Squamous Cell Carcinoma Arising From Massive Localized Lymphedema of Scrotum Mimicking Scrotal Smooth Muscle Hamartoma of Dartos

Abstract

Massive localized lymphedema (MLL) is an uncommon benign skin lesion typically presenting with prominent edema and vascular proliferation in the adipose tissue of lower limbs. When rarely occurring in scrotum, it instead is characterized by a striking proliferation of dermal smooth muscle bundles mimicking acquired smooth muscle hamartoma of dartos. The authors report a rare case of scrotal MLL. A 57-year-old obese man with a history of previous surgery for rectal adenocarcinoma, 20 years earlier, presented with progressive nodular enlargement of the scrotum for 2 years, causing discomfort, difficulty in ambulation, and cosmetic problems. The preoperative radiographic investigation revealed thickening of the scrotal wall with multiple soft-tissue nodules. The patient underwent a wide excision of the scrotal wall, perineum, and penile skin. The pathological examination showed a scrotal MLL associated with well-differentiated squamous cell carcinoma. The authors speculate that prior radiotherapy and surgery together with morbid obesity led to long-standing lymphedema that triggered the proliferation of smooth muscle cells, chronic epidermal change, and finally squamous cell carcinoma.

Concurrent bariatric surgery and surgical resection of massive localized lymphedema of the thigh. A case report

“…If left untreated, MLL may progress to angiosarcoma, which is seen in 13% of all reported MLL [5]. Squamous cell carcinoma has also been reported to arise from MLL [10].…”

Labia Majora Share

“…SCC has rarely been found to be associated with chronic lymphedema. So far, 4 congenital cases and 9 acquired cases, all arising in the extremities [ 7 , 8 ], as well as 3 cases with penoscrotal involvement [ 9 , 10 , 11 ], have been reported. To the best of our knowledge, this study is the first to report involvement of the labia.…”

Massive Localized Lymphedema Arising from Abdominal Wall: A Case Report and Review of the Literature

“…Since the underlying cause of MLL is lymphatic obstruction possibly due to morbid obesity, persistence or even recurrence of these lesions is expected after the surgical resection although recurrence with an aggressive manner has not been documented [ 1 ]. Recently, cases of squamous cell carcinoma and cutaneous angiosarcoma arising from long-standing MLL have been reported [ 12 , 13 ] probably through similar pathomechanism as Stewart–Treves syndrome. Hence long term follow-up is still recommended.…”