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      Leydig cell tumor in grey zone: A case report

      case-report

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          Highlights

          • Leydig cell tumor is a testicular tumor with a low incidence accounting for 1–3% of testicular neoplasms.

          • Only about 10% of them show malignant behavior in the form of metastatic disease.

          • When diagnosed and treated early, long-term favorable outcomes are seen even with its potential metastatic behavior.

          Abstract

          Introduction

          Leydig cell tumor constitutes only about 1–3% of testicular neoplasms. There is apparently increased incidence in the last few years; one possible explanation for this phenomenon is the widespread use of ultrasound technology and the subsequent increased early detection of smaller lesions that have not been found in historical series.

          Case presentation

          We report a case of Leydig cell tumor of testis in a patient presenting with painless long standing slowly growing left scrotal mass who found to have intrapulmonary nodule and multiple enlarged retroperitoneal lymph nodes on staging work up. The mass was managed by radical orchiectomy. Pathological diagnosis was Leydig cell tumor.

          Discussion

          Orchiectomy is the accepted mode of treatment but follow-up every 3–6 months with physical examination, hormone assays, scrotal and abdominal ultrasonography, chest radiography, and CT scans is essential in such a case with a potential for malignant behavior.

          Conclusion

          Inguinal orchiectomy is the therapeutic decision of choice and long-term follow-up is necessary to exclude recurrence or metastasis. Cases which fall in the grey zone like ours need to be followed up carefully for metastasis instead of rushing into an early retroperitoneal lymph node dissection, with its potential risks and complications.

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          Most cited references15

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          The SCARE Statement: Consensus-based surgical case report guidelines.

          Case reports have been a long held tradition within the surgical literature. Reporting guidelines can improve transparency and reporting quality. However, recent consensus-based guidelines for case reports (CARE) are not surgically focused. Our objective was to develop surgical case report guidelines.
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            Leydig cell tumors of the testis. A clinicopathological analysis of 40 cases and review of the literature.

            The clinical and pathological features of 40 Leydig cell tumors of the testis were analyzed. The patients ranged from 2 to 90 (average 46.5) years of age. The most common initial manifestation was testicular swelling, which was sometimes associated with gynecomastia; 15% of the patients presented because of gynecomastia and were found to have palpable testicular tumors. All three children were brought to the physician because of isosexual pseudoprecocity. The tumors, one of which was asynchronously bilateral, ranged from 0.5 to 10.0 (average 3) cm in greatest diameter. They were usually well circumscribed, but in seven of them the margin with the adjacent testis was ill-defined. On microscopic examination the most common pattern was that of diffuse sheets of neoplastic cells, but insular, trabecular, pseudotubular, and ribbon-like patterns were also encountered. The neoplastic cells were most often large and polygonal with abundant eosinophilic, slightly granular cytoplasm; occasionally the cytoplasm was abundantly vacuolated. In eight tumors some of the cells were spindle-shaped, and in six some had scanty cytoplasm. Crystalloids of Reinke were identified in 35% of the tumors. Conspicuous nuclear atypicality was present in 12 tumors and the mitotic rate ranged from less than 1 to 32/10 high-power fields. Blood vessel invasion, lymphatic invasion, or both were identified in four tumors. Follow-up of 2 months to 22 years (average 4 years) was available for 30 patients. Five of them died as a result of spread of their tumor. A comparison of the clinically malignant tumors with those associated with survival for 2 or more years postoperatively revealed that the former occurred in older patients and were accompanied by symptoms of shorter duration and an absence of endocrine manifestations. The malignant tumors were larger, often had an infiltrative margin and had spread beyond the testis, frequently exhibited blood vessel or lymphatic invasion, and had a greater degree of cellular atypia and necrosis and a higher mitotic rate than the benign tumors.
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              Does retroperitoneal lymph node dissection have a curative role for patients with sex cord-stromal testicular tumors?

              Sex cord-stromal tumors account for < 5% of all adult testicular tumors, and 10% are malignant. Due to the limited reported experience, there is no agreement on the best management, especially in patients who have tumors with malignant pathologic features or who present with metastatic disease. The authors attempt to evaluate the role of retroperitoneal lymph node dissection (RPLND) in the management of patients with these malignant sex cord-stromal tumors. Reviewing the Indiana University testis cancer registry revealed 17 patients who underwent RPLND for sex cord-stromal tumors. Pathology was reviewed for features suggestive of malignancy. The data examined included clinical and pathologic stage, surgical procedure, additional therapy received, and outcome. Pathology included Leydig tumors in six patients, Sertoli tumors in four patients, sex cord-stromal tumors in five patients, a granulosa cell tumor in one patient, and a poorly differentiated non-germ cell tumor in one patient. Nine patients had histologic features suggestive of malignancy. Clinical stage at surgery was Stage I in nine patients and Stage IIA-IIIA in eight patients. Patients underwent modified or bilateral RPLND. Nine patients had pathologic Stage I tumors, and the remaining eight patients and had pathologic Stage IIB-IIIA tumors. Follow-up ranged from 8 months to 11 years. Of the eight patients with Stage II-III disease, six patients eventually died of metastatic disease despite additional radiotherapy and/or chemotherapy. Sex cord-stromal tumors have a potentially aggressive malignant behavior that is difficult to predict based on clinical and pathologic features. Although the therapeutic role of RPLND in patients with small-volume metastatic retroperitoneal tumors is unclear, RPLND remains an option to be performed immediately after orchiectomy, especially in patients who have tumors with malignant features and/or small-volume metastatic disease. Copyright 2003 American Cancer Society.
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                Author and article information

                Contributors
                Journal
                Int J Surg Case Rep
                Int J Surg Case Rep
                International Journal of Surgery Case Reports
                Elsevier
                2210-2612
                04 April 2017
                2017
                04 April 2017
                : 35
                : 12-16
                Affiliations
                [a ]Division of Urology, Department of Special Surgery, Jordan University Hospital, The University of Jordan, Jordan
                [b ]Department of Pathology and Microbiology and Forensic Medicine, Jordan University Hospital, The University of Jordan, Jordan
                [c ]Department of Radiology, Jordan University Hospital, The University of Jordan, Jordan
                [d ]General Practitioner, Jordan
                Author notes
                [* ]Corresponding author. Mhelan87@ 123456hotmail.com
                Article
                S2210-2612(17)30175-X
                10.1016/j.ijscr.2017.03.043
                5394201
                28419904
                14e3ee7e-6e22-4da5-a84f-f77b6da0525e
                © 2017 The Authors

                This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/).

                History
                : 14 January 2017
                : 31 March 2017
                : 31 March 2017
                Categories
                Case Report

                testicular neoplasms,leydig cell tumor,inguinal orchiectomy,rplnd,malignant behavior

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