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    Hydroa vacciniforme-like cutaneous T-cell lymphoma in a child: A case report.
    Medicine (Baltimore) 2018 04;97(15):e0319
    Department of Pediatrics, West China Second University Hospital.
    Rationale: Hydroa vacciniforme (HV)-like T-cell lymphoma is a rare malignancy in childhood associated with Epstein-Barr virus infection.

    Patient Concerns: A 6-year old girl presented with complaint of 3-year history of recurrent skin lesions, 3 months of fever accompanied by cough for 8 days.

    Diagnoses: Skin biopsy revealed a HV-like lymphoma presentation and positive signals of EBER were detected by in situ hybridization. Read More

    GATA2 Deficiency and Epstein-Barr Virus Disease.
    Front Immunol 2017 22;8:1869. Epub 2017 Dec 22.
    Medical Virology Section, Laboratory of Infectious Diseases, National Institute of Allergy and Infectious Diseases, National Institutes of Health, Bethesda, MD, United States.
    GATA2 is a transcription factor that binds to the promoter of hematopoietic genes. Mutations in one copy of the gene are associated with haploinsufficiency and reduced levels of protein. This results in reduced numbers of several cell types important for immune surveillance including dendritic cells, monocytes, CD4, and NK cells, as well as impaired NK cell function. Read More

    Characterization of skin blister fluids from children with Epstein-Barr virus-associated lymphoproliferative disease.
    J Dermatol 2018 Apr 20;45(4):444-449. Epub 2018 Jan 20.
    Department of Pediatrics, School of Medicine, Institute of Medical, Pharmaceutical and Health Sciences, Kanazawa University, Kanazawa, Japan.
    Epstein-Barr virus (EBV)-associated T- or natural killer (NK)-cell lymphoproliferative disease (LPD) is a heterogeneous group of disorders characterized by chronic proliferation of EBV-infected lymphocytes. Patients may present with severe skin manifestations, including hypersensitivity to mosquito bites (HMB) and hydroa vacciniforme (HV)-like eruption, which are characterized by blister formation and necrotic ulceration. Skin biopsy specimens show inflammatory reactions comprising EBV-infected lymphocytes. Read More

    A distinct subtype of Epstein Barr virus positive T/NK-cell lymphoproliferative disorder: Adult patients with chronic active Epstein Barr virus infection-like features.
    Haematologica 2017 Dec 14. Epub 2017 Dec 14.
    Department of Pathology, Kurume University School of Medicine, Kurume, Japan.
    The characteristics of adult patients with chronic active Epstein-Barr virus infection (adult-onset CAEBV) are poorly recognized, hindering early diagnosis and an improved prognosis. Adult-onset CAEBV (n = 54) diagnosed between 2005 and 2015 were conducted. Adult-onset was defined as an estimated age of onset ≥15 years. Read More

    Primary cutaneous T-cell lymphoma: experience from the Peruvian National Cancer Institute.
    An Bras Dermatol 2017 Sep-Oct;92(5):649-654
    Medical Oncology Service, Instituto Nacional de Enfermedades Neoplásicas (INEN) - Surquillo, Peru.
    Background: Primary cutaneous T-cell lymphomas constitute a heterogeneous and rare group of diseases with regional particularities in Latin America.

    Objective: To determine the clinicopathological features, relative frequency and survival among patients from a Peruvian institution.

    Methods: Primary cutaneous T-cell lymphomas were defined based on the absence of extracutaneous disease at diagnosis. Read More

    Epstein-Barr Virus-Associated Vesiculopapular Eruption on the Face of a Patient with Natural Killer T Cell Lymphoma.
    Ann Dermatol 2017 Oct 25;29(5):618-620. Epub 2017 Aug 25.
    Department of Dermatology, Seoul National University Hospital, Seoul, Korea.
    Unlike typical hydroa vacciniforme (HV), Epstein-Barr virus (EBV)-associated HV-like eruption is more variable in its clinical manifestations. In some patients, progression to lymphoma or leukemia has been reported, which are characterized by the T-cell immunophenotype. Here, we report the first Korean case of EBV-associated vesiculopapular eruption on the face of a patient with natural killer (NK)/T cell lymphoma. Read More

