Plain English summary

A cryptoglandular anal fistula is a small tunnel connecting the inner surface of the anus or rectum to the overlying skin, which happens as a result of chronic infection and can be difficult to treat. Patients typically suffer from pain and continuous discharge which can greatly impact quality of life. Despite this, few studies have conducted a detailed investigation into what it is like to live with a fistula. We conducted interviews with fistula patients in the UK and Netherlands and found that the effects were broad, influencing work, relationships, daily life and activities and there were certain aspects of medical care that patients found challenging. This has implications for future assessment of fistula, which should expand beyond the focus on physiological and clinical symptoms and should incorporate those outcomes that are important to patients.

Introduction

An anal fistula is an abnormal epithelialised connection between the anal canal and the perianal skin, affecting 1–2 people per 10,000 per year [1]. Most cases occur due to an infection of the mucus glands lining the anal canal, which discharges via the perianal skin. Chronic, ongoing infection of this pathway leads to the formation of the epithelialised fistula tract, as described by the cryptoglandular hypothesis [2].

Most of the research into cryptoglandular anal fistula is concerned with evaluating the physiological and clinical outcomes of surgical treatment [3] including healing and continence. However, anal fistulae have a frequent and profound negative impact on quality of life (QoL) [4]. Patients typically experience perianal pain and ongoing discharge from the external opening, in addition to infective episodes and the development of recurrent abscesses requiring surgical drainage. These symptoms drive a significant QoL burden and require considerable practical adjustments [4,5,6,7]. When asked about the most important QoL issues associated with fistula treatment, patients were more likely to prioritise factors relating to psycho-social wellbeing [8]. In contrast, surgeons and most of the known literature focussed primarily on the impact of physical symptoms [8]. Surgical treatment as been demonstrated to improve QoL [6], even in patients who see a minor deterioration in continence as a result [9, 10].

Despite patient prioritisation of the psycho-social impact of a fistula [8], only a minority of interventional studies measure this as an outcome of treatment [3]. To our knowledge, a detailed assessment of the reality of living with an anal fistula is seldom reported in the literature. Recently, qualitative exploration of patient experience in those with an anal fistula secondary to Crohn’s disease has found that the burden of disease has far-reaching consequences for emotional, physical and social wellbeing [11]. It is unknown whether similar themes can be found in those with a cryptoglandular anal fistula, however, given that the majority of fistulae have a cryptoglandular aetiology, this is an essential area of research.

The aim of this study is to further understand the impact of cryptoglandular anal fistula on patient quality of life through a detailed, qualitative investigation of the patient experience.

Methods

This qualitative study is reported according to the Standards for Reporting Qualitative Research (SRQR) [12]. Appropriate ethical approval was obtained in the UK (REC reference 19/WM/0296) and the Netherlands (METC 2018-0913).

Qualitative approach and research paradigm

We aimed to conduct an exploration of patient experience of anorectal fistula by conducting semi-structured qualitative interviews. A pragmatic constructivist approach was used to investigate the subjective experiences of anal fistula patients, without assuming a single objective reality [13].

Researcher characteristics

Interviews were conducted by clinician–researchers with experience in the assessment of patients with anal fistula. In order to minimise biases in questioning and interpretation, interviewers were not known to, nor had any pre-existing clinical relationship to the participants. Interviewers were trained and supervised by qualitative researchers on the study management team who also oversaw data analysis and interpretation. Furthermore, in order to minimise the influence of interviewer’s clinical backgrounds on the line of questioning and collection of data, interview guides and coding strategies were reviewed by qualitative researchers and patient representatives prior to implementation and analysis.

Context

Patients were recruited from outpatient clinics at tertiary referral centres in the UK and the Netherlands. This provided an opportunity to recruit and interview patients during a period of relative symptom and disease stability, in that they had not been subjected to any recent emergency intervention or acute deterioration of symptoms that may have influenced study results.

