Unilateral acute maculopathy is a rare inflammatory macular disorder believed to be caused by viral infection, especially Coxsackievirus. It most commonly affects young healthy adults. This is a case report of unilateral acute maculopathy in a 28-year-old man with concurrent hand, foot and mouth disease. Although the typical acute manifestation of the disease is sudden, severe, unilateral central vision loss, most patients achieve full visual recovery over the course of several weeks without therapy.