    Recurrent scarring papulovesicular lesions on sun-exposed skin in a 22-year-old man.
    J Am Acad Dermatol 2018 Mar 28;78(3):637-642. Epub 2017 Sep 28.
    Laboratory of Cancer Biology and Genetics, National Cancer Institute, National Institutes of Health, Bethesda, Maryland. Electronic address:

    Epstein-Barr Virus-associated Lymphoproliferative Disorders in the Skin.
    Surg Pathol Clin 2017 Jun 18;10(2):429-453. Epub 2017 Mar 18.
    Haematological Malignancy Diagnostic Services (HMDS), Level 3, Bexley Wing, St James's University Hospital, Leeds LS9 7TF, UK. Electronic address:
    Epstein-Barr virus (EBV)-associated lymphoproliferations involving the skin are a rare but important group of diseases with a broad spectrum of behavior, ranging from self-limiting spontaneously resolving disorders to highly aggressive malignancies. They may be of B, T, or natural killer (NK) cell type and include EBV-positive mucocutaneous ulcer, lymphomatoid granulomatosis, EBV-positive diffuse large B-cell lymphoma, hydroa vacciniforme-like lymphoproliferative disorder, and extranodal NK/T-cell lymphoma of nasal type. Recognition and distinction of these entities is important in view of their differing prognoses and treatments. Read More

    Hydroa Vacciniforme-Like Lymphoma in Tibetan Children: 2 Cases and a Literature Review.
    Am J Dermatopathol 2018 May;40(5):358-361
    Department of Dermatovenereology, West China Hospital, Sichuan University, Sichuan, China.
    Hydroa vacciniforme-like lymphoma shows a marked predilection for the pediatric population, especially in Latin America and Asia. Here, we present 2 pediatric patients from Tibet of China, which is the first report from this area. Read More

    Hydroa Vacciniforme-Like T-Cell Lymphoma: A Further Brazilian Case.
    Am J Dermatopathol 2018 Mar;40(3):201-204
    Hydroa vacciniforme (HV)-like lymphoma is a rare, usually fatal Epstein-Barr virus-driven lymphoproliferative disease affecting children from Asia, Mexico, and South America. Cutaneous manifestations imitate HV, a benign photodermatosis in which systemic symptoms are not observed, and spontaneous regression occurs later in adolescence or young adulthood. We report a case of HV-like lymphoma in a 12-year-old girl, descendent from an ancient Amazon indigenous tribe that, as far as we know, represents the second Brazilian case ever reported in the medical literature. Read More

    How we treat chronic active Epstein-Barr virus infection.
    Int J Hematol 2017 Apr 16;105(4):406-418. Epub 2017 Feb 16.
    Department of Hematology/Oncology, Osaka Medical Center and Research Institute for Maternal and Child Health, 840 Murodo, Izumi City, Osaka, 594-1101, Japan.
    Chronic active Epstein-Barr virus infection (CAEBV) is a prototype of the EBV-associated T- or NK-cell lymphoproliferative diseases, which also include hypersensitivity to mosquito bites and severe-type hydroavacciniforme. The manifestations of CAEBV are often self-limiting with minimum supportive care or only prednisolone and cyclosporine A with or without etoposide. However, allogeneic hematopoietic stem cell transplantation (HSCT) is the only cure, without which patients with CAEBV die within several years. Read More

    [The understanding of Epstein-Barr virus associated lymphoproliferative disorder].
    Zhonghua Bing Li Xue Za Zhi 2016 Dec;45(12):817-821
    Department of Pathology, Beijing Friendship Hospital Capital Medical University, Beijing 100050, China.
    In recent years, there are increasing articles concerning Epstein-Barr virus associated lymphoproliferative disorder (EBV+ LPD), and the name of EBV+ LPD is used widely. However, the meaning of EBV+ LPD used is not the same, which triggered confusion of the understanding and obstacles of the communication. In order to solve this problem. Read More

