Thematic synthesis of the experiences of people with hidradenitis suppurativa: a systematic review *

Although hidradenitis suppurativa (HS) is known to affect quality of life, little summative knowledge exists on how HS impacts people living with the condition.

What does this study add?
• We show the breadth of physical, psychological and social challenges of living with HS.
• We illustrate the stigma felt by people with HS, how individuals attempt to conceal their condition, and how social support and psychological acceptance can help people cope.
• Early diagnosis, management by multidisciplinary teams, access to social support networks and improved communication with patients about managing HS could lead to improvements in care.
Hidradenitis suppurativa (HS) is a long-term inflammatory skin condition that results in abscesses, usually occurring around the groin, buttocks, breasts and armpits.Prevalence estimates vary from 0Á05% to 4Á1%, and age of onset is typically in young adulthood. 1][4][5][6] HS is often difficult to manage and has limited evidencebased treatment options. 7There is evidence of poor quality of life, psychological impact and disability associated with HS. 8 The James Lind Alliance priority setting partnership for hidradenitis suppurativa identified 'What is the impact of HS and the treatments on people with HS (physical, psychological, financial, social, quality of life)' as a top ten research priority of patients and healthcare professionals. 9This study goes some way to answering this prioritized research question, and highlights gaps in current knowledge.
The aim of this work is to systematically review the qualitative literature reporting the experiences of people living with HS, as well as to: (i) collate what is known about the experience of people living with HS; (ii) collate what is known about the experience of treatment for HS; and (iii) synthesize the data to offer interpretations of the experiences of people with HS.

Literature search strategy
Searches on MEDLINE, PsycINFO, Embase and CINAHL databases were conducted on 17 April 2020.Terms for HS were searched using free text (hidradenitis and acne inversa) and using the relevant subject heading in each database, with no limit on date.The search was not limited in any other way.
An information specialist (D.G.) helped determine the search strategy, full details of which can be found in Table S1 (see Supporting Information).

Inclusion and exclusion criteria
The review included studies conducted globally in any language.All studies that used a qualitative approach to data collection and analysis were included.No article was excluded based on language.Studies had to be relevant to understanding some aspect of experience for people with HS and could include participants with any severity and of any age or sex.Full text needed to be available, which included research letters but not conference abstracts.

Data screening
After duplicates were removed, two reviewers (L.H. and M.M.) independently screened title and abstract of all studies retrieved.Any discrepancies were discussed, and if the discrepancy could not be resolved a third reviewer (N.L.) was consulted.Two reviewers (L.H. and M.M. or N.L.) independently screened full-text articles.Any discrepancies were discussed and a third reviewer (N.L. or M.M.) was consulted when a discrepancy could not be resolved.

Data extraction
A data extraction form was developed and piloted.Information about study characteristics and study quality was extracted.Study quality was assessed by two reviewers (L.H. and M.M. or N.L. or P.L.) independently using the National Institute for Health and Care Excellence quality appraisal checklist for qualitative studies. 10Discrepancies were discussed, and a third reviewer (M.M. or N.L.) was consulted when discrepancies were difficult to resolve.

Evidence synthesis
Thematic synthesis involved an iterative three-stepped approach: (i) coding findings line by line to translate the concepts between studies and begin the process of synthesis; (ii) developing descriptive themes by looking for similarities and differences between codes, and grouping into a hierarchical structure; and (iii) generating analytical themes that interpret findings beyond the present data and answer the review questions. 11ynthesis was performed by L.H. (qualitative methodology and health psychology background).Descriptive themes were secondarily coded by P.L. for five of 14 studies that were purposefully selected to cover a range of topics, study quality and country.There were few discrepancies, but each was discussed and resolved.All authors were included in discussions about the descriptive and analytic themes to ensure interpretation was considered from a variety of perspectives (expertise within the team includes health psychology, sociology, dermatology, nursing, patient experience, applied research and qualitative methodology).

Patient involvement
Patient involvement aimed to aid interpretation of the findings, as well as to assess the acceptability of language and content.K.H-S was asked to read and comment on early versions of analysis and was involved in ongoing discussions and writing.Patient involvement resulted in changing wording that had negative connotations (e.g.replacing 'smell' with 'odour'); it added interpretation to the analysis (e.g.highlighted the language used around the idea of 'acceptance'); and added ideas to include in the discussion (e.g.impact of comorbidities).

Data management
References were stored in an EndNote (Clarivate, Philadelphia, PA, USA) library and uploaded to Rayyan, a web app for systematic reviews (https://rayyan.qcri.org/),for data screening.Studies included in the metasynthesis were imported into NVivo 12 (NVivo software; QSR International, Burlington, MA, USA) for coding.

