Background: Pilonidal disease is considered a spectrum of disease ranging from acute abscesses to a chronic state most often associated with draining sinuses in the gluteal fold. An increasing prevalence is seen in children with 3 out of every 250,000 children developing Pilonidal Sinus Disease (PSD). Although this is a common disease within general and paediatric surgery departments worldwide, there is yet to be an agreement on a ‘Gold Standard ‘of treatment. Minimally invasive treatment techniques such as EPSiT are a more recent development for pilonidal disease that are showing promising results.

Method: This study is a retrospective case series of children and adolescents who underwent excision of pilonidal disease using EPSiT by a single paediatric surgeon in Western Australia. The primary outcome measure is recurrence, secondary outcomes include complications, length of hospital stay, time to return to school/work, post-operative pain, and time to complete wound healing.

Results: No patients suffered recurrence or wound complications at mean follow up of thirteen months. No patients were readmitted to hospital for procedure related complications. All patients reported complete wound healing at an average of twenty-eight days and definitive total wound closure at six weeks post operatively. All patients reported the ability to resume daily activities at 2-4 days post operatively.

Conclusion: PEPSiT is a promising method of treating paediatric PSD and may represent a new `Gold Standard’ of treatment in these patients. Our results were comparable to other studies in the literature. However, more studies and large-scale randomized control trials are necessary in the paediatric patient cohort.

• PEPSiT; Endoscopic treatment of pilonidal sinus disease; Case report; Complications

Gera P, O’Sullivan JL (2024) Endoscopic Treatment of Pilonidal Sinus Disease in Paediatric Patients – in Western Australia. JSM Pediatr Surg 3(1): 1019.

PEPSiT: Paediatric Endoscopic Pilonidal Sinus Treatment; EPSiT: Endoscopic Pilonidal Sinus Treatment; PS: Pilonidal Sinus; PSD: Pilonidal Sinus Disease; PC: Primary Closure; PMC: Primary Midline Closure; OW: Open Wound; EHSI: Excision and Healing by Secondary Intention; ASCRS: American Society of Colorectal Surgeons; LA: Local Anaesthetic; RCT: Randomised Control Trial; VAC: Vacuum Assisted Closure; ADL: Activities of Daily Living; LOS: Length of Stay; MLF: Midline Flap; LF: Limberg Flap; KF: Karydakis Flap; VAAFT: Video Assisted Anal Fistula Treatment; VAS: Visual Analogue Scale 

Pilonidal sinus disease is a chronic disease affecting the sacrococcygeal area and natal cleft [1]. It has a broad spectrum of disease ranging from acute abscesses to chronic draining sinuses [2]. It is a common disease in both paediatric and general surgery departments with a prevalence of approximately 13 out of every 50,000 adults [3]. The aetiology is not fully understood with various theories regarding the pathophysiology of PSD. Many believe PSD formation is due the ‘congenital theory’ of sinuses existing as a remnant of the caudal aspect of the neural tube during embryonic development [4]. The ‘acquired theories’ are more widely accepted, such as the Bascom theory of ‘midline pits’, or the ‘Karydakis theory’ of an inflammatory foreign body response, however the consensus is that chronic irritation and recurrent infection of the area at the level of the hair follicle results in a low-grade inflammatory response [5,6]. This disease is found to affect predominately males aged 15-25 years old, however, it is being seen more frequently in the paediatric cohort [7]. The risk factors include excess hair in the natal cleft, obesity, sedentary lifestyle, ethnicity, and family history of PSD [8]. The surge of PSD in children may be partly explained by rising paediatric obesity rates globally as well as more time spent sitting at a desk and or computer [9].

