An international survey exploring the management of pilonidal disease

Pilonidal disease (PD) is a common debilitating condition frequently seen in surgical practice. Several available treatments carry different benefit/risk balances. The aim of this study was to snapshot the current management of PD across European countries.


INTRODUC TI ON
Pilonidal disease (PD) is a common condition encountered by clinicians in medical practice.Typically, the doctor examining a patient with PD may have little interest in this condition, with their knowledge of PD often limited to general surgery textbooks that sometimes convey wrong or obsolete information on the disease.Studies since the 1980s marked a turning point in the debate on the congenital versus acquired nature of PD, strongly supporting the theory that PD arises from ingrown hairs [1].Some physicians may take for granted that wide excision with or without primary suturing is the only way to treat PD, thereby discrediting the less invasive therapeutic approaches proposed since the 1960s and which later surged in popularity [2][3][4].
In the attempt to minimize confusion and false myths on PD, guidelines grounded in evidence-based medicine and consensus statements have been developed by dedicated national scientific societies in the United States [5] and Europe [6][7][8].However, it remains difficult to establish the 'on the floor' impact of these guidelines over the years, matched to a parallel increase in knowledge and the launch of newer technologies.In other words, it is still unclear who is treating who, what and how.Similar 'snapshot' have already been taken at a national level.For instance, 983 studies from the literature on the management of PD were the object of a recent mapping review by Kumar et al. [9], summarizing eight decades of research on the topic.A total of 36 studies were systematic reviews with/ without meta-analyses, 121 randomized controlled trials and 826 observational studies.
A recent national survey from the Netherlands on the surgical treatment of PD concluded that traditional excision techniques are still the most frequently performed in that country, but the majority of surgeons and surgical residents remain unsatisfied with the current treatments [10].
It was therefore decided to launch a survey at a continental level and under the auspices of the European Society of Coloproctology (ESCP) to snapshot the current management of PD across Europe.

Statistical analysis
Continuous variables were summarized by means and standard deviations (SDs), while categorical variables were assessed by proportions.Comparisons of categorical variables across groups were made using Pearson's chi-square test.

What does this paper add to the literature?
This is the first international survey to explore the manage- The data were statistically described according to sociodemographic traits and variables related to the question answers.
Response percentages for items were calculated according to the number of respondents per response out of the total responses for that item.Starting from the raw qualitative recorded data, after applying a data transformation in order to obtain dummy variables, we applied a logistic regression based on a stepwise procedure for variable selection [12,13].Thus, the associations between responses related to the question 'Suggested type of elective treatment of PD (first presentation)', i.e. 'Surgical' or 'Conservative'; 'Do you change your therapeutic approach in case of failure/ recurrence after the first treatment?',i.e. 'Yes' or 'No'; 'Do you think that the use of patient reported outcome measures (PROMs) could be useful in the evaluation of a patient with PD?', i.e. 'Yes' or 'No'; sociodemographic characteristics and all the other responses were evaluated.The data were used to estimate six models, one for each of the above-mentioned question answers.All the variables included in the databases, including the dependent variables, were qualitative and hence treated as dummy variables [12].The categories within each variable were grouped to obtain a sufficient sample size.Because the grouping procedure dichotomized the dependent variables, a logistic regression model was used with a stepwise procedure to select the explanatory variables based on the Akaike information criterion [12,13].The denominator of the percentages of respondents was the total number of respondents who eventually completed the survey.Adjustment to the p-values was not performed.However, considering the number of tests performed, p-values of <0.05 were critically appraised to consider the risk of false positives.Statistical analysis was performed using MATLAB R2019a (The MathWorks Inc.).

