The aims of this study were to examine abnormalities in brain structure in adolescents and young adults with very low birth weight (VLBW, <1,500 g) and associations of these abnormalities with neuropsychological outcomes. The sample of 108 participants from 14 to 19 years of age included 37 participants with <750 g birth weight, 35 with 750-1,499 g birth weight, and 36 normal birth weight (NBW) controls. One or both of the VLBW groups had smaller brain volumes, larger lateral ventricles, and a small surface area of the corpus callosum than the NBW controls. Group differences in white matter (WM) structures, subcortical gray matter (GM), and the cerebellum were found even when controlling for whole brain volume (WBV), and were most pronounced in the <750 g group. WM reductions in the two VLBW groups relative to NBW controls were associated with more pervasive cognitive deficits than were reductions in subcortical GM. Associations of cognitive outcomes with structural abnormalities remained when controlling for WBV or neonatal risks. The results are consistent with previous findings of residual brain abnormalities in adolescents and young adults with VLBW and provide new information on their cognitive correlates.