{"paper_id":"b349abfe-a1e1-45ee-9112-fc0ae60a3622","body_text":"Articles published in Obstet Gynecol Sci are open-access, distributed under the terms of \nthe Creative Commons Attribution Non-Commercial License (http://creativecommons.\norg/licenses/by-nc/3.0/) which permits unrestricted non-commercial use, distribution, \nand reproduction in any medium, provided the original work is properly cited.\nCopyright © 2015 Korean Society of Obstetrics and Gynecology \nCase Report\nObstet Gynecol Sci 2015;58(4):319-322\nhttp://dx.doi.org/10.5468/ogs.2015.58.4.319\npISSN 2287-8572 · eISSN 2287-8580\nwww.ogscience.org 319\nIntroduction\nPost hysterectomy complications at the vault site such as a \nbleeding incident can be commonly observed at a short term \npostoperative period. Other delayed complications often occur \nas a hematoma, granuloma, keloid, incisional hernia, and or \nvascular formation at the vault [1,2]. Many of these complica-\ntions may be accompanied with bleeding symptoms. However \na sudden massive bleeding occurring after more than a year \npostoperatively is rare. \nHere we report a case of sudden vaginal vault bleeding in \na post-hysterectomy state patient with no known history of \nendometriosis, hysterectomy undergone prior 13 months ago, \nproven to be a rare case of vault endometriosis. Hence, we \nwould like to present how such a rare complication might be \nmisdiagnosed and moreover increase the awareness of such \ncomplication of the vault in a post-hysterectomy patient.\nCase report\nEarly on October 2014, a 45-year-old woman was presented \nat our outpatient gynecology clinic with sudden lower pelvic \ndiscomfort and vaginal bleeding symptoms. The patient had \na history of hysterectomy 13 months ago due to symptomatic \nmultiple leiomyomas and adenomyosis. \nThe previous surgery was conducted as a single-port ap -\nproach laparoscopic-assisted vaginal hysterectomy in July 2013. \nSurgical findings showed an enlarged uterus of about a 14 \nweeks gestational age size. Both adnexa were grossly normal \nin appearance and the peritoneum was clear with no signs of \nendometriosis. The vaginal vault was sutured vaginally using \na 1-0 vicryl. No complications were observed during the post-\noperative period and the patient was discharged as scheduled. \nThe histology of the uterus was confirmed as adenomyosis with \nleiomyoma with a secretory phase endometrium. Follow up \nexaminations at 3 and 6 months showed no complications and \nthus no additional follow-ups were required afterwards.\nHowever on October 2014, after more than a year from her \nlast check up, the patient appeared at our clinic complaining \nReceived: 2014.12.9.   Revised: 2015.1.11.   Accepted: 2015.1.13.\nCorresponding author: Woo Young Kim\nDepartment of Obstetrics and Gynecology, Kangbuk Samsung Hospital, \nSungkyunkwan University School of Medicine, 29 Saemunan-ro, \nJongno-gu, Seoul 110-746, Korea\nTel: +82-2-2001-2499  Fax: +82-2-2001-2187\nE-mail: obgykim@gmail.com\nA rare case of post-hysterectomy vault site iatrogenic \nendometriosis\nCha Hien Choi\n1\n, Jeong Jin Kim\n1\n, Woo Young Kim\n1\n, Kyeung Whan Min\n2\n, Dong Hoon Kim\n2\nDepartments of \n1\nObstetrics and Gynecology, \n2\nPathology, Kangbuk Samsung Hospital, Sungkyunkwan University School of Medicine, Seoul, Korea\nA 45-year-old woman with a prior history of hysterectomy due to adenomyosis and leiomyomas was presented at our \noutpatient gynecology clinic 13 months later with sudden lower pelvic discomfort and vaginal bleeding symptoms. \nThe patient underwent vaginal vault biopsy however diagnosis was still uncertain. Additional evaluation was required \ndue to massive rebleeding incidents. After an emergent explorative laparoscopic operation with total excision of \nthe vault, a diagnosis of vaginal vault endometriosis was made. Our theory is that a possible transplantation of \nendometrial cells during morcellation of the adenomyotic uterus which then may have progressed to iatrogenic \nendometriosis of the vaginal vault. Therefore, vault endometriosis must be considered in incidences of delayed \nmassive bleeding occurring in post-hysterectomy patients when other diagnoses have been excluded.\nKeywords: Hysterectomy; Morcellation; Vagina vault endometriosis\n\nwww.ogscience.org320\nVol. 58, No. 4, 2015\nof sudden pelvic discomfort and vaginal bleeding symptoms. \nPelvic examinations showed no signs of active bleeding. Yet, \na dark wine colored papule suggestive of a small hematoma \nor ulcerative lesion was observed upon the previous operative \nvaginal vault site (Fig. 1.).