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Abstract
Introduction Pulmonary arterial hypertension (PAH) is a complex condition affecting quality of life, characterised by high blood pressure in the pulmonary arteries leading to heart strain. PAH’s impact extends beyond physical symptoms, influencing emotional and social well-being, particularly in women where it affects sexual health and pregnancy outcomes. Despite medical advancements, the disease’s full impact on women’s lives is under-researched, especially regarding sexual experiences and health. This systematic review and qualitative meta-synthesis aim to address this gap by examining women’s experiences with PAH, informing clinical practice and patient-centred care. The protocol outlines a methodology for synthesising qualitative research to amplify women’s voices and enhance our understanding of PAH’s impact on female patients.
Methods and analysis Search strategy: A systematic review and thematic meta-synthesis of qualitative research studies and qualitative components of mixed-methods studies will be conducted. The systematic review will be guided by the Joanna Briggs Institute methodological framework. A comprehensive search will encompass seven electronic databases and search engines (Ovid Medline, CINAHL, EMBASE, APA PsycINFO, Cochrane Database of Systematic Reviews, PubMed, Scopus) and grey literature sources (ProQuest Dissertations, ClinicalTrials.gov). Building on insights from previous reviews centred on symptom-related experience and sexual health. Two review authors will independently conduct the screening and data extraction processes. Discrepancies will be resolved through consensus or discussion with a third review author. The review will include English studies from database inception. Findings will be presented graphically and tabularly, together with a narrative description.
Assessment of confidence and data synthesis: The meta-synthesis used thematic analysis, importing study transcripts and notes into NVivo software. Data-derived codes formed categories, which were then iteratively refined into broader themes reflecting women’s experiences with sex and childbirth while living with PAH.
Ethics and dissemination External ethical approval is not required as this review is a retrospective review, which is undertaking secondary analysis of publicly available primary data. The results will be disseminated in peer-reviewed journals and presented in conference papers and elsewhere. This protocol is registered with PROSPERO prospective database of systematic review.
PROSPERO registration number CRD42024529342.
- Health
- Cardiovascular Disease
- Patient-Centered Care
- Nursing Care
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STRENGTHS AND LIMITATIONS OF THIS STUDY
The systematic review will employ a comprehensive methodology aligned with the Cochrane methodological framework, underpinned by the framework of Hilary Thomson and Mhairi Campbell with the support of Dario Sambunjak and colleagues from Cochrane’s People Services Department.
The heterogeneity in patients with pulmonary arterial hypertension across gender and countries regarding age, roles and symptoms means significant diversity in their experience and response affecting the disease; we hope to capture this through the review findings as an important link between female patients’ response in influencing sexual health and health-related quality of life.
Two reviewers will independently screen titles/abstracts and full-text articles using ReviewManager (RevMan) and then extract data using an adapted Cochrane data extraction template.
To ensure rigour and transparency, the review will document each step and any discrepancies between reviewers will be resolved through consensus or third-party arbitration, ensuring a thorough and unbiased synthesis of the evidence.
This systematic review focused exclusively on English-language publications, which may introduce potential biases that could limit the reliability and validity of the study findings.
Background
Pulmonary arterial hypertension (PAH) is a disease marked by elevated blood pressure within the pulmonary arteries. This condition can cause the narrowing, obstruction or destruction of lung blood vessels, which in turn slows blood flow and raises pulmonary arterial pressure. As a result, the heart has to exert more effort to circulate blood through the lungs. If left untreated, this increased workload can ultimately result in right ventricular failure and death.1 Despite advancements in right heart catheterisation technology and new targeted drugs, PAH continues to significantly impact patients’ physical, psychological and social well-being. Patients experience symptoms such as dyspnoea, fatigue, chest pain, anxiety, depression and financial burden.2 3. Sexual health is a particularly important aspect of quality of life for women with PAH, as the majority of patients with idiopathic PAH—nearly 80%—are female, with many in their reproductive years.4 For women with PAH, the physical symptoms may limit sexual activity, while psychological symptoms can affect desire and relationship dynamics, even maternal injuries during pregnancy and childbirth can be severe, permanent and even fatal.5 Moreover, previous studies have found that high degrees of sexual dysfunction were observed in women treated with parenteral prostanoids.6 All these can severely impair health-related quality of life (HRQoL).7
Sexual health encompasses a broad range of issues, including sexual function, satisfaction and overall well-being.8 The symptoms and treatment modalities can have profound implications on the sexual experiences of patients with PAH. Furthermore, the fear of intimacy due to concerns about disease progression or the potential impact on pregnancy adds another layer of complexity to their sexual health.9 Understanding the lived experiences of female patients with PAH is crucial for healthcare providers to deliver patient-centred care and improve quality of life that addresses the holistic needs of patients.10 Despite the recognised importance of sexual health in the overall well-being of individuals with chronic illnesses, the voices of women living with PAH often go unheard, highlighting a need for research that focuses on their unique perspectives and experiences in this under-researched domain. This study focuses on women with PAH as pregnancy and childbirth can significantly affect their condition, an aspect that is less explored in studies including male patients.11 Several qualitative studies have explored the experiences of patients with PAH, highlighting challenges in diagnosis, daily life impact, coping mechanisms and emotional well-being.12–17 A synthesis by Rawlings and colleagues included articles reflecting the experiences of over 1900 individuals with PH, identifying themes related to diagnosis, treatment, prognosis, healthcare professionals, the impact of PH and coping strategies.18 This review emphasises the role of educational and psychological therapies in supporting patients with PAH.
