Epidermodysplasia-Verruciformis-Like Eruption Associated with Gamma-Papillomavirus Infection in a Patient with Adult T-Cell Leukemia

Abstract

Epidermodysplasia verruciformis (EV) is a genodermatosis characterized by widespread and persistent cutaneous lesions caused by β-papillomaviruses. Rare cases of acquired EV-like eruption associated with β-papillomavirus infection have been reported in immunosuppressed patients. We report a case of acquired EV-like eruption in an immunosuppressed patient with adult T-cell leukemia. The cutaneous lesions clinically resembled pityriasis versicolor and exhibited the typical histological features of EV, but in some areas of the same biopsy specimen characteristic homogeneous intracytoplasmic inclusion bodies were observed. Although β-papillomavirus was not detected by highly sensitive polymerase chain reaction, a putative novel type of γ-papillomavirus was identified. This is the first documentation of an association between EV-like eruption and γ-papillomavirus infection.

Epidermodysplasia Verruciformis in Human Immunodeficiency Virus–Infected Patients

“…COMMENT Epidermodysplasia verruciformis-like eruptions have rarely been described in immunodeficient patients, unlike HPV-associated anogenital malignant diseases. [5][6][7][8][9] The EV phenotype and EV-HPV genotypes [30][31][32] were first reported in kidney transplant recipients and then in hematological malignant diseases, [33][34][35] iatrogenic immunodeficiency, 36,37 and CD4 ϩ T lymphocytopenia. 38,39 In HIVpositive patients, HAART resulted in a decrease in the incidence of opportunistic infections, but the prolonged survival was associated to the emergence of cutaneous and mucosal HPV-related diseases, especially anogenital intraepithelial neoplasia.…”

“…32 Gamma HPV-65 and HPV-38 found in patient 6 had previously been described in genetic EV 41 and in an immunocompromised patient. 34 Unlike patients with genetic EV who are only susceptible to ␤-HPV, HIV-positive patients with EV often displayed other HPV-related diseases including mucosal HPV in 64% of cases. Most of these mucosal HPV were oncogenic, highlighting the risk of intraepithelial cervical neoplasia observed in HIV-positive women.…”

Acquired Epidermodysplasia Verruciformis: A Comprehensive Review and a Proposal for Treatment

“…Recently an EV‐like syndrome has been described in patients with impaired cell‐mediated immunity, and the term “acquired EV” has been introduced. This form has been described in immunosuppressed patients with the human immunodeficiency virus; organ transplantation; lepromatous leprosy; Hodgkin's disease; systemic lupus erythematosus; common variable immunodeficiency; warts, immunodeficiency, lymphedema, anogenital dysplasia syndrome (WILD syndrome); immunoglobulin M deficiency; and adult T‐cell leukemia and in the setting of graft‐versus‐host disease . Treatment with biological agents may also increase the risk of HPV infections, including acquired EV …”

“…In half of individuals with EV, these lesions, especially when present on sun-exposed areas, progress to cutaneous malig-*Department of Dermatology, Policlinico A. Gemelli, Universit a Cattolica del Sacro Cuore, Rome, Italy; † Department of Dermatology, University of Padua, Padua, Italy 14,15 Recently an EV-like syndrome has been described in patients with impaired cell-mediated immunity, and the term "acquired EV" has been introduced. This form has been described in immunosuppressed patients with the human immunodeficiency virus; 3,4,16,17 organ transplantation; 18,19 lepromatous leprosy; 20 Hodgkin's disease; 21 systemic lupus erythematosus; 5,22,23 common variable immunodeficiency; 24 warts, immunodeficiency, lymphedema, anogenital dysplasia syndrome (WILD syndrome); 25 immunoglobulin M deficiency; 26 and adult T-cell leukemia 27 and in the setting of graft-versus-host disease. 28 Treatment with biological agents may also increase the risk of HPV infections, including acquired EV.…”

Multimodal Treatment of Epidermodysplasia Verruciformis in an HIV-Positive Man

“…[1][2][3] Acquired cases have been reported in patients lacking immunocompetency, including transplant recipients and patients living with HIV. [4][5][6][7][8][9][10][11] We present the case of a patient with HIV-associated EDV who was treated successfully with intralesional Candida albicans antigen, oral acitretin, and cryotherapy.A 56-year-old man presented for evaluation of several cutaneous lesions that had developed over several months on the neck and over many years on the hands and feet. He had a 16-year history of HIV, Castleman disease, and primary effusion lymphoma in remission that was treated with rituximab, etoposide phosphate, prednisone, vincristine sulfate, cyclophosphamide, and doxorubicin hydrochloride 10 or more years ago.…”