In view of the scarcity in number of the reports on clinical cases of torsion spasm, the authors believe that the case presented here will be of some interest. This patient was a 15-year old boy who had had, since early part of his twelfth year, the episodes of the torsion on the anterior backbone accompanied by that peculiar twisting and turning along with cramping of muscles, which alternately increased or decreased, facial expression blank and entire movement simple and sluggish, without any apparent causative factors. He had been clinically diagnosed as having torsion spasm reactions, but he soon died of somatic weakening. The autopsy findings of this case were the destruction of the lense nucleus, the caudate nucleus, the thalamus, the dentate nucleus, and a marked proliferation of neuroglia, especially, the appearance of giant glia as well as liver cirrhosis; and it was diagnosed as Wilson-pseudosclerosis. Thereupon, the authors have attempted to present their opinions on this particular case here.