Sources and consequences of self-doubt in patients with Ehlers-Danlos syndrome: a qualitative study in Europe and North America

Abstract

Objectives

To examine how clinicians’ scepticism regarding patients’ self-reports of subjective symptoms can be internalised, leading to psychosocial and medical harms.

Design

In-depth, semi-structured qualitative interviews with the resulting data analysed using reflexive thematic analysis.

Participants

43 individuals with Ehlers-Danlos syndrome (EDS) from Europe and North America completed a pre-survey, and 39 of those participants completed interviews for this study. Purposive sampling was used to obtain approximately equal numbers of participants with hypermobile EDS and the molecularly defined types of EDS.

Results

Patients with both hypermobile and molecularly defined types of EDS reported high levels of self-doubt, with 73% of survey respondents questioning the extent—and even reality—of their private experiences of pain. Participants attributed much of their self-doubt to repeated dismissal and minimisation of their symptoms in healthcare settings, especially during childhood. Ultimately, self-doubt transformed not merely how they communicated their symptoms but also how they recognised, evaluated and even experienced them at a phenomenological level. While some participants developed coping strategies, others withdrew from the conventional medical system altogether.

Conclusion

These findings have important implications for clinicians, who may inadvertently reinforce self-doubt through discussion of diagnostic uncertainty. Doubt need not be delegitamising. Recognising and mitigating these potential harms requires epistemic humility and attention to the psychosocial dynamics of patient-provider interaction.

STRENGTHS AND LIMITATIONS OF THIS STUDY.

• While previous literature has focused almost exclusively on external acts of clinician scepticism and doubt (like medical gaslighting or invalidation), this study addresses the generally understudied phenomenon of patient self-doubt.

• The qualitative nature of the study provides significant data on sources of doubt, self-doubt and coping strategies in individuals with Ehlers-Danlos syndrome.

• The participant sample was predominantly white and well-educated, which may impact how our results apply to individuals from other demographic groups.

• The participants included individuals receiving healthcare either in North America or Europe. This prevents the study from linking self-doubt to the specific infrastructural features of any single national healthcare model.

Introduction

Chronic pain is a critical issue facing medicine, bioethics and the sociology of illness, among many other disciplines. It eludes the objective measures on which evidence-based medicine relies: It lacks precise and universal biomarkers,¹ thwarts attempts at robust imaging² and presents considerable obstacles to quantification.^{3 4} Individuals experience painful stimuli with profound differences in sensitivity.⁵ As an inherently private experience, chronic pain raises not only diagnostic challenges but social and epistemic challenges as well. In clinical parlance, it is a subjective symptom par excellence, whose presence and severity can only be evaluated through the self-report of those experiencing it. Therefore, its appropriate care depends on both the capacity of patients to report their pain and the capacity of their clinicians to trust them.

The evidentiary gap between the objective measures favoured by modern medicine and the subjective character of chronic pain leads to institutionally motivated scepticism. This doubt about patients’ chronic pain is both a critical and dangerous resource in its treatment. It enables clinicians to discriminate between expert and lay analyses and has played a central role in policy responses to the opioid epidemic, as seen through opioid patient-prescriber agreements and prescription-drug monitoring programmes.^{6 7} For patients, however, doubt can appear as a form of epistemic injustice—in particular as a form of testimonial injustice, whereby individuals are disenfranchised as legitimate and credible conveyors of knowledge due to their social or demographic identity.⁸ This experience can be emotionally and psychically damaging,⁹,¹¹ and disproportionately affects marginalised groups, including women^{12 13} and disabled individuals,¹⁴ compounding existing disparities that these populations face.

But such doubt has more than epistemic effects. When clinicians invalidate their patients’ self-report of pain, it creates material barriers as well: causing diagnostic delays, increasing healthcare utilisation and costs, impeding access to governmental and other resources and decreasing self-worth.¹¹¹⁵,¹⁷ Invalidation can also lead to higher levels of pain-associated disability,¹⁸ which in turn restricts the pursuit of education and career opportunities.¹⁹ Moreover, patients living with chronic pain experience increasing levels of stigma, which negatively impacts numerous facets of their professional and personal lives.^{20 21}

Particularly disturbing, however, is the internalisation of this doubt. It has been suggested that patients may come to adopt the perspective of their sceptical clinicians and discredit or dismiss their interpretation of their own subjective experiences.^{22 23} This self-doubt has the potential to interfere with a patient’s sincere testimony, a significant harm given that self-report is the ‘gold standard’ for pain assessment.²⁴ Freeman and Stewart refer to this as ‘secondary epistemic harm’, as it limits the patient’s authority to make claims to knowledge and leads to ‘broad social ramifications’ insofar as they are labelled as unreliable or untrustworthy.²² However, it may also have more profound and existential consequences. At that level, self-doubt has additionally been linked to dissociative affect and an otherwise unfounded fear of mental illness.^{13 25} Although highly worrisome in its potential to cause harm, the problem of patients’ self-doubt has been understudied.

