The etiology of craniofacial asymmetry secondary to positional plagiocephaly with or without concomitant congenital muscular torticollis has been well established. It has been proposed that the craniofacial asymmetry secondary to congenital superior oblique palsy involves a similar etiology. The causal relationship has been thought to be a result of the adoption of certain head and neck positioning, which predisposes the infant to develop preferential resting positions during supine sleep. We present a single subject with ocular torticollis and resulting plagiocephaly, and we distinguish the resultant craniofacial findings from those seen in patients with congenital muscular torticollis-associated deformational plagiocephaly. The distinctions that exist between the resultant asymmetries observed in ocular torticollis with superior oblique palsy and those found with congenital muscular torticollis suggest that the facial hemihypoplasia observed in conjunction with ocular torticollis may be the result of gravitational forces rather than compressive forces.