{"paper_id":"eda2d38f-a70b-4fb8-8651-ad2ff8aafa33","body_text":"C A S E R E P O R T Open Access\nUmbilical endosalpingiosis: a case report\nTheodossis S Papavramidis 1*, Konstantinos Sapalidis 1, Nick Michalopoulos 1, Georgia Karayannopoulou 2,\nAngeliki Cheva 2, Spiros T Papavramidis 1\nAbstract\nIntroduction: Endosalpingiosis describes the ectopic growth of Fallopian tube epithelium. Pathology confirms the\npresence of a tube-like epithelium containing three types of cells: ciliated, columnar cells; non-ciliated, columnar\nsecretory mucous cells; and intercalary cells.\nWe report the case of a woman with umbilical endosalpingiosis and examine the nature and characteristics of\ncutaneous endosalpingiosis by reviewing and combining the other four cases existing in the international\nliterature.\nCase presentation: A 50-year-old Caucasian, Greek woman presented with a pale brown nodule in her umbilicus.\nThe nodule was asymptomatic, with no cyclical discomfort or variation in size. Her personal medical, surgical and\ngynecologic history was uneventful. An excision within healthy margins was performed under local anesthesia.\nA cystic formation measuring 2.7×1.7×1 cm was removed. Histological examination confirmed umbilical\nendosalpingiosis.\nConclusions: Umbilical endosalpingiosis is a very rare manifestation of the non-neoplasmatic disorders of the\nMüllerian system. It appears with cyclic symptoms of pain and swelling of the umbilicus, but not always. The\ndisease is diagnosed using pathologic findings and surgical excision is the definitive treatment.\nIntroduction\nEndosalpingiosis is a rare clinical entity that describes\nthe ectopic growth of Fallopian tube epithelium [1].\nEndosalpingiosis, endometriosis and endocervicosis con-\nstitute the triad of non-neoplastic disorders of the Mül-\nlerian system. These pathologies are found in isolation,\nbut are more commonly found in association with one\nanother [2,3]. The diagnosis of these pathologies is\nmade histologically. In the case of endosalpingiosis,\npathology confirms the presence of a tube-like epithe-\nlium containing three types o f cells: ciliated, columnar\ncells; non-ciliated, columnar mucous secretor cells; and\nthe so-called intercalary or peg cells [4,5].\nWe report the case of an adult woman with umbilical\nendosalpingiosis and elucidate the nature and character-\nistics of cutaneous endosal pingiosis by reviewing and\ncombining the four cases existing in the international\nliterature.\nCase presentation\nA 50-year-old Caucasian, Greek woman presented with\na pale brown nodule in her umbilicus. The nodule was\nasymptomatic, with no cyclical discomfort or variation\nin size. Her personal medical, surgical and gynecologic\nhistory was uneventful. An excision within healthy mar-\ngins was performed under local anesthesia. A cystic for-\nmation measuring 2.7×1.7 ×1 cm was removed. There\nwere no signs of malignancy and no evidence of endo-\nmetrial component in her surrounding tissue. Pathologic\nexamination showed a unilocular cyst with papillary\nprojections into the lumen (Figures 1 and 2). The cyst\nwas surrounded by edematous fibrous tissue. The lining\nconsisted of epithelium cells, both cuboidal and ciliate\n(Figure 3). Immunohistochemical staining showed posi-\ntivity for keratins AE1/AE3 (Figure 4). Finally, a histolo-\ngical examination posed the diagnosis of cutaneous\nendosalpingiosis.\nDiscussion\nThe term endosalpingiosis was employed for the first\ntime by Sampson et al . in 1930. Under that term, the\nauthor designated any unusual growth and invasion of\n* Correspondence: papavramidis@hotmail.com\n1Department of Surgery, AHEPA University Hospital, Aristotle University of\nThessaloniki, Thessaloniki, Greece\nFull list of author information is available at the end of the article\nPapavramidis et al . Journal of Medical Case Reports 2010, 4:287\nhttp://www.jmedicalcasereports.com/content/4/1/287 JOURNAL OF MEDICAL\nCASE REPORTS\n© 2010 Papavramidis et al; licensee BioMed Central Ltd. This is an Open Access article distributed under the terms of the Creative\nCommons Attribution License (http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and\nreproduction in any medium, provided the original work is properly cited.