{"paper_id":"229952da-b5a0-4735-af07-cbf38a0c1d59","body_text":"Tubocutaneous Fistula due to Endometriosis – A\nDifferential Diagnosis in Cutaneous Fistulas with\nCyclic Secretion\nFístula tubocutânea secundária à endometriose– diagnóstico\ndiferenciado em fístulas cutâneas com secreção cíclica\nEdinari Nunes de Sousa Lopes 1 L i aC r u zV a zd aC o s t aD a m á s i o1 Laio Santana Passos 1\n1 Department of Gynecology, Universidade Federal do Piauí - UFPI,\nTeresina, Brazil\nRev Bras Ginecol Obstet 2017;39:31 –34.\nAddress for correspondence Edinari Nunes de Sousa Lopes, MD,\nDepartmento de Ginecologia, Universidade Federal do Piauí - UFPI, Av.\nUniversitária – Ininga, Teresina, PI, Brazil 64049-550\n(e-mail: edinarinunes@hotmail.com).\nIntroduction\nEndometriosis is a benign disease de fined by the presence of\nendometrial glands and stroma outside the uterus.1 The average\nage at diagnosis is between 25 and 35 years. 2,3 Endometriosis\noutside the pelvis is rare, and most cases occur in surgical scars\nafter procedures involving the female genital tract. 4 In this\nreport, we present a rare case of tubocutaneous fistula due to\nendometriosis that developed after a cesarean section. The\nfistula stretched from the left uterine tube to the left inguinal\nregion along the anatomical path of the round ligament.\nCase Report\nA 34-year-old woman sought the surgery service of the\nUniversity Hospital of Teresina in August 2015 due to a\nhistory of discharge from a cutaneous opening in the left\niliac fossa that had varied in color from citric yellow to red\nKeywords\n► gynecology\n► endometriosis\n► fistula\nAbstract The development of a tubocutaneous fistula due to endometriosis in a post-cesarean\nsection surgical scar is a rare complication that generates signi ficant morbidity in the\naffected women. Surgery is the treatment of choice in these cases. Hormonal therapies\nmay lead to an improvement in symptoms, but do not eradicate such lesions. In this\nreport, we present a 34-year-old patient with a cutaneous fistula in the left iliac fossa\nwith cyclic secretion. Anamnesis, a physical examination, and supplementary tests led\nus to suggest endometriosis as the main diagnosis, which was con firmed after surgical\nintervention.\nPalavras-chave\n► ginecologia\n► endometriose\n► fístula\nResumo O desenvolvimento de fístula tubocutânea secundária à endometriose em cicatriz\ncirúrgica após cesariana é uma complicação rara, que gera importante morbidade às\nmulheres acometidas. A cirurgia é o tratam ento de escolha nesses casos. Terapias\nhormonais podem conduzir a uma melhora dos sintomas, mas, de forma alguma,\nlevam à erradicação de tais lesões. No presente relato, temos uma paciente de 34 anos\nde idade que apresentava uma fístula cutânea em fossa ilíaca esquerda com secreção\ncíclica. Anamnese, exame físico e exames complementares nos levaram a aventar como\nprincipal hipótese diagnóstica a endometriose, que foi con firmada após intervenção\ncirúrgica.\nreceived\nJune 11, 2016\naccepted\nNovember 17, 2016\nDOI http://dx.doi.org/\n10.1055/s-0036-1597754.\nISSN 0100-7203.\nCopyright © 2017 by Thieme-Revinter\nPublicações Ltda, Rio de Janeiro, Brazil\nTHIEME\nCase Report 31\n\n\nand had exhibited cyclical behavior over the course of six\nyears. She presented with the following obstetrical history:\none miscarriage late in 1999; two vaginal deliveries at term,\none in 2002 and another in 2003; and one cesarean section in\n2008, which interrupted a pregnancy of between 35 and\n36 weeks, due to history of anemia and severe thrombocyto-\npenia associated with maternal-fetal Rh incompatibility.\nRegarding the medical history related to the cesarean birth,\nthe patient reported that six months after the cesarean section,\nshe resumed regular menstrual cycles, which were associated\nwith pain and redness in the left iliac fossa, abdominal disten-\nsion, and fever. At the time, fluid collection in the left inguinal\nregion was diagnosed, and drainage was performed with seros-\nanguineous secretion. The patient showed a partial improve-\nment in the symptoms, and, a few months later, a serous\ndischarge in the collection area began, which was cyclical,\nand it appeared eight days before menstruation and lasted until\nthe end of the menstrual period. Thereafter, she was subjected\nto two more surgical procedures, including adhesiolysis in the\nfistula tract and drainage collection. The patient was in posses-\nsion of the histopathological results of the latter approach\n(performed in 2013), which showed a foreign body-type chronic\ngranulomatous inflammation and nonspecificl y m p h a d e n i t i s .