\nHer vitals were stable and she showed no signs of fever. The \npain was tolerable during manipulation of the vault site. The \nulcerative lesion was suspected as an old hematoma or granu-\nlation formation of the previous hysterectomy vault or possibly \ndue to an unknown malignancy or cancerous change. \nFor further investigation, a quick excision biopsy using sharp \nscissors was performed under local anesthesia at the outpatient \noperation room. Bleeding control was done with sutures and \ntampon gauzes were inserted intravaginally. Nevertheless, the \npatient returned to the clinic within a few hours with excessive \nbleeding. Upon re-examination, active bleeding was visible at \nthe vault site, and additional maneuvers including sutures were \nperformed for hemostasis. \nThe pathology reports showed normal vaginal tissue with \nnon-specific loss of surface epithelium and subepithelial hemor-\nrhage. The diagnosis was uncertain but any kind of malignancy \ncould be ruled out. \nTen days later, the patient revisited our clinic with another \nincidence of massive vaginal bleeding with large clots. She \nlooked pale and anemic in general with vital signs showing an \nincreased pulse with a decreased blood pressure at an initial \n130/70 to 100/60. The patient also complained of symptoms \nof dizziness. Her lab results showed a hemoglobin level of 6.9 \ng/dL and hematocrit of 20.2%. Further attempts to achieve \nhemostasis with ball electrode cauterization were insufficient. \nThe lesion healed only for a short while and the fragile tissue \ncontinued to bleed. Because of the massive bleeding, a clinical \ndiagnosis of a possible vascular malformation or uterine artery \npseudoaneurysm was considered. \nAn emergent exploratory laparoscopic operation was per -\nformed to control vault bleeding and to evaluate any possible \nvascular complication or collateral arterial bleeding. Due to ane-\nmic conditions, transfusion with 2 pints of packed red blood \ncells were necessary prior to the operation. \nUpon surgery, other peritoneal structures including both \novaries were grossly normal and showed no signs of endome-\ntriosis. The pelvic side vault site was securely sealed with normal \nperitoneum covering the vault with no dehiscence or any other \ncomplication. Under conventional laparoscopy, after dissecting \nthe anterior rectal wall and bladder peritoneum, a full thickness \nexcision of approximately 2 cm in diameter was performed at \nthe vault site and resealed with sutures. \nPathology results of the excised lesion was confirmed to be \nconsistent with endometriosis (Fig. 2.). Hence, our difficult di-\nagnosis of delayed vault site bleeding turned out to be a rare \ncase of vault site iatrogenic endometriosis.\nDiscussion\nEndometriosis is defined as the presence of endometrial tissue \nlesions or nodules that are similar to the endometrium but are \npresent at sites outside the uterus [3]. Common sites of endo-\nmetriosis include the ovaries, pelvic viscera and the peritoneum \nand the presentations can vary from a few minimal lesions \non pelvic organs to massive endometric ovary cysts or even \nextensive adhesions involving subperitoneal spaces, intestinal \nsystem, and urinary system [4,5].\nRare cases of extrapelvic endometriosis may result from vas-\ncular or lymphatic dissemination of endometrial cells to many \ngynecologic and non gynecologic sites. It has been reported \nthat endometriosis of the skin and soft tissue makes up about \n3.5% of cases of extrapelvic endometriosis with a majority of \nsuch cases occurring in surgical scars following operations of \nthe uterus [5-7].\nAlthough rare, there have been few reports of vaginal vault \nendometriosis with patients presenting with irregular or cyclic \nmenstrual bleeding several months or years after hysterectomy \n[8]. However, those cases had a history of a functional endo-\nmetriosis at the ovaries with adhesions or a fistulous tract to Fig. 1. Wine colored lesion on vault site at initial pelvic examination.\n\n\nwww.ogscience.org 321\nCha Hien Choi, et al. Vault site iatrogenic endometriosis\nthe vault or even some endometriotic spots left behind near \nthe vault site [9].