Literature DB >> 27148940

Cystic fibrosis newborn screening programs: implications of the CFTR variant spectrum in nonwhite patients.

Lynn Pique1, Steve Graham2, Michelle Pearl3, Martin Kharrazi4, Iris Schrijver1,5.   

Abstract

PURPOSE: Cystic fibrosis newborn screening (CFNBS) has been offered across the United States since 2010. However, as compared with white patients with CF, CFTR variant identification in nonwhite populations remains inequitable. Utilizing the recent characterization of the nonwhite CF variant spectrum, we examined the effectiveness of current CFNBS molecular panels in identifying affected nonwhite newborns.
METHODS: Based on a cross-sectional evaluation of genotyping data from the CF Foundation Patient Registry that compared 3,496 nonwhite with 22,206 white CF patients, the current CFNBS algorithms used in the 50 states and the District of Columbia were analyzed. We assessed the percentage of CF patients of Hispanic, African, Asian, and Native American heritage who would not be identified by the molecular panels most commonly used.
RESULTS: Compared with whites, variant detection was significantly lower in Hispanic, black, and Asian newborns (P ≤ 0.0001 each), as well as in Native American newborns (P values ranged from 0.001 to 0.0003), for the most common CFNBS panels.
CONCLUSION: This study provides a perspective on the applicability of current panels to a diverse population and enables CFNBS programs to consider more inclusive test approaches to facilitate diagnosis, timely clinical intervention, and enhanced prognosis for CF patients of nonwhite and mixed ethnicities.Genet Med 19 1, 36-44.

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Year:  2016        PMID: 27148940     DOI: 10.1038/gim.2016.48

Source DB:  PubMed          Journal:  Genet Med        ISSN: 1098-3600            Impact factor:   8.822


  38 in total

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Review 2.  Newborn screening for cystic fibrosis: an opportunity to improve care and outcomes.

Authors:  Preston W Campbell; Terry B White
Journal:  J Pediatr       Date:  2005-09       Impact factor: 4.406

3.  Cost-effectiveness of 4 neonatal screening strategies for cystic fibrosis.

Authors:  M Elske van den Akker-van Marle; Hinke M Dankert; Paul H Verkerk; Jeannette E Dankert-Roelse
Journal:  Pediatrics       Date:  2006-09       Impact factor: 7.124

Review 4.  The diagnosis of cystic fibrosis: a consensus statement. Cystic Fibrosis Foundation Consensus Panel.

Authors:  B J Rosenstein; G R Cutting
Journal:  J Pediatr       Date:  1998-04       Impact factor: 4.406

5.  Clinical investigational studies for validation of a next-generation sequencing in vitro diagnostic device for cystic fibrosis testing.

Authors:  Daniel S Grosu; Lynda Hague; Manjula Chelliserry; Kristina M Kruglyak; Ross Lenta; Brandy Klotzle; Jonathan San; Wendy M Goldstein; Sharmili Moturi; Patricia Devers; Julie Woolworth; Eric Peters; Barbara Elashoff; Jay Stoerker; Daynna J Wolff; Kenneth J Friedman; W Edward Highsmith; Erick Lin; Frank S Ong
Journal:  Expert Rev Mol Diagn       Date:  2014-06       Impact factor: 5.225

Review 6.  Newborn screening for cystic fibrosis: evaluation of benefits and risks and recommendations for state newborn screening programs.

Authors:  Scott D Grosse; Coleen A Boyle; Jeffrey R Botkin; Anne Marie Comeau; Martin Kharrazi; Margaret Rosenfeld; Benjamin S Wilfond
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7.  A comprehensive assay for CFTR mutational analysis using next-generation sequencing.

Authors:  Ahmad N Abou Tayoun; Christopher D Tunkey; Trevor J Pugh; Tristen Ross; Minita Shah; Clarence C Lee; Timothy T Harkins; Wendy A Wells; Laura J Tafe; Christopher I Amos; Gregory J Tsongalis
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8.  Novel CFTR variants identified during the first 3 years of cystic fibrosis newborn screening in California.

