Cited by 346

The DOI matches its Crossref record; 2 of the 2 references it cites were found in a registry or an index.

The Cystic Fibrosis Foundation Patient Registry. Design and Methods of a National Observational Disease Registry

Knapp, Emily A., Fink, Aliza K., Goss, Christopher H., Sewall, Ase, Ostrenga, Josh, Dowd, Christopher, Elbert, Alexander, Petren, Kristofer M., Marshall, Bruce C.·2016·Annals of the American Thoracic Society

Abstract

Rationale: The Cystic Fibrosis Foundation Patient Registry (CFFPR) is an ongoing patient registry study that collects longitudinal demographic, clinical, and treatment information about persons with cystic fibrosis (CF) in the United States. CF is a life-shortening genetic disorder that occurs in approximately 1 in 3,500 births in the United States. High-quality observational data is important for clinical research, quality improvement, and clinical management. Objectives: To describe the data collection, patient population, and key limitations of the CFFPR. Methods: Inclusion criteria for the CFFPR include diagnosis with CF or a CFTR-associated disorder, care at an accredited care center program, and provision of informed consent. Data from clinic visits and hospitalizations are collected through a secure website. Loss to follow-up and generalizability were examined using several methods. The accuracy of CFFPR data was evaluated with an audit of 2012 CFFPR data compared to the medical record. Measurements and Main Results: Since 1986, the CFFPR contains the records of 48,463 individuals with CF. Participation among individuals seen at accredited care centers is high, and loss to follow-up is low. An audit of 2012 CFFPR data suggests that the CFFPR contains 95% of clinic visits and 90% of hospitalizations found in the medical record for these patients, and nearly all of the audited fields were highly accurate. Conclusions: Registries such as the CFFPR are important tools for research, clinical care, and tracking incidence, mortality and population trends.

Overview

Integrity signals

Registry

Crossref · 10.1513/annalsats.201511-781oc

Cites

2 of 2 references verified0 citation errors · 0 not checkable

Statistics

Recomputing p-values needs the paper’s full text.

Verification

Claims the paper makes

This paper’s full text isn’t openly available, so its claims and self-check weren’t run. The registry and citation checks still apply.

Claims other papers make about this one

Each is checked against this paper’s own text: does it say what the citing paper says it does?

