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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...

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Autores principales: Shu, Hongyan, Chi, Jingwei, Li, Jing, Zhang, Wei, Lv, Wenshan, Wang, Jie, Deng, Yujie, Hou, Xu, Wang, Yangang
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
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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.
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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
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