Sampling strategy and ethical issues

Participants were recruited by clinician-researchers who later conducted the interviews. Patients aged 18 years and older with a new or previously treated anal fistula not known to have any underlying aetiology were approached in the outpatient clinic and offered written study information, a consent form, and contact details of the researcher so that they could address any queries arising from the participant information sheet. Potential participants were encouraged to take the information away to read fully prior to returning the completed consent form if they wished to participate, however some provided informed consent within the same clinical care episode. Purposive sampling was used to ensure the selection of patients who had just been referred and were therefore earlier into their treatment journey, as well as those with longstanding symptoms, aiming for equal numbers of male and female participants ranging in geographical location. The sample size was determined by the point at which saturation was reached, identified when no new themes emerged from three consecutive interviews.

Data collection methods, instruments and data processing

As patients attending tertiary centres were travelling from geographically distant sites, interviews were conducted via telephone from 1 to 31st December 2019. An interview guide was developed by the study management team and approved by patient representatives (Online Appendix 1). This was designed to cover all aspects of the disease process, from early symptoms and diagnosis, to post-treatment adjustments and desired outcomes. The interview guide was largely used to provide prompts for discussion, with deeper questioning and exploration of the issues raised by participants made by the interviewers where possible. All interviews were audio-recorded on to devices used specifically for study purposes. Prior to starting the recording, interviewers confirmed consent for participation and advised participants to avoid quoting identifiable information during the course of the interview, in order to maintain participant anonymity. All interviews were transcribed verbatim. Dutch transcripts were translated into English using the translation function in Microsoft Word. The accuracy was assessed by bilingual members of the study management team, who reviewed both Dutch and English transcripts as well as the subsequent coding of interviews by referring to the original transcripts. Interview translation was found to be accurate with no apparent loss of data identified between the original and translated manuscripts.

Data analysis

Given the semi-structured nature of the interviews and the aim of obtaining a descriptive overview of patient experience, the framework method was used to conduct thematic analysis of transcribed interviews [14]. Open coding with an inductive approach was used, where data were collected and analysed outside of any pre-existing theoretical framework given the lack of detailed qualitative exploration in this field. Two researchers independently coded the first three interviews, after which members of the study team convened. Coding was compared, and preliminary categories were identified. Subsequent transcripts were then indexed using these codes and categories, which were charted into a matrix using Microsoft Excel. Within and between case analyses were conducted and overall themes were developed from the initial coded categories. Credibility of data analysis was enhanced by having two members of the study management team coding three interviews and comparing coding between reviewers. Member checks of the analysed interviews were not performed due to the time investment this would have required from the participants.

Results

Interviews were conducted with twenty patients (11 male, median age 48.5 Interquartile range 38.5–54.5) (Table 1), all patients were at various stages of treatment. Interview duration ranged from 23 to 72 min. Categories were organised into four broad themes, shown in Fig. 1, with illustrative quotes detailed in Table 2.

Table 1 Participant demographics
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Fig. 1
figure 1

Central themes and sub-themes deduced from patient interviews regarding experiences of living with cryptoglandular anal fistula

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Table 2 Interview themes and sub-themes with illustrative quotes
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Physical symptoms

Pain and discharge

The pain originated from the fistula itself and also as a result of treatment and dressing changes. Pain and discomfort varied in intensity, often described as ‘extreme’ or ‘debilitating’ at their worst. Although a separate symptom, discharge often contributed towards discomfort. Some described a cyclical nature to their symptoms, whereas others commented on the difficulty of managing their unpredictability.

Systemic symptoms

These would sometimes reflect an underlying infective process, manifesting as fever, chills and a ‘flu-like illness’ largely described prior to fistula diagnosis, or in the presence of a recurrent abscess. However, intense fatigue and tiredness were universal to all stages of the disease.

Abscess

Patients described having symptoms of an abscess prior to fistula diagnosis, with some describing recurrent episodes with an established fistula. Patients could recognise the presence of an abscess by increasing intensity of pain and a palpable swelling.

Understanding the condition

Diagnosis

The period prior to diagnosis was often fraught with a lack of recognition of the underlying cause of symptoms, particularly when presenting to primary care. As a result, patients might have had several appointments or surgeries to drain abscesses prior to a definitive diagnosis. Patients described this period as confusing, experiencing relief when the underlying diagnosis was determined and other chronic illnesses were ruled out.