\nBased on this clinical background, the patient was referred to\nthe hospital’s gynecology team with a diagnosis of tubocuta-\nneous fistula due to endometriosis. A gynecological examina-\ntion revealed the presence of a pfannenstiel scar and a\nlongitudinal scar on the left iliac fossa, and a speculum exami-\nnation showed no communication of the vaginal walls with the\nfistula. The patient underwent additional tests. An abdominal\nultrasound examination showed: a hypoechoic tract with\na diameter of 0.5 cm, located in the left inguinal region\nFig. 1 Abdominal ultrasound showing a hypoechoic tract located in the left inguinal region (panel A); a hypoechoic area located in the\nsubcutaneous tissue, suggesting fluid collection, and showing communication with the external environment through the aforementioned tract\n(panel B); and a second hypoechoic area, showing communication with the aforementioned lesion, located close to the abdominal and internal\noblique rectal muscles, suggestive of fluid collection (panel C).\nRev Bras Ginecol Obstet Vol. 39 No. 1/2017\nTubocutaneous Fistula due to Endometriosis Lopes et al.32\n\n\n(►Fig. 1A ); a hypoechoic area, with partially de fined limits,\nlocated in the subcutaneous tissue, measuring 5.2 /C21.2 cm,\nsuggesting fluid collection, and showing communication with\nthe external environment through the aforementioned tract\n(►Fig. 1B ); and a second hypoechoic area, with partially\ndefined limits, showing communication with the aforemen-\ntioned lesion, located close to the abdominal and internal\noblique rectal muscles, measuring 4.5 /C22.5 cm, suggestive of\nfluid collection (►Fig. 1C ). Magnetic resonance imaging (MRI)\nof the pelvis showed laminar pelvic fluid collection on the\nposterior aspect of the rectus abdominis muscle on the left, next\nto the pubis, measuring /C246.2 /C20.9 cm, extending to the\nipsilateral iliac fossa. In this area, a fistulous tract could be\nobserved that pierced the musculature, forming another sub-\ncutaneous laminarfluid collection area measuring/C245.3 /C21.0\ncm and draining at its lower portion toward the cutaneous\nfistula (\n►Fig. 2 ). Fistulography revealed a contrast-filled cavi-\ntation in the subcutaneous area of the inguinal region, extend-\ning laterally to the left iliac fossa for /C248t o1 0c m ,w i t h o u t\ncommunication with the viscera or deep planes (►Fig. 3 ).\nGiven that the patient had no cutaneous opening dis-\ncharge at that time, she was discharged from the hospital\nwith guidance to return at the first sign of secretion drainage,\nwhich occurred 10 days afterwards ( ►Fig. 4 ). She was\nreadmitted and underwent surgery, which was performed\nover two sessions by gynecology, general surgery, and urol-\nogy specialists on September 21, 2015. Initially, a surgical\nhysteroscopy was performed, in which a normal uterine\ncavity was observed with tubal ostia, and no presence of\nlesions was detected. However, an outlet of solution was\nobserved, which was used for distension during the proce-\ndure, via the cutaneous opening of the fistula. Methylene\nblue was not visible after introduction into the fistula hole.\nLater, a laparotomy was performed with supra-aponeurotic\nresection and excision of the entire wall and fistulous tract,\nwhich exhibited the discharge of a chocolatey secretion. At\nthe opening of the aponeurosis, a new extraperitoneal\ncollection area could be seen closely adhering to the left\nhorn. A left salpingectomy was performed with the excision\nof the entire wall of the collection area. The histopathology\nresults con firmed the diagnosis of endometriosis.\nThe patient was readmitted on the 14th postoperative\nday for drainage of the purulent secretion via the surgical\nwound. She was given antibiotic therapy guided by the\nsecretion culture; she recovered well, and was discharged.\nSeven weeks after the procedure, she was readmitted at an\noutpatient basis and exhibited complete healing of the\nwound without secretion drainage or pain complaints; the\npatient was in amenorrhea due to the continued use of\ndesogestrel.\nFig. 4 Physical examination showing secretion drainage of the\ncutaneous opening in the left iliac fossa.