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A Quality Assessment of the ARM-Net Registry Design and Data Collection.
Hageman, Isabel C; van der Steeg, Hendrik J J; Jenetzky, Ekkehart; Trajanovska, Misel; King, Sebastian K; de Blaauw, Ivo; van Rooij, Iris A L M.
Afiliación
  • Hageman IC; Department of Surgery-Pediatric Surgery, Radboudumc Amalia Children's Hospital, Nijmegen, the Netherlands; Surgical Research, Murdoch Children's Research Institute, Melbourne, Australia. Electronic address: isabel.hageman@radboudumc.nl.
  • van der Steeg HJJ; Department of Surgery-Pediatric Surgery, Radboudumc Amalia Children's Hospital, Nijmegen, the Netherlands.
  • Jenetzky E; Faculty of Health/School of Medicine, Witten/Herdecke University, Witten, Germany; Department of Child and Adolescent Psychiatry and Psychotherapy, University Medical Center of the Johannes-Gutenberg-University, Mainz, Germany.
  • Trajanovska M; Surgical Research, Murdoch Children's Research Institute, Melbourne, Australia; Department of Paediatrics, University of Melbourne, Melbourne, Australia.
  • King SK; Surgical Research, Murdoch Children's Research Institute, Melbourne, Australia; Department of Paediatrics, University of Melbourne, Melbourne, Australia; Department of Paediatric Surgery, The Royal Children's Hospital, Melbourne, Australia.
  • de Blaauw I; Department of Surgery-Pediatric Surgery, Radboudumc Amalia Children's Hospital, Nijmegen, the Netherlands.
  • van Rooij IALM; Department for Health Evidence, Radboudumc, Nijmegen, the Netherlands.
J Pediatr Surg ; 58(10): 1921-1928, 2023 Oct.
Article en En | MEDLINE | ID: mdl-37045715
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.
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Enfermedades Raras / Malformaciones Anorrectales Tipo de estudio: Guideline Aspecto: Patient_preference Límite: Child / Humans Idioma: En Revista: J Pediatr Surg Año: 2023 Tipo del documento: Article Pais de publicación: Estados Unidos
Texto completo
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Texto completo: 1 Colección: 01-internacional Base de datos: MEDLINE Asunto principal: Enfermedades Raras / Malformaciones Anorrectales Tipo de estudio: Guideline Aspecto: Patient_preference Límite: Child / Humans Idioma: En Revista: J Pediatr Surg Año: 2023 Tipo del documento: Article Pais de publicación: Estados Unidos