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PMID: 9654209 Published · ppublish English Case Reports Journal Article Research Support, Non-U.S. Gov't

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

Human genetics ·Vol. 102 ·No. 5 ·1998-05-00 ·Pages 582-6

Mekus F, Ballmann M, Bronsveld I, Dörk T, Bijman J, Tümmler B, Veeze HJ

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.

MeSH Terms
Adult Cystic Fibrosis/diagnosis,genetics Cystic Fibrosis Transmembrane Conductance Regulator/genetics DNA Mutational Analysis Diagnosis, Differential Electrophysiology Haplotypes Humans Phenotype
Chemicals
CFTR protein, human Cystic Fibrosis Transmembrane Conductance Regulator
Authors & Affiliations
7 authors, click to expand affiliations / ORCID
Mekus F
Klinische Forschergruppe Molekulare Pathologie der Mukoviszidose, Medizinische Hochschule, Hannover, Germany.
Ballmann M
Bronsveld I
Dörk T
Bijman J
Tümmler B
Veeze H J
Article Info
Journal
Human genetics
Abbr.
Hum Genet
ISSN
0340-6717
Published
1998-05-00
Pages
582-6
Language
English
Region
Germany
NLM ID
7613873
Subset
IM
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