Information from healthcare professionals

The information given to patients was variable both between patients and within their individual experiences. Information regarding the nature of the fistula and treatment strategy was limited in the early stages of the disease, with some patients sensing reluctance from doctors to offer details. Experiences of good communication occurred in the tertiary setting, where patients spoke favourably of not feeling rushed, being given time to ask questions, and being given information specific to their own disease.

Patients felt that information about surgery, aftercare and signs of complications was lacking, and some found that hospitals and primary care gave conflicting advice. Patients particularly felt uninformed immediately after their surgery, where there was a lack of information regarding operative findings until subsequent follow up in the outpatient clinic.

Patients expressed that communication could be improved, particularly in complex cases. It was suggested that written communication and diagrams were helpful, but noted that the three-dimensional nature of the disease was challenging to comprehend.

Information gathering by patients

Patients frequently sought information regarding their diagnosis from online sources, particularly when information from healthcare professionals was conflicting or absent. Some specifically sought out surgical literature to increase understanding of treatment options to inform their decisions, especially when such detail was not divulged by their surgeon.

Practical and emotional support were found in online forums and social media groups. Learning from other patients' experiences and working through the emotional impact and practicalities of daily management meant that these communities were a highly valued resource and helped to fill a clear void.

Chronicity and complexity of condition

Understanding the full extent of disease was an ongoing process. Patients described a point of realisation where they recognised and accepted that there was no ‘quick fix’, and became aware that their recovery could be prolonged. There was frustration and disbelief that the disease could be so complicated and resistant to treatment.

Life impact

Impact on work

Patients required significant time away from work as a result of undergoing investigations, treatment and recovery. This was measured in weeks rather than days and was amplified in patients with a delayed diagnosis or those who required multiple procedures. Aftercare had an additional disruptive impact when time was needed for regular dressing changes.

Patients noted that the direct impact of symptoms required them to make adjustments at work, such as avoiding prolonged periods of sitting, working from home and finding somewhere to change dressings. However, an indirect impact was also noted, due to being mentally pre-occupied with managing symptoms and fatigue affecting the ability to work efficiently.

Some patients found that the personal nature of the disease made it difficult to disclose to colleagues, using reasons such as back pain to explain physical adjustments and would endeavour to continue at work despite feeling unwell. For some, the fistula influenced long-term decisions, such as taking on freelance work, changing jobs and retiring early.

Impact on daily living and activities

One of the significant impacts on daily life was the need for regular dressing changes. Patients reported needing a constant supply of equipment to do this and developed a routine around dressing changes, particularly when managing bowel motions and being away from home.

Daily adjustments were also required to manage fistula symptoms, such as finding a comfortable sitting or sleeping position and being mindful of what colour clothing to wear. Patients felt restricted in the range of activities they could participate in, and actively avoided doing things that would exacerbate their symptoms, such as bike- and horse-riding and swimming. Simple activities such as walking long distances and long car journeys would also be affected, having a more pervasive effect on social activities.

Impact on relationships and intimacy

Often family and friends offered practical and emotional support, but a lack of understanding was noted regarding the chronicity of the condition, and limitations on travel and attending social gatherings meant that friends became distant over time.

Due to the intimate nature of the disease, patients spoke about the impact it had on their relationship with their partners. Patients expressed an increased dependency on their partner to provide assistance in managing the fistula, particularly when adjusting to new symptoms, hygiene demands and dressing changes in a body area that they could not visualise themselves. Patients felt guilty for its impact on their partner, who needed to make their own lifestyle adjustments around surgery and the recovery period.

Patients also spoke of difficulties with intimacy, largely due to a lack of desire and feeling that the fistula itself was a barrier to being intimate. Patients were highly conscious of the presence of fistula discharge, the smell and the need for dressings, which perpetuated feelings of shame and self-consciousness, resulting in little desire for sexual activity. For those who weren’t in relationships, the fistula and the perceived difficulty of explaining it to a potential partner were described as significant barriers to initiating relationships.

