A novel mouse hemorrhagic hydrocephalus mutation (hhy) inherited in an autosomal recessive manner on chromosome 12 has been found at the Osaka Prefecture University. The hhy homozygous mutant had dilated lateral ventricles and a communicating aqueduct, with no histological abnormalities either in the subarachnoid space or in the choroid plexus. Multiple hemorrhages in the meninges and throughout the brain parenchyma of the mutant were relevant to advanced stages of hydrocephalus. Subcortical heterotopia was detected unexceptionally in the mutants. Thus, the hhy mutation is characterized by three different abnormalities, i.e. hydrocephalus, intracranial hemorrhage and subcortical heterotopia.