Abstract
Since apoE allele status is the predominant Alzheimer's disease (AD) genetic risk factor, functional single nucleotide polymorphisms (SNPs) in brain apoE receptors represent excellent candidates for association with AD. Recently, we identified a SNP, rs688, as modulating the splicing efficiency of low-density lipoprotein receptor (LDLR) exon 12 in female human liver and in minigene-transfected HepG2 cells. Moreover, the rs688T minor allele was associated with significantly higher LDL and total cholesterol in women within the Framingham Offspring Study cohort. Since LDLR is a major apoE receptor in the brain, we hypothesized that rs688 modulates LDLR splicing in neural tissues and associates with AD. To evaluate this hypothesis, we first transfected LDLR minigenes into SH-SY5Y neuroblastoma cells and found that the rs688T allele reduces exon 12 inclusion in this neural model. We then evaluated the association of rs688 allele with exon 12 splicing efficiency in vivo by quantifying LDLR splicing in human anterior cingulate tissue obtained at autopsy; the rs688T allele is associated with decreased LDLR exon 12 splicing efficiency in aged males, but not females. Lastly, we evaluated whether rs688 associates with AD by genotyping DNA from 1457 men and 2055 women drawn from three case-control series. The rs688T/T genotype was associated with increased AD odds in males [recessive model, odds ratio (OR) of 1.49, 95% confidence interval (CI) of 1.13-1.97, uncorrected P = 0.005], but not in females. In summary, these studies identify a functional apoE receptor SNP that is associated with AD in a sex-dependent fashion.
MeSH Terms
Aged
Aged, 80 and over
Alzheimer Disease/genetics,metabolism
Apolipoproteins E/metabolism
Brain/metabolism
Case-Control Studies
Cell Line, Tumor
Exons
Female
Humans
Male
Mutagenesis, Site-Directed
Odds Ratio
Polymorphism, Single Nucleotide
RNA Splicing
Receptors, LDL/genetics
Reverse Transcriptase Polymerase Chain Reaction
Sex Characteristics
Chemicals
Apolipoproteins E
Receptors, LDL
Authors & Affiliations
16 authors, click to expand affiliations / ORCID
Zou Fanggeng
Department of Neuroscience, Mayo Clinic, 4500 San Pablo Road, Jacksonville, FL 32224, USA.
Gopalraj Rangaraj K
Lok Johann
Zhu Haiyan
Ling I-Fang
Simpson James F
Tucker H Michael
Kelly Jeremiah F
Younkin Samuel G
Dickson Dennis W
Petersen Ronald C
Graff-Radford Neill R
Bennett David A
Crook Julia E
Younkin Steven G
Estus Steven
References (26)
26 references, click to expand
-
Linking C5 deficiency to an exonic splicing enhancer mutation.
J Immunol. 2005 Apr 1;174(7):4172-7
PMID: 15778377
-
Molecular genetics of the LDL receptor gene in familial hypercholesterolemia.
Hum Mutat. 1992;1(6):445-66
PMID: 1301956
-
Joe Goldstein and Mike Brown: from cholesterol homeostasis to new paradigms in membrane biology.
Trends Cell Biol. 2003 Oct;13(10):534-9
PMID: 14507481
-
A common polymorphism decreases low-density lipoprotein receptor exon 12 splicing efficiency and associates with increased cholesterol.
Hum Mol Genet. 2007 Jul 15;16(14):1765-72
PMID: 17517690
-
The expression of several mitochondrial and nuclear genes encoding the subunits of electron transport chain enzyme complexes, cytochrome c oxidase, and NADH dehydrogenase, in different brain regions in Alzheimer's disease.
Neurochem Res. 1999 Jun;24(6):767-74
PMID: 10447460
-
Genetic association of low density lipoprotein receptor and Alzheimer's disease.
Neurobiol Aging. 2005 Jan;26(1):1-7
PMID: 15585340
-
Clinical diagnosis of Alzheimer's disease: report of the NINCDS-ADRDA Work Group under the auspices of Department of Health and Human Services Task Force on Alzheimer's Disease.
