Literature DB >> 9654209

Cystic-fibrosis-like disease unrelated to the cystic fibrosis transmembrane conductance regulator.

F Mekus1, M Ballmann, I Bronsveld, T Dörk, J Bijman, B Tümmler, H J Veeze.   

Abstract

Cystic fibrosis (CF) is considered to be a monogenic disease caused by molecular lesions within the cystic fibrosis transmembrane conductance regulator (CFTR) gene and is diagnosed by elevated sweat electrolytes. We have investigated the clinical manifestations of cystic fibrosis, CFTR genetics and electrophysiology in a sibpair in which the brother is being treated as having CF, whereas his sister is asymptomatic. The diagnosis of CF in the index patient is based on highly elevated sweat electrolytes in the presence of CF-related pulmonary symptoms. The investigation of chloride conductance in respiratory and intestinal tissue by nasal potential difference and intestinal current measurements, respectively, provides no evidence for CFTR dysfunction in the siblings who share the same CFTR alleles. No molecular lesion has been identified in the CFTR gene of the brother. Findings in the investigated sibpair point to the existence of a CF-like disease with a positive sweat test without CFTR being affected. Other factors influencing sodium or chloride transport are likely to be the cause of the symptoms in the patient described.

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Year:  1998        PMID: 9654209     DOI: 10.1007/s004390050744

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


  9 in total

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Authors:  Colin Wallis
Journal:  J R Soc Med       Date:  2003       Impact factor: 5.344

2.  Potential benefits of the UK Cystic Fibrosis Database.

Authors:  G Mehta; E J Sims; F Culross; J D McCormick; A Mehta
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3.  Best practice guidelines for molecular genetic diagnosis of cystic fibrosis and CFTR-related disorders--updated European recommendations.

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4.  The relevance of sweat testing for the diagnosis of cystic fibrosis in the genomic era.

Authors:  Avantika Mishra; Ronda Greaves; John Massie
Journal:  Clin Biochem Rev       Date:  2005-11

Review 5.  Cystic fibrosis: terminology and diagnostic algorithms.

Authors:  K De Boeck; M Wilschanski; C Castellani; C Taylor; H Cuppens; J Dodge; M Sinaasappel
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Review 6.  Consensus on the use and interpretation of cystic fibrosis mutation analysis in clinical practice.

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Review 7.  [Molecular genetics principles in cystic fibrosis. An example of genetic illness in pneumology].

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8.  Identification of CFTR variants in Latino patients with cystic fibrosis from the Dominican Republic and Puerto Rico.

Authors:  Andrew M Zeiger; Meghan E McGarry; Angel C Y Mak; Vivian Medina; Sandra Salazar; Celeste Eng; Amy K Liu; Sam S Oh; Thomas J Nuckton; Deepti Jain; Thomas W Blackwell; Hyun Min Kang; Goncalo Abecasis; Leandra Cordero Oñate; Max A Seibold; Esteban G Burchard; Jose Rodriguez-Santana
Journal:  Pediatr Pulmonol       Date:  2019-10-30

9.  Association of cystic fibrosis transmembrane conductance regulator with epithelial sodium channel subunits carrying Liddle's syndrome mutations.

Authors:  Arun K Rooj; Estelle Cormet-Boyaka; Edlira B Clark; Yawar J Qadri; William Lee; Ravindra Boddu; Anupam Agarwal; Richa Tambi; Mohammed Uddin; Vladimir Parpura; Eric J Sorscher; Cathy M Fuller; Bakhrom K Berdiev
Journal:  Am J Physiol Lung Cell Mol Physiol       Date:  2021-05-26       Impact factor: 6.011

  9 in total

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