Dear Editor
We read with interest the article by Liang and colleagues who reported from a pooled analysis that women with a history of infertility, miscarriages and stillbirths were at an increased risk of ischaemic or haemorrhagic stroke.[1] This finding generated considerable media coverage and public discussion, including among those who have experienced miscarriage and stillbirth and/or provide support following miscarriage and stillbirth. We thank the researchers for their post-publishing support for the bereaved parent community and their willingness to answer concerns and queries that were received.
While the topic addressed in this study is important and the findings are intended to be used by health care professionals to improve appropriate monitoring and provide action for women at risk, the media coverage generated by the study has led to unnecessary distress and heightened anxiety for some women who are experiencing the grief of pregnancy loss. Furthermore, the narrative in the article promotes a stigmatising and victim-blaming view of pregnancy loss (e.g., Women with recurrent pregnancy loss are also more likely to have unhealthy behaviours (e.g., smoking), to have obesity, and to experience depression …).
The patient and public involvement (PPI) statement in the article indicates that no “patients” (or members of the stillbirth or pregnancy loss community) were involved in any aspect of this pooled analysis. The absence of PPI is disappointing given the sensitive nature of the topic addressed. As organisations that support and advocate for perinatally bereaved parents, we wish to note the potential consequences of the absence of parent involvement in this research.
PPI in research aims to ensure the voices of those who are most affected by research are heard, included, and able to inform the research itself. Stillbirth and other types of pregnancy loss are highly sensitive topics and the distress experienced by women and families can be immense and long-lasting. Bereaved parents and families offer unique and valuable insights as research is prioritised, conducted, and translated, helping to ensure research quality and relevance. The inclusion of parent voices in a meaningful way is fundamentally important to ensure that research includes an understanding of lived experience and does not add to individuals’ distress. Studies of participants’ experiences of research on sensitive topics suggest that opportunities to participate are valued and that there may be benefits for participants.[2] We are strongly of the view that the engagement of people with lived experience of pregnancy loss would have led to more sensitive communication of results with greater attention to the context in which the messages would be received.
While current levels of experience and knowledge of PPI throughout the research process, especially analysis, are limited, evidence is growing around the benefits of co-creation. [3,4] The importance of PPI extends to desk-based/literature review research, not just research directly interacting with parents. We appreciate the BMJ’s commitment to PPI which acknowledges that research should be carried out with patients, carers, or members of the public, not on them.
Unfortunately, the article by Liang and colleagues does not align with this commitment. It provides a strong example of the need for PPI in research.
References
[1] Liang C, Chung HF, Dobson AJ, Hayashi K, van der Schouw YT, Kuh D, Hardy R, Derby CA, El Khoudary SR, Janssen I, Sandin S. Infertility, recurrent pregnancy loss, and risk of stroke: pooled analysis of individual patient data of 618 851 women. BMJ. 2022 Jun 22;377. https://www.bmj.com/content/377/bmj-2022-070603
[2] Alexander S, Pillay R, Smith B. A systematic review of the experiences of vulnerable people participating in research on sensitive topics. International Journal of Nursing Studies. 2018 Dec 1;88:85-96.
[3] Banner D, Bains M, Carroll S, Kandola DK, Rolfe DE, Wong C, Graham ID. Patient and public engagement in integrated knowledge translation research: are we there yet? Research Involvement and Engagement. 2019 Dec;5(1):1-4.
[4] South A, Hanley B, Gafos M, Cromarty B, Stephens R, Sturgeon K, Scott K, Cragg WJ, Tweed CD, Teera J, Vale CL. Models and impact of patient and public involvement in studies carried out by the Medical Research Council Clinical Trials Unit at University College London: findings from ten case studies. Trials. 2016 Dec;17(1):1-3.
Response to article: Infertility, recurrent pregnancy loss, and risk of stroke: pooled analysis of individual patient data of 618 851 women