Please use this identifier to cite or link to this item:
https://hdl.handle.net/2440/128575
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Type: | Journal article |
Title: | Development of a core outcome set for fetal Myelomeningocele (COSMiC): study protocol |
Author: | Altoukhi, S. Whitehead, C.L. Ryan, G. Deprest, J. Joyeux, L. Gallagher, K. Drake, J. Church, P. Horn, D. Diambomba, Y. Carvalho, J.C.A. Van Mieghem, T. |
Citation: | Trials, 2020; 21(1):732-1-732-6 |
Publisher: | Springer Nature |
Issue Date: | 2020 |
ISSN: | 1745-6215 1745-6215 |
Statement of Responsibility: | Samar Altoukhi, Clare L. Whitehead, Greg Ryan, Jan Deprest, Luc Joyeux, Katie Gallagher, James Drake, Paige Church, Daphne Horn, Yenge Diambomba, Jose C.A. Carvalho, and Tim Van Mieghem |
Abstract: | Background: Open spina bifida (OSB) is one of the most common congenital central nervous system defects and leads to long-term physical and cognitive disabilities. Open fetal surgery for OSB improves neurological outcomes and reduces the need for ventriculoperitoneal shunting, compared to postnatal surgery, but is associated with a significant risk of prematurity and maternal morbidity. Fetoscopic surgery comes with less maternal morbidity, yet the question remains whether the procedure is neuroprotective and reduces prematurity. Comparison of outcomes between different treatment options is challenging due to inconsistent outcome reporting. We aim to develop and disseminate a core outcome set (COS) for fetal OSB, to ensure that outcomes relevant to all stakeholders are collected and reported in a standardised fashion in future studies. Methods: The COS will be developed using a validated Delphi methodology. A systematic literature review will be performed to identify outcomes previously reported for prenatally diagnosed OSB. We will assess maternal (primary and subsequent pregnancies), fetal, neonatal and childhood outcomes until adolescence. In a second phase, we will conduct semi-structured interviews with stakeholders, to ensure representation of additional relevant outcomes that may not have been reported in the literature. We will include patients and parents, as well as health professionals involved in the care of these pregnancies and children (fetal medicine specialists, fetal surgeons, neonatologists/paediatricians and allied health). Subsequently, an international group of key stakeholders will rate the importance of the identified outcomes using three sequential online rounds of a modified Delphi Survey. Final agreement on outcomes to be included in the COS, their definition and measurement will be achieved through a face-to-face consensus meeting with all stakeholder groups. Dissemination of the final COS will be ensured through different media and relevant societies. Discussion: Development and implementation of a COS for fetal OSB will ensure consistent outcome reporting in future clinical trials, systematic reviews and clinical practice guidelines. This will lead to higher quality research, better evidence-based clinical practice and ultimately improved maternal, fetal and long-term childhood outcomes. |
Keywords: | Spina bifida; Myelomeningocele; Myeloschisis; fetal surgery; fetoscopic surgery; core outcome set; Delphi study, open spina bifida |
Rights: | © The Author(s). 2020. Open Access This article is licensed under a Creative Commons Attribution 4.0 International License, which permits use, sharing, adaptation, distribution and reproduction in any medium or format, as long as you give appropriate credit to the original author(s) and the source, provide a link to the Creative Commons licence, and indicate if changes were made. The images or other third party material in this article are included in the article's Creative Commons licence, unless indicated otherwise in a credit line to the material. If material is not included in the article's Creative Commons licence and your intended use is not permitted by statutory regulation or exceeds the permitted use, you will need to obtain permission directly from the copyright holder. To view a copy of this licence, visit http://creativecommons.org/licenses/by/4.0/. The Creative Commons Public Domain Dedication waiver (http://creativecommons.org/publicdomain/zero/1.0/) applies to the data made available in this article, unless otherwise stated in a credit line to the data. |
DOI: | 10.1186/s13063-020-04668-6 |
Grant ID: | http://purl.org/au-research/grants/nhmrc/1142380 |
Published version: | http://dx.doi.org/10.1186/s13063-020-04668-6 |
Appears in Collections: | Aurora harvest 4 Obstetrics and Gynaecology publications |
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hdl_128575.pdf | Published Version | 636.26 kB | Adobe PDF | View/Open |
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