Duane Syndrome (DS) in its classic form is characterized by congenital onset limitation of horizontal eye movements with globe retraction and narrowing of palpebral fissure (PF) on adduction.[1] Despite significant limitation of horizontal ocular motility, the ocular deviation in primary position is lesser than would occur in muscle palsies.[1] Upshoot or downshoot in adduction is commonly associated.[1] A congenital sixth nerve or congenital lateral rectus (LR) palsy is rare and may be related to birth trauma.[2] The title of a Souza-Dias publication stated: “Congenital VIth nerve is Duane’s Syndrome until disproven”, and it also reflects the rarity of congenital sixth nerve paresis.[3,4] Here, we present a case of an adolescent, with a congenital sixth nerve palsy presenting as Type I DS.[5] To our knowledge, a similar case has not been reported in literature (Medline search). Our patient was successfully managed with a single muscle surgery.

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