Kawai Kosuke, Ward Bryan K, Toivonen Joonas, Poe Dennis S
Department of Medicine Statistics Core, David Geffen School of Medicine, University of California los Angeles, 1100 Glendon Avenue, Los Angeles, CA, 90024, USA.
Department of Otolaryngology-Head and Neck Surgery, Johns Hopkins University School of Medicine, Baltimore, MD, USA.
BMC Med Res Methodol. 2025 Mar 11;25(1):68. doi: 10.1186/s12874-025-02523-3.
The nested frailty model, a random effects survival model that can accommodate data clustered at two hierarchical levels, has been rarely used in practice. We aimed to evaluate the utility of the Bayesian nested frailty modeling approach in the context of a study to examine the effects of various surgical procedures for patients with patulous Eustachian tube dysfunction (PETD).
A nested frailty model was employed to account for the correlation between each pair of ears within patients and the correlation between multiple event times within each ear. Some patients underwent multiple different surgical treatments in their affected ears. We incorporated two nested lognormal frailties into the Cox proportional hazards model. The Bayesian Monte Carlo Markov Chain approach was utilized. We examined the consequences of ignoring a multilevel structure of the data.
The variances of patient-level and ear-level random effects were both found to be significant in the nested frailty model. Shim insertion and patulous Eustachian tube reconstruction using Alloderm or cartilage were associated with a lower risk of recurrence of PETD symptoms than calcium hydroxyapatite injection.
Bayesian nested frailty models provide flexibility in modeling hierarchical survival data and effectively account for multiple levels of clustering. Our study highlights the importance of accounting for all levels of hierarchical clustering for valid inference.
嵌套脆弱模型是一种随机效应生存模型，可处理两个层次水平上的数据聚类，在实际应用中很少被使用。我们旨在评估贝叶斯嵌套脆弱建模方法在一项研究中的效用，该研究旨在考察各种手术程序对咽鼓管异常开放功能障碍（PETD）患者的影响。
采用嵌套脆弱模型来解释患者双耳之间的相关性以及每只耳内多个事件时间之间的相关性。一些患者在患耳接受了多种不同的手术治疗。我们将两个嵌套的对数正态脆弱性纳入Cox比例风险模型。使用了贝叶斯蒙特卡洛马尔可夫链方法。我们研究了忽略数据的多层次结构所产生的后果。
在嵌套脆弱模型中，患者水平和耳水平随机效应的方差均被发现具有显著性。与羟基磷灰石注射相比，插入硅胶管以及使用同种异体真皮或软骨进行咽鼓管重建与PETD症状复发风险较低相关。
贝叶斯嵌套脆弱模型在对分层生存数据进行建模时提供了灵活性，并有效地解释了多层次聚类。我们的研究强调了考虑所有层次聚类以进行有效推断的重要性。
