Rationale: This case report describes an unusual presentation of recurrent Wernicke encephalopathy (WE) in a patient undergoing hemodialysis, with distinctive neuroimaging findings that provide new insights into the disease. Patient concerns: The patient presented with progressive gait ataxia, dysarthria, and mild memory impairment, with recurrent neurological symptoms following thiamin discontinuation. Diagnoses: WE was diagnosed based on characteristic MRI findings including the rare lentiform fork sign and other atypical brain lesions, supported by symptom recurrence after thiamin cessation and radiological reversibility with treatment. Interventions: The patient received immediate thiamin supplementation therapy, close neurological monitoring, and regular hemodialysis. Outcomes: Neurological symptoms significantly improved with treatment, and MRI showed reversal of the lentiform fork sign and other atypical lesions, demonstrating the importance of sustained thiamin management during follow-up. Lessons: This case highlights the lentiform fork sign as a valuable neuroimaging marker for WE, suggests that blood–brain barrier dysfunction may be pathogenic, and emphasizes the need for continuous thiamin management in high-risk patients to prevent recurrence.
Liang et al. (2026) studied this question.