Purpose: To report a case of visual loss immediately after hand, foot, and mouth disease and demonstrate the high-resolution optical coherence tomography findings. Methods: A retrospective case report of a 19-year-old nursery worker with resolving hand, foot, and mouth disease and acute unilateral visual loss. Results: The clinical features were characteristic of unilateral acute idiopathic maculopathy. High-resolution optical coherence tomography demonstrated highly reflective subretinal material at the macula of one eye with disruption of the photoreceptor inner segment/outer segment junction. Vision remained poor for 4 weeks when there was rapid recovery coinciding with reconstitution of the inner segment/outer segment junction on optical coherence tomography. Conclusion: Unilateral acute idiopathic maculopathy may be caused by Coxsackievirus infection. Optical coherence tomography and clinical findings suggest an acute viral retinal pigment epitheliitis to be the main pathologic feature.