Literature DB >> 10947197

Defective folding and rapid degradation of mutant proteins is a common disease mechanism in genetic disorders.

N Gregersen1, P Bross, M M Jørgensen, T J Corydon, B S Andresen.   

Abstract

Many disease-causing point mutations do not seriously compromise synthesis of the affected polypeptide but rather exert their effects by impairing subsequent protein folding or stability of the folded protein. This often results in rapid degradation of the affected protein. The concepts of such 'conformational disease' are illustrated by reference to cystic fibrosis, phenylketonuria and short-chain acyl-CoA dehydrogenase deficiency. Other cellular components such as chaperones and proteases, as well as environmental factors, may combine to modulate the phenotype of such disorders and this may open up new therapeutic approaches.

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Year:  2000        PMID: 10947197     DOI: 10.1023/a:1005663728291

Source DB:  PubMed          Journal:  J Inherit Metab Dis        ISSN: 0141-8955            Impact factor:   4.982


  37 in total

1.  Chloride conductance expressed by delta F508 and other mutant CFTRs in Xenopus oocytes.

Authors:  M L Drumm; D J Wilkinson; L S Smit; R T Worrell; T V Strong; R A Frizzell; D C Dawson; F S Collins
Journal:  Science       Date:  1991-12-20       Impact factor: 47.728

Review 2.  Monogenic traits are not simple: lessons from phenylketonuria.

Authors:  C R Scriver; P J Waters
Journal:  Trends Genet       Date:  1999-07       Impact factor: 11.639

Review 3.  Defective protein folding as a basis of human disease.

Authors:  P J Thomas; B H Qu; P L Pedersen
Journal:  Trends Biochem Sci       Date:  1995-11       Impact factor: 13.807

4.  Missense mutations in the phenylalanine hydroxylase gene (PAH) can cause accelerated proteolytic turnover of PAH enzyme: a mechanism underlying phenylketonuria.

Authors:  P J Waters; M A Parniak; B R Akerman; A O Jones; C R Scriver
Journal:  J Inherit Metab Dis       Date:  1999-05       Impact factor: 4.982

5.  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

6.  Partial characterization and three-dimensional-structural localization of eight mutations in exon 7 of the human phenylalanine hydroxylase gene associated with phenylketonuria.

Authors:  E Bjørgo; P M Knappskog; A Martinez; R C Stevens; T Flatmark
Journal:  Eur J Biochem       Date:  1998-10-01

7.  A rare disease-associated mutation in the medium-chain acyl-CoA dehydrogenase (MCAD) gene changes a conserved arginine, previously shown to be functionally essential in short-chain acyl-CoA dehydrogenase (SCAD).

Authors:  B S Andresen; P Bross; T G Jensen; V Winter; I Knudsen; S Kølvraa; U B Jensen; L Bolund; M Duran; J J Kim
Journal:  Am J Hum Genet       Date:  1993-09       Impact factor: 11.025

8.  Processing of mutant cystic fibrosis transmembrane conductance regulator is temperature-sensitive.

Authors:  G M Denning; M P Anderson; J F Amara; J Marshall; A E Smith; M J Welsh
Journal:  Nature       Date:  1992-08-27       Impact factor: 49.962

Review 9.  Familial hypertrophic cardiomyopathy: from mutations to functional defects.

Authors:  G Bonne; L Carrier; P Richard; B Hainque; K Schwartz
Journal:  Circ Res       Date:  1998-09-21       Impact factor: 17.367

10.  Intracellular turnover of cystic fibrosis transmembrane conductance regulator. Inefficient processing and rapid degradation of wild-type and mutant proteins.

Authors:  C L Ward; R R Kopito
Journal:  J Biol Chem       Date:  1994-10-14       Impact factor: 5.157

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  16 in total

1.  Genotype, enzyme activity, glutathione level, and clinical phenotype in patients with glutathione synthetase deficiency.

Authors:  Runa Njålsson; Ellinor Ristoff; Katarina Carlsson; Andreas Winkler; Agne Larsson; Svante Norgren
Journal:  Hum Genet       Date:  2005-02-17       Impact factor: 4.132

Review 2.  Garrod's foresight; our hindsight.

Authors:  C R Scriver
Journal:  J Inherit Metab Dis       Date:  2001-04       Impact factor: 4.982

3.  Molecular consequences of the ACVR1(R206H) mutation of fibrodysplasia ossificans progressiva.

Authors:  Gin-Ah Song; Hyun-Jung Kim; Kyung-Mi Woo; Jeong-Hwa Baek; Gwan-Shik Kim; Jin-Young Choi; Hyun-Mo Ryoo
Journal:  J Biol Chem       Date:  2010-05-12       Impact factor: 5.157

4.  Implication of the extracellular disulfide bond on myelin protein zero expression.

Authors:  G Pfend; J M Matthieu; N Garin; M Tosic
Journal:  Neurochem Res       Date:  2001-05       Impact factor: 3.996

5.  Y65C missense mutation in the WW domain of the Golabi-Ito-Hall syndrome protein PQBP1 affects its binding activity and deregulates pre-mRNA splicing.

Authors:  Victor E Tapia; Emilia Nicolaescu; Caleb B McDonald; Valeria Musi; Tsutomu Oka; Yujin Inayoshi; Adam C Satteson; Virginia Mazack; Jasper Humbert; Christian J Gaffney; Monique Beullens; Charles E Schwartz; Christiane Landgraf; Rudolf Volkmer; Annalisa Pastore; Amjad Farooq; Mathieu Bollen; Marius Sudol
Journal:  J Biol Chem       Date:  2010-04-21       Impact factor: 5.157

6.  Deletion of Ser-171 causes inactivation, proteasome-mediated degradation and complete deficiency of human transaldolase.

Authors:  Craig E Grossman; Brian Niland; Christina Stancato; Nanda M Verhoeven; Marjo S Van Der Knaap; Cornelis Jakobs; Lawrence M Brown; Sandor Vajda; Katalin Banki; Andras Perl
Journal:  Biochem J       Date:  2004-09-01       Impact factor: 3.857

Review 7.  Human gene mutation in pathology and evolution.

Authors:  D N Cooper
Journal:  J Inherit Metab Dis       Date:  2002-05       Impact factor: 4.982

8.  Mutations in the gene for the E1beta subunit: a novel cause of pyruvate dehydrogenase deficiency.

Authors:  Ruth M Brown; Rosemary A Head; Ivan I Boubriak; James V Leonard; Neil H Thomas; Garry K Brown
Journal:  Hum Genet       Date:  2004-05-11       Impact factor: 4.132

9.  The Hsp70 and TRiC/CCT chaperone systems cooperate in vivo to assemble the von Hippel-Lindau tumor suppressor complex.

Authors:  Mark W Melville; Amie J McClellan; Anne S Meyer; Andre Darveau; Judith Frydman
Journal:  Mol Cell Biol       Date:  2003-05       Impact factor: 4.272

10.  Mutation and biochemical analysis in carnitine palmitoyltransferase type II (CPT II) deficiency.

Authors:  S E Olpin; A Afifi; S Clark; N J Manning; J R Bonham; A Dalton; J V Leonard; J M Land; B S Andresen; A A Morris; F Muntoni; D Turnbull; M Pourfarzam; S Rahman; R J Pollitt
Journal:  J Inherit Metab Dis       Date:  2003       Impact factor: 4.982

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