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Review

Recurrence Rates of Pilonidal Sinus Disease Are High in Children and Recurrences Occur Exceedingly Early

by
Christina Oetzmann von Sochaczewski
1,* and
Dietrich Doll
2
1
Chirurgische Klinik, Universitätsklinikum Bonn, 53127 Bonn, Germany
2
Abteilung für Proktochirurgie und Pilonidalsinus, St. Marienhospital Vechta, 49377 Vechta, Germany
*
Author to whom correspondence should be addressed.
Surgeries 2024, 5(3), 726-737; https://doi.org/10.3390/surgeries5030057
Submission received: 5 July 2024 / Revised: 27 July 2024 / Accepted: 2 August 2024 / Published: 19 August 2024
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Abstract

:
Pilonidal sinus disease increasingly affects children and adolescents. While recurrence rates in adults are lower, with a minimum of 2% per year, and are essentially a function of the surgical approach over time, this is totally different in children. Many studies investigating children and adolescents report much higher recurrence rates. We aimed to gather and concentrate the available data on this matter in our review by searching PubMed, MEDLINE, and Scopus. The crude median recurrence rate of pilonidal sinus disease in children and adolescents was 13% (interquartile range, 7–20%) in our sample of 3599 patients in 42 studies, with a median study size of 52 (interquartile range, 27–96 patients) patients. Time to recurrence was available from eight studies with 1418 patients and its mean or median time to recurrence was usually much less than one year. For both parameters, an association with study size or patient age was not evident. Although treatment algorithms have been developed to adapt treatment to this dire situation, relevant improvements can only be expected if the underlying factors that cause these recurrence dynamics in children, which are substantially different from those in adults, were to be uncovered.

1. Introduction

A long standing belief in surgery was that pilonidal sinus disease affects primarily adults and is scarce in children and adolescents, reflected by the paucity of studies in the literature on this disease in the latter age groups even in the first decade of the 21st century [1]. However, pilonidal sinus disease, even in infants, is well documented in the past [2] and present [3] literature. Recent studies demonstrated a rising incidence of pilonidal sinus disease on both local [4,5,6,7] and global [8,9] levels to twice the rates reported almost 30 years ago [10]. Pilonidal sinus disease has a relevant negative impact on the daily activities of young people [11], and the proportion of patients that have been previously treated for pilonidal sinus disease exceeds 20% in many series [12,13,14,15,16], up to 41% in one study that included consecutive patients [17], indicating a relevant burden of disease due to recurrences.
In adults, large meta-analyses established thresholds for recurrence rates of 2% per year for some surgical techniques, which fared better than others [8,18,19]. This research demonstrated that recurrence in adults is essentially a function of surgical approach and time. The overall recurrence rate in adults is still high given the young age of the affected population: while the burden of disease of pilonidal sinus disease may exceed that of inguinal hernias in some age groups [18], recurrence rates are lower in inguinal hernia repair [20], although the affected population is often of older age. This is substantially different in children and adolescents, as Maasewerd et al. [21] pointed out first that recurrences in these age groups occur exceedingly early compared to adults. The issue of early recurrences in children and adolescents had not been addressed at all in systematic reviews [22,23] and had only been mentioned once before because the causative factors of this substantial difference in time to recurrence compared to adults remain unknown [16].
This prompted us to curate the currently available evidence in a narrative on both increased recurrence rates and time to recurrence in children and adolescents in order to raise awareness of this important aspect for patient care in the surgical community. We therefore searched PubMed, MEDLINE, and Scopus for studies of pilonidal sinus disease in children or adolescents that reported a recurrence rate or a time to recurrence irrespective of the employed surgical technique without time or language restrictions. We also snowballed the reference lists of included studies for additional studies that might have escaped our database search.