With myriad treatment options now available, the HRQoL has been greatly improved in disease management.19 Despite a more consistent focus on HRQoL in PAH research, we do not fully understand the sexual health-related quality of life (SHRQoL) of patients with PAH especially of female patients with PAH.20 21 This systematic review and qualitative meta-synthesis will conduct a comprehensive search and aim to fill this gap by synthesising the qualitative research on the experiences of women living with PAH. We aim to elucidate the challenges encountered, coping mechanisms adopted, and support needed by these individuals.
This protocol outlines the methodology and approach that will be taken to conduct a systematic review and qualitative meta-synthesis of the symptom-related experiences and sexual health of female patients with PAH. It sets the stage for a research endeavour that seeks to amplify the voices of women with PAH and contribute to a more patient-centred understanding of the impact and management on the disease.
Methods
Design
The protocol was registered with the International Prospective Register of Systematic Reviews (PROSPERO) in April 2024, and the registration number is CRD42024529342. This protocol was designed following the guidelines set forth by the Cochrane Effective Practice and Organization of Care: Qualitative Evidence Synthesis,22 while the reporting follows the Preferred Reporting Items for Systematic Review and Meta-Analysis Protocols (PRISMA-P) 2015 statement23 shown in online supplemental appendix I. The study is tentatively scheduled to run from 1 April 2024 to 31 January 2025. In the event of this protocol is registered but not yet published, we will update the registration record by submitting a modification request through the PROSPERO portal, detailing the changes and their rationale, ensuring all revisions are documented and auditable within the registry.
Supplemental material
Search strategy
The search strategy will be used for the literature search by three steps.24 For step 1, a preliminary search of the PubMed database will be performed on 1 April 2024, analysing the titles, abstracts, subject terms and MeSH terms in the articles. For step 2, a comprehensive search will be undertaken across all included databases using keywords, subject word and random word; the databases will include Ovid Medline, CINAHL, EMBASE, APA PsycINFO, Cochrane Database of Systematic Reviews, PubMed, Scopus and grey literature sources (ProQuest Dissertations, ClinicalTrials.gov). This task will be finished on 1 May 2024. For step 3, all reference lists of the included literature published before 1 June 2024 will be searched to complement other relevant literature. The search strategy for databases is shown in online supplemental appendix II. The data synthesis for this study is expected to begin in December 2024.
Eligibility criteria
The inclusion criteria for literature in this study are qualitative studies. The types of participants focus on female patients diagnosed with PAH (aged ≥18 years), regardless of their pregnancy status. The eligible research is in English, and the full text can be obtained. Studies focusing on PAH that do not include female patients will be excluded.
Identification and selection of studies
The results of the electronic database search will be imported to EndNote V.X9 reference management software and will use the automated ‘Find Duplicates’ function to exclude any duplicates. The literature will be independently screened by two researchers (LY, YH) trained in systematic review methodology, who will read the titles and abstracts for the preliminary screening and then read the full texts for secondary screening and eligibility using a standardised form. Ineligible studies will be excluded, and the reason for exclusion will be recorded. Eligible studies will be included in the analysis. Differences encountered during the screening process will be resolved by the two investigators through negotiation or by discussion with a third investigator (FM). The screening process will be reported using a PRISMA flow chart. We plan to complete the selection of studies on 31 October 2024.
Assessment of methodological quality of included studies
The GRADE-CERQual (Confidence in the Evidence from Reviews of Qualitative Research) evidence grading method will be used to grade the quality of evidence of the synthesised findings.25 The GRADE CERQual approach is designed to help researchers of qualitative evidence make informed judgments about the trustworthiness and applicability of the findings.26 27 The Critical Appraisal Skills Programme checklist for qualitative research will be used to assess methodological limitations of qualitative research28 of which the utilisation methods is presented in table 1. The final confidence rating would be classified into one of the following categories: ‘high’, ‘moderate’, ‘low’ or ‘very low’.
