Objective　To evaluate the accuracy and value of the ultrasonography and magnetic resonance imaging (MRI) in the diagnosis of agenesis of the corpus callosum in the newborns. Methods　Conventional cerebral ultrasound screening was performed in 8086 newborns admitted to NICU in our hospital from January 2012 to June 2014 and agenesis of the corpus callosum was suspected or confirmed in 31 newborns. The diagnostic accuracy of ultrasonography was assessed through the comparison between the results of ultrasonography and MRI. Results　In 31 cases with suspected agenesis of the corpus callosum, ultrasonography showed 14 cases of complete agenesis of which 13 cases had the same diagnosis with MRI except one case of partial agenesis, meanwhile, ultrasonography showed 16 cases of partial agenesis of which 15 cases had the same diagnosis with MRI except one case of complete agenesis. One case of abnormal corpus callosum determined by ultrasonography was diagnosed as partial agenesis by MRI. MRI showed there were other brain malformations in 14 cases. Conclusions　Ultrasonography and MRI has a high consistency in the diagnosis of agenesis of the corpus callosum in neonates, so the former can be used as a routine screening and the latter can be used as a method of accurate diagnosis. A combination of both has an important role in early diagnosis and clinical evaluation.