The management of pilonidal disease: A systematic review

YJPSU-59150; No of Pages 12 Journal of Pediatric Surgery xxx (xxxx) xxx

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Review Article

The management of pilonidal disease: A systematic review Julia Grabowski a,⁎, Tolulope A. Oyetunji b, Adam B. Goldin c, Robert Baird d, Ankush Gosain e, Dave R. Lal f, Akemi Kawaguchi g, Cynthia Downard h, Juan E. Sola i, L. Grier Arthur j, Julia Shelton k, Karen A. Diefenbach l, Lorraine I. Kelley-Quon m, Regan F. Williams e, Robert L. Ricca n, Roshni Dasgupta o, Shawn D. St. Peter b, Stig Sømme p, Yigit S. Guner q, Tim Jancelewicz e a

Division of Pediatric Surgery, Ann and Robert H. Lurie Children's Hospital, Northwestern University, Chicago, IL Department of Surgery, Children's Mercy Hospital, Kansas City, MO c Division of Pediatric General and Thoracic Surgery, Seattle Children's Hospital, Seattle, WA d Division of Pediatric Surgery, British Columbia Children's Hospital, University of British Columbia, Vancouver, Canada e Division of Pediatric Surgery, Le Bonheur Children's Hospital, University of Tennessee Health Science Center, Memphis, TN f Division of Pediatric Surgery, Department of Surgery, Medical College of Wisconsin, Milwaukee, WI g University of Texas McGovern Medical School and Children's Memorial Hermann Hospital, Houston, TX h Division of Pediatric Surgery, Hiram C. Polk, Jr, MD Department of Surgery, University of Louisville, Louisville, KY i Division of Pediatric Surgery, University of Miami Miller School of Medicine, Miami, FL j Division of Pediatric Surgery, St. Christopher's Hospital for Children, Philadelphia, PA k Division of Pediatric Surgery, University of Iowa Stead Family Children's Hospital, Iowa City, IA l Department of Pediatric Surgery, Nationwide Children's Hospital, The Ohio State University, Columbus, OH m Department of Surgery, Children's Hospital Los Angeles, Keck School of Medicine, University of Southern California, Los Angeles, CA n Division of Pediatric Surgery, Naval Medical Center, Portsmouth, VA o Division of Pediatric General and Thoracic Surgery, Cincinnati Children's Hospital Medical Center, University of Cincinnati, Cincinnati, OH p Division of Pediatric Surgery, Children's Hospital Colorado, University of Colorado, Aurora, CO q Division of Pediatric Surgery, Children's Hospital of Orange County, Orange, CA b

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Article history: Received 19 December 2018 Received in revised form 2 February 2019 Accepted 27 February 2019 Available online xxxx Keywords: Pilonidal Pediatric Adolescent Sinusectomy

a b s t r a c t Objective: The goal of this systematic review by the American Pediatric Surgical Association Outcomes and Evidence-Based Practice Committee was to derive recommendations from the medical literature regarding the management of pilonidal disease. Methods: The PubMed, Cochrane, Embase, Web of Science, and Scopus databases from 1965 through June 2017 were queried for any papers addressing operative or non-operative management of pilonidal disease. The Preferred Reporting Items for Systematic reviews and Meta-Analyses (PRISMA) guidelines were followed. Consensus recommendations were derived for three questions based on the best available evidence, and a clinical practice guideline was constructed. Results: A total of 193 articles were fully analyzed. Some non-operative and minimally invasive techniques have outcomes at least equivalent to operative management. Minimal surgical procedures (Gips procedure, sinusectomy) may be more appropriate as first-line treatment than radical excision due to faster recovery and patient preference, with acceptable recurrence rates. Excision with midline closure should be avoided. For recurrent or persistent disease, any type of flap repair is acceptable and preferred by patients over healing by secondary intention. There is a lack of literature dedicated to the pediatric patient. Conclusions: There is a definitive trend towards less invasive procedures for the treatment of pilonidal disease, with equivalent or better outcomes compared with classic excision. Midline closure should no longer be the standard surgical approach. Type of study: Systematic review of level 1–4 studies. Level of evidence: Level 1–4 (mainly level 3–4). © 2019 Elsevier Inc. All rights reserved.

⁎ Corresponding author at: 225 East Chicago Box 25, Chicago IL 60611. E-mail address: [email protected] (J. Grabowski). https://doi.org/10.1016/j.jpedsurg.2019.02.055 0022-3468/© 2019 Elsevier Inc. All rights reserved.

Please cite this article as: J. Grabowski, T.A. Oyetunji, A.B. Goldin, et al., The management of pilonidal disease: A systematic review, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.02.055

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J. Grabowski et al. / Journal of Pediatric Surgery xxx (xxxx) xxx

Contents 1.

Methods . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 1.1. Research questions . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 1.2. Search methods . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 1.3. Study selection . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 1.4. Full review process . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 2. Results . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 2.1. Question 1: Are non-operative and minimally invasive management strategies effective treatment for pilonidal disease? . . . . . . . . . . 2.1.1. Standard hair removal (non-epilation) and hygiene without surgery . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 2.1.2. Epilation (laser or intense pulse light hair removal) as primary treatment . . . . . . . . . . . . . . . . . . . . . . . . . . . 2.1.3. Postoperative epilation . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 2.1.4. Phenol . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 2.1.5. Alternative minimally invasive methods: fibrin glue, laser probes, radiofrequency ablation . . . . . . . . . . . . . . . . . . . . 2.2. What are the indications, contraindications, and associated complications with various operative techniques for pilonidal disease? . . . 2.3. Minimal surgery . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 2.3.1. Incision and drainage (I&D) . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 2.3.2. Sinusectomy . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 2.3.3. Endoscopic . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 2.3.4. Excision . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 2.3.5. Flaps . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 2.4. What operative and non-operative approaches for the treatment of pilonidal disease are associated with the highest quality-of-life and/or patient satisfaction? . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 2.4.1. Wide excisional versus limited excisional, minimal surgical approaches, or non-operative approaches . . . . . . . . . . . . . . . . 2.4.2. Flaps . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . 3. Discussion . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . References . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . . .

Pilonidal disease (PD) is a common and often debilitating infectious and inflammatory condition of the gluteal cleft and sacrococcygeal region. It is characterized by sinus and abscess formation, typically in association with midline openings that entrap hair and debris [1]. Patients may have significant pain, drainage, and bleeding. Chronicity and recurrence are common. The disease process can range in severity from small, asymptomatic pits to multiple tracts and fistulization away from the midline. PD is frequently encountered by pediatric surgeons because it disproportionately affects adolescents and young adults, with a peak incidence between 14 and 25 years of age [2]. Although it occurs equally in males and females during adolescence, it is more common in males than females after the teenage years [2,3]. There are innumerable reported approaches to the surgical management of PD, ranging in complexity from simple drainage to intricately designed multi-flap closures. For decades, standard definitive care has consisted of excision with either secondary healing or primary closure of the wound; these approaches were originally derived largely from military hospital experience with “Jeep riders' disease” [4,5]. Until recently, little has changed over time in the basic surgical approach to PD, despite a surge in the volume of literature on the topic. The purpose of this systematic review was to identify and summarize the best available literature regarding PD in order to provide an updated set of guidelines for management. The study focused on newer operative and nonoperative approaches, and, where possible, on the concerns of the pediatric surgeon and patient.