RE SULTS
Four hundred and fifty-two subjects located in 48 different countries answered the questionnaire (Appendix S4).Most of them [121 (24.8%)] came from Italy, with 13.9% and 13.5% from Spain and Turkey, respectively, and 6.4% from the United Kingdom.Their main sociodemographic characteristics are shown in Table 1.
More than a third [170 (38%)] were exclusively affiliated to the ESCP, with a fair representation of other coloproctological scientific societies, especially the Italian Society of Colorectal Surgery and the Turkish Society of Colon and Rectal Surgery, accounting for 12% of respondents (Figure 1).
Respondents were mostly men (77%), age range 26-60 years, practising in both academic and public hospitals, with a heterogeneous experience in the treatment of PD.Over a third of respondents [175 (39%)] declared that they generally treated between 11 and 100 cases per year; 3% dealt with more than 100 cases per year.Almost all the respondents were surgeons, with an even distribution between colorectal (51%) and general (48%) surgeons.
More than two-thirds [331 (73%)] of respondents preferred surgical treatment even at first presentation of the disease, whereas 141 (31%) opted for first-line conservative treatment.
Up to 80% of these recommended antibiotic therapy, irrespective of the type of presentation and 95% did not use any disease classification.
A primary closure technique was the most preferred procedure among respondents (29%), followed by the open technique (22%), flap (7%), sinusectomy (7%), marsupialization (7%) and others (less than 6% each, reported in Table 2).Most respondents stated their willingness to change their therapeutic approach according to the presentation of the disease, number and side of pits and type/extension of PD (limited or extensive).
Approximately 27% of subjects would recommend the same surgical technique even after a failure.Almost half of respondents (46%) performed office-based treatments, with 27% adopting 1-day surgery (overnight stay) and 21% an outpatient (walk in/walk out) setting.About two-thirds (66%) of respondents routinely scheduled surgery 4-8 weeks after an acute episode (Table 3).Most respondents (40%) reported recurrences of less than 5%, followed by 30% reporting a recurrence rate of 5%-10%.
The vast majority (74%) declared that they perform direct physical examination on follow-up visits, in 35% of cases at 1 year from  4-6.No significant associations were found between the other questions and answers and all the other predictors.
A conservative approach was negatively associated with acute presentation of PD (e.g.pilonidal abscess).On the contrary, it was associated with surgeons aged between 41 and 60 years working in public, private and university hospitals (Table 4).
For those respondents who were unwilling to change their practice in case of failure (27%), the negative associations found in Table 5 show that respondents were less prone to not change their therapeutic approach when the recurrence rate was about 11%-15% and when PD presented with acute abscess, number and site of pits and in the presence of limited or extensive PD.On the other hand, positive associations found between the same item and flap and open techniques as first-line treatment strategies, with respondents shown to be more prone to not change their therapeutic approach in such situations (Table 5).
Finally, PROMs were rarely evaluated as useful by surgeons in cases of a low PD recurrence rate (<5%), when patients were followed-up with physical examination and when the speciality of the respondent was general surgery (Table 6).No significant associations were found between the other questions and answers and all the other predictors and evidence related to other factors (e.g.geographical variation, case volume, further demographic aspects, healing times) (Table 6).

DISCUSS ION AND CON CLUS ION
This is the first international survey to evaluate trends in the management and treatment of PD.One of the most representative findings of this study is that 95% of the respondents did not use any classification system for PD [14].Indeed, even though PD is a common disorder that every surgeon faces at least once in his or her career, only a tiny minority follow a classification system.This involves both those who do not usually deal with PD and those who perform a large number of procedures each year.Classifications have the potential role of predicting prognosis, guiding treatment and making outcomes comparable for research purposes.Without a universally acceptable classification system, comparative trials carry unacceptable selection and outcome-reporting bias.A standardization of the diagnosis and classification based on location and severity might be helpful for comparing techniques and results, and therefore in the identification of the most effective kind of assessment.This is likely to apply to the subgroup of patients with advanced disease who may be poor candidates for a given intervention and at the same time respond differently to different surgical strategies [14,15].On the other hand, as acknowledged by national guidelines, no validated classification of PD exists even if sinus characteristics may modify surgical decision-making and postoperative outcomes.Features to be considered before recommending conservative treatment or surgery should cover the number of pits, their location in relation to the midline, the distance of the most caudal pits from the anal verge and the presence of previous incisions or scars [8].Therefore, it would be desirable to collect data and create a classification applicable to all professionals who encounter PD, both for clinical-therapeutic and research reasons.However, recurrence of disease and the correct choice of surgical technique remain crucial topics in the management of PD.In an Australian survey conducted in 2018 [16], the authors registered 64% of respondents who reported recurrence rates above 5% and 37% surgeons with rates exceeding 10%.Six per cent reported no recurrences at all.Five per cent reported recurrence rates over 20%, but 24% stated that over one-fifth of their practice consisted of recurrent disease.In our survey, none of the respondents registered 'no recurrences', while the vast majority (40%) reported a recurrence rate of less than 5%, 30% had recurrence rates of 5%-10% and 16% of the surgeons acknowledged recurrence rates of 11%-15%.The fact that more than 50% of participants were dedicated colorectal surgeons may partly explain this finding.Given that the Karydakis procedure was the most frequently performed operation in the Australian study, this indicates that the recurrence rate outside of clinical trials and published series may be substantially higher than previously assumed.
Most respondents in our study reported using an open or a closed technique (with a primary closure).Even though recently numerous publications have demonstrated the technical feasibility and favourable short-term results of endoscopic and laser techniques [17], these procedures respectively accounted for just 5.5% and Overall, more than 10 different techniques were considered in our survey (Table 2).Interestingly, almost 6% and 1% of surgeons would perform a pit picking technique (Bascom technique) or Gips procedure, respectively, as a first line of treatment.Most studies recommend the use of pit picking and its variations in previously untreated patients with minimal disease.In some case series it is evident that once the learning curve has been accomplished, indications can be also extended to recurrent disease, but reliable longterm results are lacking [18,19].One of the substantial advantages could be the low cost of these procedures and the resulting high cost-effectiveness [18].
Notably, procedures requiring intensive postoperative healthcare involvement (e.g.regular dressings or packing) will have significant healthcare staff and consumable costs in addition to societal costs, including time off work, in a young, active working patient population.Increased equipment costs for interventions such as endoscopic pilonidal sinus treatment and laser therapy or consumable costs such as fibrin glue must be also be included in the costeffectiveness calculations [20].
In our cohort of respondents, 31% would recommend a conservative approach at first presentation of PD.The German guidelines TA B L E 4 Stepwise regression model of 'Conservative approach' response as the suggested type of elective treatment of pilonidal disease in relation to significant variable answers in 452 respondents.Prolonged wound healing and time out of work, as well as nonnegligible recurrence rates, are the main drawbacks of this approach.The Italian guidelines stated that open healing should be limited to complex cases, since the benefits concerning recurrence are not clear and postoperative recovery may be longer [8].