Patients with Ehlers-Danlos syndrome (EDS) represent a particularly illuminating population in which to study this phenomenon. EDS refers to 13 types of hereditary disorders of connective tissue. While heterogeneous in presentation, most forms are characterised by joint hypermobility, skin hyperextensibility and tissue fragility,²⁶ but the most frequent complaint with which these patients present in clinic is chronic pain.²⁷ The most common type, hypermobile EDS (hEDS), is primarily diagnosed in women,²⁸ a demographic particularly vulnerable to testimonial injustice.²⁹ Research has found a high prevalence of medical and psychosocial harms among these patients due to clinicians’ scepticism,³⁰,³² and one study has suggested that internalised self-doubt may be a driving factor of these harms.³³

While patients with EDS may be particularly subject to and burdened by self-doubt, existing research has been limited to the related but analytically distinct constructs. These include epistemic injustice,³⁴ relative human dignity,³¹ medical gaslighting,³⁵ symptom invalidation,³⁰ social delegitimisation³⁶ and clinician-associated traumatisation.³³ These analyses focus on the source or act of the moral and epistemic harms, rather than the internalisation of those external perspectives and its consequences. To our knowledge, no prior study has rigorously or systematically examined self-doubt as a discrete phenomenon in this population. To address this gap, we conducted in-depth, qualitative interviews with patients living with both hypermobile and molecularly defined types of EDS. In these conversations, we explored the sources, consequences and coping strategies related to self-doubt. We have found not only epistemic harms but also negative effects on self-worth, sensation and perception and health-seeking behaviour.

Methods

Editorial policies and ethical considerations

Ethics approval for this research was received from the Indiana University Institutional Review Board. All participants were informed about the goals of the research and provided their informed consent.

Research team

The research team was multidisciplinary: The primary investigator (CMEH) is a male bioethicist and anthropologist with doctoral-level training in qualitative methods. SLV is a female research assistant whose work focuses on promoting the care of patients with rare disease. CAF is a female medical geneticist with extensive experience in the care of patients living with EDS and hypermobility spectrum disorder. TD is a male bioethicist with a doctorate in philosophy and a postdoctoral fellow in the Center for Bioethics.

Sampling and recruitment

All participants were enrolled in an ongoing study on the lived experiences of patients with EDS and had completed at least one interview with the research team prior to the current study. Because recruiting individuals with rare and ultra-rare types of EDS is challenging, participants were initially recruited through a variety of avenues: clinician referral from a single academic medical centre in the American Midwest; the Ehlers-Danlos Society (https://www.ehlers-danlos.com/), a non-profit organisation promoting advocacy and research for all types of EDS; and Annabelle’s Challenge (https://www.annabelleschallenge.org/), a charity dedicated in particular to patients with vascular EDS (vEDS). As such, participants were recruited from both Europe and North America.

As much research on EDS focuses solely on or over-represents hEDS perspectives in characterising EDS experience, we used purposive sampling to recruit approximately equal numbers of participants with hEDS and the molecularly defined types of EDS (hereafter referred to as mdEDS). Candidates were first invited by email to complete the pre-survey. They were eligible if they had been diagnosed with any type of EDS by a clinician, were 18 years of age or older and were comfortable communicating in English. A random subset of survey respondents was then invited to complete an interview. Consistent with interpretive, qualitative research, recruitment was not determined in advance by constructs of saturation, but rather continued until team members agreed that our results were deep and broad enough to allow for the recognition of themes and patterns within a rich context.³⁷ Interviewees were offered a US$25 Amazon gift card for the completion of the interview.

Data collection

The survey instrument was developed based on knowledge gaps identified in our previous qualitative work^{30 33} as well as a review of the literature. Items included both closed, Likert and open-ended questions and asked respondents to define terms related to doubt and self-doubt; characterise how others assess their subjective symptoms, including pain; and reflect on the impact of self-doubt on their lives and relationships, among other topics. Surveys were developed and circulated in Qualtrics, and collection lasted from February to April 2024. The survey instrument has also been provided as a supplementary file (online supplemental file 1—Survey).

Reflecting on the results of the surveys, we developed items for the interview guide with the goal of investigating topics in greater detail, contextualising them and ascertaining perceptions of causality in these topics. For instance, questions examined issues of others’ and one’s own doubt of pain complaints, strategies for communicating private symptoms, the impact of self-doubt on healthcare-seeking behaviours and means of validation. The interview guide was reviewed by researchers and clinicians who both specialise in EDS and those who do not, and revisions were made to it on those bases. The interview guide has also been provided as a supplementary file (online supplemental file 2—Interview Guide). One author (CMEH) conducted all interviews between June and October 2024 over either Zoom or telephone, based on participant preference. Interviews were audio-recorded, and field notes were taken. Verbatim transcriptions were produced using the AI-based application Whisper, implemented on a local, password-protected server that did not transmit recordings or transcripts to external servers, and all associated data were deleted from it immediately after processing, in a manner compliant with the US Health Insurance Portability and Accountability Act (HIPAA) and the EU General Data Protection Regulation (GDPR).³⁸

