دورية أكاديمية
Data accuracy, consistency and completeness of the national Swiss cystic fibrosis patient registry: Lessons from an ECFSPR data quality project.
| العنوان: | Data accuracy, consistency and completeness of the national Swiss cystic fibrosis patient registry: Lessons from an ECFSPR data quality project. |
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| المؤلفون: | Wolf L; Department of Respiratory Medicine, University Children's Hospital Zurich, Switzerland; Departments of Pulmonology and Paediatric Pulmonology, Cantonal Hospital Winterthur, Switzerland., Usemann J; Department of Respiratory Medicine, University Children's Hospital Zurich, Switzerland; Department of Pulmonology, University Children's Hospital Basel, Switzerland., Collaud E; Department of Respiratory Medicine, University Children's Hospital Zurich, Switzerland., Derkenne MF; Department of Pulmonology, Lausanne University Hospital, Switzerland., Fischer R; Quartier Bleu, Pulmonology Outpatient Clinic, Berne, Switzerland., Hensen M; Department of Paediatrics, Cantonal Hospital Fribourg, Switzerland., Hitzler M; Department of Paediatric Pulmonology, Cantonal Hospital Lucerne, Switzerland., Hofer M; Departments of Pulmonology and Paediatric Pulmonology, Cantonal Hospital Winterthur, Switzerland., Inci D; Department of Respiratory Medicine, University Children's Hospital Zurich, Switzerland., Irani S; Department of Pulmonology, Cantonal Hospital Aarau, Switzerland., Jahn K; Clinic of Respiratory Medicine and Pulmonary Cell Research, University Hospital Basel, Switzerland., Koutsokera A; Department of Pulmonology, Lausanne University Hospital, Switzerland., Kusche R; Department of Pulmonology, Cantonal Hospital Aarau, Switzerland; Department of Paediatric Pulmonology, Cantonal Hospital Aarau, Switzerland., Kurowski T; Department of Pulmonology, University Hospital Zurich, Switzerland., Latzin P; Division of Paediatric Respiratory Medicine and Allergology, Department of Paediatrics, Inselspital Berne University Hospital, Switzerland., Lin D; Department of Pulmonology, Inselspital Berne University Hospital, Switzerland., Mioranza L; Paediatric Pulmonology and Cystic Fibrosis Unit, Division of Paediatrics, Department Woman-Mother-Child, Lausanne University Hospital, Switzerland., Moeller A; Department of Respiratory Medicine, University Children's Hospital Zurich, Switzerland., Mornand A; Department of Paediatric Pulmonology, Geneva University Hospital, Switzerland., Mueller-Suter D; Department of Paediatric Pulmonology, Cantonal Hospital Aarau, Switzerland., Murer C; Department of Pulmonology, Cantonal Hospital Lucerne, Switzerland., Naehrlich L; Department of Paediatrics, Justus-Liebig-University Giessen, Germany; European Cystic Fibrosis Society Patient Registry, Karup, Denmark., Plojoux J; Deparment of Pulmonology, Geneva University Hospital, Switzerland., Regamey N; Department of Paediatric Pulmonology, Cantonal Hospital Lucerne, Switzerland., Rodriguez R; Division of Paediatric Respiratory Medicine and Allergology, Department of Paediatrics, Inselspital Berne University Hospital, Switzerland., Rochat I; Paediatric Pulmonology and Cystic Fibrosis Unit, Division of Paediatrics, Department Woman-Mother-Child, Lausanne University Hospital, Switzerland., Sauty A; Department of Pulmonology, Réseau Hospitalier Neuchâtelois, Switzerland., Schuurmans M; Department of Pulmonology, University Hospital Zurich, Switzerland., Semmler M; Department of Pulmonology, Inselspital Berne University Hospital, Switzerland., Trachsel D; Department of Pulmonology, University Children's Hospital Basel, Switzerland., Walter AL; Department of Pulmonology, Cantonal Hospital St. Gallen, Switzerland., Jung A; Department of Respiratory Medicine, University Children's Hospital Zurich, Switzerland; Departments of Pulmonology and Paediatric Pulmonology, Cantonal Hospital Winterthur, Switzerland; European Cystic Fibrosis Society Patient Registry, Karup, Denmark. Electronic address: andreas.jung@kispi.uzh.ch. |
