Schizophrenia has been conceptualized to be a neurodevelopmental disorder. Neuroimaging evidence was generally findings of volumetric reductions in various brain structures. The shape analysis of the insula can uncover unique structural deformity in the neurodevelopmental disorder, which cannot be revealed from a simple volume measurement. The objective of this study was to demonstrate a subtle change of the insula in schizophrenia using our special shape analysis technique. Subjects were 23 patients with schizophrenia and 23 normal healthy subjects. A landmark-based structural and surface shape analysis of the insula was performed using high-spatial resolution magnetic resonance imaging. A characteristic finding was that the frontotemporal sides of the right insula were deformed in the patients with schizophrenia compared with normal controls. This deformation can be associated with abnormal development of the frontal and temporal lobes in schizophrenia.