Since introduction of population-based neonatal screening for congenital hypothyroidism (CH), cases of long-term untreated inborn hypothyreosis have become very rare whereas its clinical symptoms are currently not well known. We report a patient with CH due to an ectopic thyroid. The presented case is exceptional due to both advanced age of diagnosis and unusual clinical presentation, as the anomaly was detected during evaluation of suspected acromegaly at the age of 62. This report indicates that there is still necessity of considering, in spite of its rarity, congenital form in differential diagnosis of hypothyroidism.