Literature DB >> 8101042

Prenatal diagnosis and a donor splice site mutation in fibrillin in a family with Marfan syndrome.

M Godfrey1, N Vandemark, M Wang, M Velinov, D Wargowski, P Tsipouras, J Han, J Becker, W Robertson, S Droste.   

Abstract

The Marfan syndrome, an autosomal dominant connective tissue disorder, is manifested by abnormalities in the cardiovascular, skeletal, and ocular systems. Recently, fibrillin, an elastin-associated microfibrillar glycoprotein, has been linked to the Marfan syndrome, and fibrillin mutations in affected individuals have been documented. In this study, genetic linkage analysis with fibrillin specific markers was used to establish the prenatal diagnosis in an 11-wk-gestation fetus in a four-generation Marfan kindred. At birth, skeletal changes suggestive of the Marfan syndrome were observed. Reverse transcription-PCR amplification of the fibrillin gene mRNA detected a deletion of 123 bp in one allele in affected relatives. This deletion corresponds to an exon encoding an epidermal growth factor-like motif. Examination of genomic DNA showed a G-->C transversion at the +1 consensus donor splice site.

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Year:  1993        PMID: 8101042      PMCID: PMC1682369     

Source DB:  PubMed          Journal:  Am J Hum Genet        ISSN: 0002-9297            Impact factor:   11.025


  42 in total

1.  Clustering of fibrillin (FBN1) missense mutations in Marfan syndrome patients at cysteine residues in EGF-like domains.

Authors:  H C Dietz; J M Saraiva; R E Pyeritz; G R Cutting; C A Francomano
Journal:  Hum Mutat       Date:  1992       Impact factor: 4.878

2.  Identification of an altered splice site in Ashkenazi Tay-Sachs disease.

Authors:  E Arpaia; A Dumbrille-Ross; T Maler; K Neote; M Tropak; C Troxel; J L Stirling; J S Pitts; B Bapat; A M Lamhonwah
Journal:  Nature       Date:  1988-05-05       Impact factor: 49.962

3.  Severe Hb S-beta zero-thalassaemia with a T----C substitution in the donor splice site of the first intron of the beta-globin gene.

Authors:  J M Gonzalez-Redondo; T A Stoming; F Kutlar; A Kutlar; V C McKie; K M McKie; T H Huisman
Journal:  Br J Haematol       Date:  1989-01       Impact factor: 6.998

4.  Single-step method of RNA isolation by acid guanidinium thiocyanate-phenol-chloroform extraction.

Authors:  P Chomczynski; N Sacchi
Journal:  Anal Biochem       Date:  1987-04       Impact factor: 3.365

5.  The 5' splice site: phylogenetic evolution and variable geometry of association with U1RNA.

Authors:  M Jacob; H Gallinaro
Journal:  Nucleic Acids Res       Date:  1989-03-25       Impact factor: 16.971

6.  RNA splice junctions of different classes of eukaryotes: sequence statistics and functional implications in gene expression.

Authors:  M B Shapiro; P Senapathy
Journal:  Nucleic Acids Res       Date:  1987-09-11       Impact factor: 16.971

7.  Fetal Marfan syndrome: prenatal ultrasound diagnosis with pathological confirmation of skeletal and aortic lesions.

Authors:  M Koenigsberg; S Factor; S Cho; A Herskowitz; H Nitowsky; R Morecki
Journal:  Prenat Diagn       Date:  1981-10       Impact factor: 3.050

8.  Donor splice site mutation in the apolipoprotein (Apo) C-II gene (Apo C-IIHamburg) of a patient with Apo C-II deficiency.

Authors:  S S Fojo; U Beisiegel; U Beil; K Higuchi; M Bojanovski; R E Gregg; H Greten; H B Brewer
Journal:  J Clin Invest       Date:  1988-11       Impact factor: 14.808

9.  Identification of a mutation that causes exon skipping during collagen pre-mRNA splicing in an Ehlers-Danlos syndrome variant.