For the last few decades, many techniques have been used to treat this common disease including Excision with Packing and Healing by Secondary Intention (EHSI), Primary Closure (PC), flap techniques such as Karydakis and more recently minimally invasive methods such as trephination, sinusectomy and endoscopic treatment [10]. The primary goal of treatment is to reduce recurrence, with secondary outcomes being reduced morbidity, shorter length of stay in hospital, reduced pain scores and improved quality of life [11]. The goal of surgical management is to treat not only the lesion but also address all risk factors associated with recurrence such as re-growth of hair in the natal cleft and poor perineal hygiene [8]. The historically used open, closed or flap based excisional treatments are associated with prominent levels of both recurrence and morbidity which is particularly distressing in the paediatric cohort [12,13]. Due to the unsatisfactory outcomes they were having in their paediatric patients treated with traditional methods for PSD, Meneiro and Milrone et al concomitantly developed the endoscopic pilonidal sinus disease treatment technique based on their experience with the VAAFT procedure in anal fistulas. This minimally invasive technique has been shown to reduce recurrence, morbidity, post- operative pain, and length of hospital stay with faster time to complete healing [14,15]. This procedure could represent a new ‘Gold Standard’ of treatment and significantly improve quality life in paediatric patients with pilonidal sinus disease.

This study presents a single-surgeon’s experience of endoscopic treatment for pilonidal disease in paediatric patients in Perth, Western Australia.

Prior to undertaking research, ethical approval was granted independently via HREA by St John of God Healthcare ethical approval boards. All participants gave written, informed consent to be included in this study. All participants were contacted via phone for the study questionnaire and given corresponding information via e-mail. Participants were given the option to opt-out of the study at any time point during the study period. We retrospectively reviewed the data of 5 consecutive paediatric patients who underwent endoscopic treatment of pilonidal sinus diseases over a 12-month period. All surgeries were performed in Perth, Western Australia by a single surgeon. Patients over 16 years of age at time of surgery, those who presented with acute pilonidal abscesses and those who had prior surgical intervention for PSD were excluded from the study. Data on patients presenting complaints, phenotype and operative procedure were gained from prospectively maintained patient records. After consent had been gained, patients were contacted to gather information on recurrence, wound infection, wound healing time, length of hospital stay, post-operative pain, time to return to activities of daily living and overall patient satisfaction. Follow up was at a single point in time, on average at 13 months post-surgery. A hairy body in this populations was defined as males or females that have visibly long hair, dark colour and feels coarse when touched, localized to the thighs, buttocks, and other parts of the Body. The surgical success rate was complete wound healing during the at 60 days post operatively. Recurrence was considered when symptoms of swelling, discharge, or local pain re-occurred post total wound closure or >4 months post total wound healing. Post-operative pain score was assessed using the Wong-Baker FACES visual pain scoring system [16].

The surgical equipment includes the specially designed MENEIRO Fistuloscope (Karl Storz SE& Co), a unipolar electrode connected to a high-frequency electrosurgical generator, a fistula brush and an optical forceps. The fistuloscope with incorporated fibreoptic light transmission is equipped with a straight working channel which is also used for irrigation. The length of the fistuloscope shaft is 18 cm; the use of a handle reduces it to an effective length of 14 cm.

All patients were recommended to undergo laser epilation both before and after surgery. All patients were admitted to hospital on the day of surgery. All procedures were performed under General Anaesthesia. The surgical and anaesthetic technique were standardized throughout the procedure. The patient is positioned prone with their legs apart. The buttocks are separated by two large elastic tape bandages. The surgeon may position themselves either to the right of the patient or in between their legs depending on the position of the sinus and or surgeons’ preferences.

