Sarcomas of the testis are extremely rare tumors, their incidence being difficult to assess accurately. The authors report a case of a 21-year-old male, presented with painless scrotal swelling that increased in size insidiously within three months. The examination revealed a 10 cm right testicular swelling—hard, not tender and oval in shape. Testicular ultrasound revealed right heterogenous testicular mass. Tumor markers (Human chorionic gonadotrophin (HCG) and alpha fetoprotein) were normal. CT chest and abdomen were normal. Radical orchidectomy was done through an inguinal approach. Histopathology showed testicular fibrosarcoma with spindle cells. After one month, the patient noticed reappearance of a swelling in the right hemiscrotum. Right inguinal exploration with massive resection of the recurrent scrotal mass was done. Histopathology revealed inflammatory process dominated by stitch granuloma. In conclusion, the recurrence rate of testicular sarcomas is high following radical orchidectomy, but still there was a place of non-malignant massthat looks like recurrence; it might occur like nonspecific inflammatory mass and stich granuloma

AbuIdris, Dafalla

Ahmed, Mohammed El Imam Mohammed

Awadalla, Mohamed Daffalla

Mokhtar, Nadia M.

Osman, Yassin Mohammed

Taha, Sami Mahjoub

Sudan Journal of Medical Sciences

English

Sarcomas of the testis are extremely rare tumors, their incidence being difficult to assess accurately. The authors report a case of a 21-year-old male, presented with painless scrotal swelling that increased in size insidiously within three months. The examination revealed a 10 cm right testicular swelling—hard, not tender and oval in shape. Testicular ultrasound revealed right heterogenous testicular mass. Tumor markers (Human chorionic gonadotrophin (HCG) and alpha fetoprotein) were normal. CT chest and abdomen were normal. Radical orchidectomy was done through an inguinal approach. Histopathology showed testicular fibrosarcoma with spindle cells. After one month, the patient noticed reappearance of a swelling in the right hemiscrotum. Right inguinal exploration with massive resection of the recurrent scrotal mass was done. Histopathology revealed inflammatory process dominated by stitch granuloma. In conclusion, the recurrence rate of testicular sarcomas is high following radical orchidectomy, but still there was a place of non-malignant mass that looks like recurrence; it might occur like nonspecific inflammatory mass and stich granuloma.Keywords: testicular tumor, testicular fibrosarcoma, spindle cells, recurrent testicular sarcom

