{"paper_id":"a7520684-0cb1-4de5-94f6-1445187a3be5","body_text":"524Copyright © All rights are reserved by Kariman Ghazal. \n Interventions in Gynaecology  & \nWomen’s Healthcare\nCase ReportISSN: 2637-4544\nDOI: 10.32474/IGWHC.2022.05.000211\nAtipical presentations of endometriosis: \na case series\nKariman Ghazal1,2,3*, Ahmad Bayrouti1, Georges Yared1,5,6 and Jihad El Hasan1,4\n1Obstetrics and Gynaecology Department, Lebanese University, Beirut, Lebanon \n2Obstetrics and Gynaecology Department, Al Zahraa Hospital University Medical Centre, Beirut, Lebanon \n3Obstetrics and Gynaecology Department, Makassed General Hospital, Beirut, Lebanon\n4Head of Obstetrics and Gynecology Department, Al Zahraa Hospital University Medical Center, Beirut, Lebanon\n5Head of Obstetrics and Gynecology Department, Rafik Hariri Hospital University Medical Center, Beirut, Lebanon\n6Assistant Professor Obstetrics and Gynecology Department, American Lebanese University, Beirut, Lebanon\n*Corresponding author: Kariman Ghazal, Obstetrics and Gynaecology Department, Lebanese University, Lebanon\nReceived: \n  April 18, 2022                                                                                                                        Published: \n  April 25, 2022\nIntroduction\nEndometriosis is the growth of endometrium outside the \nuterus [1]. Endometriosis lesions are mainly located in the pelvis \nthough it could occur at other sites [2]. It is estrogen-dependent \nand occurs in around 10% of women in the reproductive age group \n[3]. Endometriosis has a broad spectrum of presentation [4]. \nIt could be internal located at the walls of the uterus or external \nlocated anywhere in the pelvis such as the ovaries, pouch of \nDouglas, or rectovaginal [5]. It could also develop in scars after \nsurgical procedures affecting the endometrium such as cesarean \nsection, hysterectomy, and episiotomy after normal delivery [6-8]. \nSymptoms vary among women though some could be asymptomatic. \nYet, it is often associated with chronic pelvic pain especially during \nmenses [1, 2]. Other symptoms include dysmenorrhea, dyspareunia, \nand infertility [9]. The diagnosis of endometriosis could be \nchallenging due to its variable presentations [6]. Ultrasonography,  \n \ncomputerized tomography, and magnetic resonance imaging can \nassist in preoperative diagnosis; however, they are associated \nwith some degree of uncertainty [10]. The mainstay of diagnosis \nis the visualization of lesions surgically whether by laparoscopy \nor laparotomy [3]. Nevertheless, histologic examination is needed \nto confirm the presence of endometriosis [1]. We hereby report a \nseries of multiple endometriosis presentations appearing during \npregnancy, at episiotomy and perineum, as well as after cesarean \ndelivery and hysterectomy.\nCases Series\nCase 1: Endometriosis during pregnancy and \nEndometriosis during pregnancy\n A 23-year-old woman gravida 0 para 0 presented with pain \nand amenorrhea 2. Pelvis ultrasound revealed 3 cm pelvic mass \n    Abstract\nEndometriosis has a broad spectrum of presentation The purpose of this article is to familiarize the gynecologist with the wide \nspectrum of pelvic and extra pelvic endometriosis and to review the distinctive imaging findings. Its clinical diagnosis could be \nchallenging since it is often confused with infection, abscess, hematoma, and tumors. We hereby report a series of multiple endome-\ntriosis presentations appearing during pregnancy, at episiotomy and perineum, as well as after cesarean delivery and hysterectomy. \nThe primary modality of treatment is surgical removal of the lesions, though hormonal therapy is also applied.\nKeywords: Endometriosis; Episiotomy; Hysterectomy\n\nCitation: Kariman Ghazal*, Ahmad Bayrouti, Georges Yared and Jihad El Hasan. Atipical presentations of endometriosis: a case series. Int \nGyn & Women’s Health 5(3)- 2022. IGWHC.MS.ID.000211. DOI: 10.32474/IGWHC.2022.05.000211.