Introduction: Tacrolimus induced optic neuropathy (TION) is a rare condition seen in transplant patients leading to severe vision loss caused by damage to the optic pathway. The underlying pathophysiology is thought to be a combination of ischemic damage due to vasoconstriction of the cerebral microvasculature and direct neurotoxicity.

Method: We describe a case of a 51-year old male, combined multivisceral and renal transplant recipient who developed severe, bilateral TION 3 years after transplantation. Furthermore, a literature research was performed for all published cases describing TION after organ transplantation.

Results: Optic tract inflammation was clearly detected on MRI (Figure). Treatment with intravenous corticosteroids and immunoglobulins was started. Tacrolimus was reduced but not withdrawn completely to avoid rejection, especially of the intestinal component of the graft. Everolimus was associated to maintain sufficient immunosuppression. After three months, vision had recovered completely. The patient experienced no signs of rejection in any transplanted organ during this period and organ function remained stable. Seven other reports in various organs were found in literature (Table). In most, tacrolimus was discontinued completely and outcomes were poor.

Conclusion: Our report demonstrates the importance of swift treatment to reverse optic tract inflammation and highlights the possibility to add everolimus to the immunosuppressive regimen to allow safe reduction of tacrolimus exposure in intestinal transplant patients. By contrast, results from literature show sporadic use of anti-inflammatory medication and poor long-term vision outcomes, often related to delayed diagnosis and treatment (Table).