Neurology. 1984 Jul;34(7):939-44
PMID: 6610841
-
Pre-mRNA splicing and human disease.
Genes Dev. 2003 Feb 15;17(4):419-37
PMID: 12600935
-
Lack of LDL receptor aggravates learning deficits and amyloid deposits in Alzheimer transgenic mice.
Neurobiol Aging. 2006 Nov;27(11):1632-43
PMID: 16236385
-
Single-step method of RNA isolation by acid guanidinium thiocyanate-phenol-chloroform extraction.
Anal Biochem. 1987 Apr;162(1):156-9
PMID: 2440339
-
Cholesterol paradox: is high total or low HDL cholesterol level a risk for Alzheimer's disease?
J Neurosci Res. 2003 Apr 15;72(2):141-6
PMID: 12671988
-
Primary hypercholesterolemia: genetic causes and treatment of five monogenic disorders.
Expert Rev Cardiovasc Ther. 2003 May;1(1):107-19
PMID: 15030301
-
No association between low density lipoprotein receptor genetic variants and Alzheimer's disease risk.
Am J Med Genet B Neuropsychiatr Genet. 2006 Jul 5;141B(5):541-3
PMID: 16741934
-
The Lebanese allele at the low density lipoprotein receptor locus. Nonsense mutation produces truncated receptor that is retained in endoplasmic reticulum.
J Biol Chem. 1987 Jan 5;262(1):401-10
PMID: 3025214
-
Apolipoprotein E facilitates neuritic and cerebrovascular plaque formation in an Alzheimer's disease model.
Ann Neurol. 2000 Jun;47(6):739-47
PMID: 10852539
-
Potential genetic markers of sporadic Alzheimer's dementia.
Psychiatr Genet. 2001 Sep;11(3):115-22
PMID: 11702052
-
Alzheimer's disease: the cholesterol connection.
Nat Neurosci. 2003 Apr;6(4):345-51
PMID: 12658281
-
The role of common single-nucleotide polymorphisms on exon 9 and exon 12 skipping in nonmutated CFTR alleles.
Hum Mutat. 2004 Aug;24(2):120-9
PMID: 15241793
-
Effects of gender and menopausal status on the association of apolipoprotein E phenotype with plasma lipoprotein levels. Results from the Framingham Offspring Study.
Arterioscler Thromb. 1994 Jul;14(7):1105-13
PMID: 8018666
-
The low density lipoprotein receptor regulates the level of central nervous system human and murine apolipoprotein E but does not modify amyloid plaque pathology in PDAPP mice.
J Biol Chem. 2005 Jul 8;280(27):25754-9
PMID: 15888448
-
Lack of association of hepatic lipase polymorphisms with late-onset Alzheimer's disease.
Neurobiol Aging. 2008 May;29(5):793-4
PMID: 17175070
-
The neuronal sortilin-related receptor SORL1 is genetically associated with Alzheimer disease.
Nat Genet. 2007 Feb;39(2):168-77
PMID: 17220890
-
Functional interaction between APOE4 and LDL receptor isoforms in Alzheimer's disease.
J Med Genet. 2005 Feb;42(2):129-31
PMID: 15689450
-
The LDLR locus in Alzheimer's disease: a family-based study and meta-analysis of case-control data.
Neurobiol Aging. 2007 Jan;28(1):18.e1-4
PMID: 16378661
-
Disruption of exonic splicing enhancer elements is the principal cause of exon skipping associated with seven nonsense or missense alleles of NF1.
Hum Mutat. 2004 Dec;24(6):491-501
PMID: 15523642
-
Elevated amyloid beta protein (Abeta42) and late onset Alzheimer's disease are associated with single nucleotide polymorphisms in the urokinase-type plasminogen activator gene.
Hum Mol Genet. 2005 Feb 1;14(3):447-60
PMID: 15615772