\n                                                                                                                                                          Volume 5 - Issue 3Int Gyn & Women’s Health Copyrights @ Kariman Ghazal 525\nat the right ovary with heterogeneous endometrioma-like aspect \n(Figure1) and luteinique cyst on the left ovary 3cm. After 3 months, \nthe patient became pregnant. During pregnancy, the patient had \nintermittent severe pain treated with analgesics (paracetamol and \nnon-steroidal anti-inflammatory drugs). There was no evidence \nfor cyst during pregnancy. At 37th week the patient had cesarean \nsection (CS) for fetal distress. During CS, there were multiple \nlesions of endometriosis in the pelvis (utero sacral ligament, pouch \nof Douglas, and vesico uterine space) as well as a ruptured ovarian \nmass that was diagnosed before pregnancy with diffuse opaque \ndark fluid in the pelvis (Figure 2). Cauterization and hemostasis \nwere done for some lesions. The newborn had an Apgar score of \n8 but was admitted to the Neonatal Intensive Care Unit (NICU) for \nrespiratory distress.\nFigure 1: Ultrasound image showing endometrioma on the on the right ovary before pregnancy. endometrioma (arrow) with \nhomogeneous low-level echoes no color flow will be seen and luteinique cyst on the left ovary.\nFigure 2: During CS, there were multiple lesions of endometriosis in the pelvis (utero sacral ligament, pouch of Douglas, and \nvesico uterine space) as well as a ruptured ovarian mass that was diagnosed before pregnancy Dark red or bluish cysts or \nnodules on the surface of peritoneal and pelvic organs.\nCase 2: Recurrent endometriosis on the cesarean section \nscar \nA 27-year-old female gravida 2 para2 who had two previous \ncesarean deliveries due to fetopelvic disproportion without \nany complication. She had a history of severe bleeding during \nmenses without experiencing severe pain. She takes estrogen and \nprogesterone contraceptives. The patient presented with pain on \nthe cesarean wound after 2 years from first cesarean. She stopped \ncontraceptive pills 5 months ago. Upon examination, a nodule on \nthe wound was found an approximately 3 cm wide, tender, strict, \n\nCitation: Kariman Ghazal*, Ahmad Bayrouti, Georges Yared and Jihad El Hasan. Atipical presentations of endometriosis: a case series. Int \nGyn & Women’s Health 5(3)- 2022. IGWHC.MS.ID.000211. DOI: 10.32474/IGWHC.2022.05.000211.\n                                                                                                                                                          Volume 5 - Issue 3 Copyrights @Kariman GhazalInt Gyn & Women’s Health 526\nand immobile right subcutaneous mass beneath the low segment \ncesarean scar. Pelvic and abdominal ultrasound showed a 3 cm mass \nwith heterogeneous echo structure (Figure 3a). Surgery was done \nto remove the mass (Figure 3b). Histology confirmed the diagnosis \nof endometriosis. After two years, the patient presented with pain. \nUltrasound examination revealed a 3 cm nodule on other side of the \nwound. (Figure 4a). She had surgery for recurrent endometriosis \n(Figure 4b).\nFigure 3a: Pelvic and abdominal ultrasound showed a 3 cm × 3 cm × cm, oval-shaped heterogeneous mass within the right \nrectus abdominus muscle, with no abnormalities of the uterus and ovaries Partly cystic and partly solid intramuscular lesion \nin the abdominal wall.\nFigure 3b: Excised mass having brown aspect inside.\nFigure 4a: pelvic and abdominal Ultrasound image showing heterogenous aspect mass under the skin of cesarean scar at the \nleft side: solid intramuscular lesion in the abdominal wall.\nFigure 4b: Excised mass having brown aspect inside.