1. Methods 1.1. Research questions The American Pediatric Surgical Association (APSA) Outcomes and Evidence Based Practice (OEBP) committee selected the following questions for this systematic review: 1. Are non-operative and minimally invasive management strategies effective treatment for pilonidal disease? 2. What are the indications, contraindications, and associated complications with various operative techniques for pilonidal disease?

0 0 0 0 0 0 0 0 0 0 0 0 0 0 0 0 0 0 0 0 0 0 0 0

3. What operative and non-operative approaches for the treatment of pilonidal disease are associated with the highest quality-of-life and/ or patient satisfaction? 1.2. Search methods With the assistance of a biosciences and bioinformatics librarian, the PubMed, Cochrane, Embase, Web of Science, and Scopus databases from 1965 through June 2017 were queried for any papers addressing operative or non-operative management of pilonidal disease. All non-English papers, animal studies, case reports or small case series, clinical trial designs, abstracts without manuscripts, studies of non-gluteal pilonidal disease, and articles written earlier than 1965 were excluded. We included all ages in the search given the paucity of studies focusing solely on the pediatric and adolescent populations. Search terms included “pilonidal disease or sinus” and the following terms: “surgery or surgical or hair or laser or hygiene or epilation or wound or wounds or debridement or healing or marsupial or excision or excised or drainage or Limberg or flap or flaps or closure.” We included additional articles when identified in the references of selected articles (i.e. snowballing). The Preferred Reporting Items for Systematic reviews and MetaAnalyses (PRISMA) guidelines were followed [6]. 1.3. Study selection A PRISMA flow diagram of the search and exclusion process is presented in Fig. 1. The list of 2380 titles and/or abstracts generated by the automated search was reviewed independently by three authors (J.G., T.J., and T.A.O.) and 1710 articles were excluded, as they did not address any of the study questions. The remaining 670 abstracts were allocated to the most appropriate question for full review. Accepted articles (N = 532) were allocated to the most appropriate study question and then reviewed for quality of evidence and pertinence to the questions. Some articles were analyzed for multiple questions. 1.4. Full review process Each article was assessed for level of evidence based on Oxford Centre for Evidence-Based Medicine (OCEBM) study design (Table 1),

Please cite this article as: J. Grabowski, T.A. Oyetunji, A.B. Goldin, et al., The management of pilonidal disease: A systematic review, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.02.055

J. Grabowski et al. / Journal of Pediatric Surgery xxx (xxxx) xxx

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Fig. 1. PRISMA flow diagram of the search and exclusion process.

population definition and source, study period, intervention or exposure, comparison measures, outcome measures, sample size, and findings. A total of 193 articles that met inclusion criteria were fully analyzed (Fig. 1). Many exclusions of low-level studies occurred where level 1 and 2 evidence was available for a particular topic. A consensus statement of recommendations in response to each study question was developed based on the results of our study synthesis. Based on the best collected evidence from this study, a clinical practice guideline was generated.

2. Results 2.1. Question 1: Are non-operative and minimally invasive management strategies effective treatment for pilonidal disease? Minimally invasive procedures were defined as non-excisional procedures performed either in a clinic or operating room setting. This included hair removal and hygiene both in lieu of surgery or postoperatively, phenol injection, and others procedures including fibrin injection, laser therapy, and radio-frequency ablation (all excisional and flap procedures were assessed in Question 2). The procedures described focused primarily on the management of pilonidal disease in patients who presented without an abscess or as an adjunct treatment after

incision and drainage in patients presenting with an abscess. Thirtyfour articles addressed this question. Table 2 summarizes these data. 2.1.1. Standard hair removal (non-epilation) and hygiene without surgery Two papers examined standard hair removal in lieu of surgery. The management algorithms included shaving, gluteal cleft hygiene, and patient education. A 1994 study in Archives of Surgery used a military population in a two-part analysis including a pilot study where excisional operations were performed for all cases of symptomatic pilonidal disease. This initial population was then compared to a prospective cohort who were treated with a conservative approach: shaving, hygiene, patient education, exercise restrictions and incision and drainage when needed for abscesses. Data on the follow-up cohort were collected over the next 17 years. The authors found a total of 240 operations in 229 patients in the pilot study compared to only 23 operations on severe recalcitrant pilonidal disease in an estimated 350,000 at risk patients in the subsequent years. They concluded that the conservative approach could avoid operations in the majority of patients. [7] A 2012 study retrospectively reviewed 235 patients initially managed non-operatively over 10 years [8]. The management of these patients included shaving and daily application of a chlorine solution to the gluteal crease. Patients who had persistent symptoms went on to surgical excision. The authors determined that 57% of patients failed to resolve with non-operative measures. Significant risk factors for failed

Table 1 Oxford Centre for Evidence-Based Medicine (OCEBM) 2011 Levels of Evidence. Levels of Evidence

Grades of Recommendation

1 2 3 4 5

A B C D

Systematic review of randomized trials or n-of-1 trials Randomized trial or observational study with dramatic effect Non-randomized controlled cohort/follow-up study Case-series, case–control studies, or historically controlled studies Mechanism-based reasoning (expert opinion)

Consistent Level 1 studies Consistent Level 2 or 3 studies or extrapolation from Level 1 studies Level 4 studies or extrapolations from Level 2 or 3 studies Level 5 evidence or inconsistent or inconclusive studies

Adapted from OCEBM Levels of Evidence Working Group*. “The Oxford Levels of Evidence 2”. Oxford Centre for Evidence-Based Medicine. http://www.cebm.net/index.aspx?o=5653 *OCEBM Levels of Evidence Working Group = Jeremy Howick, Iain Chalmers (James Lind Library), Paul Glasziou, Trish Greenhalgh, Carl Heneghan, Alessandro Liberati, Ivan Moschetti, Bob Phillips, Hazel Thornton, Olive Goddard and Mary Hodgkinson.

Please cite this article as: J. Grabowski, T.A. Oyetunji, A.B. Goldin, et al., The management of pilonidal disease: A systematic review, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.02.055

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J. Grabowski et al. / Journal of Pediatric Surgery xxx (xxxx) xxx

Table 2 Interpretation of the literature regarding outcomes of various non-operative procedures approaches.