2. High Recurrence Rates in Children and Adolescents

Recurrence rates in children and adolescents varied highly between studies and ranged from 0% to 42% (Table 1). We did not separate the results by surgical approach because the large studies did not consistently identify an advantage for any technique [12,14,16,21], which is in line with the results from a systematic review [22]. Only cohorts for whom complete summary statistics were available were included in the review because reporting only a subgroup might have biased the results even more. Thus, some relevant studies [17,24,25,26,27] had to be excluded, and we included 42 studies with 3599 patients spanning 22 years in the literature (Table 1).
Of the included 3599 patients, 524 (15%) developed a recurrence (Table 1), and the median crude recurrence rate was 13% (interquartile range, 7–20 percent). The median study size was 52 patients (interquartile range, 27–96 patients) (Figure 1).
Although the recurrence rates span the whole spectrum from 0% to 42%, an obvious relationship between study size and crude recurrence rate cannot be deduced (Figure 2). This is also true for the relationship between patient age and crude recurrence rate (Figure 2).
We noted a considerable growth in research interest in pilonidal sinus disease in recent years, as 62% (26/42) of the studies were published within the last six years, and 67% (28/42) of the manuscripts were published by just five journals (Figure 3).

3. Recurrences in Children and Adolescents Occur Very Early Compared to Adults

Provisioning a concise summary of time to recurrence is even more difficult, as this information is often not provided within the manuscript. Nevertheless, the available data demonstrated that recurrences of pilonidal sinus disease in children and adolescents occur exceedingly early compared to what could be expected from the adult data (Table 2).
We were able to include information from just eight studies with 1418 patients (39% of the total number of patients included in the estimation of the crude recurrence rate), who had 190 recurrences (36% of the total recurrences included in the estimation of the crude recurrence rate). Summary statistics for this result could not be calculated because the different reported statistics could not be summarised. However, additional studies that could not be included in our study due to statistics that were only reported for subgroups, a lack of variability measures, or the reporting of only Kaplan–Meier plots indicate the same result: the majority of recurrences occur in the first postoperative year [14,15,26,27,34,44,61,63,64], while studies that report longer median times to recurrence were scarce [50,58,62].

