Rivolta Carina M, Esperante Sebastián A, Gruñeiro-Papendieck Laura, Chiesa Ana, Moya Christian M, Domené Sabina, Varela Viviana, Targovnik Héctor M
Laboratorio de Biología Molecular, Cátedra de Genética y Biología Molecular, Facultad de Farmacia y Bioquímica, Universidad de Buenos Aires, Buenos Aires, Argentina.
Hum Mutat. 2003 Sep;22(3):259. doi: 10.1002/humu.9175.
Thyroid peroxidase (TPO) defects, typically transmitted as autosomal recessive traits, result in hypothyroid goiters with failure to convert iodide into organic iodine. We analyzed the TPO gene in 14 unrelated patients with clinical evidence of iodide organification defects. Seven of the affected individuals harbored mutations in the TPO gene; one was compound heterozygous, the others were simply heterozygous for TPO mutations. Five novel mutations have been identified, one of which was found to be a single nucleotide deletion, while the other four were single nucleotide substitutions. A frameshift mutation c.387delC was detected in exon 5 which leads to an early termination signal in exon 7 (p.N129fsX208). Two missense mutations were identified in exon 8. The first, a c.920A>C transversion that results in a p.N307T substitution, was found in two patients. The second, a c.1297G>A transition, results in p.V433M. A c.1496C>T transition was detected in exon 9 that caused the substitution p.P499L. Finally, in exon 14 a c.2422T>C transition was identified, causing a p.C808R change. In addition, the previously reported GGCC duplication in exon 8 (c.1186_1187insGGCC; p.R396fsX472) was also detected in two affected individuals, one of whom was a compound heterozygous (p.R396fsX472/p.V433M).
甲状腺过氧化物酶(TPO)缺陷通常以常染色体隐性遗传特征传递,会导致甲状腺功能减退性甲状腺肿,无法将碘化物转化为有机碘。我们分析了14名有碘化物有机化缺陷临床证据的非亲属患者的TPO基因。7名受影响个体的TPO基因存在突变;1名是复合杂合子,其他是TPO突变的单纯杂合子。已鉴定出5种新突变,其中1种是单核苷酸缺失,其他4种是单核苷酸替换。在外显子5中检测到移码突变c.387delC,导致外显子7中出现早期终止信号(p.N129fsX208)。在外显子8中鉴定出2种错义突变。第一种是c.920A>C颠换,导致p.N307T替换,在2名患者中发现。第二种是c.1297G>A转换,导致p.V433M。在外显子9中检测到c.1496C>T转换,导致p.P499L替换。最后,在外显子14中鉴定出c.2422T>C转换,导致p.C808R变化。此外,在2名受影响个体中还检测到先前报道的外显子8中的GGCC重复(c.1186_1187insGGCC;p.R396fsX472),其中1名是复合杂合子(p.R396fsX472/p.V433M)。