Attenuation of an adult T-cell leukemia skin lesion after treatment of a concomitant herpes simplex infection: a case study
© Tomita et al.; licensee BioMed Central Ltd. 2012
Received: 3 April 2012
Accepted: 26 September 2012
Published: 1 October 2012
We report the development and treatment of eczema herpeticum in a 51-year-old male suffering from adult T-cell leukemia (ATL). Lesions of eczema herpeticum coexisted with the skin lesions of ATL. Treatment of eczema herpeticum resulted in a concomitant improvement in the symptoms of ATL, including a reduction in the size of the ATL plaques, for over 2 months before relapse.
KeywordsAdult T-cell leukemia virus type 1 Herpes simplex Eczema herpeticum
Eczema herpeticum is an uncommon viral infection caused by the herpes simplex virus (HSV). It affects patients with a pre-existing primary dermatological condition, such as atopic dermatitis, psoriasis, mycosis fungoides, and/or burns . Although HSV infections occur frequently in adult T-cell leukemia (ATL) patients, eczema herpeticum at ATL skin lesions has never been reported before in the literature. In this Case Report, we describe the occurrence and treatment of eczema herpeticum in a patient suffering from ATL.
Small vesicles, crusts, erosion, and infiltrated itchy erythemas suddenly developed in the almost every pre-existing ATL skin lesions on the entire trunk (Figure 1b). Examination of a biopsy specimen revealed acantholysis and ballooning degeneration of epidermal keratinocytes in the vesicles (Figure 2c and d). These degenerated epidermal cells were positive for an anti-HSV antibody that recognized both HSV-1 and HSV-2 (Figure 2e). In addition, Pautrier’s microabscesses were observed adjacent to the erosion in the biopsied sample (Figure 2c). An enzyme immunoassay revealed levels of serum immunoglobulin G and M antibodies against HSV of over 128 (normal range <2.0) and 0.77 (normal range <0.8), respectively. These results indicated a recurrent HSV infection that was strictly limited to the pre-existing ATL skin lesions.
Development of eczema herpeticum appears to be linked to defects in cellular immunity and disruption of the skin barrier. A report suggested that the number of regulatory T (Treg) cells increases in ATL patients . Treg cells hamper the development of immunity against viral infections, such as HSV [3, 4]. In addition, HTLV-1 activates cellular genes, including the cytokine gene IL-4 . High levels of IL-4 allow the development of eczema herpeticum by suppressing the expression of cathelicidin peptide LL-37, which exhibits activity against HSV . It is also noteworthy that IL-17 mRNA is highly expressed in HTLV-1-infected T-cells  and that IL-17-mediated inhibition of natural killer cell activity induces eczema vaccinatum in mice . Together with disruption of the skin barrier in the ATL lesions, UV-irradiation and topical steroid treatment may also participate in HSV infection. Furthermore, increased Treg cells and high levels of IL-4 and/or IL-17 associated with ATL infection may lead skin conditions vulnerable to HSV infection.
A study has shown that adenovirus vaccine injection into the tumoral lesions of cutaneous T-cell lymphoma leads to tumor reduction by the induction of interferon gamma, a subtype 1 helper T (Th1) cell cytokine . In the present case, the Th1-dominant immune response for anti-viral immunity  may have been activated following HSV infection, whereupon it contributed to the attenuation of the ATL skin lesions.
HSV infection affected our patient only within the confines of pre-existing ATL skin lesions on his torso. Interestingly, these ATL skin lesions improved in parallel with the symptoms of HSV infection upon treatment with acyclovir. This uncommon phenomenon observed in the present case may shed a light to a development of novel strategies for treatment of ATL.
Written informed consent was obtained from the patient for publication of this Case Report and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal.
Adult T-cell leukemia
Hematoxylin and eosin
Herpes simplex virus
Human T-cell leukemia virus type 1
Subtype 1 helper T
- Santmyire-Rosenberger BR, Nigra TP: Psoriasis herpeticum: three cases of Kaposi’s varicelliform eruption in psoriasis. J. Am. Acad. Dermatol. 2005, 53: 52-56. 10.1016/j.jaad.2005.01.140.PubMedView Article
- Toulza F, Heaps A, Tanaka Y, Taylor GP, Bangham CR: High frequency of CD4 + FoxP3+ cells in HTLV-1 infection: inverse correlation with HTLV-1-specific CTL response. Blood. 2008, 111: 5047-5053. 10.1182/blood-2007-10-118539.PubMedPubMed CentralView Article
- Rouse BT, Suvas S: Regulatory cells and infectious agents: detentes cordiale and contraire. J Immunol. 2004, 173: 2211-2215.PubMedView Article
- Fernandez MA, Puttur FK, Wang YM, Howden W, Alexander SI, Jones CA: T regulatory cells contribute to the attenuated primary CD8+ and CD4+ T cell responses to herpes simplex virus type 2 in neonatal mice. J Immunol. 2008, 180: 1556-1564.PubMedView Article
- Li-Weber M, Giaisi M, Chlichlia K, Khazaie K, Krammer PH: Human T cell leukemia virus type I Tax enhances IL-4 gene expression in T cells. Eur J Immunol. 2001, 31: 2623-2632. 10.1002/1521-4141(200109)31:9<2623::AID-IMMU2623>3.0.CO;2-4.PubMedView Article
- Howell MD, Wollenberg A, Gallo RL, Flaig M, Streib JE, Wong C, Pavicic T, Boguniewicz M, Leung DY: Cathelicidin deficiency predisposes to eczema herpeticum. J Allergy Clin Immunol. 2006, 117: 836-841. 10.1016/j.jaci.2005.12.1345.PubMedPubMed CentralView Article
- Dodon MD, Li Z, Hamaia S, Gazzolo L: Tax protein of human T-cell leukaemia virus type 1 induces interleukin 17 gene expression in T cells. J Gen Virol. 2004, 85: 1921-1932. 10.1099/vir.0.79921-0.PubMedView Article
- Kawakami Y, Tomimori Y, Yumoto K, Hasegawa S, Ando T, Tagaya Y, Crotty S, Kawakami T: Inhibition of NK cell activity by IL-17 allows vaccinia virus to induce severe skin lesions in a mouse model of eczema vaccinatum. J Exp Med. 2009, 206: 1219-1225. 10.1084/jem.20082835.PubMedPubMed CentralView Article
- Whittaker SJ, Foss FM: Efficacy and tolerability of currently available therapies for the mycosis fungoides and Sezary syndrome variants of cutaneous T-cell lymphoma. Cancer Treat Rev. 2007, 33: 146-160. 10.1016/j.ctrv.2006.08.006.PubMedView Article
- Suryawanshi A, Veiga-Parga T, Rajasagi NK, Reddy PB, Sehrawat S, Sharma S, Rouse BT: Role of IL-17 and Th17 cells in herpes simplex virus-induced corneal immunopathology. J Immunol. 2011, 187: 1919-30. 10.4049/jimmunol.1100736.PubMedPubMed CentralView Article
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