Calcineurin inhibitors (CNIs) are regarded as a corner stone in immunosuppressive therapy after solid organ transplantation. However, neurotoxicity is a common side effect of CNIs, resulting in a wide range of neurological symptoms such as headache, tremor and seizures. In this case report, we describe a patient who developed severe motor and sensory neuron dysfunction related to CNIs after bilateral lung transplantation, which resolved after halting CNI and switching to a mammalian Target of Rapamycin-inhibitor.