Objective: The EPISTOP project is a multi-center prospective European study tracking epileptogenesis, epilepsy and neurodevelopment in infants with Tuberous TSC. Here we report the preliminary findings of serial neuropsychological evaluations in the first 24 months of life Methods: Between 2013 and 2016, 101 TSC infants younger than 5 months were enrolled; all were seizure-free and on no anti-epileptic drugs. Neuropsychological evaluations through Bayleys Scales of Infant Development (BSID) was performed for the first time at 6 months, while Autism Diagnostic Observation Schedule (ADOS) to screen autistic features was initiated at 12 months. All tests have been repeated every 6 months up to 24 months of age. At the time of the first data analysis (31/10/2016) BSID data have been collected at 6, 12, 18 and 24 months respectively for 32, 27, 17 and 15 patients; ADOS data have been collected at 12, 18 and 24 months for 12, 15 and 12 patients. Results: BSID revealed a normal cognitive developmental quotient (DQ) in 52%, 46% and 22% infants at the age respectively of 6, 12 and 24 months and a mild delay in 24%, 24%, and 45% children at the same age. A normal language DQ was observed in 38%, 24% and 11% children respectively at 6, 12 and 24 months and a normal motor DQ in 24%, 18% and 11% children respectively at 6, 12 and 24 months. ADOS revealed a high risk for autism in 8%, 23% and 29% children at 12, 18 and 24 months. Conclusion: Our findings showed that motor development was the first to be impaired with anomalies already detected at 6 months, and a clear deviation from the normal neurodevelopmental trajectory evident in all the domains from 12 months. Also an increase of the rate of children with high risk for autism over time have been observed.