirmed by pathology \n[3, 6, 8]. In all reported cases, surgical excision was the \nmanagement of choice. In one report, the patient received \nsix months of treatment with a luteinizing hormone \nreleasing hormone analogue with a good evolution [8]. \nAs there are few such cases reported in literature, there is \nno good data to comment on complication or recurrence \nrates.\nCONCLUSION\nOur patient presents with a unique, spontaneous case \nof isolated vulvar endometriosis unrelated to prior trauma \nor surgery, with intra-abdominal sites of disease ruled out \nlaparoscopically.\nKeywords: Endometriosis, Vulvar endometriosis, Vul -\nvar mass\nHow to cite this article\nAli AA, Musselman K, Smithgall M, Pepin K. A unique \ncase of isolated vulvar endometriosis. J Case Rep \nImages Obstet Gynecol 2022;8(2):4–6.\nArticle ID: 100124Z08AA2022\n*********\ndoi: 10.5348/100124Z08AA2022CI\nREFERENCES\n1. Gustofson RL, Kim N, Liu S, Stratton P. Endometriosis \nand the appendix: A case series and comprehensive \nreview of the literature. Fertil Steril 2006;86(2):298–\n303.\n2. Jenkins S, Olive DL, Haney AF. Endometriosis: \nPathogenetic implications of the anatomic distribution. \nObstet Gynecol 1986;67(3):335–8.\n3. Gocmen A, Inaloz HS, Sari I, Inaloz SS. Endometriosis \nin the Bartholin gland. Eur J Obstet Gynecol Reprod \nBiol 2004;114(1):110–1.\n4. Brug P, Gueye NA, Bachmann G. Vulvar endometriosis \npresenting with dyspareunia: A case report. J Reprod \nMed 2012;57(3–4):175–7.\n5. Eyvazzadeh AD, Smith YR, Lieberman R, Quint EH. A \nrare case of vulvar endometriosis in an adolescent girl. \nFertil Steril 2009;91(3):929.e9–11.\n6. Heijink T, Bogers H, Steensma A. Endometriosis of \nthe Bartholin gland: A case report and review of the \nliterature. J Med Case Rep 2020;14(1):85.\n7. Maillard C, Cherif Alami Z, Squifflet JL, et al. Diagnosis \nand treatment of vulvo-perineal endometriosis: A \nsystematic review. Front Surg 2021;8:637180.\n8. Hakimi I, Benabdejlil Y, Kouach J, Moussaoui D, \nDehayn M. Endometriosis in the Bartholin gland: A \ncase report. J Obstet Gynaecol 2014;2(5):75–6.\n*********\nAcknowledgments\nThe authors would like to thank Dr. Evelyn Cantillo, MD, \nMPH, Gynecologic Oncologist and Assistant Professor at \nNew York Presbyterian Hospital/Weill Cornell Medical \nCenter for her diligent care of this patient.\nAuthor Contributions\nAyesha Aziz Ali – Design of the work, Acquisition of \ndata, Drafting the work, Revising the work critically for \nimportant intellectual content, Final approval of the \nversion to be published, Agree to be accountable for all \naspects of the work in ensuring that questions related \nto the accuracy or integrity of any part of the work are \nappropriately investigated and resolved\nKelsey Musselman – Design of the work, Acquisition \nof data, Drafting the work, Revising the work critically \nfor important intellectual content, Final approval of the \nversion to be published, Agree to be accountable for all \naspects of the work in ensuring that questions related \nto the accuracy or integrity of any part of the work are \nappropriately investigated and resolved\nMarie Smithgall – Analysis of data, Interpretation of data, \nRevising the work critically for important intellectual \ncontent, Final approval of the version to be published, \nAgree to be accountable for all aspects of the work in \nensuring that questions related to the accuracy or integrity \nof any part of the work are appropriately investigated and \nresolved\n\nJournal of Case Reports and Images in Obstetrics and Gynecology, Volume 8, Issue 2, 2022; Pages 4–6. ISSN: 2582-0249\nJ Case Rep Images Obstet Gynecol 2022;8(2):4–6. \nwww.ijcriog.com\nAli et al. 6\nKristen Pepin – Conception of the work, Revising the work \ncritically for important intellectual content, Final approval \nof the version to be published, Agree to be accountable for \nall aspects of the work in ensuring that questions related \nto the accuracy or integrity of any part of the work are \nappropriately investigated and resolved\nGuarantor of Submission\nThe corresponding author is the guarantor of submission.\nSource of Support\nNone.\nConsent Statement\nWritten informed consent was obtained from the patient \nfor publication of this article.\nConflict of Interest\nThe authors declare that they have no conflicts of interest \nand nothing to disclose. These data have not been \npublished or presented elsewhere and the manuscript is \nnot under review at any other journal.\nData Availability\nAll relevant data are within the paper and its Supporting \nInformation files.\nCopyright\n© 2022 Ayesha Aziz Ali et al. This article is distributed \nunder the terms of Creative Commons Attribution \nLicense which permits unrestricted use, distribution \nand reproduction in any medium provided the original \nauthor(s) and original publisher are properly credited. \nPlease see the copyright policy on the journal website for \nmore information.\nAccess full text article on\nother devices\nAccess PDF of article on\nother devices\n\nSubmit your manuscripts at\nwww.edoriumjournals.com","source_license":"CC0","license_restricted":false}