Leydig cell tumors of the testis: a case report

BMC Research Notes

Ancuta Augustina Gheorghisan-Galateanu¹ 

[1] Department of Cellular and Molecular Medicine, Carol Davila University of Medicine and Pharmacy, 8 Eroii Sanitari Blvd, Bucharest 050474, Romania

关键词: Subdiaphragmatic micro lymphadenopathy;    Estradiol;    Gynecomastia;    Overweight;    Leydig cell tumor;   

Others  :  1129371
DOI  :  10.1186/1756-0500-7-656

 received in 2014-06-13, accepted in 2014-09-16,  发布年份 2014

【 摘 要 】

Background

Leydig cell tumors are the most common non-germ cell gonadal tumors with apparent increased incidence in the last few years. They are usually benign tumors. We report a case of Leydig cell tumor of testis in a patient presenting atypical features.

Case presentation

A 29-year-old Caucasian man, born with right cryptorchidism, corrected without medical treatment before the age of two years, was diagnosed with Leydig cell tumor. Two years after diagnosis was identified moderately elevated estradiol serum level, in the context of a significant overweight, hormonal changes which had maintained after unilateral orchiectomy and after the patient's return to normal weight. Four years after unilateral orchiectomy, elevated value of estradiol persisted and subdiaphragmatic micro lymphadenopathy was observed.

Conclusions

Despite the favorable evolution of the patient four years after unilateral orchiectomy, long-term follow-up is necessary to exclude recurrence or metastasis to the testis. The endocrine profile and imaging investigations need to be repeated periodically. The changes in the hormonal assay and any new aspects on computed tomography scan can be used as a marker of tumor recurrence and require careful screening and the correct therapeutic decisions.

【 授权许可】

   
2014 Gheorghisan-Galateanu; licensee BioMed Central Ltd.

【 图 表 】

Figure 1.

Figure 2.

