Purpose: To study the clinical presentation and surgical outcome of CSOP. Materials and Methods: A retrospective analysis of 15 cases of unilateral CSOP in the age group of 7-20 years between 2004-2006 visiting the pediatric ophthalmology department of a tertiary eye care centre in south India was done. Results: Patients presented with anomalous head posture (AHP), strabismus and facial asymmetry. Among 15 cases,12 cases underwent one muscle surgery, and 3 cases underwent two muscle surgery. The mean deviation reduced from 15 ± 8 pd preoperatively to 4 ± 2 pd postoperatively which was statistically significant by paired t test. Conclusion: Isolated Inferior Oblique (IO) weakening is a safe and effective treatment option for CSOP of up to 20 PD of vertical deviation in primary position and two muscle surgery should be reserved for larger deviations. Good anatomical alignment was obtained in all patients with satisfactory cosmetic results with either total resolution of head posture or only a mild head tilt postoperatively.