Mesothelioma of the Tunica Vaginalis Testis: Report of 2 Cases with Asbestos Occupational Exposure

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1 International Journal of Surgical Pathology 13(2): , 2005 Mesothelioma of the Tunica Vaginalis Testis: Report of 2 Cases with Asbestos Occupational Exposure Giuseppe Gorini, MD,* Marco Pinelli, MD,* Vincenzo Sforza, MD,t Umberto Simi, MD,* Andrea Rinnovati, MD, and Giuseppe Zocchi, MD" We repnrt iiere 2 cases of malignant mesothelioma of the tunica vaginalis testis. A 67- year-uld man with a left testicular mass was referred for left inguinal orchidectomy. Histologic examination showed a malignant mesothelioma ol epithelial type. There is no evidence of recurrence at 2-year follow-up. The patient had been exposed to asbestos 12 years previously for a 30-year period. An 80-year-oId man presented a 5- year fiistory of scrotal swelling. Clinical examination revealed a hydrocele. The patient underwent resection of the tunica vaginalis through scroial incision. Microscopic examination showed a malignant mesothelioma of hiphasic type. There is no evidence ol recurrence ai 2-year follow-up. The patient had been exposed to asbestos 52 years previously lor a 5-year [leriod. Inl J Stsr^ Pathol!3(2):2! 1-214, 2005 Key words: malignant mesotheliiima, tunica vajiiiuilis testis, asbestos exposure. Malignant mesotbelioma of the tunica vaginalis testis is a rare disease with an unfavorable course in most cases [1]. Among malignanl mcsotheliomas, a very small perentage (< 5%) arise in the tunica vaginalis testis [2. Mesotbelioma of the tunica vaginalis most commonly presents between the fifth and seventh decades, hut 10% of the cases have occurred in patients younger tban 25 years [1]. Asbestos exposure and tratima have been implicated as risk factors for the development of this tu- *E[iviri)riiiiL'nUil Jiid Oeciipatioiial Epidfiiiiolony Uiiil, CSPO, FlortMice; ^Pjllinlogy Unit, USL 8, Are/zo: 'Paihology Unil. USL 6. Leghorn; ^Siirgfry Unit. USL 8, Bibbicna Hospiial, Arc//o; and "*Surgcry Unit, USL 6, Ct'cina Hosiiilal. Lfjihorn, Italy. Rcprini reqiicsis: Giuseppe Gorini, MD, Tnstan Mesothelinnia Registry, Finvironmeinal and Occupational Epkicniiology Unit, Centt-r forslndy and Prevention of Cancer (CSPO), via di S.Saivi, Florence, Italy. mor l,3,4, which is associated with hydrocele in 56.3% of the cases [5]. Accurate preoperative diagnosis is difficult. We report here 2 cases of this rare tumor, boih with a clearly defined occupational asbestos exposure. Materials and Methods The cases were retrieved from the consultation files of the population-based Tuscan Malignant Mesothelioma Register (ARTMM). Since 1988 the ARTMM has recorded cases of malignant mesothelioma occurring in Tuscan residents (3.5 million total population, 1991 Census) 16-8]. ft is )art of the Italian Natitnial Mesothelioma Register (ReNaM), set up in 1993 [9,10]. Occupational history, lifestyle habits, and areas of residence are obtained by interviewing patients directly, or their relatives, using a standard questionnaire, administered by trained in- 211

