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A novel mutation in PCK2 gene causes primary angle-closure glaucoma

Primary angle-closure glaucoma (PACG) is an ophthalmic genetic disease characterized by direct contact between the iris and trabecular meshwork, resulting in an obstructed outflow of aqueous humor from the eye. However, it is unclear as to what role genetics plays in the development of PACG. The pre...

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Autores principales: Xu, Menghan, Yang, Jin, Sun, Jiayue, Xing, Xuemei, Liu, Zheng, Liu, Tao
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Impact Journals 2021
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8544327/
https://www.ncbi.nlm.nih.gov/pubmed/34650006
http://dx.doi.org/10.18632/aging.203627
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author Xu, Menghan
Yang, Jin
Sun, Jiayue
Xing, Xuemei
Liu, Zheng
Liu, Tao
author_facet Xu, Menghan
Yang, Jin
Sun, Jiayue
Xing, Xuemei
Liu, Zheng
Liu, Tao
author_sort Xu, Menghan
collection PubMed
description Primary angle-closure glaucoma (PACG) is an ophthalmic genetic disease characterized by direct contact between the iris and trabecular meshwork, resulting in an obstructed outflow of aqueous humor from the eye. However, it is unclear as to what role genetics plays in the development of PACG. The present study investigated the disease-causing mutation in a five-generation Chinese PACG family using whole-genome sequencing. A novel heterozygous missense mutation c.977C>T in PCK2 gene was identified in five affected family members, but not in any unaffected and 86 unrelated healthy individuals. This nucleotide substitute is predicted to result in a proline to leucine substitution p.Pro326Leu. Furthermore, the function of this mutation was analyzed through various in vitro assays using the RGC-5 cell line. Our results demonstrate that the p.Pro326Leu mutation induces RGC-5 cell cycle arrest and apoptosis with a decreased BcL-XL. The increasing P53, P27, P21, AKT, and P-GSK3α were also detected in the cells transfected with c.977C>T mutation, suggesting that this mutation within PCK2 gene cause PACG through impairment of AKT/GSK3α signaling pathway.
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spelling pubmed-85443272021-10-26 A novel mutation in PCK2 gene causes primary angle-closure glaucoma Xu, Menghan Yang, Jin Sun, Jiayue Xing, Xuemei Liu, Zheng Liu, Tao Aging (Albany NY) Research Paper Primary angle-closure glaucoma (PACG) is an ophthalmic genetic disease characterized by direct contact between the iris and trabecular meshwork, resulting in an obstructed outflow of aqueous humor from the eye. However, it is unclear as to what role genetics plays in the development of PACG. The present study investigated the disease-causing mutation in a five-generation Chinese PACG family using whole-genome sequencing. A novel heterozygous missense mutation c.977C>T in PCK2 gene was identified in five affected family members, but not in any unaffected and 86 unrelated healthy individuals. This nucleotide substitute is predicted to result in a proline to leucine substitution p.Pro326Leu. Furthermore, the function of this mutation was analyzed through various in vitro assays using the RGC-5 cell line. Our results demonstrate that the p.Pro326Leu mutation induces RGC-5 cell cycle arrest and apoptosis with a decreased BcL-XL. The increasing P53, P27, P21, AKT, and P-GSK3α were also detected in the cells transfected with c.977C>T mutation, suggesting that this mutation within PCK2 gene cause PACG through impairment of AKT/GSK3α signaling pathway. Impact Journals 2021-10-14 /pmc/articles/PMC8544327/ /pubmed/34650006 http://dx.doi.org/10.18632/aging.203627 Text en Copyright: © 2021 Xu et al. https://creativecommons.org/licenses/by/3.0/This is an open access article distributed under the terms of the Creative Commons Attribution License (https://creativecommons.org/licenses/by/3.0/) (CC BY 3.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited.
spellingShingle Research Paper
Xu, Menghan
Yang, Jin
Sun, Jiayue
Xing, Xuemei
Liu, Zheng
Liu, Tao
A novel mutation in PCK2 gene causes primary angle-closure glaucoma
title A novel mutation in PCK2 gene causes primary angle-closure glaucoma
title_full A novel mutation in PCK2 gene causes primary angle-closure glaucoma
title_fullStr A novel mutation in PCK2 gene causes primary angle-closure glaucoma
title_full_unstemmed A novel mutation in PCK2 gene causes primary angle-closure glaucoma
title_short A novel mutation in PCK2 gene causes primary angle-closure glaucoma
title_sort novel mutation in pck2 gene causes primary angle-closure glaucoma
topic Research Paper
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8544327/
https://www.ncbi.nlm.nih.gov/pubmed/34650006
http://dx.doi.org/10.18632/aging.203627
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