Sections of this chapter have been reproduced from Strong et al.30. This is an Open Access article distributed in accordance with the terms of the Creative Commons Attribution (CC BY 4.0) licence, which permits others to distribute, remix, adapt and build upon this work, for commercial use, provided the original work is properly cited. See: https://creativecommons.org/licenses/by/4.0/. The text below includes minor additions and formatting changes to the original text.
Methods
Design
A multiple-case design was employed to compare more than one data type between and within more than one person.59 The case study was nested in the observational cohort, with two embedded units of analysis at baseline and 6 months: qualitative longitudinal semistructured interviews and quantitative cohort data.
Participants
Sampling was purposive and sought to recruit PITSTOP cohort participants with symptomatic PSD referred for elective surgical treatment. This sampling method aimed for maximum variation based on the following: Wysocki classification (an indicator of disease severity), surgical procedure (excision and closure techniques) and NHS Foundation Trust. Initial contact was made by telephone. All potential participants were e-mailed the participant information sheet and provided verbal consent prior to participation in the interview.
Data collection
At baseline, participants completed a patient-reported experience measure (PREM) of SDM. SDM is scored 0 to 9 (0 indicating poor SDM; 9 indicating good SDM).60 At 6 months, participants completed a PREM of DR. Using a 5-point scale, healthcare DR is scored 0 (low DR) to 100 (high DR).61 In addition, the following outcome measures were collected: pain, length of time to healing and post-surgery complications. Semistructured telephone interviews were conducted between June 2019 and September 2020. A minimum of 20 interviews was considered adequate to understand common perceptions and experiences of treatment choices, thereby achieving data saturation.62,63 A topic guide was designed using the coping in deliberation (CODE) and Sekhon’s Acceptability framework.64,65 Baseline interviews adapted key ‘choice’ (e.g. ‘did you let the surgeon choose your treatment?’) and ‘options’ (e.g. ‘did the surgeon talk you through the risks and benefits?’) questions from the CODE framework.64 At 6 months, the interview guide asked CODE questions related to decision ‘consolidation’ (e.g. ‘was this the right decision?’). Throughout, probing questions covered dimensions of Sekhon’s acceptability framework,65 as well as intervention attributes, to inform the discrete choice experiment (DCE). The interviews were recorded using an encrypted digital recorder and transcribed verbatim.
Patient and public involvement
One patient representative participated in a pilot interview. The aim of the pilot interview was to assess the apprehension of the topic guide questions and review interview cohesion. No amendments were made to the topic guide.
Data analysis
Transcripts were analysed using the National Centre for Social Research ‘Framework’ analysis approach. This approach was chosen because it allows for coding of a priori and de novo themes.66 After familiarising ourselves with the data set, we independently coded a sample of transcripts using the CODE and Acceptability frameworks64,65 on NVivo (QSR International, Warrington, UK) version 11 before conferring. During the analysis and interpretation, integration of qualitative and quantitative data occurred to understand: (1) how disease characteristics and surgeon preferences interacted with patient values in treatment choices; and (2) how participants appraised treatments given particular outcomes. We used joint display tables to look for convergences and divergences between cohort data (disease features/treatment choices/outcomes) with experiences, views and values.67,68 Patient experts were invited to provide feedback on a lay summary of triangulated results to assess acceptability.
Results
Sample
An initial expression of interest was made by 266 cohort participants. The final sample comprised 20 patients (median age 28; range 20–64) from 13 NHS Foundation Trusts in the UK (Table 14). Only 13 participants completed baseline (median 16 minutes, range 6–47 minutes) and follow-up interviews (median 18 minutes, range 11–37 minutes).
