Dural sinus malformation occasionally results in the development of giant venous lakes involving torcular herophili. Such dilatation can result in the formation of a massive venous thrombus leading to ventriculomegaly/hydrocephalus. Although, majority of patients have an unremarkable clinical course owing to spontaneous resolution of the thrombus, significant morbidity and mortality has been documented. We report the first case of torcular thrombosis in a preterm infant who survived severe anemia, consumption coagulopathy and high-output cardiac failure during the neonatal period and had a normal neurological outcome.