Impact of treatment

Although surgical treatment provided hope of relieving symptoms and improving QoL, patients stated that treatment itself had emotional, mental and physical effects. In the immediate recovery, frustration at the prolonged period of inactivity was described, and patients were thankful for minimally invasive methods that resulted in rapid recovery time. Procedures requiring a large wound meant increased frequency and intensity of physical symptoms and further limitation of mobility and activity. Dressing changes were seen as highly inconvenient and the associated pain and discomfort were experienced as psychologically traumatic, particularly when dressing changes were required daily.

Multiple treatments had a cumulative effect on work and also in planning for life events such as holidays. Extra time away from work and special travel arrangements were required for follow up appointments with surgical teams, and travel to and from distant tertiary centres imposed additional time and financial burdens.

There was also mention of the actual impact of surgery on faecal continence. Anxieties were shared regarding the potential impact on long-term continence and whether repeated surgeries were going to result in the need for a stoma bag.

Impact of seton

The impact of a seton was separate to that of other treatments, as its presence required additional adjustments. Some patients reported positive experiences, noting that their symptoms changed from being dominated by pain to ongoing discharge, which some found easier to manage, although this was not universal. A seton may have psychological benefits, particularly when it prevents abscesses. This was especially true for patients who had negative experiences of being unwell with an abscess. Those who had experienced a seton for a prolonged period of time had made necessary adjustments around it, and as a result, had reached a stage of seton acceptance.

In contrast the seton brought its own set of unique symptoms in addition to those from the fistula itself. It was frequently felt to be uncomfortable, and in the most extreme cases resulted in rubbing, chafing and skin blistering and further restriction of mobility. Managing the seton required additional adjustments, particularly when maintaining hygiene after bowel motions. Furthermore, patients had to be conscious of not pulling on the seton and several had experiences of the seton falling out, needing further surgery for replacement. Several patients spoke about the negative psychological impact of a seton, which was seen as unnatural and a clear deviation from normality. Persistent anxiety about keeping it clean and its impact on their intimate relationships was an additional psychological burden. These patients expressed fears of living with a permanent seton, and had difficulty accepting it as long-term treatment.

Psychological impact

The psychological effects of having a fistula were wide reaching, and underlies many of the previous themes. The stress of managing physical symptoms drove a constant state of low-level anxiety. Patients would worry in anticipation of symptoms, particularly those who were fearful about developing an abscess. These patients had heightened anxiety around sudden and unexplained worsening of symptoms, leading to paranoia and frequent checking for any clinical signs of an abscess. The need to make daily adjustments to manage their symptoms required additional mental effort to plan for the day ahead in terms of activities they could tolerate, whether any additional dressings or equipment were needed, and what clothes it was ‘safe’ to wear. Patients described the mental exhaustion this caused, in addition to being a constant reminder that they were unwell.

Patients who had undergone multiple surgeries described the emotional toll this took. Recovery was painful and prolonged, and often represented a period of time in which there was uncertainty regarding the procedure and the eventual outcome. Inconsistencies in aftercare provided an additional stressor. Negative experiences were amplified for patients who later learned that surgery had been ineffective, where the prospect of further surgery and a repeat of these experiences resulted in more anxiety.

Whilst the psychological impact varied at different stages of the disease, feelings of uncertainty about the future, anxiety, frustration, embarrassment and isolation and low mood and depression were consistent in many accounts. Most reported the need for mental resilience to be able to process the psychological impact of an anal fistula, and some patients found professional help in the form of therapy and counselling highly valuable.

Treatment

Surgical decision-making

Patients who had undergone several procedures became accustomed to the fact that no treatment was perfect, and reflected upon the thought processes that guided them to a specific treatment. Many described assessing the risks and benefits of a procedure, where the chance of relieving the worst aspects of their current symptoms were weighed against the worst outcome of treatment, of which incontinence and the need for a stoma were most frequently mentioned. Patients also noted that the weight they gave each of these scenarios changed over time, with some stating that they were more likely to choose low-risk options early on in their diagnosis. After repeated procedures, patients were more likely to consider higher-risk options if it meant a greater chance that their fistula could be cured.

