Dear Editor, Spastic dystonia is defined as tonic involuntary muscle activation at rest superimposed over spastic paresis [1]. It occurs in different pathological conditions, ranging from dopamine-dependent dystonia [2] to post-stroke deforming spastic hemiparesis [1]. Even though important burden is associated to spastic dystonia, therapeutic options are scarce and mostly limited to intramuscular botulinum toxin injection and surgical partial nerve section. Repetitive transcranial magnetic stimulation (rTMS) has been proposed as an interesting therapeutic option, but with inconsistent results [3]. We suggest that optimized targeting based on functional brain imaging could enhance the results of rTMS in schizencephaly-related dystonia and improve our knowledge about the technical procedure to become more widely applicable in neurodevelopmental disorders.Here we report the case of 41 years old woman suffering from openlip schizencephaly-related left hemiparesis with spastic dystonia. Schizencephaly, a disorder of cortical development, consists of a gray matter polymicrogyri-lined cleft of one cerebral hemisphere connecting extra-axial subarachnoid space and ventricle [4]. The patient was born after eight months pregnancy without reported complication. During childhood, congenital limb length discrepancy and equinus foot deformity were surgically corrected. A left spastic hemiparesis appeared in early childhood. It was associated to abnormal synkinesia that referred to involuntary movement of right hand and to a lesser extent of left foot that occurred with voluntary movement of left hand and vice versa (Supplementary Video 1). Neither spastic paresis nor other movement disorder was found in the right-side extremities. A painful dystonic gait pattern began on both left-side extremities around forty years of age and severely compromised the patient's autonomy. Its management by intramuscular botulinum toxin injections was painful and only partially effective, so her physician referred her for rTMS treatment trial. Based on a literature review, we hypothesized that reducing the excitability of primary motor cortex involved in motor control of left-side extremities would be effective in spastic dystonia treatment.First, we measured the motor evoked potentials (MEP) for left abductor pollicis brevis (APB) muscle in a relaxed state in order to determine motor threshold (MT). Single-pulse TMS was applied with a biphasic waveform using a figure-of-eight coil connected to a MagPro R30 MagOption stimulator (MagVenture, Denmark). The coil was positioned tangentially with the handle rotated posterior-laterally 45°to the sagittal plane to induce a posterior-anterior current flow in the brain. Motor responses were recorded using MEP Monitor (MagVenture, Denmark) and pre-gelled surface electrodes. It is noteworthy that no MEP was evoked in left APB when the motor cortex of the right hemisphere facing the schizencephaly cleft was stimulated, even when stimulation intensity was increased to 100% of maximal stimulator output (MSO)...