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Holmes tremor is a rare movement phenomenon, with atypical low-frequency tremor at rest and when changing postures, often related to brainstem pathology. We report a 70-year-old female patient who was presented with dystonic head and upper limb tremor after brainstem hemorrhage. The patient had experienced a sudden onset of left hemiparesis and right facial paralysis. Brain magnetic resonance imaging showed an acute hemorrhage from the brachium pontis through the dorsal midbrain on the right side. Several months later, the patient developed resting tremor of the head and left arm, which was exacerbated by a sitting posture and intentional movement. The tremor showed a regular low-frequency (1–2 Hz) for the bilateral sternocleidomastoid and cervical paraspinal muscles at rest. The patient’s symptoms did not respond to propranolol or clonazepam, but gradually improved with levodopa administration. Although various remedies were attempted, overall, the results were poor. We suggest that levodopa might be a useful remedy for Holmes tremor. The curative or relieving effect of the dopaminergic agent in Holmes tremor needs more research.

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