The utilisation of the CASP tool for assessing methodological limitations of qualitative studies
Two researchers (LY, YH) will independently use the 10-item Critical Appraisal Checklist for qualitative research to assess the methodological quality of the included studies.26 Each question is rated ‘yes’, ‘no’, ‘cannot tell’. It was necessary to achieve a minimum affirmative response in at least six of the domains. Studies fully meeting the above criteria, indicating a low risk of bias, will be graded A. Studies partially meeting the minimum criteria, indicating a moderate risk of bias, will be graded B. Finally, studies not meeting the above quality standards at all, indicating a high risk of bias, will be graded C. Studies with grades of A and B will be included in the qualitative meta-synthesis. Differences encountered by the two investigators (LY, YH) during the evaluation process will be resolved through negotiation or discussion with a third investigator (FM).
Data extraction
Data extraction was performed by two reviewers (ZY, ZR) using a standardised extraction form. The form was pilot-tested on a sample of articles and refined as necessary. In addition to parameters required for quality appraisal of included studies and thematic analyses, data extraction will contain the following data parameters:
Study identifiers (including authors, publication date and country)
Context
Aims and objectives
Study design
Sampling frame and recruitment
Participants
Method of data collection and analysis
Summary of major study findings
Data synthesis
The meta-synthesis process involved a thematic analysis approach.29 Transcripts and field notes from the included studies were imported into NVivo (Lumivero) qualitative analysis software. Codes were inductively generated from the data and organised into categories.30 Through an iterative process, these categories were refined and synthesised into overarching themes that captured the essence of the women’s experiences with sex and delivery during pregnancy with PAH.
Ethics and dissemination
External ethical approval is not required as this review is a retrospective review, which is undertaking a secondary analysis of publicly available primary data. The results will be disseminated in peer-reviewed journals and presented in conference papers and elsewhere.
Patient and public involvement
The qualitative meta-synthesis will involve a secondary analysis of publicly available primary data; consequently, there will be no participant or public involvement.
Potential bias and countermeasures
In the realm of qualitative research synthesis, potential biases can undermine the dependability and potency of study outcomes. These biases include information bias from inaccurate variable measurement, sampling bias when the sample fails to mirror the intended population, and both researcher and reporting biases that can skew result interpretation or lead to selective outcome reporting.31 To mitigate such risks, we’ve implemented standardised training for our research team, enforced consistent data collection protocols and employed a strategy of collaborative, independent analysis by multiple researchers.
Discussion
This synthesis will establish what is known about the components and processes of experience and sexual health concerns for female patients with PAH. It is hoped that the conclusion of this study can provide a theoretical basis for medical staff to combine multiple resources for patients with PAH and provide a new perspective for improving their HRQoL.
Strengths and limitations of this study
The strengths of this study include the adoption of a comprehensive methodology in line with the Cochrane methodological framework. We aim to capture the significant diversity in experiences and responses among female patients with PAH, particularly focusing on the relationship between female patients’ responses and their sexual health and quality of life. The review process will be rigorous, with two independent reviewers using RevMan to screen titles/abstracts and full-text articles, and an adapted Cochrane data extraction template to ensure consistency. Discrepancies will be resolved through consensus or third-party arbitration, maintaining the integrity and transparency of the review. However, a limitation is the focus on English-language publications, which may introduce biases affecting the reliability and validity of the study’s findings.
Significance and impact of this review
In the published literature, pulmonary hypertension-related qualitative research has mainly focused on social, cultural and psychological aspects and the impact of disease-related symptoms on emotional and sexual health, and some qualitative research has explored these attributes and behaviours by individuals experiencing and reporting symptoms. This review represents a novel approach to illness perception research. The synthesis and interpretation of the preliminary research of this study will increase the breadth and depth of the findings of the contribution, listen to and understand the feelings and needs of patients with pulmonary hypertension, and provide reference for better healthcare and guidance for them.
Ethics statements
Patient consent for publication
References
Footnotes
Contributors FM and LY contributed equally to this work. YH was responsible for study design and data integration. FM assisted with study design and literature search as well as editing the manuscript. LY and ZY were responsible for literature analysis and data extraction. All authors approved the final version of the manuscript submitted and agreed to be accountable for all aspects of the work. FM is responsible for the overall content as guarantor.
Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.
Competing interests None declared.
Patient and public involvement Patients and/or the public were not involved in the design, or conduct, or reporting, or dissemination plans of this research.
Provenance and peer review Not commissioned; externally peer reviewed.
Supplemental material This content has been supplied by the author(s). It has not been vetted by BMJ Publishing Group Limited (BMJ) and may not have been peer-reviewed. Any opinions or recommendations discussed are solely those of the author(s) and are not endorsed by BMJ. BMJ disclaims all liability and responsibility arising from any reliance placed on the content. Where the content includes any translated material, BMJ does not warrant the accuracy and reliability of the translations (including but not limited to local regulations, clinical guidelines, terminology, drug names and drug dosages), and is not responsible for any error and/or omissions arising from translation and adaptation or otherwise.