The authors present a case report of Duane retraction syndrome (DRS) a
ssociated with Rubinstein-Taybi syndrome (RTS). RTS is a multisystem c
ondition characterized by psychomotor developmental delay, broad thumb
s and great toes, short stature, and characteristic facies with a beak
ed nose. DRS is an ocular disorder characterized by retraction of the
globe with narrowing of the palpebral fissure in attempted adduction,
with abduction and/or adduction deficiency, and, frequently, upshoot o
r downshoot of the affected eye on adduction. The case report describe
d represents the first reported case of Das associated with RTS, an as
sociation not previously recognized. This association raises the possi
bility that the abnormal CNS development in RTS may occasionally invol
ve the ocular motor nerves.