\n\ntubal epithelium in tubal stumps, in subjects who had\nundergone previous salpingec tomy or tubal sterilization\n[1]. The different theories for the pathogenesis of endo-\nsalpingiosis are similar to those for endometriosis, since\nthose two entities, together w ith endocervicosis, consti-\ntute the non-neoplastic disorders of the Müllerian sys-\ntem. The different models can be traced back to two\nbasic ideas. One group of theories is based on the fact\nthat endometrial cells (or their precursors) are trans-\nported by various routes (transtubal, hematogenous,\nlymphogenous or by direct apposition) and implanted in\nthe affected organ. The other group of theories suggests\nthat Müllerian ectopias are the result of metaplastic pro-\ncesses in the target organ (co elomic metaplasia theory,\nsecondary Müllerian system) or from scattered embryo-\nnic rest [6-8]. We believe that the first group of theories\nis inadequate when explaining the origin of endosalpin-\ngiosis in our case report, since she has a free gynecolo-\ngic, obstetric and surgical hi story. In our case report, it\nis likely that the Müllerian ectopia resulted either from\nmetaplastic processed or scattered embryonic rest.\nNon-neoplastic glandular proliferation showing spon-\ntaneous Müllerian differentiation has been described in\nmany sites including the vagina [9], uterine cervix [10],\nurinary bladder [11,12], appendix [13], peritoneum\n[14,15], abdominal wall (in guinal channel, umbilicus)\n[2,16], and the lymph nodes [17]. However, to the best\nof our knowledge, this is the first case of a patient with\nspontaneous endosalpingiosis presenting as a nodule on\nthe abdominal wall.\nThe differential diagnosis of nodular umbilical lesions\nshould include a wide range of diseases; such as hernia,\nkeloid, abscess, lipoma, hematoma, sebaceous cyst, ade-\nnocarcinoma [primary of metastatic (Sister Joseph\nnodule)], melanoma, suture granuloma, pyogenic granu-\nloma, desmoid tumor, sarcoma, lymphoma, endometrio-\nsis, and endosalpingiosis. Of course, the final diagnosis\nshould be made by pathology [18].\nIn the international literature, this is the fifth case of\numbilical endosalpingiosis. The other four cases refer to\npatients with previous medical history of gynecologic\nprocedures [5,19,20], while this is the first case of spon-\ntaneous appearance. These lesions appear as nodules of\nthe umbilicus and are usually brownish in colour. The\nmain symptoms (besides the es thetic) are pain and size\nfluctuation with menstruat ion. The treatment of choice\nis surgical excision. Our opinion is that excision has to\nbe made under local anesthesia, in order to minimize\nmorbidity and hospitalization. However, the patient has\nto be notified that in the case of a reappearance of\nFigure 1 Unilocular cyst with papillary projections into the lumen (Hematoxylin and Eosin ×4) .\nFigure 2 Unilocular cyst with papillary projections into the lumen (Hematoxylin and Eosin ×10) .\nPapavramidis et al . Journal of Medical Case Reports 2010, 4:287\nhttp://www.jmedicalcasereports.com/content/4/1/287\nPage 2 of 4\n\nabdominal pain (especially in the lower quadrant), a\nlaparoscopy should be performed in order to exclude\nabdominal endometriosis.\nConclusions\nUmbilical endosalpingiosis is a very rare manifestation\nof the non-neoplasmatic disorders of the Müllerian sys-\ntem. Normally, it appears with cyclic symptoms of pain\nand swelling of the umbilicus, but not always. The dis-\nease is diagnosed using pathologic findings and surgical\nexcision is the definitive treatment.\nConsent\nWritten informed consent was obtained from the patient for publication of\nthis case report and any accompanying images. A copy of the written\nconsent is available for review by the Editor-in-Chief of this journal.\nCompeting interests\nThe authors declare that they have no competing interests.\nAuthors’ contributions\nTSP analyzed and interpreted the patient data and drafted the manuscript.\nKS received the patient in our out-patient department and was the principal\nsurgeon. NM received the patient in our out-patient department, was the\nauxillary surgeon and drafted the manuscript. GK performed the\npathological examination and was a major contributor in writing the\nmanuscript. AC performed the pathological examination. STP was\nresponsible for the overall treatment of the patient and corrected the\nmanuscript. All authors read and approved the final manuscript.\nAuthor details\n1Department of Surgery, AHEPA University Hospital, Aristotle University of\nThessaloniki, Thessaloniki, Greece. 