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Chin Med J 2010;123(19):2748-2749 2748 Images for diagnosis Basal cell carcinoma of the scrotum: report of a case and review of the literature WANG Jian-wei, MAN Li-bo, HE Feng, HUANG Guang-lin, LI Gui-zhong and WANG Hai-dong Keywords: scrotum; basal cell carcinoma; risk factors B asal cell carcinoma (BCC) is the most common cancer of the skin. However, BCC occurring on non-sun-exposed sites, especially the genital regions such as scrotum and labia, are very rare. An estimated annual incidence of basal cell carcinoma of the scrotum is 1 per 1 000 000 population.1 Here, we report a man with scrotal BCC with the lesion for 51 years, the longest one in the documents. He was successfully treated by circumferential excision. CASE REPORT A 74-year-old Chinese man was hospitalized with the symptom of itching on the skin of the scrotum for 51 years. Initially, an erythema with itching and desquamating was found on the middle of the bottom of the scrotum in 1953. Then the patient left China and went for working as a photographer for 2 years in Africa without consulting his doctor. However, the erythema was broken, an ulcer 2 cm in diameter was surrounded by a well defined pearly border in the third year after onset. The necrosis tissues with lots of white and yellow mucous fluids bring the ulceration odd smell. The situation got worse in summer and some relief in winter. As a photographer, he had the history of benzolism. He did not have history for sexually transmitted disease, trauma to this area, radiotherapy and chemical or arsenic exposure. No enlarged lymph node was found in inguinal areas, and no positive findings of thoracic X-ray, pelvic CT and abdominal B-ultrasonic scanning. The lesion was excised with a margin of 2.0 cm of normal skin. Pathological examination of the specimen verified that was a basal cell carcinoma (nodular type, Figure). No any positive findings were found in thoracic X-ray, pelvic CT and abdominal B-ultrasonic scanning during 3 years’ follow-up. DISCUSSION Scrotal carcinomas are uncommon, and less than 5% of scrotal carcinomas are basal cell origin.2 Although basal cell carcinoma occurs very rarely in pudendum, to our knowledge, 47 cases of scrotal BCC have been reported as series or clinical case reports mainly in the urological and dermatological literatures.1-15 The average age of the patients was 66.6 (42–82) years. Clinically the lesions presented mainly as nodular or ulceration and there was a Figure. Peripheral palisading basaloid cells invade the deep dermis (HE, original magnification ×200). wide variation of 3 months to 51 years, in the duration of the lesion before presentation.3 Based on data collected in those literatures, 5 (10.6%) patients have been reported to develop metastatic scrotal BCC. Metastasis arose 2–3 years after the onset of the primary disease which is shorter than the average of 11 years mentioned earlier in metastatic BCC at other sites.14 Exposure to ultraviolet (UV) radiation is considered to be the major etiological factor in the development of BCC,3,16,17 and the vast majority of lesions occur on the sun-exposed sites such as head and neck. However, the causative factors of BCC in areas not exposed to the sun are still unclear. It is possible that a generalized reduction in immunosurveillance with advancing age, or following excessive UV exposure elsewhere, predispose to their development.9,11 For example, renal transplant patients have a 10-fold increased risk of developing BCC as compared with the general population.17 Whether or not, ultraviolet is the most important factor in lesion development and progression. However, the rationale of the impairment of immune surveillance caused by ultraviolet radiation is still unclear. The study of human papillomavirus (HPV) in BCC specimens has been limited. About 10 cutaneous biopsy DOI: 10.3760/cma.j.issn.0366-6999.2010.19.026 Urology Department, Beijing Jishuitan Hospital, Beijing 100035, China (Wang JW, Man LB, He F, Huang GL, Li GZ and Wang HD) Correspondence to: Dr. MAN Li-bo, Urology Department, Beijing Jishuitan Hospital, Beijing 100035, China (Email: mmanlibo@ sohu.com) Chinese Medical Journal 2010;123(19):2748-2749 samples of scrotal BCC were tested for the presence of human papilloma virus by in situ hybridization or PCR,10,18 and failed to reveal DNA of HPV types. Although HPV DNA has been occasionally detected in reports of single cases or small series of patient with BCCs on sites other than the scrotum, a significant relationship between HPV and BCCs in the general population has yet to be established. 2749 4. 5. 6. 