Anosognosia for hemiplegia is the denial of the contralesional motor deficits that may follow brain damage. Although this disturbance has been reported in the neurological literature since the beginning of the last century, only few longitudinal studies have addressed the issue of the anatomical substrate of the disorder. Here we present a comprehensive review of the literature on anosognosia for hemiplegia from 1938 to 2001, taking into account some of its clinical, epidemiological and anatomical aspects. In particular, an attempt has been made to identify the intra-hemispheric lesion locations most frequently associated to the denial behaviour. Our review shows that anosognosia for hemiplegia most frequently occurs in association to unilateral right-sided or bilateral lesions of different brain areas (cortical and/or subcortical). It seems to be equally frequent when the damage is confined to frontal, parietal or temporal cortical structures, and may also emerge as a consequence of subcortical lesions. Interestingly, the probability of occurrence of anosognosia is highest when the lesion involves parietal and frontal structures in combination, if compared to other combinations of lesioned areas. This pattern of lesions suggests the existence of a complex cortico-subcortical circuit underlying awareness of motor acts that, if damaged, can give raise to the anosognosic symptoms.