Literature DB >> 9250667

Complementation of null CF mice with a human CFTR YAC transgene.

A L Manson1, A E Trezise, L J MacVinish, K D Kasschau, N Birchall, V Episkopou, G Vassaux, M J Evans, W H Colledge, A W Cuthbert, C Huxley.   

Abstract

We have made transgenic mice carrying a 320 kb YAC with the intact human cystic fibrosis transmembrane regulator (CFTR) gene. Mice that only express the human transgene were obtained by breeding with Cambridge null CF mice. One line has approximately two copies of the intact YAC. Mice carrying this transgene and expressing no mouse cftr appear normal and breed well, in marked contrast to the null mice, where 50% die by approximately 5 days after birth. The chloride secretory responses in these mice are as large or larger than in wild-type tissues. Expression of the transgene is highly cell type specific and matches that of the endogenous mouse gene in the crypt epithelia throughout the gut and in salivary gland tissue. However, there is no transgene expression in some tissues, such as the Brunner's glands, where it would be expected. Where there are differences between the mouse and human pattern of expression, the transgene follows the mouse pattern. We have thus defined a cloned fragment of DNA which directs physiological levels of expression in many of the specific cells where CFTR is normally expressed.

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Year:  1997        PMID: 9250667      PMCID: PMC1170049          DOI: 10.1093/emboj/16.14.4238

Source DB:  PubMed          Journal:  EMBO J        ISSN: 0261-4189            Impact factor:   11.598


  41 in total

1.  Identification of the cystic fibrosis gene: genetic analysis.

Authors:  B Kerem; J M Rommens; J A Buchanan; D Markiewicz; T K Cox; A Chakravarti; M Buchwald; L C Tsui
Journal:  Science       Date:  1989-09-08       Impact factor: 47.728

2.  Identification of the cystic fibrosis gene: cloning and characterization of complementary DNA.

Authors:  J R Riordan; J M Rommens; B Kerem; N Alon; R Rozmahel; Z Grzelczak; J Zielenski; S Lok; N Plavsic; J L Chou
Journal:  Science       Date:  1989-09-08       Impact factor: 47.728

3.  In vivo cell-specific expression of the cystic fibrosis transmembrane conductance regulator.

Authors:  A E Trezise; M Buchwald
Journal:  Nature       Date:  1991-10-03       Impact factor: 49.962

4.  Genomic DNA sequence of the cystic fibrosis transmembrane conductance regulator (CFTR) gene.

Authors:  J Zielenski; R Rozmahel; D Bozon; B Kerem; Z Grzelczak; J R Riordan; J Rommens; L C Tsui
Journal:  Genomics       Date:  1991-05       Impact factor: 5.736

5.  A yeast artificial chromosome contig encompassing the cystic fibrosis locus.

Authors:  R Anand; D J Ogilvie; R Butler; J H Riley; R S Finniear; S J Powell; J C Smith; A F Markham
Journal:  Genomics       Date:  1991-01       Impact factor: 5.736

6.  Modulation of disease severity in cystic fibrosis transmembrane conductance regulator deficient mice by a secondary genetic factor.

Authors:  R Rozmahel; M Wilschanski; A Matin; S Plyte; M Oliver; W Auerbach; A Moore; J Forstner; P Durie; J Nadeau; C Bear; L C Tsui
Journal:  Nat Genet       Date:  1996-03       Impact factor: 38.330

7.  Identification of the cystic fibrosis gene: chromosome walking and jumping.

Authors:  J M Rommens; M C Iannuzzi; B Kerem; M L Drumm; G Melmer; M Dean; R Rozmahel; J L Cole; D Kennedy; N Hidaka
Journal:  Science       Date:  1989-09-08       Impact factor: 47.728

8.  Liposome-mediated CFTR gene transfer to the nasal epithelium of patients with cystic fibrosis.

Authors:  N J Caplen; E W Alton; P G Middleton; J R Dorin; B J Stevenson; X Gao; S R Durham; P K Jeffery; M E Hodson; C Coutelle
Journal:  Nat Med       Date:  1995-01       Impact factor: 53.440

9.  Submucosal glands are the predominant site of CFTR expression in the human bronchus.

Authors:  J F Engelhardt; J R Yankaskas; S A Ernst; Y Yang; C R Marino; R C Boucher; J A Cohn; J M Wilson
Journal:  Nat Genet       Date:  1992-11       Impact factor: 38.330

10.  Hyperabsorption of Na+ and raised Ca(2+)-mediated Cl- secretion in nasal epithelia of CF mice.

Authors:  B R Grubb; R N Vick; R C Boucher
Journal:  Am J Physiol       Date:  1994-05
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  25 in total

1.  Genomic sequence analysis of Fugu rubripes CFTR and flanking genes in a 60 kb region conserving synteny with 800 kb of human chromosome 7.

Authors:  H Davidson; M S Taylor; A Doherty; A C Boyd; D J Porteous
Journal:  Genome Res       Date:  2000-08       Impact factor: 9.043

2.  An arrayed human genomic library constructed in the PAC shuttle vector pJCPAC-Mam2 for genome-wide association studies and gene therapy.

Authors:  John Fuesler; Yasunori Nagahama; Joseph Szulewski; Joshua Mundorff; Stephanie Bireley; Jonathon S Coren
Journal:  Gene       Date:  2012-01-24       Impact factor: 3.688

3.  A transgenic mouse model engineered to investigate human brain-derived neurotrophic factor in vivo.

Authors:  Fabrice Guillemot; Italina Cerutti; Charles Auffray; Marie-Dominique Devignes
Journal:  Transgenic Res       Date:  2007-01-16       Impact factor: 2.788

4.  Generation of a conditional null allele for Cftr in mice.

Authors:  Craig A Hodges; Calvin U Cotton; Mark R Palmert; Mitchell L Drumm
Journal:  Genesis       Date:  2008-10       Impact factor: 2.487

5.  A dicistronic construct allows easy detection of human CFTR expression from YAC DNA in human cells.

Authors:  G Vassaux; C Huxley
Journal:  Nucleic Acids Res       Date:  1997-10-15       Impact factor: 16.971

Review 6.  RNA Splicing and Disease: Animal Models to Therapies.

Authors:  Matías Montes; Brianne L Sanford; Daniel F Comiskey; Dawn S Chandler
Journal:  Trends Genet       Date:  2018-11-19       Impact factor: 11.639

Review 7.  Size matters: use of YACs, BACs and PACs in transgenic animals.

Authors:  P Giraldo; L Montoliu
Journal:  Transgenic Res       Date:  2001-04       Impact factor: 2.788

Review 8.  Animal models for cystic fibrosis liver disease (CFLD).

Authors:  Romina Fiorotto; Mariangela Amenduni; Valeria Mariotti; Massimiliano Cadamuro; Luca Fabris; Carlo Spirli; Mario Strazzabosco
Journal:  Biochim Biophys Acta Mol Basis Dis       Date:  2018-07-30       Impact factor: 5.187

9.  Rescue of the embryonic lethal hematopoietic defect reveals a critical role for GATA-2 in urogenital development.

Authors:  Y Zhou; K C Lim; K Onodera; S Takahashi; J Ohta; N Minegishi; F Y Tsai; S H Orkin; M Yamamoto; J D Engel
Journal:  EMBO J       Date:  1998-11-16       Impact factor: 11.598

10.  Prospects for the use of artificial chromosomes and minichromosome-like episomes in gene therapy.

Authors:  Sara Pérez-Luz; Javier Díaz-Nido
Journal:  J Biomed Biotechnol       Date:  2010-08-24
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