Facial asymmetry is an underrecognized association or sequela of torticollis in congenital or very early onset superior oblique muscle palsy, which when present in an adult, helps to confirm chronicity and prevent unnecessary neurologic evaluation. Nineteen cases of superior oblique muscle palsy were studied prospectively. Of 12 unilateral palsies presenting in adulthood, 9 were considered congenital by history and 7 (77%) of these had facial asymmetry. Large vertical fusional vergence amplitudes and torticollis, on review of childhood photographs, confirmed the early onset of the palsy in each facial asymmetry patient. In addition, two children presenting at age 18 months and 3 years of age with constant head tilting had facial asymmetry. If torticollis is corrected before maturation of the facial structure, resolution of the asymmetry may occur.