<h4>Introduction</h4>We report the first documented case of bilateral neurotrophic keratitis potentially associated with Gilteritinib therapy.<h4>Case presentation</h4>A 52-year-old male patient presented with bilateral blurred vision and photophobia due to neurotrophic keratitis, which developed after two years of treatment with Gilteritinib - a second-generation tyrosine kinase inhibitor (TKI) used for relapsed or refractory acute myeloid leukemia (AML) with a confirmed FMS-like tyrosine kinase 3 (FLT3) mutation. The gene discussed is FLT3; the disease is acute myeloid leukemia.