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Major stoma related morbidity in young children following stoma formation and closure: A retrospective cohort study

Open AccessPublished:November 27, 2021DOI:https://doi.org/10.1016/j.jpedsurg.2021.11.021

      Highlights

      • What is currently known about this topic?.
      • Altough sometimes necessary, stomas can lead to increased morbidity.
      • What new information is contained in this article?.
      • Major stoma related morbidity (Clavien-Dindo ≥III) occurs in approximately 25% following stoma formation and closure each. Taking into account both operations, 39% experiences a major stoma related complication. Patients with an ileostomy are significantly most at risk.

      Abstract

      Background

      Little is known about stoma related morbidity in young children. Therefore, the aim of this study is to assess major morbidity after stoma formation and stoma closure and its associated risk factors.

      Methods

      All consecutive young children (age ≤ three years) who received a stoma between 1998 and 2018 at our tertiary referral center were retrospectively included. The incidence of major stoma related morbidity (Clavien-Dindo grade ≥III) was the primary outcome. This was separately analysed for stoma formation alone, stoma closure alone and all stoma interventions combined. Non-stoma related morbidity was excluded. Risk factors for major morbidity were identified using multivariable logistic regression analysis.

      Results

      In total 336 young children were included with a median follow-up of 6 (IQR:2–11) years. Of these young children, 5% (n = 17/336) received a jejunostomy, 57% (n = 192/336) an ileostomy, and 38% (n = 127/336) a colostomy. Following stoma formation, 27% (n = 92/336) of the young children experienced major stoma related morbidity, mainly consisting of high output stoma, prolapse and stoma stenosis. The major morbidity rate was 23% (n = 66/292) following stoma closure, most commonly comprising anastomotic leakage/stenosis, incisional hernia and adhesive obstructions. For combined stoma interventions, major stoma related morbidity was 39% (n = 130/336). Ileostomy was independently associated with a higher risk of developing major morbidity following stoma formation (OR:2.5; 95%-CI:1.3–4.7) as well as following closure (OR:2.7; 95%-CI:1.3–5.8).

      Conclusions

      Major stoma related morbidity is a frequent and severe clinical problem in young children, both after stoma formation and closure. The risk of morbidity should be considered when deliberating a stoma.

      Keywords

      1. Introduction

      Stoma creation can be necessary and lifesaving in young children with a bowel perforation, necrosis or obstruction [
      • Hunter C.J.
      • Chokshi N.
      • Ford H.R.
      Evidence vs experience in the surgical management of necrotizing enterocolitis and focal intestinal perforation.
      ,
      • Steinau G.
      • et al.
      Enterostomy complications in infancy and childhood.
      ]. In these critically ill children, a stoma is often created instead of a primary anastomosis to avoid the risk of anastomotic leakage. Stoma formation can result in major stoma related morbidity such as stoma necrosis, stenosis, prolapse, and incisional or parastomal hernia [
      • Eeftinck Schattenkerk L.D.
      • et al.
      Incisional hernia after abdominal surgery in young children: A retrospective analysis of incidence and risk factors.
      ]. Additionally, excessive fluid loss might lead to dehydration and failure to thrive most specifically in case of ileostomies. [
      • Zani A.
      • et al.
      The Timing of Stoma Closure in Young children with Necrotizing Enterocolitis: A Systematic Review and Meta-Analysis.
      ,
      • Bradnock T.J.
      • et al.
      The use of stomas in the early management of Hirschsprung disease: Findings of a national, prospective cohort study.
      ] Most stomas are reversed, which means that these young children undergo a second surgery, after which anastomotic leakage or other complications might still occur. Overall stoma related morbidity is reported to occur in 20–38% of pediatric patients taking into account both stoma formation and closure [
      • Steinau G.
      • et al.
      Enterostomy complications in infancy and childhood.
      ,
      • Eeftinck Schattenkerk L.D.
      • et al.
      Incisional hernia after abdominal surgery in young children: A retrospective analysis of incidence and risk factors.
      ,
      • O'Connor A.
      • Sawin R.S.
      High morbidity of enterostomy and its closure in premature young children with necrotizing enterocolitis.
      ]. The true incidence of major stoma related morbidity cannot reliably be established based on the currently available studies because of small cohort sizes [
      • Steinau G.
      • et al.
      Enterostomy complications in infancy and childhood.
      ,
      • Eeftinck Schattenkerk L.D.
      • et al.
      Incisional hernia after abdominal surgery in young children: A retrospective analysis of incidence and risk factors.
      ,
      • O'Connor A.
      • Sawin R.S.
      High morbidity of enterostomy and its closure in premature young children with necrotizing enterocolitis.
      ].
      Identification of risk factors for major stoma related morbidity could aid in the development of preventative strategies, or might lead to changes in surgical approaches. Previous studies identified a lower weight at stoma closure as a risk factor for postoperative morbidity in patients treated for necrotizing enterocolitis [
      • Eeftinck Schattenkerk L.D.
      • et al.
      Incisional hernia after abdominal surgery in young children: A retrospective analysis of incidence and risk factors.
      ]. However, other studies could not confirm low weight as a risk factor for morbidity following stoma closure [
      • Aguayo P.
      • et al.
      Stomal complications in the newborn with necrotizing enterocolitis.
      ,
      • Mattioli G.
      • et al.
      Risk management in pediatric surgery.
      ,
      • Bælum J.K.
      • et al.
      Enterostomy complications in necrotizing enterocolitis (NEC) surgery, a retrospective chart review at Odense University Hospital.
      ]. In addition, prematurity and underlying inflammatory disease have been reported to be correlated to stoma morbidity [
      • Bradnock T.J.
      • et al.
      The use of stomas in the early management of Hirschsprung disease: Findings of a national, prospective cohort study.
      ,
      • Lee J.
      • et al.
      Enterostomy closure timing for minimizing postoperative complications in premature young children.
      ,
      • Mochizuki K.
      • et al.
      Timing and outcome of stoma closure in very low birth weight young children with surgical intestinal disorders.
      ]. Large cohort studies are needed to more reliably define relevant risk factors.
      Thus, the aim of this study was to assess major stoma related morbidity and its associated risk factors, both after stoma formation and stoma closure.

