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Mild pericardial effusion is common in severe form of hypothyroidism but massive pericardial effusion with or without cardiac tamponade is rare.  A 49 year-old male with a short stature, large head and facial puffiness, was brought to the emergency department because of progressive altered mental status and severe dyspnea. Cardiac tamponade and hypothyroidism were diagnosed in work ups. Lack of thyroid tissue in his imagines, confirmed the diagnosis of thyroid agenesis. With pericardiocenthesis, hydration and starting levothyroxine his clinical symptoms were resolved dramatically. Our case was presented with full-blown clinical manifestations and irreversible consequences of congenital hypothyroidism. This shows us the important role of newborn screening tests.