Kutoku Yumiko, Ohsawa Yutaka, Kuwano Ryozo, Ikeuchi Takeshi, Inoue Haruhisa, Ataka Suzuka, Shimada Hiroyuki, Mori Hiroshi, Sunada Yoshihide
Department of Neurology, Kawasaki Medical School, Japan.
Intern Med. 2015;54(2):205-8. doi: 10.2169/internalmedicine.54.3021. Epub 2015 Jan 15.
A 59-year-old woman developed early-onset, slowly progressive dementia and spastic paraplegia. positron emission tomography (PET) imaging revealed a large reduction in the level of glucose uptake without amyloid deposition in the cerebral cortex. We identified a homozygous microdeletion within the amyloid β (Aβ) coding sequence in the amyloid precursor protein (APP) gene (c.2080_2082delGAA, p.E693del) in three affected siblings and a heterozygous microdeletion in an unaffected sibling. The identical mutation was previously reported in the first Alzheimer's pedigree without amyloid deposits. Furthermore, an increase in high-molecular-weight Aβ-reactive bands was detected in the patient's CSF. Our findings suggest that soluble Aβ-oligomers induce neuronal toxicity, independent of insoluble Aβ fibrils.
一名59岁女性出现早发性、缓慢进展性痴呆和痉挛性截瘫。正电子发射断层扫描(PET)成像显示大脑皮质葡萄糖摄取水平大幅降低,且无淀粉样蛋白沉积。我们在三名患病兄弟姐妹的淀粉样前体蛋白(APP)基因的淀粉样β(Aβ)编码序列中鉴定出一个纯合微缺失(c.2080_2082delGAA,p.E693del),在一名未患病的兄弟姐妹中鉴定出一个杂合微缺失。此前在首个无淀粉样蛋白沉积的阿尔茨海默病家系中报道过相同的突变。此外,在患者脑脊液中检测到高分子量Aβ反应性条带增加。我们的研究结果表明,可溶性Aβ寡聚体可诱导神经元毒性,与不溶性Aβ原纤维无关。