      2. Methods

      2.1 Patients and management

      All young children (age ≤ three years) that received a stoma between January 1998 and December 2018 at our tertiary referral center were retrospectively identified. The ethical committee of the Amsterdam university medical center approved the study protocol (reference number: W18_233#18.278). Patients and parents received an opt-out letter for consent. Following consent, patient records were checked for eligibility. Data was retrieved and stored in an electronic database (Castor EDC).

      2.2 Data extraction

      Baseline characteristics and complications are presented for all patients. Diseases with N<10 were included as “Other” and are specified in supplementary Table 1.
      The primary outcome included the incidence of major stoma related morbidity, which was defined as Clavien-Dindo grade ≥III complications (morbidity that led to redo-surgery, intensive care admission or death) [
      • Dindo D.
      • Demartines N.
      • Clavien P.A.
      Classification of surgical complications: a new proposal with evaluation in a cohort of 6336 patients and results of a survey.
      ]. Non-stoma related morbidity and stoma related complications with a Clavien-Dindo grade I or II were excluded. Patients that died within 30 days following formation or closure to non-stoma related causes were excluded from the analysis. Stoma related morbidity included: central line sepsis, high output stoma, stoma prolapse, stoma necrosis, stoma stenosis, adhesive obstruction, parastomal hernia, fistula to stoma and anastomotic leakage. Furthermore, following stoma closure, any morbidity related to the initial stoma site which resulted in surgery (e.g. correction of scar tissue or abscess drainage under general anesthesia) was included [
      • Dindo D.
      • Demartines N.
      • Clavien P.A.
      Classification of surgical complications: a new proposal with evaluation in a cohort of 6336 patients and results of a survey.
      ]. Major stoma related morbidity was noted for both types of stoma interventions (formation and closure) and classified according to the moment of occurrence following surgery (within 30 days or after 30 days).
      Patients with a high-output stoma were only included if they underwent redo surgery for insertion of a central venous line. Central line sepsis was only included if the blood culture was positive in concordance with clinical suspicion which led to replacement of a new central venous line under general anesthesia or admission to the intensive care. Adhesive obstructions were identified based on the combined information from the electronic patient file, and surgical and radiological reports, and were recorded only if leading to redo surgery. In case of uncertainty based on the reports, a pediatric surgeon (JD) was consulted. Parastomal hernias were included based on physical examination or ultrasound results and the need for a re-intervention. For anastomotic leakage, the definition and classification of the International Study Group of Rectal Cancer (ISREC) was used [
      • van Helsdingen C.P.
      • et al.
      Consensus on the definition of colorectal anastomotic leakage: A modified Delphi study.
      ,
      • Rahbari N.N.
      • et al.
      Definition and grading of anastomotic leakage following anterior resection of the rectum: a proposal by the International Study Group of Rectal Cancer.
      ]. There is no consensus on a definition for anastomotic stenosis. For this reason, anastomotic stenosis was presumed in patients with obstructive symptoms and description of a stenosis at the anastomotic site seen on contrast enema or during surgery. To distinguish anastomotic stenosis from post disease strictures in necrotizing enterocolitis, an anastomotic stenosis was only included if the stenosis was described at the site of the anastomosis in the surgical report.
      Risk factors for the overall occurrence of major stoma related morbidity (any patient who experienced one or more high grade complications either during or after 30 days) were analysed following stoma formation and following stoma closure separately. Factors that were analysed included gender, surgery before stoma formation, underlying inflammatory disease (necrotizing enterocolitis and meconium peritonitis patients), vasopressin usage post-surgery, history of prematurity (defined as gestational age < 37 weeks), weight at surgery, location of the stoma (ileostomy, jejunostomy or colostomy), duration of presence of a stoma in days and suture resorption speed (defined as normal (Vicryl©, Novosyn©, Monocryl©) or slow (PDS and Monosyn plus©)) used for fascial suturing. These factors were chosen based on previous literature as well as clinical significance aiming to identify general, rather than disease specific, risk factors.

