Transitional Care for Patients with Congenital Colorectal Diseases: An EUPSA Network Office, ERNICA, and eUROGEN Joint Venture

Cunera M C de Beaufort; Dalia Aminoff; Ivo de Blaauw; Célia Crétolle; Jens Dingemann; Natalie Durkin; Wout F J Feitz; JoAnne Fruithof; Caterina Grano; Carmen Mesas Burgos; Nicole Schwarzer; Graham Slater; Tutku Soyer; Cristiano Violani; Rene Wijnen; Paolo de Coppi; Ramon R Gorter; Transition of Care study group

doi:10.1016/j.jpedsurg.2023.06.008

Transitional Care for Patients with Congenital Colorectal Diseases: An EUPSA Network Office, ERNICA, and eUROGEN Joint Venture

J Pediatr Surg. 2023 Dec;58(12):2319-2326. doi: 10.1016/j.jpedsurg.2023.06.008. Epub 2023 Jun 14.

Authors

Cunera M C de Beaufort¹, Dalia Aminoff², Ivo de Blaauw³, Célia Crétolle⁴, Jens Dingemann⁵, Natalie Durkin⁶, Wout F J Feitz⁷, JoAnne Fruithof⁸, Caterina Grano⁹, Carmen Mesas Burgos¹⁰, Nicole Schwarzer¹¹, Graham Slater¹², Tutku Soyer¹³, Cristiano Violani¹⁴, Rene Wijnen¹⁵, Paolo de Coppi⁶, Ramon R Gorter¹⁶; Transition of Care study group

Collaborators

Transition of Care study group:
Olivier Abbo, Rossella Angotti, Sanja Sindjic Antunovic, Stefan Anzelewicz, Christoph Arneitz, Marion Arnold, Zaki Assi, Emrah Aydin, Quentin Ballouhey, Patricia Barila, Sonia Basson, Manuel Besendorfer, Antonio Di Cesare, Anne Dariel, Inga Dekeryte, Ivona Djordjevic, Vincenzo Domenichelli, Lise Duranteau, Cigdem Durakbasa, Anne-Karoline Ebert, Verena Ellerkamp, Francesco Fascetti Leon, Margit Fisch, Araceli García Vazquez, Christoph Gielok, Gulnur Gollu Bahadir, Frank-Martin Haecker, Gundela Holmdahl, Sabine Irtan, Ceri Jones, Mohit Kakar, Ali Keshtgar, Kolja Kvist, Francesco Laconi, Marie Laurance, Eduardo Lopez Candel, Johanna Ludwiczek, Barbara Ludwikowski, Francesco Macchini, Mark Malota, Lucas Matthyssens, Benjamin Martin, Leopoldo Martinez, Paola Midrio, Marc Miserez, Giovanni Mosiello, Francisco Murcia Pascual, Bernardo Nunez, Roger Olén, Can İhsan Öztorun, Ayse Parlak, Alessio Pini Prato, Anna Poupalou, Marcos Prada Arias, Jordi Prat, Carlos Pueyo Gil, Luis Felipe Avila Ramirez, Bertram Reingruber, Giovanna Riccipetitoni, Silke Riechardt, Toni Risteski, Wojciech Rogula, Udo Rolle, Carmelo Romeo, Frank-Mattias Schäfer, Gabriella Scirè, Rony Sfeir, Georg Singer, Pim Sloots, Michael Stanton, Pernilla Stenström, Maria Stockinger, M Surhan, Janne Suominen, Julia Syed, Isabelle Talon, Halil Ibrahim Tanriverdi, Osman Uzunlu, Laura Valfre, Marieke Witvliet, Elisa Zambaiti, Cinzia Zanatta, Augusto Zani

