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To report a case of a branch retinal vein occlusion secondary to a retinal arteriolar macroaneurysm (RAM).Retrospective case report describing examination findings, treatment outcome and unique multimodal imaging features demonstrated on fluorescein angiography, optical coherence tomography, optical coherence tomography angiography and adaptive optics photography of the retinal vessels and RAM.A 61-year-old man presented with 20/200 vision in the right eye because of a branch retinal vein occlusion secondary to a RAM. After sector panretinal photocoagulation and a course of 24 intravitreal antivascular endothelial growth factor injections over 4 years, visual acuity improved to 20/25. Fluorescein angiography showed filling of the RAM even after 4 years. Optical coherence tomography angiography demonstrated venous collateral vessels in both the superficial and deep capillary plexuses, and adaptive optics imaging revealed a gap between the RAM wall and occluded vein.Multimodal imaging of this unusual presentation illustrated a novel mechanism of branch retinal vein occlusion in which a primary RAM adjacent to the junction of two retinal veins led to obstruction of venous flow without evidence of direct compression. This supports the theory that perianeurysmal microenvironment changes may be of importance in the pathogenesis of venous occlusion.