\nCase 3: Endometriosis in the groin near cesarean section \nscar\n A 27-year-old woman gravida 3 para2 1 abortion (1 NVD and \n1 CS) patient presented with a painful swelling in her groin. She \nhad been symptomatic for 8 months. Inguinal hernia was initially \nsuspected. She reported that her groin pain increased during \nher periods. She had a cesarean delivery 1year ago. Abdominal \nexamination revealed a palpable 3 cm mass in the groin area \nat the right side and lateral to her CS scar. MRI was done and \nshowed a 3 cm mass with a mixed signal intensity near her CS scar. \nEndometriosis was suspected. The patient had a laparotomy at her \ngroin during which lesions were found and completely removed. \nHistology confirmed the diagnosis of endometriosis.\nCase 4: Endometriosis on the episiotomy \n A 26-year-old woman gravida 2 para1 1 abortion (1 NVD) \npatient presented with a painful nodule over the episiotomy site \nfor two years. She had forceps delivery seven years ago. Upon \nexamination, a tender, irregular, dark-colored nodule measuring \n4 cm was found in the right perineal region over the previous \nepisiotomy scar. She underwent surgery during which the scar and \nthe nodule were excised. Histopathology showed endometriosis \nwith chronic inflammation at the episiotomy scar. The patient \nwas relieved of her presenting complaints after the surgery. She \ntook progestin medication Dienogest (Visanne) for endometriosis \ntreatment for 6 months and then she became pregnant without any \ncomplication.\n\nCitation: Kariman Ghazal*, Ahmad Bayrouti, Georges Yared and Jihad El Hasan. Atipical presentations of endometriosis: a case series. Int \nGyn & Women’s Health 5(3)- 2022. IGWHC.MS.ID.000211. DOI: 10.32474/IGWHC.2022.05.000211.\n                                                                                                                                                          Volume 5 - Issue 3Int Gyn & Women’s Health Copyrights @ Kariman Ghazal 527\nCase 5: Deep perineal endometriosis behind episiotomy \n A previously healthy 28-year-old woman gravida 2 para1 1 \nabortion had a 1-year history of a painful palpable lesion within the \ndeep left perineum. The pain was correlating with her menstrual \nperiod. Gynecological ultrasound examination was normal. \nPhysical examination revealed a deep firm mass in the perineum \ninferior to the left labia majora. A punch biopsy was performed. \nHistopathology showed multiple foci of endometrial glands and \ndense stroma surrounded by tissue. The patient was diagnosed as \nhaving endometriosis and was treated with Dienogest for 6 months. \nShe was asymptomatic but when she stopped the medication the \nsevere pain returned. She was advised to undergo surgery during \nwhich multiple lesions of endometriosis were excised. The patient \nnow is asymptomatic and is being treated with contraceptives and \nDienogest.\nCase 6: Endometriosis of the vaginal vault \nA 41-year-old woman gravida 0 para0 presented with prolonged \nlower pelvic discomfort, constipation, and severe vaginal bleeding. \nThe patient had a history of abdominal hysterectomy 3 months \nago due to symptomatic adenomyosis and multiple leiomyomas. \nAfter 2 months of surgery, the patient started to have severe \nvaginal bleeding. She consulted a physician who detected polypoid \nlesions protruding from the suture of previous hysterectomy at \nthe vaginal vault (Figures 5a & 5b). Her vital signs were stable \nwithout fever. Pelvic and vaginal exam induced severe pain during \nmanipulation of the vault site and polypoid lesions were suspected \nas an old hematoma of the hysterectomy site. Pelvic and abdominal \nultrasound examinations showed no signs of active bleeding \nat the surgery site. For further investigation, under general \nanesthesia, polypoid lesions were removed with sponge forceps \nand Di thermocoagulation. Bleeding was controlled with sutures \nand tampon gauzes that were inserted intra-vaginally and were \nremoved after one day. Histology confirmed endometriosis on the \nscar. The patient underwent treatment with Dienogest.