Ismail Said I, Mahmoud Ismail S, Al-Ardah Mahmoud, Abdelnour Amid, Younes Nidal A
Department of Biochemistry, Faculty of Medicine, University of Jordan, Amman 11942, Jordan.
J Genet. 2009 Aug;88(2):233-8. doi: 10.1007/s12041-009-0032-z.
Five patients, four brothers and their paternal aunt, presented with a history of overt hyperthyroidism and goiter. Hyperthyroidism in this family was remarkable for its poor response to carbimazole (30-50 mg/d). The thyroid ultrasound showed a diffusely enlarged gland in all the affected members, and thyroid stimulating antibodies (TSAB) were negative. Screening for germline mutations in thyroid stimulating hormone (TSH) receptor (TSHR) gene was performed by direct sequencing of genomic DNA extracted from peripheral blood leukocytes of all family members. The sequence analysis of all TSHR gene exons and intron borders revealed two genomic variants. The first was a single nucleotide polymorphism (SNP) within exon seven (Asn187Asn), whereas the other was located in intron seven (IVS7+68TG). All affected members, two asymptomatic brothers with sub-clinical hyperthyroidism, and their father were heterozygous for those two genomic variants. Anti-thyroid drug treatment for several months successfully relieved symptoms in one subject, whereas the remaining patients required total thyroidectomy to control their disease. This is the first Jordanian family with familial non-autoimmune hyperthyroidism, with mutations affecting the TSHR gene.
五名患者,四兄弟及其姑姑,有明显的甲状腺功能亢进和甲状腺肿病史。该家族中的甲状腺功能亢进对卡比马唑(30 - 50毫克/天)反应不佳,这一点很显著。甲状腺超声显示所有受影响成员的甲状腺弥漫性肿大,甲状腺刺激抗体(TSAB)为阴性。通过对从所有家庭成员外周血白细胞中提取的基因组DNA进行直接测序,对促甲状腺激素(TSH)受体(TSHR)基因的种系突变进行筛查。对所有TSHR基因外显子和内含子边界的序列分析揭示了两个基因组变异。第一个是外显子七内的单核苷酸多态性(SNP)(Asn187Asn),另一个位于内含子七(IVS7 + 68T→G)。所有受影响成员、两名患有亚临床甲状腺功能亢进的无症状兄弟及其父亲对于这两个基因组变异均为杂合子。一名患者接受数月的抗甲状腺药物治疗成功缓解了症状,而其余患者需要进行甲状腺全切除术来控制病情。这是首个约旦家族性非自身免疫性甲状腺功能亢进病例,其突变影响TSHR基因。