    Cutaneous EBV-related lymphoproliferative disorders.
    Semin Diagn Pathol 2017 Jan 7;34(1):60-75. Epub 2016 Dec 7.
    Hematopathology, National Cancer Institute (NCI), Bethesda, MD, USA.
    This article will focus on the cutaneous lymphoproliferative disorders associated with EBV, with an emphasis on the upcoming changes in the revised 4th Edition of the WHO classification of tumors of the hematopoietic system, many of which deal with cutaneous disorders derived from NK-cells or T-cells. Extranodal NK/T-cell lymphoma usually presents in the upper aerodigestive tract, but can involve the skin secondarily. EBV-associated T- and NK-cell lymphoproliferative disorders (LPD) in the pediatric age group include the systemic diseases, chronic active EBV infection (CAEBV) and systemic EBV+ T-cell lymphoma of childhood. Read More

    Photodermatoses: Kids are not just little people.
    Clin Dermatol 2016 Nov - Dec;34(6):724-735. Epub 2016 Jul 12.
    University of Connecticut School of Medicine, Department of Dermatology, Farmington, CT.
    Photodermatoses are a group of skin disorders caused by abnormal reaction to ultraviolet radiation. Photodermatoses are divided into four groups: (1) immunologically mediated photodermatoses; (2) chemical- and drug-induced photodermatoses; (3) photoaggravated dermatoses; and (4) hereditary photodermatoses. This contribution discusses differences in the approach and diagnosis of pediatric and adult patients with suspected photodermatoses, focusing on immunologically mediated photodermatoses and chemical- and drug-induced photodermatoses. Read More

    Differential diagnosis of herpetiform vesicles by a non-invasive, molecular method using crusts or blister roofs: Sensitivity, specificity and likelihood ratio.
    J Dermatol Sci 2016 Dec 3;84(3):358-359. Epub 2016 Oct 3.
    Department of Dermatology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, 700-8558, Okayama, Japan. Electronic address:

    Hydroa vacciniforme and hydroa vacciniforme-like T-cell lymphoma: an uncommon event for transformation.
    J Cutan Pathol 2016 Dec 9;43(12):1102-1111. Epub 2016 Sep 9.
    Department of Dermatology, College of Medicine, National Cheng Kung University and Hospital, Tainan, Taiwan.
    Background: Hydroa vacciniforme (HV) is associated with Epstein-Barr virus (EBV) infection and a risk of transformation to lymphoma.

    Methods: We retrospectively analyzed six HV cases for EBV association and transformation to HV-like T-cell lymphoma. Clinicopathologic features were reviewed and cases were assessed for EBV-encoded RNA (EBER) by in situ hybridization, double staining with immunohistochemistry and EBER and for T-cell clonality. Read More

    Association of GATA2 Deficiency With Severe Primary Epstein-Barr Virus (EBV) Infection and EBV-associated Cancers.
    Clin Infect Dis 2016 Jul 11;63(1):41-7. Epub 2016 May 11.
    Laboratorie of Clinical Infectious Diseases, National Institute of Allergy and Infectious Diseases.
    Background: Most patients infected with Epstein-Barr virus (EBV) are asymptomatic, have nonspecific symptoms, or have self-limiting infectious mononucleosis. EBV, however, may result in severe primary disease or cancer.

    Methods: We report EBV diseases associated with GATA2 deficiency at one institution and describe the hematology, virology, and cytokine findings. Read More

    Clinicopathological categorization of Epstein-Barr virus-positive T/NK-cell lymphoproliferative disease: an analysis of 42 cases with an emphasis on prognostic implications.
    Leuk Lymphoma 2017 01 9;58(1):53-63. Epub 2016 May 9.
    a Department of Pathology , Seoul National University Hospital, Seoul National University College of Medicine , Seoul , South Korea.
    Epstein-Barr virus-positive T/NK-cell lymphoproliferative diseases (EBV-T/NK-LPDs) include several overlapping EBV-related conditions with variably aggressive courses. For prognostic categorization, we retrospectively analyzed 42 EBV-T/NK-LPD cases. Male (79% [33/42]), young (≤40 years; 83% [35/42]) patients and T-cell lineage (81% [34/42]; CD8/CD4 = 1. Read More