\nFigure 5(a b): Polypoid lesions protruding from the suture of previous hysterectomy at the vaginal vault.\nDiscussion \n Endometriosis is a gynecologic pathology that most commonly \ndevelop in the pelvis, such as ovaries, pouch of Douglas, uterosacral \nligaments, and anterior abdominal wall [4,5]. The endometrial \nlesions are composed of glands and stroma that functionally respond \nto exogenous and endogenous hormonal stimuli. The presentation \nand evolution are variable, ranging from few lesions on pelvic \norgans to massive extensive adhesions involving the intestinal \nand urinary systems [3-5]. Endometriosis occurs in women of \nreproductive age. It may also develop during pregnancy [11-14]. \nBean et al. reported that the prevalence of ovarian endometriomas \nand deep endometriosis in women in their early pregnancy was \n4.9% [15]. Ovarian endometriomas during pregnancy are usually \ndiagnosed by ultrasonography due to its high accuracy and safety \n[11]. Complications of endometriosis during pregnancy are rare \n[11]. These include increased risk of placenta previa, placental \nabruption, hypertension, spontaneous abortion, preterm birth, \nand cesarean delivery [14]. However, there is no evidence that \nendometriosis has a detrimental effect on pregnancy outcome \n[11,12]. In our case, the patient did not have any complication. She \nonly had diffuse pain that was medically treated. On the other hand, \nendometriosis can develop in the scar after obstetric or gynecologic \nsurgeries [6,16,17]. \nThe incidence of scar endometriosis after cesarean delivery \nwas reported to range between 0.08% and 0.95% [10,18]. The \nsymptoms of scar endometriosis are nonspecific, usually involving \npain and swelling at the incision site [6,17]. It can be clinically \nmisdiagnosed as granuloma, hernia, abscess, lipoma, or hematoma \n[6,10]. Preliminary diagnosis is made through medical history and \nphysical examination in addition to imaging. Ultrasonography is \ncommonly used. Sonographic features are generally not specific \nshowing irregular borders, heterogeneous echotexture, and \nincreased vascularity [6,16]. Accurate diagnosis can be done by \nhistopathological examination of the tissue [6,10,17]. Treatment \ninvolves the surgical removal of the lesion [6,10,17]. Kaplanoglu et \nal. suggested that the complete excision of the lesion together with \n\nCitation: Kariman Ghazal*, Ahmad Bayrouti, Georges Yared and Jihad El Hasan. Atipical presentations of endometriosis: a case series. Int \nGyn & Women’s Health 5(3)- 2022. IGWHC.MS.ID.000211. DOI: 10.32474/IGWHC.2022.05.000211.\n                                                                                                                                                          Volume 5 - Issue 3 Copyrights @Kariman GhazalInt Gyn & Women’s Health 528\naround 1 cm of healthy tissue could help to prevent local recurrence \n[6] since recurrence rate ranged from 1.5% to 9.1% [19, 20]. In our \nsecond case, the patient had recurrent scar endometriosis but on a \ndifferent side since the first lesion was on the right of the scar while \nthe second lesion was central. Scar endometriosis could also rarely \noccur in scars resulting from episiotomy [8,21,22]. The incidence \nof endometriosis at episiotomy site after normal vaginal delivery \nwas reported 0.01%-0.04% [6,21]. It may be misdiagnosed as \ngranuloma, cyst or abscess [23]. The presence of a perineal nodule \nor tender mass, along with cyclic perineal pain with a history of \nan episiotomy, could be highly indicative of endometriosis [8]. \nPhysical examination usually reveals a dark blue perineal mass \n[5,8]. The first choice of treatment is complete excision of the \nperineal endometrial tissue. Other treatment modalities include \nhormonal suppression [8]. Early treatment is essential since \nextensive perineal endometriosis can extend to the anal sphincter \nnecessitating primary sphincteroplasty in addition to surgical \nexcision [24]. Another rare type of endometriosis is vaginal vault \nendometriosis that could develop after hysterectomy [7,25,26].