We share our experience with stereotactic gamma knife thalamotomy (GKT) for medically refractory Holmes tremor (HT). A 22-year-old patient underwent gamma knife thalamotomy at ventrointermediate nucleus for disabling HT of the right upper limb. A single 4-mm isocenter was used to target the ventral intermediate nucleus with 130 Gy radiation. At 4 months follow up, we observed 84% improvement in his Fahn-Tolosa-Marin (FTM) rating scale with significant improvement in the right upper limb dystonic tremor. There was only subtle improvement in the ataxic component of the right lower limb. At 1 year after stereotactic GKT, there was sustained neurological improvement with no side effect, We present the stereotactic GKT as a treatment modality for drug-resistant HT. Moreover, it may be considered an alternate treatment modality especially in patients reluctant or contraindicated for any invasive surgical technique. CLINICAL TRIAL REGISTRATION NUMBER: Not required.