Background Facial rejuvenation and reconstruction with autologous fat injection are a common and effective procedure used worldwide. Most surgeons and patients are satisfied with the favorable outcomes. However, catastrophic complications from arterial and venous occlusion resulting in visual loss and stroke may occur. Case presentation We herein report a case of isolated venous occlusion from fat embolism. The patient developed acute painful proptosis and blurred vision of her right eye while undergoing an esthetic autologous fat injection into her forehead. Based on her clinical manifestations and radiologic findings, the patient was diagnosed with superior ophthalmic vein occlusion. Symptomatic and supportive treatments were given. Spontaneous clinical improvement occurred without secondary complications. Therefore, the initially planned endovascular therapy with transfemoral transvenous embolectomy of the right superior ophthalmic vein was canceled. Conclusions Facial augmentation with autologous fat injection can cause superior ophthalmic vein embolism. Surgeons should therefore perform this procedure very cautiously. Prompt ophthalmological evaluation and proper management are important for improving clinical outcomes. Level of Evidence V This journal requires that authors assign a level of evidence to each article. For a full description of these Evidence-Based Medicine ratings, please refer to the Table of Contents or the online Instructions to Authors www.springer.com/00266.