Memory and Aging Center, Department of Neurology, UCSF Weill Institute for Neurosciences, University of California, San Francisco, CA 94158, USA.
Department of Neurology and Neurological Sciences, Stanford University, Stanford, CA 94304, USA.
Brain. 2022 Nov 21;145(11):4080-4096. doi: 10.1093/brain/awac217.
Focal anterior temporal lobe degeneration often preferentially affects the left or right hemisphere. While patients with left-predominant anterior temporal lobe atrophy show severe anomia and verbal semantic deficits and meet criteria for semantic variant primary progressive aphasia and semantic dementia, patients with early right anterior temporal lobe atrophy are more difficult to diagnose as their symptoms are less well understood. Focal right anterior temporal lobe atrophy is associated with prominent emotional and behavioural changes, and patients often meet, or go on to meet, criteria for behavioural variant frontotemporal dementia. Uncertainty around early symptoms and absence of an overarching clinico-anatomical framework continue to hinder proper diagnosis and care of patients with right anterior temporal lobe disease. Here, we examine a large, well-characterized, longitudinal cohort of patients with right anterior temporal lobe-predominant degeneration and propose new criteria and nosology. We identified individuals from our database with a clinical diagnosis of behavioural variant frontotemporal dementia or semantic variant primary progressive aphasia and a structural MRI (n = 478). On the basis of neuroimaging criteria, we defined three patient groups: right anterior temporal lobe-predominant atrophy with relative sparing of the frontal lobes (n = 46), frontal-predominant atrophy with relative sparing of the right anterior temporal lobe (n = 79) and left-predominant anterior temporal lobe-predominant atrophy with relative sparing of the frontal lobes (n = 75). We compared the clinical, neuropsychological, genetic and pathological profiles of these groups. In the right anterior temporal lobe-predominant group, the earliest symptoms were loss of empathy (27%), person-specific semantic impairment (23%) and complex compulsions and rigid thought process (18%). On testing, this group exhibited greater impairments in Emotional Theory of Mind, recognition of famous people (from names and faces) and facial affect naming (despite preserved face perception) than the frontal- and left-predominant anterior temporal lobe-predominant groups. The clinical symptoms in the first 3 years of the disease alone were highly sensitive (81%) and specific (84%) differentiating right anterior temporal lobe-predominant from frontal-predominant groups. Frontotemporal lobar degeneration-transactive response DNA binding protein (84%) was the most common pathology of the right anterior temporal lobe-predominant group. Right anterior temporal lobe-predominant degeneration is characterized by early loss of empathy and person-specific knowledge, deficits that are caused by progressive decline in semantic memory for concepts of socioemotional relevance. Guided by our results, we outline new diagnostic criteria and propose the name, 'semantic behavioural variant frontotemporal dementia', which highlights the underlying cognitive mechanism and the predominant symptomatology. These diagnostic criteria will facilitate early identification and care of patients with early, focal right anterior temporal lobe degeneration as well as in vivo prediction of frontotemporal lobar degeneration-transactive response DNA binding protein pathology.
额颞叶变性常优先影响左或右半球。左侧额颞叶萎缩为主的患者表现为严重的命名障碍和言语语义障碍,并符合语义变异原发性进行性失语症和语义性痴呆的标准,而早期右侧额颞叶萎缩的患者则更难诊断,因为其症状不太明确。右侧额颞叶萎缩与明显的情感和行为变化有关,患者往往符合或继续符合行为变异额颞叶痴呆的标准。早期症状的不确定性和缺乏全面的临床解剖学框架继续阻碍对右侧额颞叶疾病患者的正确诊断和护理。在这里,我们研究了一个大型、特征明确的、纵向的右侧额颞叶为主的变性患者队列,并提出了新的诊断标准和分类。我们从我们的数据库中确定了临床诊断为行为变异额颞叶痴呆或语义变异原发性进行性失语症且具有结构磁共振成像(n=478)的个体。根据神经影像学标准,我们定义了三组患者:以右侧额颞叶为主的萎缩伴额叶相对保留(n=46),以额叶为主的萎缩伴右侧额颞叶相对保留(n=79)和以左侧额颞叶为主的萎缩伴额叶相对保留(n=75)。我们比较了这些组的临床、神经心理学、遗传和病理学特征。在以右侧额颞叶为主的组中,最早的症状是同理心丧失(27%)、特定于人的语义障碍(23%)和复杂的强迫和僵化的思维过程(18%)。在测试中,与额叶和左侧额颞叶为主的组相比,该组在情绪理论思维、名人识别(从名字和面孔)和面部情感命名(尽管保留了面部感知)方面表现出更大的损伤。疾病的前 3 年的临床症状具有高度的敏感性(81%)和特异性(84%),可将右侧额颞叶为主的组与额叶为主的组区分开来。额颞叶退行性变-转染反应 DNA 结合蛋白(84%)是右侧额颞叶为主的组最常见的病理学表现。右侧额颞叶为主的退行性变的特征是早期同理心和特定于人的知识丧失,这些损伤是由与社会情感相关的概念的语义记忆进行性下降引起的。根据我们的结果,我们概述了新的诊断标准,并提出了“语义行为变异额颞叶痴呆”的名称,强调了潜在的认知机制和主要的症状。这些诊断标准将有助于对早期、局灶性右侧额颞叶退行性变患者进行早期识别和护理,并对额颞叶退行性变-转染反应 DNA 结合蛋白病理学进行体内预测。