{"paper_id":"35195a20-1c75-41b3-a8df-cdbe1ca0ae61","body_text":"Review began\n 03/06/2022 \nReview ended\n 03/07/2022 \nPublished\n 03/08/2022\n© Copyright \n2022\nSabri et al. This is an open access article\ndistributed under the terms of the Creative\nCommons Attribution License CC-BY 4.0.,\nwhich permits unrestricted use, distribution,\nand reproduction in any medium, provided\nthe original author and source are credited.\nVaginal Endosalpingiosis: A Case Report and\nLiterature Review\nAhmed Sabri \n \n, \nMargarita Loxas \n \n, \nLinnea Banker \n \n, \nKevin Zhang \n \n, \nWayne Penka \n1.\n Pathology and Laboratory Medicine, Creighton University School of Medicine, Omaha, USA\nCorresponding author: \nAhmed Sabri, \nahmsab91@gmail.com\nAbstract\nEndosalpingiosis is a benign condition with unclear pathogenesis and clinical significance and is defined as\nthe presence of ectopic fallopian tube-like epithelium. It can be found in multiple locations, most commonly\nin the pelvic peritoneum covering the ovaries, uterus, and fallopian tubes, and less commonly found in the\nlymph nodes, omentum, appendix, cervix, vulva, or vagina.\nIt is difficult to distinguish from endometriosis by gross appearance or localization, and theories propose\nthat tissues of the secondary Mullerian system may undergo a metaplastic transformation, for example, from\nendosalpingiosis to endometriosis, which contributes to the debated association of endosalpingiosis with\nchronic pelvic pain. Additionally, there is evidence demonstrating a close association with reproductive\ntract neoplasms.\nWe report the clinical course, diagnosis including pathology, follow-up, and the treatment plan of vaginal\nendosalpingiosis in a 34-year-old woman presenting with a chronic painful right-sided vaginal mucosal\nulceration, dyspareunia, and foul-smelling vaginal discharge. To our knowledge, this is the second reported\ncase of vaginal endosalpingiosis and the first case with this presentation.\nCategories:\n Obstetrics/Gynecology, Pathology\nKeywords:\n gynecology, vagina, gynecologic pathology, benign, ectopic epithelium, vaginal endosalpingiosis,\nendosalpingiosis\nIntroduction\nEndosalpingiosis (ES) is, by definition, the presence of ectopic fallopian tube-like epithelium \n[1]\n. Distinct\nfrom endometriosis, ES is characterized by ciliated glandular epithelium, absent endometrial stroma, and\nusually no inflammatory component \n[2]\n. It is most commonly encountered in the ovary and is rarely seen in\nareas such as the myometrium or the pelvic peritoneum. The pathogenesis and clinical significance of ES are\nambiguous, as ES has been widely assumed to be an incidentally discovered and benign condition since it\nwas first described in 1930. However, in recent years, there is growing evidence of the potential of increased\nrisk for malignancy in patients with ES \n[3]\n. As a result, it is imperative to grow familiar with the features of\nthis diagnosis. Here, we present a case of a woman with a five-year history of vaginal ulceration and foul-\nsmelling discharge. The tissue displayed ciliated tubal epithelium on biopsy and the patient was given the\ndiagnosis of vaginal ES.\nCase Presentation\nA 34-year-old woman presented with a right-sided vaginal mucosal ulceration as well as a foul-smelling\nvaginal discharge for the last five years. The ulceration was painful to palpation and resulted in a constant\ngeneral pelvic discomfort. She also reported pain with sexual intercourse, describing the pain as burning and\nrubbing sensation. She denied any bleeding between menstrual cycles, post-coital bleeding, or bowel or\nbladder symptoms.\nThe patient had been previously diagnosed with recurrent bacterial vaginosis with no resolution of\nsymptoms with treatment. She had a history of three vaginal deliveries, one of which, in 2008, required\nforceps assistance and resulted in a fourth-degree vaginal laceration. The injury was repaired with a suture.\nThe patient subsequently underwent MRI to assess for rectovaginal fistula, which did not show evidence of\nanatomic abnormality or further injury. She did not have a history of sexually transmitted infections (STIs).\nUpon physical exam, a 3 cm vaginal ulceration of the right vaginal sidewall was noted. It was described to be\nlocated about 3 cm from the vaginal introitus. The ulcer was described as having a puckered and thickened\nborder, which was exquisitely tender to palpation and bled when touched. There was a small area of beefy-\nred granulation tissue present near the ulcer. The lesion did not have the appearance of necrosis or acute\ninfection. A large ectropion and several nabothian cysts of the cervix were also visualized.