SE
Interestingly, even if not supported by international recommendations, 29% of respondents preferred a primary closure of the wound after excision.There is a strong consensus against midline wound closure as it correlates to a high incidence of dehiscence and an increased recurrence rate.German guidelines recommend Karydakis or Limberg flaps as off-midline procedures; however, their use instead of open healing is still not advocated [6].
Another relevant consideration regarding PD concerns the category of patients most affected by the disease, i.e. young adults at an age where body image is more important than at other stages of life, when relationships are formed and attendance at study or work is crucial to progression in life.How, if at all, were patients involved in designing these guidelines and their perspectives?In the study by Strong et al. [21] investigating patient decision-making and regret after surgery, the burden of wound care and the disparity between anticipated and actual recovery times were the main reasons for decisional regret.
Long-term healing may not be compatible with a young adult daily life, especially not at the cost of disfiguring scaring, packing or time away from normal activities.According to 36% of our respondents, patients needed 3-4 weeks to recover, 28% of them considered 1-2 months as the recovery time, while 18% of the surgeons considered 2 weeks enough for recovery.Of course, this depends on the technique chosen and the severity of PD at presentation.Attention should be paid to counselling patients, especially regarding the burden of wound care and the risks of recurrence associated with different surgical approaches.
Several studies have demonstrated a lower postoperative recurrence rate with hair removal after complete wound healing, and the superiority of permanent laser compared with razor/cream epilation has been demonstrated in terms of recurrence.More than half (57%) of respondents to our survey would recommend permanent laser epilation after surgery.
Interestingly, in a recent publication about the role of telemedicine in proctology, teleconsultation was deemed appropriate only for the diagnosis and management of PD [22].Indeed, the study high-