\nChoi et al. and Chen et al. reported two cases of vaginal vault \nendometriosis post vaginal hysterectomy who presented with \nsudden vaginal bleeding without having previous evidence of \nendometriosis. A possible pathophysiology could be endometrial \nimplantation during hysterectomy [25,26]. Similar to other forms \nof endometriosis, the primary treatment is total surgical excision \n[25,26]. Although surgical removal of the lesions is recommended; \nthe use of medications such as Dienogest has shown to be effective \n[27]. Dienogest is a progestin that binds to progesterone receptors \nand inhibits gonadotropin secretion. It also has anti-inflammatory \nand antiproliferative effects on endometriotic lesions; thereby, \nproviding several advantages over combined hormonal \ncontraceptives [27].\nConclusion\nEndometriosis is associated with considerable morbidity. It \nis still an underdiagnosed disease. Further studies are needed to \ndetermine preventative measures. Clinical trials are also necessary \nto determine effective medical therapies, along with surgical \ntreatment, to prevent disease progression, minimize pain, and \nimprove fertility.\nConflict of Interest\nNo conflict of interest was reported. \nFunding Information\nNone.\nReferences\n1. Lapp T (2000) ACOG issues recommendations for the management of \nendometriosis. American Family Physician 62(6): 1431-1432.\n2. Khan KN, Kitajima M, Hiraki K, Fujishita A, Nakashima M, et al. (2014) \nVisible and occult microscopic lesions of endometriosis. Gynecology and \nMinimally Invasive Therapy 3(4): 109-114.\n3. Giudice LC, Kao LC (2004) Endometriosis. Lancet 364: 1789-1799.\n4. Sud S, Buxi TB, Sheth S, Ghuman SS (2021) Endometriosis and Its Myriad \nPresentations: Magnetic Resonance Imaging-Based Pictorial Review. \nIndian Journal of Radiology and Imaging. 31(1): 193-202.\n5. Acién P , Velasco I (2013) Endometriosis: a disease that remains \nenigmatic. ISRN Obstetrics and Gynecology.\n6. Kaplanoglu M, Kaplanoğlu DK, Dincer Ata C, Buyukkurt S (2014) \nObstetric scar endometriosis: retrospective study on 19 cases and \nreview of the literature. International Scholarly Research Notices pp: \n1-5.\n7. Mahendru R, Siwach S, Aggarwal D, Rana P , Duhan A, et al. (2013) A rare \ncase of endometriosis in vaginal hysterectomy scar. Annals of Surgical \nInnovation and Research 7: 6.\n8. Tam T , Huang S (2012) Perineal endometriosis in an episiotomy scar: \ncase report and review of literature. Journal of Endometriosis 4(2): 93-\n96.\n9. Zondervan KT , Becker CM, Missmer SA (2020) Endometriosis. New \nEngland Journal of Medicine 382: 1244-1256.\n10. Ozel L, Sagiroglu J, Unal A, Unal E, Gunes P , et al. (2012) Abdominal \nwall endometriosis in the cesarean section surgical scar: a potential \ndiagnostic pitfall. Journal of Obstetrics and Gynaecology Research 38(3): \n526-530.\n11. Leone Roberti Maggiore U, Ferrero S, Mangili G, Bergamini A, Inversetti \nA, et al. (2016) A systematic review on endometriosis during pregnancy: \ndiagnosis, misdiagnosis, complications, and outcomes. Human \nReproduction Update 22(1): 70-103.\n12. Mekaru K, Masamoto H, Sugiyama H, Asato K, Heshiki C, et al. (2014) \nEndometriosis and pregnancy outcome: are pregnancies complicated \nby endometriosis a high-risk group? European Journal of Obstetrics & \nGynecology and Reproductive Biology 172: 36-39.