Hygiene/standard hair removal Pre-operative epilation hair removal Post-operative epilation Phenol Laser Fibrin injection Radio Frequency Ablation

Complications

Initial Success

Recurrence

None None Minimal Minimal Minimal Minimal Minimal

Poor Moderate Moderate Excellent Moderate Moderate Excellent

Poor Poor Moderate Excellent Moderate Moderate Moderate

non-operative management included male gender, previous treatment at other facility, previous abscess, and sinus length N 3 cm. 2.1.1.1. Recommendations. Standard hair removal and hygiene may be used as first-line therapy for pilonidal disease, but has a substantial failure rate when used alone. Level 3–4 evidence; grade C recommendation. 2.1.2. Epilation (laser or intense pulse light hair removal) as primary treatment Epilation techniques in this section included laser hair removal (LHR) and intense pulsed light (IPL). The most common lasers described in the literature are the Alexandrite and neodymium-doped yttrium aluminum garnet (Nd:YAG) lasers. IPL differs from LHR in that it is less precise and risks damage to surrounding tissue. There was no standard technique between papers for amount of hair removed or number of sessions. Data regarding cost were lacking in most studies, though most authors commented that epilation was not covered by insurance. Follow up was variably reported. Three papers studied the use of epilation as primary treatment. One prospectively studied patients between 2008 and 2012 who underwent IPL for management of pilonidal disease as both primary treatment and after surgical management. Overall, there was a 13.3% recurrence rate. The recurrence rate included operative and non-operative patients and the number of non-operative patients treated with only IPL was not specifically reported [9]. A retrospective study compared excision, LHR, and observation for the years 2005–2013. Recurrence trended higher in the LHR group compared to the surgical excision group, but, due to small numbers, this difference was not significant despite the large effect size (55.6% v. 34.0%, respectively, p = 0.11) [10]. Finally, a 2009 retrospective study mentioned patients with asymptomatic pits who underwent LHR alone; of the 3 patients who only had LHR, none developed symptomatic pilonidal disease [11]. 2.1.2.1. Recommendations. Epilation may be used in lieu of surgery but there may be an increased rate of recurrence. Level 3–4 evidence; grade C recommendation. 2.1.3. Postoperative epilation Six papers were included that evaluated epilation used postoperatively to prevent disease recurrence. There were two randomized controlled trials, one prospective study, and three papers with level of evidence 4. Overall, there were 275 patients within these studies and the recurrence rate was between 0 and 20% [11–16]. Two randomized control trials were identified with contradictory results. The first randomized 60 post-excisional patients after a Karydakis flap to either no hair removal or LHR [12]. After 1 year follow-up, the authors found that the LHR patients had a 20% recurrence rate compared to only 4% recurrence rate in those who did not have LHR. This result was statistically significant. The authors concluded that LHR does not decrease risk of recurrence in post-operative patients, but recommended that larger studies be performed as this was contrary to their hypothesis. Conversely, a prospective trial randomized 86 post-operative patients to either LHR or “conventional razors or depilatory” hair removal [13]. All patients had undergone surgery with excision and closure by

Comments Number of treatments and area of hair removal varied Conflicting data Type and percentage of phenol uncertain

secondary intention. At a mean follow-up of 2 years, there were no recurrences in the LHR group and two recurrences (4.4%) in the conventional hair removal group. Four remaining studies focused on post-operative LHR. A 2009 paper reported a prospective nonrandomized trial of patients who underwent excision of a pilonidal sinus and then chose either LHR or no hair removal. At an average follow-up of 12–23 months, there was a 70% recurrence rate in the control group compared to 0% recurrence rate in those who underwent LHR [14]. The 2009 study by Lukish et al. (discussed above) included 25 patients who underwent LHR post-operatively. Of these 25 patients, one (4.4%) had a recurrence at a mean follow-up of 24 months [11]. Another retrospective review reported on 60 patients who had post-operative LHR. This study observed a 13% recurrence rate at a mean follow-up of 4.8 years. Most recurrences were in those patients with pre-operative abscess [15]. Another study described 19 patients who underwent post-operative LHR and reported a 0% recurrence rate at mean follow-up of 4 years. Notably, there was a 36% rate of wound complications, and this risk was highest in obese patients [16]. 2.1.3.1. Recommendations. There is conflicting data on whether postoperative laser hair removal decreases the rate of recurrence of pilonidal disease. Hair removal may be attempted prior to or after other interventions. Level 2–4 evidence; grade C recommendation. 2.1.4. Phenol Phenol, or phenolic acid, for the treatment of pilonidal disease has been described in the literature since 1964 and has most commonly been performed in European and Middle Eastern countries. Although there are some differences in the methods used, the general technique involves cleaning of the sinus, instillation of phenol into pits, and suctioning or denaturing the phenol. The procedure is often repeated to achieve success. Because of the toxic nature of phenol, complications including a dull sensation in the area, abscess, or cellulitis are described [17]. Seventeen papers examined the effectiveness of phenol injections in the treatment of pilonidal disease. Outcomes studied included wound healing rate and recurrence. Several studies compared the effectiveness of different concentrations of phenol, while others evaluated the use of phenol in combination with other procedures. The most robust study looking at phenol was a prospective randomized trial that included 140 patients who underwent either crystallized phenol injection or operative excision with open healing from 2011 to 2014 [18]. The primary endpoint was complete wound healing. All relevant outcomes favored phenol, including complete wound healing, median operative time and healing time. Recurrence rate was less than 20% in both groups and not statistically different between groups. In two retrospective studies, phenol injection was compared to operative excision; the first compared 132 patients who underwent either operative excision with Limberg flap or application of crystallized phenol [19]. The recurrence was 8% in those who had excision and flap, and 12% in those who had phenol application and this difference was not statistically significant. Given the higher rate of infections, wound complications, and cosmetic complaints in the flap group, these authors favored phenol as first-line treatment. The second study compared 117 children who were treated with either operative excision with primary

Please cite this article as: J. Grabowski, T.A. Oyetunji, A.B. Goldin, et al., The management of pilonidal disease: A systematic review, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.02.055