    Epstein-Barr virus reactivation is induced, but abortive, in cutaneous lesions of systemic hydroa vacciniforme and hypersensitivity to mosquito bites.
    J Dermatol Sci 2016 Jun 5;82(3):153-9. Epub 2016 Mar 5.
    Department of Dermatology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, 2-5-1 Shikata-cho, Kita-ku, Okayama 700-8558, Japan. Electronic address:
    Background: Epstein-Barr virus (EBV)-associated T/natural killer (NK)-lymphoproliferative disorders (LPDs) include hydroa vacciniforme (HV) and hypersensitivity to mosquito bites (HMB). The pathomechanisms of these diseases are still unclear.

    Objective: To understand the inflammatory process, we examined EBV reactivation markers, BZLF1 and BDRF1 mRNA in the tissue and blood from patients with EBV-associated T/NK-LPDs. Read More

    Mature T- and NK-cell non-Hodgkin lymphoma in children and young adolescents.
    Br J Haematol 2016 05 15;173(4):573-81. Epub 2016 Mar 15.
    Department of Pathology and Laboratory Medicine, Hospital of the University of Pennsylvania, University of Pennsylvania, Philadelphia, PA, USA.
    Mature T/Natural killer (NK)-cell neoplasms of children and the young adolescent population exhibit higher prevalence in Central and South American and Asian populations and many are associated with Epstein-Barr virus (EBV). They are represented in large part by extranodal T/NK cell lymphomas- nasal-type or extra nasal-type, chronic lymphoproliferative disorders of T/NK cells or chronic active EBV disease, systemic EBV-positive lymphoproliferative disorders of childhood, hydroa vacciniforme-like lymphoma, hepatosplenic T-cell lymphoma and primary cutaneous gamma/delta T-cell lymphoma among others. Many T/NK cell neoplasms in this age group are derived from cells of the innate immune system, in contrast to adults where they are predominantly from the adaptive immune system. Read More

    Hydroa Vacciniforme-Like EBV-Positive Cutaneous T-Cell Lymphoma, First Report of 2 Cases in Ecuador.
    Am J Dermatopathol 2016 May;38(5):e57-9
    Servicio de Patología, Hospital Metropolitano, Quito, Ecuador.
    Hydroa vacciniforme-like cutaneous lymphoma is a very rare Epstein-Barr virus positive peripheral T-cell lymphoma affecting Asian and Hispanic children and young adults with a defective cytotoxic immune response to EBV predisposing to the development of the disease. We report on 2 Ecuadorian patients with papulovesicular and ulcerated crusted lesions on the face, upper and lower extremities and abdomen, with aggressive clinical course and, in one case, a fatal outcome. The histological and molecular profiles (immunohistochemistry and in situ hybridization) established a diagnosis of hydroa vacciniforme-like Epstein-Barr virus-encoded small RNAs + cutaneous T-cell lymphoma in both cases. Read More

    Progressive scarring facial lesions in a boy.
    Clin Case Rep 2016 Feb 6;4(2):120-2. Epub 2015 Dec 6.
    Dermatology Service KK Women's & Children's Hospital Singapore Singapore.
    Hydroa vacciniforme is a photosensitivity disorder characterized by the childhood onset of necrotic vesiculopapules on exposed areas. We present a case of a 10-year-old boy with a 1-year history of papular lesions over his face. In this report, we discuss the in-depth histology of hydroa vacciniforme, and the management options. Read More

    Chronic active EBV infection: the experience of the Samsung Medical Center in South Korea.
    Bol Med Hosp Infant Mex 2016 Jan - Feb;73(1):10-17. Epub 2016 Feb 1.
    Department of Pathology, Samsung Medical Center, Sungkyunkwan University, Seoul, Korea. Electronic address:
    Background: Chronic active EBV infection (CAEBV) of T-cell or NK-cell type is an EBV+ polyclonal, oligoclonal or often monoclonal lymphoproliferative disorder (LPD) recognized as representing the spectrum of EBV-associated T-cell and NK-cell LPD with different clinical presentations; one systemic and two cutaneous disorders including hydroa vacciniforme-like T-cell LPD and mosquito bite hypersensitivity. The systemic form of the disease is characterized by fever, persistent hepatitis, hepatosplenomegaly and lymphadenopathy, which shows varying degrees of clinical severity depending on the immune response of the host and the EBV viral load.

