Sunthornthepvarakul T, Likitmaskul S, Ngowngarmratana S, Angsusingha K, Kitvitayasak S, Scherberg N H, Refetoff S
Department of Medicine, Rajavithi Hospital, Bangkok, Thailand.
J Clin Endocrinol Metab. 1998 May;83(5):1448-54. doi: 10.1210/jcem.83.5.4815.
We report the abnormal albumin in members of a Thai family that presented with high serum total T3 but not T4 when measured by radioimmunoassay. In contrast, total T3 values were very low when measured by ELISA and chemiluminescence. The subjects have no goiter, and clinically euthyroid. Their serum free T4, free T3, and TSH were normal. Spiking of T3 to affected serum showed good recovery by radioimmunoassay, but very poor recovery by ELISA and by chemiluminescence. The immunoprecipitation with labeled T3 bound to albumin showed high percent precipitation in affected serum. T3-binding studies showed that the association constant of serum albumin in affected subjects was 1.5 x 10(6) M-1 or 40-fold that of unaffected relatives of 3.9 x 10(4) M-1. In contrast, the Ka of HSA for T4 in an affected subject was only 1.5-fold that of a normal. Albumin complementary DNA from leukocytes of affected member was amplified and sequenced. We found the second nucleotide of normal codon 66 (CTT), a thymine, was substituted by a cytosine (CCT), resulting in the replacement of the normal leucine by proline. This is the first report of variant albumin causing familial dysalbuminemic hypertriiodothyroninemia.
我们报告了一个泰国家庭成员中白蛋白异常的情况,这些成员通过放射免疫测定法检测时血清总T3升高但T4正常。相比之下,通过酶联免疫吸附测定法(ELISA)和化学发光法检测时,总T3值非常低。这些受试者没有甲状腺肿大,临床甲状腺功能正常。他们的血清游离T4、游离T3和促甲状腺激素(TSH)均正常。向受影响的血清中加入T3后,放射免疫测定法显示回收率良好,但ELISA和化学发光法的回收率很差。用与白蛋白结合的标记T3进行免疫沉淀显示,受影响血清中的沉淀百分比很高。T3结合研究表明,受影响受试者血清白蛋白的缔合常数为1.5×10⁶ M⁻¹,是未受影响亲属的3.9×10⁴ M⁻¹的40倍。相比之下,受影响受试者中血清白蛋白(HSA)对T4的缔合常数(Ka)仅是正常人的1.5倍。对受影响成员白细胞中的白蛋白互补DNA进行了扩增和测序。我们发现正常密码子66(CTT)的第二个核苷酸胸腺嘧啶被胞嘧啶(CCT)取代,导致正常的亮氨酸被脯氨酸取代。这是关于变异白蛋白导致家族性异常白蛋白血症性高甲状腺素血症的首次报告。