Shinkai A, et al. - Since the link between anti-estrogen drugs and macular telangiectasia type 2 (MacTel-2) remains unknown, researchers reported a case with anti-estrogen maculopathy resembling MacTel-2 with improved visual function and macular morphology following cessation of anti-estrogen drugs. A 53-year-old woman in both eyes presented a 5-month history of central loss of vision and anorthopia. She had been given 69 months of oral tamoxifen followed by toremifene. In this case, visual acuity, macular morphology, and retinal pigment epithelium (RPE) impairment improved after cessation of anti-estrogen drugs, indicating the association between retinal toxicity of anti-estrogen drugs and the development of MacTel-2-like findings. The toxicity of both photoreceptor and RPE cells caused by anti-estrogen drugs will contribute to the development of anti-estrogen maculopathy similar to MacTel-2 from these findings and previous observations.