Lymphangioma circumscriptum (LC) is a rare, benign vascular malformation induced by abnormal lymphatic vessels of the skin. LC might be either congenital or acquired, and is predominantly located on the trunk, buttock, axillary region, or thighs. Penile LC is rare. This case report describes a patient with acquired LC associated with high-grade penile intraepithelial neoplasia induced by human papillomavirus type 66. As the patient had multifocal lesions on the glans penis and prepuce we decided to perform circumcision, followed by electrocoagulation of the lesions on the glans. Electrocautery should be considered as a first choice for treatment of LC located at surgically challenging regions such as the glans penis.
Keywords: Human papillomavirus; circumcision; treatment.