Large-scale GWAS of strabismus identifies risk loci and provides support for a link with maternal smoking

Abstract Strabismus is a common pediatric eye misalignment and has complex genetic and environmental causes. Previous genome-wide association studies (GWAS) encountered difficulties in identifying strabismus risk variants due to heterogeneity and small samples. We performed large meta-analyses of 11...

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Main Authors: Weixiong He, Peter J. van der Most, Jue-Sheng Ong, Liang-Dar Hwang, Yeda Wu, Morten S. Magnø, Jelle Vehof, Kristi Krebs, Laura Mauring, Katrin Õunap, Erik Abner, Nicholas G. Martin, Denis Plotnikov, Chen Jiang, Ronald B. Melles, Puya Gharahkhani, Harold Snieder, Teele Palumaa, Kuldar Kaljurand, Jeremy A. Guggenheim, David A. Mackey, Elizabeth C. Engle, Hélène Choquet, Lifelines Cohort Study, Stuart MacGregor
Format: Article
Language:English
Published: Nature Portfolio 2025-08-01
Series:Nature Communications
Online Access:https://doi.org/10.1038/s41467-025-62456-9
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author Weixiong He
Peter J. van der Most
Jue-Sheng Ong
Liang-Dar Hwang
Yeda Wu
Morten S. Magnø
Jelle Vehof
Kristi Krebs
Laura Mauring
Katrin Õunap
Erik Abner
Nicholas G. Martin
Denis Plotnikov
Chen Jiang
Ronald B. Melles
Puya Gharahkhani
Harold Snieder
Teele Palumaa
Kuldar Kaljurand
Jeremy A. Guggenheim
David A. Mackey
Elizabeth C. Engle
Hélène Choquet
Lifelines Cohort Study
Stuart MacGregor
author_facet Weixiong He
Peter J. van der Most
Jue-Sheng Ong
Liang-Dar Hwang
Yeda Wu
Morten S. Magnø
Jelle Vehof
Kristi Krebs
Laura Mauring
Katrin Õunap
Erik Abner
Nicholas G. Martin
Denis Plotnikov
Chen Jiang
Ronald B. Melles
Puya Gharahkhani
Harold Snieder
Teele Palumaa
Kuldar Kaljurand
Jeremy A. Guggenheim
David A. Mackey
Elizabeth C. Engle
Hélène Choquet
Lifelines Cohort Study
Stuart MacGregor
author_sort Weixiong He
collection DOAJ
description Abstract Strabismus is a common pediatric eye misalignment and has complex genetic and environmental causes. Previous genome-wide association studies (GWAS) encountered difficulties in identifying strabismus risk variants due to heterogeneity and small samples. We performed large meta-analyses of 11 European-ancestry GWAS (7 sources), analysing broad strabismus (20,464 cases, 954,921 controls) and subtypes (esotropia/exotropia). We discovered 4 loci (e.g., NPLOC4-TSPAN10-PDE6G-FAAP100, COL6A1) for strabismus and 5 additional loci (e.g., CHRNA4, MAD1L1) for strabismus subtypes and we successfully replicated the previously reported strabismus variant near NPLOC4-TSPAN10-PDE6G-FAAP100. Using mendelian randomisation, we found genetic evidence supporting maternal smoking as a causal risk factor for strabismus in offspring.