Data analysis

Mixed methods were used to analyse the survey and interview data. Likert and demographic items were analysed using descriptive statistics, including the calculation of mean and mode. Free-text survey items and interviews were analysed through reflexive thematic analysis³⁹,⁴¹ in order to identify how participants experience similar situations differently. Following the iterative, recursive phases of this method, team members began by familiarising themselves with transcripts in the coding platform Dedoose (http://www.dedoose.com). Transcripts were read thoroughly and discussed in multiple initial analysis meetings. In subsequent meetings, researchers developed brief codes to label classes of information relevant to the research question of self-doubt, with inclusion and exclusion criteria. After all transcripts were coded by three team members (CMEH, SLV, TD), the aggregated data were interpreted, identifying relationships between codes and patterns of shared meaning, which were constructed into coherent themes providing a ‘thick description’⁴² of the experiences of our participants. Survey and interview data were synthesised during this final analysis using mixed-methods sequential explanatory design, with qualitative results aiding in refining and contextualising quantitative results and providing richer and deeper participant perspectives.⁴³ Themes were then defined and refined and finally were used to create a narrative to fit the data. In the final phase, all members of the team collaborated to write the results.

Results

43 individuals completed the pre-survey (81% response rate), 25 with hEDS and 18 with mdEDS. Subsequently, 39 individuals completed an interview, 21 with hEDS and 18 with mdEDS. One interviewee did not complete a pre-survey. The interviews averaged just over an hour, at 65 min in length (range 36–104 min), for a total of 45 hours of conversation across all participants. The average participant identified as a white female, was in her early 40s and had at least a bachelor’s degree. The demographics of our participants can be found in greater detail in table 1.

Table 1. Demographics.

		Survey (n=43)	Interview (n=39)
Age	Average	42 years	41 years
	Range	21–68 years	21–68 years
Gender	Female	32 (74%)	29 (74%)
	Male	6 (14%)	6 (15%)
	Other	5 (11%)	4 (11%)
Race and ethnicity	White	37 (86%)	33 (85%)
	Asian	3 (7%)	3 (8%)
	Black	0	1 (3%)
	Hispanic	1 (2%)	1 (3%)
	Multiracial	2 (5%)	1 (3%)
Education	High school or less	4 (9%)	3 (8%)
	Some college or associate’s degree	8 (19%)	7 (18%)
	Bachelor’s degree	14 (33%)	13 (33%)
	Graduate degree	17 (40%)	16 (41%)
Years from diagnosis to data collection	Average	10 years
	Range	1–42 years

We use three-digit numerical pseudonyms to protect participants’ confidentiality while allowing analytic distinctions by cohort and age, both of which are relevant to our final analysis of the data. Pseudonyms in the 100-range represent individuals with hEDS, and those in the 200-range represent those with mdEDS. The tens digit indicates the participants’ age in decades, and the final digit has been randomly assigned. For example, the pseudonym 230 would refer to a participant with mdEDS in their 30s.

The construct of self-doubt

73% of our survey respondents (68% of hEDS and 79% of mdEDS participants) said that they doubted or second-guessed whether they were truly in pain at least some of the time. In interviews, we asked those individuals who lacked this experience to expand on their responses, and some mentioned that they doubted the severity but not the reality of their pain. Others stated that they doubted the cause but not the reality, primarily worrying that the pain was psychogenic rather than due to EDS. “I didn't necessarily doubt that I was in pain. I knew that I was in pain. I guess it was more […] there is no concrete reason for me to be experiencing pain every day. […] The alternative is this is all the result of all of my trauma growing up, the CPTSD, the central sensitization” (231). One woman stated that she had come not to notice her pain at all. Finally, only two interviewees stated that they had never had any doubt about any aspect of their pain.

In defining self-doubt, they identified several dimensions of the construct: They primarily saw it as an interference in their decision-making abilities, limiting their confidence in their judgments. But they also identified self-doubt as a failure of their trust in their sensations and perception of their severity. In that vein, one man defined it as “second-guessing your feelings” (242). Similarly, some described a decrease in self-efficacy, doubting their capacity to engage in activities in which they had previously succeeded. For instance, one woman stated that she had developed a “low belief in my abilities […] I question whether I can do something” (144). It was seen as an ungrounded belief that one is physically incapable of accomplishing some task or goal. “Self-doubt is when we tell ourselves we can’t do something, even though we have no evidence” (143).

However, a third, significant group of respondents linked self-doubt to a yet more existential questioning of their “reality” (141) and “human experience” (144), leading them to worry about their psychological well-being

Sources of doubt and self-doubt

Sources of doubt

Participants believed that their self-doubt arose in large part due to the doubt they encountered from others, most prominently, from their clinicians. In particular, they reported neurologists and, to a lesser extent, cardiologists as the most suspicious specialties. They believed that they were doubted for a number of reasons: The chronic nature of their pain meant that they did not respond in an expected way, leading clinicians to doubt the severity of their pain: “You wouldn’t be sitting here talking to me in this way if you were in that amount of pain,” one woman stated, voicing the disbelief expressed by her physician (143).

