Sunthornthepvarakul T, Angkeow P, Weiss R E, Hayashi Y, Refetoff S
Department of Medicine, University of Chicago, IL 60637.
Biochem Biophys Res Commun. 1994 Jul 29;202(2):781-7. doi: 10.1006/bbrc.1994.1998.
Familial dysalbuminemic hyperthyroxinemia (FDH) is the most common form of inherited increase of serum thyroxine in Caucasians. It is the result of increased thyroxine-binding to serum proteins and is inherited as a dominant trait. The entire coding region of the albumin gene of a subject with FDH was sequenced. A single nucleotide substitution, G to A transition in codon 218, was found in one of the two alleles, resulting in the replacement of the normal Arg with His. This mutation was found in 9 affected family members but not in 8 unaffected relatives and 18 unrelated normal individuals. The same missense mutation was found in 12 other subjects with FDH belonging to 7 unrelated families. In every individual with FDH, the mutation was associated with the Sac I+ polymorphism in the albumin gene, strongly suggesting a founder effect.
家族性异常白蛋白血症性高甲状腺素血症(FDH)是白种人中最常见的遗传性血清甲状腺素升高形式。它是甲状腺素与血清蛋白结合增加的结果,呈显性遗传。对一名患有FDH的受试者的白蛋白基因整个编码区进行了测序。在两个等位基因中的一个中发现了一个单核苷酸替换,即密码子218处的G到A转换,导致正常的精氨酸被组氨酸取代。在9名受影响的家庭成员中发现了这种突变,但在8名未受影响的亲属和18名无关的正常个体中未发现。在另外12名来自7个无关家族的患有FDH的受试者中也发现了相同的错义突变。在每个患有FDH的个体中,该突变都与白蛋白基因中的Sac I+多态性相关,强烈提示存在奠基者效应。