Abramowicz M J, Targovnik H M, Varela V, Cochaux P, Krawiec L, Pisarev M A, Propato F V, Juvenal G, Chester H A, Vassart G
Institut de Recherche Interdisciplinaire en Biologie Humaine et Nucléaire, Free University of Brussels, Belgium.
J Clin Invest. 1992 Oct;90(4):1200-4. doi: 10.1172/JCI115981.
Thyroid peroxidase (TPO) is the key enzyme in the synthesis of thyroid hormones, and the TPO defects are believed to be the most prevalent causes of the inborn errors of thyroid metabolism. We investigated an adopted boy with iodide organification defect, who presented with florid hypothyroidism at the age of 4 mo, poorly complied with thyroxine treatment, and developed a compressive goiter necessitating partial resection at the age of 12 yr. Biochemical studies revealed the absence of TPO activity in the resected tissue. Genomic DNA studies identified a 4 base-pair insertion in the eighth exon of the TPO gene, and showed that the patient was homozygous for this frameshift mutation. The direct genetic diagnosis of this mutation can be made by digestion of polymerase chain reaction products with NaeI restriction enzyme. This will help assessing its prevalence among the heterogenous genetic group of TPO defects.
甲状腺过氧化物酶(TPO)是甲状腺激素合成中的关键酶,TPO缺陷被认为是先天性甲状腺代谢紊乱最常见的原因。我们调查了一名患有碘有机化缺陷的领养男孩,他在4个月大时出现明显的甲状腺功能减退,对甲状腺素治疗依从性差,并在12岁时出现压迫性甲状腺肿,需要进行部分切除。生化研究显示切除组织中缺乏TPO活性。基因组DNA研究在TPO基因的第八外显子中发现了一个4个碱基对的插入,并表明该患者为这种移码突变的纯合子。通过用NaeI限制酶消化聚合酶链反应产物可以对这种突变进行直接基因诊断。这将有助于评估其在TPO缺陷异质基因组中的患病率。