Four infants with obstructive hydrocephalus caused by space occupying third ventricle and mesencephalic cysts are reported. Despite immediate shunt insertion in all patients, there was either lack of clinical improvement or late onset of clinical deterioration. Neuroimaging (CT, MRI, and ventriculography) diagnosed the presence of non-communicating midline outpouchings of the CSF pathways causing obstruction of aqueductal CSF flow and brainstem signs. The cysts were of different origin. In one patient it was caused by a previous thalamic haemorrhage, in another patient by neonatal Escherichia coli meningoventriculitis. In two cases with obstructive hydrocephalus at birth, the aetiology is unclear. Direct puncture and drainage of the cysts led to clinical improvement. The cysts were poorly visualised on CT and could be misinterpreted as an enlarged third ventricle, simulating congenital aqueduct stenosis. Careful neuroradiological investigation is necessary to establish an accurate diagnosis and neurosurgical management. In such cases with hydrocephalus and persisting ventricular enlargement despite shunting, CT ventriculography is a useful tool.
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