Cargando…
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...
Autores principales: | , , , , , |
---|---|
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 |
_version_ | 1784589791397937152 |
---|---|
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. |
format | Online Article Text |
id | pubmed-8544327 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2021 |
publisher | Impact Journals |
record_format | MEDLINE/PubMed |
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 |
work_keys_str_mv | AT xumenghan anovelmutationinpck2genecausesprimaryangleclosureglaucoma AT yangjin anovelmutationinpck2genecausesprimaryangleclosureglaucoma AT sunjiayue anovelmutationinpck2genecausesprimaryangleclosureglaucoma AT xingxuemei anovelmutationinpck2genecausesprimaryangleclosureglaucoma AT liuzheng anovelmutationinpck2genecausesprimaryangleclosureglaucoma AT liutao anovelmutationinpck2genecausesprimaryangleclosureglaucoma AT xumenghan novelmutationinpck2genecausesprimaryangleclosureglaucoma AT yangjin novelmutationinpck2genecausesprimaryangleclosureglaucoma AT sunjiayue novelmutationinpck2genecausesprimaryangleclosureglaucoma AT xingxuemei novelmutationinpck2genecausesprimaryangleclosureglaucoma AT liuzheng novelmutationinpck2genecausesprimaryangleclosureglaucoma AT liutao novelmutationinpck2genecausesprimaryangleclosureglaucoma |