{"paper_id":"815abf05-2b3e-46b6-b20c-dd9257416ab1","body_text":"Archive of SID\nCASE REPORT  \n*Corresponding Author: Loabat Geranpayeh, MD. \nDepartment of Surgery, Sina Hospital, Tehran University of Medical Sciences, 14155-6537 Tehran, Iran \nTel: +98 21 66495948, Fax: +98 21 66495948, E-mail: geranpay@sina.tums.ac.ir \nSpontaneous Endometriosis of the Abdominal Wall \nLoabat Geranpaye1*, Mohsen Fadaei-Araghi1, Shirin Irani2, and Behnam Shakiba2\n1 Department of Surgery, Sina Hospital, School of Medicine, Tehran University of Medical Sciences, Tehran, Iran \n2 Students’ Scientific Research Center, School of Medicine, Tehran University of Medical Sciences, Tehran, Iran \nReceived:  8 Feb. 2008; Received in revised form:  3 Apr. 2008; Accepted:  18 May 2008  \nAbstract- Abdominal wall endometriosis, the commonest type of extra-pelvic endometriosis, may rarely \ndevelop spontaneously in the absence of prior surgical scar. We aim to report 3 cases with this unique charac-\nteristic, two of them with a diagnosis of unusual development of umbilical endometriosis and one with ingui-\nnal endometriosis, from the perspective of a general surgeon. Because of the potential pitfalls in its diagnosis, \nendometriosis should be emphasized in the differential diagnosis of abdominal wall masses. \n© 2009 Tehran University of Medical Sciences. All rights reserved. \nActa Medica Iranica 2009; 47(2): 154-156. \n \nKey words: Endometriosis, Spontaneous, umbilicus, inguinal \nIntroduction \n \nEndometriosis is characterized by the abnormal growth \nof endometrial tissue outside the uterine cavity. Most \nendometrial deposits are found in the pelvis including \novaries, peritoneum, uterosacral ligaments, pouch of \nDouglas, and rectovaginal septum, but it may rarely oc-\ncur in extra pelvic locations. \nThese include most body cavities, as well as organs \nincluding lung, gallbladder, bowel, kidney, central nerv-\nous system, extremities, perineum, and abdominal wall \n(1-4). Extrapelvic endometriosis may occur in up to 12 \npercent of women with endometriosis (5). Umbilical \nendometriosis is rare with an estimated incidence of 0.5 \nto 1.0 percent of all patients with endometrial ectopia.6 \nMore commonly, cutaneous endometriosis occurs in a \nsurgical scar from abdominal or pelvic procedures, \nwhich include hysterectomy, cesarean sections, episiot-\nomy and laparoscopy (7,8). As endometriosis is rarely \nseen by general surgeons and is often diagnosed on his-\ntological examination postoperatively due to a large \nnumber of potential pitfalls in its diagnosis, we report 3 \ncases ,two of them with a diagnosis of unusual devel-\nopment of umbilical endometriosis and one with ingui-\nnal endometriosis, from the perspective of a general sur-\ngeon. \n \nCase reports \n \nCase 1 \nA 40-year-old woman was referred to our general \nsurgery clinic due to a five month history of bleeding \nfrom her umbilicus which was concurrent with her men-\nstruation period. In her past medical history, she had the \ndiagnosis of uterine fibroma due to the mid cycle spot-\nting from 4 years ago. She had the history of 3 Natural \nvaginal deliveries. She had no history of pain or previ-\nous caesarean section or any other surgery. Ultrasono-\ngraphy (USG) revealed a myomatosis uterus with a right \nsubserosal myoma. She underwent abdominal hysterec-\ntomy and left oophorectomy. The uterus had a big sub-\nserosal myoma in the left fundus and two intramural \nmyomas in the corpus of the uterus. It was also a 1×0.5 \ncm mass in the umbilicus, which was totally excised. It \nwas confirmed to be endometriosis with histopathology. \nShe was discharged from the hospital after 1 day without \nany complication. \n \nCase 2 \nA 37-year-old woman presented with a blue-violet um-\nbilical nodule with bloody and pussy discharge. She had \ngynecological visit the previous year for the abnormal \nmenstruation