Stopkova Pavla, Saito Takuya, Papolos Demitri F, Vevera Jan, Paclt Ivo, Zukov Ilja, Bersson Yonina B, Margolis Benjamin A, Strous Rael D, Lachman Herbert M
Psychiatric Clinic, First Medical Faculty, Charles University, Prague, Czech Republic.
Biol Psychiatry. 2004 May 15;55(10):981-8. doi: 10.1016/j.biopsych.2004.01.014.
Genes involved in phosphoinositide (PI) lipid metabolism are excellent candidates to consider in the pathogenesis of bipolar disorder (BD) and schizophrenia (SZ). One is PIK3C3, a member of the phosphatidylinositide 3-kinase family that maps closely to markers on 18q linked to both BD and SZ in a few studies.
The promoter region of PIK3C3 was analyzed for mutations by single-strand conformation polymorphism analysis and sequencing. A case-control association study was conducted to determine the distribution of variant alleles in unrelated patients from three cohorts. Electromobility gel shift assays (EMSA) were performed to assess the functional significance of variants.
Two polymorphisms in complete linked disequilibrium with each other were identified, -432C- > T and a "C" insert at position -86. The -432T allele occurs within an octamer containing an ATTT motif resembling members of the POU family of transcription factors. In each population analyzed, an increase in -432T was found in patients. EMSAs showed that a -432T containing oligonucleotide binds to brain proteins that do not recognize -432C.
A promoter mutation in a PI regulator affecting the binding of a POU-type transcription factor may be involved in BD and SZ in a subset of patients.
参与磷酸肌醇(PI)脂质代谢的基因是双相情感障碍(BD)和精神分裂症(SZ)发病机制研究中值得考虑的优秀候选基因。其中一个是PIK3C3,它是磷脂酰肌醇3激酶家族的成员,在一些研究中,该基因与18q上与BD和SZ相关的标记紧密连锁。
通过单链构象多态性分析和测序对PIK3C3的启动子区域进行突变分析。进行病例对照关联研究以确定来自三个队列的不相关患者中变异等位基因的分布。进行电泳迁移率凝胶阻滞试验(EMSA)以评估变异的功能意义。
鉴定出两个处于完全连锁不平衡状态的多态性,-432C>T和-86位的“C”插入。-432T等位基因出现在一个八聚体中,该八聚体包含一个类似于POU转录因子家族成员的ATTT基序。在每个分析的人群中,患者中-432T的比例增加。EMSA显示,含有-432T的寡核苷酸与不识别-432C的脑蛋白结合。
PI调节因子中的启动子突变影响POU型转录因子的结合,可能在一部分患者的BD和SZ中起作用。