In-person qualitative interviews and focus groups are traditional methods for exploring patients’ behaviours, perceptions, beliefs, unmet needs, and knowledge gaps [16,17,18]. However, as a result of significant technological advancements and changes in people’s comfort level with technology, it has become more common practice to engage patients online [19, 20]; this was amplified during the COVID-19 pandemic when patients and caregivers had to use more virtual channels of engagement. The use of OBB was thus advantageous in this research, which was conducted in mid-2020 shortly after the pandemic had started, thereby enabling caregiver participation from different geographies without the need to travel and/or risk their health.

Moreover, the US Food and Drug Administration has recommended using online qualitative research methods to enable the participation and inclusion of patients from different regions with different impairments and limitations, which would enhance the generalisability of the data [21].

In general, the caregiver experience depends on the stage of illness, type of cancer treatment, patient and caregiver demographic characteristics, the healthcare system and the familial and governmental support network [22]. Caregivers face many challenges when caring for patients with advanced cancer diagnoses and poor prognoses [23, 24]. A patient with advanced cancer and their caregiver can experience physical, emotional, social, spiritual and functional issues [2, 5]. These factors are a burden for caregivers, which might result in emotional distress, often causing them to develop depression and burnout [25]. In many cases, caregivers of patients with advanced cancer experience a higher level of depression compared to the patients themselves [26]. Additionally, caregivers may have increased morbidity and mortality due to the burden of caregiving [23]. Caring for patients with advanced cancer significantly affects caregivers’ lives and choices, along with a high impact on their financial and work status. Research on the burden of caring for patients with advanced cancer suggests insufficient support for informal caregivers, possibly due to the lack of knowledge about their needs [27,28,29].

Patients with higher-risk MDS are predominantly elderly with clinical comorbidities and have limited treatment options. HMAs and transfusion therapy offer limited efficacy with burdensome administration and/or side effects. More effective treatments such as alloHSCT are available to few patients and present a significant treatment burden. A recently published study suggested that MDS has a significant impact on the QoL and mental health of patients with MDS and their caregivers [30]. Interestingly, the burden of MDS was found to be more severe for caregivers than for patients in some areas, such as planning and uncertainty about the future. The study also reported lower average emotional, social, and functional well-being among caregivers compared with patients, but higher physical well-being [30].

We believe that for better patient care, caregivers have a vital role. Findings that give weight to the importance of the role of caregivers from an outcomes-based perspective were observed in a prospective cohort study of 164 care partners at the Cleveland Clinic in the USA. In-hospital care partners were those who intended to spend 5 or more days per week with the adult patient during their hospital stay as they underwent allogeneic bone marrow transplant between 2003 and 2008. Results showed that having the consistent support of an in-hospital care partner was associated with significantly better overall survival (P = 0.017) and relapse-free survival (P = 0.020) versus those without a care partner [31]. Although not yet studied among patients with higher-risk MDS in real-world settings, these findings give weight to a strategy supporting both patients and caregivers to potentially improve outcomes in patients with higher-risk MDS whilst ensuring the caregiver’s well-being.

The findings of this OBB research show that caregivers’ experiences in higher-risk MDS are multifactorial. There is a significant impact and burden on caregivers of patients with higher-risk MDS, which varied depending on the stage of the disease, the choice (or lack of choice) of treatments and the caregiver’s personal situation. The level of care and burden fluctuates along the disease pathway. For example, although the caregiver role for more recently diagnosed patients was perceived as minimal effort, their role increased significantly if a stem cell transplant occurred and/or if there were changes in the patient’s health status (e.g. infection, medication change, managing side effects).

Caregivers are protective of the patient and absorb the patient’s physical, functional and emotional needs; this leads to a range of emotions for the caregiver, such as loneliness, feeling a lack of empathy and that their life has been put on hold.

“We moved to a different state in order to be close to a Centre of Excellence. I knew no one and my whole support system was gone.” (Caregiver B, USA).

Most caregivers were able to manage the physical and functional aspects of care; however, many stated that the bigger unmet need for both patient and caregiver was emotional support, which has not typically been part of the standard of care provided to patients with MDS worldwide. Emotional stress was pervasive, especially at the diagnosis and prognosis stage, and when told to ‘watch and wait’. This stress was amplified when HCPs were perceived to lack knowledge/expertise about MDS, resulting in an additional burden to patients and caregivers as they sought more appropriate care. Additionally, the incomplete picture of MDS provided by clinicians to patients and their families often leads to online information and emotional seeking behaviour [32].

Mental health emerged strongly as a high area of unmet need both for patients and caregivers. There is a lack of mental health support offered, and this seemed to be offered only when the patient was in urgent need (e.g. considered suicide).

There is an opportunity for healthcare systems to provide better support in these areas of emotional and mental health burden.

Additional stress factors include the uncertainty of employment and financial burden; the time burden when balancing other commitments; and the impact on their own health, particularly if they have their own health issues to manage.

Disease experience is negative where there is a lack of knowledge about MDS and what to expect; this is compounded when there is poor communication from HCPs and leads to some caregivers feeling helpless/paralysed, whilst others are motivated to seek further information and second opinions.

“In my darkest times, I called [the] MDS [Foundation] for support. Later after the transplant, I called BeTheMatch for caregiver support. There you get matched with another caregiver who has had a similar experience. That was extremely helpful. At the hospital, we also talked to the social worker and to volunteers who had had a transplant. That was always very emotional but very helpful.” (Caregiver B, USA).

