Correspondence Address:
Barnita Saha
Department of Skin and VD, Pt. B. D. Sharma PGIMS, Rohtak, Haryana
India

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Dear Editor,

A 25-year-old unmarried man presented with multiple asymptomatic skin colored to reddish-purple raised skin lesions on the shaft of the penis since 4 years. These lesions had started abruptly as small spots and had gradually increased in size and number. There was an occasional history of bleeding on touching the lesions. There was no history of high-risk sexual behavior in the patient and he had not received any treatment. Cutaneous examination revealed a plaque of size 3 cm × 2 cm with multiple studded skin colored to reddish-purple hyperkeratotic papules of size 2–4 mm on the margin located on the middle aspect of the shaft of penis on the dorsal surface [Figure 1]. Lesions were soft to firm in consistency, nontender, slightly compressible, nonpulsatile, and free from the underlying structure. There was no involvement of the scrotum and inguinal lymphadenopathy was absent. A punch biopsy was taken from one of the papules that revealed epidermal hyperkeratosis and dilated blood vessels in the papillary dermis [Figure 2]. Ultrasonography of abdomen, pelvis, and genital area was performed, which showed mild thickening of skin and subcutis on the dorsum of the penile shaft with slightly raised echogenicity and slow flow on color Doppler in the penile shaft area suggestive of venous malformation [Figure 3] and [Figure 4]. On the basis of clinical, histopathological, and radiological features, a diagnosis of angiokeratoma of Fordyce on the shaft of the penis with underlying venous malformation was made.{Figure 1}{Figure 2}{Figure 3}{Figure 4}

Angiokeratomas are benign cutaneous vascular proliferations characterized by dilated thin-walled blood vessels lying in the upper dermis, with hyperkeratosis at the surface. Several clinical variants of angiokeratomas exist: the Mibelli type, the solitary and multiple (papular type), the Fordyce type, the angiokeratoma circumscriptum, and the angiokeratoma corporis diffusum. Solitary or multiple (papular types) usually involves the lower extremities.[1] Angiokeratoma corporis diffusum is associated with a systemic disease such as Fabry's disease or fucosidosis.[2] Angiokeratoma circumscriptum usually presents as papules with a tendency to confluence into plaques.[3] Angiokeratoma of Mibelli characterized by papules or verrucoid nodules is more commonly present in men and involves bony prominences.[4] Angiokeratomas of Fordyce is most often observed on the scrotum or vulva but has also been noted on the glans penis and clitoris.[1] Angiokeratomas of the penis are rare vascular lesions that are most commonly present as multiple lesions along the coronal rim of the glans penis.[5] Our patient presented with multiple angiokeratoma on the shaft of penis; the morphologic features of the lesion were most suggestive of angiokeratoma of Fordyce. There are only six cases of angiokeratoma of Fordyce on shaft of penis reported in the literature as summarized in [Table 1], but literature search could not reveal any similar case in our search efforts like that of angiokeratoma on the shaft of the penis with underlying venous malformation.[2],[8],[9],[10],[11] The pathogenesis of genital angiokeratomas remains to be definitively established. Among the proposed hypothesis are congenital defects in venule walls, malformations, and acquired injury to vessel walls due to trauma or long-standing venous hypertension.[6] The common clinical differentials affecting this area include seborrheic keratoses, melanocytic nevi, dermatofibroma, bowenoid papulosis, molluscum contagiosum, condyloma acuminata, and lymphangioma circumscriptum. The histopathological differentials include lymphangioma (dilated lymphatics with clear fluid in dermis), pyogenic granuloma (polypoidal outgrowth of angiomatous tissue with mixed inflammatory cells in deeper part), and cherry angioma, hemangioma (lacks epidermal hyperkeratosis and acanthosis). The treatment of genital angiokeratomas mainly consists of observation as they are benign and asymptomatic. However, for lesions that are cosmetically undesirable or symptomatic, multiple treatment modalities are available such as cryotherapy, electrocoagulation, laser, sclerotherapy, and surgical excision.[7] We report our case because of the rarity of its occurrence.{Table 1}

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