and spotting with pain and abdominal mass \nsensation. USG revealed a 9x5 cm cystic lesion in the \nright adnexa and a multiseptate cystic lesion in the left. \nIt also revealed a solid lesion accompanied by a small \ncystic part which was extended from the right adnexa to \nthe abdominal wall which was suspected to be a uterine \nfibroma. During the surgery with sub and circumumbili-\ncal incision, fibromatous uterus was detected while two \nbig and subserosal fibromas were excised. The ovaries \nwere polycystic which the big ones were undergone cys-\ntectomy. There were also two prominent mass (ap-\nproximately 1×0.5 cm) in the umbilicus which were to-\nwww.SID.ir\n\nArchive of SID\nL. Geranpayeh, et al. \nActa Medica Iranica, Vol. 47, No. 2 (2009) 155 \ntally excised. The pathologic report from the ovaries and \numbilical masses was respectively endometriotic cysts \nand cutaneous endometriosis. She had bloody and pussy \ndischarge from the umbilicus after 5 months which ac-\ncompanied with pain and discomfort. It was revealed a \n2x2 painful black mass on examination with the above-\nmentioned characteristics. The patient again underwent a \nsurgery and the aforesaid mass which contained granula-\ntion tissue and hair was entirely excised. It was extended \nup to the peritoneum. The pathology reported endome-\ntriosis. She was discharged from hospital after 5 days \nwithout any complication. \n \nCase 3 \nA 38-year-old woman was referred to the surgical clinic \ndue to a painful inguinal mass. She had pain and dis-\ncomfort in the right inguinal region for about a year. She \nunderwent a surgery with the preoperative diagnosis of \nirreducible sliding hernia. There was no history of any \nprior surgery or caesarian section. During the surgery a \nmass with bloody discharge was detected on the medial \nside of the indirect hernia sac. The hernia sac was ex-\ncised with mentioned mass and the pathology report \nrevealed endometriosis of the hernia sac. She was dis-\ncharged from hospital after 2 days without any compli-\ncation. \n \nDiscussion \n \nDifferent pathophysiological theories concerning the \norigins of endometriosis have been proposed, including \nthe implantation or reflux, direct extension, coelomic \nmetaplasia (claiming that endometriosis develops from \nmetaplasia of peritoneum), induction (suggesting that \nsloughed endometrium results in endometriosis), em-\nbryonic rest (claiming a specific stimulus to a Mullerian \norigin cell nest produces endometriosis) and lymphatic \nand vascular metastasis (9). But we didn’t recognize a \nnet etiology for our three cases. The diagnosis of extra \ngenital endometriosis could be difficult due partly to a \nlow index of suspicion by both general practitioners and \nspecialists. Endometriosis of the skin is a well-\nrecognized pathologic entity usually encountered clini-\ncally as a periumbilical lesion or within abdominal sur-\ngical scars after cesarean section or other gynecologic \nprocedures (10). Umbilical endometriosis may also de-\nvelop in the surgical scar after abdominal procedures or \nthe trocar site of laparoscopic procedures, but it may \nrarely occur spontaneously in the physiologic scar of the \numbilicus (11), while our first two cases had this kind of \nendometriosis. Inguinal endometriosis is also rare and \nmay be difficult to recognize. Often it is confused with \nother more common disorders of the groin, such as lym-\nphadenopathy, hernia, granuloma, neuroma, abscess, \nlipoma, hematoma, soft-tissue tumor, metastatic cancer \nand subcutaneous cyst. The majority of cases are be-\nlieved to be caused by progression of pelvic endometrio-\nsis down the round ligament into the inguinal canal. In-\nguinal endometriosis has been described after gyneco-\nlogic surgery, and only a minority of cases has been as-\nsociated with a hernial sac (12), such as our third pa-\ntient. She