Effect of Ascertainment Bias on Estimates of Patient Mortality in Inherited Cardiac Diseases

Eline A. Nannenberg; Ingrid A. W. van Rijsingen; Paul A. van der Zwaag; Maarten P. van den Berg; J. Peter van Tintelen; Michael W. T. Tanck; Michael J. Ackerman; Arthur A. M. Wilde; Imke Christiaans

doi:10.1161/CIRCGEN.117.001797

Effect of Ascertainment Bias on Estimates of Patient Mortality in Inherited Cardiac Diseases

Eline A. Nannenberg
, Ingrid A. W. van Rijsingen
, Paul A. van der Zwaag
, Maarten P. van den Berg
, J. Peter van Tintelen
, Michael W. T. Tanck
, Michael J. Ackerman
, Arthur A. M. Wilde
, Imke Christiaans

Research output: Contribution to journal › Article › Academic › peer-review

14 Downloads (Pure)

Abstract

BACKGROUND: Accurate estimates of survival are indispensable for cardiologists, clinical geneticists, and genetic counselors dealing with families with an inherited cardiac disease. However, a bias towards a more severe disease with a worse outcome in the first publications may not accurately represent the actual survival forecast. We, therefore, evaluated the effect of ascertainment bias on survival in 3 different inherited cardiac diseases (idiopathic ventricular fibrillation, SCN5A overlap syndrome, and arrhythmogenic cardiomyopathy) caused by a founder mutation.

Original language	English
Pages (from-to)	e001797
Journal	Circulation. Genomic and precision medicine
Volume	11
Issue number	10
DOIs	https://doi.org/10.1161/CIRCGEN.117.001797
Publication status	Published - 2018

UN SDGs

This output contributes to the following UN Sustainable Development Goals (SDGs)

SDG 3 Good Health and Well-being

Access to Document

10.1161/CIRCGEN.117.001797

Effect-of-ascertainment-bias-on-estimates-of-patient-mortality-in-inherited-cardiac-diseases.pdfFinal published version, 171 KBLicence: Taverne

Cite this

Nannenberg, E. A., van Rijsingen, I. A. W., van der Zwaag, P. A., van den Berg, M. P., van Tintelen, J. P., Tanck, M. W. T., Ackerman, M. J., Wilde, A. A. M., & Christiaans, I. (2018). Effect of Ascertainment Bias on Estimates of Patient Mortality in Inherited Cardiac Diseases. Circulation. Genomic and precision medicine, 11(10), e001797. https://doi.org/10.1161/CIRCGEN.117.001797

@article{12d812f44c3d4ec09127b0b678dd6e4a,

title = "Effect of Ascertainment Bias on Estimates of Patient Mortality in Inherited Cardiac Diseases",

abstract = "BACKGROUND: Accurate estimates of survival are indispensable for cardiologists, clinical geneticists, and genetic counselors dealing with families with an inherited cardiac disease. However, a bias towards a more severe disease with a worse outcome in the first publications may not accurately represent the actual survival forecast. We, therefore, evaluated the effect of ascertainment bias on survival in 3 different inherited cardiac diseases (idiopathic ventricular fibrillation, SCN5A overlap syndrome, and arrhythmogenic cardiomyopathy) caused by a founder mutation.",

author = "Nannenberg, \{Eline A.\} and \{van Rijsingen\}, \{Ingrid A. W.\} and \{van der Zwaag\}, \{Paul A.\} and \{van den Berg\}, \{Maarten P.\} and \{van Tintelen\}, \{J. Peter\} and Tanck, \{Michael W. T.\} and Ackerman, \{Michael J.\} and Wilde, \{Arthur A. M.\} and Imke Christiaans",

year = "2018",

doi = "10.1161/CIRCGEN.117.001797",

language = "English",

volume = "11",

pages = "e001797",

journal = "Circulation. Genomic and precision medicine",

issn = "2574-8300",

publisher = "Lippincott Williams and Wilkins Ltd.",

number = "10",

}

TY - JOUR

T1 - Effect of Ascertainment Bias on Estimates of Patient Mortality in Inherited Cardiac Diseases

AU - Nannenberg, Eline A.

AU - van Rijsingen, Ingrid A. W.

AU - van der Zwaag, Paul A.

AU - van den Berg, Maarten P.

AU - van Tintelen, J. Peter

AU - Tanck, Michael W. T.

AU - Ackerman, Michael J.

AU - Wilde, Arthur A. M.

AU - Christiaans, Imke

PY - 2018

Y1 - 2018

N2 - BACKGROUND: Accurate estimates of survival are indispensable for cardiologists, clinical geneticists, and genetic counselors dealing with families with an inherited cardiac disease. However, a bias towards a more severe disease with a worse outcome in the first publications may not accurately represent the actual survival forecast. We, therefore, evaluated the effect of ascertainment bias on survival in 3 different inherited cardiac diseases (idiopathic ventricular fibrillation, SCN5A overlap syndrome, and arrhythmogenic cardiomyopathy) caused by a founder mutation.

AB - BACKGROUND: Accurate estimates of survival are indispensable for cardiologists, clinical geneticists, and genetic counselors dealing with families with an inherited cardiac disease. However, a bias towards a more severe disease with a worse outcome in the first publications may not accurately represent the actual survival forecast. We, therefore, evaluated the effect of ascertainment bias on survival in 3 different inherited cardiac diseases (idiopathic ventricular fibrillation, SCN5A overlap syndrome, and arrhythmogenic cardiomyopathy) caused by a founder mutation.

UR - https://www.scopus.com/pages/publications/85055599623

UR - https://www.ncbi.nlm.nih.gov/pubmed/30354299

U2 - 10.1161/CIRCGEN.117.001797

DO - 10.1161/CIRCGEN.117.001797

M3 - Article

C2 - 30354299

SN - 2574-8300

VL - 11

SP - e001797

JO - Circulation. Genomic and precision medicine

JF - Circulation. Genomic and precision medicine

IS - 10

ER -

Effect of Ascertainment Bias on Estimates of Patient Mortality in Inherited Cardiac Diseases

Abstract

UN SDGs

Access to Document

Other files and links

Fingerprint

Cite this