Congenital strabismus affects 3% of world population. Millions of persons suffer this condition, but still its origin or the reasons why not all patients respond to the traditional treatment are unknown.

Until very recently, it was believed that congenital strabismus had no relation to cortical alterations; therefore, neuroimaging studies were only required when strabismus was present in premature infants or when brain damage was suspected. A preliminary study on strabismal patients in 1968 provided some insight into the incidence of the different presentations of strabismus in our institution, as well as the correlation among the various clinical signs. Based on this experience we decided to enlarge our sample. Using conventional EEG and digitized brain mapping (DBM) methods, we analyzed 195 young patients with clinical diagnosis of congenital strabismus–111 females (56.92%) and 84 males (43.08%); the age range was from 2 to 14 years. The DBM approach was done in real time. Given its low cost, security and availability, DBM turned to be a useful tool to evince some alterations in cerebral cortex related to congenital strabismus, especially dissociated strabismus. We also employed complementary neuroimaging methods for research purposes. From 195 DBM images, 56.4% exhibited various neuroelectric alterations, whereas 43.6% were considered normal. Abnormal DBM were more frequent in the dissociated strabismus group (64.95%) than in non-dissociated strabismus patients (42.6%); the rate of altered DBM images was higher in horizontal dissociated deviation cases (73.3%). Based on these findings, we recommend the use of DBM in patients with dissociated strabismus, and in some cases the treatment must go beyond surgery and glasses. Some of our patients were subjected to different neuroimaging methods, such as single Photon emission tomography (SPECT), magnetic resonance imaging (MRI), granulometry, and proton nuclear magnetic resonance spectroscopy (1H NMRS) with the aim of correlating this data and gain further understanding on the origin of congenital strabismus, particularly dissociated strabismus cases. This chapter addresses aspects of congenital strabismus, as well as some of its cortical implications–neuroelectric, neurometabolic and morphometric. The illustrations are meant to make this interesting and scarcely-explored topic more accessible.