Citations

Cites

Flagged first, then by citations

Show all 2 references

Research works printed but not linked: 30

  1. Farrell, 2008, Guidelines for diagnosis of cystic fibrosis in newborns through older adults: Cystic Fibrosis Foundation consensus report, J Pediatr, 153, S4, 10.1016/j.jpeds.2008.05.005 · DOI
  2. Doershuk, 2001, Cystic fibrosis in the 20th century: people, events, and progress
  3. Gliklich, 2014, Registries for evaluating patient outcomes: a user’s guide, 336
  4. Marshall, 2009, Cystic fibrosis foundation: achieving the mission, Respir Care, 54, 788, 10.4187/002013209790983223 · DOI
  5. Schechter, 2014, The Cystic Fibrosis Foundation Patient Registry as a tool for use in quality improvement, BMJ Qual Saf, 23, i9, 10.1136/bmjqs-2013-002378 · DOI
  6. Mall MA, 2014, Cystic fibrosis, 332, 10.1183/1025448x.erm6414 · DOI
  7. Mogayzel, 2014, Improving chronic care delivery and outcomes: the impact of the cystic fibrosis Care Center Network, BMJ Qual Saf, 23, i3, 10.1136/bmjqs-2013-002363 · DOI
  8. Knapp, 2015, Estimating the number of individuals with cystic fibrosis in the United States [abstract 502], Pediatr Pulmonol, 50, S381
  9. Dowd, 2015, The Cystic Fibrosis Foundation Patient Registry (CFFPR) data quality audit program: an analysis of data completeness and data accuracy [abstract 538], Pediatr Pulmonol, 50, S396
  10. FitzSimmons, 1994, The changing epidemiology of cystic fibrosis, Curr Probl Pediatr, 24, 171, 10.1016/0045-9380(94)90034-5 · DOI
  11. Martin, 2013, Births: final data for 2012, Natl Vital Stat Rep, 62, 1
  12. Kaye, 2006, Newborn screening fact sheets, Pediatrics, 118, e934, 10.1542/peds.2006-1783 · DOI
  13. Comeau, 2004, Population-based newborn screening for genetic disorders when multiple mutation DNA testing is incorporated: a cystic fibrosis newborn screening model demonstrating increased sensitivity but more carrier detections, Pediatrics, 113, 1573, 10.1542/peds.113.6.1573 · DOI
  14. Sontag, 2005, Two-tiered immunoreactive trypsinogen-based newborn screening for cystic fibrosis in Colorado: screening efficacy and diagnostic outcomes, J Pediatr, 147, S83, 10.1016/j.jpeds.2005.08.005 · DOI
  15. Rock, 2005, Newborn screening for cystic fibrosis in Wisconsin: nine-year experience with routine trypsinogen/DNA testing, J Pediatr, 147, S73, 10.1016/j.jpeds.2005.08.004 · DOI
  16. Prach, 2013, Novel CFTR variants identified during the first 3 years of cystic fibrosis newborn screening in California, J Mol Diagn, 15, 710, 10.1016/j.jmoldx.2013.05.006 · DOI
  17. Feuchtbaum, 2012, Birth prevalence of disorders detectable through newborn screening by race/ethnicity, Genet Med, 14, 937, 10.1038/gim.2012.76 · DOI
  18. Korzeniewski, 2011, Variation in immunoreactive trypsinogen concentrations among Michigan newborns and implications for cystic fibrosis newborn screening, Pediatr Pulmonol, 46, 125, 10.1002/ppul.21330 · DOI
  19. Larsson, 2012, Use of 13 disease registries in 5 countries demonstrates the potential to use outcome data to improve health care’s value, Health Aff (Millwood), 31, 220, 10.1377/hlthaff.2011.0762 · DOI
  20. Sykes, 2016, A standardized approach to estimating survival statistics for population-based cystic fibrosis registry cohorts, J Clin Epidemiol, 70, 206, 10.1016/j.jclinepi.2015.08.026 · DOI
  21. Mendelsohn, 2015, Characterization of missing data in clinical registry studies, Ther Innov Regul Sci, 49, 146, 10.1177/2168479014532259 · DOI
  22. Liou, 2007, Lung transplantation and survival in children with cystic fibrosis, N Engl J Med, 357, 2143, 10.1056/NEJMoa066359 · DOI
  23. Goss, 2004, Effect of ambient air pollution on pulmonary exacerbations and lung function in cystic fibrosis, Am J Respir Crit Care Med, 169, 816, 10.1164/rccm.200306-779OC · DOI
  24. Psoter, 2015, Fine particulate matter exposure and initial Pseudomonas aeruginosa acquisition in cystic fibrosis, Ann Am Thorac Soc, 12, 385, 10.1513/AnnalsATS.201408-400OC · DOI
  25. Nick, 2010, Effects of gender and age at diagnosis on disease progression in long-term survivors of cystic fibrosis, Am J Respir Crit Care Med, 182, 614, 10.1164/rccm.201001-0092OC · DOI
  26. Ren, 2015, Outcomes of infants with indeterminate diagnosis detected by cystic fibrosis newborn screening, Pediatrics, 135, e1386, 10.1542/peds.2014-3698 · DOI
  27. Goss, 2015, Children and young adults with CF in the USA have better lung function compared with the UK, Thorax, 70, 229, 10.1136/thoraxjnl-2014-205718 · DOI
  28. Motheral, 2003, A checklist for retrospective database studies–report of the ISPOR Task Force on Retrospective Databases, Value Health, 6, 90, 10.1046/j.1524-4733.2003.00242.x · DOI
  29. Psoter, 2013, Opportunities and pitfalls of registry data for clinical research, Paediatr Respir Rev, 14, 141
  30. Heltshe, 2016, Short-term and long-term response to pulmonary exacerbation treatment in cystic fibrosis, Thorax, 71, 223, 10.1136/thoraxjnl-2014-206750 · DOI

Cited by

Sorted by citations

See all 346

Metadata

Authors
Knapp, E. A., Fink, A. K., Goss, C. H., Sewall, A., Ostrenga, J., Dowd, C., Elbert, A., Petren, K. M., & Marshall, B. C.
Affiliations
  • Cystic Fibrosis Foundation, Bethesda, Maryland
  • Department of Medicine, University of Washington, Seattle, Washington; and
  • Sewall, Inc., Bethesda, Maryland
Venue
Annals of the American Thoracic Society
Published
2016-07-01
Volume
13
Issue
7
Pages
1173-1179
ISSN
  • 2329-6933
  • 2325-6621
Cited by
346
References
2
Language
en

About these checks

Registry, retraction, Cites and statistics results are computed, not judged, with one exception: a printed reference that no index lists is read once by a model before it counts as a citation error. Claims are extracted by a model and kept only when their quote appears verbatim in the text; each cited source is then checked, with a model’s help, for whether it says what the claim relies on. Those verdicts can be wrong — read the claim’s quote and the cited source before relying on one.

Statistics and arithmetic are recomputed, not judged.

Confidence bands are shown only for fields calibrated on our annotated test set. None is calibrated yet, so no band is shown.

What counts as a claim ›How we measure confidence ›How we check this ›

Are you an author, or do you see a problem with this page? Request a review