PurposeRecognize a rare yet existing risk of severe visual loss as a postoperative complication of bilateral lung transplant.ObservationsA 62-year-old male had undergone bilateral lung transplant for end-stage idiopathic pulmonary fibrosis and emphysema overlap syndrome. The operation was initially off-pump; however, during the left lung transplantation, cardiopulmonary bypass conversion was necessary to maintain intraoperative hemodynamic stability. On post-operative day 4, shortly after extubation and full recovery from sedation, the patient reported bilateral no light perception vision. There were no other associated neurologic symptoms. A computed tomographic (CT) of the head, cranial magnetic resonance (MR) scan of the head, MR angiogram of the circle of Willis and neck were negative. Neuro-ophthalmologic examination revealed no light perception vision in both eyes(OU). The pupils were non-reactive to light (amaurotic pupils). The intraocular pressure measured 18 mm Hg OU, and complete bilateral ophthalmoplegia was present. The fundus exam showed bilateral pallid optic disc edema, cherry red spots, with arteriolar attenuation, and mildly dilated and tortuous veins. Stroke work up was negative.Conclusions and importanceA case of post-operative visual loss and ophthalmoplegia carrying significant and permanent quality of life implications. It questions the role disruption of homeostasis during cardiopulmonary bypass contributes for this outcome.