A patient with Duane's syndrome and congenital sensory dysfunction of
the ipsilateral trigeminal nerve is presented. Electrophysiological st
udies of trigeminal function did not reveal any abnormalities. Hypopla
sia of the caudal spinal trigeminal tract is the most likely underlyin
g pathology. Duane's syndrome results from abnormal embryological deve
lopment of the oculomotor nerves and may occasionally affect other cra
nial nerves. Involvement of the trigeminal nerve, however, has not bee
n reported previously.