Authors:  D Weil; M Bernard; N Combates; M K Wirtz; D W Hollister; B Steinmann; F Ramirez
Journal:  J Biol Chem       Date:  1988-06-25       Impact factor: 5.157

10.  A new mutation in IVS-1 of the human beta globin gene causing beta thalassemia due to abnormal splicing.

Authors:  G F Atweh; C Wong; R Reed; S E Antonarakis; D Zhu; P K Ghosh; T Maniatis; B G Forget; H H Kazazian
Journal:  Blood       Date:  1987-07       Impact factor: 22.113

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  15 in total

1.  Familial occurrence of typical and severe lethal congenital contractural arachnodactyly caused by missplicing of exon 34 of fibrillin-2.

Authors:  M Wang; C L Clericuzio; M Godfrey
Journal:  Am J Hum Genet       Date:  1996-11       Impact factor: 11.025

2.  Marfan Database (second edition): software and database for the analysis of mutations in the human FBN1 gene.

Authors:  G Collod-Béroud; C Béroud; L Adès; C Black; M Boxer; D J Brock; M Godfrey; C Hayward; L Karttunen; D Milewicz; L Peltonen; R I Richards; M Wang; C Junien; C Boileau
Journal:  Nucleic Acids Res       Date:  1997-01-01       Impact factor: 16.971

Review 3.  Marfan syndrome in the third Millennium.

Authors:  Gwenaëlle Collod-Béroud; Catherine Boileau
Journal:  Eur J Hum Genet       Date:  2002-11       Impact factor: 4.246

4.  Double mutant fibrillin-1 (FBN1) allele in a patient with neonatal Marfan syndrome.

Authors:  M Wang; P Kishnani; M Decker-Phillips; S G Kahler; Y T Chen; M Godfrey
Journal:  J Med Genet       Date:  1996-09       Impact factor: 6.318

5.  A mutation in FBN1 disrupts profibrillin processing and results in isolated skeletal features of the Marfan syndrome.

Authors:  D M Milewicz; J Grossfield; S N Cao; C Kielty; W Covitz; T Jewett
Journal:  J Clin Invest       Date:  1995-05       Impact factor: 14.808

6.  Abnormal morphology of fibrillin microfibrils in fibroblast cultures from patients with neonatal Marfan syndrome.

Authors:  M Godfrey; M Raghunath; J Cisler; C L Bevins; A DePaepe; M Di Rocco; J Gregoritch; K Imaizumi; P Kaplan; Y Kuroki
Journal:  Am J Pathol       Date:  1995-06       Impact factor: 4.307

Review 7.  Identification of defects in the fibrillin gene and protein in individuals with the Marfan syndrome and related disorders.

Authors:  D M Milewicz
Journal:  Tex Heart Inst J       Date:  1994

8.  A compound-heterozygous Marfan patient: two defective fibrillin alleles result in a lethal phenotype.

Authors:  L Karttunen; M Raghunath; L Lönnqvist; L Peltonen
Journal:  Am J Hum Genet       Date:  1994-12       Impact factor: 11.025

9.  An extra cysteine in one of the non-calcium-binding epidermal growth factor-like motifs of the FBN1 polypeptide is connected to a novel variant of Marfan syndrome.

Authors:  C Ståhl-Hallengren; T Ukkonen; K Kainulainen; U Kristofersson; T Saxne; K Tornqvist; L Peltonen
Journal:  J Clin Invest       Date:  1994-08       Impact factor: 14.808

10.  Analyses of truncated fibrillin caused by a 366 bp deletion in the FBN1 gene resulting in Marfan syndrome.

Authors:  M Raghunath; C M Kielty; K Kainulainen; A Child; L Peltonen; B Steinmann
Journal:  Biochem J       Date:  1994-09-15       Impact factor: 3.857

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