had no history of any prior surgery or caesar-\nian section. Although the reasons for right-sided pre-\ndominance have not been identified, over 90% of ingui-\nnal endometriosis occurs on the right (13) and the disor-\nder occurred on the right side of our case too. This case \nmay be of interest to the general surgeon who commonly \nmanages patients with groin masses but does not deal \noften with endometriosis. Hernias associated with en-\ndometriosis may not be clinically detectable. even \nthough no proven explanation can be offered; the scar-\nring reaction that surrounds endometrial foci may reduce \ntissue elasticity, thus hindering hernia detection on \nphysical examination (14). \nA blue-violet painful nodule with symptoms of \nbleeding or discharge synchronous with the menstrual \ncycle is pathognomonic for endometriosis. Only one \n(case 1) of our patients had these characteristics. Pain \nwas a remarkable complaint in two (case 2 and 3) of our \npatients, but they didn’t have a cyclic pain which could \nhelp in establishing the diagnosis. A preoperative ultra-\nsonographic examination has helped to determine the \nsize of the lesion and whether the mass is cystic or solid \nand has been useful in excluding underlying intra-\nabdominal pathologic factors (15,16). But the reported \nappearance of endometriosis on ultrasonographic ex-\namination is nonspecific and may change during the \ncourse of a menstrual cycle (17-19). Altogether ultra-\nsonography should be applied preoperatively in a suspi-\ncious case of endometriosis. We had also applied USG \nfor all our three cases. Computed tomographic scans and \nmagnetic resonance imaging are also useful in ruling out \nincisional hernias and showing a direct association of \nthe mass and the abdominal wall, but they do not pro-\nvide a definitive preoperative diagnosis (15,16,20), we \ndidn’t use these techniques preoperatively. Fine-needle \naspiration biopsy had been reported in some studies to \nhelp confirm the diagnosis and effectively eliminate the \npossibility of malignancy (16,21), but in other studies it \nhas been reported of inconclusive help or needless \n(20,22), or even to increase the risk of recurrence (23). \nA large retrospective study has concluded that fine-\nwww.SID.ir\n\nArchive of SID\nSpontaneous Endometriosis \n156 Acta Medica Iranica, Vol. 47, No. 2 (2009) \nneedle aspiration (FNA) or percutaneous biopsy should \nnot be suggested when endometriosis is suspected (24), \nso we did not perform FNA in our patients. Simple sur-\ngical excision of the endometriosis is the treatment of \nchoice, with sparing of the umbilicus when possible, in \nthe case of umbilical endometriosis (25) Local recur-\nrence after adequate surgical excision is uncommon. \nMalignancy of ectopic endometrial tissue has also \nbeen reported (26). Thus wide excision of endometriosis \nin the present cases enabled us to gain clear surgical \nmargins to avoid leaving the rest of the ectopic tissue. \nThe possibility of coexisting pelvic endometriosis \nshould be investigated by Postoperative follow-up with a \ngynecologist. Endometriosis should be considered in any \nwoman of childbearing age with a painful or tender ab-\ndominal mass. Imaging studies such as ultrasound, CT \nor MRI are non-specific; thus, a biopsy is necessary to \nmake a definitive diagnosis. Endometriosis should be \nconsidered in the differential diagnosis of abdominal \nwall lesions even if not associated with a scar of a previ-\nous operation. Biopsy is diagnostic and surgical excision \nis curative (27). In conclusion, although there are very \nfew cases reported in the literature in whom a surgical \nhistory was absent (13), all our three patients had this \nunique characteristic. And since it is often diagnosed \nonly upon histological examination postoperatively, it \nshould be emphasized in the differential diagnosis of \nabdominal wall masses. \n \nReferences  \n \n1. Steck WD, Helwig EB. Cutaneous endometriosis. Clin \nObstet Gynecol 1966; 9:373-383. \n2. Franklin RR, Navarro C. Extragenital endometriosis. Prog \nClin Biol Res 1990; 323:289-295.  \n3. Yu CH, Perez-Reyes M, Brown JJ, Borrello JA. MR ap-\npearance of umbilical endometriosis. J Comput Assist To-\nmogr 1994; 18:267-271. \n4. Sataloff DM, La Vorgna KA, McFarland MM. Extrapelvic \nendometriosis presenting as a hernia: clinical reports and \nreview of the literature. Surgery 1989; 105:109-112. \n5. Franklin RR, Navarro C. Extragenital endometriosis. In: \nChadha DR, Buttran VC, eds. Current Concepts in Endo-\nmetriosis. New York: Alan R. Liss, Inc. 1990, p. 289. \n6. Michowitz M, Baratz M, Stavorovsky M. Endometriosis of \nthe umbilicus. Dermatologica 1983; 167(6):326-330. \n7. Steck WD. Cutaneous endometriosis. JAMA 1965; 191:167.  \n8. Albrecht LE, Tron V, Rivers JK. Cutaneous endometriosis. \nInternational Journal of Dermatology 1995 Apr; 34(4):261-\n262. \n9. Witz CA. Current concepts in the pathogenesis of endome-\ntriosis. Clin Obstet Gynecol 1999; 42:566-585. \n10. Fair KP, Patterson JW, Murphy RJ, Rudd RJ. Cutaneous \ndeciduosis. J Am Acad Dermatol 2000; 43:102-107. \n11. Majeski J, Craggie J. Scar Endometriosis Developing After \nan Umbilical Hernia Repair with Mesh. Southern Medical \nAssociation 2004; 97:532-534. \n12. Brzezinski A, Durst AL. Endometriosis presenting as an \ninguinal hernia. AmJ Obstet Gynecol 1983; 146:982-983. \n13. Candiani GB, Vercellini P, Fedele L,Nicoletta V, Carinelli \nS, Scagkione V. Inguinalendometriosis: pathogenetic and \nclinical implications. Obstet Gynecol 1991; 78:191-194. \n14. Boggi U, Del Chiaro M, Pietrabissa A, Mosca F. \nExtrapelvic endometriosis associated with occult groin \nhernias. Canadian Medical Association 2001; 44(3):224. \n15. Yu CY, Perez-Reyes M, Brown JJ, Borrello JA. MR ap-\npearance of umbilical endometriosis. J Comput Assist To-\nmogr 1994; 18:267-271. \n16. Wolf GC, Singh KB. Cesarean scar endometriosis: a \nreview. Obstet Gynecol Surv 1989; 44:89-95. \n17. Patterson GK, Winburn GB. Abdominal wall endometrio-\nmas: report of eight cases. Am Surgeon 1999; 65:36-39. \n18. Alexiadis G, Lambropoulou M, Deftereos S, \nGiatromanolaki A, Sivridis E, Manavis J. Abdominal wall \nendometriosis: ultrasound research: a diagnostic problem. \nClin Exp Obstet Gynecol 2001; 28:121-122. \n19. Nirula R, Greaney GC. Incisional endometriosis: an under-\nappreciated diagnosis in general surgery. J Am Coll Surg \n2000; 190:404-407.  \n20. Dwivedi AJ, Agrawal SN, Silva YJ. Abdominal wall \nendometriomas. Dig Dis Sci 2002; 47:456-4561.  \n21. Simsir A, Thorner K, Waisman J, Cangiarella J. Endome-\ntriosis in abdominal scars: a report of three cases diagnosed \nby fine-needle aspiration biopsy. Am Surgeon 2001; \n67:984-986. \n22. Koger KE, Shatney CH, Hodge K, McClenathan JH. \nSurgical scar endometrioma. Surg Gynecol Obstet 1993; \n177:243-246. \n23. Matthes G, Zabel DD, Nastala CL, Shestak KC. Endo-\nmetrioma of the abdominal wall following combined ab-\ndominoplasty and hysterectomy: case report and review of \nthe literature. Ann Plast Surg 1998; 40:672-675. \n24. Zhao X, Lang J, Leng J, Liu Z, Sun D, Zhu L. Abdominal \nwall endometriomas. International Journal of Gynecology \nand Obstetrics 2005; 90:218-222. \n25. Purvis RS, Tyring SK.Cutaneous and subcutaneous endo-\nmetriosis surgical and hormonal therapy. Journal of Derma-\ntologic Surgery and Oncology 1994 Oct; 20(10):693-695. \n26. Hitti IF,Glasberg SS,Lubicz S. Clear cell carcinoma arising \nin extraovarian endometriosis: report of three cases and \nreview of the literature. Gynecol Oncol 1990; 39:314-320. \n27. Ideyi SC, Schein M, Niazi M, Gerst PH. Spontaneous \nEndometriosis of the Abdominal Wall. Digestive Surgery \n2003; 20:246-248. \nwww.SID.ir","source_license":"CC0","license_restricted":false}