A man in his mid-40s who had been recently started on alirocumab (a human monoclonal antibody which inhibits proprotein convertase subtilisin/kexin type 9) due to his strong familial cardiovascular risk and refractory hypercholesterolaemia presented with a few-hour history of acute-onset left-sided blurred vision. The best-corrected visual acuities were 6/6 bilaterally and slit-lamp examination was normal. However, optical coherence tomography revealed serous subretinal fluid in the left macula. Optos ultra-widefield retinal imaging and fundus autofluorescence, along with a set of blood tests, did not reveal any alternative causes. A diagnosis of alirocumab-associated uveitis was diagnosed. Alirocumab was stopped and he was followed up in uveitis clinic. Within 4 months following alirocumab cessation, the subretinal fluid resolved completely. This case report emphasises the importance of early multidisciplinary team involvement, since novel therapeutic agents can have unexpected adverse events.