4. Discussion

Pilonidal sinus disease in children and adolescents had long been out of the focus of the surgical community [1] because it collectively believed that this disease seldom occurs in these age groups. Even when this disease gained more attention, the primary focus of research studies was the identification of the best surgical technique to treat it. When systematic reviews addressed this question, the researchers found that all techniques led to equally poor results in children and adolescents [22,23]. Compared to the results in adults, which are better than those in children in terms of recurrence [18,19], the recurrence rates of pilonidal sinus disease in adults are still high compared to other diseases that require surgery, such as inguinal hernia [20]. This resulted in a substantial shift in the treatment approach: while it had been previously assumed that the surgical treatment approach in children should follow that in adults [65], this has changed substantially. Due to the expected increase in recurrence rate in paediatric age groups, minimally invasive techniques conducted in-office or single-day surgeries are preferred. These procedures result in the smallest tissue disruption and cause the least impact on the daily life activities of patients [23,66,67]. This aspect is also of importance because the extent of tissue disruption due to repeated surgeries has been demonstrated to reduce the success rates of subsequent surgeries [68]. The old surgical adage that if there are multiple procedures available to treat a disease, none is ideal [69] fits perfectly with paediatric pilonidal sinus disease. A substantial improvement in the treatment of adolescents and children with pilonidal sinus disease is the long-awaited trial on postoperative laser hair depilation [70] that demonstrated a relevant reduction in recurrence rates due to the intervention, but recurrence rates remain high [71].
In addition to the issue of the ideal surgical approach, the underlying factors that cause this difference in recurrence dynamics remain unknown. Although many theories were developed in the last century to put forward a congenital origin of pilonidal sinus disease [72,73,74,75,76], the universal belief now is that pilonidal sinus disease is an acquired disease. It is assumed to be caused by sharp hair fragments that bury themselves in the tissue, resulting in an inflammatory reaction, which forms the basis of disease [77]. Hormonal factors were investigated but were shown to be irrelevant [78]. Although this idea was brought up again recently in the context of polycystic ovary syndrome with its characteristic increased levels of androgens [79], it remains doubtful if this is the explanatory variable. Otherwise, one would expect a significant number of female infants with adrenogenital syndrome to be affected due to the androgen excess. However, there are no reports in the literature on this condition, which precludes the assumption that this might be the holy grail of the pathogenesis.
Another factor that was investigated as a potential factor affecting recurrence is obesity, which was first suggested to be of relevance in a study of 14 patients, of which 6 were overweight, 2 were obese, and 6 were non-overweight; this prompted the authors to conclude that a high body mass index might be a risk factor for pilonidal sinus disease [61]. Another study in 86 patients demonstrated that recurrences were higher in the 27 patients that were overweight compared to the non-overweight patients [35]. Finally, in a multivariable study including 307 patients, the patients that were overweight had a 67% higher chance of experiencing a recurrence compared to the non-overweight patients [14]. By contrast, there are nine studies that found no association between body mass index and recurrence [21,26,29,30,31,37,45,51,64]. Although one cannot base the interpretation of relevance of a risk factor solely on the number of studies or included patients, one may expect contrary findings to a certain extent due to the underlying probability distribution. Nonetheless, the evidence is more compatible with the assumption that an increased body mass index is not associated with recurrences. The potential role of smoking in recurrences has not been investigated so far, but despite the lack of data, 10% of respondents in a Turkish survey of paediatric surgeons believe that smoking is a causative factor in pilonidal sinus disease recurrence [80]. It has only been demonstrated that there is an association between smoking and hiradenitis suppurativa [81,82], which is, in turn, associated with pilonidal sinus disease [83].
Another potential issue of paediatric pilonidal sinus disease could be the development of a pilonidal sinus disease carcinoma due to the long-standing disease. However, a recent analysis of studies in the literature revealed that although the latency from the onset of pilonidal sinus disease to the diagnosis of a pilonidal sinus carcinoma is 20 years [84], the median patient age is 56 years [85], indicating no relationship between pilonidal sinus disease onset as a child or adolescent and its progression towards a pilonidal sinus disease carcinoma. This is supported by the finding that in recent years, the interval between pilonidal sinus disease onset and diagnosis of a pilonidal sinus disease carcinoma was even shorter, at just 6.9 years [86].
