There are rare reports of children with hydromyelia in association wit
h arachnoid cysts at the foramen of Magendie, and these cases have uni
formly been associated with hydrocephalus. We report a case of a 45-ye
ar-old woman with a posterior fossa cyst associated with hydromyelia a
nd normal ventricles. This was successfully treated with a cystoperito
neal shunt. We believe this unusual condition is of interest in elucid
ating potential mechanisms of hydromyelia.