Modeling of atrophy size trajectories: variable transformation, prediction and age-of-onset estimation.

Age-of-onset estimation Age-related macular degeneration Box-Cox transformation Geographic atrophy Mixed-effects models Prediction

Journal

BMC medical research methodology
ISSN: 1471-2288
Titre abrégé: BMC Med Res Methodol
Pays: England
ID NLM: 100968545

Informations de publication

Date de publication:
17 08 2021
Historique:
received: 29 12 2020
accepted: 22 07 2021
entrez: 18 8 2021
pubmed: 19 8 2021
medline: 28 8 2021
Statut: epublish

Résumé

To model the progression of geographic atrophy (GA) in patients with age-related macular degeneration (AMD) by building a suitable statistical regression model for GA size measurements obtained from fundus autofluorescence imaging. Based on theoretical considerations, we develop a linear mixed-effects model for GA size progression that incorporates covariable-dependent enlargement rates as well as correlations between longitudinally collected GA size measurements. To capture nonlinear progression in a flexible way, we systematically assess Box-Cox transformations with different transformation parameters λ. Model evaluation is performed on data collected for two longitudinal, prospective multi-center cohort studies on GA size progression. A transformation parameter of λ=0.45 yielded the best model fit regarding the Akaike information criterion (AIC). When hypertension and hypercholesterolemia were included as risk factors in the model, they showed an association with progression of GA size. The mean estimated age-of-onset in this model was 67.21±6.49 years. We provide a comprehensive framework for modeling the course of uni- or bilateral GA size progression in longitudinal observational studies. Specifically, the model allows for age-of-onset estimation, identification of risk factors and prediction of future GA size. A square-root transformation of atrophy size is recommended before model fitting.

Sections du résumé

BACKGROUND
To model the progression of geographic atrophy (GA) in patients with age-related macular degeneration (AMD) by building a suitable statistical regression model for GA size measurements obtained from fundus autofluorescence imaging.
METHODS
Based on theoretical considerations, we develop a linear mixed-effects model for GA size progression that incorporates covariable-dependent enlargement rates as well as correlations between longitudinally collected GA size measurements. To capture nonlinear progression in a flexible way, we systematically assess Box-Cox transformations with different transformation parameters λ. Model evaluation is performed on data collected for two longitudinal, prospective multi-center cohort studies on GA size progression.
RESULTS
A transformation parameter of λ=0.45 yielded the best model fit regarding the Akaike information criterion (AIC). When hypertension and hypercholesterolemia were included as risk factors in the model, they showed an association with progression of GA size. The mean estimated age-of-onset in this model was 67.21±6.49 years.
CONCLUSIONS
We provide a comprehensive framework for modeling the course of uni- or bilateral GA size progression in longitudinal observational studies. Specifically, the model allows for age-of-onset estimation, identification of risk factors and prediction of future GA size. A square-root transformation of atrophy size is recommended before model fitting.

Identifiants

pubmed: 34404346
doi: 10.1186/s12874-021-01356-0
pii: 10.1186/s12874-021-01356-0
pmc: PMC8369742
doi:

Types de publication

Journal Article Research Support, N.I.H., Extramural Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Pagination

170

Subventions

Organisme : Deutsche Forschungsgemeinschaft
ID : FL658/4-2
Organisme : Deutsche Forschungsgemeinschaft
ID : PF950/1-1
Organisme : Deutsche Forschungsgemeinschaft
ID : Ho 1926/1-3

Informations de copyright

© 2021. The Author(s).

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Auteurs

Charlotte Behning (C)

Department of Medical Biometry, Informatics and Epidemiology, University Hospital Bonn, Venusberg-Campus 1, Bonn, 53127, Germany. charlotte.behning@imbie.uni-bonn.de.

Monika Fleckenstein (M)

John A. Moran Eye Center, University of Utah, Salt Lake City, USA.

Maximilian Pfau (M)

Ophthalmic Genetics and Visual Function Branch, National Eye Institute, Bethesda, MD, USA.

Christine Adrion (C)

Institute for Medical Information Processing, Biometry and Epidemiology, Ludwig-Maximilians-University, Munich, Germany.

Lukas Goerdt (L)

Department of Ophthalmology, University Hospital Bonn, Bonn, Germany.

Moritz Lindner (M)

Department of Ophthalmology, University Hospital Bonn, Bonn, Germany.

Steffen Schmitz-Valckenberg (S)

John A. Moran Eye Center, University of Utah, Salt Lake City, USA.

Frank G Holz (FG)

Department of Ophthalmology, University Hospital Bonn, Bonn, Germany.

Matthias Schmid (M)

Department of Medical Biometry, Informatics and Epidemiology, University Hospital Bonn, Venusberg-Campus 1, Bonn, 53127, Germany.

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