Participants also identified biases, such as misogyny and ageism, as leading to clinicians’ doubt. For instance, they stated that children are often treated as “overdramatic” (238), and “whiny” (121), as though they were malingering and simply trying to avoid school or chores. They stressed that encountering doubt during the psychosocially formative period of childhood and their teenage years in particular led to an enduring lack of self-confidence and self-trust. “I feel like it’s something that was sort of like a button that was installed in childhood, and doctors push it and then I’m sort of triggered into self-doubt” (263).

While acknowledging the harms caused by clinicians’ doubt, the majority of participants (79% overall, 68% of hEDS and 93% of mdEDS) asserted that it was acceptable for medical experts to doubt their patients, at least under certain circumstances. “Doubt’s an integral part of [their] job,” said one interviewee (122). Many participants even found uncritically supportive clinicians as potentially “detrimental” (141). They contrasted the unintentional harm caused by clinicians’ doubt—the epistemic injustice and clinician-associated traumatisation it caused—with ‘medical gaslighting’, which they generally defined as the intentional and manipulative invalidation of a patient’s perceptions.

However, participants stressed that doubt should not be a clinician’s first response. Moreover, our participants stated that clinicians should not doubt personal experiences—such as pain, fatigue or personal biography. How doubt is communicated was stressed as critical, with interviewees underscoring that clinicians should do so “delicately” (134) while keeping in mind that “in the majority of cases, [patients] just want to get better” (237).

To be sure, clinicians did not make up the sole class of person who doubted our participants’ descriptions of their experiences. Many mentioned family–parents, siblings, spouses, etc.–expressing consistent scepticism about the reality and severity of their pain. One woman said her husband was so dismissive of her not feeling well that they eventually divorced. Her ex-husband’s attitude, she said, had made her wonder: “Is it all in my head? That’s self-doubt” (262).

Sources of self-doubt

A principal cause of self-doubt for our participants consisted of the doubt that they had experienced from clinicians. Such doubt was particularly impactful because of clinicians’ socially esteemed position as experts. “That’s where the self-doubt comes in, that I am not a doctor. I haven’t been to medical school. […] I don’t necessarily have the big picture” (263). A clinician “looks at you and is like, ‘Hey, you’re actually a liar,” one participant said, explaining the feeling of being doubted, “you kind of are like, ‘Well, I’m supposed to be, like, lesser than them. I don’t understand as much as they do, so maybe they’re right. And so, the more doctors keep saying that to you, the more you start to believe it” (121). Some interviewees also recalled how their parents preferred to endorse a clinician’s assessment of their child’s symptoms, rather than validate the self-assessments offered by their child. Such experiences had been traumatic for these interviewees and were seen as an enduring alignment of previously trusted loved ones against their children and their children’s suffering. One interviewee with mdEDS remembered the impact of her clinician’s doubt in this way: “My family just dismissed everything that I said from that point on, because, you know, what does a child know?” (241).

Some clinicians reportedly badgered participants until they stopped resisting and deflated their testimony so that it matched these experts’ expectations and interpretations. One woman described such an interaction with a neurologist: “She was questioning everything I was saying, and I couldn’t trust what I was saying because– I’m sorry; I’m getting upset,” she cried quietly as she recounted the story in our interview. “[She was] making me feel like what I was saying, it was either wrong or not helpful. I don’t know, but it felt like nothing I was saying was the right thing” (245). This can be cumulative as well, as another woman described:

“I’ve had so many dismissal responses that you think, ‘Is it me? Am I making this up? Am I being too into this?’ You do doubt yourself once you’ve had enough responses telling you […] this is so rare, it doesn’t exist. And you think, ‘Do I exist? Is this really me?’ […] You should be able to trust them, but they aren’t giving you the response that you’re expecting, and you do doubt yourself then” (241).

Or as another interviewee described his experience: “You have been questioned so many times, doubted by other people, [that] you inherit that. You take that onto yourself. And then it becomes your first gut reaction to things like that. I think it is very hard to stay true to your belief if the entire world around you just constantly is at odds with the belief you hold” (221).

Clinicians’ lack of or limited response to patients’ subjective complaints also led to feelings of self-doubt. They often failed to react with the urgency that patients expected, and this blasé attitude prompted a sense that “my pain didn’t matter or else somebody else would be doing something” to treat or manage it (133). Another participant reported a similar feeling of self-doubt as the result of being sent home with “low-level pain medication” after an ocular injury (234). At first blush, the clinicians’ dismissal of this interviewee’s pain left him confused, but soon he began to doubt himself: “I guess they’re right;” his pain, he came to believe, really was not as severe as it had felt (234).