Search results
The PRISMA flow diagram in Figure 1 shows 8820 records were initially identified in searches.Fourteen studies were considered eligible for inclusion in the metasynthesis.Information on the 14 included studies (referred to as Articles 1-14) is provided in Table 1.Participant demographics are reported in Table 2.Not all included studies contain independent data, and Table 2 illustrates which studies used the same dataset.These studies were all included in the metasynthesis, as it was deemed appropriate to include all available data.
Studies were conducted in the USA, Denmark, Ireland and Israel.All articles were written in English.Qualitative data collection was reported via semistructured interviews in seven studies; via semistructured interviews and focus groups in four studies; via a patient engagement event in one study; by asking a single question in one study; and by reviewing Facebook posts and comments in one study.Of the studies that reported sex, there was a predominance of female over male participation (ratio 3 : 1); of the studies that reported disease severity, there were fewer participants with mild severity; and of the studies that reported age, participant age ranged from 18Á4 to 74 years.

Study quality
Overall, four studies were considered to fulfil most of the quality checklist criteria; eight studies were considered to fulfil some of the quality checklist criteria; and two studies were considered to fulfil few of the checklist criteria. 10Table S2 (see Supporting Information) contains the quality ratings of each article, but notable findings are summarized here.Of the 14 studies included, 12 defined clear aims and/or objectives; 13 reported ethical conduct appropriately; eight used an appropriate research design; nine used reliable methods; 10 used a sufficiently rigorous data analysis process; and 11 provided convincing findings.Lack of reporting in articles resulted in uncertainties in study qualities, notably including being unsure how defensible the methodology was in four studies; unsure how appropriately the data collection was carried out in four studies; how rich the data were in eight studies; and how reliable the analysis was in six studies.Furthermore, only two were judged to describe clearly the role of the researcher; only four were judged to describe clearly the context; and eight did not discuss limitations encountered.

Descriptive themes
Table 3 outlines the descriptive themes collating the data on living with HS, supported by illustrative quotations.Data are presented in two areas: (i) HS symptoms and the impact of HS and (ii) adjustment to life with HS.Table 4 outlines the descriptive theme collating the data on healthcare and treatment experiences.Analytic themes synthesize the descriptive themes and offer interpretations of the data.The themes 'go beyond' the initial data sources by contextualizing the findings, which was informed by published literature and theory. 11

Analytic themes
Theme 1: Putting the brakes on life The symptoms of HS were reported to be, at times, unbearable.While there were many symptoms associated with HS, pain was reported as a central symptom of active disease that can feel both relentless and extreme.
'What happens is the pain increases and then you get to the point where it's the most unbearable thing you could imagine.' (Article 3) The symptoms of HS also affected functioning in multiple ways, making everyday activities a struggle.The symptoms of HS are socially and psychologically challenging.The discharge from the boils can have a strong unpleasant odour, and people reported being keen to avoid embarrassment.They reported avoiding social situations where they feel their HS lesions may be exposed.It was common to read of people retreating to bed and waiting for the flare to subside before they dared to venture out of the house.
'When I have my flare-ups I just like being in the bed.I can't stand being around people.' (Article 4) The coping strategy of hiding, waiting, and avoiding situations because of both the impact from the symptoms themselves, and the perceived negative reactions from others, seemed to have negative psychological consequences.'I have a party tonight.I bought a new dress and have waited so long for this event.But two ugly abscesses have appeared under my chest and armpit, and I can't lift my arm or wear the dress.This disease won again, one-nil HS.I don't want to go.All I want to do is stay home in bed and cry.Instead of putting on makeup and styling my hair, I will be busy bandaging myself.Why do I get this?What did I do wrong?Why me?Why now?I feel no hope with HS.It isolates me from everything.I deal only with it all the time, and I am so tired.' (Article 14) These periods of avoiding situations such as work or social events may have a cumulative effect.The cumulative lifecourse impairment is a theoretical construct referring to the nonreversible burden of a chronic skin disease over time that was originally observed in patients with psoriasis. 12For example, job choices to avoid challenges around functioning can have longer-term career impacts; avoiding first dates can result in not developing relationships or having children; and  Kirby, 28 Thorlacius, 29   Kirby, 30 Sisic, 31 Kirby 32 (USA) Kirby, 28 Thorlacius, 29 Thorlacius, 33   (Danish) Patel 34 Sarfo 35 Kimball 36 Esmann 37 Shukla 38 Senthilnathan 39 Fisher 40

