Cargando…
A novel indel variant in LDLR responsible for familial hypercholesterolemia in a Chinese family
Familial hypercholesterolemia (FH) is an inherited disorder characterized by elevation of serum cholesterol bound to low-density lipoprotein. Mutations in LDLR are the major factors responsible for FH. In this study, we recruited a four-generation Chinese family with FH and identified the clinical f...
Autores principales: | , , , , , , , , |
---|---|
Formato: | Online Artículo Texto |
Lenguaje: | English |
Publicado: |
Public Library of Science
2017
|
Materias: | |
Acceso en línea: | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5724832/ https://www.ncbi.nlm.nih.gov/pubmed/29228028 http://dx.doi.org/10.1371/journal.pone.0189316 |
_version_ | 1783285423014936576 |
---|---|
author | Shu, Hongyan Chi, Jingwei Li, Jing Zhang, Wei Lv, Wenshan Wang, Jie Deng, Yujie Hou, Xu Wang, Yangang |
author_facet | Shu, Hongyan Chi, Jingwei Li, Jing Zhang, Wei Lv, Wenshan Wang, Jie Deng, Yujie Hou, Xu Wang, Yangang |
author_sort | Shu, Hongyan |
collection | PubMed |
description | Familial hypercholesterolemia (FH) is an inherited disorder characterized by elevation of serum cholesterol bound to low-density lipoprotein. Mutations in LDLR are the major factors responsible for FH. In this study, we recruited a four-generation Chinese family with FH and identified the clinical features of hypercholesterolemia. All affected individuals shared a novel indel mutation (c.1885_1889delinsGATCATCAACC) in exon 13 of LDLR. The mutation segregated with the hypercholesterolemia phenotype in the family. To analyze the function of the indel, we established stable clones of mutant and wild-type LDLR in Hep G2 cells. The mutant LDLR was retained in the endoplasmic reticulum (ER) and failed to glycosylate via the Golgi. Moreover, the membrane LDLR was reduced and lost the ability to take up LDL. Our data also expand the spectrum of known LDLR mutations. |
format | Online Article Text |
id | pubmed-5724832 |
institution | National Center for Biotechnology Information |
language | English |
publishDate | 2017 |
publisher | Public Library of Science |
record_format | MEDLINE/PubMed |
spelling | pubmed-57248322017-12-15 A novel indel variant in LDLR responsible for familial hypercholesterolemia in a Chinese family Shu, Hongyan Chi, Jingwei Li, Jing Zhang, Wei Lv, Wenshan Wang, Jie Deng, Yujie Hou, Xu Wang, Yangang PLoS One Research Article Familial hypercholesterolemia (FH) is an inherited disorder characterized by elevation of serum cholesterol bound to low-density lipoprotein. Mutations in LDLR are the major factors responsible for FH. In this study, we recruited a four-generation Chinese family with FH and identified the clinical features of hypercholesterolemia. All affected individuals shared a novel indel mutation (c.1885_1889delinsGATCATCAACC) in exon 13 of LDLR. The mutation segregated with the hypercholesterolemia phenotype in the family. To analyze the function of the indel, we established stable clones of mutant and wild-type LDLR in Hep G2 cells. The mutant LDLR was retained in the endoplasmic reticulum (ER) and failed to glycosylate via the Golgi. Moreover, the membrane LDLR was reduced and lost the ability to take up LDL. Our data also expand the spectrum of known LDLR mutations. Public Library of Science 2017-12-11 /pmc/articles/PMC5724832/ /pubmed/29228028 http://dx.doi.org/10.1371/journal.pone.0189316 Text en © 2017 Shu et al http://creativecommons.org/licenses/by/4.0/ This is an open access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/4.0/) , which permits unrestricted use, distribution, and reproduction in any medium, provided the original author and source are credited. |
spellingShingle | Research Article Shu, Hongyan Chi, Jingwei Li, Jing Zhang, Wei Lv, Wenshan Wang, Jie Deng, Yujie Hou, Xu Wang, Yangang A novel indel variant in LDLR responsible for familial hypercholesterolemia in a Chinese family |
title | A novel indel variant in LDLR responsible for familial hypercholesterolemia in a Chinese family |
title_full | A novel indel variant in LDLR responsible for familial hypercholesterolemia in a Chinese family |
title_fullStr | A novel indel variant in LDLR responsible for familial hypercholesterolemia in a Chinese family |
title_full_unstemmed | A novel indel variant in LDLR responsible for familial hypercholesterolemia in a Chinese family |
title_short | A novel indel variant in LDLR responsible for familial hypercholesterolemia in a Chinese family |
title_sort | novel indel variant in ldlr responsible for familial hypercholesterolemia in a chinese family |
topic | Research Article |
url | https://www.ncbi.nlm.nih.gov/pmc/articles/PMC5724832/ https://www.ncbi.nlm.nih.gov/pubmed/29228028 http://dx.doi.org/10.1371/journal.pone.0189316 |
work_keys_str_mv | AT shuhongyan anovelindelvariantinldlrresponsibleforfamilialhypercholesterolemiainachinesefamily AT chijingwei anovelindelvariantinldlrresponsibleforfamilialhypercholesterolemiainachinesefamily AT lijing anovelindelvariantinldlrresponsibleforfamilialhypercholesterolemiainachinesefamily AT zhangwei anovelindelvariantinldlrresponsibleforfamilialhypercholesterolemiainachinesefamily AT lvwenshan anovelindelvariantinldlrresponsibleforfamilialhypercholesterolemiainachinesefamily AT wangjie anovelindelvariantinldlrresponsibleforfamilialhypercholesterolemiainachinesefamily AT dengyujie anovelindelvariantinldlrresponsibleforfamilialhypercholesterolemiainachinesefamily AT houxu anovelindelvariantinldlrresponsibleforfamilialhypercholesterolemiainachinesefamily AT wangyangang anovelindelvariantinldlrresponsibleforfamilialhypercholesterolemiainachinesefamily AT shuhongyan novelindelvariantinldlrresponsibleforfamilialhypercholesterolemiainachinesefamily AT chijingwei novelindelvariantinldlrresponsibleforfamilialhypercholesterolemiainachinesefamily AT lijing novelindelvariantinldlrresponsibleforfamilialhypercholesterolemiainachinesefamily AT zhangwei novelindelvariantinldlrresponsibleforfamilialhypercholesterolemiainachinesefamily AT lvwenshan novelindelvariantinldlrresponsibleforfamilialhypercholesterolemiainachinesefamily AT wangjie novelindelvariantinldlrresponsibleforfamilialhypercholesterolemiainachinesefamily AT dengyujie novelindelvariantinldlrresponsibleforfamilialhypercholesterolemiainachinesefamily AT houxu novelindelvariantinldlrresponsibleforfamilialhypercholesterolemiainachinesefamily AT wangyangang novelindelvariantinldlrresponsibleforfamilialhypercholesterolemiainachinesefamily |