\nAnaerobic and aerobic cultures were obtained. The area was then anesthetized. When analgesia was\nachieved, a Tischler forceps was used to obtain the biopsy. It is also important to mention that the exam and\n1\n1\n1\n1\n1\n \n Open Access Case\nReport\n \nDOI:\n 10.7759/cureus.22949\nHow to cite this article\nSabri A, Loxas M, Banker L, et al. (March 08, 2022) Vaginal Endosalpingiosis: A Case Report and Literature Review. Cureus 14(3): e22949. \nDOI\n10.7759/cureus.22949\n\nbiopsy were somewhat limited due to the anatomical location and tenderness of the lesion. For the pathology\n(Figures \n1\n, \n2\n), the sections from the vaginal lesion biopsy showed ectopic glands lined by fallopian tube\nciliated epithelium with three types of cells: ciliated columnar, non-ciliated columnar, and intercalary cells.\nThose glands were strongly positive for estrogen receptor (ER) and vimentin stains, with patchy p16 staining\nconsistent with the normal staining pattern of benign tubal epithelium. Carcinoembryonic antigen (CEA)\nstain was negative.\nFIGURE\n 1: Right vaginal lesion biopsy on the hematoxylin and eosin\n(H&E) stains.\nOne area pictured from lower magnification (10x) to higher magnification (40x) showed ectopic glands lined by\nfallopian tube ciliated epithelium. At the higher magnification (40x), three types of cells can be appreciated: ciliated\ncolumnar, non-ciliated columnar, and intercalary cells.\n2022 Sabri et al. Cureus 14(3): e22949. DOI 10.7759/cureus.22949\n2\n of \n5\n\nFIGURE\n 2: Right vaginal lesion biopsy on the immunohistochemical\nstains.\nGlands are strongly positive for estrogen receptor (ER) and vimentin stains. Carcinoembryonic antigen (CEA)\nstain is negative. Glands are showing patchy p16 staining. The findings are consistent with the normal staining\npattern of benign tubal epithelium.\nThe patient was referred to the gynecologic oncology service for further workup, which was negative for STI,\ncervical dysplasia, and rectovaginal fistula. The patient will undergo wide local excision of the lesion with\nthe hope of symptomatic improvement.\nDiscussion\nES was first described in 1930 as “misplaced Mullerian mucosa…from the stump of the truncated fallopian\ntube” based on the local proliferative and invasive nature of tubal mucosa following salpingectomy \n[4]\n. True\nto the initial description, ES is histologically defined as the presence of ectopic tubal epithelium containing\nthree cell types: ciliated glandular epithelium, non-ciliated mucous secreting cells, and intercalary or peg\ncells \n[5,6]\n. The histological findings are differentiated from those of endometriosis by the presence of ciliated\nglandular epithelium as well as a lack of endometrial stroma and inflammatory components \n[7]\n. Macroscopic\nfeatures include simple cysts or complex papillary structures containing psammoma bodies \n[8]\n. ES is\ngenerally not hemorrhagic, unlike hormone-responsive endometriosis \n[6]\n.\nES can be found in multiple locations, most commonly in the pelvic peritoneum covering the ovaries, uterus,\nand fallopian tubes \n[3]\n. It is less commonly found in the lymph nodes, omentum, appendix, cervix, vulva, or\nvagina. It is unclear why certain sites are more susceptible to the ectopic tubal epithelium, but proximity to\nthe fallopian tube may be a factor.\nTwo main mechanisms giving rise to ES have been theorized: multifocal metaplastic process arising from\nperitoneal cells \n[9]\n or peritoneal implantation of sloughed tubal epithelial hyperplasia \n[10]\n. Regardless of the\nmechanism, ES is highly associated with chronic pelvic inflammatory insult or prior intrapelvic surgery \n[10]\n.\nSurgical intervention on the fallopian tubes carries a theoretical risk of ectopically seeding tubal epithelial\ncells. One study found nearly 80% of patients with ES had a prior history of tubal ligation or intrapelvic\nsurgery \n[3]\n.\nThe mean age at diagnosis is 43 years \n[5]\n. A recent study found 40% of cases occur in postmenopausal\nwomen, which was previously unrecognized \n[11]\n. ES is found in 1.4% to 12.5% of women undergoing\nlaparoscopy for a variety of gynecological symptoms \n[3,12,13]\n. The high variability in incidence may be\nrelated to patient selection and tissue sampling in each study. Intraoperative presentation ranges from\n2022 Sabri et al. Cureus 14(3): e22949. DOI 10.7759/cureus.22949\n3\n of \n5\n\nperitoneal nodular changes to omental or pelvic masses \n[14]\n. The heterogeneous appearance may mimic\nperitoneal tuberculosis or metastases of ovarian cancer \n[15]\n and is also difficult to distinguish from\nendometriosis by gross appearance or localization \n[5]\n. Diagnosis is rarely made preoperatively, but\ndisseminated pelvic calcifications can sometimes be found by radiologic imaging \n[16,17]\n.