A 45 year old woman presented with right sided ovarian mass with multiple omental deposits and liver metastases. The right ovary was enlarged and showed a partly cystic partly solid cut surface. Histological picture showed clear cell carcinoma with areas of mucinous cystadenoma and endometriosis. Clear cell carcinoma is known to be associated with endometriosis. To the best of author\u27s knowledge, it\u27s association with mucinous cystadenoma has been described only once in the literature, where clear cell carcinoma was shown to be associated with mucinous cystadenoma without any evidence of endometriosis

Hasan, Sheema H

Kayani, Naila

Uddin, Zeeshan

Yaqoob, Nausheen

English

eCommons@AKU

eCommons@AKUDepartment of Pathology and Laboratory Medicine Medical College, PakistanJuly 2007Clear cell carcinoma of ovary with associatedmucinous cystadenoma and endometriosisZeeshan UddinAga Khan University, zeeshan.uddin@aku.eduNausheen YaqoobAga Khan UniversityNaila KayaniAga Khan University, naila.kayani@aku.eduSheema H. HasanAga Khan University, sheema.hasan@aku.eduFollow this and additional works at: https://ecommons.aku.edu/pakistan_fhs_mc_pathol_microbiolPart of the Microbiology Commons, and the Oncology CommonsRecommended CitationUddin, Z., Yaqoob, N., Kayani, N., Hasan, S. H. (2007). Clear cell carcinoma of ovary with associated mucinous cystadenoma andendometriosis. Journal of Pakistan Medical Association, 57(7), 373-5.Available at: https://ecommons.aku.edu/pakistan_fhs_mc_pathol_microbiol/791Case ReportClear cell carcinoma of ovary with associated mucinous cystadenoma andendometriosisZeeshan-ud-din, Nausheen Yaqoob, Naila Kayani, Sheema H HasanDepartment of Pathology & Microbiology, The Aga Khan University Hospital, Karachi.AbstractA 45 year old woman presented with right sidedovarian mass with multiple omental deposits and livermetastases. The right ovary was enlarged and showed apartly cystic partly solid cut surface. Histological pictureshowed clear cell carcinoma with areas of mucinouscystadenoma and endometriosis. Clear cell carcinoma isknown to be associated with endometriosis. To the best ofauthor's knowledge, it's association with mucinouscystadenoma has been described only once in the literature,where clear cell carcinoma was shown to be associated withmucinous cystadenoma without any evidence ofendometriosis.IntroductionClear cell carcinoma is a relatively uncommon typeof ovarian tumour which comprises 2.4% of ovarianepithelial neoplasms.1 Although they were initially calledmesonephromas and mesonephric carcinomas, theirepithelial nature is now accepted due to their highassociation with pelvic endometriosis.2,3We describe a case of clear cell carcinoma of theovary in association with mucinous cystadenoma andendometriosis. To the best of our knowledge, only one suchcase has been published previously in the literature by NDutt et al4, in which clear cell carcinoma was describedassociated with mucinous cystadenoma without anyevidence of endometriosis.4 This association was criticizedby WG Mccluggage as according to him the mucinousareas represented mucinous metaplasia of theendometriotic cyst of ovary.5 However in our case, therewere separate foci of mucinous cystadenoma andVol. 57, No. 7, July 2007 373endometriosis coexisting with areas of clear cellcarcinoma, supporting the case of N Dutt et al.Case ReportA 45 year old woman presented to the clinician withabdominal pain and distension since one month. Ultrasoundexamination revealed a right-sided ovarian mass withmultiple omental deposits and liver metastases. Per-operatively, in addition to the enlarged right ovary, the leftovary was also found to be enlarged, and the omentumstudded with tumour deposits. Bilateral salpingo-ophorectomy was performed along with an omental biopsywhich was sent for histological evaluation.Grossly the right ovary was enlarged, measuring 9.5x 9 x 6 cm. with ruptured capsule and a partly cystic, partlysolid cut surface. The solid areas were pale brown, soft,necrotic and spongy. The cystic spaces were filled withmucinous secretions and included a haemorrhagic cyst. Theleft ovary measured 6 x 6 x 4 cm. and showed cystic spacesfilled with dark brown haemorrhagic material.Histological examination of the right ovary revealedan infiltrating neoplasm composed of solid sheets and nestsof large polyhedral clear cells having pleomorphic nucleiwith clumped chromatin and prominent nucleoli (Figure 1).In some areas multiple layers of these cells were seen liningthe cystic spaces with hobnail appearance focally (Figure2). Tumour cells showed cytoplasmic glycogen on specialstaining and immunohistochemically, they were shown tobe positive for cytokeratin CAM 5.2 and AE1/AE3 andwere negative for alphafetoprotein. Cystic spaces lined by asingle layer of tall columnar mucinous cells were seen inclose proximity to the tumour. There was no cytologicalatypia or complexity of architecture in these areas. Therewere foci of endometriosis in the adjacent ovarian tissue.Based on the features, described a diagnosis of clear cellcarcinoma of the ovary associated with benign mucinouscystadenoma and endometriosis