Hepatosplenic αβ T-Cell Lymphoma as Second Malignancy in Young Adult Patient With Previously Undiagnosed Ataxia-Telangiectasia

J Pediatr Hematol Oncol. 2020 Aug;42(6):e463-e465. doi: 10.1097/MPH.0000000000001537.

Abstract

Ataxia-telangiectasia is a rare autosomal recessive neurodegenerative disease characterized by ataxia, radiosensitivity, telangiectases, and increased risk for hematologic malignancies. We present a case of a female individual diagnosed with T-cell acute lymphocytic leukemia at 13 years and subsequently with αβ subtype of hepatosplenic T-cell lymphoma (HSTCL) at 20 years. During her diagnostic work up for HSTCL, paired tumor-germline sequencing identified a diagnosis of ataxia-telangiectasia. We also describe a very refractory clinical course of her αβ HSTCL, including only a brief response to multiagent chemotherapy and an allogenic bone marrow transplant.

Publication types

  • Case Reports
  • Research Support, N.I.H., Extramural

MeSH terms

  • Adolescent
  • Adult
  • Antineoplastic Combined Chemotherapy Protocols / therapeutic use
  • Ataxia Telangiectasia / complications*
  • Female
  • Humans
  • Liver Neoplasms / drug therapy
  • Liver Neoplasms / etiology
  • Liver Neoplasms / pathology*
  • Lymphoma, T-Cell / drug therapy
  • Lymphoma, T-Cell / etiology
  • Lymphoma, T-Cell / pathology*
  • Neoplasms, Second Primary / drug therapy
  • Neoplasms, Second Primary / etiology
  • Neoplasms, Second Primary / pathology*
  • Prognosis
  • Receptors, Antigen, T-Cell, alpha-beta / immunology*
  • Splenic Neoplasms / drug therapy
  • Splenic Neoplasms / etiology
  • Splenic Neoplasms / pathology*
  • Young Adult

Substances

  • Receptors, Antigen, T-Cell, alpha-beta