Evidence map›Paper›PMID 41334963›Full record

ArticleStatistical methods in medical research2025

Modeling the effect of longitudinal markers on left-truncated time-to-event outcomes in twin studies.

Annah Muli, Mar Rodriguez-Girondo, Jeanine Houwing-Duistermaat

Abstract read
In one paragraph

Article in Statistical methods in medical research, 2025. The graph could read no effect estimate from its abstract, so it casts no vote on the map. Cited by 1 paper.

0numbers the graph read from it
0cells of the map it votes in
1citing papers in PubMed
–field-weighted citation impact
1 · What the graph read from it

What it found

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2 · The registry

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3 · Its place in the literature

Who cites it

1 citing paper in PubMed.

  1. A generalized approach for estimating the Pace of Aging.The journals of gerontology. Series A, Biological sciences and medical sciences · 2026
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4 · The record

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5 · Who and what money

Authors and funding

3 authors.

Annah MuliDepartment of Statistics, University of Leeds, UK.ORCID 0000-0002-9706-1525
Mar Rodriguez-GirondoDepartment of Biomedical Data Sciences, Leiden University Medical Center, The Netherlands.ORCID 0000-0003-0414-870X
Jeanine Houwing-DuistermaatDepartment of Statistics, University of Leeds, UK.ORCID 0000-0002-4505-7137

Funding

No grant is acknowledged in the PubMed record.

6 · The paper itself

Abstract

The identification of biomarkers for disease onset in longitudinal studies necessitates precise estimation of the association between longitudinal markers and survival outcomes. Currently, methods for estimating these associations in the context of left-truncated and clustered survival outcomes are lacking. In this study, we propose a novel model tailored to this scenario and develop several estimation methods: last observation carried forward, regression calibration, and a two-stage likelihood approach for joint modeling of longitudinal and survival processes. Simulation results indicate that the last observation carried forward method performs well only with a dense grid and no marker measurement error. For less dense grids and low measurement error, regression calibration approaches are preferred. Joint modeling approaches outperform calibration methods in the presence of measurement error, although they may suffer from numerical instability. In cases of numerical instability, calibration methods might be a good alternative. We applied these methodologies to the TwinsUK data to estimate the effect of bone mineral density (BMD) as a longitudinal marker on fracture incidence in 766 elderly females, 138 of whom experienced a fracture. The survival model included a shared gamma-distributed frailty to account for correlation between the times to fracture of twin pairs. Estimates obtained using calibration and joint modeling approaches indicated a larger BMD effect compared to the last observation carried forward method, likely due to the irregular BMD measurement process and minimal measurement error. Overall, our methods offer valuable tools for modeling the effect of a longitudinal marker on survival outcomes in complex designs.

Indexed as

BiomarkersModels, StatisticalTwin Studies as TopicAgedBone DensityComputer SimulationFemaleFractures, BoneHumansLikelihood FunctionsLongitudinal StudiesSurvival AnalysisBiomarkersdelayed entryJoint modellongitudinal dataregression–calibrationshared frailty modeltwins

Identifiers

PMID41334963
PMCPMC12669408

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Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.