was made. The right ovaryshowed features consistent with mucinous cystadenomaalong with areas of endometriosis. Deposits of clear cellcarcinoma were identified in the omental biopsy. DiscussionClear cell carcinoma was first described in 1939 bySchiller, who called it a "mesonephroma" of ovary due to itspresumptive origin from the mesonephric rests in the femalegenital tract.3,4 In view of their association with pelvicendometriosis and endometriotic cysts, the frequentadmixture with typical endometroid carcinoma, andultrastructural similarities with mullerian epithelial cells,their epithelial origin is now generally accepted.2,6 Clearcell carcinoma comprises 2.4% of ovarian epithelialneoplasms and 7.4% of ovarian carcinomas.1 The patientsare usually in their fifth or sixth decades of life.1,7Interestingly, they are the most common epithelial ovarianneoplasms to be associated with paraneoplastichypercalcaemia.1 Clear cell carcinoma has an incidence ofassociated pelvic endometriosis in 50% and endometriosisin the same ovary in 24% of the cases.2,4,8 Some authorsbelieve that this incidence of association may be muchhigher if ovarian clear cell carcinomas are extensivelysampled because of the possible obliteration ofendometriosis by the tumour.5,8 Benign and borderline clearcell tumours of ovary  are very rare and most are malignantwith a 5 year survival rate ranging from 37-47%.4,7,9Histologically different patterns have been described out ofwhich solid and tubulopapillary are most common.1,7,9,10The cells are polygonal with abundant clear cytoplasmseparated by delicate fibrovascular septae with papillarycores often exhibiting hyalinization. In the tubulopapillarypattern, the cells show a hobnail appearance. CytoplasmicFigure 1. Clear cell carcinoma of ovary showing sheets of polygomal cells with clear cytoplasm. X40.Figure 2. Ovarian tissue showing clear cell carcinoma (long arrow) with area exhibiting liningepithelium of mucinous cystadenoma (short arrow). X20.374 J Pak Med Assocglycogen in the favour of PAS positive, diastase resistanthyaline globules is seen.1,7 Immunohistochemically, thetumour cells are positive for cytokeratins, epithelialmembrane antigen and vimentin and are negative for alpha-fetoprotein. The differential diagnosis includes yolk sactumour, dysgerminoma and metastatic renal cellcarcinoma.1The presence of intracytoplasmic lumina withmucinous inclusions are described in clear cell carcinomawhich, according to some authors, support the hypothesisthat clear cell carcinoma should be regarded as an endstage transformation, which may arise from any of theother epithelial tumours.4 Association of clear cellcarcinoma with mucinous lesions has been described onlyinfrequently. One such case was described by N Dutt et al.where clear cell carcinoma was seen associated withmucinous cystadenoma.4 This association was criticizedby WG Mccluggage, according to whom the mucinousareas were in fact reproductive mucinous metaplasia ofthe endometriotic cyst of ovary.5 According to him,mucinous metaplasia in endometriotic cysts can be soextensive that a diagnosis of mucinous cystadenoma maybe considered.5 However, in our case there were separatefoci of mucinous cystadenoma and endometriosiscoexisting with areas of clear cell carcinoma. There wasno continuity between the areas of endometriosis andmucinous cystadenoma in our case. This favours the caseof N Dutt et al who suggested the origin of clear cellcarcinoma in a background of mucinous cystadenoma.References1. Robert JK. Blaustein's Pathology of Female Genital Tract. 5th ed. New Delhi(India): Springer; 2002. p. 873-8.2. Brescia RJ, Dubin N, Demopoulos RI. Endometrioid and clear cell carcinomaof the ovary. Factors affecting survival. Int J Gynecol Pathol 1989;8:132-8.3. Schiller W. Mesonephroma ovarii. Am J Cancer1939;35:1-21.4. Dutt N, Berney DM. Clear cell carcinoma of the ovary arising in mucinouscystadenoma. J Clin Pathol 2000;53:938-9.5. McCluggage WG. Clear cell carcinoma of the ovary arising in a mucinouscystadenoma. J Clin Pathol 2001;54:655-6.6. Scully RE, Barlow JF. Mesonephroma of ovary. Tumour of mullerian naturerelated to the endometrioid carcinoma. Cancer 1967;20:1405-17.7. Rosai J. Rosai and Ackerman's Surgical Pathology. 9th ed. St. Louise (MO):Elsevier; 2004. pp. 1669-71.8. Imachi M, Tsukamoto N, Shimamoto T, Hirakawa T, Uehira K, Nakano H.Clear cell carcinoma of the ovary: a clinicopathologic analysis of 34 cases. IntJ Gynecol Cancer 1991;1:113-9.9. Czernolbilsky B, Silverman BB, Enterline HT. Clear cell carcinoma of theovary. A clinicopathologic analysis of pure and mixed forms and comparisonwith endometriod carcinoma. Cancer 1970;25:762-72.10. Montag AG, Jenison EL, Griffiths CT, Welch WR, Lavin PT, Knapp RC.Ovarian clear cell carcinoma. A clinicopathologic analysis of 44 cases. Int JGynecol 1989;8:85-96.Vol. 57, No. 7, July 2007 375

Clear cell carcinoma of ovary with associated mucinous cystadenoma and endometriosis

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Clear cell carcinoma of ovary with associated mucinous cystadenoma and endometriosis

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