Congenital vertical ocular motor apraxia

The case of a 4 1/2-year-old boy with congenital vertical ocular motor apraxia who was otherwise developmentally and neurologically normal is reported. The presence of perinatal hypoxia in this patient may have been etiologic. While the presence of a supranuclear vertical ocular motor abnormality usually suggests a serious, acquired neurologic or systemic disease it may rarely occur as an isolated congenital finding, as demonstrated in this case.