A
24-item survey (Appendix S1) was designed and developed by the authors using an online platform ['Online survey' (formerly BOS-Bristol Online Survey), developed by the University of Bristol] in accordance with the Consolidated Criteria for Reporting Qualitative Research (COREQ) and the Checklist for Reporting Results of Internet E-Surveys (the CHERRIES statement; Appendix S2) [11].The questionnaire was constructed with careful consideration of the areas of open debate in various published guidelines on pilonidal disease.Each question was included to address specific aspects related to the condition and treatment.Proprietary survey software and local servers were used to ensure data protection.The fully deidentified dataset was kept on password-protected computers.The authors piloted the survey, assessed the design and checked the feasibility and validity of the questions.The estimated mean time required to complete the survey was 6 min.The finalized online survey was made available for 6 weeks, from 10 February to 24 March 2022, and disseminated among members of the ESCP.Participants could be identified only via their valid email address; no other identifying information was collected.A link to the survey was posted on social media (i.e.LinkedIn and Twitter) to capture further potential respondents.Participation was entirely voluntary, with no compensation offered.Informed consent was obtained from all those agreeing to complete a survey.The survey aimed to capture the current treatments for PD and was structured in the following two sections: 1. Demographics (Q1-Q7): including respondents' gender, age range, city and country of clinical practice, type of hospital, speciality and affiliation to coloproctological societies.2. Treatments for PD (Q8-Q24): including yearly case volume, firstchoice treatment, use of antibiotics, reasons for changing the original therapeutic plan, time interval from acute presentation to definitive treatment, rate of partial or complete dehiscence after closed approaches, operative setting, recurrence rate, time to recover, use of advanced dressings or postoperative permanent laser epilation, follow-up modalities and usefulness of patient reported outcome measures (PROMs).All questions were set as mandatory fields with real-time validation and automated skip logic to prevent missing data and avoid illogical or incompatible responses.No randomization of items was used.Quantitative data were automatically collected by the software and exported to a tabulated format.All those who completed the survey were included as collaborators (Appendix S3).
ment of pilonidal disease.It shows that newer minimally invasive procedures have not yet achieved widespread distribution.Although increasingly discredited, excision and leave open techniques are still supported by a fifth of respondents as first-line therapy.

4 .
9% of choices in our survey, implying that they have not yet fully entered the surgeon's armamentarium.The endoscopic method has the advantage of treating the tracts under vision in contrast to pit picking or laser treatment.In their mapping review on PD, Kumar et al. claimed the need for trials comparing minimally invasive procedures with standard care [9].The authors also confirmed and highlighted the absence of clear, front-running surgical interventions for PD and the presence in the literature of a great heterogeneity in the definition, measurements and clinical outcomes, as pointed out in a recent systematic review [17].
Table depictingstepwise regression model of 'conservative approach' response as suggested type of elective treatment for PD in relation to significant variable answers in 452 respondents.Positive and negative associations are reported in normal and bold type, respectively.Abbreviations: CL, confidence limit; PD, pilonidal disease; SE, standard error.TA B L E 5 Stepwise regression model of 'No change of therapeutic approach' response in case of failure/recurrence after the first treatment in relation to significant variable answers in 452 respondents.What type of surgery is your first line treatment for PD: flap What type of surgery is your first line treatment for PD: open technique Do you change your therapeutic approach according to: Presentation of PD (e.g.acute abscess), number and site of pits, limited or extensive PD Do you change your therapeutic approach according to: presentation of PD (e.g.acute abscess), number and site of pits, limited or extensive PD, other Table depicting stepwise regression model of 'No change of therapeutic approach' response in case of failure/recurrence after the first treatment in relation to significant variable answers in 452 respondents.Positive and negative associations are reported in normal and bold type, respectively.Abbreviations: CL, confidence limit; PD, pilonidal disease; SE, standard error.published in 2021 recommend no treatment in the case of asymptomatic disease.In most patients with pilonidal abscess, incision and drainage is recommended, followed by elective surgery after resolution of the acute inflammation [6].Despite the slow spread in the use of minimally invasive and offmidline procedures, the open healing technique is still one of the most frequently used methods worldwide.Around 22% of respondents would choose this procedure for surgical treatment of PD.
lighted poor acceptability of telemedicine as first-line assessment TA B L E 6 Stepwise regression model of 'Yes' response for the usefulness of patient reported outcome measures in the evaluation of a patient with pilonidal disease in relation to significant variable answers in 452 respondents.Table depicting stepwise regression model of 'Yes' response for the usefulness of patient reported outcome measures in the evaluation of a patient with pilonidal disease in relation to significant variable answers in 452 respondents.Positive and negative associations are reported in normal and bold type, respectively.Abbreviations: CL, confidence limit; GP, general practitioner; SE, standard error.
Distribution of participants based on national and international scientific society.
E 1 TA B L E 2 Clinical and surgical approaches.n% How many cases of PD do you examine/treat each year Abbreviations: EPSiT, endoscopic pilonidal sinus treatment; FiLaC, fistula laser closure; PD, pilonidal disease; VAAPS, video-assisted ablation of pilonidal sinus.TA B L E 3 Setting, failure and follow-up.n%