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Purpose To use new adaptive optics scanning laser ophthalmoscopy (AOSLO) technology to better image macular pathology in poppers maculopathy. Observations A 40-year-old patient was found to have poppers maculopathy. Best corrected visual acuity was decreased to 20/40 OD and 20/50 OS, spectral domain optical coherence tomography found outer retinal disruption of the fovea, and AOSLO imaging showed significant decrease in cone density of the fovea of both eyes. Conclusions and importance Poppers maculopathy is a rare, but visually significant, complication of popper abuse. AOSLO technology demonstrates significant cone damage in poppers maculopathy. The striking loss of cones revealed by AOSLO imaging shows how AOSLO imaging can elucidate macular pathology., Highlights • Poppers maculopathy can cause a significant decrease in BCVA despite relatively minimal changes observed on DFE and SD-OCT. • AOSLO imaging in this case of poppers maculopathy reveals a striking decrease in central cone density. • This report demonstrates that AOSLO can elucidate macular pathology not seen in more widely used retinal imaging modalities.