\n13. Ueda Y, Enomoto T , Miyatake T , Fujita M, Yamamoto R, et al. (2010) A \nretrospective analysis of ovarian endometriosis during pregnancy. \nFertility and Sterility 94(1): 78-84.\n14. Exacoustos C, Lauriola I, Lazzeri L, De Felice G, Zupi E (2016) \nComplications during pregnancy and delivery in women with untreated \nrectovaginal deep infiltrating endometriosis. Fertility and Sterility \n106(5): 1129-1135.\n15. Bean E, Naftalin J, Horne A, Saridogan E, Cutner A, et al. (2021) Prevalence \nof deep and ovarian endometriosis in early pregnancy: an ultrasound \ndiagnostic study. Ultrasound in Obstetrics & Gynecology 59(1): 107-113.\n16. Tatli F, Gozeneli O, Uyanikoglu H, Uzunkoy A, Yalcın HC, et al. (2018) The \nclinical characteristics and surgical approach of scar endometriosis: \nA case series of 14 women. Bosnian Journal of Basic Medical Sciences \n18(3): 275-278.\n17. Sumathy S, Mangalakanthi J, Purushothaman K, Sharma D, Remadevi \nC, et al. (2017) Symptomatology and surgical perspective of scar \nendometriosis: a case series of 16 women. The Journal of Obstetrics and \nGynecology of India 67(3): 218-223.\n18. Minaglia S, Mishell Jr DR, Ballard CA (2007) Incisional endometriomas \nafter Cesarean section: a case series. The Journal of Reproductive \nMedicine 52(7): 630-634.\n19. Ding Y, Zhu J (2013) A retrospective review of abdominal wall \nendometriosis in Shanghai, China. International Journal of Gynecology \n& Obstetrics 121(1): 41-44.\n20. Bektaş H, Bilsel Y, Sarı YS, Ersöz F, Koç O, et al. (2010) Abdominal wall \nendometrioma; a 10-year experience and brief review of the literature. \nJournal of Surgical Research 164(1): e77-e81.\n\nCitation: Kariman Ghazal*, Ahmad Bayrouti, Georges Yared and Jihad El Hasan. Atipical presentations of endometriosis: a case series. Int \nGyn & Women’s Health 5(3)- 2022. IGWHC.MS.ID.000211. DOI: 10.32474/IGWHC.2022.05.000211.\n                                                                                                                                                          Volume 5 - Issue 3Int Gyn & Women’s Health Copyrights @ Kariman Ghazal 529\n21. Leite GK, Carvalho LF, Korkes H, Guazzelli TF, Kenj G, et al. (2009) Scar \nendometrioma following obstetric surgical incisions: retrospective \nstudy on 33 cases and review of the literature. Sao Paulo Medical Journal \n127(5): 270-277.\n22. Demir M, Yildiz A, Ocal I, Yetimalar MH, Kilic D, et al. (2014) Endometriosis \nin episiotomy scar: a case report. J Cases Obstet Gynecol 1: 8-10.\n23. Dadhwal V, Sharma A, Khoiwal K, Nakra T (2018) Episiotomy scar \nendometriosis. Medical Journal Armed Forces India. 74(3): 297-299.\n24. Chen N, Zhu L, Lang J, Liu Z, Sun D, et al. (2012) The clinical features and \nmanagement of perineal endometriosis with anal sphincter involvement: \na clinical analysis of 31 cases. Human Reproduction 27(6): 1624-1627.\n25. Chen X, Zhu J (2018) Vaginal cuff endometriosis after laparoscopic-\nassisted vaginal hysterectomy: a case report and literature review. \nInternational Journal of Clinical and Experimental Medicine 11: 6336-\n6339.\n26. Choi CH, Kim JJ, Kim WY, Min KW, Kim DH (2015) A rare case of \npost-hysterectomy vault site iatrogenic endometriosis. Obstetrics & \nGynecology Science 58(4): 319-322. \n27. Ally Murji, Kutay Biberoğlu, Jinhua Leng, Michael D. Mueller, Thomas \nRömer (2020) Use of dienogest in endometriosis: a narrative literature \nreview and expert commentary. Current Medical Research and Opinion.\nInterventions in Gynecology and  \nWomen’s Healthcare\nAssets of Publishing with us\n• Global archiving of articles\n• Immediate, unrestricted online access\n• Rigorous Peer Review Process\n• Authors Retain Copyrights\n• Unique DOI for all articles\nThis work is licensed under Creative\nCommons Attribution 4.0 License\nTo Submit Y our Article Click Here:       \nSubmit Article\nDOI: 10.32474/IGWHC.2022.05.000211","source_license":"CC0","license_restricted":false}