Holmes tremor (HT) is a rare movement disorder characterized by low-frequency tremor, inferior to 5 Hz, at rest and during postural and intentional conditions (1). It severely alters the functional abilities. The pathophysiology of tremor is unknown. However, dysfunctions of cortico-ponto-cerebello-thalamo-cortical loops were hypothesized by Bucy in 1944 and confirmed since notably in HT (2,3), involving contralateral sensorimotor and prefrontal cortices and the ipsilateral cerebellum. Treatments of HT include levodopa, dopamine agonists, anticholinergics, propranolol, primidone, levetiracetam, more recently botulinum toxin and deep brain stimulation, with variable and unpredictable responses (4). Hence we built a personalized rehabilitation program with intensive robotic rehabilitation, as proposed after stroke (5), associated to classical human-guided rehabilitation, in one case of HT. The lesions were scrutinized using structural magnetic resonance imaging (MRI). Both clinical effects and functional MRI (fMRI) activations were studied before and after rehabilitation, willing to explore underlying neural correlates.