Familial hemiplegic migraine and its associated signs and symptoms hav
e previously been well described. The condition demonstrates autosomal
dominant inheritance and has been recently assigned to a locus on chr
omosome 19. Previous reports of familial hemiplegic migraine have indi
cated that expression always occurs early in life. We describe a patie
nt who presented with multiple episodes of reversible hemiplegia follo
wed by headache at the age of 75. Seven other family members were foun
d to have been affected in a similar manner. We believe this case repr
esents a unique description of familial hemiplegic migraine presenting
in later life.