Department of Neurology, Miller School of Medicine, University of Miami, Miami, FL, USA.
Department of Psychiatry and Human Behaviour, Alpert Medical School of Brown University, Providence, RI, USA.
Nat Rev Neurol. 2024 Jun;20(6):364-376. doi: 10.1038/s41582-024-00961-z. Epub 2024 May 20.
Increasing appreciation of the phenotypic and biological overlap between amyotrophic lateral sclerosis (ALS) and frontotemporal dementia, alongside evolving biomarker evidence for a pre-symptomatic stage of disease and observations that this stage of disease might not always be clinically silent, is challenging traditional views of these disorders. These advances have highlighted the need to adapt ingrained notions of these clinical syndromes to include both the full phenotypic continuum - from clinically silent, to prodromal, to clinically manifest - and the expanded phenotypic spectrum that includes ALS, frontotemporal dementia and some movement disorders. The updated clinical paradigms should also align with our understanding of the biology of these disorders, reflected in measurable biomarkers. The Miami Framework, emerging from discussions at the Second International Pre-Symptomatic ALS Workshop in Miami (February 2023; a full list of attendees and their affiliations appears in the Supplementary Information) proposes a classification system built on: first, three parallel phenotypic axes - motor neuron, frontotemporal and extrapyramidal - rather than the unitary approach of combining all phenotypic elements into a single clinical entity; and second, biomarkers that reflect different aspects of the underlying pathology and biology of neurodegeneration. This framework decouples clinical syndromes from biomarker evidence of disease and builds on experiences from other neurodegenerative diseases to offer a unified approach to specifying the pleiotropic clinical manifestations of disease and describing the trajectory of emergent biomarkers.
对肌萎缩侧索硬化症 (ALS) 和额颞叶痴呆之间表型和生物学重叠的认识不断提高,加上疾病前阶段的生物标志物证据不断发展,以及观察到该疾病阶段并不总是临床无症状,这对这些疾病的传统观点提出了挑战。这些进展突出表明需要调整这些临床综合征的固有观念,既要包括从临床无症状到前驱期到临床表现的全表型连续体,也要包括扩展的表型谱,包括 ALS、额颞叶痴呆和一些运动障碍。更新的临床范例还应与我们对这些疾病生物学的理解保持一致,这反映在可测量的生物标志物中。迈阿密框架源自于 2023 年 2 月在迈阿密举行的第二次肌萎缩侧索硬化症前临床研讨会(参加者的完整名单及其所属机构见补充资料)的讨论,提出了一个分类系统,基于:首先,三个平行的表型轴——运动神经元、额颞叶和锥体外系,而不是将所有表型元素组合成单一临床实体的单一方法;其次,反映神经退行性变潜在病理学和生物学不同方面的生物标志物。该框架将临床综合征与疾病的生物标志物证据分离,并借鉴其他神经退行性疾病的经验,为指定疾病的多效临床表现和描述新兴生物标志物的轨迹提供了一种统一的方法。