We report a young patient who presented with nonarteritic anterior ischaemic optic neuropathy associated with the sticky platelet syndrome (SPS, a congenital prothrombotic disorder). A 15-year-old boy presented with a sudden, painless onset of a right inferior altitudinal visual field defect and marked optic disc oedema. Cranial magnetic resonance imaging (including orbits), orbital ultrasound and haematological investigations including a routine hypercoagulability work-up were negative. Further evaluation showed a positive assay for SPS which was confirmed on repeat testing. The SPS (characterized by platelet hyperaggregability and vascular occlusion) may be a potential thrombophilic risk factor for nonarteritic anterior ischaemic optic neuropathy.