Literature DB >> 8530006

Direct molecular analysis of the fragile X syndrome in a sample of Egyptian and German patients using non-radioactive PCR and Southern blot followed by chemiluminescent detection.

A A el-Aleem1, I Böhm, S Temtamy, M el-Awady, M Awadalla, J Schmidtke, M Stuhrmann.   

Abstract

Molecular genetic analysis of individuals from 6 Egyptian and 33 German families with fragile X syndrome and 240 further patients with mental retardation was performed applying a completely non-radioactive system. The aim of our study was the development of a non-radioactive detection method and its implementation in molecular diagnosis of the fragile X syndrome. Furthermore, we wanted to assess differences in the mutation sizes between Egyptian and German patients and between Egyptian and German carriers of a premutation. Using non-radioactive polymerase chain reaction (PCR), agarose gel electrophoresis and blotting of the PCR products, followed by hybridisation with a digoxigenin-labelled oligonucleotide probe (CGG)5 and chemiluminescent detection, we identified the fragile X full mutation (amplification of a CGG repeat in the FMR-1 gene ranging from several hundred to several thousand repeat units) in all patients. We observed no differences in the length of the CGG repeat between the Egyptian and German patients and carriers, respectively. However, in one prenatal diagnosis, we detected only one normal sized allele in a female fetus using the PCR-agarose assay, whereas Southern blot analysis with the digoxigenin labelled probe StB 12.3 revealed presence of a full mutation. Our newly established nonradioactive genomic blotting method is based on the conventional radioactive Southern blot analysis. Labelling of the probe StB 12.3 with digoxigenin via PCR allowed the detection of normal, premutated and fully mutated alleles. For exact sizing of small premutated or large normal alleles, we separated digoxigenin labelled PCR products through denaturing polyacrylamide gelelectrophoresis (PAGE) and transfered them to a nylon membrane using a gel dryer. The blotted PCR-fragments can easily be detected with alkaline phosphate-labelled anti-digoxigenin antibody. The number of trinucleotide repeat units can be determined by scoring the detected bands against a digoxigenated M13 sequencing ladder. Our newly developed digoxigenin/chemiluminescence approach using PCR and Southern blot analysis provides reliable results for routine detection of full fragile X mutations and premutations.

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Year:  1995        PMID: 8530006     DOI: 10.1007/bf00197414

Source DB:  PubMed          Journal:  Hum Genet        ISSN: 0340-6717            Impact factor:   4.132


  27 in total

1.  Mapping of DNA instability at the fragile X to a trinucleotide repeat sequence p(CCG)n.

Authors:  E J Kremer; M Pritchard; M Lynch; S Yu; K Holman; E Baker; S T Warren; D Schlessinger; G R Sutherland; R I Richards
Journal:  Science       Date:  1991-06-21       Impact factor: 47.728

2.  Fragile sites on human chromosomes: demonstration of their dependence on the type of tissue culture medium.

Authors:  G R Sutherland
Journal:  Science       Date:  1977-07-15       Impact factor: 47.728

3.  A simple salting out procedure for extracting DNA from human nucleated cells.

Authors:  S A Miller; D D Dykes; H F Polesky
Journal:  Nucleic Acids Res       Date:  1988-02-11       Impact factor: 16.971

4.  Precursor arrays for triplet repeat expansion at the fragile X locus.

Authors:  M C Hirst; P K Grewal; K E Davies
Journal:  Hum Mol Genet       Date:  1994-09       Impact factor: 6.150

5.  Variation of the CGG repeat at the fragile X site results in genetic instability: resolution of the Sherman paradox.

Authors:  Y H Fu; D P Kuhl; A Pizzuti; M Pieretti; J S Sutcliffe; S Richards; A J Verkerk; J J Holden; R G Fenwick; S T Warren
Journal:  Cell       Date:  1991-12-20       Impact factor: 41.582

6.  Genotype prediction in the fragile X syndrome.

Authors:  M C Hirst; Y Nakahori; S J Knight; C Schwartz; S N Thibodeau; A Roche; T J Flint; J M Connor; J P Fryns; K E Davies
Journal:  J Med Genet       Date:  1991-12       Impact factor: 6.318

7.  Absence of expression of the FMR-1 gene in fragile X syndrome.

Authors:  M Pieretti; F P Zhang; Y H Fu; S T Warren; B A Oostra; C T Caskey; D L Nelson
Journal:  Cell       Date:  1991-08-23       Impact factor: 41.582

8.  Population incidence and segregation ratios in the Martin-Bell syndrome.

Authors:  T P Webb; S E Bundey; A I Thake; J Todd
Journal:  Am J Med Genet       Date:  1986 Jan-Feb

9.  Physical mapping across the fragile X: hypermethylation and clinical expression of the fragile X syndrome.

Authors:  M V Bell; M C Hirst; Y Nakahori; R N MacKinnon; A Roche; T J Flint; P A Jacobs; N Tommerup; L Tranebjaerg; U Froster-Iskenius
Journal:  Cell       Date:  1991-02-22       Impact factor: 41.582

10.  Molecular cloning and analysis of the fragile X region in man.

Authors:  A Dietrich; P Kioschis; A P Monaco; B Gross; B Korn; S V Williams; D Sheer; D Heitz; I Oberle; D Toniolo
Journal:  Nucleic Acids Res       Date:  1991-05-25       Impact factor: 16.971

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