    Case Reports: We described the clinicopathological findings of two children with CAEBV with a brief review of the literature. Read More

    Clinicopathologic Features of Hydroa Vacciniforme-Like Lymphoma: A Series of 9 Patients.
    Am J Dermatopathol 2016 Jan;38(1):20-5
    *Centre for Dermatology & Dermatopathology and School of Medicine, Universidad Nacional Autónoma de México/Hospital General de México "Eduardo Liceaga," México, DF, Mexico; and †Research Unit Dermatopathology, Department of Dermatology, Medical University of Graz, Graz, Austria.
    Hydroa vacciniforme-like lymphoma is a recently recognized cutaneous T-cell lymphoma associated with Epstein-Barr virus. The disease is observed in children of Latin American or Asian ethnicity. The authors report the clinical, histopathological, and immunophenotypical features of 9 new Mexican patients (M:F = 2:1; mean age, 14. Read More

    Chronic active Epstein-Barr virus infection with cutaneous and sinus lymphoproliferation in a white female patient with 25 years' follow-up: an original case report.
    Br J Dermatol 2015 Nov 8;173(5):1266-70. Epub 2015 Sep 8.
    Department of Dermatology, CHU Caen, Université de Caen Basse Normandie, 14000, Caen, France.
    Chronic active Epstein-Barr virus infection (CAEBV) is characterized by chronic infectious mononucleosis-like symptoms associated with very high viral load, as assessed by quantitative polymerase chain reaction. We present an unusual case in a French woman who was followed up over 25 years with cutaneous and sinus lymphoproliferation. This white woman presented with a long history of recurrent cutaneous necrotic papules of the skin, which started during childhood and healed spontaneously with depressed scars. Read More

    A patient with elderly-onset atypical hydroa vacciniforme with an indolent clinical course.
    Br J Dermatol 2015 Sep 22;173(3):801-5. Epub 2015 Jul 22.
    Department of Dermatology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, Okayama, Japan.
    Hydroa vacciniforme (HV) is a rare photodermatosis that mainly affects children and manifests as vesiculopapular eruptions in sun-exposed areas without systemic symptoms. HV-like lymphoma (HVLL) is one of the Epstein-Barr virus (EBV)-associated lymphoproliferative disorders (LPD) of childhood. Its diagnosis is based on monoclonal T-cell proliferation; however, its degree of malignancy is controversial owing to its variable prognosis. Read More

    Paediatric cutaneous lymphomas: a review and comparison with adult counterparts.
    J Eur Acad Dermatol Venereol 2015 Sep 25;29(9):1696-709. Epub 2015 Feb 25.
    Department of Dermatology, University Hospital Zürich, Zürich, Switzerland.
    Primary cutaneous lymphomas (CL) in children is rare. Only a few studies focused specifically on paediatric CL and therefore little is known whether primary CL in children are similar to or different from their adult counterparts with respect to the clinicopathological presentation, behaviour and prognosis. An extensive literature search using PubMed/MEDLINE from January 1995 through July 2014 was undertaken for articles reporting cases of paediatric CL. Read More

    Epstein-Barr virus: dermatologic associations and implications: part II. Associated lymphoproliferative disorders and solid tumors.
    J Am Acad Dermatol 2015 Jan;72(1):21-34; quiz 35-6
    Cooper Medical School of Rowan University, Camden, New Jersey; Perelman School of Medicine at the Hospital of the University of Pennsylvania, Philadelphia, Pennsylvania.
    Epstein-Barr virus (EBV) was the first human virus to be associated with oncogenesis. Over the past few decades, cumulative research has revealed that latent EBV infection may be implicated in the pathogenesis of a heterogeneous group of lymphoproliferative disorders and malignancies occurring in both immunocompetent and immunocompromised hosts. Many of these diseases have either primary or secondary cutaneous manifestations. Read More