AJOL - African Journals Online

Sudan Journal of Medical SciencesVolume 13, Issue no. 2, DOI 10.18502/sjms.v13i2.2638Production and Hosting by Knowledge ECase ReportPrimary Fibrosarcoma of the Testicle withPuzzling Post-surgery Presentation:Mimicking RecurrenceSami Mahjoub Taha1, Yassin Mohammed Osman2, Dafalla AbuIdris3,Mohammed El Imam Mohammed Ahmed1, Mohamed Daffalla Awadalla4,and Nadia M. Mokhtar51Department of Urology, Faculty of Medicine, University of Gezira, Sudan2Department of Urology, Gezira Hospital for Renal Disease and Surgery, Sudan3Department of Oncology, National Cancer Institute, University of Gezira, Sudan4Faculty of Medicine, University of Gezira, Sudan5Department of Pathology, National Cancer Institute, Cairo University, EgyptAbstractSarcomas of the testis are extremely rare tumors, their incidence being difficultto assess accurately. The authors report a case of a 21-year-old male, presentedwith painless scrotal swelling that increased in size insidiously within three months.The examination revealed a 10 cm right testicular swelling—hard, not tender andoval in shape. Testicular ultrasound revealed right heterogenous testicular mass.Tumor markers (Human chorionic gonadotrophin (HCG) and alpha fetoprotein) werenormal. CT chest and abdomenwere normal. Radical orchidectomywas done throughan inguinal approach. Histopathology showed testicular fibrosarcoma with spindlecells. After one month, the patient noticed reappearance of a swelling in the righthemiscrotum. Right inguinal exploration with massive resection of the recurrentscrotal mass was done. Histopathology revealed inflammatory process dominatedby stitch granuloma. In conclusion, the recurrence rate of testicular sarcomas is highfollowing radical orchidectomy, but still there was a place of non-malignant massthat looks like recurrence; it might occur like nonspecific inflammatory mass and stichgranuloma.Keywords: testicular tumor, testicular fibrosarcoma, spindle cells, recurrent testicularsarcoma1. IntroductionTesticular cancer represents about 1.5% of male neoplasms and 5% genitourinarytumors. Most testicular tumors are diagnosed in 20th and 30th years of life. The his-tological type varies, although there is a clear predominance (90–95%) of germ cellHow to cite this article: Sami Mahjoub Taha, Yassin Mohammed Osman, Dafalla AbuIdris, Mohammed El Imam Mohammed Ahmed, MohamedDaffalla Awadalla, and Nadia M. Mokhtar (2018)“Primary Fibrosarcoma of the Testicle with Puzzling Post-surgery Presentation: MimickingRecurrence,” Sudan Journal of Medical Sciences, vol. 13, issue no. 2, 78–85. DOI 10.18502/sjms.v13i2.2638Page 78Corresponding Author: SamiMahjoub Taha; email:samimahj@gmail.comReceived 26 March 2018Accepted 15 June 2018Published 28 June 2018Production and Hosting byKnowledge ESami Mahjoub Taha etal. This article is distributedunder the terms of theCreative CommonsAttribution License, whichpermits unrestricted use andredistribution provided thatthe original author andsource are credited.Editor-in-Chief:Prof. Mohammad A. M.IbnoufSudan Journal of Medical Sciences Sami Mahjoub Taha et altumors [1]. Testicular sarcomas are extremely rare tumors, their incidence being diffi-cult to assess accurately. Although uncommon, these tumors have been recorded asthemain urogenital site of sarcomas in the elderly, whereas primary testicular sarcomais rare neoplasm, in which the diagnosis is made after exclusion of the more commontype of sarcomas like paratesticular sarcomas [2, 3]. Furthermore, the rarity of primaryfibrosarcoma in the testis further justifies the following report.2. Case ReportA 21-year-old male, presented to the urology department with painless right testicularswelling that increased in size insidiously within three months. No fever and no urinarysymptoms were reported. Left scrotum was free. On examination, about 10 cm righttesticular swelling was found—hard, not tender and oval in shape. Scrotal skin andanother testis were normal. Testicular ultrasound showed right heterogenous testic-ular mass. Tumor markers like HCG and alpha fetoprotein were normal. CT chest andabdomen were normal. Radical orchidectomy was done through inguinal approach,and the spermatic cord was chopped at the internal inguinal ring.Histopathology revealed fibrosarcoma of the testis, themicroscopic appearancewasmalignant hypercellular sacromatous tumor composed of long fascicle of fusiformspindle-shaped cells arranged in herringbone pattern. Tumors have spindle-shapednuclei and the tumor cells are separated by interwoven collagen fibers arranged inparallel fashion. There was mild to moderate anaplasia with scattered mitotic activity,Figures 1–6, evident by immunohistochemistry (SMA, S100, Desmin were negative andKi67was positive in 10%denoting low proliferation index). After onemonth, he noticedswelling in the same side of previous surgery, which was painless and increased insize. Testicular ultrasound showed right testicular tumor and right inguinal lymph nodeenlargement.Right inguinal exploration with massive resection of the recurrent scrotal mass