2 212 International Journal of Surgical Pathology Vol. 13 No. 2 April 2005 terviewers. Asbestos exposure is classified by an industrial bygienist using a standard grid, according to national guidelines [10. Further details can be fotind elsewbere [6,8]. Hematoxylin and eosin-stained slides were reviewed and immunohistochemical stainings on formalin-fixed, paraffin-embedded tissues were performed in hoth cases. Follow-up data of patients were obtained by contacting primary physicians and patbologists. Case 1 Results A 67-year-old man was referred to Bihbiena I^ospital, Arezzo, Italy, in May 2002 because of an umbilical bernia and a left groin hernia. On clinical examination, he was found to have a firm, stiff, and painless left testicular mass, highly suggestive of tumor. Ultrasound examination confirmed the presence of a mass in the left testicle. Alpbafeto-protein and chorionic gonadotropin (HCG) levels were within normal limits. A left inguinal orchidectomy was perfortned. Grossly, the tunica vaginalis appeared diffusely thickened by a whitish, firm tissue. On tnicroscopic examination, there was an atypical epithelial pro]iferation with a papillary, exophytic pattern of growth (Fig. 1), witb easily visible scattered psammoma bodies (Fig. 2). Cholesterol-like clefts and granulomatous reaction in the underlying stroma were also present. There was no evidence of testicular or spermatic cord invasion. Immunohistochemical examination of the ttimor showed positivity for AE1/AE3 cytokeratins (Zymed, 1:50), EMA (Bio-Genex, 1:150), HBMFl (Dako, 1:50), and vimentin (Bio-Genex 1:100). Carcinoembryotic antigen (CEA; Bio-Genex, 1:100) was negative. The tumor was diagnosed as mesothelioma of epithelial type. There is no evidence of recurrence at 2 year follow-up after orchidectomy. During the period the patient was etnployed in the railway transports. During this 30-year period be had heen regularly exposed to asbestos when carrying out repairing and maintenance of locomotives. Case 2 An 80-year-old man presented to Cecina Hospital, Leghorn, Italy, in May 2002, with a 5-year history of a painless right-sided scrotal swelling. Clinical examination revealed a hydrocele with a palpably nt)rmal testis. These findings were confirmed on ultrasound scan. Serum alpbaleto-prolein and chorionic gonadotropin (HCG) levels were within normal limits. Tbe patient underwent resection of the tunica vaginalis through scrotal incision. Grossly, tbe tunica vaginalis was diffusely thickened and showed a central whitish nodule of about I cm diameter. Microscopically, the tunica vaginalis was fibrotic, with a mild inflammatory infiltrate formed mostly of lymphocytes and plasma cells (Figs. 3, 4). A nodule was formed by a proliferation of 2 distinct populations of neoplastic cells, which created a distinct hiphasic pattern. Tbe most superficial popu]ation was composed of cuboidal monomorpbic cells arranged around papihary structures. Mitoses were scanty. These cells infiltrated the connective tissue as thin reticulated tubular strtictures. The nuclei were round or oval with Fig. 1. Case 1: Atypical epithelial proliferation with a papillary exophytic pattern ol growth (HE xloo). Fig. 2. Case 1: Atypical epithelial proliferation with scattered psammoma bodies (HE x25o). I

3 Mesothelioma of the Tunica Vaginaiis Testis Gorinietai, 213 Fig. 3. Cdsc 2: Superficial portion of neoplasia, showing a papillary pattern of growth with no aiypia in the proliferating tells (HExlO). Fig. 4. Case 2: The deeper infiltrating portion of the same tumor shows anaplastic pseudosartomatous features (HE X25). unilonii granular chromatin and small or inapparent nudeoli. The deeper portion of the nodule was composed of highly anaplastic cells wilh epitheiioid and spindle patterns. Mitoses were increased and the connective tissue around the neoplastic cells showed reactive fibrosis. Immunohistochemical examination showed ihat both neoplastic components strongly reacted with monoclonal antibodies to MNII6 cytokeratin (Dako, 1:50), EMA (Dako, 1:50), calretinin incubated overnight (Netimarkers, 1:2000), and trombomodulin (Neomarkers, 1:100). All the other antibodies tested (SMA, SlOO, CD34, HHF35, caponin) were not expressed. The histologic picture and the immunohistochemical profile were consistent with biphasic malignant mesothelioma of the tunica vaginalis. During the period ihe patient had been regularly exposed to asbestos when carrying out repairing and maintenance of tractors with asbestos friction brakes. Discussion Among all the serosal-covercd suffaces of the body, the tunica vaginalis testis is the least common site for mesothelioma [1]. Since its initial description by Barbera and Rubino in 1957 \\\\, about 80 case reports of mesothelioma of the tunica vaginalis have been described. It was reported that 34.2% of patients with tunical mesothelioma have a positive history of asbestos exposure, even if the real prevalence could be underestimated, as sufficient information was given in only 51.4% of the reported cases [1 ]. According to a recent review, asbestos exposure was traced in 23-41''X. of cases [12]. Some authors have suggested that direct effect of asbestt)s fibers on the tunica vaginalis or the deposition of asbestos libers within the serosa, causing chronic irritation of the tissue, could be the pathophysiologic mechanisms underlying the development of tunical mesothelioma [13. The 2 cases reported here are the oniy ones that have been recorded as mesothelioma of the tunica vaginalis in ARTMM from 1988 to 2003 (16 years), with both patients having been exposed to asbestos. The presence of asbestos in railway locomt>tives and in the vehicle repairing and maintenance business is well documented 16,10]. This fact highlights the need for collecting accurate information on occupational history, lifestyle habits, and areas of residence of patients witb mesothelioma, including these with a location in the tunica vaginalis. Acknowledgments We thank Valentina Cacciarini and Anna Maria Badiali (Occupational and Environmental Epidemiology Unit, CSPO, Elorence) who interviewed patients with mesotheiioma, and Stefano Silvestri (Occupational and Environmental Epidemiology Unit, CSPO, Florence) who attributed asbestos exposure.