Frustration with the healthcare system

Many patients commented on how the path to diagnosis was prolonged, with many barriers to accessing specialised care. In some instances, referral to secondary care was difficult to obtain. The wait for a specialist consultation was frustrating, particularly when this was followed by the need for more investigations. In some instances, patients cited poor communication and co-ordination between care providers, particularly for secondary and tertiary referrals. This was also demonstrated in patients who suffered from post-operative complications. These were monitored in primary care and only addressed by operating surgeons when the situation became an emergency or at formal follow up occurring much later. In some cases, dismissive attitudes expressed by doctors and surgeons, particularly when addressing concerns over complications, led to a mistrust of healthcare professionals.

Patients also felt that clinical expertise in and support for anal fistula was lacking. Several related the impression that the majority of surgeons were not practiced in the area and couldn’t offer specialised procedures. Patients expressed feelings of abandonment and frustration.

Aftercare

Patients spoke about a lack of comprehensive aftercare when recovering from surgery. They felt poorly prepared for managing their wounds or setons, were reassured that community services would direct their care, but found that even these providers had limited information and experience, and their practice could sometimes conflict with instructions given by the surgeon. The uncertainty of whether the wound was being cared for appropriately added to the psychological distress experienced in the recovery period, and some later reflected on whether inadequate aftercare contributed towards treatment failure.

Goals of treatment

Ideal outcomes from future therapies were mostly related to an absence of physical symptoms and physical reminders of the fistula, such as an external opening. This also included preservation of continence and the avoidance of permanent reminders of disease, such as a seton or a stoma. The importance of restoring QoL and ‘returning to normal’ was also emphasised. This meant restoring mobility and normal activities without considering the impact of the fistula or making adjustments for symptoms, dressings or painkillers. Patients who had experienced multiple surgeries explained how they were more likely to accept that the fistula wouldn’t be fully healed if it meant they could lead some semblance of a normal life.

Discussion

The impact of having a cryptoglandular anal fistula expands far beyond physical symptoms. In research studies, impact on QoL is sometimes acknowledged as an outcome of intervention. Several studies have demonstrated that worsening faecal continence after anal fistula surgery results in deterioration of QoL scores [4, 7], however others have demonstrated that patients treated for simple fistula did not show any worsening of their Faecal Incontinence Quality of Life (FIQL) scores despite showing minor worsening of continence [10]. Data obtained from our interviews demonstrates the complexity of making this risk-benefit assessment, where some patients who experience the worse end of the spectrum of fistula symptoms would take bigger risks in treatment, including impairment of continence, if it meant relief from their fistula. Owen et al. found that fistula patients demonstrated impaired QoL on all domains of the Short Form-36 (SF-36) questionnaire compared to the general population [4] and along with other studies, have shown that patients with recurrent disease had worse QoL compared to those with a primary fistula [4, 15]. However, overall QoL as measured with the Gastrointestinal Quality of Life Index (GIQLI) did not differ between patients with a fistula and age-matched controls [16]. Furthermore, there is suggestion that people with a prolonged disease course exhibit better QoL scores than those with a shorter time with clinical symptoms due to the extensive adaptations made around the disease [17]. Adjustment around symptoms was seen in many of our sub-themes, including in activities of daily living, adjustments made at work and around a seton, with participants essentially describing a ‘new normal’ way of living. In summary, although previous studies have shown variation of QoL and alluded to the broad themes identified in this study [8, 18, 19], to our knowledge this is the first in-depth exploration of what it means to live with a cryptoglandular anal fistula. The qualitative techniques we have employed have allowed us to identify how these themes are inter-related, for example, how physical symptoms require readjustment of daily activities and working patterns, the negative impact this has on psychological wellbeing and the additive effect of these additional stressors on managing an unpredictable illness, providing some explanation as to why other studies sometimes see conflicting outcomes.

A similar qualitative investigation has been conducted in patients living with a Crohn’s anal fistula, finding similar themes [11]. However, there are subtle differences in that the side effects of long-term medication addressing the underlying inflammatory nature of Crohn’s fistulae were a significant concern for patients with Crohn’s Disease, who also cited a loss of independence due to requiring regular infusions in hospital. Apart from this, the negative psycho-social effects were similar in both diseases.

