Literature DB >> 14500307

Genetic counselling after carrier detection by newborn screening when one parent carries DeltaF508 and the other R117H.

L Curnow1, R Savarirayan, J Massie.   

Abstract

Newborn screening (NBS) for cystic fibrosis (CF) has been carried out in Victoria, Australia since 1989. The primary screen is immunoreactive trypsinogen (IRT) followed by DeltaF508 mutation analysis. As part of this process, carrier babies are detected and their parents are routinely offered carrier testing as part of their follow up. The DeltaF508 parent is identified and the other parent has an extended mutation analysis performed in case they are also a carrier. One of the mutations in the extended analysis is R117H which is associated with a broad phenotypic range, from CF with suppurative lung disease, to no clinical disease. We present four healthy DeltaF508 carrier babies identified by our NBS service with both parents identified as carriers, one DeltaF508 and the other R117H. Owing to the variable phenotype associated with R117H we have developed an approach to this difficult genetic counselling situation. Centres offering or considering NBS for CF will need an approach to this problem.

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Year:  2003        PMID: 14500307      PMCID: PMC1719305          DOI: 10.1136/adc.88.10.886

Source DB:  PubMed          Journal:  Arch Dis Child        ISSN: 0003-9888            Impact factor:   3.791


  14 in total

1.  Pancreatic function and extended mutation analysis in DeltaF508 heterozygous infants with an elevated immunoreactive trypsinogen but normal sweat electrolyte levels.

Authors:  R J Massie; B Wilcken; P Van Asperen; S Dorney; M Gruca; V Wiley; K Gaskin
Journal:  J Pediatr       Date:  2000-08       Impact factor: 4.406

Review 2.  The diagnosis of cystic fibrosis: a consensus statement. Cystic Fibrosis Foundation Consensus Panel.

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Journal:  J Pediatr       Date:  1998-04       Impact factor: 4.406

3.  Sweat testing following newborn screening for cystic fibrosis.

Authors:  J Massie; K Gaskin; P Van Asperen; B Wilcken
Journal:  Pediatr Pulmonol       Date:  2000-06

Review 4.  Molecular mechanisms of CFTR chloride channel dysfunction in cystic fibrosis.

Authors:  M J Welsh; A E Smith
Journal:  Cell       Date:  1993-07-02       Impact factor: 41.582

5.  Neonatal screening for cystic fibrosis: a comparison of two strategies for case detection in 1.2 million babies.

Authors:  B Wilcken; V Wiley; G Sherry; U Bayliss
Journal:  J Pediatr       Date:  1995-12       Impact factor: 4.406

6.  Mutations in CFTR associated with mild-disease-form Cl- channels with altered pore properties.

Authors:  D N Sheppard; D P Rich; L S Ostedgaard; R J Gregory; A E Smith; M J Welsh
Journal:  Nature       Date:  1993-03-11       Impact factor: 49.962

7.  Genetic basis of variable exon 9 skipping in cystic fibrosis transmembrane conductance regulator mRNA.

Authors:  C S Chu; B C Trapnell; S Curristin; G R Cutting; R G Crystal
Journal:  Nat Genet       Date:  1993-02       Impact factor: 38.330

8.  A mutation in CFTR produces different phenotypes depending on chromosomal background.

Authors:  S Kiesewetter; M Macek; C Davis; S M Curristin; C S Chu; C Graham; A E Shrimpton; S M Cashman; L C Tsui; J Mickle
Journal:  Nat Genet       Date:  1993-11       Impact factor: 38.330

9.  Sweat chloride concentrations in infants homozygous or heterozygous for F508 cystic fibrosis.

Authors:  P M Farrell; R E Koscik
Journal:  Pediatrics       Date:  1996-04       Impact factor: 7.124

10.  The impact of newborn screening on cystic fibrosis testing in Victoria, Australia.

Authors:  M E Balnaves; L Bonacquisto; I Francis; J Glazner; S Forrest
Journal:  J Med Genet       Date:  1995-07       Impact factor: 6.318

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  3 in total

Review 1.  A systematic review of the effects of disclosing carrier results generated through newborn screening.

Authors:  R Z Hayeems; J P Bytautas; F A Miller
Journal:  J Genet Couns       Date:  2008-10-28       Impact factor: 2.537

2.  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

3.  Decision making with uncertain information: learning from women in a high risk breast cancer clinic.

Authors:  Caren J Frost; Vickie Venne; Dianne Cunningham; Ruth Gerritsen-McKane
Journal:  J Genet Couns       Date:  2004-06       Impact factor: 2.537

  3 in total

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