The aim of this study was to determine the neuropsychologic development in patients with congenital (primary) hypothyroidism (CH) detected by screening and treated from early age. Seven studies were evaluated, including 675 CH patients and 570 controls. Criteria for inclusion of the studies were: 1) age of patients and controls > or = 5 y; 2) control group well defined in the original article. A meta-analysis of the data on the intellectual development was performed. All analyzed studies show a trend toward lower intelligence quotient (IQ) and--as far as investigated--poorer motor skills in CH patients compared with controls. Pooling of data showed a significant deficit of the mean IQ of 6.3 (95% confidence interval: 4.7-7.8). The most important independent risk factor for the eventual outcome appears to be the severity of CH (defined by initial thyroxine at the moment of diagnosis and by skeletal maturation); treatment variables do not seem to have an important effect the cognitive development. We conclude that CH, despite early detection and treatment, results in an IQ deficit; the severity of CH seems to be the most important individual risk factor. Our second conclusion is that, although biomedical risk factors are well investigated, the individual weight of these factors is yet largely unknown. This may be due to the variability of definitions and test tools, especially for the investigation of motor skills, that have been used.