Linkage of a gene for familial hypobetalipoproteinemia to chromosome 3p21.1-22.

Familial hypobetalipoproteinemia (FHBL) is an apparently autosomal dominant disorder of lipid metabolism characterized by less than fifth percentile age- and sex-specific levels of apolipoprotein beta (apobeta) and low-density lipoprotein-cholesterol. In a minority of cases, FHBL is due to truncation-producing mutations in the apobeta gene on chromosome 2p23-24. Previously, we reported on a four-generation FHBL kindred in which we had ruled out linkage of the trait to the apobeta gene. To locate other loci containing genes for low apobeta levels in the kindred, a genomewide search was conducted. Regions on 3p21.1-22 with two-point LOD scores >1.5 were identified. Additional markers were typed in the region of these signals. Two-point LOD scores in the region of D3S2407 increased to 3.35 at O = 0. GENEHUNTER confirmed this finding with an nonparametric multipoint LOD score of 7.5 (P=.0004). Additional model-free analyses were conducted with the square root of the apobeta level as the phenotype. Results from the Loki and SOLAR programs further confirmed linkage of FHBL to 3p21.1-22. Weaker linkage to a region near D19S916 was also indicated by Loki and SOLAR. Thus, a heretofore unidentified genetic susceptibility locus for FHBL may reside on chromosome 3.

[1]  P. Kwok,et al.  Known mutations of apoB account for only a small minority of hypobetalipoproteinemia. , 1999, Journal of Lipid Research.

[2]  E. Wijsman,et al.  Multipoint oligogenic analysis of age-at-onset data with applications to Alzheimer disease pedigrees. , 1999, American journal of human genetics.

[3]  E. Keeffe,et al.  Asymptomatic elevation of aminotransferase levels and fatty liver secondary to heterozygous hypobetalipoproteinemia , 1998, American Journal of Gastroenterology.

[4]  P. Wilson,et al.  Frequency of ApoB and ApoE gene mutations as causes of hypobetalipoproteinemia in the framingham offspring population. , 1998, Arteriosclerosis, thrombosis, and vascular biology.

[5]  L. Almasy,et al.  Multipoint quantitative-trait linkage analysis in general pedigrees. , 1998, American journal of human genetics.

[6]  Jingshi Wu,et al.  Genetic heterogeneity in familial hypobetalipoproteinemia: linkage and non-linkage to the apoB gene in Caucasian families. , 1998, American journal of medical genetics.

[7]  S. Heath Markov chain Monte Carlo segregation and linkage analysis for oligogenic models. , 1997, American journal of human genetics.

[8]  A. Lonardo,et al.  Fatty liver in heterozygous hypobetalipoproteinemia caused by a novel truncated form of apolipoprotein B. , 1996, Gastroenterology.

[9]  L Kruglyak,et al.  Parametric and nonparametric linkage analysis: a unified multipoint approach. , 1996, American journal of human genetics.

[10]  K Lange,et al.  Descent graphs in pedigree analysis: applications to haplotyping, location scores, and marker-sharing statistics. , 1996, American journal of human genetics.

[11]  A A Schäffer,et al.  Faster sequential genetic linkage computations. , 1993, American journal of human genetics.

[12]  Robert V Farese,et al.  Apolipoprotein B gene mutations affecting cholesterol levels , 1992, Journal of internal medicine.

[13]  S. Giampaoli,et al.  A form of familial hypobetalipoproteinaemia not due to a mutation in the apolipoprotein B gene , 1991, Journal of internal medicine.

[14]  H. Hobbs,et al.  Evidence for a dominant gene that suppresses hypercholesterolemia in a family with defective low density lipoprotein receptors. , 1989, The Journal of clinical investigation.

[15]  D. Levy,et al.  Cholesterol and mortality. 30 years of follow-up from the Framingham study. , 1987, JAMA.

[16]  J. Ott,et al.  Strategies for multilocus linkage analysis in humans. , 1984, Proceedings of the National Academy of Sciences of the United States of America.

[17]  J. Polonovski,et al.  [Structure and metabolism of plasma lipoproteins]. , 1983, Pathologie-biologie.

[18]  P. Wilson,et al.  Reference intervals for plasma apolipoprotein B determined with a standardized commercial immunoturbidimetric assay: results from the Framingham Offspring Study. , 1996, Clinical chemistry.

[19]  G. Schonfeld The hypobetalipoproteinemias. , 1995, Annual review of nutrition.

[20]  Charles R.scriver,et al.  The Metabolic basis of inherited disease , 1989 .