Neurological complications are common after allogeneic hematopoietic stem cell transplantation (HSCT) but it is often difficult to define the cause among infections, Graft- versus-Host Disease (GvHD), adverse effects from medications or nervous system disease (for instance autoimmune disease). We describe a rare case of ocular GvHD associated with the reduction of tacrolimus in a female patient, three months after allogeneic HSCT for acute myeloblastic leukemia in second remission. The multidisciplinary investigations led to the diagnosis of retrobulbar optic neuritis associated with the expression of auto (allo)immune markers. Quickly after rising dosage of tacrolimus to therapeutic level, the ocular symptoms disappeared without relapse thereafter.