Isolated penile lymphedema: a case report and review of the literature

An 18-year-old man with a previous history of orchitis presented with a 2-year history of persistent penile edema that had not resolved since its onset. The edema developed two weeks prior to an appendectomy for acute appendicitis. It began acutely and gradually worsened. The patient did not recall any precipitating factors. The edema was more severe in the morning, often causing mild pain with micturition. He denied a history of penile discharge or dermatoses, erectile pain, trauma, sexually transmitted infections, and weight gain. Two lymph node biopsies, previously performed by urology, did not demonstrate granuloma, necrosis, or malignancy. A recent colonoscopy was within normal limits. Physical examination revealed edema extending from the penile base to the urethral meatus (Figure 1). Additionally, there were scattered illdefined mildly erythematous patches with sparing of the skin folds. The mons pubis, crural and inguinal folds, scrotal sac, and the skin of the perineum were within normal limits. A punch biopsy from the penile shaft demonstrated a proliferation of dilated lymphatic vessels, which were highlighted by a D2-40 stain, and a slight increase in interstitial edema (Figure 2). A complete blood count, metabolic panel, urinalysis, and urinary culture were within normal limits. A computed tomography scan of the abdomen and pelvis did not demonstrate any abnormalities. These results were suggestive of a diagnosis of idiopathic penile lymphedema.Treatment with pimecrolimus cream has resulted in improvement of the erythematous penile patches. He is currently seeking care with a reconstructive urologist for possible lymphangiectomy.