Hydroa vacciniforme-like lymphoma: a chronic EBV+ lymphoproliferative disorder with risk to develop a systemic lymphoma

Blood. 2013 Oct 31;122(18):3101-10. doi: 10.1182/blood-2013-05-502203. Epub 2013 Aug 27.

Abstract

Hydroa vacciniforme-like lymphoma (HVLL) is an Epstein-Barr virus (EBV)-positive T-cell lymphoproliferative disorder of childhood that occurs mainly in Central and South America and Asia. We present the clinicopathological features of 20 Mexican children with HVLL with a median age of 8 years at diagnosis (range, 1-15). All patients presented with skin lesions involving sun-exposed areas, but not exclusively. Fever, lymphadenopathy, and hepatosplenomegaly were often observed. Most patients were treated with immunomodulators and/or immunosuppressive agents, resulting in temporary remission. For 13 patients follow-up was available for a median of 3 years (range, 1 month-13 years). Three patients with long follow-up (9-13 years) are alive with disease. Four patients died, 2 after developing systemic lymphoma. Histologically, the skin showed a predominantly angiocentric and periadnexal Epstein-Barr early RNA+ lymphoid infiltrate with variable atypia and subcutaneous involvement. Fifteen patients showed a T-cell phenotype (12, αβ; 2, γδ; 1, silent phenotype) and monoclonal T-cell receptor-γ rearrangements, whereas 6 exhibited a natural killer (NK)-cell phenotype. Four patients had hypersensitivity to mosquito bites. One patient showed both phenotypes. HVLL is an EBV-associated lymphoproliferative disorder of αβ-, γδ-, or NK-cell phenotype with a broad clinical spectrum, usually prolonged clinical course, and risk for progression to systemic disease.

Publication types

  • Research Support, Non-U.S. Gov't

MeSH terms

  • Adolescent
  • Child
  • Child, Preschool
  • Epstein-Barr Virus Infections / complications
  • Epstein-Barr Virus Infections / drug therapy
  • Epstein-Barr Virus Infections / pathology*
  • Female
  • Follow-Up Studies
  • Herpesvirus 4, Human / drug effects
  • Herpesvirus 4, Human / genetics
  • Herpesvirus 4, Human / metabolism
  • Humans
  • Hydroa Vacciniforme / complications
  • Hydroa Vacciniforme / drug therapy
  • Hydroa Vacciniforme / pathology*
  • Immunohistochemistry
  • Immunosuppressive Agents / therapeutic use
  • In Situ Hybridization
  • Infant
  • Lymphoma, T-Cell, Cutaneous / complications
  • Lymphoma, T-Cell, Cutaneous / drug therapy
  • Lymphoma, T-Cell, Cutaneous / pathology*
  • Lymphoproliferative Disorders / complications
  • Lymphoproliferative Disorders / drug therapy
  • Lymphoproliferative Disorders / pathology*
  • Male
  • Mexico
  • RNA, Viral / genetics
  • Receptors, Antigen, T-Cell / genetics
  • Steroids / therapeutic use
  • T-Lymphocytes / drug effects
  • T-Lymphocytes / metabolism
  • T-Lymphocytes / virology
  • Thalidomide / therapeutic use
  • Viral Proteins / metabolism

Substances

  • Immunosuppressive Agents
  • RNA, Viral
  • Receptors, Antigen, T-Cell
  • Steroids
  • Viral Proteins
  • Thalidomide