\nSome studies have reported symptomatic pain in up to one-third of patients \n[18,19]\n, while others have\nconcluded that ES presents asymptomatically \n[5,20]\n. The debated association with chronic pelvic pain is\ncomplicated by the high concordance of ES with endometriosis, which ranges from 4.4% to 67% \n[3,12]\n.\nInterestingly, one theory proposes that tissues of the secondary Mullerian system may undergo a\nmetaplastic transformation, for example, from ES to endometriosis \n[21]\n. Thus, further examination of this\nrelationship may demonstrate whether ES is an independent risk for pain or infertility. While there is an\nestablished clinical correlation between endometriosis and infertility, existing literature reports either low\nor no association between ES and infertility \n[3,11]\n.\nWhile ES has been hypothesized to be an incidental intraoperative finding, there is evidence demonstrating\na close association with reproductive tract neoplasms. Of particular importance is the increased association\nbetween premenopausal women with ES and gynecologic malignancy \n[11,22]\n. Gynecologic malignancy is\nfound in nearly half of patients with ES \n[11]\n, most commonly ovarian serous neoplasm (21%) and cervical\ncancer (18%) \n[3]\n. A large retrospective study found a significant increase of both uterine and ovarian cancer,\nbut not cervical cancer, in patients with ES \n[3]\n. This study confirmed prior reported association with ovarian\ncancers \n[23-26]\n. Of interest is the newly found association with uterine cancer, which may further strengthen\nthe hypothesis that tissues sharing a Mullerian origin have the capacity to undergo metaplastic conversion\nto other types of glandular epithelium that predispose to neoplastic transformation.\nIn terms of the literature, another case of vaginal ES is previously reported by Câmara et al. \n[27]\n. The patient\npresented with intermenstrual bleeding for several months without any suggestive history \n[27]\n. The physical\nexamination revealed a soft, polypoid, and hemorrhagic neoplasm in the posterior vaginal cuff \n[27]\n. The\npatient underwent polypectomy with electrocoagulation, the tissue was sent to pathology and revealed\nvaginal ES, and the subsequent follow-ups did not show any new symptoms \n[27]\n. In our case, the patient had\na different clinical presentation for the same underlying etiology. She presented with a painful vaginal ulcer\nwith dyspareunia and foul-smelling vaginal discharge, and the pathology showed similar findings to the\npreviously reported case of ES. Our patient will undergo wide local excision of the lesion with the hope of\nsymptomatic improvement.\nConclusions\nTo the authors’ knowledge, this is a rare case of vaginal ES with unique clinical features and gross findings.\nThis is still a poorly understood entity and provides a challenging diagnosis to pathologists. Our aim is to\nreport this rare entity in the literature in hopes of establishing a better understanding of the\npathophysiology, clinical course, and burden on patients.\nAdditional Information\nDisclosures\nHuman subjects:\n Consent was obtained or waived by all participants in this study. \nConflicts of interest:\n In\ncompliance with the ICMJE uniform disclosure form, all authors declare the following: \nPayment/services\ninfo:\n All authors have declared that no financial support was received from any organization for the\nsubmitted work. \nFinancial relationships:\n All authors have declared that they have no financial\nrelationships at present or within the previous three years with any organizations that might have an\ninterest in the submitted work. \nOther relationships:\n All authors have declared that there are no other\nrelationships or activities that could appear to have influenced the submitted work.\nReferences\n1\n. \nZangmo R, Singh N, Kumar S, Vatsa R: \nSecond look of endosalpingiosis: a rare entity\n. 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Gynecol Oncol. 2014, 132:316-21.\n10.1016/j.ygyno.2013.12.007\n27\n. \nCâmara S, Mendinhos G, Madureira R, Martins A, Veríssimo C: \nVaginal endosalpingiosis case report: a rare\nentity presenting as intermenstrual bleeding\n. Case Rep Obstet Gynecol. 2017, 2017:2424392.\n10.1155/2017/2424392\n2022 Sabri et al. Cureus 14(3): e22949. DOI 10.7759/cureus.22949\n5\n of \n5","source_license":"CC0","license_restricted":false}