| ID | Male/female | Age | Baseline | Surgery | 6 month | Follow-up interview? | |||
|---|---|---|---|---|---|---|---|---|---|
| Prior operations | Severity | SDM | Excision/closure | DR a | Recurrenceb | ||||
| Complete data set | |||||||||
| 1 | Male | 31 | 0 | 1 | 2.6 | LE/MC | 10 | No | Y |
| 3 | Male | 29 | 1 | 1 | 4 | PP/LO | 10 | Yes | Y |
| 5 | Male | 28 | 0 | 1 | 3 | LE/LC | 5 | No | Y |
| 6 | Female | 20 | 3 | 1 | 4 | LE/LO | 30 | No | Y |
| 8 | Male | 27 | 2 | 4 | 3 | LE/LO | 20 | No | Y |
| 9 | Male | 25 | 0 | 1 | 4 | LE/LC | 5 | No | Y |
| 10 | Male | 64 | 0 | 3 | 3.6 | Se only | 5 | No | Y |
| 11 | Male | 27 | 1 | 4 | 3 | Cu/LO | 5 | No | Y |
| 14 | Male | 23 | 0 | 2 | 2 | EPSiT/LO | 15 | No | Y |
| 16 | Female | 26 | 2 | 4 | 4 | LE/LC(K) | 0 | No | Y |
| 17 | Male | 22 | 2 | 3 | 4 | LE/LC(K) | 0 | No | Y |
| 18 | Female | 27 | 0 | 1 | 4 | PP/LO | 50 | No | Y |
| 19 | Male | 40 | 0 | 4 | 2.66 | LE/MC,M,LC | 0 | No | Y |
| Incomplete data set | |||||||||
| 2 | Female | 31 | 2 | 4 | 4 | LE/LC(K) | LTFU | LTFU | Refused |
| 7 | Female | 33 | 1 | 2 | 4 | Cu,PP/FG | 0 | No | LTFU |
| 12 | Male | 49 | 0 | 1 | 4 | Cu/FG | LTFU | LTFU | LTFU |
| 13 | Male | 28 | 0 | 1 | 4 | Cu/FG | LTFU | LTFU | LTFU |
| 15 | Male | 44 | 0 | 2 | 2.33 | LE/Se,Fl | LTFU | LTFU | Refused |
| 20 | Male | 34 | 0 | 4 | 4 | LE/Fl | LTFU | Yes | LTFU |
| 21 | Female | 25 | 1 | 2 | 2 | LE/MC | 0 | No | LTFU |
Coping in deliberation framework: health threat
Newly diagnosed participants were unfamiliar with, and expressed confusion about, the cause and prognosis of PSD. They detailed their experience of soreness, inflammation, discharge and odour which disrupted employment, physical activity and relationships. Participants discussed the impact this had on psychosocial well-being.
I was told initially, 'Oh that could be it, and then it might go away' … but once you get it once, that’s it: it’s coming back … If I was a bit more aware of that I would have probably started to look into the surgeries quicker.
Some participants were reluctant to address their condition, deciding to tolerate distress and delay treatment. Often, an exacerbation of symptoms would cause participants to present to emergency services for treatment.
I said to [my girlfriend], 'Look, I can’t really see it properly. Is it still getting bigger?' And, she said, 'Oh bloody hell … get in the car.' So, we went straight to [hospital].
Other participants discussed barriers to secondary care treatment referrals due to their GP not taking their condition seriously.
… just gave me some antibiotics … it just kept getting more painful and worse … I went back three times … then she put me on sort of the path to go back to surgery, but she didn’t send me [as] an urgent patient … so I had to wait for maybe like five months.
Another saw their GP numerous times over 25 years and was repeatedly dissuaded from surgery.
he basically sort of said to me that it’s a very precarious operation … that the success rate wasn’t very high … that it was something that if I could live with.
Coping in deliberation framework: choice
Once referred to secondary care, 9/20 participants were offered a choice of treatment. Absence of choice was rarely viewed negatively. Due to their own limited knowledge of the condition, some participants viewed healthcare professionals as best placed to make decisions about their care, especially where emergency surgery was concerned. If offered a choice, participant preference was based on one or more of the following factors: previous experience of surgery (n = 3); surgeon’s guidance (n = 3); invasiveness of the treatment (n = 3); or anticipated recovery time (n = 2). One participant rejected their consultant’s treatment advice to undergo a ‘leave open’ procedure due to the employment opportunity costs – they would require additional time off work (Table 15). Other participants utilised social networks (significant others, friends and relatives) or the internet to support decision-making, with some acquiring a sense of control from independently researching the condition and treatment options.
