A Quality Assessment of the ARM-Net Registry Design and Data Collection.

Hageman IC, van der Steeg HJ, Jenetzky E, Trajanovska M, King SK, de Blaauw I, van Rooij IA, Aminoff D, Amerstorfer E, Till H, Bagolan P, Iacobelli B, Çavuşoğlu H, Ozen O, Deluggi S, Ludwiczek J, Divarci E, Fanjul M, Fascetti-Leon F, Vázquez AG, Giné C, Gorter R, de Jong J, Goseman J, Lacher M, Grano C, Grasshoff-Derr S, Haanen M, Leva E, Morandi A, Lisi G, Makedonsky I, Marcelis C, Midrio P, Miserez M, Mohideen M, PiniPrato A, Reck-Burneo C, Reutter H, Rohleder S, Samuk I, Schmiedeke E, Schwarzer N, Sloots P, Stenström P, Verhaak C, Vilanova-Sánchez A, Volk P, Witvliet M (2023)

Publication Type: Journal article

Publication year: 2023

Journal

Book Volume: 58

Pages Range: 1921-1928

Journal Issue: 10

DOI: 10.1016/j.jpedsurg.2023.02.049

Abstract

BACKGROUND: Registries are important in rare disease research. The Anorectal Malformation Network (ARM-Net) registry is a well-established European patient registry collecting demographic, clinical, and functional outcome data. We assessed the quality of this registry through review of the structure, data elements, collected data, and user experience. MATERIAL AND METHODS: Design and data elements were assessed for completeness, consistency, usefulness, accuracy, validity, and comparability. An intra- and inter-user variability study was conducted through monitoring and re-registration of patients. User experience was assessed via a questionnaire on registration, design of registry, and satisfaction. RESULTS: We evaluated 119 data elements, of which 107 were utilized and comprised 42 string and 65 numeric elements. A minority (37.0%) of the 2278 included records had complete data, though this improved to 83.5% when follow-up elements were excluded. Intra-observer variability demonstrated 11.7% incongruence, while inter-observer variability was 14.7%. Users were predominantly pediatric surgeons and typically registered patients within 11-30 min. Users did not experience any significant difficulties with data entry and were generally satisfied with the registry, but preferred more longitudinal data and patient-reported outcomes. CONCLUSIONS: The ARM-Net registry presents one of the largest ARM cohorts. Although its collected data are valuable, they are susceptible to error and user variability. Continuous evaluations are required to maintain relevant and high-quality data and to achieve long-term sustainability. With the recommendations resulting from this study, we call for rare disease patient registries to take example and aim to continuously improve their data quality to enhance the small, but impactful, field of rare disease research. LEVEL OF EVIDENCE: V.

Involved external institutions

Radboud University Nijmegen Medical Centre / Radboudumc of voluit Radboud Universitair Medisch Centrum (UMC) Netherlands (NL) Medizinische Universität Graz Austria (AT) Ospedale Pediatrico Bambino Gesu Italy (IT) Gazi University Turkey (TR) Kepler Universitätsklinikum (KUK) Austria (AT) University Hospital Leuven (UZ) / Universitaire ziekenhuizen Leuven Belgium (BE) Murdoch Childrens Research Institute Australia (AU) Medizinische Universität Wien Austria (AT) Schneider Children's Medical Center Israel (IL) Klinikverbund Bremen (Gesundheit Nord) Germany (DE) Ege University / Ege Üniversitesi Turkey (TR) Hospital General Universitario Gregorio Marañón Spain (ES) University of Padua / Universita degli Studi di Padova Italy (IT) Hospital Universitario 12 de Octubre Spain (ES) Vall d'Hebron University Hospital / Hospital Universitari Vall d'Hebron Spain (ES) Amsterdam University Medical Centers (Amsterdam UMC) / Amsterdam Universitair Medische Centra Netherlands (NL) Universitätsklinikum Leipzig Germany (DE) Università degli studi "La Sapienza" Italy (IT) Bürger­hospital Frankfurt Germany (DE) Fondazione IRCCS Ca' Granda - Ospedale Maggiore Policlinico Italy (IT) Ospedale Civile Santo Spirito Italy (IT) SoMA Austria Austria (AT) Azienda Ospedaliera Nazionale SS.Antonio e Biagio e C.Arrigo Italy (IT) Rheinische Friedrich-Wilhelms-Universität Bonn Germany (DE) SoMA e.V. Germany (DE) Erasmus MC - Sophia Children’s Hospital Netherlands (NL) Lund University / Lunds universitet Sweden (SE) Hospital Universitario La Paz Spain (ES) Universitätsklinikum Heidelberg Germany (DE) Wilhelmina Kinderziekenhuis Netherlands (NL) Universität Witten/Herdecke Germany (DE) Dnipropetrovsk Regional Clinical Hospital named after I.I. Mechnykov / Dnepropetrovsk Children's Municipal Clinical Hospital #5 Ukraine (UA) Ospedale Ca' Foncello Italy (IT)

How to cite

APA:

Hageman, I.C., van der Steeg, H.J., Jenetzky, E., Trajanovska, M., King, S.K., de Blaauw, I.,... Witvliet, M. (2023). A Quality Assessment of the ARM-Net Registry Design and Data Collection. Journal of Pediatric Surgery, 58(10), 1921-1928. https://dx.doi.org/10.1016/j.jpedsurg.2023.02.049

MLA:

Hageman, Isabel C., et al. "A Quality Assessment of the ARM-Net Registry Design and Data Collection." Journal of Pediatric Surgery 58.10 (2023): 1921-1928.

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