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issn 2041-1723
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publisher Nature Portfolio
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series Nature Communications
spelling doaj-art-022eb5fe224c4ddfa1f9cb4a15ccd6492025-08-24T11:37:43ZengNature PortfolioNature Communications2041-17232025-08-0116111410.1038/s41467-025-62456-9Large-scale GWAS of strabismus identifies risk loci and provides support for a link with maternal smokingWeixiong He0Peter J. van der Most1Jue-Sheng Ong2Liang-Dar Hwang3Yeda Wu4Morten S. Magnø5Jelle Vehof6Kristi Krebs7Laura Mauring8Katrin Õunap9Erik Abner10Nicholas G. Martin11Denis Plotnikov12Chen Jiang13Ronald B. Melles14Puya Gharahkhani15Harold Snieder16Teele Palumaa17Kuldar Kaljurand18Jeremy A. Guggenheim19David A. Mackey20Elizabeth C. Engle21Hélène Choquet22Lifelines Cohort StudyStuart MacGregor23QIMR Berghofer Medical Research InstituteDepartment of Epidemiology, University Medical Center Groningen, University of GroningenQIMR Berghofer Medical Research InstituteInstitute for Molecular Bioscience, University of QueenslandQIMR Berghofer Medical Research InstituteDepartment of Ophthalmology, University Medical Center Groningen, University of GroningenDepartment of Twin Research and Genetic Epidemiology, King’s College LondonEstonian Genome Center, Institute of Genomics, University of TartuGenetics and Personalized Medicine Clinic, Tartu University HospitalGenetics and Personalized Medicine Clinic, Tartu University HospitalEstonian Genome Center, Institute of Genomics, University of TartuQIMR Berghofer Medical Research InstituteCentral Research Laboratory, Kazan State Medical UniversityDivision of Research, Kaiser Permanente Northern CaliforniaDivision of Research, Kaiser Permanente Northern CaliforniaQIMR Berghofer Medical Research InstituteDepartment of Epidemiology, University Medical Center Groningen, University of GroningenEstonian Genome Center, Institute of Genomics, University of TartuEye Clinic, Tartu University HospitalSchool of Optometry & Vision Sciences, Cardiff UniversityLions Eye Institute, Centre for Ophthalmology and Visual Science, University of Western AustraliaDepartment of Ophthalmology, Boston Children’s Hospital and Harvard Medical SchoolDivision of Research, Kaiser Permanente Northern CaliforniaQIMR Berghofer Medical Research InstituteAbstract Strabismus is a common pediatric eye misalignment and has complex genetic and environmental causes. Previous genome-wide association studies (GWAS) encountered difficulties in identifying strabismus risk variants due to heterogeneity and small samples. We performed large meta-analyses of 11 European-ancestry GWAS (7 sources), analysing broad strabismus (20,464 cases, 954,921 controls) and subtypes (esotropia/exotropia). We discovered 4 loci (e.g., NPLOC4-TSPAN10-PDE6G-FAAP100, COL6A1) for strabismus and 5 additional loci (e.g., CHRNA4, MAD1L1) for strabismus subtypes and we successfully replicated the previously reported strabismus variant near NPLOC4-TSPAN10-PDE6G-FAAP100. Using mendelian randomisation, we found genetic evidence supporting maternal smoking as a causal risk factor for strabismus in offspring.https://doi.org/10.1038/s41467-025-62456-9
spellingShingle Weixiong He
Peter J. van der Most
Jue-Sheng Ong
Liang-Dar Hwang
Yeda Wu
Morten S. Magnø
Jelle Vehof
Kristi Krebs
Laura Mauring
Katrin Õunap
Erik Abner
Nicholas G. Martin
Denis Plotnikov
Chen Jiang
Ronald B. Melles
Puya Gharahkhani
Harold Snieder
Teele Palumaa
Kuldar Kaljurand
Jeremy A. Guggenheim
David A. Mackey
Elizabeth C. Engle
Hélène Choquet
Lifelines Cohort Study
Stuart MacGregor
Large-scale GWAS of strabismus identifies risk loci and provides support for a link with maternal smoking
Nature Communications
title Large-scale GWAS of strabismus identifies risk loci and provides support for a link with maternal smoking
title_full Large-scale GWAS of strabismus identifies risk loci and provides support for a link with maternal smoking
title_fullStr Large-scale GWAS of strabismus identifies risk loci and provides support for a link with maternal smoking
title_full_unstemmed Large-scale GWAS of strabismus identifies risk loci and provides support for a link with maternal smoking
title_short Large-scale GWAS of strabismus identifies risk loci and provides support for a link with maternal smoking
title_sort large scale gwas of strabismus identifies risk loci and provides support for a link with maternal smoking
url https://doi.org/10.1038/s41467-025-62456-9
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