Monitors are placed in view of the surgeon, depending on which positioning he/she chooses. Local anaesthesia is applied to the sinus openings. IV Cefazolin is administered prophylactically. The procedure consists of a diagnostic and a therapeutic phase. The diagnostic phase involves identifying the anatomy of the sinus, the sinus’ extension, secondary tracts and or any cysts present. The fistuloscope enters the sinus via the external opening. If there are multiple pits, the lower most sinus is opened and the fistuloscope is inserted. If the sinus opening is too small, it is enlarged using a spreading clamp. Continuous irrigation of 0.9% Normal Saline is used to facilitate better endoscopic view and the removal of waste and debris. The therapeutic phase aims to ablate and clean the area. This consists of the removal of all hair, follicles, and debris by the grasping forceps. The sinus is the debrided and the cavity is curetted using the endobrush. Haemostasis is achieved using monopolar coagulation. Monopolar is also used for ablation of granulation tissue in the sinus tract. Debris is then removed and debrided using the endobrush.

Post-operative

Post operatively a PICO dressing is applied. This is kept in situ for 7 days and then replaced with a sterile dressing. Simple analgesia- acetaminophen and ibuprofen were used for pain management. No antibiotics are administered post operatively. All patients are discharged on the day of surgery. Regular Sitz baths are recommended for perineal hygiene. Information and recommendations are given on laser epilation and maintenance of good perineal hygiene once the wounds are healed.

Follow up

All patients were followed up by the operating surgeon at one and six weeks post operatively.

Statistics

Data underwent statistical analysis using Microsoft Excel. Graphs and tables were generated from Excel software.

Five paediatric patients underwent endoscopic treatment of pilonidal sinus disease that were eligible for inclusion in the study. One patient did not consent to be included: therefore, four patients were recruited. Our study consisted of 3 male and 1 female patients. The mean age at time of treatment was 15 years. Two of our patients reported excess hair at the natal cleft. All patients were of Caucasian descent.

All patients reported chronic discharging sinuses at the natal cleft. None of the patients included in our study reported acute abscesses. All our study participants had received no prior treatment for PSD. All patients had one pilonidal sinus opening. All pilonidal sinus openings were in the midline. All patients underwent the surgical procedure described above under general anaesthesia. All patients had a PICO negative pressure dressing applied to the operative site in the operating theatre. All patients included in our study were discharged on the day of the procedure. All patients reported a pain score of 0-2 on the Wong- Baker FACES pain score index [16]. Only one patient reported the need to use simple analgesia. The average time off school was two days. This was not applicable to one patient as the procedure was completed during school holidays and therefore, could not be assessed. No patients suffered recurrence at a mean follow up of thirteen months. No patients reported wound complications- haematoma, seroma, infection, or dehiscence. No patients were admitted to hospital for procedure related complications. All patients attended their routine follow up at one and six weeks post operatively. All patients had their PICO dressing removed at one week follow up and replaced by simple dressings.

All patients reported complete wound healing at an average of twenty-eight days. All had definitive total wound closure at their six week follow up appointment. All patients reported the ability to resume daily activities at two- four days post operatively, with none of the participants in this study reporting pain as a limiting factor to resumption of ADLs. All patients reported significant improvement in quality-of-life post treatment with PEPSiT. All our patients said they would recommend PEPSiT for the treatment of PSD.

The treatment of PSD remains controversial and paediatric surgeons are yet to agree on a ‘Gold Standard’ approach. The technique chosen is determined by both surgeon preference and their perception of which method has the lowest recurrence, rates of complication and fastest healing time [17]. Many treatment methods are used, including excisional techniques such as en- bloc resection, healing by secondary intention, flap techniques and marsupialization, all of which have been described with differing success rates [10].

However, all techniques demonstrated high recurrence rates and a high frequency of surgical complications, which are unacceptable in the treatment of such a prevalent disease [10]. Patients treated using the above methods often have a long and painful post-operative period with much time spent off school and work [10]. The ideal treatment for PSD should have a clear, reproducible surgical procedure with a low incidence of recurrence, morbidity, minimal pain and quick return to activities of daily living [18].

