Pickard Ben S, Malloy M P, Porteous D J, Blackwood D H R, Muir W J
Department of Psychiatry, University of Edinburgh, Kennedy Tower, Royal Edinburgh Hospital, Morningside Park, UK.
Am J Med Genet B Neuropsychiatr Genet. 2005 Jul 5;136B(1):26-32. doi: 10.1002/ajmg.b.30204.
A mother and daughter diagnosed with schizophrenia and schizophrenia co-morbid with mild learning disability, respectively, possess a balanced reciprocal translocation t(9,14)(q34.2;q13). Fluorescence in situ hybridization (FISH) with YAC, BAC, and cosmid probes indicate that the chromosome 14q13 breakpoint disrupts a large gene, NPAS3, encoding a CNS expressed transcription factor of the basic helix-loop-helix PAS (bHLH-PAS) gene family. By analogy with other members of the bHLH-PAS family, the putative truncated protein generated from the disrupted gene locus may have a dominant negative effect. The 14q13 region was previously identified by a linkage study of an inherited neurodegenerative condition, idiopathic basal ganglia calcification (IBGC or Fahr syndrome, OMIM:213600/606656), which is often co-morbid with psychosis. Sequencing of the gene in a third patient diagnosed with IBGC, schizophrenia, and mild learning disability did not reveal functional mutations.
一位被诊断为精神分裂症的母亲和一位被诊断为精神分裂症合并轻度学习障碍的女儿,均拥有一种平衡的相互易位t(9,14)(q34.2;q13)。使用酵母人工染色体(YAC)、细菌人工染色体(BAC)和黏粒探针进行的荧光原位杂交(FISH)表明,14号染色体q13断点破坏了一个大基因NPAS3,该基因编码一种在中枢神经系统表达的碱性螺旋-环-螺旋PAS(bHLH-PAS)基因家族的转录因子。与bHLH-PAS家族的其他成员类似,由被破坏的基因位点产生的推定截短蛋白可能具有显性负效应。14q13区域先前是通过对一种遗传性神经退行性疾病——特发性基底节钙化(IBGC或法尔综合征,OMIM:213600/606656)的连锁研究确定的,该疾病常与精神病合并存在。对第三位被诊断为IBGC、精神分裂症和轻度学习障碍的患者的该基因进行测序,未发现功能性突变。