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• This systematic review evaluates data obtained from Brazilian congenital toxoplasmosis studies. • Children born from treated mothers have better outcomes than those born from untreated mothers, thus, gestational treatment is a watershed • Congenital toxoplasmosis is diagnosed late in Brazil, despite existing government guidelines. Congenital toxoplasmosis is a zoonosis caused by the intracellular Apicomplexa protozoan Toxoplasma gondii. This infection causes subclinical or clinical lesions, such as retinochoroiditis and central nervous system lesions. The severity of fetal infection is related to the stage of pregnancy and the efficacy of the gestational treatment on fetal infection, whether it is achieved, or if it starts early. South America is the region with the highest burden of congenital toxoplasmosis and the most pathogenic genotypes. Here, we present the results of a comprehensive systematic review and meta-analysis of the congenital toxoplasmosis in Brazil. PubMed, Web of Science, and CAPES databases were used to search for relevant studies that were published between 1 January 2007 and 31 December 2018. The final searching process yielded 21 papers. The studies accounted for 469 children with congenital toxoplasmosis. Of these, 269 (57%) had a diagnosis in the postnatal period. Concerning mothers, 209 (44.6%) underwent prenatal care, but 47 (22.5%) did not receive any drug for toxoplasmosis treatment. There were 226 (48.2%) children with retinochoroiditis; 83 (17.7%) with brain calcifications; 9 (1.9%) with neurosensory auditory dysfunction; and 2 (0.42%) with human immunodeficiency virus coinfection. A total of 460 (98%) children had a medical and multidisciplinary follow-up for at least one year and the most frequent genotype was #11(BRII), found in seven children. There was a statistical correlation between the mother's treatment and asymptomatic children. The gestational treatment seems to protects the fetus since children of mothers who received anti-T. gondii medications have a better prognosis. The retinochoroiditis was the main finding among children, followed by brain calcifications. [ABSTRACT FROM AUTHOR]