The surgical procedure is one of the clear advantages of the endoscopic treatment of PSD. The direct endoscopic view of the sinus cavity obtained by the fistulascope during the procedure, allows for straight forward exploration, identification and removal of any hair and debris, as well as identification of any sinus tracts or abscess cavities that are present, while achieving effective haemostasias [14,19]. The use of pre-existing sinus openings as the site of entry minimizes tissue disruption and local trauma, reducing surgical morbidity [20]. This method is seen to significantly reduces post-operative pain, with only a single patient in our study reporting the need to use simple analgesia, and all reporting a pain scope of <2 on the FACES paediatric pain score index [16]. Larger scale studies such as that by Milone et al report comparable results with an average VAS score of 1-2, in comparison to studies of conventional methods which show average pain scores of 7 in these patients [15]. All patients reported that their daily activities were not impacted by post-operative pain. Traditional surgical methods are associated with prominent levels of recurrence, varying in different techniques from 4-31% [18]. A study by Abraham et al found recurrence rates in both Primary Closure (PC) and Excision and Healing by Secondary Intention (EHSI) to be 11-18% in their group of 135 patients [18]. Research undertaken by Fike et al has demonstrated high recurrence in both EHSI (25%) and in PC (25%), showing that almost a quarter of patients treated by these methods can expect recurrence, with PC having the highest rate of wound complications [17]. Concerningly, some European countries have advocated for abandonment of primary midline closure due to reported recurrence rates as high as 48% and over half of patients suffering wound dehiscence during the recovery phase [21]. Although most of the data describing recurrence rates thus far is heterogenous with large differences in results, papers analysing EPSiT versus conventional methods show the clear advantages of endoscopic treatment [1,15,19,20]. A comparison study by Milone et al found recurrence rates in those treated with EPSiT to be 7.8% at four years compared to 25% in those treated with sinusectomy [15].

Although out study is relatively small scale in comparison to the above, we reported no recurrence over an average follow up of 13 months, which is comparable to those in the literature. Large scale studies assessing the outcome of treatment using EPSiT over a longer period show a recurrence rate of <5% over a 36month period in those treated with EPSiT [22]. This clearly demonstrates the superiority of EPSiT over other methods with regards recurrence. The second consideration in the treatment of paediatric pilonidal sinus disease is the rate of complications.

Traditional methods are associated with high morbidity and significant post-operative complications. This may be due to their lengthy post-operative course, large cavities at substantial risk of seroma or haematoma formation and frequent dressing changes [15,17,23,24]. A European study of paediatric patients comparing OW and Karydakis procedure by Roldon Golet et al found Karydakis to be the superior method with regards recurrence (4% vs 28.6%), however, they found high rates of wound infection at 15.4%, with over a quarter of patients having some type of wound complication (Claven-Dindo Grade I-III) [24,25]. These rates are lower than a study by Bali et al which found complications as high as 23% in their population [26]. Fike et al demonstrated flap techniques to be the most successful operative intervention with the lowest rates of recurrence, and complications however they still had considerable recurrence and complication rates at 15% and 21% [17]. In our study no patients suffered wound complications with a time to complete wound healing being approximately 28 days. The literature states complication rates in those treated with EPSiT to be approximately 5%, with significantly reduced rates of post- operative infection, with a comparative complete healing time on average being 28.5 days [22]. A promising study by Esposito et al reported a wound infection rate of 0% when EPSiT was used in conjunction with laser epilation and either silver nitrate or oil-based dressings [27]. The low complication rates may be explained by the minimal tissue disruption caused during surgery, the small surgical site, and the lack of interaction with the site post operatively by means of daily dressings and surgical drains, all which pose an infection risk. Our patients underwent wound review at 1 and 6 weeks post operatively, with little interaction with the surgical site. The prompt time to complete healing also has a positive effect on reducing wound related complications with complete healing in endoscopic treatment occurring on average in 3.1 weeks compared to 6.1 weeks in EPC and 14.9 weeks in EHSI [1].