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closure (N = 77) or crystallized phenol injection (N = 40). Patients who underwent operative excision had significantly higher number of days requiring dressings, longer operating room times, and longer hospitalizations. There was also a higher rate of intravenous pain medication administration in the operative group. Between the operative and phenol groups, there was a trend towards lower rates of complications (10.4% vs 2.5%) and recurrence (13% vs 2.5%) in the phenol group, though these did not reach statistical significance. The authors noted that there was a significantly longer follow-up period in the operative group (44 months) compared to the phenol group (8.1 months) [20]. Two randomized control trials were identified that compared the effectiveness and complications of different concentrations of liquid phenol. These studies had contradictory results. One compared a single application of 80% phenol to 30% phenol and showed a statistically significantly higher recovery rate in the 80% phenol group. There was no difference in recurrence or complications between groups at mean follow-up of 1 year [21]. In contrast, the other trial randomized 112 patients to either 40% or 80% liquid phenol. At a mean follow-up of 2.8 years, they found 40% phenol had significantly higher rates of initial success, lower number of recovery days required, as well as lower rates of recurrence and complications (abscess, skin necrosis, and fatty tissue necrosis) [22]. Seven studies examined the use of either liquid or crystallized phenol and reported descriptive statistics alone. In these studies, a total of 318 patients were treated with phenol. Success rates varied between 59 and 95.1% [17,23–28]. Finally, three papers evaluated the success of phenol in combination with other procedures. One analyzed endoscopic pilonidal sinus excision with the addition of crystallized phenol in 23 patients and found no recurrences at 12 months [29]. Another combined phenol application with laser hair removal in 42 patients and found no recurrences at 42 months [30]. The third described a procedure in which a substantial pit excision was combined with 80% phenol use and found that 86.7% of patients were asymptomatic at an average follow-up of 25.7 months [31]. 2.1.4.1. Recommendations. Phenol is effective in the management of pilonidal disease with low recurrence and complication rates. Further studies are warranted to compare percentage and type of phenol applied and the applicability in the pediatric population. Combining phenol with hair removal or excisional procedures may increase efficacy. Level 2–4 evidence; grade B recommendation. 2.1.5. Alternative minimally invasive methods: fibrin glue, laser probes, radiofrequency ablation 2.1.5.1. Fibrin glue. Although use of fibrin glue is described as a therapy for pilonidal disease in several different ways, including as an adjunct to excisional surgery and flap procedures, this section focuses on fibrin used as a primary treatment without excision. All evaluated studies entailed cleaning and shaving the diseased area, followed by pit excision and injection of fibrin (Tisseal™). A prospective study of 40 patients who underwent pit clearing with fibrin instillation found a 10% recurrence at a mean follow-up of 18 months [32]. A retrospective study in 2013 reported on 93 patients who underwent fibrin injection as monotherapy. The authors found that 74% of patients required no further treatment and 71% of patients returned to normal activities by 2 weeks [33]. Exact duration of follow up was not reported. A retrospective comparison of lateralizing flap procedure (N = 10) to those who had undergone pit excision with fibrin instillation (N = 26) over the years 2006–2013 was performed in pediatric patients. Recurrence after primary procedure was similar between those who underwent surgical excision and those who had fibrin instillation: 20% vs 15% (p = 0.63). Similarly, overall recurrence was not statistically different between treatment groups. Operating time was

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significantly lower in the fibrin group with no difference in wound complications or infection [34]. 2.1.5.2. Laser Probes. Several different types of laser probes have been used as an alternative means to treat pilonidal disease. The aim of laser treatment is to use high energy at the tip of the probe to destroy the squamous epithelium of the pilonidal sinus and induce contraction/obliteration of the tract [35]. This technique is currently being used to treat varicose veins, anal fistulae and hemorrhoids. The different lasers described vary in wavelength and amount of energy delivered. A retrospective series of 40 patients treated with the FILAC™ radial laser probe between 2014 and 2015 documented an 87.5% success rate with 2.9% recurrence. The mean follow-up period was 234 days. Four patients presented with complications: 2 hematomas (5%) and 2 abscesses (5%), which were all medically treated [35]. A retrospective review of 70 patients that underwent epilation utilizing the Sharplan laser probe included patients with acute abscesses as well as those with inflamed or chronic pilonidal disease [36]. Their technique involved a small incision overlying the diseased area, hair removal and curettage and instillation of hydrogen peroxide. The laser probe was then used on the affected tissue. The small wound was packed and patients were followed for an average of 12 months. The overall recurrence rate was 11.4%. Of these recurrences, half were treated with another laser procedure while 4% went on to wide local excision. Complications included pain, bleeding, and skin bridging in 5.7% of patients. A retrospective series of 37 patients who underwent Nd:YAG laser treatment from 2006 to 2009 included patients who underwent laser treatment of pits at 1 month intervals for an average of 5.1 months. At follow-up, 28 of the 37 patients (75.7%) reported no symptoms without additional interventions, and 30 patients (81%) were symptom-free after further laser treatment and minimal surgery. The median followup time was 15.2 months. Some patients described temporary soreness and redness at the site of laser treatment [37]. 2.1.5.3. Radiofrequency ablation. Radiofrequency ablation (RFA) has also been described as a minimally invasive technique to treat pilonidal disease [38]. An RFA electrode is inserted into the sinus pits and skin and subcutaneous tissue is excised with a minimal margin. The goal is to make a small, cone shaped wound which is then covered by a dry dressing and closes by secondary intention. One group in India extensively studied and published the utility of RFA probe excision in pilonidal disease between 2004 and 2005. This method of excision utilizes radiofrequency electrodes of different sizes and shapes to simultaneously cut and coagulate tissues in a minimally traumatic fashion. The 3 studies identified included level 2, 3 and 4 evidence. Comparison of RFA excision to wide excision with either marsupialization or open healing favored the RFA procedure with respect to operative time, postoperative pain, hospital stay, and time off from work (p b 0.05). There was no difference in recurrence rate in these three studies [38–40]. 2.1.5.3.1. Recommendations. Alternative minimally invasive techniques, including fibrin injection, laser probes, and radio frequency ablation may be used in patients with pilonidal disease with rates of healing and recurrence comparable to other methods. Level 2,3,4 evidence; grade C recommendation. 2.2. What are the indications, contraindications, and associated complications with various operative techniques for pilonidal disease? Surgical treatment of PD is frequently complicated by: (1) surgical site infection (SSI); (2) delayed or failed wound healing; (3) pain and protracted convalescence; and (4) recurrence of disease. Most included studies address some or all these specific outcomes. After exclusions, there were three level 1 studies, six systematic reviews, 51 randomized controlled trials (RCT, level 2), 10 prospective cohort studies or large database studies (level 3), 12 case–control studies, 63 retrospective cohort

Please cite this article as: J. Grabowski, T.A. Oyetunji, A.B. Goldin, et al., The management of pilonidal disease: A systematic review, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.02.055

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studies, and 122 case series (level 4) assessed. Publication dates were 1965–2017. Very few reports focused on the pediatric patient or included a pediatric subgroup analysis. The majority of studies excluded patients who presented with an abscess, and there is no validated standard method to report severity of presentation [41]. Thus, the below recommendations regarding surgical management should, unless otherwise stated, be considered to refer to patients of all ranges of severity who either did not have an abscess or had any abscess drained with resolution of acute inflammation prior to more definitive surgical intervention. 2.3. Minimal surgery 2.3.1. Incision and drainage (I&D) There is near-universal consensus that the presence of a pilonidal abscess merits at least incision and drainage (I&D) as primary treatment. Recurrence rates after simple I&D range from 10 to 79% [42,43]. One RCT examining simple I&D versus I&D with curettage for patients with an abscess showed that curettage was associated with faster and more likely healing, and a lower recurrence rate at up to 5 years (11% versus 42%, P b 0.001) [44]. 2.3.2. Sinusectomy Excision of skin around pits (i.e. sinusectomy) with curettage of the space or “pit picking” has been promoted as an effective initial approach for all types of presentation (abscess, draining sinus). A version of this technique without curettage was described by Lord and Millar in 1965, with recurrence rates highly dependent on the quality of postoperative wound care [45]. Bascom's simple surgery involves midline sinusectomy with a separate off-midline incision and curettage of the pilonidal space, followed by closure of the sinusectomy site(s); this approach has a b10% recurrence rate [46,47]. Neither of these approaches has been assessed in higher-quality level 2 studies.

