Bagyinszky Eva, Park Sun Ah, Kim Hyung Jun, Choi Seong Hye, An Seong Soo A, Kim Sang Yun
Department of BioNano Technology, Gachon University, Seongnam-si.
Department of Neurology, Soonchunhyang University Bucheon Hospital, Bucheon.
Clin Interv Aging. 2016 Oct 12;11:1433-1440. doi: 10.2147/CIA.S111821. eCollection 2016.
In this study, we report a first 226leucine (Leu) mutation to phenylalanine (Phe) in (PSEN1, CTC>TTC, L226F) in Asia from a Korean early-onset Alzheimer's disease (EOAD) patient. Polymerase chain reaction (PCR)-single strand conformation polymorphism, sequencing, and in silico predictions were performed. Previously, L226F was reported in EOAD patients by Zekanowski et al and Gómez-Tortosa et al. Disease phenotypes appeared in their thirties, and family history was positive in both cases. In our patient, age of onset was similar (37 years of age), but the mutation seemed to be de novo, since no affected family member was found. This leucine to phenylalanine substitution may cause additional stresses inside the transmembrane region due to large aromatic side chain and increased hydrophobic interactions with hydrocarbon chains in the membrane and its binding partners. Clinical phenotype of the mutation was aggressive progression into neurodegeneration, resulting in rapid cognitive decline. One of the patients was initially diagnosed with frontotemporal dementia, but the diagnosis was revised to AD upon postmortem studies in which Aβ plaques were seen. A second mutation, L226R, was found for the L226 residue. Similar to L226F, the patient with L226R also developed the first symptoms in his 30s, but EOAD was diagnosed in his 40s. These findings suggested that L226 might be an important residue in PSEN1, since mutations could result in neurodegenerative disease phenotypes at relatively young ages. There are mutations, such as L226F, which may not present clear clinical symptoms for the definitive diagnosis between frontotemporal dementia and AD. In addition, the similarities in the phenotypes could also be possible between AD and frontotemporal dementia, suggesting difficulties in differential diagnosis of various neurodegenerative diseases.
在本研究中,我们报告了亚洲一名韩国早发性阿尔茨海默病(EOAD)患者的早老素1(PSEN1)基因中首次出现的226位亮氨酸(Leu)突变为苯丙氨酸(Phe)(CTC>TTC,L226F)。进行了聚合酶链反应(PCR)-单链构象多态性分析、测序及计算机模拟预测。此前,Zekanowski等人和Gómez-Tortosa等人在EOAD患者中报道过L226F突变。这两位患者均在三十多岁时出现疾病表型,且家族史均为阳性。在我们的患者中,发病年龄相似(37岁),但该突变似乎是新发的,因为未发现受影响的家庭成员。这种亮氨酸到苯丙氨酸的替换可能会因大的芳香族侧链以及与膜及其结合伴侣中的烃链增加的疏水相互作用而在跨膜区域内引起额外的压力。该突变的临床表型是向神经退行性变的侵袭性进展,导致快速认知衰退。其中一名患者最初被诊断为额颞叶痴呆,但在尸检研究中发现了Aβ斑块后,诊断被修订为阿尔茨海默病。对于226位残基还发现了第二个突变L226R。与L226F相似,携带L226R突变的患者也在30多岁时出现了最初症状,但在40多岁时被诊断为EOAD。这些发现表明,226位可能是PSEN1中的一个重要残基,因为突变可能在相对年轻时导致神经退行性疾病表型。存在一些突变,如L226F,可能不会表现出明确的临床症状以用于额颞叶痴呆和阿尔茨海默病之间的明确诊断。此外,阿尔茨海默病和额颞叶痴呆之间在表型上也可能存在相似之处,这表明各种神经退行性疾病的鉴别诊断存在困难。