Benign cystic mesothelioma is a rare tumor originated from mesenchymal cells of the abdominal and pelvic peritoneum. This rare tumor should be considered in the differential diagnosis of young women presenting with abdominal pain and a multicystic abdominal mass.Keywords: Benign cystic mesothelioma; pelvic pain; dispareuni

Capp, Edison

Corleta, Helena von Eye

Komlós, Marcia

Lopes, Maria Francisca

English

Archives of the Faculty of Veterinary Medicine UFRGS

Revista HCPA 2004;24(1)Volume 24 (1)     •   32INTRODUCTIONBenign cystic mesothelioma (BCM) is a rare tumorthat originates in the peritoneum and may involve thesurface of the bladder, rectum, ovary or cul de sac. Theincidence is of 0.1-1.0 case per million people every year(1), and occurs mostly in women of reproductive age. Thewomen-men rate is 5:1.The present study reports a case of BCM diagnosedin a 21-year-old female presenting with abdominal painand a pelvic cystic mass.CASE REPORTA 21-year-old nulligravida, white, single, sexuallyactive woman had a history of pelvic pain and dispareuniaPELVIC PAIN AND DISPAREUNIA: A BENIGN CYSTICMESOTHELIOMA CASE REPORTDOR PÉLVICA E DISPAREUNIA: RELATO DE UM CASO DE MESOTELIOMACÍSTICO BENIGNOHelena von Eye Corleta1, Marcia Komlós2, Edison Capp1, Maria Francisca Lopes3ABSTRACTBenign cystic mesothelioma is a rare tumor originated from mesenchymal cells of theabdominal and pelvic peritoneum. This rare tumor should be considered in the differentialdiagnosis of young women presenting with abdominal pain and a multicystic abdominal mass.Key words: Benign cystic mesothelioma, pelvic pain, dispareunia.RESUMOO mesotelioma cístico benigno é um tumor raro originado de células mesenquimais doperitônio abdominal e pélvico. Este tumor raro deve ser considerado no diagnóstico diferencialde mulheres jovens apresentando dor abdominal e massa abdominal multicística.Unitermos: Mesotelioma cístico benigno, dor pélvica, dispareunia.1 Department of Obstetrics and Gynecology, School of Medicine, Universidade Federal do Rio Grande do Sul (UFRGS);Service of Gynecology and Obstetrics, Hospital de Clínicas de Porto Alegre, Porto Alegre, RS, Brazil; Clínica Gerarde Fertilização Assistida, Porto Alegre, RS, Brazil.2 Department of Obstetrics and Gynecology, School of Medicine, Universidade Federal do Rio Grande do Sul (UFRGS);Service of Gynecology and Obstetrics, Hospital de Clínicas de Porto Alegre, Porto Alegre, RS, Brazil.3 ANATPAT, Laboratory of Surgical Pathology, Porto Alegre, RS, Brazil.Correspondence: Dr. Helena Corleta, Rua Ramiro Barcelos, 910 cj 905, 90035-001, Porto Alegre, RS, Brazil. Fax: (+55-51) 3311-6588. E-mail: hcorleta@portoweb.com.br.for 6 months. Two previous ultrasounds were suggestiveof hydrosalpinx, and the patient had been treated forinflammatory pelvic disease, without improvement. Thepatient had been using oral contraceptives for 2 years.Abdominal examination on admission revealedabdominal distention; gynecologic examination wassuggestive of a pelvic mass. Videolaparoscopy wasperformed and revealed multiple cysts containing clearand gelatinous material, the largest with 5 cm in diameter.Fallopian tubes and ovaries were normal, as well as therest of the peritoneal cavity. The cysts were aspirated,and the capsule was sent to examination. The pelvicperitoneum of the cul de sac was cauterized. Pathologyreport revealed BCM.After surgery, the patient showed improvement ofsymptoms, and two years later, was asymptomatic, withoutevidence of recurrence.lato de CasoVolume 24 (1)     •   33BENIGN CYSTIC MESOTHELIOMA CASE REPORTDISCUSSIONMesotheliomas are mesenchymal tumors originatedfrom cells that cover serous cavities. In the peritoneum(site affected in 20% of cases), mesotheliomas may manifestthemselves as benign adenomatoid tumors, malignantmesotheliomas or BCM (2,3). Patients presenting withdiffuse malignant mesotheliomas show an overall mediansurvival of 6 to 10 months. Adenomatoid tumors are rareand are most commonly found in the genital tract. Theyare benign and do not tend to recur. BCM is a borderlinelesion between these two entities (4). Mennemeyer & Smith were the first to report themesothelial origin of these tumors, in 1979 (4). The etiologyremains obscure (3); however, a possible role of chronicperitoneal irritation and previous laparotomy has beendescribed (5). Reactive origin, previous history ofabdominal surgery, endometriosis, or pelvic inflammatorydisease may also be involved (6). The present patient hadbeen submitted to an abdominal surgery two years beforethe diagnosis of