Sinusectomy using trephines with curettage, peroxide irrigation, and open healing without packing, also known as minimal incision surgery or minimally invasive pilonidal excision (MIPE), may be an optimal initial approach because of its simplicity as well as rapid recovery and low recurrence rates, and it is gaining popularity amongst pediatric surgeons [48,49]. The original retrospective cohort study of 1358 patients by Gips et al. in 2008 reported a recurrence rate after 1 year of 6.5%; 5 years, 13.2%; and 10 years, 16.2%. Only 39.1% of patients required analgesics postoperatively [50]. There was no difference in morbidity or recurrence rates between patients presenting with an abscess and those with chronic disease. The recurrence rate in an even larger study of 2347 patients from 2016 was 5.8% at a median follow-up of 16 months [51]. As yet, there are no level 2 studies evaluating this procedure. Implementation of a Gips-type minimal approach at a children's hospital was associated with a recurrence rate of 28% that was not significantly different from a wide-excision approach, but patients had fewer days on analgesics (0 versus 2.5, P = 0.005), fewer patient sick days (4 versus 14, P b 0.001), and fewer days to full activity (10 versus 45, P b 0.001) [49]. The median time to reoperation was 12 (6–15) months in the minimal group and 28.5 (24–33) months in the excisional group (P = 0.041). Follow-up time was shorter for the minimal group at 28 (17–37) months, compared with 50 (41–52) months for the excisional group (P b 0.001).

2.3.3. Endoscopic Endoscopic pilonidal sinus treatment (EPSiT) or video-assisted ablation of pilonidal sinus (VAAPS) involves use of a cystoscope and irrigation to clean and debride the pilonidal space under direct visualization, with healing of the small opening(s) by secondary intention. In addition to adult case series [29,52–57] and one pediatric case series [58] showing low recurrence rates, there is an RCT comparing VAAPS with the Bascom cleft lift (Bascom II, Fig. 2) [59]. There was less time off work

1

A

B

C

B

2

C A

Rhomboid (Limberg) flap

Karydakis flap

Bascom cleft lift

Fig. 2. Drawings of the three best-studied flap closures in pilonidal disease: the rhomboid or Limberg flap (rotational flap), the Karydakis advancement flap, and the Bascom cleft lift (Bascom II) advancement flap. The modified Limberg is shown here, with the inferiormost apex created off-midline; the short dashed lines repreent the flap incisions. The Karydakis closure is ~2 cm off-midline. With the cleft lift procedure, the area of apposition of the buttocks is reduced afterwards (lateral dashed lines), along with elimination of the gluteal cleft. For each diagram, the midline/gluteal cleft is depicted by the long dashed lines with the anus inferiorly. Solid lines and circles show the area that is excised. Arrows indicate the direction of flap mobilization. Diagonal lines show the region that is typically mobilized/undermined for the cleft lift.

Please cite this article as: J. Grabowski, T.A. Oyetunji, A.B. Goldin, et al., The management of pilonidal disease: A systematic review, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.02.055

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and less pain with VAAPS, and no difference in recurrence rates at 1 year. 2.3.3.1. Recommendations. The initial surgical approach to PD with an abscess or draining sinus should be at least incision and drainage. For patients with an abscess, adjunctive curettage and/or minimal surgical procedure should be strongly considered because of both lower recurrence rates and faster healing time than I&D alone. A minimal surgical procedure such as the Gips/sinusectomy (or MIPE) has at least equivalent recurrence rates compared with classic excisional approaches, and is a reasonable first surgical option for patients with or without an abscess. Level 2–4 evidence; grade B recommendation. 2.3.4. Excision Removal of affected tissue with en bloc resection, followed either by primary closure or healing by secondary intention, has been the most common definitive surgical treatment of PD for decades, but is associated with longer healing time as well as greater pain and disability than less radical procedures [60]. Despite the heterogenous findings in the literature, there is level 1 and 2 evidence regarding several key aspects of the excisional approach. A summary of all excision-related level 1 and 2 evidence is provided in Table 3, and evidence supporting the key comparisons in surgical management of pilonidal disease is provided in Table 4. 2.3.4.1. Primary closure versus healing by secondary intention. A Cochrane review showed that after wide excision, open healing by secondary intention reduces the risk of recurrence by 35% when compared with any closed method, though SSI rates are not different and healing time

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is faster with primary closure [61]. However, a more recent, large systematic review (the German national guidelines) found that open healing was inferior to off-midline primary closure (e.g. Karydakis flap) [60]. This study cited two RCTs published after the Cochrane review that found the off-midline Karydakis flap closure and Limberg rotational flap (Fig. 2) had lower recurrence, shorter healing time, and fewer complications than open healing [62,63]. There is also level 1 evidence that less radical excision (sinusectomy, sinotomy, or unroofing) with open healing results in less time off work and less associated pain than radical excision with open healing with no difference in recurrence (Tables 3-5, see question 3). Conservative excision/sinusectomy/unroofing with open healing represents a quarter of the recurrence risk compared to wide excision with primary closure (relative risk 0.27; 95% CI, 0.11–0.63) [64]. Marsupialization of the wound after excision may be done in order to reduce the size of the wound and is associated with low recurrence rates, but level 2 evidence suggests it is associated with prolonged healing times and poor cosmetic results compared with other types of closure, and does not effectively ameliorate the drawbacks of open healing [60,65–67]. 2.3.4.2. Midline versus off-midline closure. As summarized in Table 4, there is general consensus amongst three level 1 studies, plus multiple systematic reviews, that midline closure is inferior to any type of offmidline closure (including flaps) in terms of SSI, healing time, wound dehiscence, and possibly recurrence rates, though not all data show significance and many included studies with high risk of bias [61,64,68]. Based on the included studies, the Cochrane review estimates that offmidline procedures are associated with significantly fewer recurrences

Table 3 Summary of all Level 1 evidence and high-quality systematic reviews that address the surgical management of pilonidal disease. Paper

Design

Patient age

Al-Khamis 2010

Meta-analysis of RCTs

N14 years N 1 year

Horwood 2012

Meta-analysis of RCTs Meta-analysis of RCTs

Adult

1–39 months 641

Adult

Variable

Enriquez-Navascues 2014

Follow-up time

Lee 2008

Systematic review

Variable

Variable

Steele 2013

Systematic review

Variable

Variable

Iesalnieks 2016

Systematic review

Variable

Variable

N

Comparisons (N patients)

