Objective: Horner's syndrome (HS) is a rare complication of high-intensity focused ultrasound (HIFU) and radiofrequency ablation for thyroid nodules. We present such a case and discuss how to avoid this complication in the future. Methods: This case occurred during HIFU treatment of a benign thyroid nodule (BTN). Ultrasound and fine-needle aspiration cytology (FNAC) were performed before the procedure. Volume reduction was evaluated at 6 weeks, 3, 6, and 12 months. Technical success was !50% reduction at 6 months. Results: A 30-year-old woman presented with a solitary symptomatic thyroid nodule. Her thyroid stimulating hormone was 1.16 (ref 0.4-3.6) mUI/mL), ultrasound found a 13 mL right-thyroid EU-TIRADS 4 nodule. Two FNACs were read as Bethesda II. The subsequent HIFU procedure was conducted with local 2% lidocaine anesthesia. The procedure was painful (visual analogic scale 10/10) and ipsilateral partial ptosis occurred during the procedure. Volume reduction at 12 months was 34.6% of the initial volume with persisting functional and cosmetic complaints, discomfort, and partial ptosis. As the volume reduction was 50%, the procedure was a technical failure. A new FNAC was read as Bethesda IV. A right lobectomy was performed without postoperative outcomes and without requiring hormonal replacement therapy. Pathological evaluation found no malignant cells. Conclusion: HS is a rare complication of HIFU for management of BTNs. It may be symptomatic and have sequalae that persist for months. Severe neck pain may by associated, but further investigation is needed.