We have read with interest the article “Pseudoverrucous Lesions of Recent Appearance on the Vulva” in which the authors report the case of an adult woman with acquired vulvar lymphangioma.1 In their article they stress the need to correctly diagnose the condition and to avoid confusion with other disorders, such as genital warts, in order to identify the appropriate treatment and the underlying cause (lymphadenectomy, radiation therapy for pelvic carcinoma, Crohn disease, or tuberculosis).1,2

In the clinical history, the authors note as an interesting fact that the patient had been diagnosed 5 years earlier with genital warts, which were treated successfully with imiquimod 5% cream. In our opinion, the value of the article would have been even greater if the authors had analyzed the possible role of imiquimod therapy in the pathogenesis of the lymphangioma.

It is possible that the imiquimod therapy may, while resolving the infectious process, have given rise to lymphedema and that this condition then favored the development of the lymphangioma. Edema and lymphedema are adverse effects reported to be associated with imiquimod treatment. These conditions may resolve rapidly (for example, in the genital area),3 or persist for months or even years (reported in 1 case on the cheek following prolonged use of imiquimod to treat lentigo maligno melanoma).4 The influence of lymphedema on the development of lymphangiomas is also well known.2,5

We do not know whether the initial diagnosis of genital warts was confirmed by histopathology. If that diagnosis was not confirmed, it is possible that the earlier lesions were, in fact, lymphangioma. In that event, the interest of the case would be in the partial response to treatment with imiquimod. Imiquimod has been shown to be an effective therapy in some cases of lymphangioma circumscriptum. Occasionally, as occurred in this case, the lesion recurs months later.6 It is thought that imiquimod has an antiangiogenic effect and induces apoptosis of the tumor cells.6,7

In conclusion, we found this recent report of a cases of acquired lymphangioma circumscriptum very interesting. It would be important to know what imiquimod regimen was used and the duration of treatment, as this information could be used to hypothesize about the possible role of imiquimod in the development of the lymphangioma.