One might assume that higher recurrence rates in children and adolescents might be caused by a higher parental awareness of recurrences compared to adults, who might adapt to living with a chronic pilonidal sinus. It is, however, unclear whether that might be a relevant aspect or if the recurrence rate in adults is underestimated: in a recent survey of adult pilonidal sinus surgeons in the United Kingdom, 29% of respondents assumed their personal recurrence rate to be between 16% and 30% [87], and another 37% of surgeons assumed their personal recurrence rate to be between 6% and 15% [87]. This is interesting in so far as the same respondents stated that recurrent disease accounted for 20% of all cases [87]. Results from a survey conducted in Austria and Switzerland, in which 19% of pilonidal sinus surgeries were reported to be performed for recurrent disease, is in agreement with aforementioned results [88]. These differences might be caused by adults seeking treatment at a different clinic for recurrent pilonidal sinus disease, which has been observed as a typical pattern in inguinal hernia surgery [89]. This might be more difficult for children and adolescents, given the rarity of paediatric surgical departments compared to general surgery departments. Moreover, the only report comparing results in children and adolescents reported similar crude recurrence rates [90], although this report is likely to be biased by the vast majority of adults being treated with primary closure, which should be avoided in adults due to a high recurrence rate [91,92,93].
A specific issue in the treatment of children is the negative impact of pilonidal sinus disease on school attendance, which affects a relevant portion of patients [11,94]. Closely linked to the affected children’s absence from school is the impairment of a parent’s ability to attend work if their children need additional care regardless of whether the absence is caused by a more extensive resection and necessary postoperative wound care or by recurrent clinic visits for repeated minimally invasive interventions. Apart from work attendance, a chronic disease of a child inevitably has negative impacts on the family and parents in several domains of life [95]. Chronic illness is irksome for the affected children, and it is well known that they may tend to deny being chronically ill and stop taking their medicaments in order to avoid being confronted with their chronic illness [96]. Although the necessity for recurrent clinic visits for follow-ups during a minimally invasive treatment protocol [97,98,99] or multiple laser depilation sessions as adjunctive care [67,100] might not be equivalent to taking daily medication, it is still a visible and notable sign of being chronically ill. Although it has been demonstrated that social support is available and much more prevalent than the affected patients fear [101], the presumed negative effect for patients is still present and has relevant consequences in their daily lives, as they tend to abstain from their daily life activities [11]. Consequently, adherence to therapy is affected by multiple factors, including social support, organisational strategy, and behavioural and motivational interventions, which work by increasing the patient’s resilience [102]. When these interventions were included in a structured treatment protocol for pilonidal sinus disease, they increased patients’ resilience, and those with a higher resilience were less likely to experience recurrent disease [103]. In addition to the psychological negative impact of a chronic disease on children, adolescents, and their parents, the school absence of patients and subsequent work absenteeism of their parents have relevant negative financial impacts on the families [104]. Chronic illness due to recurrent disease with necessary re-interventions increases this financial burden owing to higher insurance prices or, even worse, not being eligible for health insurance, which negatively affects adherence to therapy [105], except in the countries that have statutory health insurance covering the whole population [106].
Taken together, children and adolescents affected by pilonidal sinus disease have higher recurrence rates when compared to adults. Moreover, their recurrences occur exceedingly early, usually within the first year after primary surgery. As the surgical community is currently unaware of the causative factors of these differences in recurrence dynamics, the adjustments in treatment algorithms are just an adaptation to this different situation, but not a solution to reduce the burden of disease due to recurrences. This might only be achieved if we were able to identify additional causative factors in order to be able to reduce the negative impact on children’s and adolescents’ daily life activities in their formative phases of life. Moreover, the specific needs of patients affected by recurrent disease needs to be addressed beyond surgical care in order to minimise the negative effects of recurrent disease on the lives of these patients.

