Journal of Clinical Virology 56 (2013) 163–166 Contents lists available at SciVerse ScienceDirect Journal of Clinical Virology j ourna l ho mepage: www.elsevier.com/locate/jcv Case report Double integration band of HTLV-1 in a young patient with infective dermatitis who developed an acute form of adult T-cell leukemia/lymphoma Pedro D. Oliveira a , Marcelo Magalhães b , Juliana M. Argolo b , Achiléa L. Bittencourt c,d , Lourdes Farre b,∗,d a Service of Dermatology, Complexo Hospitalar Universitário Prof. Edgard Santos, Federal University of Bahia, Rua Dr. Augusto Viana, s/n, Canela, CEP: 40.110-060, Salvador, Bahia, Brazil b Laboratory of Experimental Pathology (LAPEX), CPQGM–FIOCRUZ, Bahia, Rua Waldemar Falcão 121, Candeal, CEP: 40296-710, Salvador, Bahia, Brazil c Service of Pathology, Complexo Hospitalar Universitário Prof. Edgard Santos, Federal University of Bahia, Rua Dr. Augusto Viana, s/n, Canela, CEP: 40.110-060, Salvador, Bahia, Brazil a r t i c l e i n f o Article history: Received 3 July 2012 Received in revised form 17 October 2012 Accepted 18 October 2012 Keywords: HTLV-1 Adult T-cell leukemia/lymphoma Infective dermatitis Proviral integration a b s t r a c t Few cases of acute adult T-cell leukemia/lymphoma (ATL) have been diagnosed in young patients. This report is the first to describe a young girl with infective dermatitis associated with HTLV-1 that progressed to acute ATL with Southern blot hybridization and gamma-TCR-rearrangement revealing a monoclonal pattern with two copies of the provirus. © 2012 Elsevier B.V. All rights reserved. 1. Why this case is important? Adult T-cell leukemia/lymphoma (ATL) generally occurs after a long period of human T-cell lymphotropic virus type 1 (HTLV- 1) infection. 1 However, rare cases of ATL have been described in patients with infective dermatitis associated with HTLV-1 (IDH) prior to 20 years of age. 2–4 Only one of these patients, already in remission of IDH, developed the acute form of ATL. 3 To the best of our knowledge, this report represents the first case of IDH associated with acute ATL with a double band of HTLV-1 integration. 2. Case report A 19-year-old girl born and living in Bahia, Brazil presented at the Federal University of Bahia Teaching Hospital with recurrent eczema on her scalp, retroauricular area, face and neck that had been present since she was three years old. She reported a history of papules all over her body for the past six months. She informed that she had used oral corticosteroids to treat the lesions. Polymerase ∗ Corresponding author at: Laboratory of Experimental Pathology, Centro de Pesquisa Gonc ¸ alo Moniz-Fundac ¸ ão Oswaldo Cruz. Rua Waldemar Falcão, 121, Brotas – CEP: 40.295-001, Salvador, Bahia, Brazil. Tel.: +55 7131762228; fax: +55 7131762228. E-mail address: lfarre@bahia.fiocruz.br (L. Farre). d These authors contributed equally to this study. chain reaction (PCR) was performed using DNA from peripheral blood mononuclear cells (PBMC) to confirm the presence of HTLV- 1 infection. 5 Physical examination revealed erythematous, scaly and crusty lesions on the scalp and retroauricular region associ- ated with disseminated erythematous macules and papules, some of which were ulcerated. The palms of her hands and the soles of her feet were also affected. Most of the papules were concentrated on her forehead and on the back of her neck (Fig. 1). She also pre- sented with blepharoconjunctivitis and hepatosplenomegaly. She reported having lost 3 kg of body weight. Laboratory tests showed marked lymphocytosis (428,000 lymphocytes/mm 3 ), neutrophilia, eosinophilia, thrombocytopenia and anemia. The presence of 23% of atypical cells was found, including 8% of flower cells in smears of peripheral blood. There was hypercalcemia and a marked increase in lactate dehydrogenase (LDH) (7 times the upper normal limit). Peripheral blood was acquired in FACScalibur (Becton-Dickinson, San Jose, CA, USA) for immunophenotyping, 6 which showed 97% of small and large CD2+ T lymphocytes, as well as CD3+, CD4+, CD7-, CD8- and CD25+ lymphocytes, suggesting a lymphocytic proliferative disease, compatible with ATL. 6 Bone marrow biopsy showed no infiltration by malignant cells. Skin biopsy revealed atypical medium and large lymphocytes infiltrating the upper and middle dermis. The immunohistochemical study was per- formed on formalin-fixed, paraffin-embedded sections using a standard streptavidin–biotin–peroxidase technique, 7 and revealed CD2+, CD3+, CD4+, CD5-, CD8-, CD20-, CD25+ and CD79a- cells, the same phenotype of peripheral blood. A marked epider- motropism with Pautrier’s abscesses was also observed (Fig. 2). 1386-6532/$ – see front matter © 2012 Elsevier B.V. All rights reserved. http://dx.doi.org/10.1016/j.jcv.2012.10.010