Work Package 7: Efficacy and acceptability of fetal surgery

Professor Neil Marlow, Professor Paolo De Coppi, Professor Jan Deprest, Professor Anna David

About this work package

This work package explores the efficacy and acceptability of fetal surgery. To achieve this, we have created an ethical framework for fetal surgery in the UK and will evaluate the acceptability of this technology to affected women, as well as develop condition-specific outcome measures for future research. We will also explore the context of fetal surgery, including policies in place for adverse events and the perceived support available to families considering fetal surgery.    

Advancing technology must be accompanied by the assessment of the impact on our patients and their families. Ensuring the appropriateness of innovative treatments is the core aim of this group, which needs to be based on sound ethical principles. We are developing key measures which will allow researchers to compare outcomes across studies for our target conditions. We are then evaluating the family experience to ensure what we are proposing is presented to patients in an ethical and open manner and will be something that they find an acceptable alternative. 

We are working within GIFT-Surg and with other clinical groups to develop not just an assessment framework, but also support for clinicians working in this area, in order to ensure that families are actively engaged in the decisions that need to be made. Working in this way will produce the best outcomes for our child and the whole family.

Professor Neil Marlow

Work package tasks

Ethical framework

Our review of legal frameworks governing fetal surgery in the jurisdictions of the United Kingdom, European Court of Human Rights, and the United States of America has been completed and published. 

Acceptability of fetal surgery

Following diagnosis of a congenital malformation such as diaphragmatic hernia, or spina bifida, prospective parents face a range of uncertainties regarding the future of their unborn child, and the options provided require major ethical considerations. In the situation under study, termination of pregnancy may be for some parents an alternative option to expectant prenatal management. Fetal therapy provides a tantalising third option for some.

We know little about the processes that women experience in choosing a particular course when fetal surgery is a potential option to improve long-term outcome, but also carries a significant risk to the woman and her fetus. This study therefore will explore the acceptability of fetal interventions to women and their families, and the concepts and strategies women employ in assessing which pathway to choose. Interviews have conducted with parents at different time points to gain a better understanding on how acceptability of fetal surgery is perceived by parents and how this shapes their decision.  

Inclusion has now ceased for the group of parents pregnant with a fetus diagnosed with spina bifida, who have opted for fetal surgery. The results of the analysis of this group are currently processed for publication. 

Core Outcome Set for Myelomeningocele (the COSMiC study)

There has been rapid development in recent prenatal surgical approaches and clear and robust evidence is required to determine their safety and efficacy. To date, clinical trials in this area report multiple outcomes, hindering comparison between trials. To facilitate comparison between studies, a core outcome set is required, which identifies the minimum outcomes recommended for routine measurement and reporting in clinical trials, whilst allowing researchers to add further outcomes to address specific questions. Our goal is therefore to develop, disseminate and implement a core outcome set (COS) for prenatally diagnosed myelomeningocele (COSMiC study), setting the minimum standards when reporting outcomes in spina bifida research. The COS will incorporate the views of key stakeholders including healthcare professionals, researchers, parents and young adults.  

The protocol is complete and has been published in Trials. The systematic review of outcomes is complete and results are currently being written up for publication. Ethical approval for interviews with key stakeholders is underway, to inform the Delphi process planned for mid-2021.  

Parental online support for Fetal Surgery: Spina Bifida

Surgical innovation now allows for a range of fetal surgical options for prospective parents which were not previously routinely available to treat spina bifida, a severe non-lethal congenital anomaly. These choices, however, are complex, as whilst fetal survival and morbidity have improved, surgery presents a range of serious consequential outcomes for both the fetus and the mother. Prospective parents frequently turn to online social support groups for information and to help with their decision making, although little information exists about the nature and content of these groups, and any potential impact they have on parents’ decision making preferences. This study aims to explore the content of one specific UK-based social media platform to identify the nature of support and information shared amongst parental peers. We hope to identify how healthcare professional counselling may be improved, and to understand more about parental concerns that may not be expressed in medical settings. 

UCL Research Ethics Committee application has now been submitted and reviewed and amendments are being made prior to approval. Data collection is anticipated to take place in December 2020. 

Fetal Resuscitation During Fetal Surgery for Spina Bifida: a global review of policy and practice

Surgical innovation now allows for a range of options to prospective parents for their unborn baby diagnosed with spina bifida. Fetal survival and morbidity have improved, however, surgery now presents a range of consequential outcomes for both the fetus and the mother. One such risk is of fetal demise during surgery, which may require in utero fetal resuscitation. If in-utero resuscitation measures fail to stabilise fetal circulation, or in the case of maternal circulatory collapse, emergent fetal delivery and subsequent neonatal resuscitation may be required to increase both maternal and fetal chances of survival. There are few policies available which provide guidance on how to approach the scenario of resuscitation of a viable infant during fetal surgery for spina bifida. The purpose of this survey is to explore current policies and practices for the anticipation of an emergent fetal delivery during fetal surgery for spina bifida repair. 

The UCL Research Ethics Committee has reviewed and approved the study and our questionnaire ready to disseminate to all centres globally. The anticipated completion date is May 2021. 

Sign up for our studies

Fetal surgery interview study: Parental perceptions of fetal surgery is still open for recruitment of parents who have received a diagnosis of a congenital malformation, such as congenital diafragmatic hernia or spina bifida, which may benefit from fetal surgery and who opt for termination of pregnancy, or for postnatal surgery. 

Find out more

 

The COSMiC study will recruit parents of children who have had myelomeningocele (MMC) repair (pre and post natally), and young adults living with spina bifida. We aim to recruit participants from Canada, Australia and the USA following a similar study in the UK. Participation will involve qualitative interviews exploring their experiences and opinions towards outcomes of MMC repair. These interviews will contribute towards the list of outcomes entered into the Delphi process, which will ultimately identify the main outcomes to be reported in spina bifida research. Please contact Katie Gallagher for further information.

Contact Katie Gallagher

Key publications

The legal frameworks that govern fetal surgery in the United Kingdom, European Union, and the United States.  Cao KX, Booth A, Ourselin S, David AL, Ashcroft. (2018).  Prenatal Diagnosis, 2018 Jun, 38(7):475-481. doi: 10.1002/pd.5267

Development of a Core Outcome Set for Fetal MyelomenIngoCele (COSMiC): study protocol. Altoukhi S, Whitehead CL, Ryan G, Deprest J, Joyeux L, Gallagher K, et al. (2020). Trials, 21; 732. Open Access. 

Core outcome sets in women’s and newborn health. A systematic review, methodological quality assessment, and reporting quality assessment informing high impact actions for core outcome set developers and wider stakeholdersGallagher K, Dromey B, David A, Marlow N, Ourselin S, Duffy J. (under review; preprint available at: doi: 10.22541/au.159819265.57456701)