The investigation into patient experience prior to diagnosis and throughout the treatment journey has highlighted important issues regarding clinical management. A common theme throughout all interviews was dissatisfaction with the information, support and aftercare given by healthcare providers. The presentation of anorectal abscesses can be insidious and is not always associated with a fistula. However, patients reported that doctors failed to investigate incomplete wound healing or recurrent abscesses, requiring them to take ownership of their own care to ask for specialist referrals and further investigations. Even within secondary care, patients expressed frustration that the information and treatment options provided seemed limited, and felt that cryptoglandular disease was given less emphasis in healthcare provision. Furthermore, inconsistencies in aftercare and a lack of feedback from surgeons fueled the uncertainty that patients experienced, which contributed to the negative psychological impact of treatment and recovery.

Previous studies have shown how patients and clinicians have different priorities when it comes to the outcome of fistula treatment [8]. Whilst patients reported wanting an improvement in the physiological aspects of the disease, there was also a significant focus on reducing the adjustments required to manage their fistula and the psychological impact of constantly thinking about and planning a routine around their symptoms. This suggests that there is some dissonance between clinician and patient expectations of treatment, as judged by the most frequently measured outcomes in interventional studies [3].

Taken together, these findings suggest that changes are required in clinical practice and future research to improve care for these patients. First, improvement in clinical practice needs to be driven by greater education amongst healthcare professionals within all care settings regarding the diagnosis and management of cryptoglandular anal fistula, to ensure that patients receive a timely diagnosis and are referred for further investigations and treatment appropriately. This should be expanded to include allied healthcare professionals and nursing staff involved in providing wound and post-procedural care for patients, to ensure that best clinical practice is followed and that patients are reassured with continuity of care. Furthermore, an emphasis on providing emotional and psychological support should be made, ensuring a more holistic approach to fistula treatment. Second, if the complexity of the disease is beyond the scope of practice within one centre, referral to a specialist centre, with a wider range of treatment strategies and access to experimental therapies may be appropriate. In addition, patient experience of fistula treatment can be greatly improved by ensuring they receive adequate, easy to understand and complete information regarding the extent of disease and operative management in a timely manner following surgery as well as consistency in wound care advice. With regards to future research directions, greater emphasis on the nature and chronicity of cryptoglandular perianal disease is needed, as well as studies that work towards clarifying optimal management pathways, addressing the heterogeneity of current clinical practice [20, 21]. Finally, the patient experience of disease and desired outcomes, particularly the impact on QoL should receive greater emphasis in the design and methodology of future research studies, ensuring that the outcomes that are most valued by patients are given due consideration.

There are some limitations to this study. All patients were recruited from tertiary referral centres, meaning that disease will naturally be more complex, and one of the issues raised by participants was the prolonged time to diagnosis and treatment, which may be specific to this particular group. As a result, some early or less impactful disease related issues could have been overlooked in our study and may require further, focused investigation. However, we were able to include some participants who had not yet undergone treatment and some symptoms, and therefore the impact on patients, would be similar to those with simple fistulae attending primary or secondary care, regardless of duration of disease. Furthermore, our findings have value in identifying the issues that occur prior to diagnosis and in the immediate post-operative period, which would be common to all patients. The youngest patient in our sample was 30 years old, and so our findings may not be generalisable to the youngest age groups who suffer from this condition, and the impact on areas such as educational attainment could have been overlooked. Finally, we conducted this study across two different countries which may vary in cultural attitudes and access to healthcare services, which could influence patient perceptions of illness and disease. Whilst the aim of this study was to provide a general overview of patient experience rather than to investigate cultural differences, we accept that this would be an interesting area of future research. However, this should be investigated using a dedicated study with questioning focused on this aim and an adequate sample of patients from different cultural groups and countries. Despite this, we have conducted a detailed and extensive exploration of cryptoglandular anal fistula, which has not been reported before.

Conclusion

Cryptoglandular anal fistula has an extensive impact on all aspects of daily living and a consequent effect on psychological and emotional wellbeing. Patients frequently felt that information and treatment options for their condition were limited. The ultimate aim of treatment for many was to be able to live as normal a life as possible. This suggests that future studies of fistula treatment should have a more holistic focus on outcomes and individualised patient goals. Clinicians can address deficiencies in care by providing clinical and practical information, timely referral where there is limited experience of disease and direct patients towards helpful resources such as online support groups and sources of psychological support.