\nFig. 3 Fistulography showing a contrast- filled cavitation in the\nsubcutaneous area of the inguinal region, extending laterally to the\nleft iliac fossa for /C248 to 10 cm, without communication with the\nviscera or deep planes.\nFig. 2 Magnetic resonance imaging of the pelvis showing laminar\npelvic fluid collection on the posterior aspect of the rectus abdominis\nmuscle on the left, extending to the ipsilateral iliac fossa. In this area, a\nfistulous tract could be observed that pierced the musculature,\nforming another subcutaneous laminar fluid collection area and\ndraining at its lower portion toward the cutaneous fistula.\nRev Bras Ginecol Obstet Vol. 39 No. 1/2017\nTubocutaneous Fistula due to Endometriosis Lopes et al. 33\n\n\nDiscussion\nExtrapelvic endometriosis may be associated with a wide\nvariety of cyclic symptoms re flecting the affected organs. 1\nPhysical findings, when present, are related to the location\nand extent of the disease, 5 and greater diagnostic sensitivity\nis present when the patient is investigated during menstru-\nation.6 The present case showed a clinical background highly\nsuggestive of endometriosis due to the cyclical character of\nthe presence of fistula debit and the characteristics of the\ndrained content, which varied in color from citric yellow to\nbright red.\nDevelopment of endometrioma in the surgical scar after\ncesarean section is a rare complication, with a reported\nfrequency of no more than 0.4%.\n7 The possible mechanisms\ninvolved in the formation of female genital tract fistulas\ninclude previous pelvic surgery, the use of drains, surgical\nwound dehiscence, and invasive endometriosis.\n8–10 In the\npresent case, the possible triggering factors included the\noccurrence of a previous cesarean section and the use of\ndrains in the left iliac fossa collection area. The endometrial\ntissue may have been implanted in the surgical scar, causing\nthe erosion of the underlying tissue through a cyclical\ninflammatory process.\nMagnetic resonance imaging is superior to transvaginal\nultrasound for detecting the peritoneal implants of endome-\ntriosis and collections, but it still identi fies only 30 –40% of\nthe lesions observed during surgery.\n11 The patient in ques-\ntion was submitted to the cited additional tests, which\ndiagnosed the presence of two liquid collection areas, but\ndid not show the connection of these collection areas with\nthe ipsilateral fallopian tube.\nSurgery is the treatment of choice in cases of endometri-\notic fistula. Hormone therapy may lead to an improvement in\nsymptoms, but does not eradicate such lesions.\n12,13 To avoid\nrecurrence and the emergence of additional complications,\nthe patient underwent complete surgical excision of the\nlesion. Because the patient was young, and in order to avoid\nchronic problems resulting from estrogen deprivation, we\ndecided to preserve the ovaries.\nIn summary, we present an extremely rare case of an\nendometriotic tubocutaneous fistula, the description and\nliterature review of which provide greater awareness of\nthis clinical entity, offering useful information for the correct\ndiagnosis and treatment.\nReferences\n1 Fritz MA, Speroff L. Endocrinologia, ginecologia e infertilidade.\nRio de Janeiro: Revinter; 2015\n2 Kuohung W, Jones GL, Vitonis AF, et al. Characteristics of patients\nwith endometriosis in the United States and the United Kingdom.\nFertil Steril 2002;78(4):767 –772\n3 Hediger ML, Hartnett HJ, Louis GM. Association of endometriosis\nwith body size and figure. Fertil Steril 2005;84(5):1366 –1374\n4 Taff L, Jones S. Cesarean scar endometriosis. 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Acta Obstet\nGynecol Scand 1984;63(2):183 –184\n10 Gupta SK, Shukla VK, Varma DN, Roy SK. Uterocutaneous fistula.\nPostgrad Med J 1993;69(816):822 –823\n11 Stratton P, Winkel C, Premkumar A, et al. Diagnostic accuracy of\nlaparoscopy, magnetic resonance imaging, and histopathologic\nexamination for the detection of endometriosis. Fertil Steril 2003;\n79(5):1078–1085\n12 Purvis RS, Tyring SK. Cutaneous and subcutaneous endometriosis.\nSurgical and hormonal therapy. J Dermatol Surg Oncol 1994;20-\n(10):693–695\n13 Rivlin ME, Das SK, Patel RB, Meeks GR. Leuprolide acetate in the\nmanagement of cesarean scar endometriosis. Obstet Gynecol\n1995;85(5 Pt 2):838 –839\nRev Bras Ginecol Obstet Vol. 39 No. 1/2017\nTubocutaneous Fistula due to Endometriosis Lopes et al.34","source_license":"CC0","license_restricted":false}