‘Nobody Has Ever Spoken to Me About PCD and Fertility Issues’: Fertility Experiences of People With Primary Ciliary Dyskinesia and Their Family Caregivers

IF 3.2 3区医学 Q2 HEALTH CARE SCIENCES & SERVICES

Health Expectations Pub Date : 2025-06-05 DOI:10.1111/hex.70316

Leonie D. Schreck, Sophie Meyer, Eva S. L. Pedersen, Yin Ting Lam, Hansruedi Silberschmidt, Sara Bellu, Living with PCD patient advisory group, Sofía C. Zambrano, Claudia E. Kuehni, Myrofora Goutaki

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引用次数: 0

Abstract

Background

Primary ciliary dyskinesia (PCD) affects fertility in both females and males. To understand the impact and concerns among people with PCD and parents of affected children (family caregivers), we explored how they report their experiences with fertility.

Methods

We used qualitative data from a questionnaire on fertility from Living with PCD, an international participatory study. In optional open-ended comment fields, participants shared their thoughts and experiences related to fertility. We adopted conventional content analysis and analysed the data inductively.

Results

Out of 384 survey respondents, 206 (54%) provided free-text comments that we included in this analysis. We identified five categories illustrating participants' experiences with fertility: (1) challenging experiences of fertility care, ranging from insufficient fertility evidence to poor care from treating physicians, leading to an overall perception of inadequate care related to fertility; (2) PCD-related reproductive concerns, such as pregnancy risks, heritability of PCD and delayed PCD diagnosis; (3) non-PCD-related factors complicating fertility, such as challenges in accessing fertility treatment, age as a limiting factor for fertility and other diseases affecting fertility; (4) psychological impact of infertility, marked by emotional distress, grief and coping strategies; and (5) family caregivers as gatekeepers of fertility information, reflecting their role in managing, delaying, or shaping how and when children learn about fertility.

Conclusion

We need enhanced support and standardised reproductive counselling and healthcare for people with PCD to enable informed decisions on fertility and to reduce the fertility-related concerns and psychological impact faced by many.

Patient or Public Contribution

This participatory study was co-designed with people with PCD and a patient advisory group. Patients actively shaped research priorities, contributed to study design and questionnaire development, and played a key role in data interpretation and dissemination.

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“没有人跟我谈过PCD和生育问题”：原发性纤毛运动障碍患者及其家庭照顾者的生育经历

背景：原发性纤毛运动障碍（PCD）影响男性和女性的生育能力。为了了解PCD患者和受影响儿童的父母（家庭照顾者）的影响和担忧，我们探讨了他们如何报告他们的生育经历。方法采用国际参与性研究《伴PCD生活》中生育问卷的定性数据。在可选的开放式评论栏中，参与者分享了他们与生育有关的想法和经历。采用常规的含量分析方法，对数据进行归纳分析。在384名受访者中，206名（54%）提供了我们纳入本分析的自由文本评论。我们确定了五个类别来说明参与者的生育经历：(1)生育护理的挑战性经历，从生育证据不足到治疗医生的护理不良，导致与生育有关的护理不足的总体看法；(2)与PCD相关的生殖问题，如妊娠风险、PCD遗传和PCD延迟诊断；(3)使生育复杂化的非pcd相关因素，例如难以获得生育治疗、年龄是生育的限制因素以及影响生育的其他疾病；(4)不孕症的心理影响，表现为情绪困扰、悲伤和应对策略；(5)家庭照顾者作为生育信息的守门人，反映了他们在管理、延迟或塑造儿童如何以及何时了解生育方面的作用。结论：我们需要加强对PCD患者的支持和标准化的生殖咨询和保健，以使他们能够在生育方面做出知情决定，并减少许多人面临的与生育有关的担忧和心理影响。这项参与性研究是由PCD患者和患者咨询小组共同设计的。患者积极塑造研究重点，为研究设计和问卷开发做出贡献，并在数据解释和传播中发挥关键作用。

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来源期刊

Health Expectations 医学-公共卫生、环境卫生与职业卫生

CiteScore

5.20

自引率

9.40%

发文量

251

审稿时长

>12 weeks

期刊介绍： Health Expectations promotes critical thinking and informed debate about all aspects of patient and public involvement and engagement (PPIE) in health and social care, health policy and health services research including: • Person-centred care and quality improvement • Patients'' participation in decisions about disease prevention and management • Public perceptions of health services • Citizen involvement in health care policy making and priority-setting • Methods for monitoring and evaluating participation • Empowerment and consumerism • Patients'' role in safety and quality • Patient and public role in health services research • Co-production (researchers working with patients and the public) of research, health care and policy Health Expectations is a quarterly, peer-reviewed journal publishing original research, review articles and critical commentaries. It includes papers which clarify concepts, develop theories, and critically analyse and evaluate specific policies and practices. The Journal provides an inter-disciplinary and international forum in which researchers (including PPIE researchers) from a range of backgrounds and expertise can present their work to other researchers, policy-makers, health care professionals, managers, patients and consumer advocates.