BCM. In contrast with what is observed inmalignant mesotheliomas, no relation with asbestosis wasfound.The most common symptoms associated with BCMinclude chronic pelvic or abdominal pain, distention, andpresence of abdominal mass on examination (6,7,8).Dispareunia, urinary urgency and frequency may also bepresent (2,6). Sometimes, abdominal mass is absent, andthe tumor is discovered incidentally on laparotomy. Thesetumors may grow in the peritoneal cavity and compressother organs. Invasion of organs has been observed but isdifficult to explain (7). Symptoms differ from those ofmalignant mesotheliomas, which present ascites in mostcases. Although the diagnosis of BCM may be suggestedby ultrasonography (US), computerized tomography (CT)and magnetic resonance (MR), definitive diagnosis is madeonly after histological examination (7).The cysts may range from several millimeters toseveral centimeters in diameter (7). They are filled with aclear, watery fluid, sometimes serous to gelatinous.Microscopic appearance consists of cystic spaces lined withone to several layers of cubical or flattened cells embeddedin a delicate fibrous stroma (figure 1A), with numerousmicrovilli and well formed desmosomal junctions. Theyoften have a “hobnail” or “picked fence” appearance (figure1B). Focuses of epithelial hyperplasia may also be noticed.Some atypias are often present and may lead to misdiagnosisof serous carcinoma or malignant mesothelioma (4).Differential diagnosis should be made with cysticmalignant mesothelioma, ovarian cystadenoma,cystadenocarcinoma, cystic lymphangioma, angiosarcomaof serous membranes, cystic smooth muscle tumors, visceralcysts, pancreatic neoplasms, endometriosis andadenomatoid tumors (7,8). Many of these pathologies aresimilar in terms of image findings and clinical appearanceand can be differentiated only by histological examination.Treatment consists of complete surgical resection,and preservation of pelvic organs is indicated (6).Recurrence may occur in as much as 50% of cases, andusually takes place 1 to 27 years after the primary treatment.Recurrence is also managed with resection, and morbidityis related to local dissemination and recurrence (2,7,8).Metastasis or malignant degeneration has never beenreported. Fatal complications related to local recurrenceand surgery have been described (9,10).In conclusion, the present study reported a case ofperitoneal BCM in a 21-year-old patient. The exact etiologywas unclear, but could be associated with a previousFigure 1. (A) Detail of one of the mesothelial-lined cysts embedded in a delicate fibrous stroma (H&E - 250X). (B) Hobnail-shaped cells in an areaof mesothelial hyperplasia (H&E-400X).A BRevista HCPA 2004;24(1)Volume 24 (1)     •   34surgery. Although peritoneal BCM is rare in occurrence, itshould be considered in the differential diagnosis of youngwomen presenting with abdominal pain and a multicysticabdominal mass (10).REFERENCES1. Toma GD, Nicolanti V, Plocco M. Cystic peritonealmesothelioma: report of a case. Surg Today2000;30(1):98-100.2. O’Neil JD, Ros PR, Storm BL, Buck JL, Wilkinson EJ.Cystic mesothelioma of the peritoneum. Radiology1989;170:333-7.3. Hatsube Y, Mukai K, Silverberg SG. Cysticmesothelioma of the peritoneum - report of five casesand review of the literature. Cancer 1982;50:1615-22.4. Mennemeyer R, Smith M. Multicystic, peritonealmesothelioma. Cancer 1979;44:692-8.5. Takenouchi Y, Oda K, Takahara O, et al. Report of acase of benign cystic mesothelioma. Am JGastroenterol 1995;90(7):1165-7.6. Schneider V, Partridge JR, Gutierrez F, Hurt WG,Maizels MS, Demay RM. Benign cystic mesotheliomainvolving the female genital tract: report of four cases.Am J Obstet Gynecol 1983;145(3):355-9.7. Ozgen A, Akata D, Akhan O, Tez M, Gedikoglu G,Ozmen MN. Giant benign cystic peritonealmesothelioma: US, CT, and MRI findings. AbdomImaging 1998;23:502-4.8. Omeroglu A, Husain A, Multilocular peritonealinclusion cyst (benign cystic mesothelioma). ArchPathol Lab Med 2001;125(8):1123-4.9. De Toma G, Nicolanti V, Plocco M, Cavallaro G,Amato D, Letizia C. Cystic peritoneal mesothelioma:report of a case. Surg Today, 2000; 30: 98-100.10. Weiss SW, Tavassoli FA. Multicystic mesothelioma. Ananalysis of pathologic findings and biologic behavior in37 cases. Am J Surg Pathol 1988;12:737-46.

PELVIC PAIN AND DISPAREUNIA: A BENIGN CYSTIC MESOTHELIOMA CASE REPORT

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PELVIC PAIN AND DISPAREUNIA: A BENIGN CYSTIC MESOTHELIOMA CASE REPORT

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