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To investigate the morphology of the optic disc and retinal vessels in children with surgically treated hydrocephalus.A prospective, population-based study was performed in 69 children (median age 9.6 years) with early surgically treated hydrocephalus. All children were examined by ophthalmoscopy. Additionally, optic disc and retinal vessel morphology was evaluated in 55 children by digital image analysis of ocular fundus photographs.Optic atrophy was found in 10 of 69 children (14%). In comparison with a reference group, the median optic-disc area was significantly smaller (p = 0.013) in the children with hydrocephalus. There was no corresponding difference in cup area, so the rim area was significantly smaller in the hydrocephalic children (p = 0.002). Children with hydrocephalus had an abnormal retinal vascular pattern, with significantly straighter retinal arteries and fewer central vessel branching points compared with controls (p<0.001 and p<0.001, respectively).Hydrocephalus is associated with subnormal optic disc and rim areas and an abnormal vascular pattern, indicating a pre/perinatal disturbance of the development of these structures. A promising finding is that the frequency of optic atrophy in the present study was lower than previously reported, most likely reflecting improved perinatal care and better regulation of the intracranial pressure.