INTRODUCTION

Penile angiokeratomas are rare, benign capillary lesions typically with small, multiple, dark spots on the glans penis or penile skin. The patients generally present with cosmetic concerns or bleeding. Histopathology reveals the presence of vascular dilatation in the papillary dermis, flanked by acanthotic rete ridges and overlying hyperkeratosis.¹ Angiokeratomas occur in localized and systemic forms. The localized types include solitary papular angiokeratoma, localized angiokeratoma of the scrotum and vulva (Fordyce type), angiokeratoma circumscriptum naeviforme and bilateral angiokeratoma (Mibelli type). These lesions are generally acquired and rarely exist on penile skin.^1,2 We report herein a case of larger angiokeratoma of the penile shaft that was treated successfully.

CASE PRESENTATION

A 49-year-old circumcised man presented with a 2-year history of penile lesion on the lateral side of the penis. The lesion persisted for approximately 2 years and was asymptomatic.

Physical examination revealed 20 mm solitary, firm, dark purple-red nontender erythematous papules on the shaft of the penis (fig. 1). There were no similar lesions or any other abnormality on the physical examination elsewhere. The laboratory values including hemogram and biochemical panel were within normal range. The patient had no predisposing factors such as inherited metabolic disorder (lysosomal storage diseases) or any systemic disease.

The patient underwent punch biopsy on the penile shaft 2 years ago and pathological examination revealed cavernous hemangioma. After the biopsy, the lesion had been slowly expanding within 2 years. Also, the patient felt discomfort during sexual intercourse because of the lesion. On penile magnetic resonance imaging, a lesion measuring 30 mm diameter on the right side of penile skin was described. The lesion did not invade the cavernous bodies (fig. 2). The patient decided to undergo surgical treatment of angiokeratoma.

In the supine position under general anesthesia, the incision line was drawn around the elliptical margin using a marking pen. Under 3.5× magnification, the mass was completely excised. After excision, the edges of skin were approximated (fig. 3). The final pathological examination revealed angiokeratoma.

DISCUSSION

Figure 1. Two cm, red-colored lesion on the penile shaft.

Figure 2. Magnetic resonance imaging of the lesion.

Figure 3. Penile skin imaging after the excision.

Angiokeratoma of the scrotum was initially described by John Addison Fordyce in 1896.³ Angiokeratomas are benign lesions characterized by vascular ectasia in the upper dermis, generally with acanthosis and hyperkeratosis. They generally present as a red and/or dark blue-domed hyperkeratotic papule and range in size from 0.5 to 5 mm.¹ Fordyce type angiokeratomas are generally located on the glans penis. In a previous review, 11 cases with penile angiokeratomas were located on the penile shaft.¹ In this review, all lesions were smaller than 15 mm. Unlike the previous cases, the lesion size on the penile shaft was 20 mm in our case.

Different treatment options such as excision, cryotherapy, Nd-YAG laser and pulsed dye laser were reported.¹ However, the optimum treatment procedure for regional therapy of angiokeratoma is unknown. Because of the larger size and patient preference, the lesion was excised surgically in our case. Also, some benign and malignant lesions such as infectious, epithelial, fibrous, vascular lesions can mimic the angiokeratomas. Angiokeratomas are difficult to diagnose. In a study, correct diagnosis of angiokeratomas has been reported at just 3%.⁴ The excision and histopathological evaluation are more appropriate for the diagnosis. In the followup, it was reported that 10% of patients had recurrence after therapy for penile angiokeratomas.¹ Two men after excision and 1 man after cryotherapy had recurrence. No recurrence developed after the excision in our case until now.

The etiology of genital angiokeratoma is still controversial but some factors such as long-standing venous blood pressure, primary degeneration of vascular elastic tissues and capillary injury are considered.⁵ The majority of the patients’ angiokeratomas were asymptomatic as in our case. Most of the cases had multiple lesions. The whole genital region should be evaluated by physical examination.

To our knowledge, this is the first case with an angiokeratoma of more than 15 mm localized unifocal on the penile shaft and successfully treated with surgery. Penile angiokeratomas are rare benign lesions and larger lesions can be treated successfully with surgery.