Rare variant association analysis in case-parents studies by allowing for missing parental genotypes

Abstract Background The development of next-generation sequencing technologies has facilitated the identification of rare variants. Family-based design is commonly used to effectively control for population admixture and substructure, which is more prominent for rare variants. Case-parents studies,...

Full description

Bibliographic Details
Main Authors: Yumei Li, Yang Xiang, Chao Xu, Hui Shen, Hongwen Deng
Format: Article
Language:English
Published: BMC 2018-01-01
Series:BMC Genetics
Subjects:
Online Access:http://link.springer.com/article/10.1186/s12863-018-0597-8
_version_ 1828528193401782272
author Yumei Li
Yang Xiang
Chao Xu
Hui Shen
Hongwen Deng
author_facet Yumei Li
Yang Xiang
Chao Xu
Hui Shen
Hongwen Deng
author_sort Yumei Li
collection DOAJ
description Abstract Background The development of next-generation sequencing technologies has facilitated the identification of rare variants. Family-based design is commonly used to effectively control for population admixture and substructure, which is more prominent for rare variants. Case-parents studies, as typical strategies in family-based design, are widely used in rare variant-disease association analysis. Current methods in case-parents studies are based on complete case-parents data; however, parental genotypes may be missing in case-parents trios, and removing these data may lead to a loss in statistical power. The present study focuses on testing for rare variant-disease association in case-parents study by allowing for missing parental genotypes. Results In this report, we extended the collapsing method for rare variant association analysis in case-parents studies to allow for missing parental genotypes, and investigated the performance of two methods by using the difference of genotypes between affected offspring and their corresponding “complements” in case-parent trios and TDT framework. Using simulations, we showed that, compared with the methods just only using complete case-parents data, the proposed strategy allowing for missing parental genotypes, or even adding unrelated affected individuals, can greatly improve the statistical power and meanwhile is not affected by population stratification. Conclusions We conclude that adding case-parents data with missing parental genotypes to complete case-parents data set can greatly improve the power of our strategy for rare variant-disease association.
first_indexed 2024-12-11T21:48:52Z
format Article
id doaj.art-779486edd3c54718afb53bfc18d93035
institution Directory Open Access Journal
issn 1471-2156
language English
last_indexed 2024-12-11T21:48:52Z
publishDate 2018-01-01
publisher BMC
record_format Article
series BMC Genetics
spelling doaj.art-779486edd3c54718afb53bfc18d930352022-12-22T00:49:32ZengBMCBMC Genetics1471-21562018-01-011911810.1186/s12863-018-0597-8Rare variant association analysis in case-parents studies by allowing for missing parental genotypesYumei Li0Yang Xiang1Chao Xu2Hui Shen3Hongwen Deng4School of Mathematics and Computational Science, Huaihua UniversitySchool of Mathematics and Computational Science, Huaihua UniversityCenter for Bioinformatics and Genomics, Department of Global Biostatistics and Data Science, Tulane UniversityCenter for Bioinformatics and Genomics, Department of Global Biostatistics and Data Science, Tulane UniversityCenter for Bioinformatics and Genomics, Department of Global Biostatistics and Data Science, Tulane UniversityAbstract Background The development of next-generation sequencing technologies has facilitated the identification of rare variants. Family-based design is commonly used to effectively control for population admixture and substructure, which is more prominent for rare variants. Case-parents studies, as typical strategies in family-based design, are widely used in rare variant-disease association analysis. Current methods in case-parents studies are based on complete case-parents data; however, parental genotypes may be missing in case-parents trios, and removing these data may lead to a loss in statistical power. The present study focuses on testing for rare variant-disease association in case-parents study by allowing for missing parental genotypes. Results In this report, we extended the collapsing method for rare variant association analysis in case-parents studies to allow for missing parental genotypes, and investigated the performance of two methods by using the difference of genotypes between affected offspring and their corresponding “complements” in case-parent trios and TDT framework. Using simulations, we showed that, compared with the methods just only using complete case-parents data, the proposed strategy allowing for missing parental genotypes, or even adding unrelated affected individuals, can greatly improve the statistical power and meanwhile is not affected by population stratification. Conclusions We conclude that adding case-parents data with missing parental genotypes to complete case-parents data set can greatly improve the power of our strategy for rare variant-disease association.http://link.springer.com/article/10.1186/s12863-018-0597-8Rare-variant association analysisCase-parent triosCollapsing method
spellingShingle Yumei Li
Yang Xiang
Chao Xu
Hui Shen
Hongwen Deng
Rare variant association analysis in case-parents studies by allowing for missing parental genotypes
BMC Genetics
Rare-variant association analysis
Case-parent trios
Collapsing method
title Rare variant association analysis in case-parents studies by allowing for missing parental genotypes
title_full Rare variant association analysis in case-parents studies by allowing for missing parental genotypes
title_fullStr Rare variant association analysis in case-parents studies by allowing for missing parental genotypes
title_full_unstemmed Rare variant association analysis in case-parents studies by allowing for missing parental genotypes
title_short Rare variant association analysis in case-parents studies by allowing for missing parental genotypes
title_sort rare variant association analysis in case parents studies by allowing for missing parental genotypes
topic Rare-variant association analysis
Case-parent trios
Collapsing method
url http://link.springer.com/article/10.1186/s12863-018-0597-8
work_keys_str_mv AT yumeili rarevariantassociationanalysisincaseparentsstudiesbyallowingformissingparentalgenotypes
AT yangxiang rarevariantassociationanalysisincaseparentsstudiesbyallowingformissingparentalgenotypes
AT chaoxu rarevariantassociationanalysisincaseparentsstudiesbyallowingformissingparentalgenotypes
AT huishen rarevariantassociationanalysisincaseparentsstudiesbyallowingformissingparentalgenotypes
AT hongwendeng rarevariantassociationanalysisincaseparentsstudiesbyallowingformissingparentalgenotypes