Literature DB >> 30112042

Novel compound heterozygous mutations in low density lipoprotein receptor gene causes a severe phenotype in a Chinese hypercholesterolemia family.

Xinyao Cheng1, Yifang Huang2, Xueping Qiu2, Xiaohuan Cheng2, Yalei Jin3, Yafei Hu4, Bing Yang4, Jingbo Zhao4, Yuhua Lei4, Fang Zheng2.   

Abstract

Mutations in the low density lipoprotein receptor (LDLR) gene serve a causative role in the pathophysiology of familial hypercholesterolemia (FH), a common autosomal inherited disorder characterized by abnormal lipid metabolism. The aim of the present study was to investigate genetic defects in a Chinese family with FH. Clinical features and family histories were collected, as were the results of various laboratory tests, including determinations of serum lipid concentrations, ultrasonography and angiography results. Potential mutations in LDLR were screened using direct polymerase chain reaction (PCR) sequencing. Multiple sequence alignments, structure and hydrophobicity predictions were performed in silico. Novel compound heterozygote mutations in LDLR of the proband were identified, with a Trp577Term-bearing maternal allele and a Pro685Leu-bearing paternal allele. The proband, a 27-year-old male, had severe and diffuse coronary stenosis and non-ST segment elevation myocardial infarction, as well as multiple skin xanthomas and high serum lipid levels. The allele-dosage-dependent clinical features, including hypercholesterolemia and peripheral arterial atherosclerosis, were observed in the proband and the other heterozygous patients. Therefore, the coexistence of Pro685Leu and Trp577Term mutations in LDLR is a novel compound heterozygosis in Chinese patients and may lead to a severe FH phenotype. The explanation for the existence of compound heterozygous mutations instead of homozygous mutations in this particular family requires further study.

Entities:  

Keywords:  LDLR gene; atherosclerosis; coronary artery disease familial hypercholesterolemia; xanthomas

Year:  2018        PMID: 30112042      PMCID: PMC6090432          DOI: 10.3892/etm.2018.6205

Source DB:  PubMed          Journal:  Exp Ther Med        ISSN: 1792-0981            Impact factor:   2.447


  29 in total

Review 1.  Monogenic hypercholesterolemia: new insights in pathogenesis and treatment.

Authors:  Daniel J Rader; Jonathan Cohen; Helen H Hobbs
Journal:  J Clin Invest       Date:  2003-06       Impact factor: 14.808

2.  Genetic risk for metabolic syndrome: examination of candidate gene polymorphisms related to lipid metabolism in Japanese people.

Authors:  Y Yamada; S Ichihara; K Kato; T Yoshida; K Yokoi; H Matsuo; S Watanabe; N Metoki; H Yoshida; K Satoh; Y Aoyagi; A Yasunaga; H Park; M Tanaka; W Lee; Y Nozawa
Journal:  J Med Genet       Date:  2007-08-31       Impact factor: 6.318

3.  Rare intracranial cholesterol deposition and a homozygous mutation of LDLR in a familial hypercholesterolemia patient.

Authors:  Haoxian Li; Yanghui Zhang; Xianda Wei; Ying Peng; Pu Yang; Hu Tan; Chen Chen; Qian Pan; Desheng Liang; Lingqian Wu
Journal:  Gene       Date:  2015-04-29       Impact factor: 3.688

4.  Molecular basis of autosomal dominant hypercholesterolemia: assessment in a large cohort of hypercholesterolemic children.

Authors:  Anouk van der Graaf; Hans J Avis; D Meeike Kusters; Maud N Vissers; Barbara A Hutten; Joep C Defesche; Roeland Huijgen; Sigrid W Fouchier; Frits A Wijburg; John J P Kastelein; Albert Wiegman
Journal:  Circulation       Date:  2011-03-07       Impact factor: 29.690

5.  Genotypic and phenotypic features in homozygous familial hypercholesterolemia caused by proprotein convertase subtilisin/kexin type 9 (PCSK9) gain-of-function mutation.

Authors:  Hiroshi Mabuchi; Atsushi Nohara; Tohru Noguchi; Junji Kobayashi; Masa-aki Kawashiri; Takeshi Inoue; Mika Mori; Hayato Tada; Chiaki Nakanishi; Kunimasa Yagi; Masakazu Yamagishi; Kousei Ueda; Tadayoshi Takegoshi; Susumu Miyamoto; Akihiro Inazu; Junji Koizumi
Journal:  Atherosclerosis       Date:  2014-06-26       Impact factor: 5.162

Review 6.  Molecular genetics of the LDL receptor gene in familial hypercholesterolemia.

Authors:  H H Hobbs; M S Brown; J L Goldstein
Journal:  Hum Mutat       Date:  1992       Impact factor: 4.878

7.  Acid-dependent ligand dissociation and recycling of LDL receptor mediated by growth factor homology region.

Authors:  C G Davis; J L Goldstein; T C Südhof; R G Anderson; D W Russell; M S Brown
Journal:  Nature       Date:  1987 Apr 23-29       Impact factor: 49.962

8.  Association and linkage of LDLR gene variation with variation in plasma low density lipoprotein cholesterol.

Authors:  A P Boright; P W Connelly; J H Brunt; K Morgan; R A Hegele
Journal:  J Hum Genet       Date:  1998       Impact factor: 3.172

9.  Familial hypercholesterolemia in China. Identification of mutations in the LDL-receptor gene that result in a receptor-negative phenotype.

Authors:  X M Sun; D D Patel; J C Webb; B L Knight; L M Fan; H J Cai; A K Soutar
Journal:  Arterioscler Thromb       Date:  1994-01

Review 10.  Mechanisms of disease: genetic causes of familial hypercholesterolemia.

Authors:  Anne K Soutar; Rossi P Naoumova
Journal:  Nat Clin Pract Cardiovasc Med       Date:  2007-04
View more
  1 in total

1.  Prevalence of genetically defined familial hypercholesterolemia and the impact on acute myocardial infarction in Taiwanese population: A hospital-based study.

Authors:  Yen-Ju Chen; I-Chieh Chen; Yi-Ming Chen; Tzu-Hung Hsiao; Chia-Yi Wei; Han-Ni Chuang; Wei-Wen Lin; Ching-Heng Lin
Journal:  Front Cardiovasc Med       Date:  2022-09-12
  1 in total

北京卡尤迪生物科技股份有限公司 © 2022-2023.