A patient with left thalamo-mesencephalic infarction presented with micrographia in the right hand as the only motor sign. Brain MRI and 99mTc ethyl cysteinate dimer (ECD) perfusion SPECT revealed ischemic lesions in the left midbrain and the anterior thalamus, but not in the basal ganglia, whereas [123I]-IPT SPECT demonstrated decreased activity of the [123I]-IPT in the left striatum. The patient's micrographia may be related to a dysfunctional nigrostriatal dopaminergic system secondary to ischemic damage to the substantia nigra.