This study evaluated the frequencies of ophthalmic abnormalities in Turkish children with a history of congenital hypothyroidism in a retrospective study of 121 children. Median age was nine months (one to 216) at initial ophthalmic examination. Forty children were ex-premature and five had retinopathy of prematurity. Nine patients had thyroid agenesis or hypoplasia. Remaining patients had dyshormonogenesis and iodine deficiency. Visual acuity was measured in 78 children – the remaining children had positive results on preferential looking. Mean flash VEP p100 latencies were delayed in 74.4% but over 60% of these had history of retinopathy of prematurity (ROP); 40.4% had ophthalmic pathology including significant refractive error and strabismus; 87 patients were emmetropic and six were anisometropic. Eleven had esotropia, four exotropia and one hypertropia; 74.3% had systemic abnormalities including congenital heart defects, neurological abnormalities, Down syndrome and atopic dermatitis. Limitations of this study were its retrospective design, some patients were euthyroid, some were on treatment and the duration of treatment was not evaluated. The authors recommend routine ophthalmic screening for such children.