| المصدر: | Journal of cystic fibrosis : official journal of the European Cystic Fibrosis Society [J Cyst Fibros] 2024 May; Vol. 23 (3), pp. 506-511. Date of Electronic Publication: 2023 Nov 22. |
| نوع المنشور: | Journal Article |
| اللغة: | English |
| بيانات الدورية: | Publisher: Elsevier Country of Publication: Netherlands NLM ID: 101128966 Publication Model: Print-Electronic Cited Medium: Internet ISSN: 1873-5010 (Electronic) Linking ISSN: 15691993 NLM ISO Abbreviation: J Cyst Fibros Subsets: MEDLINE |
| أسماء مطبوعة: | Original Publication: Amsterdam ; New York : Elsevier, c2002- |
| مواضيع طبية MeSH: | Cystic Fibrosis*/epidemiology , Registries*/statistics & numerical data , Data Accuracy*, Humans ; Switzerland/epidemiology ; Child ; Male ; Female ; Adult ; Adolescent ; Reproducibility of Results |
| مستخلص: | Background: Good data quality is essential when rare disease registries are used as a data source for pharmacovigilance studies. This study investigated data quality of the Swiss cystic fibrosis (CF) registry in the frame of a European Cystic Fibrosis Society Patient Registry (ECFSPR) project aiming to implement measures to increase data reliability for registry-based research. Methods: All 20 pediatric and adult Swiss CF centers participated in a data quality audit between 2018 and 2020, and in a re-audit in 2022. Accuracy, consistency and completeness of variables and definitions were evaluated, and missing source data and informed consents (ICs) were assessed. Results: The first audit included 601 out of 997 Swiss people with CF (60.3 %). Data quality, as defined by data correctness ≥95 %, was high for most of the variables. Inconsistencies of specific variables were observed because of an incorrect application of the variable definition. The proportion of missing data was low with <5 % for almost all variables. A considerable number of missing source data occurred for CFTR variants. Availability of ICs varied largely between centers (10 centers had >5 % of missing documents). After providing feedback to the centers, availability of genetic source data and ICs improved. Conclusions: Data audits demonstrated an overall good data quality in the Swiss CF registry. Specific measures such as support of the participating sites, training of data managers and centralized data collection should be implemented in rare disease registries to optimize data quality and provide robust data for registry-based scientific research. Competing Interests: Declaration of Competing Interest The authors declare the following financial interests/personal relationships which may be considered as potential competing interests: Kathleen Jahn, Andreas Jung, Philipp Latzin, Alexander Moeller, Christian Murer, Lutz Naehrlich, Alain Sauty and Jakob Usemann have received honoraria from Vertex Pharmaceuticals. Andreas Jung has received consulting fees and honoraria from EffRx Pharmaceuticals and Viatris. Philipp Latzin has received honoraria from CSL Vifor. (Copyright © 2023. Published by Elsevier B.V.) |
| فهرسة مساهمة: | Keywords: Cystic fibrosis; Data quality; Data quality audit; Patient registries |
| تواريخ الأحداث: | Date Created: 20231123 Date Completed: 20240617 Latest Revision: 20240617 |
| رمز التحديث: | 20240618 |
| DOI: | 10.1016/j.jcf.2023.08.015 |
| PMID: | 37996316 |
| قاعدة البيانات: | MEDLINE |
Full Text via ClinicalKey 
Full Text Finder | تدمد: | 1873-5010 |
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| DOI: | 10.1016/j.jcf.2023.08.015 |