Facial manifestations of Epstein-Barr virus-related lymphoproliferative disease in childhood acute lymphoblastic leukemia in remission: Two atypical presentations

Pediatr Blood Cancer. 2016 Nov;63(11):2042-5. doi: 10.1002/pbc.26102. Epub 2016 Jul 8.

Abstract

Epstein-Barr virus-related lymphoproliferative disease (EBV-LPD) rarely occurs in patients with acute lymphoblastic leukemia (ALL), who have not received hematopoietic transplantation. We describe EBV-LPD manifesting as facial lesions in two children with ALL in remission. One patient was a 16-year-old male with T-cell ALL with an EBV-positive angiocentric polymorphous lip lesion presenting as right-sided facial swelling. The other patient was a 12-year-old male with B-cell ALL with an EBV-positive polymorphous lymphoplasmacytic infiltrate presenting as bilateral dacryoadenitis. Neither patient had known primary immunodeficiencies. Both cases improved with immunosuppressant de-escalation. These cases suggest that immunosuppression induced by maintenance chemotherapy is sufficient to promote EBV-LPD.

Keywords: Epstein-Barr virus; acute lymphoblastic leukemia; lymphoproliferative disease; maintenance chemotherapy.

Publication types

  • Case Reports

MeSH terms

  • Adolescent
  • Child
  • Dacryocystitis / etiology*
  • Epstein-Barr Virus Infections / complications*
  • Humans
  • Lymphoproliferative Disorders / complications*
  • Male
  • Precursor Cell Lymphoblastic Leukemia-Lymphoma / complications*