To report the first case of centromedian nucleus (CMN) responsive neurostimulation (RNS) in the treatment of adult-onset Rasmussen’s Encephalitis (RE).

RE is a neuroinflammatory disorder characterized by uni-hemispheric atrophy, drug-resistant epilepsy, and progressive hemiplegia with cognitive deterioration. Typically, a disease of childhood, adult-onset RE is rare and treatment is limited. Hemispherotomy offers seizure control but risks functional impairment. Immunotherapy is rarely effective. Although RNS have been used to treat seizures in RE, cases are highly selective to less than 2 epileptogenic foci. The use of RNS implants to the CMN have not been explored.

We describe a 26-year-old male with medically refractory multifocal epilepsy. Semiologies include right-sided clonic movements, which can progress to bilateral tonic-clonic seizures. Since seizure onset at age 19, the frequency increased to several daily along with gradual right-sided hemiparesis and spasticity. Serial MRI Brains revealed worsening left frontal T2 hyperintensities and frontoparietal atrophy. Autoimmune cerebrospinal fluid studies were unremarkable. Brain biopsy showed gliosis, though it was obtained likely during the remote stage of disease. He failed seven anti-seizure medications and immunomodulatory therapies. Pre-surgical evaluation with stereo-electroencephalography (sEEG) recorded at least 5 seizure foci in the left frontal lobe. Deep brain stimulation of the CMN was considered but denied by insurance. Thus, RNS of the CMN was placed. Over two years, the device parameters were adjusted in four sessions, at three, four, eight months and current, have all been clinically seizure-free, respectively.