2Department of Pathology, AHEPA\nUniversity Hospital, Aristotle University of Thessaloniki, Thessaloniki, Greece.\nReceived: 23 October 2009 Accepted: 24 August 2010\nPublished: 24 August 2010\nReferences\n1. Sampson JA: Postsalpingectomy endometriosis (endosalpingiosis). Am J\nObstet Gynecol 1930, 20:443-480.\n2. Apostolidis S, Michalopoulos A, Papavramidis TS, Papadopoulos VN,\nParamythiotis D, Harlaftis N: Inguinal endometriosis: three cases and\nliterature review. South Med J 2009, 102:206-207.\n3. Edmondson JD, Vogeley KJ, Howell JD, Koontz WW, Koo HP, Amaker B:\nEndosalpingiosis of bladder. J Urol 2002, 167:1401-1402.\n4. Butterworth S, Stewart M, Clark JV: Heterotopic ciliated epithelium -\nMüllerian origin? Lancet 1970, 1:1400-1401.\n5. Redondo P, Idoate M, Corella C: Cutaneous umbilical endosalpingiosis\nwith severe abdominal pain. J Eur Acad Dermatol Venereol 2001,\n15:179-180.\n6. Sinkre P, Hoang MP, Albores-Saavedra J: Mullerianosis of inguinal lymph\nnodes: report of a case. Int J Gynecol Pathol 2002, 21:60-64.\n7. Onybeke W, Brescia R, Eng K, Quagliarello J: Symptomatic endosalpingiosis\nin a postmenopausal woman. Am J Obstet Gynecol 1987, 156:924-926.\n8. Keltz MD, Kliman HJ, Arici AM, Olive DL: Endosalpingiosis found at\nlaparoscopy for chronic pelvic pain. Fertil Steril 1995, 64:482-485.\n9. Martinka M, Allaire C, Clement PB: Endocervicosis presenting as a painful\nvaginal mass: a case report. Int J Gynecol Pathol 1999, 18:274-276.\n10. Young RH, Clement PB: Endocervicosis involving the uterine cervix: a\nreport of four cases of a benign process that may be confused with\ndeeply invasive endocervical adenocarcinoma. Int J Gynecol Pathol 2000,\n19:322-328.\nFigure 3 The lining of the cyst consisted of epithelium cells cuboidal and ciliate (Hematoxylin and Eosin ×40) .\nFigure 4 Immunohistochemical staining shows positivity for keratins AE1/AE3 (Keratins AE1/AE3 ×40) .\nPapavramidis et al . Journal of Medical Case Reports 2010, 4:287\nhttp://www.jmedicalcasereports.com/content/4/1/287\nPage 3 of 4\n\n11. Nazeer T, Ro JY, Tornos C, Ordonez NG, Ayala AG: Endocervical type\nglands in urinary bladder: a clinicopathologic study of six cases. Hum\nPathol 1996, 27:816-820.\n12. Clement PB, Young RH: Endocervicosis of the urinary bladder. a report of\nsix cases of a benign müllerian lesion that may mimic adenocarcinoma.\nAm J Surg Pathol 1992, 16:533-542.\n13. Cajigas A, Axiotis CA: Endosalpingiosis of the vermiform appendix. Int J\nGynecol Pathol 1990, 9:291-295.\n14. Lauchlan SC: The secondary müllerian system. Obstet Gynecol Surv 1972,\n27:133-146.\n15. Zinsser KR, Wheeler JE: Endosalpingiosis in the omentum: a study of\nautopsy and surgical material. Am J Surg Pathol 1982, 6:109-117.\n16. Horton JD, Dezee KJ, Ahnfeldt EP, Wagner M: Abdominal wall\nendometriosis: a surgeon ’s perspective and review of 445 cases. Am J\nSurg 2008, 196:207-212.\n17. Ferguson BR, Bennington JL, Haber SL: Histochemistry of mucosubstances\nand histology of mixed müllerian pelvic lymph node glandular\ninclusions. Evidence of histogenesis by müllerian metaplasia of coelomic\nepithelium. Obstet Gynecol 1969, 33:617-625.\n18. Papavramidis TS, Sapalidis K, Michalopoulos N, Karayanopoulou G,\nRaptou G, Tzioufa V, Kesisoglou I, Papavramidis ST: Spontaneous\nabdominal wall endometriosis: a case report. Acta Chir Belg 2009,\n109:778-781.\n19. Doré N, Landry M, Cadotte M, Schürch W: Cutaneous endosalpingiosis.\nArch Dermatol 1980, 116:909-912.\n20. Perera GK, Watson KM, Salisbury J, Du Vivier AW: Two cases of cutaneous\numbilical endosalpingiosis. Br J Dermatol 2004, 151:924-925.\ndoi:10.1186/1752-1947-4-287\nCite this article as: Papavramidis et al .: Umbilical endosalpingiosis: a\ncase report. Journal of Medical Case Reports 2010 4:287.\nSubmit your next manuscript to BioMed Central\nand take full advantage of: \n• Convenient online submission\n• Thorough peer review\n• No space constraints or color figure charges\n• Immediate publication on acceptance\n• Inclusion in PubMed, CAS, Scopus and Google Scholar\n• Research which is freely available for redistribution\nSubmit your manuscript at \nwww.biomedcentral.com/submit\nPapavramidis et al . Journal of Medical Case Reports 2010, 4:287\nhttp://www.jmedicalcasereports.com/content/4/1/287\nPage 4 of 4","source_license":"CC0","license_restricted":false}