| Literature DB >> 33166271 |
Joakim Crona1, Eric Baudin2, Massimo Terzolo3, Alexandra Chrisoulidou4, Anna Angelousi5, Cristina L Ronchi6,7, Cristina Lamas Oliveira8, Els J M Nieveen van Dijkum9, Filippo Ceccato10, Françoise Borson-Chazot11, Giuseppe Reimondo12, Guido A M Tiberi13, Hester Ettaieb14, Andreas Kiriakopoulos15, Canu Letizia16, Darko Kastelan17, Esthr Osher18, Eugenia Yiannakopoulou19, Giorgio Arnaldi20, Guillaume Assié21,22, Isabel Paiva23, Isabelle Bourdeau24, John Newell-Price25, Karolina M Nowak26, M Tous Romero27, Maria Cristina De Martino28, Maria João Bugalho29, Mark Sherlock30, Marie-Christine Vantyghem31, Michael Conall Dennedy32, Paula Loli33, Patrice Rodien34, Richard Feelders35, Ronald de Krijger36,37, Sam Van Slycke38, Simon Aylwin39, Valentina Morelli40, Laurent Vroonen41, Zulfiya Shafigullina42, Irina Bancos43, Małgorzata Trofimiuk-Müldner44, Marcus Quinkler45, Michaela Luconi16, Matthias Kroiss7,46, Mitsuhide Naruse47, Peter Igaz48,49, Radu Mihai50, Silvia Della Casa51, Alfredo Berruti52, Martin Fassnacht7,46, Felix Beuschlein53,54.
Abstract
Adrenocortical carcinoma (ACC) is an orphan disease lacking effective systemic treatment options. The low incidence of the disease and high cost of clinical trials are major obstacles in the search for improved treatment strategies. As a novel approach, registry-based clinical trials have been introduced in clinical research, so allowing for significant cost reduction, but without compromising scientific benefit. Herein, we describe how the European Network for the Study of Adrenal Tumours (ENSAT) could transform its current registry into one fit for a clinical trial infrastructure. The rationale to perform randomized registry-based trials in ACC is outlined including an analysis of relevant limitations and challenges. We summarize a survey on this concept among ENSAT members who expressed a strong interest in the concept and rated its scientific potential as high. Legal aspects, including ethical approval of registry-based randomization were identified as potential obstacles. Finally, we describe three potential randomized registry-based clinical trials in an adjuvant setting and for advanced disease with a high potential to be executed within the framework of an advanced ENSAT registry. Thus we, therefore, provide the basis for future registry-based trials for ACC patients. This could ultimately provide proof-of-principle of how to perform more effective randomized trials for an orphan disease.Entities:
Year: 2021 PMID: 33166271 DOI: 10.1530/EJE-20-0800
Source DB: PubMed Journal: Eur J Endocrinol ISSN: 0804-4643 Impact factor: 6.664