| Participant information | Decision-making | |||
|---|---|---|---|---|
| ID | Number of prior procedures | Key outcome | CollaboRATE score | Sample quote (coding) |
| 21 | 1 | Recurrence | 2 | ‘[The surgeon] said they’d cut like a flap out, get everything out and sort of stitch it back up … that was the only option … that or managing with medication … I was like, yeah, do what you have to do.’ (Presentation of choice) |
| 14 | 0 | ADL | 2 | ‘I only really got a say in it this time … cos it was a new surgery coming though …. They offered me to do the other one if I wanted’ (Presentation of choice) |
| 15 | 0 | Recovery time | 2.33 | ‘If you’re asking me how it felt like, it felt like I didn’t have a choice.’ (Presentation of choice)‘at first I, [the consultant] sort of said, oh you might be back in a … couple of weeks and then when my friend said oh, 12 weeks for this open wound to heal, I thought … I can’t take that long off work. I can’t afford it.’ (Preference construction) |
| 1 | 0 | Recurrence | 2.6 | ‘[The surgeon] said you either don’t have the surgery and hope that it maybe sorts itself out … I took the decision that the chance of the surgery resolving the matter was worth the risk that it might still reoccur … with no other sort of major health issues that seemed like an easy enough choice’ (Presentation of choice) |
| 19 | 0 | Recurrence | 2.66 | ‘No, [the surgeon] did not give me any option. He just said, just, just he only mentioned the surgery. As I say, I wasn’t given any other options’ (Presentation of choice) |
| 5 | 0 | Pain | 3 | ‘[The doctor] said that they’ll operate and that was pretty much it … just leave it, or you could have the operation and I thought, well, best to try and get it sorted before it keeps getting infected, and gets worse’ (Presentation and interpretation of options) |
| 8 | 2 | Smell | 3 | ‘[The surgeon] give me options of what I wanted and I just wanted one, like obviously cos I had it packed last time, it healed better that way, so I asked for it that way.’ (Preference construction) |
| 11 | 1 | Pain and ADL | 3 | ‘[The consultant] explained to me that you know, we could try medication first and then if that doesn’t work, we could try surgery … it was a scraping out I think … that was something [the consultant] recommended’ (Presentation of choice) |
| 10 | 0 | (Not specified) | 3.6 | ‘I didn’t decide any treatment. The treatment was decided for me by the consultant … I’m not medically qualified, you know … I’m told what the problem is and how it can be rectified. We go along with that.’ (Decision) |
| 2 | 2 | ADL | 4 | ‘No, there was only one procedure left’ (Presentation and interpretation of options) |
| 3 | 1 | Recurrence and pain | 4 | ‘It’s not me fighting this battle … I’m just a battlefield. You guys are fighting it … by the time I got to A&E, they may have given me options, I can’t remember …. I’m quite happy to accept that I don’t know what I’m talking about, so even if I’m given options I will say to the man giving me options, what would you do’ (Presentation and interpretation of options) |
| 6 | 3 | Reducing anxiety of knocking the sinus (reduce symptoms) | 4 | ‘I saw my consultant and he said … depending on the MRI, I’ll give you a few options … one is that we do the same but obviously different in theatre and then the, the other option is to have it, like, lasered, removed’ (Health threat) |
| 7 | 1 | ADL | 4 | ‘They gave me two options but obviously because I have to get a mastectomy in September … I wouldn’t have been healed in time … my immune’s so low as well, we said that the glue one’d be more beneficial for me.’ (Preference construction) |
| 9 | 0 | Recurrence | 4 | ‘The wording was this is the best thing to go for … either don’t have the surgery and hope that it maybe it sorts itself out … or sort of cutting it out … I wasn’t really exploring every single option available’ (Preference construction) |
| 12 | 0 | Recurrence | 4 | ‘[The surgeon] gave me two or three different options that we could take i.e. stitching, gluing, leaving alone etc. and I thought the gluing one sounded the best and of course she agreed that she would like to do the gluing one anyway but she wanted me to make the choice really.’ (Presentation of choice) |
| 13 | 0 | Closing the wound | 4 | ‘It was either an option of having it packed, which the doctor said can take up to a month for it to be fully healed … obviously being self-employed, I need to be back in work … I just plumped for the one that sounded like the one that I thought would work the best and I think it was a newer procedure’ (Presentation of choice/presentation and interpretation of options) |
| 16 | 2 | ADL | 4 | ‘I could leave it and just live with it, which obviously for me wasn’t an option! … my other option was to get a cosmetic surgeon in … So, I was just kind of worried that I would always kind of be left with some sort of wound’ (Health threat) |
| 17 | 2 | Recurrence | 4 | ‘[The nurse] just told me I’d be having emergency surgery … someone looked at me that following morning and decided that I definitely had to have the incision and drainage. They didn’t go through the details of why that was, I’ll be honest … I didn’t know the ins and outs of what I had, and I didn’t know if there was any other options available.’ (Presentation of choice) |
