Zolpidem improves dystonia in "Lubag" or X-linked dystonia-parkinsonism syndrome.

tion of movement disorders secondary to cervical disc prolapse with cord compression has not been previously highlighted. Our patient presented with upper limb choreathethosis and dystonia associated with tingling sensation of the fingers. However , there was no objective sensory loss. Neurophysiologic test-ings revealed right CTS. There was no electrophysiologic evidence of posterior column abnormality on somatosensory evoked potential examination. Her movement disorders were ascribed to disc prolapse with cord compression for the following reasons: first, the almost complete resolution of her movement disorders after surgery ; second, the movements were accompanied by tingling sensation in her fingers that may be caused by underlying cervical nerve root irritation or compression. Interestingly, her hand dys-tonia showed some characteristic dystonia features such as relief by certain " sensory tricks " and rest, and aggravated by action and stress and anxiety. A number of hypothesis underlying movement disorders in patients with cervical cord lesions have been proposed. 1-4 These include altered sensory input (in particular proprioceptive pathways), abnormal processing of both input and output signals in the spinal inter-neurones, and increased excitability of the spinal motor neurones. Disruption of the somatosensory pathways or motor cortex to the striatum also may produce abnormal movements without sensory loss. It is not clear why movement disorders secondary to cord pathologies are rarely observed. It could be that these disorders are under-recognized and hence under-reported. Despite the lack of clinical and electrophysiologic evidence of sensory abnormalities in our patient, we cannot rule out sub-clinical proprioceptive or cutaneous abnormalities resulting in aberrant sensory input as a possible mechanism. Although there were no structural basal ganglia abnormalities on MRI, we speculate that subclinical basal ganglia dysfunction contributing to altered input and output signals at cortical, subcorti-cal, or spinal levels also could be a predisposing factor. Functional brain imaging with surgical correlation may be useful to address this hypothesis. The effects of the hypnotic agent zolpidem on Lubag or X-linked dystonia-parkinsonism syndrome are described in this article. Lubag afflicts adult men from the Philippine island of Panay and is often resistant to therapy. 1 Zolpidem binds to gamma-aminobutyric acid–A (GABA A) receptors, particularly the omega-1 (␻ 1) subtype receptor. 2 Zolpidem was reported to improve parkin-sonism in some patients with PD or progressive supranuclear palsy (PSP) 3,4 ; its effects on dystonia are unknown. Case reports. Patient 1. A 41-year-old man with Lubag, presented at age 37 with head pulling …