Given the long diagnostic odyssey our participants underwent, the lack of an identified, unifying cause for their symptoms was another source of self-doubt. “Over time, I guess it kind of ate away at me, after not having answers over and over and over and over again. I was like, ‘Well, maybe [my clinician] was right,’” namely, that her symptoms were not “real” after all (144). For participants with hEDS, the lack of available molecular diagnosis amplified their doubt in their private experiences. In certain instances, witnessing others with more severe pain and greater disability also compounded their self-doubt. “It’s easy to compare yourself to other people online whose conditions might be worse than yours and be like, ‘Oh, maybe mine’s not real’” (121).

Responses to self-doubt

Damage to self-worth

The growing disconnect between subjective experience and objective validation was also “very scary” and brought on a “sense of doom” (154). Self-doubt led participants to internalise their assessment of others’ opinions of them: “Doctors make you feel like you don’t deserve care, […] like you’re wasting resources for people who need it. That’s what they’re accusing you of. You kind of hate yourself, because you’re like, maybe I don’t deserve this [healthcare]. Maybe I really do suck” (121). Their self-doubt frequently led our participants to worry, “Did I take up a bed space in a hospital? Did I waste people’s time?” (233). In short, it caused them to question their self-worth and their deservingness of care.

Failure of trust in the self-perception of pain

Self-doubt primarily expressed itself as a scepticism about the severity or even reality of an individual’s pain experience. This was felt as an acute epistemic disorientation. Some participants came to feel that “maybe I am faking it” (121), or that they were secretly malingering. They began to worry that their pain was actually an unconscious manifestation of an unacknowledged urge to seek attention (262) or a mode of unintentional manipulation in some pathological pursuit of sympathy (221). “I’ll put it in my head that […] really, I’m not feeling like this, and maybe I am just looking for excuses and could really be doing all the stuff that other people could be doing,” one woman explained (134).

The high tolerance for discomfort that our participants developed in response to their chronic pain further encumbered their confidence in being able to assess their private states: “You kind of tolerate it. You get through your day, you do things, you start to question yourself, like, ‘Am I really in pain?’” (141). Even the successful relief of symptoms through treatment and therapy triggered self-doubt for some participants. For instance, one participant recounted visiting an osteopath with her mother:

He did some manual work on me […] and he was very good at his job. I walked out of the office, and I was like, “I don’t really feel any pain in my shoulders. What is happening? I was kind of quiet and not really saying anything. Then at the beginning of the drive home, I turned to my mom, like, “Mom, I don’t know if I have EDS.” And she’s like, “Well, it’s a genetic condition, so even if you feel good because of the treatment, you still have it.” “But I’m not in pain!” (123)

In extreme cases, our interviewees even described how self-doubt resulted in their experiencing something akin to dissociation: “Am I having a break with reality?” (144). They lost all trust in their ability to assess their private, internal states and subsequently began to doubt their ability to assess reality altogether. “It makes you question: Is anything even real, […] anything I experience as a human being […] any kind of experience that I was having, beyond that medical point even,” one woman explained (237). “That was making me feel so scared,” she continued, worrying that her perceptions were “essentially not true” (237). Several interviewees said that when clinicians erroneously posed a psychological explanation as the (sole) explanation for their chronic pain, they were amenable to this interpretation. “I wasn’t against it; at least it was an answer,” said one interviewee (121). One interviewee with mdEDS even dutifully took anxiolytics at his doctor’s recommendation only to later learn that he had suffered an aneurysm (234). Even after getting a diagnosis, even with that external validation, some participants continued to struggle with trusting themselves again. For instance, one woman stated that despite a clinician’s confirmation that she had vocal cord dysfunction, she persisted in thinking of her airway stridor as “just anxiety” and that she was “just nuts” (132).

Self-doubt transforms the experience of pain severity and reality.

Interviewees questioned the severity or the medical significance of their symptoms. They stated that they regularly underestimated their pain as a result of their self-doubt. “I wondered, does everyone just feel this way, and I’m just the only person expressing it in words? Is this some kind of universal thing? […] I genuinely didn’t know […] Maybe my experiences were all normal” (238).

The majority worried about precision when they attempted to describe their private experiences. One man described incorporating his clinicians’ dismissal into his own quantification of his pain experience. When he initially reported “10 out of 10” postoperative pain, “the doctors were just insistent on this being a non-painful procedure. So that kind of skewed my thoughts, like, ‘Oh, well, I must be wrong because they’re telling me that this isn’t a possibility,” so he reduced his personal assessment of his subjective experience to “a 4 or 5 out of 10” (234). He explained that from then on, he truly believed his pain should be rated at this lower level. It felt less severe and less threatening, at least until another clinician discovered that he was suffering from severe inflammation after an emergent surgery.

But this self-doubt was not just a breakdown in communication. Through self-doubt, some participants actually came to experience pain differently—or not at all. “I have figured out how to push it into the back of my mind, so I don’t even feel it” (263). One participant described her experience of “adaptive dissociat[ion],” by which she meant “compartmentalization in order to communicate effectively and receive sufficient care” (131). These participants worried they could no longer tell “what’s just normal aches and pains, and what’s significant” (161). For patients with vascular EDS (vEDS), in whom pain can indicate life-threatening vascular events, this indeterminacy felt particularly unsafe.

