Introduction: Many clinical manifestations can be related to Tropheryma whipplei infection.
Case report: We report a Tropheryma whipplei limbic encephalitis developed as a relapse of classical Whipple's disease.
Discussion: This case is to the best of our knowledge the first proof of the effective brain-blood barrier crossing of both doxycycline and hydroxychloroquine as demonstrated by direct concentration monitoring on brain biopsy.
Keywords: Emerging infectious disease; Limbic encephalitis; Tropheryma whipplei; Whipple’s disease.