| 18 | 0 | Solve the problem | 4 | ‘I wasn’t given the choice as such of which ones to do but when [the consultant] said that this is what she recommends, I completely took that on board from somebody with her kind of experience and knowledge of it’ (Presentation of choice) |
| 20 | 0 | ADL and recurrence | 4 | ‘[The GP] said you’ve got two options, I either give you some antibiotics and pain relief now or I recommend you go to hospital … I wanted to maintain as much quality of life as possible while listening to the consultant’s guidance’ (Preference construction) |
Coping in deliberation framework: key outcomes at the time of decision-making
Not all participants specified a single important outcome when undergoing surgery. The following were discussed: avoiding recurrence (n = 8), return to normal activities (n = 6) and/or the elimination of symptoms (n = 7). Six participants were not aware of procedural risks; others expressed awareness of risks presented by anaesthesia (n = 2), infection or bleeding (n = 4), the wound not healing (n = 5) and recurrence (n = 8).
Coping in deliberation framework: consolidation
After surgery, most participants were anxious of aggravating the wound and/or delaying wound healing. Many implemented adaptations, including physical (altering seating positions) and behavioural (reducing exercise duration and changing the type of exercise) changes, which negatively impacted their well-being.
It has made me reticent to engage in some activities … exercise and things like that … through the pain and discomfort, and also the chance of sort of popping the cyst...
Many participants required daily or weekly primary care wound management support via GP appointments or district nurse visits. Due to its location, most experienced difficulties managing the wound independently. They used mirrors for physical inspection or were dependent on others to help examine and manage the wound (including cleaning, dressing and packing). Some participants cited the reliance on others as a loss of independence, while others acknowledged how emotional support alleviated distress.
I think the worst part of it is that you always have to rely on someone else to do, like, a dressing for you … you can’t drive cos you can’t sit down … you basically you can’t do anything.
Acceptability framework: key outcomes at 6-month follow-up
Six months post surgery, some participants reported hoping surgery would address: pain (n = 3); recurrence (n = 5); wound healing (n = 1); the smell (n = 1); the inconvenience (n = 1); impaired ability to perform ADL (n = 1). In five cases (01, 05, 11, 14, 17; see Appendix 3, Table 33), these priorities had changed since baseline. During the recovery period, some participants accepted the recurrent nature of the condition. Others managed their own treatment expectations by considering any symptomatic improvement as an indicator of effectiveness.
I’ve still got some kind of stuff going down on there that is just a recurring thing … if I’ve had four operations, it probably won’t get rid of [it].
Some participants specified a single important outcome 6 months after surgery. The following were discussed: the wound healing in the expected time (n = 3); avoidance of recurrence (n = 4); return to ADL (n = 1). Three participants who had undergone PSD surgery for the first time found their treatment to be effective. When asked, they did not feel they would have done anything differently (see Appendix 3, Table 33).
Discussion
This substudy explored how patients make, and sometimes regret, treatment decisions for PSD. Patients are often reluctant to address the condition due to inadequate knowledge and embarrassment. GPs are often hesitant to refer patients to secondary care services as surgical approaches are perceived to be poorly evidenced. Once referred, patients may not be involved in the choice of surgical treatment. Typically, they are unconcerned with and uninformed about the burden post procedure (wound management, practical support, and risk of infection and/or recurrence). Therefore, they may experience unanticipated difficulties when trying to cope with these matters. Patients undergoing their first surgery are often overly optimistic about the effectiveness of treatment. In contrast, those with recurrent disease may experience higher treatment regret and psychosocial distress due to poorly informed decisions. Irrespective of prior experience of PSD, making decisions about surgical treatment is challenging.64 Digesting new and complex treatment information can be difficult. Insufficiently informed, patients are unable to articulate what they would have done differently, with many citing a change in outcome priorities after 6 months. Patients with a previous history of PSD who had undergone an excise-and-leave-open procedure – which is associated with high levels of pain, wound management and extended healing times – demonstrated the highest levels of DR.
