Abstract
Recently it has been reported that children with congenital hypothyroidism may have persistent elevation of TSH despite normal serum T4 level with exaggerated response to TRH, indicating a functional abnormality.*We report 2 patients showing the converse situation.- K.K.(♀, 4.9.70.) and A.M. (♂, 22.5.74.) had early symptoms of hypothyroidism and serious psychosomatic demage, later convulsions. Thyroid replacement was begun at the 4th and 2nd month of life respectively. Neither responded with TSH increase to 100 μg of i.v. TRH during their first years in repeated tests despite low T4 level. 99mTc scan showed the absence of functioning thyroid tissue. In a 3-day TSH (AMBINON) test they did not respond with T4 elevation. K.K. was able to secrete GH and FSH in DOPA and LHRH tests; A.M. heigh- and bone age were nearly normal. K.K. was 8 yrs old at the first moderate TSH increase (T4:33,8 nmol/L;TSH:4,9 μU/ml) and A.M. 5 yrs (T4:7 nmol/L;TSH:22 μU/ml) during a temporary cessation of thyroid therapy. Finally a 7-day oral TRH test (1mg/day) increased the TSH level of K.K. to 27,8 μU/ml and of A.M. to 84 μU/ml. During the last two years both children have shown nearly normalized TSH responsiveness.- In conclusion: early deficiency of thyroid hormones may alter the TSH control of man as well as in hypothyroid rats. This demage may prolonged but recovers eventually. The use of TSH alone determinations for neonatal screening of congenital hypothyroidism can miss these rare cases.
*Redmond, G.P. & Soyka, L.F.: J.Pediat.98 ,83.1981.
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Peter, F. Prolonged hypothyrotropinaemia in congenital primary hypothyroidism. Pediatr Res 15, 1555 (1981). https://doi.org/10.1203/00006450-198112000-00120
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DOI: https://doi.org/10.1203/00006450-198112000-00120