Literature DB >> 9016524

The PAH mutation analysis consortium database: update 1996.

P Nowacki1, S Byck, L Prevost, C R Scriver.   

Abstract

A website (http://www.mcgill.ca/pahdb ) is maintained by the curators for a Consortium (88 investigators, 28 countries) and all other users; it serves a relational database for human locus-specific genetic variation in a defined DNA sequence (GenBank U49897); (100 kb on human chromosome 12q24.1, gene symbol PAH). The intragenic nucleotide variation is both rare (Q< 0.01), extensive (>320 different mutations) and phenotype modifying, causing hyperphenylalaninemia by impairing phenylalanine hydroxylase function (see OMIM 261600), as well as polymorphic and neutral, the latter providing informative locus-specific haplotypes (>1200 different mutation/haplotype associations). The PAH database contains both offline core components (mutations, population associations and data source information) and several accessory online components: (i) relative frequencies of mutations by populations/regions (expanding file); (ii) data on genotype- phenotype correlations both in vitro and in vivo (new file); (iii) polymorphic haplotype structures (new file); (iv) intron sequence data (new file for design of primers); (v) description of mouse homologues (new file for mutations and phenotypes); (vi) the predicted PAH gene mutability profile (improved graphic); (vii) a clinical field for patient use (new interface with database). The website home page has been revised and a counter is recording >15 visits per day. Linkages to other mutation databases and an alliance of mutation database curators (new) are expanding. The primary 'electronic publication' reports now vastly exceed print reports. PAHdb serves as a prototype for obtaining, storing and distributing records of human genetic variation.

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Year:  1997        PMID: 9016524      PMCID: PMC146402          DOI: 10.1093/nar/25.1.139

Source DB:  PubMed          Journal:  Nucleic Acids Res        ISSN: 0305-1048            Impact factor:   16.971


  5 in total

1.  Mutation nomenclature: nicknames, systematic names, and unique identifiers.

Authors:  E Beutler; V A McKusick; A G Motulsky; C R Scriver; F Hutchinson
Journal:  Hum Mutat       Date:  1996       Impact factor: 4.878

2.  A suggested nomenclature for designating mutations.

Authors:  A L Beaudet; L C Tsui
Journal:  Hum Mutat       Date:  1993       Impact factor: 4.878

3.  PAH Mutation Analysis Consortium Database: a database for disease-producing and other allelic variation at the human PAH locus.

Authors:  L Hoang; S Byck; L Prevost; C R Scriver
Journal:  Nucleic Acids Res       Date:  1996-01-01       Impact factor: 16.971

4.  Molecular structure and polymorphic map of the human phenylalanine hydroxylase gene.

Authors:  A G DiLella; S C Kwok; F D Ledley; J Marvit; S L Woo
Journal:  Biochemistry       Date:  1986-02-25       Impact factor: 3.162

5.  Mouse models of human phenylketonuria.

Authors:  A Shedlovsky; J D McDonald; D Symula; W F Dove
Journal:  Genetics       Date:  1993-08       Impact factor: 4.562

  5 in total
  8 in total

1.  PAH Mutation Analysis Consortium Database: 1997. Prototype for relational locus-specific mutation databases.

Authors:  P M Nowacki; S Byck; L Prevost; C R Scriver
Journal:  Nucleic Acids Res       Date:  1998-01-01       Impact factor: 16.971

Review 2.  Phenylketonuria in Britain: genetic analysis gives a historical perspective of the disorder but will it predict the future for affected individuals?

Authors:  L A Tyfield
Journal:  Mol Pathol       Date:  1997-08

3.  Identification of hepatic nuclear factor 1 binding sites in the 5' flanking region of the human phenylalanine hydroxylase gene: implication of a dual function of phenylalanine hydroxylase stimulator in the phenylalanine hydroxylation system.

Authors:  X D Lei; S Kaufman
Journal:  Proc Natl Acad Sci U S A       Date:  1998-02-17       Impact factor: 11.205

4.  Population genetics of hyperphenylalaninaemia resulting from phenylalanine hydroxylase deficiency in Portugal.

Authors:  I Rivera; P Leandro; U Lichter-Konecki; I Tavares de Almeida; M C Lechner
Journal:  J Med Genet       Date:  1998-04       Impact factor: 6.318

5.  Haplotypes and linkage disequilibrium at the phenylalanine hydroxylase locus, PAH, in a global representation of populations.

Authors:  J R Kidd; A J Pakstis; H Zhao; R B Lu; F E Okonofua; A Odunsi; E Grigorenko; B B Tamir; J Friedlaender; L O Schulz; J Parnas; K K Kidd
Journal:  Am J Hum Genet       Date:  2000-04-27       Impact factor: 11.025

6.  Large heterozygous deletion masquerading as homozygous missense mutation: a pitfall in diagnostic mutation analysis.

Authors:  J Zschocke; E Quak; A Knauer; B Fritz; M Aslan; G F Hoffmann
Journal:  J Inherit Metab Dis       Date:  1999-08       Impact factor: 4.982

7.  Human phenylalanine hydroxylase mutations and hyperphenylalaninemia phenotypes: a metanalysis of genotype-phenotype correlations.

Authors:  E Kayaalp; E Treacy; P J Waters; S Byck; P Nowacki; C R Scriver
Journal:  Am J Hum Genet       Date:  1997-12       Impact factor: 11.025

8.  Structural characterization of the N-terminal autoregulatory sequence of phenylalanine hydroxylase.

Authors:  James Horne; Ian G Jennings; Trazel Teh; Paul R Gooley; Bostjan Kobe
Journal:  Protein Sci       Date:  2002-08       Impact factor: 6.725

  8 in total

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