Sclerosing cholangitis and intracranial lymphoma in a child with classical Wiskott-Aldrich syndrome

Pediatr Blood Cancer. 2017 Jan;64(1):106-109. doi: 10.1002/pbc.26196. Epub 2016 Aug 27.

Abstract

Patients with Wiskott-Aldrich syndrome (WAS) are predisposed to malignancy and autoimmunity in addition to infections. We report a male child with WAS, who had presented with recurrent pneumonia, eczema, thrombocytopenia, autoimmune hemolytic anemia, and vasculitic skin lesions. Genetic analysis revealed a classical genotype WAS 155C>T; R41X. At 2 years of follow-up, he developed persistent headache and progressive hepatomegaly. Brain imaging showed a mass in the right frontal region, which on histopathology was shown to be high-grade non-Hodgkin lymphoma. Magnetic resonance cholangiopancreatography showed features of sclerosing cholangitis. This report extends the clinical spectrum and highlights unusual manifestations of sclerosing cholangitis and intracranial lymphoma in a patient with WAS.

Keywords: CNS lymphoma; Wiskott-Aldrich syndrome; primary sclerosing cholangitis.

Publication types

  • Case Reports

MeSH terms

  • Adolescent
  • Anemia, Hemolytic, Autoimmune / etiology
  • Anemia, Hemolytic, Autoimmune / pathology*
  • Brain Neoplasms / etiology
  • Brain Neoplasms / pathology*
  • Cholangitis, Sclerosing / etiology
  • Cholangitis, Sclerosing / pathology*
  • Humans
  • Lymphoma, Non-Hodgkin / etiology
  • Lymphoma, Non-Hodgkin / pathology*
  • Male
  • Mutation / genetics
  • Prognosis
  • Thrombocytopenia / etiology
  • Thrombocytopenia / pathology*
  • Wiskott-Aldrich Syndrome / complications*
  • Wiskott-Aldrich Syndrome / pathology
  • Wiskott-Aldrich Syndrome Protein / genetics

Substances

  • Wiskott-Aldrich Syndrome Protein