Background

The term ‘women’ will be used throughout this protocol and is defined as persons assigned female at birth but includes any person who lives with uterine adenomyosis (UA), regardless of gender identity.

UA is a benign, enigmatic, gynaecological disease defined by the ectopic infiltration of uterine cavity lining (endometrium) into the uterine muscle (myometrium) and is of indeterminate aetiology. Chronic pain and abnormal, often very heavy bleeding patterns are common. Uterine enlargement can cause adjacent urinary and bowel symptoms. Infertility and poor pregnancy outcomes are possible clinical manifestations, and the chronic nature of symptoms can impact quality of life (QoL) in terms of medical, psychological and wider sociological issues.1–3 Broad symptomatology makes diagnosis challenging and as a process this can often occur over multiple years. Delays in the establishment of a diagnosis, result in missed potential opportunities to intervene (earlier). Despite recent diagnostic advances using ultrasound imaging4–6 that have created possibilities to identify UA earlier, diagnosis is often still only made following the symptomatic cure of a hysterectomy.7–9 This is especially unhelpful for those wishing to retain their uterus and with the inherent access limitations and risks of surgery, subsequent time delays can lengthen diagnostic processes.

Further compounding these delays are existing societal and cultural conventions and the universal stigma associated with menstruation and period-related health issues, which perpetuates the ongoing lack of research in this field and contribute to the diminished seriousness of women’s health problems.10 11 Breaking down this discourse averseness is vital to encourage open discussion of what diversity of experience is normal and what menstrual variations may be considered abnormal.12 13 A cultural shift is required to not only support improved knowledge and understanding of UA, thus encouraging a more advice-seeking population, but to raise awareness of such conditions within healthcare professional (HCP) communities and avoid women ‘developing more serious and potentially avoidable ill health’.12 Furthermore, inclusive cultural shifts must be acknowledged to address the challenges faced by all populations who menstruate. Only then will we start to progress with constructive discourse, equitable healthcare access and improved HCP knowledge, understanding and engagement.11

Another significant factor that contributes to diagnostic delays is the gender inequalities and distorted societal perception of women’s pain. It is widely recognised that women often face dismissive attitudes when they report pain or discomfort, with their symptoms being attributed to emotional or psychological factors rather than physical ailments. This dismissal of women’s pain stems from deeply ingrained gender stereotypes that portray women as overly emotional and less credible when it comes to their health concerns. As a result, women often experience significant delays in receiving appropriate medical attention, which ultimately leads to worsened health outcomes.14 15

Reassuringly, in the UK, focused policy regarding women’s health is being prioritised more in recent years.13 16 In 2021, with a responsibility to setting direction and leading debate in global and domestic health, the UK Department for Health and Social Care initiated a comprehensive survey aimed at gathering insights into women’s health.17 The response from nearly 100 000 individuals in England has played a pivotal role in shaping the UK government’s inaugural Women’s Health Strategy for England. This strategy incorporates personal perspectives and experiences shared by women, as well as insights from their family members, friends, partners, as well as from HCP communities.

The results of this survey have shed light on crucial areas that require further investigation, with the top five topics being gynaecological conditions (63%), fertility, pregnancy, pregnancy loss, and postnatal support (55%), the menopause (48%), menstrual health (47%), and mental health (39%). One disheartening revelation from the findings is that over 84% of women expressed feeling unheard by HCPs. Their symptoms were often disregarded or not taken seriously during initial consultations with general practitioners and other healthcare providers. Consequently, women frequently found themselves in the position of having to persistently advocate for themselves, enduring multiple visits, months and even years before securing a diagnosis. Moreover, even after receiving a diagnosis, women reported limited opportunities to discuss treatment options and felt that their preferences were frequently disregarded.17

The consequences of not taking women’s health problems seriously are far-reaching. Women may endure prolonged ill health, delayed diagnosis and inadequate treatment, all of which lead to worsened health outcomes and a reduced QoL.18 Moreover, the lack of attention and research on women’s health issues hinders medical advancements and the development of effective treatments specifically tailored to meet the unique needs of women.

Lived experience and burden of disease

Lived experience is a poorly understood concept with varied definitions that can be confused with related experiential constructs. Indeed, the wider concept of patient experience is a complex phenomenon often understood to emerge solely from healthcare interactions.19 20 This emphasis on ‘healthcare systems related’ elements only (quality and responsiveness of services, and health-related political influences), may fail to appreciate ‘patient-related’ elements (lived experience and more subjective influences such as perceptions and expectations). Zakkar presents both elements, combining all ‘determinants’ to construct patient experience and its subsequent ‘manifestations’; patient satisfaction and engagement levels.20 Similarly, it is noted that experience has been extensively debated for many decades in the conceptual discourse around the determinants that may shape an individual’s QoL.21 In the broadest sense, QoL encompasses all facets of living that shape subjective well-being and contentment with life overall,22 within the unique context of an individual’s culture, goals, beliefs, values and anxieties.23 More recently, researchers have focused on health as a single element of that complex, enigmatic process. How an individual functions in life relative to the effects of illness, within their personal perceived health-related QoL (HRQoL), is just as multidimensional and includes physical, psychological, financial and social elements.24 25 HRQoL and how disease may impact overall well-being, has been extensively debated.21 However, in comparison to a simple count of mortality rates, there is no doubt that HRQoL instruments as outcome measures of the diagnostic and treatment journey, provide a more sophisticated and useful gauge of disease status, diagnostic and therapeutic intervention success, and prevention planning.26–28 Scrutinising the expansive and converging experiential concepts further, the theoretical parallels of lived experience and HRQoL are conspicuous. Although focused to cancer specifically, Sitlinger and Zafar’s review of disease burdens (physical, psychosocial and financial) as components of HRQoL that affect ‘well-being and survival’24 draw clear similarities with Zakkar’s wider work into modelling the determinants of lived experience (beliefs, financial burden, social burden and emotional burden).20

This work is motivated from the premise that understanding the presentation of UA, patients’ journeys and the burden that the onset of the condition has on them and their lives, is key to achieving the goal to improve the healthcare experience of women living with UA. Better understanding of the lived experience could strengthen healthcare provision pathways, influence policy decisions, secure funding allocations and raise awareness.

