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KMID : 0363220170550080549
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Korean Journal of Dermatology
2017 Volume.55 No. 8 p.549 ~ p.551
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Clinical Experience of Ingenol Mebutate Therapy in Extramammary Paget Disease Accompanying Condyloma Acuminatum
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Oh Eui-Hyun
Park Hyung-Kwon
Kim Joung-Soo
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Abstract
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Extramammary Paget disease (EMPD) is a rare intraepidermal adenocarcinoma affecting apocrine gland bearing skin, which usually occurs in the anogenital area in patients over 50 years of age1. Due to its multifocal nature and indistinct demarcation, recurrences are common, even after invasive surgical treatment2. Herein, we report a case of EMPD that improved after the topical application of ingenol mebutate (IM) gel.
A 51-year-old man presented with a 1-year history of a painful erosive plaque on the scrotum and multiple verrucous papules that had subsequently developed within the plaque (Fig. 1A). His medical history was clear of any malignant tumors in internal organs. Histopathological examination of the plaque demonstrated Paget cells in the epidermis and a skin biopsy taken from the papular lesion showed thickened epidermis with papillomatosis and vacuolated cells (Fig. 1B, C). Human papilloma virus (HPV)-6 DNA was detected by oligonucleotide microarray. We diagnosed the patient with EMPD concomitant with condyloma acuminatum (CA). Given the limitations of wide surgical resection, alternative treatments were considered and topical application of IM 0.015% gel was administered once per session for 3 courses. Human papilloma virus (HPV)-related lesions were subjected to laser vaporization. During the first week of each session, the patient experienced moderate erythema with painful blistering. However, IM treatment resulted in clinical improvement of the EMPD lesion during the 2-month follow-up period and the inflammatory reaction was well-tolerated (Fig. 2). A post-treatment biopsy performed after the second session of therapy demonstrated the absence of malignant cells in the periphery of the lesion with remnant tumor cells in the central lesion (Fig. 1D∼F). He is currently under observation to be considered for further treatment including surgical resection of the remnant lesion.
The coexistence of EMPD and HPV-associated neoplasms has previously been reported3. In the present case, there was a focus of CA within the EMPD area. To the best of our knowledge, three other cases of EMPD accompanying CA have been reported4. It has been hypothesized that keratinocytes in the diseased skin have an increased susceptibility for viral infections, which might contribute to development of CA3.
Many therapeutic approaches to EMPD including surgery, chemotherapy, or irradiation have been used; however, relapse occurs in 30% or more of patients2. Recently, IM, which eradicates superficial skin cancer by inducing lesional cell death and immune responses, has been used as an efficient field therapy for actinic keratosis5. Although IM has been used for actinic keratosis or superficial basal cell carcinoma, there have been no reports on its use for treating EMPD. In our case, the EMPD lesion was a poor candidate for aggressive treatment due to its anatomic location; therefore, we applied IM gel and observed clinical and histological improvements without severe adverse effects. In fact, IM is useful for discrete lesions of dysplastic keratinocytes confined to the epidermis, and Paget cells mostly found in the basal epidermal layer could be targeted by IM. Moreover, the concept of field-directed therapy might be effective for subclinical extension of EMPD. However, longterm follow-up should be conducted to examine evidence of recurrence.
In conclusion, we recommend that IM gel be carefully considered as an adjuvant to surgery for EMPD, especially if there is extensive involvement requiring potentially mutilating wide excision.
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KEYWORD
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Condyloma acuminatum, Extramammary Paget, disease, Ingenol mebutate
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