Purpose: To report a case of peripapillary pachychoroid syndrome showing spontaneous anatomic improvement after initiation of hemodialysis and to propose a possible systemic mechanism. Methods: A single case was reviewed. Results: A 67-year-old man with diabetic nephropathy and vascular nephrosclerosis presented with peripapillary pachychoroid syndrome characterized by peripapillary choroidal thickening with associated subretinal and intraretinal fluid accumulation. After initiation of hemodialysis, the retinal fluid resolved spontaneously, accompanied by a reduction in choroidal thickness, without any ocular treatment. Conclusions: Initiation of hemodialysis may reduce systemic fluid volume and venous pressure, leading to decompression of the choroidal venous system, decreased choroidal hyperpermeability, and resolution of retinal fluid. To our knowledge, this is the first reported case of peripapillary pachychoroid syndrome showing anatomic improvement after dialysis, highlighting the potential role of systemic venous hemodynamics in pachychoroid disease activity.