Novel de novo TREX1 mutation in a patient with retinal vasculopathy with cerebral leukoencephalopathy and systemic manifestations mimicking demyelinating disease

Gabrielle Macaron, Jean Khoury, Rula A. Hajj-Ali, Richard A. Prayson, Sunil Srivastava, Justis P. Ehlers, Hafsa Mamsa, M. Kathryn Liszewski, Joanna C. Jen, Robert A. Bermel, Daniel Ontaneda

Research output: Contribution to journalLetterpeer-review

4 Scopus citations

Abstract

Retinal vasculopathy with cerebral leukoencephalopathy and systemic manifestations (RVCL-S) is a rare fatal autosomal dominant vasculopathy associated with mutations in the TREX1 gene. Only one de novo case has been reported in the literature. We report the long-term clinical, radiological, and pathological presentation of a patient with a de novo and novel mutation in this gene. Description of the clinical, genetic, imaging and pathologic characteristics is important to better characterize RVCL-S and prevent unnecessary interventions. RVCL-S should be considered in patients with tumefactive brain lesions unresponsive to immunotherapy.

Original languageEnglish
Article number103015
JournalMultiple Sclerosis and Related Disorders
Volume52
DOIs
StatePublished - Jul 2021

Keywords

  • Leukoencephalopathy
  • Novel
  • Retinal vasculopathy
  • TREX1
  • de novo

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