Focus groups
Article ID     While the majority of articles covered the impact of active HS, there were findings in some articles that acknowledged the impact of the damage left even when lesions were no longer active.Theme 2: A stigmatized identity: concealed and revealed People with HS reported an acute awareness of a stigma of HS.Goffman 13 describes a stigma as 'an attribute that is deeply discrediting'; however, he warns that the attribute is not problematic in isolation but within relationships and social contexts. 14n terms of negative impact, people reported going to great lengths to visually conceal their symptoms (both active symptoms and scars) from others, for example by wearing clothing to cover up the HS lesions.During active phases of Disengagement from services 'Many reported feeling angry because they felt that their doctors were being dismissive, which made them want to disengage from medical services.'( Going online 'Many spoke about online forums for people with HS.There were both advantages and disadvantages associated with these forums.The advantages were increased support and access to advice.The disadvantage given was that it would be distressing for people newly diagnosed as it could show how severe the condition could become.' (Article 1) 1, 10, 14 Treatment experiences.People described the experiences of seeking treatment, learning to manage the condition, concerns about treatment and the burdens of treatment.
From seeking a cure to accepting no cure '"Can I get rid of it so that it won't come back at all . ..."' ( Concern about the effectiveness of treatment 'Sadness and worry also occur when patients . . .if the disease is unaffected by the treatment provided.'(Article Self-management burdencaring for wounds, managing weight and smoking '"Instead of putting on makeup and styling my hair, I will be busy bandaging myself."'(Article the condition, they also attempted to conceal the odour.Compared with other skin conditions, HS appears to be relatively concealable.This could suggest HS is a 'concealable stigmatized identity', namely an identity that can be hidden from others, and that is socially devalued and negatively stereotyped. 15owever, it is not entirely concealable, and participants reported worrying that pus would leak and stain clothes, or that others would notice an odour.Therefore, rather than offer freedom from the effects of stigma, the concealable nature of the lesions resulted in ambiguity, anticipation of exposure, constant checking of other people's reactions and avoidance of situations. 15'll take any pain but oh God, don't let anything happen to me like . . .have an accident in public or something would burst . . .that would just be the end of me.If something like that was to happen . . .I could be housebound for a good while because the thoughts of it would probably be . . .I couldn't imagine.Yeah, that would be the worst thing for me, it would be the embarrassment and the shame of . . .oh my God.(Article 1) People reported ways of coping.There were examples of how people had maintained or re-established a positive selfidentity, using positive self-talk, acceptance and not paying attention to it.There appeared to be a trend of finding it more challenging during younger years, with some reflecting that they had found ways to accept themselves over time.
'I've had to do a lot of soul searching and pep talks and say well in spite of these scars . . .you still look good.You still can think highly of yourself and still be sexy.You just have to wear different underwear certain days of the week but that's ok.' (Article 8) Disclosing and discussing their condition with others, be that family, friends, partners, healthcare professionals or others with HS, helped individuals feel supported or understood.People were careful about who they spoke to, and it was important that the person respond supportively for this disclosure to offer benefits.'I told my friend . . .and he was so cool about it.He's like "hey that's ok.That's something that you took your time to tell us, that's fine." . . .And I got two different positive reactions.'(Article 8) Social identity theory and self-categorization theory suggests individuals try to maintain self-esteem by viewing their ingroup favourably. 16,17In the case of stigmatized identities, such as HS, where individuals often have low self-worth, it is not always possible to view the ingroup favourably. 14One strategy that can increase self-worth is to strengthen a collective identity. 14This may explain why people with HS expressed a desire to speak to others with HS, seek online support groups, and reported benefits of connecting with other people with HS.These support networks may have the distinct benefit of maintaining a positive self-identity.'Having that support there, that's what really helped me through it . ... And because I could talk to them about it as well because they knew what was going on because . . .they've seen the sores, they knew I was in an awful lot of pain.' (Article 1) This suggests a paradox: people with HS cope by hiding their condition, both verbally and visually, but this situation makes it difficult to adjust psychologically to living with HS in the longer term.
Theme 3: Falling through the cracks Findings suggest that healthcare was frequently falling short of participants' expectations and needs.Participants reported a lack of knowledge about HS amongst healthcare professionals, and there is limited access to healthcare professionals who understand HS.These problems can lead to delays in diagnosis or to misdiagnosis, which can result in irreversible tissue damage due to the scarring nature of the condition. 18,19nother way that people with HS appeared to be 'falling through the cracks' of healthcare systems is that they often reported feeling misunderstood, unheard and dismissed by professionals, which in some cases may lead to mistrust and disengagement with services.The stigmatized identity, lack of sense of personal control over HS and low self-worth may be important factors that mean poor or unclear communication from healthcare professionals can easily lead to a breakdown in therapeutic relationships.
'I used to bawl my eyes out.I used to cry.You know, you'd come back after going to the doctors and you cry because they just don't realise.' (Article 1) Like other long-term conditions that cannot be cured, HS needs to be managed.People reported finding this challenging to accept and come to terms with.'I suppose the chronic aspect of it was a bit of a shock to me because I thought that this is a boil, this is an abscess . . .you know, I'll have it treated it will be gone.And then you learn, no this will go on for decades.' (Article 1) People reported not feeling adequately supported in managing the condition.HS involves self-management (e.g.bandages, pain relief, managing weight, etc.), as well as ongoing topical and oral medication or surgery where there are some concerns about the effectiveness and side-effects of the treatments.Discussions about weight management and stopping smoking, which are advised by healthcare professionals to improve self-management of HS, were particularly challenging for participants.They reported that healthcare professionals advise these changes without understanding the challenges for the individual, leaving people feeling stigmatized and 'dehumanized'.
For some participants who identified themselves as overweight, there was another source of shame.They reported that medical professionals told them to lose weight without realizing how difficult this was for them.They maintained that this emphasized their sense of shame about being overweight (Article 1).