wasdone. Histopathology revealed inflammatory process dominated by stitch granuloma.3. DiscussionSarcomas of the testis are extremely rare. When you review the literature of pub-lished series of sarcomas they were distributed as follows: leiomyosarcoma (32%),rhabdomyosarcoma (RMS, 24%) and liposarcoma (20%) are the commonest subtypes[4, 5].DOI 10.18502/sjms.v13i2.2638 Page 79Sudan Journal of Medical Sciences Sami Mahjoub Taha et al                 (A) (B) Figure 1: Malignant hypercellular sacromatous tumor formedwith long fascicle of fusiform spindle-shapedcells arranged in herringbone pattern—spindle-shaped nuclei with scanty cytoplasm and indistinct borders(orange arrows)—Hematoxylin-eosin stain; magnification x 40 (A), magnification x 100 (B). The copyrightof the image is kept for the authors. Figure 2: Collagen fibers arranged in parallel fashion : Hematoxylin-eosin stain; magnification x 10 (left)and x 40 (right).Figure 3: Ki67 immunochemistry is positive—in about 10% of tumor cells (magnification x 40).DOI 10.18502/sjms.v13i2.2638 Page 80Sudan Journal of Medical Sciences Sami Mahjoub Taha et alFigure 4: S100 is negative—immunochemistry (magnification x 40).Figure 5: Desmin is negative—immunochemistry (magnification x 40).Gowing and Morgan studied all mesenchymal tumors arising in testicular tissues,and their relative rarity is apparent; the study consists of 22 sarcomas enrolled in a sur-vey of around 1000 patients with testicular neoplasm. The majority of the sarcomas inthis study were mesenchymal, that is, fibrosarcoma, liposarcoma, myosarcoma, rhab-domyosarcoma and leiomyosarcoma, or combinations [6]. The sarcomas may presentwith a unilateral, painless scrotal swelling with short duration as seen in this case.The commonest way of metastasis is via lymphatics to internal iliac and para-aorticlymph nodes. Distant metastasis to liver, lungs and bone by hematogenous spreadaccounts for 20% of patients in initial presentation [14]. Management of testicularfibrosarcoma has shifted with multispecialty approaches that improves the survivalchances up to 80% by incorporating chemotherapeutic medications, radiotherapy orsurgical intervention [14, 15].DOI 10.18502/sjms.v13i2.2638 Page 81Sudan Journal of Medical Sciences Sami Mahjoub Taha et alIn the current reported case, the radiological staging was N0M0, and accordingly theoncologist preferred follow-up after radical orchidectomy. Inguinal approach orchidec-tomy with ligation of spermatic cord at the internal ring is the standard surgical inter-vention as seen in the case. The site of tumor origin (testicular-paratesticular), lymphnode involvement, distant metastasis, presence or absence of residual disease aftersurgical resection will help in staging of testicular and the plan of management offibrosarcoma of the testis.The reported local relapse rate in the scrotum and groin after orchidectomy is 25–37% [8, 10, 11]. Adjuvant locoregional (ipsilateral pelvic and groin nodes and scrotum)radiation and/or surgery apparently reduces rate of recurrence [7, 9, 11, 12]. Fagun-des et al. [10] reported no local recurrence in patients receiving adjuvant radiation,compared with 37% local recurrence in those managed with the removal of the testisalone.Catton et al. [13] mentioned 14% local recurrence in patients who underwent widelocal excision and/or radiotherapy after original excision of the testicular or parates-ticular sarcomas. They also suggested that orchidectomy alone is inadequate for tes-ticular sarcomas and recommended adjuvant wide hemiscrotum excision, which mayinclude inguinal lymph nodes for those managed with orchidectomy or local excision.They found that therewasmicroscopic disease present in 27%, followingwide excisionin patients with apparently completely excised tumors.In the current case, there was an appearance of swelling in the right testicle withinone month following radical orchidectomy, which made us to think of local recurrence,and therefore the decision of re-exploration was made with wide local excision of theswelling and the hemiscrotum. The patient was followed-up for six months and therewas no local recurrence or distant metastasis.The role of retroperitoneal lymph node dissection (RPLND) in testicular sarcomasremains controversial [5, 10, 16, 17]. Catton et al. [10] recommended RPLND in theirearlier series of 21 patients, but in their subsequent report they noted that the patientswith a high risk of nodal metastasis are also at highest risk of concurrent or subse-quent systemic disease; accordingly, they felt that those patients would benefit fromsystemic rather than regional treatment [13]. Others suggest that RPLND should belimited to patients with suspicion of LN metastasis in the image [19]. In this case, weadvocate that the last suggestions is to do RPLND when there is radiological suspicionof LN residual after adjuvant chemotherapy.DOI 10.18502/sjms.v13i2.2638 Page 82Sudan Journal of Medical Sciences Sami Mahjoub Taha et alAdjuvant chemotherapy in testicular sarcomas is not fully established. In pediatrics,with testicular sarcomas—especially