This substudy is limited as postsurgical wound healing can take over 6 months and recurrence of PSD may take place over many years. Therefore, the attitudes of participants may be affected due to the short follow-up period. The COVID-19 pandemic significantly impacted our ability to follow up participants.69,70 Although remote data collection was possible, engaging patients in research was difficult as many were experiencing COVID-related barriers.71 In PSD studies, attrition rates are poorly reported and this is thought to be due to the young, mobile, mainly male population.15,72–74 High attrition rates may also result from an unwillingness to disclose negative experiences,75 or a loss of interest in research after the wound has healed and they have returned to work.76
The mixed-methods approach identified divergences and inconsistencies between different data sets – particularly, how patients reflect on their treatment decisions after surgery. Even if they are involved in SDM, if patients are not fully informed about treatment and post-surgery pathways, their expectations may not be met.77 Levels of self-reported DR in this study are in line with the 1 in 7 rate reported across 73 surgical studies, in which regret was mainly associated with type of surgery, health outcomes and absence of SDM.78 Another systematic review has flagged decisional conflict and anxiety as predictive of DR.79 Surgeons80,81 and patients82 may have reasons for avoiding SDM, and our findings complicate the common assumption that SDM leads to increased decisional satisfaction.83 Systematic reviews in other contexts suggest that unmet information needs are common and distressing.84–87 There are growing concerns that self-report measures of SDM may not capture the quality of the interaction or the multistaged nature of the process.88,89 PREMs may be compromised by social desirability or acquiescence bias,90–93 and open-ended questions may reveal significant problems from patients who report high levels of satisfaction on survey instruments.94,95 Triangulation of research methods is useful to identify such problems.96,97
Clinical teams should ensure patients are provided with sufficient information about available surgical procedures. Expectations associated with postprocedural aftercare and the uncertainties surrounding clinical outcomes should be managed. Surgeons may not actively engage in wound care discussions as this is perceived as the responsibility of primary care services.98 However, insufficient information may impact a patient’s ability to self-manage post surgery. Therefore, patients should receive tailored verbal and written information regarding treatment outcomes during relevant consultation appointments.99 Discussing expectations with patients may provide an opportunity to address false optimism.100,101 In other settings, patient expectations predict treatment satisfaction and functional outcomes post surgery.102
Awareness-raising among GPs and surgeons is needed to avoid delays in treatment where PSD is poorly recognised. Where pilonidal surgery is seen as unglamorous,103 or surgeons only specialise in one technique,104 patients with recurrent disease should be referred rapidly onward to relevant specialists. Both SDM and the consent process may be compromised if patients are poorly informed about their condition, available treatments and the probability of various outcomes.80 This is challenging when there are many available treatments supported by variable evidence.14 There are around 20 systematic reviews and meta-analyses on surgical techniques alone, and around 15 more on medical, wound care and other topics. An overview of these reviews should be an urgent research priority to adequately inform SDM and the development of decision support tools. Until then, the review by Stauffer and colleagues remains one of the most comprehensive overviews focusing on time to recurrence with different surgical techniques.13 Finally, discharge planning should begin at pre-assessment visits, involving the patient, day surgery nurses and district nurses.105–107 Postoperative wound care is enhanced by continuity of care from a limited number of community-based health professionals.108
In conclusion, ensuring people with PSD are provided with sufficient information regarding wound care management and risks of recurrence associated with different surgical approaches may facilitate decision-making and minimise treatment regret. An overview of systematic reviews is needed to inform decision support tools. Healthcare professionals should communicate the uncertainties about treatment effects in addition to the time frames, adaptations and psychosocial impact associated with recovery.