BACKGROUND Ocular manifestations in Children with Craniofacial Anomalies are an important but easily overlooked part as the patient and his family tends to be more focused on the more visible cosmetic disfigurement. The disfigurement while may be corrected in the hands of a competent surgeon at a later date, more lasting harm may be done to the patient's well-being if simple correctable refractive or other ocular conditions are left too late. This study was done to detect ocular involvement in patients with craniofacial anomalies and also to emphasise importance of routine ophthalmic examination in these patients. MATERIALS AND METHODSThis was a cross-sectional observational type of study. Study duration was dated from dated from November 2012 to October 2014. All patients who presented to the Department of Ophthalmology of Justice KS Hegde Medical College Hospital, Mangalore with craniofacial anomalies were included in this study. All patients with CFA were included in the study irrespective of age of presentation, gender or history of previous corrective surgery. The patients were evaluated at the time of presenting to the Department of Ophthalmology of Justice KS Hegde Medical College, Mangalore. Duration of this study was dated from November 2012 to October 2014. RESULTSOut of the 151 patients screened, 47.68 % (n=72) of the patients had one or the other forms of ocular anomalies. Patients with craniosynostosis had 100% ocular involvement (n=39). In patients with clefting syndrome, 29.46% of the patients had ocular involvement (n=33). Refractive error was most common ocular anomaly detected in our study. 49.01% of the patients in our study had refractive error out of which 22.51% of the patients were hypermetropes. CONCLUSIONFindings in our study suggest that there are numerous ocular associations in patients with craniofacial anomalies. A routine ophthalmic evaluation would help in early diagnosis of these ocular conditions and early management which would greatly benefit the patient.