We report on an 8-year-old boy with pituitary resistance to thyroid ho
rmone (PRTH) having a cysteine for arginine substitution at codon 320
in the TR-beta gene who was presented because of thyrotoxicosis. Due t
o its suppressive effect on the pituitary thyrotropin secretion, treat
ment with D-thyroxine (D-T4) was started. After a few days, clinical e
uthyroidism was achieved but thyroid stimulating hormone secretion was
not suppressed. Symptoms of thyrotoxicosis relapsed when therapy was
interrupted so that therapy with D-T4 was reinstituted and continued t
o date. Symptoms did not recur, and the psychomotor development procee
ded normally. D-T4 should therefore be considered in the treatment of
thyrotoxicosis in PRTH.