Treatment experience is negative when limited treatment options offer little control of the disease (e.g. HMAs) or provide significant and/or unexpected burden (e.g. transplant). As the physical and cognitive function of the patient declines, the role of the ‘decision-maker’ increases for the caregiver, who then seeks information on the internet and even a second opinion. In many cases, the caregiver acts as a decision-maker and motivator.

“[My husband is] too tired, or he doesn’t want to think about the hard questions that the doctor will be asking him.” (Caregiver A, USA).

The interaction with HCPs can be positive or negative depending on how the caregiver perceives their knowledge, communication skills, transparency and empathy. MDS CoEs provide hope for those who have doubts about their initial HCP care experience. Some caregivers prefer MDS CoEs even when factoring in the increased burdens on time and appointment logistics.

“I was solely responsible. I was [my husband’s] advocate, and being assertive to doctors and nurses was not something I was looking forward to, but I had more strength than I knew.” (Caregiver B, USA).

Information and education among different stakeholders is a dynamic area to address. Patients/caregivers have to navigate through often complex or incomplete information, use non-traditional channels like social media to fill knowledge gaps, and those of an older generation are often not comfortable asking their doctor questions. Patient organisations play a key role in supporting patients and families in this navigation. HCPs may be perceived to lack MDS knowledge, expertise and consensus with their peers. Caregivers sometimes take on the role of information gatherer when the patient cannot handle it or does not wish to know. Caregivers who are overwhelmed by caregiving tasks have less capacity to seek and understand information about disease/treatments.

“I have to explain (MDS) at every appointment, and that can be exhausting.” (Caregiver K, UK).

Although we identified many similarities in caregiver experience and challenges across the three countries, the one area where a notable difference was observed was the general sentiment of UK caregivers towards HCPs. Caregivers in the UK reported feeling invisible, unsupported and even mistreated by HCPs. They found them unsympathetic and unresponsive, lacking knowledge about MDS and an understanding/appreciation for the role of caregivers; this added frustration, stress and a feeling of isolation to the caregivers.

Reasons for the sentiment of UK caregivers towards HCPs could be attributed to dissatisfaction about the lack of clarity around treatment options and outcomes to expect—‘watch and wait’ approach or no treatment; current active treatments with limited efficacy and side effects; transplant as the only potential ‘cure’, which is not suitable for all patients and brings both risks and disappointment; clinical trials of new treatments, which bring hope but also a potential burden to participate, and restricted inclusion criteria. However, these reasons were reported across participants in all three countries.

We believe that the difference in the healthcare system and culture may play a part in the different sentiments reported in the UK. Pressures on HCPs and their availability to spend time with patients may be more challenging in the UK healthcare system, especially in smaller and non-specialist healthcare settings. A multicountry social healthcare listening research conducted to identify why patients with MDS and their caregivers go online identified that the highest motivator in the UK was emotional topics (vs clinical topics in the USA and treatment topics in Canada) [32]. This research suggests a need for more emotional support for UK patients/caregivers, which is not being met by HCP interactions or within their care setting. Hence, they feel a greater frustration towards HCPs.

The other key difference observed was in the experience of adult children of patients versus partners of patients. The burden of caregiving was greater in adult children of patients, as they had their own family lives, relationships, and employment to balance against the demands of caregiving. Whereas partners of patients accepted this was part of their commitment to the patient, and most being older, had few other responsibilities.

The findings of this research indicate a need to identify solutions to better support caregivers of patients with higher-risk MDS, which can be adapted to specific needs at specific time points in the disease journey. We present some ideas here and encourage further discussion among stakeholders who care for patients with MDS.

MDS disease and treatment education for non-specialist HCPs

MDS information/education tailored for patients/caregivers that can be accessed ‘on-demand’ in a modular approach, as well as be provided via HCPs

A digital healthcare tool to monitor/measure the patient mental health/well-being status over time as the disease evolves, with input from multiple care stakeholders (physician, nurse, caregiver, etc.)

A method to capture QoL patient-relevant symptoms and changes in between consultations, to optimise physician consultation time (ideally integrated into the same digital healthcare tool)

A caregiver toolkit that acknowledges the important role of caregivers and provides resources to support them

By providing such resources and supported by knowledgeable HCPs as part of the standard of care for patients with MDS and their caregivers, we believe that the challenges identified in this research may be (for a large number of patients) avoided and not accumulate to a point where stress, mental health and other psychosocial burden impact their physical health and QoL.

The main advantage of conducting qualitative research via an OBB was the ability to capture more informed insights through advanced technology in less time. It enabled researchers to capture the experiences, emotions and influencing factors of a group of caregivers in different geographies that go deeper than a single focus group or survey [33]. The OBB format also mitigates the risk of having participants who dominate the discussion, encourages equal participant involvement and helps ensure an ideal group dynamic [34].

There are a few limitations to this type of research. Firstly, using an OBB does not allow observation of participants, so visual and verbal cues, which may provide additional insights, were not captured. Additionally, since this research method required participants to use technology comfortably and have access to the internet, it may have excluded participants who were unable to do so and potentially excluded different perspectives/experiences. Participants were recruited via patient organisations, whose members are, by nature, more inquisitive and better informed than those who are not part of such organisations.

As a result of the complex nature of an MDS diagnosis, the recruitment would ideally be supported by the treating physicians who can confirm that the patients are classified as higher risk. However, the recruitment by patient advocate leaders knowledgeable about the disease enabled this limitation to be somewhat addressed.

Finally, even though the number of participants for an OBB was adequate in this research to gain deep insights—with five to six participants from each country and a total of 16 participants—the results may not be representative of the broader MDS caregiver population, especially since most of the caregivers were female and Caucasian/white. Further research may be required to validate the findings in a broader caregiver population and across more geographies.