ABSTRACT
SUMMARY Resistance to thyroid hormone (RTH) coexisting with ectopic thyroid is rare. Here we report a case of RTH with ectopic thyroid. A ten-year-old girl had been misdiagnosed as congenital hypothyroidism and treated with levothyroxine since she was born. Ten-year follow-up showed that the elevated thyrotropin was never suppressed by levothyroxine and no signs indicating hyperthyroidism or hypothyroidism despite elevated FT3 and FT4 levels. Therefore the girl developed no defects in physical and cognitive development. Pituitary adenoma was excluded by magnetic resonance imaging. Ultrasonography did not find the thyroid gland in the normal place, while the thyroid scan found a large lingual thyroid gland. The octreotide inhibition test showed a reduction in thyrotropin by 41.98%. No mutation was detected in the thyroid hormone receptor (THR) β, THRα, thyrotropin receptor (TSHR), and GNAS1 genes. To our knowledge, it is an interesting RTH case coexisting with lingual thyroid.
Subject(s)
Humans , Female , Child , Receptors, Thyroid Hormone/genetics , Thyroid Hormone Resistance Syndrome/complications , Thyroid Dysgenesis/complications , Thyroxine/therapeutic use , Time Factors , Tongue Diseases/diagnostic imaging , DNA/isolation & purification , Thyrotropin/analysis , DNA Mutational Analysis , Follow-Up Studies , Thyroid Hormone Resistance Syndrome/genetics , Congenital Hypothyroidism/diagnosis , Diagnostic Errors , Thyroid Dysgenesis/genetics , Thyroid Dysgenesis/diagnostic imagingABSTRACT
Resistance to thyroid hormone (RTH) coexisting with ectopic thyroid is rare. Here we report a case of RTH with ectopic thyroid. A ten-year-old girl had been misdiagnosed as congenital hypothyroidism and treated with levothyroxine since she was born. Ten-year follow-up showed that the elevated thyrotropin was never suppressed by levothyroxine and no signs indicating hyperthyroidism or hypothyroidism despite elevated FT3 and FT4 levels. Therefore the girl developed no defects in physical and cognitive development. Pituitary adenoma was excluded by magnetic resonance imaging. Ultrasonography did not find the thyroid gland in the normal place, while the thyroid scan found a large lingual thyroid gland. The octreotide inhibition test showed a reduction in thyrotropin by 41.98%. No mutation was detected in the thyroid hormone receptor (THR) ß, THRα, thyrotropin receptor (TSHR), and GNAS1 genes. To our knowledge, it is an interesting RTH case coexisting with lingual thyroid.
Subject(s)
Receptors, Thyroid Hormone/genetics , Thyroid Dysgenesis/complications , Thyroid Hormone Resistance Syndrome/complications , Child , Congenital Hypothyroidism/diagnosis , DNA/isolation & purification , DNA Mutational Analysis , Diagnostic Errors , Female , Follow-Up Studies , Humans , Thyroid Dysgenesis/diagnostic imaging , Thyroid Dysgenesis/genetics , Thyroid Hormone Resistance Syndrome/genetics , Thyrotropin/analysis , Thyroxine/therapeutic use , Time Factors , Tongue Diseases/diagnostic imagingABSTRACT
Two new mexicanolide-type limonoids, named xylomexicanin A (1) and xylomexicanin B (2), were isolated from seeds of the Chinese mangrove Xylocarpus granatum. Their structures were elucidated on the basis of spectroscopic methods. Compound 1 exhibited antiproliferative activity against human breast carcinoma cells (KT), while 2 did not show inhibitory effects on eleven human tumour cell lines tested.