Endoscopic Treatment of Pilonidal Sinus Disease in Children: A Systematic Review

Introduction

Pilonidal sinus disease (PSD) is an inflammatory disease that occurring in young and teenagers at the sacrococcygeal region with a reported incidence of 26:100,000. ¹ Often in association with obesity, hirsutism, sedentary occupation, and local irritation, the disease seems to have hirsutism as the main risk factor ² ; in fact, the disorder resulted in the occlusion of hair follicles in the natal cleft. ³ First, described by Herbert Mayo in 1833, in 1880, the disease was called by Hodges with the word “pilonidal” from the Latin words “pilus,” meaning hair, and “nidus,” meaning nest.^4,5 Symptoms vary from asymptomatic cavity to acute abscess or to chronic cyst; associated conditions with PSD are variable but with repercussions on the patients' quality of life. ⁶

In the past decades, a lot of different approaches have been proposed for the treatment of this disease both in adult and children but the most effective treatment remains debatable. The ideal surgical procedure should be focused on the eradication of the cyst and the removal and cleaning of the sinus tract and secondary tracts, to ensure the complete and long-lasting healing with acceptable cosmetic results.^7,8 According to the literature, the traditional open excision and healing by secondary intention is associated with a prolonged and painful hospital stay and high morbidity rates. ¹ Despite of shorter wound healing time, open excision with primary closure, including flap-based procedures, showed an excessive rates of problems related to the wound, including recurrence, infection, and wound dehiscence.^8,9 The most serious problem for the pediatric surgeons is the recurrence of the disease after traditional surgery ranging from 0% to 40%, for various techniques.^10,11

PSD is a frequent disease affecting pediatric population and young adults, with complication and recurrence percentages comparable to the adults.^12,13 The Minimally Invasive Endoscopic approach called Endoscopic Pilonidal Sinus Treatment (EPSiT), first reported in 2014 by Meinero et al., describes a new procedure for PSD treatment, with encouraging effects in adults such as a quick wound healing and time off work and also better pain management and cosmesis.^13,14 Esposito et al. in 2018 reported their surgical procedure for the treatment of PSD in pediatric population (PEPSiT).^15,16

Materials and Methods

Results

A total of 203 studies were obtained from the literature search but 165 articles were excluded: 122 not fitting title/abstract, 37 for article type (21 reviews/meta-analyses, 8 letters to the editor/commentary, 8 case reports/technical notes), 4 as they were not written in English, and 2 no full text available. We pooled 38 studies, but 18 of these were excluded because they were either not relevant or concerned an adult population. We report all the exclusion criteria in Figure 1. Cohort and comparative studies on the pediatric population are reported in Tables 1 and 2, respectively.

OPEN IN VIEWER

FIG. 1.

Inclusion and exclusion criteria.

Discussion

The gold standard surgical technique for pilonidal sinus in children is still under debate. ¹⁸ The current management is quite similar both for adult and pediatric patients; in fact many pediatric patients are often operated by general surgeons. In 1990, Allen-Mersh performed an extensive review about the treatment of pilonidal sinus, showing that the right surgical technique is related to the surgeon's experience and perceived results in terms of healing speed and recurrence rate. ¹⁹ Nowadays, the tendency for medical treatments to progress from standard surgery to minimally invasive techniques is represented in pilonidal sinus management. Sinusectomy, a type of barely invasive procedure, a surgical approach managed with an edged incision of the pilonidal orifices preventing wide cutaneous margins and a careful subcutaneous asportation of the sinus without closure of the wound, is performed.

This approach proposed by Soll ²⁰ was a new minimal invasive procedure for pilonidal sinus to prevent open enbloc excision in adults; this technique seems to report a low recurrence rate and a rapid return to daily activities in adults. Despite some emerging minimally invasive techniques in the adult population, open procedures, enbloc or not, give various troubles such as painful and prolonged postoperative days with late return to everyday routine and in some cases, a long and distressing healing process. Moreover these approaches reported recurrence rates higher than 30%, with a long recovery times, extended use of resources, redo surgery, and low patients' satisfaction. There are not many articles in the literature regarding PSD in children and adolescents even if PSD is a common condition in children and teenagers.

Due to the growing evidence and development of new instruments, minimally invasive surgical procedures are nowadays becoming widespread deriving from the idea that endoscopic techniques with the removal of all the infected areas by way of small circular incisions are the best choice. One of these techniques is EPSiT inspired by video-assisted anal fistula treatment (VAAFT) described by Meinero and Mori in 2011. Similar to VAAFT procedure, EPSiT was first described by Meinero et al. in 2014. Since the initial trial on pediatric population was developed by Esposito et al., the choice of the endoscopic procedure has received great consensus even in the group of pediatric surgeons. ¹⁵ Esposito et al., have in fact carried out this technique in children and teenagers, Pediatric EPSiT or PEPSiT, with modifications and very promising long-term outcomes.

As for the technique, a 10 ch fistuloscope equipped by an operative channel and an endoscopic brush, a monopolar electrode and an endoscopic grasping forceps is needed. In opposition to the procedure described by Meinero, PEPSiT adopts a continuous jet of saline or mannitol solution instead of glycine-mannitol to guarantee, during the procedure, a limpid visual field, but optimizing the economic repercussions of the surgical technique.

In pediatric population, compared to open technique, PEPSiT is associated with low-grade postoperative pain, short hospitalization (about 1 day), and good cosmetic results. The recurrence rate has a range from 0 to about 6% demonstrated by several noncomparative reports.^16,21–24 PEPSiT is considered the technique of choice in children and adolescents affected by PSD by many authors. Compared to other surgical approaches, this procedure is related to better postoperative results and better postoperative outcomes. However, some important restrictions of the present literature should be considered. First, especially in pediatric patients, the articles published in literature are mostly retrospective, with a not well-represented population. As demonstrated by Doll et al., the follow-up should be not <5 years.^25,26

In fact, an important bias to give final conclusions on this procedure is the brief follow-up of the included articles. Another significant factor to be considered is that no information is available about the learning curve and the costs of this procedure. It would be stimulating to carry out studies on the learning curve between young surgeons or residents to define the number of procedures needed to achieve good skills. Furthermore, it has been considered that the cost of the endoscopic technique could be higher than that of open surgery. However, the economic outcomes could be balanced by the high patient satisfaction, less postoperative hospital stay, and shorter time off work.

The review of the literature shows that the endoscopic approach seems to have significant postoperative advantages compared to other surgical procedures, and it should be the gold standard treatment for PSD in pediatric population. However, the lack of studies on the learning curve and available data on long-term follow-up will open to further high-quality studies to give definitive conclusions on this technique.

Conclusions

After performing an analysis of the literature, we can say that endoscopic techniques for PSD are superior to excision and primary closure (EPC) especially for the postoperative conditions and wound healing time and length of hospital. PEPSIT showed to be a very promising alternative and superior to EPC in terms of recurrence and complications.