POST-PUBERTAL TESTICULAR EPIDERMOID CYST: SURGICAL OPTIONS AND LITERATURE REVIEW Olivieri Valerio1, Fortunati Valentina2, Corongiu Emanuele3, Forte Flavio3, De Luca Francesco4, Ruggiero Gabriele1 and Griffa Daniele1 1 Division of Urology, Ivrea Hospital – ASL TO4, Ivrea (Turin), Italy 2 Division of Pathology, Hospital “Policlinico of Tor Vergata”, Rome, Italy 3 Division of Urology, Hospital “Madre Giuseppina Vannini”, Rome, Italy 4 Department of Gynecological, Obstetrics and Urological Sciences, Hospital, Policlinico Umberto I, Rome, Italy INTRODUCTION AND OBJECTIVES: Epidermoid cysts (TEC) are uncommon benign tumors accounting for around 1-2% of all testicular lesions. Incidence is higher in people between the ages of 10 and 40 years. Caucasian males are frequently involved and righ testis more than left one. Etiology is not completely understood. Clinically they present with a firm, painless palpable mass highly indistinguishable to all testicular neoplasms. Preoperative serum markers are always negative. Radiology and biopsy are important criteria for surgical planning. Scrotal sonography (US) confirm its typical “onion-ring” appearance. Magnetic resonance (MR) add more informations on vascularity and borders. Diagnosis depends on histology: frozen section may be useful in preventing unnecessary orchiectomy. Two forms of TEC have been described. Prepubertal ones are considered as a benign tumors since they’re not associated with germ cell tumor (GCT) and no cases of metastatic spread have been reported: unfavorable prognostic features are usually missing. Post-pubertal TEC or “complex” may act differently. Association with GCT has been rarely described totally representing an unfavorable prognostic features: cellular atypia, high mitotic activity, necrosis are rare but may be present. MATERIALS AND METHODS: We present the case of an adult male affected by TEC.and reviewed the literature. A 40 y.o. caucasian male presented for a mass in the left testis discovered on self-examination. No familiarity for testicular cancer was reported. Inguinal lymph nodes were negative. Serum markers (AFP, bHCG and LDH) were normal too. The testis presented with a painless firm lesion at the upper pole. US revealed a 15 mm heterogeneous well-circumscribed mass being chraracterized by an echogenic rim: MR ruled out intralesional vascularity. Testicular biopsy was offered. RESULTS: Frozen section was suggestive for mature TEC and a testing sparing surgery was offered. Macroscopically the lesion presented as indishinguable from any malignant neoplasm. On microscopic evaluation, the cyst was lined by a fibrous membrane (Fig. 1) filled with layers of cornifying squamous epithelium composing the wall. Coexisting teratomatous element were ruled out. Association with GCT and atypical features were also excluded. It was classed as post-pubertal mature TEC. Surgical enucleation was offered. CONCLUSIONS: TEC are rare benign tumors incidentally discovered during self examination or scrotal ultrasound frequently affecting young males. Sonography and testicular MR helps in characterizing the lesion. Diagnosis depends on histology. The biological behaviour of these neoplasms in the adult testis still remains unknown leading to different surgical approaches. Testis sparing surgery represent the gold standard. Radical orchiectomy is mandatory as unfavorable features coexist. BIBLIOGRAPHY: 1. Anheuser P, Kranz J, Stolle E, Höflmayer D, Büscheck F, Mühlstädt S, Lock G, Dieckmann KP. Testicular epidermoid cysts: a reevaluation. BMC Urol. 2019 Jun 11;19(1):52 2. Çakıroğlu B, Sönmez NC, Sinanoğlu O, Ateş L, Aksoy SH, Özcan F. Testicular epidermoid cyst. Afr J Paediatr Surg. 2015 Jan-Mar;12(1):89-90. 3. Cook FE Jr, Kimbrough JC. Epidermoid cysts of the testicle. J Urol. 1954 Aug;72(2):236-8. 4. Dieckmann KP, Loy V. Epidermoid cyst of the testis: A review of clinical and histogenetic considerations. Br J Urol. 1994;73:436–41. 5. Dockerty M, Priestly JY. Dermoid cysts of the testis. J Urol. 1942;48:392–7 6. Ewen SW. Epidermoid cyst of the testis. Scott Med J. 1969;14:57–8 7. Heidenreich A, Zumbe J, Vorreuther R, Klotz T, Vietsch H, Engelmann UH. Testikuläre Epidermiszyste: Orchiektomie oder Enukleationsresektion? Urologe A. 1996;35:1):1–5. 8. Huyghe E, Mazerolles C, Moran C, Khedis M, Khoury E, Nohra J, Soulie M, Plante P. Synchronous epidermoid cyst and mature teratoma of the testis: an unusual association. Urol Int. 2007;78:364–6. 9. Kenan B. Ashouri, Joshua M. Heiman, Emily F. Kelly. Testicular epidermoid cyst:: a rare case. Urol Ann. 2017 Jul-Sep; 9(3): 296–298. 10. Maizlin ZV, Belenky A, Baniel J, Gottlieb P, Sandbank J, Strauss S. Epidermoid cyst and teratoma of the testis: sonographic and histologic similarities. J Ultrasound Med. 2005;24:1403–9 11. Moch H, Cubilla AL, Humphrey PA, Reuter VE, Ulbright TM. The 2016 WHO classification of Tumours of the urinary system and male genital organs-part a: renal, penile, and testicular Tumours. Eur Urol. 2016;70:93–105. 12. Muoka OE, Dahar N. Testicular epidermoid cysts. BMJ Case Rep 2013. 2013:pii: Bcr2013009103. 13. Shah KH, Maxted WC, Chun B. Epidermoid cysts of the testis: A report of three cases and an analysis of 141 cases from the world literature. Cancer. 1981;47:577–82. 14. Ulbright TM, Srigley JR (2001) Dermoid cyst of the testis: a study of five postpubertal cases, including a pilomatrixoma-like variant, with evidence supporting its separate classification from mature testicular teratoma. Am J Surg Pathol 25: 788-793. 15. Umar SA, MacLennan GT. Epidermoid cyst of the testis. J Urol. 2008;180:335

