Background: Acute macular neuroretinopathy is a rare disease that with the help of multimodal imaging is being diagnosed more frequently.
Methods: An atypical case is presented and followed by multimodal imaging.
Results: A typical acute macular neuroretinopathy lesion was seen on biomicroscopic examination and spectral domain optical coherence tomography examination. However, near-infrared imaging revealed a lack of the characteristic finding along with a relative afferent pupillary defect and a widespread depressed multifocal electroretinogram which has not been reported previously.
Conclusion: This patient exhibits photoreceptor and outer retinal findings commonly described in acute macular neuroretinopathy lesion without classic near-infrared findings. This case may represent a severe form of acute macular neuroretinopathy.