4 214 Intemational Journal of Surgical Pathology Vol, 13 No. 2 April 2005 References 1. Plas E, Riedl CR, Pfluger H, Malignant mesothelioma of the tunica vaginalis testis. Review of the literature and assessment of prognostic parameters. Cancer 83: , Atlas ol tumor pathology. Tumors of the serosal membranes. Battifora H, McCoughey WTE (eds). AFIP, p. 23, Jones MA, Young RH, Sculiy RE. Malignant mesothelioma of the tunica vaginalis: A clinico[)atliologic analysis ol 11 cases and review of the literature, AmJSurg Pathol 19: , Atianoos RL, Gihhs AR, Primary malignant gouadal mesothcliomas and asbestos. Histopathology 37: , Ahmed M, Chari R, Muli GR. Malignant mesothelioma ot the tunica vaginalis testis diagnosed hy aspiration cytology, A case report with review of literature. Ini Urol Nephrol 28: , Gorini G, Silvestri S, Merler E, Chellini E, Cacciarini V, Scniori Costanlini A. Tuscan Mesotheiioma Registry ( ): Evaluation ol asbesios exposure. Med Lav 93 6): , Gurini G, Merler E, Chellini E, Crocetti E, Seniori Costantini A. Is the ratio of pleural mesothelioma mortality to pleural cancer mortality approximately unity for Italy? Considerations from the oldest regional mesothelioma register in Iialy, Br.J Cancer 86(12):]970-l971, Gorini G, Chellini E, Merler E, Cacciarini V, Silvestri S, Seniori Costantini A. Incidenza e mortalita in Toscana ]icr mesotclioma maligno pleurico nel periodo Epidemiol Prev 27(1):I3-17, , Marinaccio A, Nesti M, Magnani C, Ivaldi C, Dalmasso P, Mirabelli D, Terracini B, Todesco A, Gennaro V, Montanaro F, Lazzarettt) A, Bianchclli M, Celesia MV, Candela S, Romanelli A, Mangonc L, Merler E, Chellini E, Gorini G, Musti M, Cavone D, Analysis of survival of mesothelioma cases in the Italian register (ReNaM). Eur J Cancer 39: , Nesti M, Marinaccio A, Chellini E, Magnani C, Ivaldi C, Dalmasso P, Mirabelii D, Terracini B, Todesco A; Gennaro V, Montanaro F, Lazzaretto A, Bianchelli M, Celesia MV, Candela S, Romanelli A, Mangone L, Merler F, Silvestri S, Gorini G, Cacciarini V, Musti M, Cavone D. Malignant mesothelioma in Italy, Am.1 Ind Med 45(l):55-62, Barhera V, Rubino M. Papillary mesothelioma of the tunica vaginalis. Cancer 120: , WHO. Pathology and genetics of tumors of the urinary system and male genital organs. Fbele JN, Santer G, Epstein JI, Sesterhenn IA (eds). larc, Lyon, , Karunharan T. Malignant mesothelioma of the lunica vaginalis in an asbestos worker, J R Coll Surg Edinb 31: , 1986

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