rhabdomyosarcoma (RMS)—adjuvant chemother-apy had good outcome on the overall survival [20, 21]. Its use in adult testicular sar-comas has not been yet specifically addressed in a controlled study [22].The use of post-intervention chemotherapy for adult high-grade testicular fibrosar-coma is controversial. A meta-analysis of randomized trials of testicular sarcomas atdifferent centers showed that doxorubicin-based adjuvant chemotherapy significantlyimproved the time to local and distant failure [23].In conclusion, testicular fibrosarcoma is extremely rare testicular tumor. Althoughrecurrence rate is high following radical orchidectomy, but there was still a place ofnon-malignant mass that looks like recurrence that might occur like a nonspecificinflammatory mass and stich granuloma.References[1] Albers, P., Albrecht, W., Algaba, F., et al. (2014). Guidelines on Testicular Cancer.European Association of Urology Guidelines on Testicular Cancer: Update.[2] Purdue, M. P., Devesa, S. S., Sigurdson, A. J., et al. (2005). International patternsand trends in testis cancer incidence. International Journal of Cancer, vol. 115, pp.822-827.[3] Lioe, T. F. and Biggart, J. D. (1993). Tumors of the spermatic cord and paratesticulartissue: A clinicopathological study. British Journal of Urology, vol. 71, pp. 600-606.[4] Soosay, G. N., Parkinson, M. C., Paradinas, J., et al. (1996). Paratesticular sarcomasrevisited: A review of cases in the British Testicular Tumour Panel and Registry.British Journal of Urology, vol. 77, pp. 143-146.[5] Fagundes, M. A., Zietman, A. L., Althausen, A. F., et al. (1996). The management ofspermatic cord sarcoma. Cancer, vol. 77, pp. 1873-1876.[6] Dowling, K. J. and Lieb, H. E. (September 1, 1985). Fibrosarcoma of epididymis.Urology, vol. 26, no. 3, pp. 307-308.[7] Russo, P., Brady, M. S., Conlon, K., et al. (1992). Adult urological sarcoma. Journal ofUrology, vol. 147, pp. 1032-1036.[8] Blitzer, P. H., Dosoretz, D. E., Proppe, K. H., et al. (1981). Treatment of malignanttumors of the spermatic cord: A study of 10 cases and a review of literature. Journalof Urology, vol. 126, pp. 611-614.[9] Rao, C. R., Srinivasulu, M., Naresh, K. N., et al. (1994). Adult paratesticular sarcomas:A report of eight cases. Journal of Surgical Oncology, vol. 56, pp. 89-93.DOI 10.18502/sjms.v13i2.2638 Page 83Sudan Journal of Medical Sciences Sami Mahjoub Taha et al[10] Catton, C. N., Cummings, V., Fornasier, B., et al. (1991). Adult paratesticular sarcomas:A review of 21 cases. Journal of Urology, vol. 146, pp. 342-345.[11] Fagundes, M. A., Zietman, A. L., Althausen, A. F., et al. (1996). The management ofspermatic cord sarcoma. Cancer, vol. 77, pp. 1873-1876.[12] Rabbani, F., Wright, J., and McLoughlin, M. (1997). Sarcomas of the spermatic cord:Significance of wide local excision. The Canadian Journal of Urology, vol. 4, pp. 366-368.[13] Catton, C. N., Jewett, M., O’Sullivan, I., et al. (1999). Paratesticular sarcoma: Failurepattern after definitive local therapy. Journal of Urology, vol. 161, pp. 1844-1847.[14] Resim, S., Okur, N., Bakar?s, S., et al. (2009). Paratesticular embryonal rhab-domyosarcoma: Report of a case. Iranian Journal of Pediatrics, vol. 19, no. 4, pp.430-434.[15] Kumar, R., Bharti, S., Khosla, D., et al. ( Jan 1, 2012). Long-term survival inparatesticular rhabdomyosarcoma. Clinical Cancer Investigation Journal, vol. 1, no.1, p. 31.[16] Merimsky, O., Terrier, P., Bonvalot, S., et al. (1999). Spermatic cord sarcoma in adults.Acta Oncologica, vol. 38, pp. 635-638.[17] Hermans, B. P., Foster, R. S., Bihrle, R., et al. (1998). Is retroperitoneal lymphnode dissection necessary for adult paratesticular rhabdomyosarcoma? Journal ofUrology, vol. 160, pp. 2074-2077.[18] Donohue, J. P. (1991). Editorial comment. Journal of Urology, vol. 146, p. 345.[19] Goldfarb, B., Khoury, A. E., Greenberg, M. L., et al. (1994). The role of retroperitoneallymphadenectomy in localized paratesticular rhabdomyosarcoma. Journal of Urol-ogy, vol. 152, pp. 785-787.[20] Raney, R. B., Jr., Tefft, M., Lawrence, W., Jr., et al. (1987). Paratesticular sarcomain childhood and adolescence: A report from the Intergroup RhabdomyosarcomaStudies I and II, 1973-83. Cancer, vol. 60, pp. 2337-2343.[21] De Vries, J. D. (1997). Paratesticular rhabdomyosarcoma. World Journal of Urology,vol. 13, pp. 219-225.[22] Anonymous. (1997). Adjuvant chemotherapy for localized resectable soft-tissuesarcoma of adults: Meta-analysis of individual data. Sarcoma Meta-analysisCollaboration. Lancet, vol. 350, pp. 1647-1654.[23] Khoubehi, B., Mishra, V., Ali, M., et al. (Nov 1, 2002). Adult paratesticular tumours.BJU International, vol. 90, no. 7, pp. 707-715.[24] Fernando, J., Azcarretazabal, T., Torio, B. et al. (1999). Primary pure intratesticularfibrosarcoma. Pathology International, vol. 49, pp. 185-189.DOI 10.18502/sjms.v13i2.2638 Page 84Sudan Journal of Medical Sciences Sami Mahjoub Taha et al[25] Zukerberg, L. R. and Young, R. H. (1990). Primary testicular fibrosarcoma: A reportof two cases. Human Pathology, vol. 21, pp. 932-935.DOI 10.18502/sjms.v13i2.2638 Page 85

