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Exploiting the Autozygome to Support Previously Published Mendelian Gene-Disease Associations: An Update.

Authors:
Sateesh Maddirevula Hanan E Shamseldin Amy Sirr Lama AlAbdi Russell S Lo Nour Ewida Mashael Al-Qahtani Mais Hashem Firdous Abdulwahab Omar Aboyousef Namik Kaya Dorota Monies May H Salem Naffaa Al Harbi Hesham M Aldhalaan Hamad Alzaidan Hadeel M Almanea Abrar K Alsalamah Fuad Al Mutairi Samira Ismail Ghada M H Abdel-Salam Amal Alhashem Ali Asery Eissa Faqeih Amal AlQassmi Waleed Al-Hamoudi Talal Algoufi Mohammad Shagrani Aimée M Dudley Fowzan S Alkuraya

Front Genet 2020 31;11:580484. Epub 2020 Dec 31.

Department of Genetics, King Faisal Specialist Hospital and Research Center, Riyadh, Saudi Arabia.

There is a growing interest in standardizing gene-disease associations for the purpose of facilitating the proper classification of variants in the context of Mendelian diseases. One key line of evidence is the independent observation of pathogenic variants in unrelated individuals with similar phenotypes. Here, we expand on our previous effort to exploit the power of autozygosity to produce homozygous pathogenic variants that are otherwise very difficult to encounter in the homozygous state due to their rarity. The identification of such variants in genes with only tentative associations to Mendelian diseases can add to the existing evidence when observed in the context of compatible phenotypes. In this study, we report 20 homozygous variants in 18 genes (, and ) that satisfy the ACMG classification for pathogenic/likely pathogenic if the involved genes had confirmed rather than tentative links to diseases. These variants were selected because they were truncating, founder with compelling segregation or supported by robust functional assays as with the variant that we present its validation using yeast model. Our findings support the previously reported disease associations for these genes and represent a step toward their confirmation.

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http://dx.doi.org/10.3389/fgene.2020.580484DOI Listing
http://www.ncbi.nlm.nih.gov/pmc/articles/PMC7806527PMC
December 2020

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