【 参考文献 】

• [1]Dilworth JP, Farrow GM, Oesterling JE: Non-germ cell tumors of testis. Urology 1991, 37:399-417.
• [2]Emerson RE, Ulbright TM: Morphological approach to tumours of the testis and paratestis. J Clin Pathol 2007, 60:866-880.
• [3]Cheville JC: Classification and pathology of testicular germ cell and sex cord-stromal tumors. Urol Clin North Am 1999, 26:595-609.
• [4]Leonhartsberger N, Ramoner R, Aigner F, Stoehr B, Pichler R, Zangerl F, Fritzer A, Steiner H: Increased incidence of Leydig cell tumours of the testis in the era of improved imaging techniques. BJU Int 2011, 108:1603-1607.
• [5]Giacaglia LR, Kohek MB da F, Carvalho FM, Fragoso MC, Mendonca B, Latronico AC: No evidence of somatic activating mutations on gonadotropin receptor genes in sex cord stromal tumors. Fertil Steril 2000, 74:992-995.
• [6]Libe R, Fratticci A, Lahlou N, Jornayvaz FR, Tissier F, Louiset E, Guibourdenche J, Vieillefond A, Zerbib M, Bertherat J: A rare cause of hypertestosteronemia in a 68-year-old patient: a Leydig cell tumor due to a somatic GNAS (guanine nucleotide-binding protein, alpha-stimulating activity polypeptide 1)-activating mutation. J Androl 2012, 33:578-584.
• [7]Carvajal-Carmona LG, Alam NA, Pollard PJ, Jones AM, Barclay E, Wortham N, Pignatelli M, Freeman A, Pomplun S, Ellis I, Poulsom R, El-Bahrawy MA, Berney DM, Tomlinson IP: Adult leydig cell tumors of the testis caused by germline fumarate hydratase mutations. J Clin Endocrinol Metab 2006, 91:3071-3075.
• [8]Lejeune H, Habert R, Saez JM: Origin, proliferation and differentiation of Leydig cells. J Mol Endocrinol 1998, 20:1-25.
• [9]Huang Y, Song J, Xu M, Zan Q: Primary Leydig cell tumour of epididymis: a rare case report with review of literature. Andrologia 2013, 45:430-433.
• [10]Butruille C, Marcelli F, Ghoneim T, Lemaitre L, Puech P, Leroy X, Rigot JM: Management of testicular lesions in a population of infertile patients. Prog Urol 2012, 22:45-52.
• [11]Markou A, Vale J, Vadgama B, Walker M, Franks S: Testicular leydig cell tumor presenting as primary infertility. Hormones (Athens) 2002, 1:251-254.
• [12]Negri L, Benaglia R, Fiamengo B, Pizzocaro A, Albani E, Levi Setti PE: Cancer risk in male factor-infertility. Placenta 2008, 29(Suppl B):178-183.
• [13]Amatya NZM, Sapkota PK: Leydig cell tumor: a case report. Nepal J Radiol 2012, 2:39-41.
• [14]Handa U, Sood T, Punia RS: Testicular Leydig cell tumor diagnosed on fine needle aspiration. Diagn Cytopathol 2010, 38:682-684.
• [15]Henderson CG, Ahmed AA, Sesterhenn I, Belman AB, Rushton HG: Enucleation for prepubertal leydig cell tumor. J Urol 2006, 176:703-705.
• [16]Loeser A, Vergho DC, Katzenberger T, Brix D, Kocot A, Spahn M, Gerharz EW, Riedmiller H: Testis-sparing surgery versus radical orchiectomy in patients with Leydig cell tumors. Urology 2009, 74:370-372.
• [17]Passman C, Urban D, Klemm K, Lockhart M, Kenney P, Kolettis P: Testicular lesions other than germ cell tumours: feasibility of testis-sparing surgery. BJU Int 2009, 103:488-491.
• [18]Guana R, Mussa A, Lala R, Tessaris D, Tessiatore PM, Canavese F: Surgical enucleation of testicular leydigioma in a young child: case report and literature review. J Pediatr Endocrinol Metab 2011, 24:839-842.
• [19]Garcia M, Dargallo T, Palacios M, Carames J, Gomez J, Sacristan F, Vela D: Leydig cell tumour: enucleation as a therapeutic choice in a case with atypical symptoms. Arch Esp Urol 2012, 65:897-899.
• [20]Bertram KA, Bratloff B, Hodges GF, Davidson H: Treatment of malignant Leydig cell tumor. Cancer 1991, 68:2324-2329.
• [21]Iczkowski KA, Bostwick DG, Roche PC, Cheville JC: Inhibin A is a sensitive and specific marker for testicular sex cord-stromal tumors. Mod Pathol 1998, 11:774-779.
• [22]Busam KJ, Iversen K, Coplan KA, Old LJ, Stockert E, Chen YT, McGregor D, Jungbluth A: Immunoreactivity for A103, an antibody to melan-A (Mart-1), in adrenocortical and other steroid tumors. Am J Surg Pathol 1998, 22:57-63.
• [23]Bremmer F, Schweyer S, Martin-Ortega M, Hemmerlein B, Strauss A, Radzun HJ, Behnes CL: Switch of cadherin expression as a diagnostic tool for Leydig cell tumours. APMIS 2013, 121:976-981.
• [24]McCluggage WG, Shanks JH, Whiteside C, Maxwell P, Banerjee SS, Biggart JD: Immunohistochemical study of testicular sex cord-stromal tumors, including staining with anti-inhibin antibody. Am J Surg Pathol 1998, 22:615-619.
• [25]Hekimgil M, Altay B, Yakut BD, Soydan S, Ozyurt C, Killi R: Leydig cell tumor of the testis: comparison of histopathological and immunohistochemical features of three azoospermic cases and one malignant case. Pathol Int 2001, 51:792-796.
• [26]Iczkowski KA, Butler SL: New immunohistochemical markers in testicular tumors. Anal Quant Cytol Histol 2006, 28:181-187.
• [27]Dong L, Wang H, Su Z, Niu S, Wang R, Wu L, Chen G: Steroidogenic acute regulatory protein is a useful marker for Leydig cells and sex-cord stromal tumors. Appl Immunohistochem Mol Morphol 2011, 19:226-232.
• [28]Sangoi AR, McKenney JK, Brooks JD, Higgins JP: Evaluation of SF-1 expression in testicular germ cell tumors: a tissue microarray study of 127 cases. Appl Immunohistochem Mol Morphol 2013, 21:318-321.
• [29]Farkas LM, Szekely JG, Pusztai C, Baki M: High frequency of metastatic leydig cell testicular tumors. Oncology 2000, 59:118-121.
• [30]Trabert B, Zugna D, Richiardi L, McGlynn KA, Akre O: Congenital malformations and testicular germ cell tumors. Int J Cancer 2013, 133:1900-1904.
• [31]Palazzo JP, Petersen RO, Young RH, Scully RE: Deoxyribonucleic acid flow cytometry of testicular Leydig cell tumors. J Urol 1994, 152:415-417.
• [32]Carreau S, Bois C, Zanatta L, Silva FR, Bouraima-Lelong H, Delalande C: Estrogen signaling in testicular cells. Life Sci 2011, 89:584-587.
• [33]Masumori N, Kumamoto Y, Itoh N, Tsukamoto T, Miyao N, Koroku M, Hirose T, Takahashi S: Leydig cell tumor: a case report with reference to its endocrinological features. Eur Urol 1993, 24:302-304.
• [34]Rieu M, Kuhn JM, Mandard JC, Bottet P, Lemonnier D, Bekka S, Mahoudeau J: Luteinizing hormone regulation by sex steroids in men with germinal and Leydig cell tumours. Clin Endocrinol (Oxf) 1993, 38:487-493.
• [35]Tavolini IM, Rossato M, Milani C, Dal Moro F: Estradiol plasma levels elevation during follow-up for testicular Leydig-cell tumor is not an unfailing sign of recurrence. Arch Ital Urol Androl 2007, 79:141-142.
• [36]Maeda T, Itoh N, Kobayashi K, Takahashi A, Masumori N, Tsukamato T: Elevated serum estradiol suggesting recurrence of Leydig cell tumor nine years after radical orchidectomy. Int J Urol 2002, 9:659-661.