Discussion
There were 14 studies included in our metasynthesis, which illustrated multiple challenges individuals face in living with HS and in seeking treatment and support.All studies eligible for inclusion were published within the last decade, which mirrors a general trend towards increased publication in HS research over the period, and the development of new treatments for the condition. 20,21iagnostic delay of HS is recognized as a global problem, with one international study across 24 countries showing patients with HS had an average diagnostic delay of 7Á2 years compared with an average diagnostic delay of 1Á6 years for patients with psoriasis. 22A German study found that a longer delay in HS diagnosis was associated with greater disease severity, an increased number of surgically treated sites, concomitant diseases, and a higher number of days of work missed. 18This review highlights how diagnostic delay can leave individuals feeling unheard and unsupported, potentially leading to mistrust of the medical community, and disengagement with healthcare services.
'Falling through the cracks' of healthcare services has a human cost.A cohort study using the Danish national register suggests people with HS are at an increased risk of death by suicide compared with people without HS. 23Many of the consequences of HS within the qualitative literature mirror the statistical data available on the impact of HS on quality of life, psychological wellbeing, sex life and disability. 8,24ome of these impacts, such as psychological impacts, low self-esteem and high levels of felt stigma, are also seen across other skin conditions. 25This review suggests that the concept of 'cumulative life-course impairment', which was developed in the psoriasis literature, is relevant to patients with HS, with the consequences of the disease having compounding impacts that can change the life course of an individual, and it advocates for early intervention where possible. 12A key difference between HS and other skin conditions is the often progressive nature of HS, as opposed to the typically relapsing and remitting nature of others such as eczema and psoriasis; furthermore, HS lesions often leave irreversible scars. 19here are limitations to this work.Reporting of studies was frequently incomplete, which often led to uncertainty of study quality.This is likely because five of the 14 studies were research letters/editorials and, of the eight full-length articles, five were reporting qualitative analysis within a wider outcome measure development project.Adequate reporting of qualitative methods and results is required for readers to assess the credibility and transferability of the data. 26Solutions include journal editors offering full article publication for substantial qualitative studies, use of supplementary materials or making details available on publicly accessible data-sharing websites.
Included studies came from just four countries (USA, Denmark, Ireland and Israel); participants typically had more severe HS and were largely white.The data on healthcare and treatment experiences (theme 3) were sparser than for the other themes.The data collated are limited by research available but the review team did not search the grey literature, which may have contained data missing within this review.
The experience of managing HS alongside other comorbidities was not reported in the qualitative literature reviewed.Because HS is associated with several other conditions (including inflammatory bowel disease, inflammatory arthritis, pilonidal sinus, polycystic ovary syndrome, Down syndrome, obstructive sleep apnoea and pyoderma gangrenosum), many people will be living with HS alongside other conditions. 1 Interactions of managing more than one long-term condition can have additional challenges that need to be acknowledged.
Given that the onset of HS is known to occur typically at around puberty, the experience of younger people with HS is largely missing, as of the articles that reported age of participants, the youngest was 18 years old.Understanding the experiences of young people living with HS is currently a gap in the literature.
Future research directions must address development, evaluation and implementation of interventions to address the needs of people with HS.The results of this review will be useful for informing the content of interventions.Interventions to address doctor-patient communication may be required.Observing the communication between healthcare professionals and patients with HS may offer useful insights on this.Interventions to improve the diagnosis of HS among general practitioners/family doctors are required.
Future qualitative studies focusing on the experiences of healthcare and treatment of HS are needed.While some experiences of living with HS will be universal across countries, differences in healthcare systems and societal attitudes mean it is important to recognize there may be crucial differences in experiences by country, and so qualitative studies exploring experiences in different countries would be beneficial.
Grounded in the key challenges this review has highlighted, we have proposed the following four key recommendations to improve HS management that we feel may improve patient experiences of healthcare for their HS.
(i) Early diagnosis is essential: delayed diagnosis, lack of healthcare professional knowledge and limited access to services needs addressing.Early intervention is key to improving the lives of people living with HS and educational materials for diagnosis of HS should be developed, evaluated and implemented, aimed at both primary and secondary care clinicians.
(ii) A multidisciplinary team approach is needed: the impact of HS is physical, psychological and social.HS management and research should be based on a biopsychosocial model of health addressing all impacts of the condition.This could be achieved with multidisciplinary teams, and the provision of psychological and social interventions alongside pharmacological and surgical interventions.
(iii) Patients need access to social support networks: additional provision of social support groups for those with HS may help them overcome the negative effects associated with stigmatization, and help them adjust to living with HS.Clinicians should signpost to local and national support groups.
(iv) Improved communication with patients is essential: there is a need for improved communication between people with HS and healthcare professionals, particularly for having conversations about the emotional impact of the condition and the behaviour changes such as smoking and weight management, which patients report as challenging.This may be achieved through appropriate training for healthcare professionals.
In conclusion, the expressed experiences of people with HS suggest that this is a challenging condition to live with.It can have profound psychological and social impacts on an individual, impair physical functioning, and impact multiple aspects of their daily life.Social support and psychological acceptance appear to be coping strategies that facilitate adjustment to living with HS, but there need to be changes in the delivery of healthcare, with adequate psychological and social support, to enable people with HS to live their life more fully.

