BackgroundNo clinical trials have been reported on the use of focused ultrasound (FUS) for treating cervical dystonia.ObjectiveWe aimed to confirm the efficacy and safety of FUS pallidothalamic tractotomy for cervical dystonia.MethodsThis was a prospective, open‐label, non‐controlled pilot study. The primary outcome was defined as a change in the score for the Toronto Western Spasmodic Torticollis Rating Scale (TWSTRS) from baseline to 6 months after FUS pallidothalamic tractotomy. The secondary outcomes included a change in the neck scale for the Burke‐Fahn‐Marsden Dystonia Rating Scale (BFMDRS), mood scales including Beck Depression Inventory (BDI), Beck Anxiety Inventory (BAI), Apathy Evaluating Scale (AES), and adverse events. Patients were assessed for TWSTRS, BFMDRS, and adverse events at baseline, 1 week, 1 month, 3 months, and 6 months after treatment. BDI, BAI, and AES were assessed at baseline and 6 months after treatment.ResultsTen patients were enrolled in this study. The mean age of onset of dystonia was 51.6 ± 10.2 years. The TWSTRS at 6 months (29.9 ± 16.0, range: 3–55) was significantly improved by 43.4% (P < 0.001) from baseline. The BFMDRS‐Neck scales at 6 months (4.2 ± 2.8) were significantly improved by 38.2% (P < 0.001) from baseline. The BDI, BAI, and AES at 6 months were improved by 23.2%, 10.9%, and 30.3%, respectively from baseline. Reduced hand dexterity in three patients and weight gain in two patients were confirmed at the last evaluation.ConclusionThis study suggests that FUS pallidothalamic tractotomy may be an effective treatment option for patients with cervical dystonia. © 2024 International Parkinson and Movement Disorder Society.