Findings

2530

1. Primary closure versus secondary intention

1. Secondary intention recurrence is lower, healing time slower; no difference SSI or complications 2. Off-midline superior to midline for recurrence, healing time, SSI, and complications

2. Midline versus off midline closure Primary closure versus Limberg flap Limberg lower dehiscence, SSI; trend towards decreased recurrence (P = 0.07) 1. Limited excision less pain and time off work, but 2949 1. Open wide excision versus open recurrence rate and healing time not different limited excision (sinusectomy) or 2. Off-midline superior to midline for dehiscence, SSI unroofing (sinotomy) (153) 2. Midline closure versus off midline 3. Limberg v. Karydakis/Bascom: no difference in recurrence or complications closure 4. Sinusectomy/sinotomy recurrence rate lower; no 3. Advancing versus rotational flaps difference in other outcomes 4. Sinusectomy/sinotomy versus primary closure 81 Various surgical approaches 1. Treatment should be tailored to individual patients studies 2. Incision, curettage, partial lateral wall excision or marsupialization for initial surgery 3. Rhomboid flap recommended for recurrent disease – lower recurrence and complications than other approaches 1. Abscess should have only I&D regardless of 83 1. Acute disease acuity/recurrence studies 2. Chronic disease 2. Multiple options are acceptable 3. Flap procedures 3. Flaps may be most useful after failure of less invasive 4. Recurrent disease methods; no flap favored 4. Treatment should be individualized 127 General approach to pilonidal disease Highest-level evidence and consensus for: studies 1. Open wound healing: longer healing time and delayed return to work as compared to off-midline procedures, but easy and should remain a standard procedure 2. Off-midline procedures: lower recurrence than midline closure; midline closure should be abandoned 3. No outcomes differences between Karydakis flap, cleft lift, and Limberg flap

Please cite this article as: J. Grabowski, T.A. Oyetunji, A.B. Goldin, et al., The management of pilonidal disease: A systematic review, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.02.055

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J. Grabowski et al. / Journal of Pediatric Surgery xxx (xxxx) xxx

Table 4 Summary of Level 1 and 2 evidence for key comparisons in the surgical management of pilonidal disease. Comparison

Studies (N)

Level of Primary Outcomes Evidence

Summary

Limitations/caveats

Wide excision with primary closure (all types) versus wide excision with open/secondary intention or marsupialization

Al-Khamis 2010 (1231–1729) Khattak 2016 (50) Keshvari 2015 (321) Yetişir 2005 (216)

1, 2

1.) No difference 2.) Secondary intention probably superior 3.) Primary closure superior

Keshvari 2015 showed Karydakis flap superior to secondary intention for recurrence, healing time, and other complications.

Midline versus off-midline closure (including flaps)

Wide excision with open healing versus conservative limited excision or unroofing (sinusectomy) with open healing Conservative limited excision or unroofing (sinusectomy) versus wide excision with primary closure Rhomboid or Limberg flap (rotational) versus Karydakis or Bascom flap (local advancing) Simple incision & drainage versus incision & drainage with curettage

Al-Khamis 2010 (541) Enriquez-Navascues 2014 (1363) Horwood 2012 (641) Sevinc 2016 (150) Khan 2013 (120) Berkem 2005 (34) Daglar 2004 (69)

1, 2

1.) SSI 2.) Recurrence 3.) Healing time

1.) 2.) 3.) 4.)

SSI Recurrence Healing time Wound dehiscence

1.) Off-midline superior 2.) Off-midline probably superior 3.) Off-midline superior 4.) Off-midline superior

Yetişir 2005 showed Limberg flap equivalent recurrence rates to marsupialization. Sevinc 2016 and Enriquez-Navascues 2014 suggest primary tension-free midline closure (no deep sutures) may have equivalent recurrence compared with off-midline closure/flap (and easier to perform). Daglar 2004 found Limberg flap had higher recurrence than primary midline closure but lower complication rate. High risk of bias in included studies.

Enriquez-Navascues 2014 (153)

1

1.) Healing time 2.) Recurrence

1.) No difference 2.) No difference

Enriquez-Navascues 2014 (482)

1

1.) Healing time 2.) Recurrence

1.) Primary closure superior High risk of bias in included studies. 2.) Conservative excision superior

Enriquez-Navascues 2014 (951) Gupta 2017 (50) Khan 2016 (180) Sevinc 2016 (150) Bali 2015 (71) Tokac 2015 (91) Vahedian 2005 (150)

1, 2

3.) SSI 4.) Recurrence 5.) Wound dehiscence

3.) No difference 4.) No difference 5.) No difference

2

1.) Recurrence 2.) Healing time

1. I&D with curettage superior 2. I&D with curettage superior

than midline closure (1.7% vs. 10.5%, N = 574); these authors and the German national guidelines advocate abandonment of midline closure due to wound dehiscence rates of up to 74% and recurrence rates of up to 45% [60,61]. One level 1 study, however, and one subsequent RCT conclude that primary tension-free midline closure with no deep suturing may have equivalent recurrence rates as compared with offmidline closures [64,69]. 2.3.4.2.1. Recommendations. Less radical excision may be more appropriate as first-line treatment than radical excision due to faster recovery. If wide excision is performed, healing by secondary intention results in lower recurrence rates compared with primary closure, but has a longer recovery time than primary closure. If primary closure is performed, it should be an off-midline or a tension-free midline closure. Level 1–2 evidence, grade B recommendation.

2.3.5. Flaps For the patient with chronic or multiply recurrent disease, or after failure of less radical treatment, excision of PD and flap coverage may be considered. The purpose of all flaps is to obliterate the pilonidal space and remove or elevate the gluteal cleft such that there is no midline crevice for hair and debris to accumulate. The most-studied flap closures are the rhomboid or Limberg rotational flap [70], the Karydakis flap [1,71], and the Bascom cleft lift procedure [72,73] (Fig. 2). The latter two are local advancement flaps that are less complex than the Limberg flap. There are many other less commonly used or reported flaps that

Khan 2016 and Gupta 2017 showed higher SSI after Karydakis flap versus Limberg flap.

are not supported by high-quality evidence and were excluded from this review (e.g. V-Y advancement flap, Z-plasty).