Post-pubertal testicular epidermoid cyst: surgical options and literature review / Olivieri, Valerio; Fortunati, Valentina; Corongiu, Emanuele; Forte, Flavio; DE LUCA, Francesco; Ruggiero, Gabriele; Griffa, Daniele. - (2019). ((Intervento presentato al convegno II° Cogresso Nazionale NAU - Natura Ambiente Alimentazione Uomo tenutosi a Frascati (Roma).

Post-pubertal testicular epidermoid cyst: surgical options and literature review

OLIVIERI VALERIO
Primo
;
Fortunati Valentina
Secondo
;
Corongiu Emanuele;Forte Flavio;De Luca Francesco;
2019

Abstract

POST-PUBERTAL TESTICULAR EPIDERMOID CYST: SURGICAL OPTIONS AND LITERATURE REVIEW Olivieri Valerio1, Fortunati Valentina2, Corongiu Emanuele3, Forte Flavio3, De Luca Francesco4, Ruggiero Gabriele1 and Griffa Daniele1 1 Division of Urology, Ivrea Hospital – ASL TO4, Ivrea (Turin), Italy 2 Division of Pathology, Hospital “Policlinico of Tor Vergata”, Rome, Italy 3 Division of Urology, Hospital “Madre Giuseppina Vannini”, Rome, Italy 4 Department of Gynecological, Obstetrics and Urological Sciences, Hospital, Policlinico Umberto I, Rome, Italy INTRODUCTION AND OBJECTIVES: Epidermoid cysts (TEC) are uncommon benign tumors accounting for around 1-2% of all testicular lesions. Incidence is higher in people between the ages of 10 and 40 years. Caucasian males are frequently involved and righ testis more than left one. Etiology is not completely understood. Clinically they present with a firm, painless palpable mass highly indistinguishable to all testicular neoplasms. Preoperative serum markers are always negative. Radiology and biopsy are important criteria for surgical planning. Scrotal sonography (US) confirm its typical “onion-ring” appearance. Magnetic resonance (MR) add more informations on vascularity and borders. Diagnosis depends on histology: frozen section may be useful in preventing unnecessary orchiectomy. Two forms of TEC have been described. Prepubertal ones are considered as a benign tumors since they’re not associated with germ cell tumor (GCT) and no cases of metastatic spread have been reported: unfavorable prognostic features are usually missing. Post-pubertal TEC or “complex” may act differently. Association with GCT has been rarely described totally representing an unfavorable prognostic features: cellular atypia, high mitotic activity, necrosis are rare but may be present. MATERIALS AND METHODS: We present the case of an adult male affected by TEC.and reviewed the literature. A 40 y.o. caucasian male presented for a mass in the left testis discovered on self-examination. No familiarity for testicular cancer was reported. Inguinal lymph nodes were negative. Serum markers (AFP, bHCG and LDH) were normal too. The testis presented with a painless firm lesion at the upper pole. US revealed a 15 mm heterogeneous well-circumscribed mass being chraracterized by an echogenic rim: MR ruled out intralesional vascularity. Testicular biopsy was offered. RESULTS: Frozen section was suggestive for mature TEC and a testing sparing surgery was offered. Macroscopically the lesion presented as indishinguable from