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Rubral Tremors Associated with an Inferior Olivary Lesion that Developed after a Brainstem Hemorrhage

Rubral (Holmes’ or midbrain) tremor caused by a dentate projection pathway is a rare combination of 2 to 5 Hz rest, enhanced by posture and kinetic tremors of an upper extremity. Symptomatic palatal tremor is a defined movement disorder characterized as a 1-3 Hz palatal myoclonus that appears after a disruption of the Guillain-Mollaret triangle, which includes the ipsilateral red nucleus, the ipsilateral inferior olivary nucleus (ION), and the contralateral dentate nucleus. Although palatal tremor remains the only dyskinesia that is consistently related to hypertrophic olivary degeneration (HOD), some uncommon dyskinesias may occur with HOD. Both of these tremors can sometimes occur after a brainstem lesion. In this study, a 66-year-old female patient underwent a ventriculo-peritoneal shunt for hydrocephalus after a coiling ruptured paraclinoid aneurysm of the right side. At the time, the right brainstem and cerebellar hemorrhagic infarction had been identified. In a follow-up imaging, hemorrhagic infarction had improved, but the patient gradually developed a rubral tremor involving both the left upper and lower extremities, besides the head and tongue tremors. Rubral tremor occurred about three months after bleeding, and the head and tongue tremors about five months. Usually, rubral tremor is in the arm, but in this case symptoms appeared even in the legs, accompanied by head and tongue tremors. The literature and imaging findings of this uncommon condition are reviewed.