2.3.5.1. Rotational versus local advancement flaps. A total of 11 RCTs (one level 1 and multiple level 2 studies) have compared rotational (Limberg) to various advancement flaps (Table 4). In the level 1 study (which included 6 RCTs), no differences were found in SSI, recurrence, or wound dehiscence, but the Karydakis flap was associated with faster return to work [64]. The authors conclude that there is no significantly superior flap approach between the Bascom, Karydakis, and Limberg, and “the presence of lateral orifices may dictate the most appropriate type of flap to be used”. Two subsequent RCTs showed a higher rate of SSI after the Karydakis flap compared with the Limberg flap [74,75], which had been noted in one of the studies in the level 1 analysis [76]. A retrospective comparison of the Limberg with the Karydakis flap in 71 patients found no recurrences in either group at 28 months, but the Limberg flap was associated with lower complication rates, shorter length of hospital stay, earlier return to work, lower pain scores, and better complete healing duration [77]. Another study found no difference in recurrence at 2 years after Karydakis (6%) versus Limberg (6%) versus tension-free primary closure (4%) [69]. The Limberg flap is more technically difficult to perform than the Bascom flap [78,79]. Recurrence may be higher with the Limberg flap if it is not properly created off-midline, which is a common technical mistake [80]. Modification of the Limberg flap, with increased

Please cite this article as: J. Grabowski, T.A. Oyetunji, A.B. Goldin, et al., The management of pilonidal disease: A systematic review, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.02.055

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Table 5 Summary of time to return to work/school (RTW), quality of life (QOL), and/or patient satisfaction data (usually secondary outcomes) from Level 1 and 2 studies, examining key comparisons in the management of pilonidal disease. Comparison

Studies (N)

Level of Outcomes Evidence

Summary

Limitations/caveats

Wide excision with primary closure (all types) versus wide excision with open/secondary intention or marsupialization

Al-Khamis 2010 (83–1729) Khattak 2016 (50) Keshvari 2015 (321) Yetişir 2005 (216)

1, 2

1.) Faster RTW with primary closure 2.) Lower satisfaction with open healing 3.) Off-midline primary closure superior

Studies with high risk of bias; RTW not treated as time-to-event data

1.) Time to RTW 2.) Patient satisfaction 3.) QOL

1.) Time to RTW 2.) Patient satisfaction

1.) Conflicting data 2.) Off-midline possibly superior

Midline versus off-midline closure (including flaps)

Al-Khamis 2010 (146) Enriquez-Navascues 2014 (598) Horwood 2012 (641) Khan 2013 (120)

1, 2

Wide excision with open healing versus conservative limited excision or unroofing (sinusectomy) with open healing Rhomboid or Limberg flap (rotational) versus Karydakis or Bascom flap (local advancing)

Enriquez-Navascues 2014 (103)

1

1.) Time to RTW 2.) Patient satisfaction

1.) Limited excision superior 2.) Limited excision superior

Enriquez-Navascues 2014 (540) Bali 2015 (71) Tokac 2015 (91)

1, 2

1.) Time to RTW 2.) Patient satisfaction 3.) QOL

1.) Conflicting data 2.) Advancement flap possibly superior 3.) No difference

Keshvari 2015 showed RTW 10 days faster with Karydakis flap than secondary intention. Yetişir 2005 showed Limberg flap RTW faster and better satisfaction than marsupialization. Khan 2013 showed faster RTW with Limberg flap. Only one RCT showed better satisfaction with off-midline [97]. Another showed no difference in satisfaction [98]. High risk of bias. Only one RCT examined patient satisfaction [99].

Bali 2015 and Tokac 2015 showed shorter RTW with Limberg flap; 3 earlier RCT's showed shorter RTW with Karydakis flap [75, 94, 100]. Two RCTs showed better satisfaction with Karydakis flap [94,95]; 1 RCT showed equivalent satisfaction [100]. Only one RCT assessed QOL using Short Form 36 score; advancement flap patients had better mobility at postoperative day 10 but otherwise there was no difference with Limberg flap.

Simple incision & drainage versus incision & drainage with curettage Phenol versus open wide excision

Vahedian 2005 (150)

2

1.) Time to RTW

1.) No difference

Calikoglu 2017 (140)

2

1.) Time to RTW 2.) QOL

1.) Phenol superior 2.) Phenol superior

lateralization of the flap off-midline, may have lower complication rates based on level 2–4 evidence [81–83]; there are reports in the teenage population of low complication rates with use of the modified Limberg flap compared with standard excision, though follow-up time is short [84,85]. 2.3.5.1.1. Recommendations. Based on surgeon availability, preference, and experience, any of the three most commonly used flaps are a reasonable choice for coverage after excision of PD. The Limberg flap is more technically challenging than advancement flaps, the modified Limberg flap may have better outcomes than the classic Limberg flap, and the Karydakis flap may have a higher rate of SSI. Level 1–2 evidence, Grade B recommendation. 2.4. What operative and non-operative approaches for the treatment of pilonidal disease are associated with the highest quality-of-life and/or patient satisfaction? For this question, papers that assessed quality of life (QOL), patient satisfaction scores, or time to return to work or school (RTW) were included. Only a small number of studies addressed these issues, and they were typically included as secondary outcomes. Twenty-seven papers met inclusion criteria. The most common outcome reported was time to return to work or school. Most studies used informal means to assess these outcomes, though a few used validated patient satisfaction or QOL tools, which included the 36-Item Short Form Health Survey (SF-

Short Form 36 and Nottingham Health Profile scores at 3 weeks after surgery favored phenol injection.

36), the Cardiff Wound Impact Schedule, or the Nottingham Health Profile [86–88]. There were 3 level 1 studies, 9 additional RCT's, and 15 level 3 or 4 studies. The highest-level data is summarized in Table 5. 2.4.1. Wide excisional versus limited excisional, minimal surgical approaches, or non-operative approaches The level 1 Cochrane review of several RCT's, in addition to several subsequent RCT's, show that wide excision with any kind of primary closure is preferred by patients over healing by secondary intention, although the data show the biggest advantage with off-midline closures/ flaps compared with open healing (Table 5) [62,66,89]. There is a paucity of high-level evidence comparing patient satisfaction after wide excision versus non-operative or minimal surgical approaches. One level 1 study that included 2 RCT's measured time to RTW, and 1 RCT that assessed patient satisfaction, for the comparison of sinusectomy or unroofing versus en bloc excision. The less invasive technique was favored for both outcomes (Table 5) [64]. Multiple non-randomized studies tended to favor less invasive techniques over wide excisional procedures in terms of loss of productivity and patient satisfaction. A questionnaire-based study examining satisfaction in 80 patients after sinotomy compared to wide excision found much shorter times to routine activity with sinotomy [90]. A retrospective examination of the minimal incision (Gips) technique in a cohort of 3407 military officers found that minimal excision was associated with an average of 13 fewer sick days compared to other techniques [91]. In

Please cite this article as: J. Grabowski, T.A. Oyetunji, A.B. Goldin, et al., The management of pilonidal disease: A systematic review, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.02.055

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Table 6 Interpretation of the literature regarding outcomes of various operative approaches as definitive management for pilonidal disease. RTW – return to work/school.