any malignant neoplasm. On microscopic evaluation, the cyst was lined by a fibrous membrane (Fig. 1) filled with layers of cornifying squamous epithelium composing the wall. Coexisting teratomatous element were ruled out. Association with GCT and atypical features were also excluded. It was classed as post-pubertal mature TEC. Surgical enucleation was offered. CONCLUSIONS: TEC are rare benign tumors incidentally discovered during self examination or scrotal ultrasound frequently affecting young males. Sonography and testicular MR helps in characterizing the lesion. Diagnosis depends on histology. The biological behaviour of these neoplasms in the adult testis still remains unknown leading to different surgical approaches. Testis sparing surgery represent the gold standard. Radical orchiectomy is mandatory as unfavorable features coexist. BIBLIOGRAPHY: 1. Anheuser P, Kranz J, Stolle E, Höflmayer D, Büscheck F, Mühlstädt S, Lock G, Dieckmann KP. Testicular epidermoid cysts: a reevaluation. BMC Urol. 2019 Jun 11;19(1):52 2. Çakıroğlu B, Sönmez NC, Sinanoğlu O, Ateş L, Aksoy SH, Özcan F. Testicular epidermoid cyst. Afr J Paediatr Surg. 2015 Jan-Mar;12(1):89-90. 3. Cook FE Jr, Kimbrough JC. Epidermoid cysts of the testicle. J Urol. 1954 Aug;72(2):236-8. 4. Dieckmann KP, Loy V. Epidermoid cyst of the testis: A review of clinical and histogenetic considerations. Br J Urol. 1994;73:436–41. 5. Dockerty M, Priestly JY. Dermoid cysts of the testis. J Urol. 1942;48:392–7 6. Ewen SW. Epidermoid cyst of the testis. Scott Med J. 1969;14:57–8 7. Heidenreich A, Zumbe J, Vorreuther R, Klotz T, Vietsch H, Engelmann UH. Testikuläre Epidermiszyste: Orchiektomie oder Enukleationsresektion? Urologe A. 1996;35:1):1–5. 8. Huyghe E, Mazerolles C, Moran C, Khedis M, Khoury E, Nohra J, Soulie M, Plante P. Synchronous epidermoid cyst and mature teratoma of the testis: an unusual association. Urol Int. 2007;78:364–6. 9. Kenan B. Ashouri, Joshua M. Heiman, Emily F. Kelly. Testicular epidermoid cyst:: a rare case. Urol Ann. 2017 Jul-Sep; 9(3): 296–298. 10. Maizlin ZV, Belenky A, Baniel J, Gottlieb P, Sandbank J, Strauss S. Epidermoid cyst and teratoma of the testis: sonographic and histologic similarities. J Ultrasound Med. 2005;24:1403–9 11. Moch H, Cubilla AL, Humphrey PA, Reuter VE, Ulbright TM. The 2016 WHO classification of Tumours of the urinary system and male genital organs-part a: renal, penile, and testicular Tumours. Eur Urol. 2016;70:93–105. 12. Muoka OE, Dahar N. Testicular epidermoid cysts. BMJ Case Rep 2013. 2013:pii: Bcr2013009103. 13. Shah KH, Maxted WC, Chun B. Epidermoid cysts of the testis: A report of three cases and an analysis of 141 cases from the world literature. Cancer. 1981;47:577–82. 14. Ulbright TM, Srigley JR (2001) Dermoid cyst of the testis: a study of five postpubertal cases, including a pilomatrixoma-like variant, with evidence supporting its separate classification from mature testicular teratoma. Am J Surg Pathol 25: 788-793. 15. Umar SA, MacLennan GT. Epidermoid cyst of the testis. J Urol. 2008;180:335
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