Figure 1
Figure 1 PRISMA flow diagram reported; IQR, interquartile range.a Some studies contain data from the same set of participants, while one article contained data for multiple sets of participants (interview participants and focus group participants reported separately), so from the 14 included studies, there are 11 sets of participants.b Analysis of comments on Facebook group of nearly 850 members, but no detail on number of people posting comments.c The Hurley staging system is a classification to grade the severity of HS.41

' 5 , 6 , 7 , 9 ©
Time spent on treatment' (Article 12) 7, 12, 14 Concern about side-effects or symptoms from treatments 'Concern about medication and side-effects' (Article 9) 7, 9, 12 Financial burden '"I've spent hundreds and hundreds of dollars a month in, um, co-pays, stupid treatments that I was told to try again that didn't work in the first place, because nobody believes me . ... Just the overall financial burden of what's happened to me."' (Article 5) 2021 The Authors.British Journal of Dermatology published by John Wiley & Sons Ltd on behalf of British Association of Dermatologists British Journal of Dermatology (2021) 185, pp921-934

Table 1
Study characteristics HS, hidradenitis suppurativa; HiSQOL, Hidradenitis Suppurativa Quality of Life; HRQOL, health-related quality of life; PRO, patient-reported outcome; QoL, quality of life.© 2021 The Authors.British Journal of Dermatology published by John Wiley & Sons Ltd on behalf of British Association of Dermatologists British Journal of Dermatology (2021) 185, pp921-934

Table 2
Participant characteristics

Table 3
Descriptive themes on experiences of living with hidradenitis suppurativa (HS) Descriptive themes on living with HS Illustrative quotations ID of studies with data on subtheme Symptoms and impacts Physical sensations.People described physical sensations or symptoms of HS.There was clearly one set of symptoms for active disease and another relating to damage from the disease.
'Patients generally look at dating as troublesome, because they find the situation with disgusting lesions too complicated and too difficult to explain, and this makes them push people away."Usually, I have no problems talking with people, but in such situations I simply shut people off."A patient reports with regret that she has realized that "I am not going to marry anyone and in any case I am not going to have children."'(Article 6)

Table 3
© 2021 The Authors.British Journal of Dermatology published by John Wiley & Sons Ltd on behalf of British Association of Dermatologists British Journal of Dermatology (2021) 185, pp921-934 Experiences of people with HS: a systematic review, L. Howells et al. 929

Table 4
Descriptive themes on experiences of treatment for hidradenitis suppurativa (HS) People mostly described a lack of knowledge among healthcare professionals about HS.There were a few descriptions of knowledgeable healthcare practitioners who had helped them manage the condition.