We report the initial and follow-up brain findings in a 42-year-old male patient with CNS involvement with African trypansomiasis. Initial MR imaging demonstrated diffuse hyperintensity in the basal ganglia bilaterally as well as involvement of the internal capsule, external capsule, and extreme capsule. Follow-up examination at 1 year revealed decreased signal intensity in the previously affected areas; however, ventricular enlargement indicative of atrophy was readily apparent.