Affiliations

¹ Emma Children's Hospital Amsterdam UMC, Location University of Amsterdam, Department of Pediatric Surgery, Meibergdreef 9, Amsterdam, the Netherlands; Amsterdam UMC, University of Amsterdam, Gastroenterology and Hepatology, Amsterdam Gastroenterology Endocrinology Metabolism, Meibergdreef 9, Amsterdam, the Netherlands; Amsterdam UMC, University of Amsterdam, Amsterdam Reproduction and Development Research Institute, Meibergdreef 9, Amsterdam, the Netherlands. Electronic address: [email protected].
² Italian Patient's Organization for ARM (AIMAR) - Patient Organization, Via Nomentana, Rome, Italy.
³ Department of Surgery - Division of Pediatric Surgery, Radboudumc Amalia Children's Hospital, Nijmegen, the Netherlands.
⁴ Necker-Enfants Malades University Hospital, Paris, France.
⁵ Department of Pediatric Surgery, Hannover Medical School, Hannover, Germany.
⁶ Stem Cell and Regenerative Medicine, DBC and BRC NIHR, Great Ormond Street Institute of Child Health, University College London, London, United Kingdom.
⁷ Department of Urology, Division of Pediatric Urology, Radboudumc Amalia Children's Hospital, Nijmegen, the Netherlands.
⁸ EAT - Esophageal Atresia Global Support Groups, Stuttgart, Germany; VOKS - Vereniging voor Ouderen en Kinderen met een Slokdarmafsluiting, Hellendoorn, the Netherlands.
⁹ Department of Psychology, Faculty of Medicine and Psychology, University of Rome Sapienza, Rome, Lazio, Italy.
¹⁰ Department of Pediatric Surgery, Karolinska University Hospital, Stockholm, Sweden.
¹¹ SoMA, The German Patient Support Organization for Anorectal Malformations and Hirschsprung Disease, Munich, Germany.
¹² EAT - Esophageal Atresia Global Support Groups, Stuttgart, Germany; Lead ePAG (Patient Representative), ERN ERNICA.
¹³ Department of Pediatric Surgery, Hacettepe University, Ankara, Turkey.
¹⁴ Sapienza University of Rome, Rome, Italy.
¹⁵ Department of Pediatric Surgery, Erasmus MC Sophia Children's Hospital, Rotterdam, the Netherlands.
¹⁶ Emma Children's Hospital Amsterdam UMC, Location University of Amsterdam, Department of Pediatric Surgery, Meibergdreef 9, Amsterdam, the Netherlands.

PMID: 37438237
DOI: 10.1016/j.jpedsurg.2023.06.008

Abstract

Background: Transition of care (TOC; from childhood into adulthood) of patients with anorectal malformations (ARM) and Hirschsprung disease (HD) ensures continuation of care for these patients. The aim of this international study was to assess the current status of TOC and adult care (AC) programs for patients with ARM and HD.

Methods: A survey was developed by members of EUPSA, ERN eUROGEN, and ERNICA, including patient representatives (ePAGs), comprising of four domains: general information, general questions about transition to adulthood, and disease-specific questions regarding TOC and AC programs. Recruitment of centres was done by the ERNs and EUPSA, using mailing lists and social media accounts. Only descriptive statistics were reported.

Results: In total, 82 centres from 21 different countries entered the survey. Approximately half of them were ERN network members. Seventy-two centres (87.8%) had a self-reported area of expertise for both ARM and HD. Specific TOC programs were installed in 44% of the centres and AC programs in 31% of these centres. When comparing centres, wide variation was observed in the content of the programs.

Conclusion: Despite the awareness of the importance of TOC and AC programs, these programs were installed in less than 50% of the participating centres. Various transition and AC programs were applied, with considerable heterogeneity in implementation, content and responsible caregivers involved. Sharing best practice examples and taking into account local and National Health Care Programs might lead to a better continuation of care in the future.

Level of evidence: III.

Keywords: Adult care; Anorectal malformation; Hirschsprung disease; Transition of care.

MeSH terms

Adult
Anorectal Malformations*
Child
Colorectal Neoplasms*
Hirschsprung Disease*
Humans
Surveys and Questionnaires
Transitional Care*