Purpose: To present a unique and unusual case of Paracentral Acute Middle Maculopathy (PAMM) with associated cystoid macular oedema (CMO) following ocular surface surgery in a low risk patient. Observations: This observational case report describes the presentation of a single case of Paracentral Acute Middle Maculopathy following pterygium surgery. The patient harboured no risk factors and underwent uncomplicated pterygium surgery with no perioperative complications. Post-operatively the patient demonstrated marked visual acuity loss to count fingers only to the operated eye. Optical Coherence Tomography demonstrated hyper-reflective band-like lesions affecting the middle layers of the retina, pathognomonic for PAMM. Unusually, cystoid macular oedema was also evident. He was managed on topical and oral therapy, with limited visual recovery. Conclusions and importance: We report a rare and unique case of PAMM with associated cystoid macular oedema occurring in a patient deemed low risk following pterygium surgery resulting in significant permanent visual loss. To our knowledge, this is the first reported incidence of PAMM following ocular surface surgery.