Chronic diseases often progress differently across patients. Rather than randomly varying, there are typically a small number of subtypes for how a disease progresses across patients. To capture this structured heterogeneity, the Subtype and Stage Inference Event-Based Model (SuStaIn) estimates the number of subtypes, the order of disease progression for each subtype, and assigns each patient to a subtype from primarily cross-sectional data. It has been widely applied to uncover the subtypes of many diseases and inform our understanding of them. But how robust is its performance? In this paper, we develop a principled Bayesian subtype variant of the event-based model (BEBMS) and compare its performance to SuStaIn in a variety of synthetic data experiments with varied levels of model misspecification. BEBMS substantially outperforms SuStaIn across ordering, staging, and subtype assignment tasks. Further, we apply BEBMS and SuStaIn to a real-world Alzheimer's data set. We find BEBMS has results that are more consistent with the scientific consensus of Alzheimer's disease progression than SuStaIn.
翻译:慢性疾病在不同患者中的进展模式往往存在差异。这种进展通常并非随机变化,而是存在少数几种典型的疾病进展亚型。为捕捉这种结构化异质性,亚型与分期推断事件模型(SuStaIn)能够从主要基于横断面数据中估计亚型数量、各亚型的疾病进展顺序,并将每位患者分配至特定亚型。该模型已被广泛应用于揭示多种疾病的亚型分类,并深化了我们对疾病机制的理解。但其性能的稳健性如何?本文提出了一种基于贝叶斯原理的事件模型亚型推断方法(BEBMS),通过在多种模型设定存在不同程度误设的合成数据实验中,将其性能与SuStaIn进行系统比较。结果表明,在疾病进展排序、分期划分及亚型分配任务中,BEBMS均显著优于SuStaIn。进一步地,我们将BEBMS与SuStaIn应用于真实世界阿尔茨海默病数据集,发现BEBMS得出的结果比SuStaIn更符合当前科学界对阿尔茨海默病进展路径的共识。