Taub Institute for Research on Alzheimer's Disease and the Aging Brain Columbia University Medical Center, New York, NY, USA.
Taub Institute for Research on Alzheimer's Disease and the Aging Brain Columbia University Medical Center, New York, NY, USA.
Cortex. 2021 Jun;139:99-115. doi: 10.1016/j.cortex.2021.03.006. Epub 2021 Mar 19.
Although executive dysfunction is the characteristic cognitive marker of behavioral variant frontotemporal dementia (bvFTD), episodic memory deficits are relatively common, and may be present even during the prodromal disease phase. In a cohort of mutation carriers with mild behavioral and/or cognitive symptoms consistent with prodromal bvFTD, we aimed to investigate patterns of performance on an abbreviated list learning task, with a particular focus on recognition memory. We further aimed to characterize the cognitive prodromes associated with the three major genetic causes of frontotemporal dementia, as emerging evidence suggests there may be subtle differences in cognitive profiles among carriers of different genetic mutations. Participants included 57 carriers of a pathogenic mutation in microtubule-associated protein tau (MAPT, N = 23), or progranulin (GRN, N = 15), or a or a hexanucleotide repeat expansion in chromosome 9 open reading frame 72 (C9orf72, N = 19), with mild cognitive and/or behavioral symptoms consistent with prodromal bvFTD. Familial non-carriers were included as controls (N = 143). All participants completed a comprehensive neuropsychological examination, including an abbreviated list learning test assessing episodic memory recall and recognition. MAPT mutation carriers performed worse than non-carriers in terms of list recall, and had difficulty discriminating targets from distractors on the recognition memory task, primarily due to the endorsement of distractors as targets. MAPT mutation carriers also showed nonverbal episodic memory and semantic memory dysfunction (object naming). GRN mutation carriers were variable in performance and overall the most dysexecutive. Slowed psychomotor speed was evident in C9orf72 repeat expansion carriers. Identifying the earliest cognitive indicators of bvFTD is of critical clinical and research importance. List learning may be a sensitive cognitive marker for incipient dementia in MAPT and potentially a subset of GRN carriers. Our results highlight that distinct cognitive profiles may be evident in carriers of the three disease-causing genes during the prodromal disease stage.
尽管执行功能障碍是行为变异额颞叶痴呆(bvFTD)的特征性认知标志物,但情节记忆缺陷相对常见,甚至可能在疾病前驱期就已经存在。在一组轻度行为和/或认知症状符合前驱性 bvFTD 的突变携带者队列中,我们旨在研究缩写列表学习任务的表现模式,特别关注识别记忆。我们还旨在描述与额颞叶痴呆三种主要遗传病因相关的认知前驱期,因为越来越多的证据表明,不同基因突变携带者的认知特征可能存在细微差异。参与者包括 57 名微管相关蛋白 tau (MAPT) 致病性突变携带者(N=23)、颗粒蛋白前体 (GRN) 携带者(N=15)或 9 号染色体开放阅读框 72 六核苷酸重复扩增(C9orf72)携带者(N=19),这些患者有轻度认知和/或行为症状符合前驱性 bvFTD。家族性非携带者作为对照纳入(N=143)。所有参与者均完成了全面的神经心理学检查,包括评估情节记忆回忆和识别的缩写列表学习测试。MAPT 突变携带者在列表回忆方面的表现不如非携带者,在识别记忆任务中难以区分目标和干扰项,主要是因为将干扰项识别为目标。MAPT 突变携带者还表现出非言语情节记忆和语义记忆功能障碍(物体命名)。GRN 突变携带者的表现各不相同,总体上执行功能障碍最严重。C9orf72 重复扩展携带者的精神运动速度明显减慢。识别 bvFTD 的最早认知指标具有重要的临床和研究意义。列表学习可能是 MAPT 患者和潜在的 GRN 携带者发生早期痴呆的敏感认知标志物。我们的结果表明,在疾病前驱期,三种致病基因的携带者可能存在明显不同的认知特征。