Healthcare avoidance due to self-doubt

Because of the scepticism our participants had encountered in clinic, they were often quite hesitant to seek professional care. Self-doubt amplified as they interrogated their symptoms, and this interrogation eventually led them to doubt whether their symptoms were severe or persistent enough to warrant medical care. They worried that they would simply be wasting the clinician’s time and another patient’s resources. Similarly, some noted that they doubted that their pain—even sometimes extreme pain—was severe enough to warrant medication. Others were not confident enough in their assessment of their internal states to push back against clinicians’ scepticism.

Overcoming self-doubt

Many of our interviewees described ways that they had overcome their self-doubt (table 2). Participants found value in both internal and external supports. Internally, they reflected on objectively verifiable features of their experience, including scars and feats of hypermobility. They also reminded themselves of past experiences of similar symptoms which had proven to have medically significant implications. During stressful clinical encounters, they made use of mindfulness practices to ground themselves, including meditation and breathwork. After the fact, they found journaling and prayer helpful. Participants also reminded themselves that they did not receive secondary gain from “faking it” (121). For instance, when doubt creeps up on one participant while he is alone, he tells himself his pain must be real because there is no one around and “no reason to make it up to seek sympathy” (221).

Table 2. Self-doubt coping strategies.

Strategy	Description
Reality testing	Grounding oneself in “objective things that couldn’t be doubted” (238), including “visual cues” (243), like scars, and behaviours that demonstrated clinical hypermobility, asking oneself whether others would reasonably be in pain under similar circumstances
Mindfulness	Meditation, taking a moment for silence, and focusing on one’s breathing in the moment; after the fact, journaling and prayer
Peer support	Family members and other close relations notice and validate participants’ pain “even before I perceive it” (263); support groups help individuals recognise that “there are so many people out there that are experiencing the same thing. You’re not alone!” (154)
Clinician support	Sympathetic expert clinicians recognise and reassure of the reality of symptoms by providing medical ‘proof’ such as clinical confirmation and genetic testing; mental health support and talk therapy were also highlighted
Acknowledging lack of secondary gain	Reminding oneself that there is no benefit to suffering from pain

Expert supports were also identified. These included family and other loved ones, especially those who lived with our participants and had the opportunity to see their symptoms on a daily basis. These individuals were taken as particularly credible and persuasive and could notice and validate patients’ pain “even before I perceive it” (263). Support groups, in particular online communities, were also noted as sources of reassurance. Finally, expert and sympathetic clinicians, while mentioned less often, could recognise and reassure participants of the reality of their symptoms by providing medical ‘proof’ of their condition: genetic results that were a “black and white answer” (233) and descriptions of the syndrome that “made complete sense” (144). Professional mental health support and talk therapy were highlighted as helpful for similar reasons.

No second chances for doubters

The majority of survey respondents (73%) said that once their trust in a clinician was lost, it could not be regained. “Hell no!” said one participant (121), “If I don’t trust a clinician, that’s forever.” However, the minority of participants stated that a clinician had won back their trust. Admitting their mistakes and taking the time to listen and respond to their patients’ concerns helped clinicians regain their patients’ trust.

Discussion

Frequent scepticism and invalidation lead to high incidence of self-doubt

People living with EDS often encounter doubt about the severity and reality of their chronic pain and other symptoms from family, society and—most poignantly—from their clinicians. The majority of our research participants internalised that doubt: 73% overall; 68% of those with hEDS and 79% of those with mdEDS. We find it surprising that despite the availability of genetic testing for participants with mdEDS, such objective measures did not decrease their experience of doubt. Invalidation negatively impacted all participants’ sense of self-worth, trust in their subjective experiences and health-seeking behaviours. They characterised it as a harm with enduring psychological and medical consequences, shaping how they communicate and understand their own bodies and symptoms. Self-doubt is thus not merely a form of epistemic injustice,²³ as it both disenfranchises patients in their capacity as knowers as well as erodes their trust in themselves at a fundamental level, upending confidence in their ability to assess or appraise their own sensory perceptions.

The EDS community is not alone in its experience of self-doubt. Though understudied, this harmful experience has been identified in patients with other chronic pain and rare disorders. For instance, it appears prominently among patients with postural orthostatic tachycardia syndrome,⁴⁴ a common comorbidity of EDS.²⁷ In studies with patients suffering from chronic lower back pain,⁴⁵ long COVID,⁴⁶ lupus,²⁵ pemphigus⁴⁷ and rare disease more generally,⁴⁸ self-doubt has also been identified as a common experience. Our study offers a substantive and specific engagement with this phenomenon among people living with EDS, but our findings likely have broader relevance to populations with chronic pain and invisible illnesses, and they may be generalisable to related disorders already associated with high levels of self-doubt.

