Leukoencephalopathy with stroke-like presentation (LEPS) is a rare complication of various chemotherapeutic agents, and it is associated with specific findings on diffusion-weighted MRI (DW-MRI). Herein, we report a case of LEPS in a patient treated with 5-fluorouracil (5-FU) who was found to be deficient in dihydropyrimidine dehydrogenase (DPD). • While the clinical syndrome of LEPS is well known, only limited DW-MRI data in LEPS after 5-FU exposure is presently available. The histopathological basis for these DW-MRI abnormalities may consist of vacuolation secondary to splitting of the myelin intraperiod line. • Early recognition of LEPS in 5-FU recipients is important to prevent irreversible neurologic damage from prolonged exposure. A comprehensive neuroimaging evaluation including DW-MRI is recommended to spare unnecessary and inappropriate invasive procedures or therapies.
- DPD deficiency
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