    Epstein-Barr virus: dermatologic associations and implications: part I. Mucocutaneous manifestations of Epstein-Barr virus and nonmalignant disorders.
    J Am Acad Dermatol 2015 Jan;72(1):1-19; quiz 19-20
    Cooper Medical School of Rowan University, Camden, New Jersey; Perelman School of Medicine at the University of Pennsylvania, Philadelphia, Pennsylvania.
    Epstein-Barr virus (EBV) is a ubiquitous virus that has been implicated in a wide range of human diseases, many of which have mucocutaneous manifestations. As a member of the herpesviridae family, EBV causes lifelong infection by establishing latency in B lymphocytes. An intact immune response is critical in preventing progression of EBV disease, and the clinical manifestations of infection are dependent on the intricate relationship between virus and host immune system. Read More

    Antiviral treatment of a boy with EBV-associated hydroa vacciniforme.
    BMJ Case Rep 2014 Nov 24;2014. Epub 2014 Nov 24.
    Department of Dermatology and Allergy Centre, Odense University Hospital, Odense, Denmark.
    Hydroa vacciniforme is one of the rarest forms of photosensitivity disorders of the skin. Effective treatment options are scarce and mainly constitute of strict sun protection. Lately, hydroa vacciniforme has been associated with Epstein-Barr virus infection. Read More

    Systemic lymphoma arising from hydroa vacciniforme-like lymphoma: report of two cases with review of literature.
    Int J Clin Exp Pathol 2014 15;7(9):6403-8. Epub 2014 Aug 15.
    Department of Hematology, The First Affiliated Hospital of Nanjing Medical University, Jiangsu Province Hospital Nanjing 210029, China.
    Hydroa vacciniforme-like lymphoma (HVLL) is an extremely rare lymphoma described in children that occurs mainly in Asia and Latin American countries. It is an Epstein-Barr virus (EBV)-positive lymphoproliferative disease (LPD) characterized by a monoclonal proliferation of T or NK cells. In this study, we report the clinical and pathological features of two Chinese patients with HVLL showed T-cell phenotype expressing CD4. Read More

    Survival rates and prognostic factors of Epstein-Barr virus-associated hydroa vacciniforme and hypersensitivity to mosquito bites.
    Br J Dermatol 2015 Jan 10;172(1):56-63. Epub 2014 Dec 10.
    Department of Dermatology, Okayama University Graduate School of Medicine, Dentistry and Pharmaceutical Sciences, 2-5-1 Shikata-cho, Kita-ku, Okayama, 700-8558, Japan.
    Background: Epstein-Barr virus (EBV)-associated T/natural-killer lymphoproliferative disorders form a group of diseases that includes classical and systemic hydroa vacciniforme (HV) and hypersensitivity to mosquito bites (HMB). Patients with systemic HV (sHV) and HMB often have a poor prognosis, although little is known about the prognostic factors.

    Objectives: To elucidate the prognostic factors of HV and HMB. Read More

    Hydroa vacciniforme with eye involvement: report of two cases.
    Pediatr Dermatol 2015 Jan-Feb;32(1):e39-41. Epub 2014 Sep 10.
    Department of Dermatology, Razi Hospital, Tehran University of Medical Sciences, Tehran, Iran.
    We herein report two patients with hydroa vacciniforme and concomitant ophthalmic involvement, primarily in the form of keratoconjunctivitis leading to corneal opacity. Awareness of this occasional manifestation may help to prevent severe complications. Read More

    Clinicopathological characteristics of four cases of EBV positive T-cell lymphoproliferative disorders of childhood in China.
    Int J Clin Exp Pathol 2014 15;7(8):4991-9. Epub 2014 Jul 15.
    Department of Pathology, Cancer Institute and Hospital, Chinese Academy of Medical Sciences, Peking Union Medical College Beijing, 100021, China.
    A new category, "EBV positive T-cell lymphoproliferative disorders (LPD) of childhood", was proposed in the 2008 World Health Organization's (WHO) classifications of lymphoma. This series of lymphoproliferative disorders is rare. There are two major types of this series of disorders: systemic EBV positive T-cell LPD of childhood and hydroa vacciniforme-like lymphoma (HVLL). Read More

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