Squamous cell carcinoma of the penis and scrotum in a patient with chronic scrotal and penile lymphedema.

Squamous cell carcinoma arising from tissue affected by chronic lymphedema is rare, though it is recognized that a variety of malignant tumors can arise in chronic congenital or acquired lymphedema. We describe, a case of scrotal and penile squamous cell carcinoma arising in a patient with a history of chronic scrotal and penile lymphedema of filarial origin. We here discuss the management and possible etiology of this unusual case.

Introduction

Filarial genital lymphedema is a known entity involving the scrotum and the penis. Malignant changes which can arise in chronic lymphedema include Kaposi's sarcoma, melanoma, lymphoma, malignant fibrous histiocytoma, and squamous cell carcinoma.[1] The association with squamous cell carcinoma has been noted previously involving the upper limb and lower limb lymphedema, with additional factors for carcinogenesis. In this case we found no additional risk factors except for chronic lymphedema, suggesting that lymphedema itself may be a carcinogenic factor for squamous cell carcinoma.

We present here an unusual case of squamous cell carcinoma of scrotum and penis arising in a 45-year-old man with a history of filarial lymphedema.

Case Report

A 45-year-old male with no significant medical history presented to us with ulcer on the scrotum and penis of 4-month duration. He had a history of surgery for lymphedema scrotum done 8 years back, probably postfilarial; details were not available except photograph [Figure 1]. Considering this ulcer [Figure 2] which was infected, debridement was done along with an excision biopsy of the ulcer [Figures 3,4]. The biopsy report revealed squamous cell carcinoma of the scrotum and penis. Further a formal excision of the lymphedematous part of the scrotum and penis was done along with left orchidectomy and partial amputation of the penis with skin grafting of the penile shaft [Figure 5]. The histopathological report of the same showed squamous cell carcinoma with marginal clearance. The wound has healed without any complication [Figure 6]. The patient was referred to a medical oncologist for chemotherapy; at follow-up he clinically remains free from local recurrence and regional disease.

Figure 1

Lymphedema scrotum 8 years back

Figure 2

Ulcer over the scrotum and penis

Figure 3

Excision biopsy of the ulcer

Figure 4

Postoperative result after excision

Figure 5

Formal excision of the lymphedematous part of the scrotum and penis along with left orchidectomy and partial amputation of the penis with skin grafting of the penile shaft

Figure 6

Postoperative result

Discussion

Squamous cell carcinoma arising in chronic lymphedema is rare with only 11 cases reported in the literature. The majority of these are related to the limbs.[2] Squamous cell carcinoma in chronic penile lymphedema is still rarer.[3] The scarcity of reports in the literature may be taken as evidence against a casual relationship between chronic lymphedema and squamous cell carcinoma. However, the association may well be under reported. Most of the theories on carcinogenesis in lymphedema centre on immunological factors. It is believed that the deficiency in afferent lymphatic drainage prevents early recognition of the tumor-specific antigen.[4] Even a field change theory has been proposed suggesting that in some a chronic lymphedema promote carcinogenesis. This case illustrates the need for careful evaluation and follow-up of patients with chronic lymphedema. In practical terms, the potential for malignant growth in the lymphedematous tissue should be borne in mind. Chronic warty hyperkeratosis and fissuring may hide a lesion leading to delay in the presentation of a malignant ulcer. A dermatological opinion should be taken as and when necessary. Finally, the link between these two rare conditions should be remembered. Serum tumor markers for penile squamous cell carcinoma such as SCC antigen could play an essential role in surveillance of these difficult cases.[56] Patients should be instructed on good hygiene and regular monitoring of any affected area. Any new or suspicious growth should be examined and biopsied without delay.