7. Human patched, a developmental gene playing an important role in regulation of cell growth and differentiation after embryogenesis, is mutated in BCC,16 and inactivation of this gene is probably a necessary if not sufficient step for tumor formation. Mutations of P53 have been documented in up to 40% of studied BCCs, 72% of the mutations bear the signature of UV light induction.19 Scrotal carcinoma was the first recognized occupational cancer as “Chimney sweeper’s Cancer”.17 The disease was described in chimney sweeps and related to exposure to soot and dust which contain the active agent as 3–4 benzpyrene. A variety of products, such as mineral oils, coal tar, petroleum, arsenic, solvents and wool oil, have been reported to be associated with scrotal BCC. The history of benzolism of this patient may indicate benzene is one of the important additional risk factors. Moreover, radiotherapy, long-standing fungal infection and chronic irritation may also be involved in carcinogenesis of the scrotum. Although some reports revealed the correlation between chronic ulcer, inflammation and the risk for development of BCCs or other skin cancer, no such report found as to chronic ulceration or inflammation on scrotum.20,21 We know most malignancies arising from chronic wounds take the form of squamous cell carcinoma, however, a study of 125 cases found that 25% patients with BCC had concomitant chronic venous stasis, suggesting a relationship between venous disease and BCC.22 8. 9. 10. 11. 12. 13. 14. 15. 16. 17. 18. Surgical extirpation is currently the standard of cure for BCC.17 Wider excision than the currently recommended 4-mm clinical margins may be required for removing tumor lesion more effectively and completely.23 Retrospective study showed a 5-year recurrence rate of 26% for BCC not completely excised compared with a rate of 14% for BCC completely excised. 17 19. 20. 21. REFERENCES 1. 2. 3. Jose I Esquivias Gomez, Antonio Gonzalez-Lopez, Encarnacion Velasco, Pozo T, del Villar A. Basal cell carcinoma of the scrotum. Australas J Dermatol 1999; 40: 141-143. Staley Tem, Nieh PT, Ciesielski TE, Cieplinski W. Metastatic basal cell carcinoma of the scrotum. J Urol 1983; 130: 792. Nahass GT, Blauvelt A, Leonardi CL, Penneys NS. Basal cell carcinoma of the scrotum. Report of three cases and review of 22. 23. the literature. J Am Acad Dermatol 1992; 26: 574-578. Ho WS, King WW, Chan WY, Li AK. Basal cell carcinoma of the scrotum. Natl Med J India 1995; 8: 195. Schleicher SM, Milstein HJ, Ilowite R. Basal cell carcinoma of the scrotum. Cutis 1997; 59: 116. Esquivias Gómez JI, González-López A, Velasco E, Pozo T, del Villar A. Basal cell carcinoma of the scrotum. Australas J Dermatol 1999; 40: 141-143. Takahashi H. Non-ulcerative basal cell carcinoma arising on the genitalia. J Dermatol 2000; 27: 798-801. Redondo Martínez E, López AR, Cruz Benavides F, Camacho Galan R. Basal cell carcinoma of the scrotum. A rare localization linked to a bad prognosis. Arch Esp Urol 2000; 53: 642-644. Vandeweyer E, Deraemaecker R. Basal cell carcinoma of the scrotum. J Urol 2000; 163: 914. Gibson GE, Ahmed I. Perianal and genital basal cell carcinoma: A clinicopathologic review of 51 cases. J Am Acad Dermatol 2001; 45: 68-71. Chave TA, Finch TM. The scrotum: an unusual site for basal cell carcinoma. Clin Exp Dermatol 2002; 27: 68. Ribuffo D, Alfano C, Ferrazzoli PS, Scuderi N. Basal cell carcinoma of the penis and scrotum with cutaneous metastases. Scand J Plast Reconstr Surg Hand Surg 2002; 36: 180-182. Izikson L, Vanderpool J, Brodsky G, Mihm MC Jr, Zembowicz A. Combined basal cell carcinoma and Langerhans cell histiocytosis of the scrotum in a patient with occupational exposure to coal tar and dust. Int J Dermatol 2004; 43: 678-680. Kinoshita R, Yamamoto O, Yasuda H, Tokura Y. Basal cell carcinoma of the scrotum with lymph node metastasis: report of a case and review of the literature. Int J Dermatol 2005; 44: 54-56. Handa Y, Kato Y, Ishikawa H. Giant superficial basal cell carcinoma of the scrotum. Eur J Dermatol 2005; 15: 186-188. Bale AE, Yu KP. The hedgehog pathway and basal cell carcinomas. Hum Mol Genet 2001; 10: 757-762. Ceilley RI, Del Rosso JQ. Current modalities and new advances in the treatment of basal cell carcinoma. Int J Dermatol 2006; 45: 489-498. Nehal KS, Levine VJ, Ashinoff R. Basal cell carcinoma of the genitalia. Dermatol Surg 1998; 24: 1361-1363. Crowson AN. Basal cell carcinoma: biology, morphology and clinical implications. Mod Pathol 2006; 19: s127-s147. Schnirring-Judge M, Belpedio D. Malignant transformation of a chronic venous stasis ulcer to basal cell carcinoma in a diabetic patient: case study and review of the pathophysiology. J Foot Ankle Surg 2010; 49: 75-79. Samira Y, Sérgio H, Michalany NS, de Almeida FA, Jane T. Squamous cell carcinoma in chronic ulcer in lepromatous leprosy. Dermatol Surg 2009; 35: 2025-2030. Aloi F, Tomasini C, Margiotta A,Pippione M. Chronic venous stasis: not a predisposing factor for basal cell carcinoma on the leg. A histopathological study. Dermatology 1994; 188: 91-93. Wong CS, Strange RC, Lear JT. Basal cell carcinoma. BMJ 2003; 327: 794-798. (Received May 31, 2010) Edited by GUO Li-shao