Primary fibrosarcoma of the testicle with puzzling post-surgery presentation: mimicking recurrence

Sarcomas of the testis are extremely rare tumors, their incidence being difficult to assess accurately. The authors report a case of a 21-year-old male, presented with painless scrotal swelling that increased in size insidiously within three months. The examination revealed a 10 cm right testicular swelling—hard, not tender and oval in shape. Testicular ultrasound revealed right heterogenous testicular mass. Tumor markers (Human chorionic gonadotrophin (HCG) and alpha fetoprotein) were normal. CT chest and abdomen were normal. Radical orchidectomy was done through an inguinal approach. Histopathology showed testicular fibrosarcoma with spindle cells. After one month, the patient noticed reappearance of a swelling in the right hemiscrotum. Right inguinal exploration with massive resection of the recurrent scrotal mass was done. Histopathology revealed inflammatory process dominated by stitch granuloma. In conclusion, the recurrence rate of testicular sarcomas is high following radical orchidectomy, but still there was a place of non-malignant mass that looks like recurrence; it might occur like nonspecific inflammatory mass and stich granuloma

Sami Mahjoub Taha

Yassin Mohammed Osman

Dafalla AbuIdris

Mohammed El Imam Mohammed Ahmed

Mohamed Daffalla Awadalla

Nadia M. Mokhtar

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Primary Fibrosarcoma of the Testicle with Puzzling Post-surgery Presentation: Mimicking Recurrence

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Primary Fibrosarcoma of the Testicle with Puzzling Post-surgery Presentation: Mimicking Recurrence

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