Authors:  Lisa Prach; Ruth Koepke; Martin Kharrazi; Steven Keiles; Danieli B Salinas; Maria Carmen Reyes; Mark Pian; Harry Opsimos; Kimberly N Otsuka; Karen Ann Hardy; Carlos E Milla; Jacquelyn M Zirbes; Bradley Chipps; Susan O'Bra; Muhammad M Saeed; Reddivalam Sudhakar; Susan Lehto; Dennis Nielson; Gregory F Shay; Mary Seastrand; Sanjay Jhawar; Bruce Nickerson; Christopher Landon; Ann Thompson; Eliezer Nussbaum; Terry Chin; Henry Wojtczak
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9.  Next-Generation Molecular Testing of Newborn Dried Blood Spots for Cystic Fibrosis.

Authors:  Martina I Lefterova; Peidong Shen; Justin I Odegaard; Eula Fung; Tsoyu Chiang; Gang Peng; Ronald W Davis; Wenyi Wang; Martin Kharrazi; Iris Schrijver; Curt Scharfe
Journal:  J Mol Diagn       Date:  2016-02-01       Impact factor: 5.568

10.  Improving newborn screening for cystic fibrosis using next-generation sequencing technology: a technical feasibility study.

Authors:  Mei W Baker; Anne E Atkins; Suzanne K Cordovado; Miyono Hendrix; Marie C Earley; Philip M Farrell
Journal:  Genet Med       Date:  2015-02-12       Impact factor: 8.822

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  13 in total

1.  Regional variations in longitudinal pulmonary function: A comparison of Hispanic and non-Hispanic subjects with cystic fibrosis in the United States.

Authors:  Meghan E McGarry; John M Neuhaus; Dennis W Nielson; Ngoc P Ly
Journal:  Pediatr Pulmonol       Date:  2019-05-29

Review 2.  Personalized Medicine and the Power of Electronic Health Records.

Authors:  Noura S Abul-Husn; Eimear E Kenny
Journal:  Cell       Date:  2019-03-21       Impact factor: 41.582

Review 3.  Increasing life expectancy in cystic fibrosis: Advances and challenges.

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4.  Evaluating Differences in the Disease Experiences of Minority Adults With Cystic Fibrosis.

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Review 5.  Left behind: The potential impact of CFTR modulators on racial and ethnic disparities in cystic fibrosis.

Authors:  Meghan E McGarry; Elizabeth R Gibb; Gabriela R Oates; Michael S Schechter
Journal:  Paediatr Respir Rev       Date:  2021-12-22       Impact factor: 5.526

6.  Nationwide genetic analysis for molecularly unresolved cystic fibrosis patients in a multiethnic society: implications for preconception carrier screening.

Authors:  Doron M Behar; Ori Inbar; Michal Shteinberg; Michal Gur; Huda Mussaffi; David Shoseyov; Moshe Ashkenazi; Soliman Alkrinawi; Concetta Bormans; Fahed Hakim; Meir Mei-Zahav; Malena Cohen-Cymberknoh; Adi Dagan; Dario Prais; Ifat Sarouk; Patrick Stafler; Bat El Bar Aluma; Gidon Akler; Elie Picard; Micha Aviram; Ori Efrati; Galit Livnat; Joseph Rivlin; Lea Bentur; Hannah Blau; Eitan Kerem; Amihood Singer
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Review 7.  The Role of Extended CFTR Gene Sequencing in Newborn Screening for Cystic Fibrosis.

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Review 8.  CFTR Modulators: Shedding Light on Precision Medicine for Cystic Fibrosis.

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Journal:  Front Pharmacol       Date:  2016-09-05       Impact factor: 5.810

Review 9.  The influence of social determinants of health on the genetic diagnostic odyssey: who remains undiagnosed, why, and to what effect?

Authors:  Yarden S Fraiman; Monica H Wojcik
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10.  Phenotypic Presentations of Cystic Fibrosis in Children of African Descent.

Authors:  Sophie Mayer Lacrosniere; Michele Gerardin; Laurence Le Clainche-Viala; Veronique Houdouin
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