Sinusectomy, Gips/MIPE, or other minimal approach (adjunct to I&D for abscess if present) Excision & leave open Excision & midline closure Bascom cleft lift Limberg/rhomboid flap Karydakis flap

Wound complications

Recurrence

Time to RTW

Patient satisfaction or QOL

Simplicity

Comments

Excellent

Moderate

Excellent

Excellent

Excellent

*BEST INITIAL APPROACH*

Excellent Poor Moderate Moderate Moderate

Excellent Poor Excellent Excellent Excellent

Poor Moderate Moderate Moderate Moderate

Poor Moderate Moderate Moderate Moderate

Excellent Moderate Moderate Poor Moderate

contrast, a study of 583 military officers evaluated long-term patient satisfaction after different open techniques; higher satisfaction was seen if recurrence-free, regardless of type of surgery [92]. One retrospective review of 134 pediatric patients compared laser epilation to either surgical excision or simple observation [10]. There was no difference in patient satisfaction or QOL amongst the three arms. Another study compared 17 patients who underwent laser pilonidal sinus ablation versus 23 patients who received the traditional excision techniques [93]. Patients who had the laser ablation therapy returned to work 2.1 days earlier. They also reported the ability to sit without discomfort 1 day earlier. Two RCT's compared phenol versus excision. Using a non-validated questionnaire, a small trial of 40 patients noted that length of stay, time off work, time to walking, and cosmetic satisfaction were all better with phenol treatment [94]. As discussed above, a large trial of 140 patients favored phenol to excision with open healing; this same trial also used the Short Form 36 Health Survey and Nottingham Health Profile to

Abscess

analyze QOL outcomes [18]. The study reported that QOL was better after phenol using both validated assessments. 2.4.2. Flaps Despite multiple RCT's involving reconstructive flap procedures in the management of pilonidal disease, there is a very limited amount of strong evidence regarding time to RTW, patient satisfaction, and quality of life. As shown in Table 5, the data are conflicted when comparing primary closure to off-midline (flap) repair, and when comparing local advancement to rotational flaps. As with surgical outcomes, no type of flap repair is definitively superior to any other flap with regard to all of these secondary outcomes, though patient satisfaction rates and cosmesis were better after the Karydakis flap compared with the Limberg flap in two RCTs [95,96]. An RCT of 91 patients found the Karydakis and Limberg flaps to be equivalent, except for faster return to work and better cosmetic results (using a visual analog scale) after the Limberg flap [97]. Additionally, a

Pilonidal pits

Asymptomatic

Drainage Bleeding, pain

Minimally invasive intervention Incision & drainage

Observe Epilation, hygiene

Gips, sinusectomy Curettage, pit-picking Phenol

Recurrent or persistent disease

Failure of conservative treatment (including limited excision/sinusectomy)

OPTION 1

Off-midline closure Karydakis, Limberg, Bascom cleft lift

Wide / en bloc excision

OPTION 2

Tension-free midline closure OPTION 3

Open/secondary healing Fig. 3. Pilonidal disease clinical practice guideline.

Please cite this article as: J. Grabowski, T.A. Oyetunji, A.B. Goldin, et al., The management of pilonidal disease: A systematic review, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.02.055

J. Grabowski et al. / Journal of Pediatric Surgery xxx (xxxx) xxx

retrospective study comparing the Karydakis to the Limberg flap found better cosmetic results with the Karydakis flap [81]. Time off from work in the modified Limberg group (12.9 days) was significantly shorter (20 days with Karydakis and 22.5 days with classic Limberg, P b 0.05). Time to walk and sit on the toilet painlessly were shorter in the modified Limberg group (P b 0.0001). 2.4.2.1. Recommendations. Less invasive procedures are generally preferred by patients over wide excision in terms of time to return to work, overall satisfaction, and quality of life. Wound closure, ideally off-midline, is preferred over healing by secondary intention. No flap closure is definitively better than any other. Level 1–4 evidence; grade C recommendation.

3. Discussion This systematic review of the existing pilonidal disease literature has revealed a definitive recent trend towards less invasive management, with unacceptably high failure rates seen after traditional wide excision. Several new and promising techniques are available for the practicing surgeon. Based on the strongest available evidence, a summary of the advantages and disadvantage inherent to each surgical approach is provided in Table 6. Additionally, a management algorithm for pilonidal disease was constructed (Fig. 3). This simple guideline is designed for the care of patients with both acute and chronic disease. Of note, this guideline reflects the conclusion from this review that wide excision should no longer be the management of choice except possibly in the circumstance of persistently recurrent or chronic disease. Several significant limitations in the literature were noted during the review process, including extreme variability in primary outcome reporting between studies, many studies with missing details regarding specific surgical technique, poorly controlled patient selection and indications for intervention, and short follow-up time. Very few studies assessed essential secondary outcomes such as loss of productivity and quality of life. The lack of a validated standard tool to assess or report severity of pilonidal disease also hampers interpretation of the success of intervention. While many of the surgical procedures had highlevel evidence supporting their use, newer and less invasive approaches were generally supported by less robust evidence. Another limitation in pilonidal research is a still-poor understanding of the cause of the disease; a thorough contemporary analysis of possible etiologies and risk factors could inform clinical management. Furthermore, there exists a paucity of literature specific to pediatric patients or that address concerns particular to this population, such as how to best manage initial presentations or abscess presentations. Prospective studies need to focus on long-term outcomes and complications of minimally invasive excisional techniques in the pediatric and adolescent population. Such work would be aided considerably by the development of standardized severity and outcomes reporting for pilonidal disease.

References [1] Karydakis GE. Easy and successful treatment of pilonidal sinus after explanation of its causative process. Aust N Z J Surg 1992;62:385–9. [2] Lee PJ, Raniga S, Biyani DK, et al. Sacrococcygeal pilonidal disease. Colorectal Dis 2008;10:639–50 discussion 651-632. [3] Nasr A, Ein SH. A pediatric surgeon's 35-year experience with pilonidal disease in a Canadian children's hospital. Can J Surg 2011;54:39–42. [4] Nesselrod JP. Pilonidal disease; a plea for simplification of its surgical management. Q Bull Northwest Univ Med Sch 1946;20:407–10. [5] Fitzpatrick EB, Chesley PM, Oguntoye MO, et al. Pilonidal disease outcomes following surgery in a military population: how far have we really come? J Surg Res 2013;179. [6] Moher D, Liberati A, Tetzlaff J, et al. Preferred reporting items for systematic reviews and meta-analyses: the PRISMA statement. PLoS Med 2009;6:e1000097. [7] Armstrong JH, Barcia PJ. Pilonidal sinus disease. The conservative approach. Arch Surg 1994;129:914–7 discussion 917-919.

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Please cite this article as: J. Grabowski, T.A. Oyetunji, A.B. Goldin, et al., The management of pilonidal disease: A systematic review, Journal of Pediatric Surgery, https://doi.org/10.1016/j.jpedsurg.2019.02.055