Additionally, Brand and Timmons’s29 exploration into knowledge sharing in healthcare, highlights the value of direct experiential knowledge ensuring that adequate person-centred care is achieved. Indeed, compared with the worth assigned to HCPs’ clinical expertise, the authors expose the missed opportunities and significant undervalue ascribed to knowledge learnt through the direct lived experience and impact of a disease.29 Paradoxically, this is despite increasing recognition of using patient experience to drive healthcare policy improvements and optimise healthcare responses: championing the benefits of appreciating the knowledge from patients as well as from HCPs.30–32

Preliminary search

On 20 September 2022, a preliminary search was conducted in PubMed to estimate the extent and nature of current literature. The search terms used are as described in the search strategy section below. PubMed was chosen because it is a large database that includes MEDLINE and provides wide coverage for this limited exploratory search. The same inclusion criteria used for the formal review were applied to this preliminary search (Methods and analysis section provides more details). This preliminary search identified two papers describing the lived experience of patients with UA, but no literature was identified specifically dealing with perceptions of diagnostic pathways.

Using a grounded theory approach and a sample of 31 semistructured interviews, Nelsen et al33 provide the most significant contribution to date in this field, proposing their study as a first step to understanding the lived experience of UA and its specific symptoms, and acknowledging the need for further similar research. Onward citation counts via CrossRef were examined and did not identify further relevant papers.

Using a content analysis approach (Colaizzi’s 7 steps) and a sample of 18 in-depth interviews, Huang et al’s34 subsequent research into how patients manage their disease and the barriers to doing so effectively, echo some of Nelsen et al’s earlier findings. They identify HCPs’ lack of knowledge directly impacting unfavourably on patients’ QoL.

Additionally, in narrative articles by clinical groups, there is a plea for urgent research into all aspects of this ‘under-recognised, undertreated and understudied’ condition35 and acknowledgement of an urgent need for validated, quality-of-life instruments.36 No systematic reviews (SRs), scoping or otherwise, detailing this phenomenon have been found to date and the apparent limited understanding across the subject area, suggests a research gap warranting this scoping review (ScR) as the first attempt at assessing the current knowledge base.

Adenomyosis and endometriosis

UA is often considered to be the same as endometriosis and indeed, for many years, UA was referred to as endometriosis interna.7 37 Unlike UA, which is confined to the uterus, endometriosis is defined as endometrial tissue found outside the uterus. This close relationship involving ectopic endometrial tissue often gives rise to the assumption of similar symptoms, impact and healthcare needs. Certainly, in Omtvedt et al’s38 survey research exploring what components are needed to develop a multidisciplinary centre for endometriosis and adenomyosis, the authors acknowledge as a limitation of their work the possibility that ‘people with adenomyosis have other, unmet needs’. Undeniably, these conditions may coexist39 but although the pathogenic mechanism of neither condition is fully appreciated, there is an increasing acceptance that they are different entities requiring different care needs.7 37

However, there remains a notable strong association of UA with endometriosis, as historically ‘they were considered—with the exception of ovarian endometriomas—as one disease’.40 Subsequently, it is important to acknowledge the greater body of evidence into the lived experience of endometriosis.41 42 Furthermore, excluding evidence that includes both adenomyosis and endometriosis within the same study, may exclude significant background and contextual information that is considered important to this review. A preliminary search for this protocol initially excluded literature investigating UA within a population with a coexisting diagnosis of another gynaecological pelvic disease. Modifying this stance, a wider search strategy is now proposed to include literature investigating the combined diseases of endometriosis and UA within the same study population, more fully capturing and quantifying work that has been conducted to date.

ScR rationale

The ScR is becoming an established evidence synthesis methodology43 and while necessarily sharing important similarities in terms of trust and transparency, they differ from the traditional SR.43 Indeed, there are accepted and differing indications for choosing to conduct an ScR.44 45 Although not often the case,46 an ScR may be used to explore the feasibility of subsequently progressing onto an SR by assessing the size and scope of evidence available and if the evidence even exists. An ScR aims to identify the key concepts and characteristics of evidence within a much broader topic area, both in question and exploration; mapping the main concepts of the topic and identifying the main sources and nature of evidence that exists. In doing so, the most common reasons cited for conducting ScRs are to identify and assess research gaps within a field of study and influence future research projects.46 Such reviews are commonly adopted when a field of study is emerging or there is not enough homogeneity to enable fair comparisons within available evidence and differing methodological approaches.47 Subsequently, with less attention towards assessing the quality of evidence found and synthesising results to answer a focused narrow question, attention is more to gathering and producing a descriptive output of everything that exists.

A preliminary review of the literature found no SRs and a dearth of peer-reviewed studies in general. This apparent paucity of current evidence suggests the usefulness of a broad ScR to map existing knowledge; define concepts, explore methods used and tabulate study findings in terms of themes and conclusions. This will establish the extent of the research gap to guide future projects, all of which will specifically direct this postgraduate researcher’s dissertation project. As the objective of this ScR is to scope all accessible evidence, it is confidently asserted that this methodological approach is appropriate to the objective of this review.