\nUsually in cases of sudden bleeding with a history of prior \nhysterectomy, pathologic causes such as granulation tissue for-\nmation or malignancies such as cervical stump cancer must be \nexcluded [10]. Among the various complications of hysterec-\ntomy, here we report an incidence of a patient who developed \nendometriosis at the vaginal vault with no previous history of \nthe disease.\nThere have been some reports where endometriosis found \non scar tissues with no prior history of the disease. Cases such \nas scar endometriosis developing after a cesarean section are \none of them [11]. However vault endometriosis after hysterec-\ntomy is an extremely rare complication and therefore difficult \nto diagnose.\nIn our case, the previous hysterectomy had no evidence of \nendometriosis on other sites. Even the pathologist, although \nhaving suspicions for endometriosis, could not give a clear di-\nagnosis due to such a rare occurrence. However after examin-\ning the final vault excision specimen and correlating with the \nsymptoms with our patient, the pathologist confirmed both \nbiopsy results as a typical histology for endometriosis.\nThe possible pathophysiology in our particular case is sus -\npected to be endometrial fragment implantation to the vaginal \nvault site at the time of surgery. We believe a risk of develop-\nment into vault site endometriosis could be due to a uterus \nshowing adenomyosis and or maybe due to a secretory phase \nendometrium. Our patient was on the 25th to 26th day of \nher menstrual cycle during the previous surgery, where the \nendometrium thickness was about 9.2 mm and the pathology \nproved the endometrium to be at secretory phase. Implanta-\ntion during morcellation of the adenomyotic uterus or the \nsecretory phase endometrium probably had an effect on the \nliving tissues of the vagina, with transplantations to the site \nthrough cell adhesion mechanisms and then progression to \niatrogenic endometriosis of the vaginal vault [3,8]. This theory \nis highly possible although further investigation and studies are \nrequired.\nAlthough such complications are extremely rare, iatrogenic \nvault site endometriosis must be considered in delayed bleed-\ning occurring in a post-hysterectomy patient when other diag-\nnoses have been excluded. Due to the nature of these lesions \nbeing highly likely to recur, the principle of treatment as in any \nother extra pelvic endometriosis, is total surgical excision.\nConflict of interest\nNo potential conflict of interest relevant to this article was \nreported.\nReferences\n1. Guth U, Wagner S, Huang DJ, Hess TH. Pyoderma gan -\ngrenosum of the vaginal vault after vaginal hysterectomy: \nonly the correct diagnosis of a rare entity can prevent long-\nFig. 2. (A) Initial biopsy of the vault lesion showing unusual cells at the surface with sub-epithelial hemorrhage  (H&E, X40, X200). S14-41234, vagina, \nloss of surface epthelium with subepithelial hemorrhage. (B) The microscopic section of vaginal vault showing typical endometriosis  (H&E, X40, X200). \nS14-42249, vaginal vault, C/W endometriosis. C/W; compatible with\nA B\n\nwww.ogscience.org322\nVol. 58, No. 4, 2015\nterm morbidity. Arch Gynecol Obstet 2013;288:79-82.\n2. Benjamin MA, Yaakub HR, Telesinghe P , Kafeel G. A rare \ncase of abnormal uterine bleeding caused by cavernous \nhemangioma: a case report. J Med Case Rep 2010;4:136.\n3. Fraser IS. Recognising, understanding and managing en -\ndometriosis. J Hum Reprod Sci 2008;1:56-64.\n4. Berek JS. Berek and Novak’s gynecology. 15th ed. Philadel-\nphia: Lippincott Williams & Wilkins; 2012. \n5. Ramalingappa A, Reddy Y. A rare presentation of endo -\nmetriosis. J Obstet Gynaecol India 2012;62(Suppl 1):94-6.\n6. Mahendru R, Siwach S, Aggarwal D, Rana P , Duhan A, \nAggarwal T, et al. A rare case of endometriosis in vaginal \nhysterectomy scar. Ann Surg Innov Res 2013;7:6.\n7. Reich WJ, Nechtow MJ, Abrams R. Endometriosis of the \nvagina following vaginal hysterectomy. Am J Obstet Gyne-\ncol 1948;56:1192-4.\n8. Green TH Jr, Meigs JV. Pseudomenstruation from posthys-\nterectomy vaginal vault endometriosis. Obstet Gynecol \n1954;4:622-34.\n9. Aydin Y, Atis A, Ercan E, Donmez M. An endometriotic \nvault fistula presenting with monthly bleeding after hys -\nterectomy. Arch Gynecol Obstet 2009;280:1011-4.\n10. Marsden NJ, Wilson-Jones N. Scar endometriosis: a rare \nskin lesion presenting to the plastic surgeon. J Plast Re -\nconstr Aesthet Surg 2013;66:e111-3.\n11. Patil NJ, Kumar V, Gupta A. Scar endometriosis-a sequel \nof caesarean section. J Clin Diagn Res 2014;8:FD09-10.","source_license":"CC0","license_restricted":false}