While participants reported their symptoms’ being doubted by a wide variety of other individuals, including family members, coworkers and peers, the primary focus of their frustration and anxiety was clinicians’ scepticism. Over the course of their long diagnostic odysseys—regularly lasting over a decade^{30 49 50}—our participants repeatedly encountered clinicians who used their medical expertise and authority to discount patients’ subjective symptoms as ambiguous, exaggerated and unreal. This dismissal is characteristic of these patients’ experiences.^{3033 49},⁵² In illuminating how such doubt transmutes itself into self-doubt, our study extends existing accounts of testimonial injustice.

Many interviewees acknowledged that chronic pain does not present in the same way as acute pain, the type of pain they felt clinicians uncritically assumed they were experiencing. Our participants described developing a high tolerance for discomfort, paradoxically making them appear as less credible historians. The invisible nature of their symptoms led others to view them as exaggerating and even fabricating the severity and reality of their pain, as has been found in other studies as well.^{349 52},⁵⁴ Our participants subsequently internalised these perceptions.

Importantly, our participants rarely interpreted clinicians’ doubt as deliberate or malicious. In this way, they distinguished it from ‘medical gaslighting’, a popular term that they reserved for interpersonal dynamics marked by manipulation and ill intent. In fact, the majority (79%) stated that doubting patients was acceptable—under specific circumstances—as it is integral to the enactment of medical expertise.

Nonetheless, participants frequently described clinicians’ doubt as the consequence of personal and institutional biases, including misogyny, ageism and ableism. Other researchers have suggested that women with chronic pain disproportionately encounter disbelief.⁵⁵,⁵⁸ In our study, several individuals additionally noted that they found female clinicians to be more understanding and trustworthy than their male colleagues. However, we were struck by the presence of such experiences for participants of all genders. Moreover, ageism appeared a more prominent concern among our cohort. Symptoms often first become unmanageable for patients with EDS in their teenage years,⁵⁹ but because of their age, many are dismissed as malingering or simply shirking school and other social obligations. Again, we found that scepticism based on these biases was internalised, and our participants noted that self-doubt arising at such formative periods in their psychosocial development was particularly acute and enduring. In fact, children regularly face testimonial injustice and are stereotyped as irrational when they express health concerns.⁶⁰

In addition to the sheer frequency with which our participants encountered doubt, many described badgering interactions, in which clinicians insisted on explanations of their symptoms that did not align with our participants’ understanding of those experiences. They regularly noted the epistemic asymmetry between themselves and medical experts, stating that they did not feel that they had the authority—or that they would be perceived as having the authority—to contradict expert explanations. They fell silent and even adopted these self-denying views, including psychogenic explanations for their symptoms. They mirrored their clinicians’ scepticism and downgraded the severity of their pain both when reporting it to others and when reflecting on it in private. Some participants began to perceive their pain as muted or nonexistent.

Self-doubt interferes with patients’ willingness and ability to assess their subjective states.

The blunting of pain in this way is clinically worrisome. In particular, for patients with vEDS, urgent conditions such as spontaneous vascular rupture may go unidentified and unmanaged. Another study reported a woman with hEDS who did not recognise the pain associated with her ectopic pregnancy.⁵³ The inability to identify—and therefore treat—the sources of these invisible pains is a critical medical concern.

Our participants’ loss of trust in their ability to communicate and even perceive their symptoms accurately reached an apex in the form of worries that they were unconsciously malingering or seeking secondary gain. Some had experiences akin to dissociation. Many interviewees used the vernacular idiom of ‘feeling crazy’ to name the uncanny disparity between their perceptions and those of their clinicians. However, for others, the worry was not metaphorical. They feared they were actually mentally ill and that their symptoms were psychogenic or imagined rather than physical. Even in participants who did not report doubting the reality of their pain, it was found that many harboured doubts about its severity or presumed that certain aspects of their pain experiences were psychogenic. Significantly, only two of our participants expressed never having doubts about any aspect of their pain experiences. It is noteworthy that at least one study has found a correlation between self-doubt and both anxiety and depression,⁶¹ two mental health conditions that have significantly higher incidence among patients with EDS than in the general population.^{27 62} Our participants’ existentially destabilising experiences additionally mirrored the dissociative responses of trauma survivors⁶³ and may be related to the high prevalence of clinician-associated traumatisation documented in the hEDS population.³³

This deep mistrust in one’s own subjective experience was profoundly emotional for our participants. They expressed a ‘sense of doom’ and felt undeserving of care. One participant devastatingly described her internal dialogue: “Maybe I really do suck.” Self-doubt led to an existential fear for some participants. Poignantly, a study of patients with lupus included the theme of self-doubt caused by physicians, and it stated that this was “sometimes felt to be more damaging than the disease” itself.²⁵

The consequences were more than simply cognitive and emotional, though. Our participants reported that self-doubt decreased their willingness to seek healthcare. They convinced themselves that their symptoms were not severe enough and delayed going to the hospital until their symptoms were unmanageable. They felt they were undeserving of care. While these comments generally linked such avoidance to self-doubt, others mentioned a general fear of being doubted. While this specific disincentive has been reported previously among patients with EDS,³⁰ as well as among patients with lupus,²⁵ the expression of entirely internalised doubt as a barrier appears to be a novel finding of our study. Patients experiencing anxieties around seeking care may benefit from self-advocacy and empowerment workshops offered virtually by EDS ECHO,⁶⁴ and Annabelle’s Challenge also offers resources to help patients with these and similar issues.