Author Contributions

Conceptualization, C.O.v.S.; methodology, C.O.v.S. and D.D.; writing—original draft preparation, C.O.v.S.; writing—review and editing, D.D. All authors have read and agreed to the published version of the manuscript.

Funding

This research received no external funding.

Institutional Review Board Statement

Not applicable.

Informed Consent Statement

Not applicable.

Data Availability Statement

The original contributions presented in this study are included in the article; further inquiries can be directed to the corresponding author.

Conflicts of Interest

The authors declare no conflicts of interest.

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Figure 1. Case numbers per year and study.
Figure 1. Case numbers per year and study.
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Figure 2. Crude recurrence rates considering patient age and case numbers [1,12,13,14,15,16,21,28,29,30,31,32,33,34,35,36,37,38,39,40,41,42,43,44,45,46,47,48,49,50,51,52,53,54,55,56,57,58,59,60,61,62].
Figure 2. Crude recurrence rates considering patient age and case numbers [1,12,13,14,15,16,21,28,29,30,31,32,33,34,35,36,37,38,39,40,41,42,43,44,45,46,47,48,49,50,51,52,53,54,55,56,57,58,59,60,61,62].
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Figure 3. Publication year and venue of the included studies.
Figure 3. Publication year and venue of the included studies.
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Table 1. Crude recurrence rates by study irrespective of the surgical technique used, considering the cohort’s age.
Table 1. Crude recurrence rates by study irrespective of the surgical technique used, considering the cohort’s age.
StudyYearCasesCrude Recurrence RateAgeMeasure of Variability
[28]20245658% (n = 46)16.2 *8.9–18 ~
[29]20246325% (n = 16)15.5 #1.6 § (12–18) ~
[21]202321316% (n = 34)14.1 *3.7–15.6 $
[30]20232945% (n = 14)14 *10–18 ~
[16]202210616% (n = 17)16 *15–16 $
[31]20229914% (n = 16)16 *8–19 ~
[32]20223196% (n = 19)15.5 #1.4 §
[33]20222015% (n = 3)15.6 #1.64 §
[34]20211916% (n = 3)16.4 #2.1 § (12.5–19.9) ~
[35]20218616% (n = 14)15.1 #1.29 §
[36]2021490%15.7 #1.6 § (14.7–16.7) $
[37]20212928% (n = 8)15.5 #2.8 §
[38]20211712% (n = 2)17.1 *12.5–18 ~
[39]20218519% (n = 16)14.7 #1.9 §
[40]2021549% (n = 5)15 #12–17 ~
[41]20212821% (n = 6)15.6 #13–17 ~
[15]20201059% (n = 9)15.6 #10–19 ~
[42]20206020% (n = 12)14.2 #1.3 § (9–17) ~
[43]2019186% (n = 1)16 *15–17 ~
[14]201830733% (n = 102)16 *15–17 $
[44]20188418% (n = 15)16.2 *12.1–17.9 ~
[45]20184723% (n = 11)15.6 #1.2 §
[46]20184312% (n = 5)15 #1.4 §
[47]20181179% (n = 11)15.6 #12–20 ~
[48]2018229% (n = 2)16 *11–18 ~
[49]20182688% (n = 21)16 *12–18 ~
[50]20174319% (n = 8)16 *14–17 $
[51]20166042% (n = 25)15 #13–20 ~
[13]20161921% (n = 4)15 *13.8–15.8 $
[52]20154115% (n = 6)15 *12–16 ~
[53]2015250%16.1 #1.2 §
[54]2014408% (n = 3)15.2 #1.3 § (12–17) ~
[55]2013150%14 *12–18 ~
[12]201112022% (n = 26)14.9 #2.9 §
[1]201112120% (n = 24)15 #12–19 ~
[56]20112020% (n = 4)13.3 #12–15 ~
[57]20117011% (n = 8)16 #1.7 §
[58]2009542% (n = 1)16 #1.9 § (13–19) ~
[59]2008260%16.7 #14–19 ~
[60]2005687% (n = 5)13.8 #4.4 §
[61]2005147% (n = 1)15.4 §1.6 § (13–18) ~
[62]2002343% (n = 1)16.4 §13–18 ~
*, median age; #, mean age; §, standard deviation; $, interquartile range; ~, range.
Table 2. Time to recurrence in paediatric pilonidal sinus disease.
Table 2. Time to recurrence in paediatric pilonidal sinus disease.
StudyCasesCrude Recurrence RateTime to RecurrenceMeasure of Variability
[28]5658% (n = 46)9.3 * months1–41 ~ months
[29]6325% (n = 16)4.1 * months1–45 ~ months
[21]21316% (n = 34)5.8 * months 4–10 & months
[31]9914% (n = 16)14 # months10 § months
[37]2928% (n = 8)5.8 # months2.8 § months
[49]2688% (n = 21)5 * months2–28 ~ months
[51]6042% (n = 25)8 # months1–36 ~ months
[1]12122% (n = 24)6.5 # months1–13 ~ months
*, median time to recurrence; #, mean time to recurrence; §, standard deviation; ~, range; &, confidence interval.
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Oetzmann von Sochaczewski, C.; Doll, D. Recurrence Rates of Pilonidal Sinus Disease Are High in Children and Recurrences Occur Exceedingly Early. Surgeries 2024, 5, 726-737. https://doi.org/10.3390/surgeries5030057

AMA Style

Oetzmann von Sochaczewski C, Doll D. Recurrence Rates of Pilonidal Sinus Disease Are High in Children and Recurrences Occur Exceedingly Early. Surgeries. 2024; 5(3):726-737. https://doi.org/10.3390/surgeries5030057

Chicago/Turabian Style

Oetzmann von Sochaczewski, Christina, and Dietrich Doll. 2024. "Recurrence Rates of Pilonidal Sinus Disease Are High in Children and Recurrences Occur Exceedingly Early" Surgeries 5, no. 3: 726-737. https://doi.org/10.3390/surgeries5030057

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