EPSiT, due to the nature of the procedure, as well as low rates of complication and recurrence is associated with a faster return to school and work with lower pain scores than conventional methods [1,2,10,15,28]. In our study, patients reported feeling well enough to return to school at 2 days post operatively, with all patients being discharged on the day of surgery. This is in keeping with the literature which reports an average LOS in hospital at 20 hours with return daily activities being reported at between 1-5 days postoperatively [1,29]. Recent studies advocate for safety of EPSiT to be performed as a day case procedure, a significant improvement on the average 4–6-day hospital stays associated with conventional methods [27]. In conjunction with treating the disease, addressing the risk factors of PSD formation plays a key role in reducing recurrence. Hirsutism or excess hair at the natal cleft is a recognized risk factor for PSD formation [3,5,6,8]. Laser epilation, in multiple studies has been shown to reduce recurrence by eradication of the risk factors - excess hair in the natal cleft, being recommended by the ASCRS for the treatment of PSD [18,30-32]. All our patients carried out post-operative laser hair epilation post total wound closure which may contribute to the low rates of recurrence. Quality of life is a significant factor to consider, especially in the paediatric patient. In a study by a group of researchers at Stanford University comparing quality of life factors pre and post minimally invasive treatment they found that 66% of patients reported severe impact on activities of daily living by PSD, with 94% of patients reporting little to no impact of PSD post treatment and 92% experiencing improvement in sport participation, school, and work attendance as well as socializing [33]. Traditional methods such as PC or EHSI, as discussed, lead to high post-operative morbidity, length of stay in hospital, time off daily activities as well as laborious and painful dressing changes. Most concerning are the high recurrence rates. This results in many days of school or work, avoidance of social scenarios, inability to participate in sport causing significant social isolation, emotional and psychological burden on these patients and their families at a crucial age of development [10,33] .

In our study, all patients reported a significant improvement in quality-of-life post treatment, with all patients reporting that they would recommend this treatment to a person in a similar scenario. This speaks volumes of the effectiveness of the use of PEPSiT.

Limitations are noted in this study. Most notably the small sample size, by a single surgeon across two sites, which yields much bias. The follow up time is relatively short at thirteen months. Some of our results rely on patients own memory when events occurred, such as time to complete wound healing, which has significant bias. As this is carried out by a single surgeon, who has mastered the technique; therefore, this may not be representative of the paediatric general surgeon cohort. Other biases are that in Australia this treatment is not yet covered by Medicare so is limited to patients with private healthcare. This has biases as these patients have more resources available to them such as access to post-operative care including laser epilation treatment, and SITZ baths which can be costly. This study also only involved patients who had no prior treatment for PSD. Results may be different in those with prior surgical intervention, complex PSD or those presenting with acute abscesses.

Endoscopic treatment of pilonidal sinus disease in paediatric patients is an innovative approach that thus far in the literature has shown promising results. A recent paper published by Esposito et al whose objective was to standardize the PEPSIT procedure described a five-step process standardizing the PEPSiT procedure which has a success rate of 98% [25] [27]. There is a paucity of literature on the use of this method in the Australasian cohort of paediatric patients, with most prior studies being conducted by European researchers. This study shows that PEPSiT is an effective method for treatment of PSD in the paediatric cohort, with reduced recurrence, minimal pain and a fast time to return to daily activities. Our results were like other studies in the literature. However, more studies and randomized control trials are necessary to be conducted in the paediatric patient cohort. Standardization of the surgical procedure and post-operative management is necessary for the future advancement of PEPSiT, to ensure its reproducibility and adequate standard of care.

Nonetheless, even considering these factors, the results of PEPSiT have been promising, and although follow up time was on average 13 months, the initial results, and benefits of PEPSiT over other techniques is obvious. We believe PEPSiT to be a promising method of treating paediatric PSD and may represent a new `Gold Standard’ of pilonidal sinus disease treatment in these patients.

Funding: No funding received to complete this study.

Level of Evidence: IV

Conflicts of Interest: Authors have no identifiable conflicts of interest.

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