Participants developed strategies to manage and reduce self-doubt.

While many participants continued to experience self-doubt at the time of our interviews, most had developed strategies to identify and combat unreasonable scepticism about their subjective states and experiences. They identified techniques for reality testing and grounding themselves when they feel dislodged by self-doubt: They assessed objective cues, like the formation of scars that would not be expected in healthy individuals.

Our participants also engaged in reflective practices—including mindfulness, journaling and prayer—in order to calm and centre themselves when they felt badgered and epistemically unmoored. This strategy has also been successful for patients living with pemphigus.^{47 65} They told themselves that they did not expect secondary gains from attending to their symptoms: EDS, they reminded themselves, is “expensive. It’s not fun. It wrecks your life.” Older patients, who had accumulated more memories of previous interactions in which they had been proven correct despite the doubt of their clinicians, noted that for this reason age naturally reduced self-doubt.

Peer validation also helped our participants to rebuild their self-confidence. Loved ones and close friends could recognise the signs of their distress even when our participants could not or refused to see them for themselves. Existing trust, however, was identified as a critical component of this validation. Strangers and distrusted individuals were not given the epistemic authority to sway our participants’ beliefs. Similarly, witnessing the experiences of others—both those with EDS and those who were perceived as otherwise healthy—in order to judge their experiences as ‘normal’ or ‘abnormal’, providing external grounding. Other studies have suggested that social media groups may play a critical role in validating the experiences of patients with EDS and other rare diseases.⁶⁶,⁶⁸ Low-level and cautious re-entry into exercise and other physical activity has additionally been identified in other chronic pain populations as a means of overcoming self-doubt and promoting self-management skills⁴⁵ and may be appropriate for patients with EDS given that our participants expressed self-doubt related to their physical capacities, though they did not identify this coping strategy explicitly.

Finally, firing sceptical and badgering clinicians was another way our participants asserted the reality of their symptoms over and against other, self-denying perspectives. Such acts were often felt to be empowering and necessary and represented another strategy to avoid and overcome triggers of self-doubt. However, it is important to recognise that this breakdown in patient–provider trust has negative implications for continuity of care and long-term outcomes related to the management of pain and other symptoms.

Limitations

Our participants were predominantly white and well educated, which is representative of what is known of the overall demographics of patients who manage to receive EDS diagnoses.⁶⁹ However, these factors may limit the generalisability of our findings. Additionally, patients reported their diagnostic status and this was not assessed in any other way. Future research should examine how the intersectional role of minority statuses, including racialisation and poverty, impacts the experiences of self-doubt and strategies developed to mitigate such harms.

In order to achieve our recruitment goal for mdEDS participants, we drew from international societies and conducted interviews with individuals in both North America and Europe, encompassing a diverse range of distinct healthcare delivery systems. Additionally, it is important to note that, especially in Europe and Canada, obtaining appropriate and holistic care for EDS often requires patients to travel across national boundaries, thereby interacting with multiple healthcare systems in practice. While this limits our ability to link self-doubt to particular infrastructural features of any one national model, it suggests that the challenges described by our participants may be more general and more broadly shared across contexts.

Conclusion and future directions

Self-doubt is a prominent issue facing patients with EDS and entails salient epistemic, moral and clinical concerns. It should be researched in other often-neglected illnesses, rare diseases and connective tissue disorders to determine the generalisability of our findings. Psychological studies could validate the effectiveness of the techniques our participants identified for overcoming the negative consequences of self-doubt. Given the role that clinicians plays in causing patient self-doubt, data are needed regarding the causes and motivations for clinicians’ doubt. Relatedly, it is important to develop strategies for clinicians to express and navigate doubt in a manner that does not result in epistemic or existential harm to their patients. Ideally, future empirical studies would assess and analyse real-time clinical conversations occurring between patients and their clinicians in order to identify specific phrases or patterns of speech that either contribute to or help alleviate patient self-doubt.

While expert scepticism may be necessary for evidence-based medicine, it must be carefully communicated. Doubt need not be delegitimising. Our participants largely believed harmful expressions of doubt were unintentional and suggested that they arose from institutional barriers and unconscious bias. This suggests that it will be necessary ultimately to examine and correct underlying systems that perpetuate these unjust social and political-economic structures, including limited visit times, barriers to accessing specialist care and insufficient medical education on rare and invisible diseases. In the meantime, however, clinicians can mitigate their role in promoting patients’ self-doubt by practising epistemic humility: They should seek to acknowledge to themselves and their patients the limits of their own expertise and capacity while also respecting the self-knowledge of their patients’ own subjective experiences and private states.

Data availability statement

Data are available upon reasonable request.