      2.3 Statistical analysis

      Descriptive data were reported as median with interquartile range. Comparison between treatment groups were performed using chi-square test for categorical data, and the Mann-Whitney U test for non-parametric continuous data. Chi squared testing was used to analyze if overall major stoma related morbidity differed over the years comparing patients operated within a consecutive five-year period starting from 1998. Multivariable logistic regression analysis was performed separately for both stoma formation and closure in order to identify risk factors for morbidity. The assumption of linearity of the logit of ordinal variables was assessed using the Box-Tidwell test. Backward Wald selection was used for selection of variables and assessment of confounding (increase in B-coefficient of >10%) and effect modification (significant interaction term). Significant risk factors were reported in odds ratio (OR) with 95% confidence intervals (95%-CI). Additionally, the adjusted R-squared is reported to show the proportion of the variance in the occurrence of major morbidity explained by the model.

      3. Results

      Patient and stoma characteristics are displayed in ​Table 1. In total, 336 young children were included of which 35% (n = 118/336) were treated for an underlying inflammatory disease (necrotizing enterocolitis and meconium peritonitis). Of all patients, 61% (n = 205/336) was male. The median birthweight was 2640 gs and the median gestational age was 36 weeks. Of all patients, 41% (n = 137/336) was born prematurely. The type of stoma was a jejunostomy in 5% (n = 17/336), ileostomy in 57% (n = 192/336), and colostomy in 38% (n = 127/336). Mortality at the end of follow-up was 15% (n = 49/336).
      Table 1Baseline table N = 336.
      CharacteristicTotal = 336 N (%)
      Underlying disease
      Necrotising enterocolitis109 (32)
      Meconium peritonitis9 (3)
      Anorectal malformation88 (26)
      Hirschsprung disease37 (11)
      Intestinal atresia29 (9)
      Meconium ileus20 (6)
      Complex gastroschisis12 (3)
      Other
      Specified in supplementary table 1
      32 (10)
      Underlying disease infectious disease118 (35)
      Male gender, n (%)205 (61)
      Median birthweight (IQR), gram
      Birthweight unknown: 20 ΠWeight at stoma formation unknown: 53 τexcluding deceased patients ϮWeight at stoma closure unknown: 59.
      2640 (1650–2640)
      Median gestational age (IQR), weeks36 (31 - 39)
      Premature, n (%)137 (41)
      Type of stoma, n (%)
      Jejunostomy17 (5)
      Ileostomy192 (57)
      Colostomy127 (38)
      Median age at stoma formation (IQR), days8 (2 – 39)
      Median weight stoma formation (IQR), gram Π2900 (2000–3570)
      Median age at stoma closure (IQR), days142 (81 – 241)
      Median weight at stoma closure (IQR), gram Ϯ5380 (3493–7500)
      Median time to stoma closure (IQR), weeks15 (9–29)
      Major stoma related morbidity at any time, n (%)130 (39)
      Mortality at end of follow up, n (%)49 (15)
      Median follow-up after stoma formation (IQR), months τ76 (33–148)
      Specified in supplementary table 1
      low asterisk Birthweight unknown: 20ΠWeight at stoma formation unknown: 53τexcluding deceased patientsϮWeight at stoma closure unknown: 59.
      Major stoma related morbidity occurred in 15% (n = 49/336) of patients within 30 days following formation (Table 2). The most common morbidity was high output (6%, n = 19/336), stoma necrosis (3%, n = 9/336) and stoma prolapse (2%, n = 8/336). Death within 30 days to non-stoma related causes occurred in 9% (n = 29/336), leaving 307 patients to be assessed for complications risk after 30 days. Following 30 days, a major complication occurred in 19% of patients (n = 58/307). Most reported were high output (6%, n = 19/307), stoma prolapse (5%, n = 14/307), and stoma stenosis (2%, n = 6/307). Overall a complication, either within or after 30 days, occurred in 27% of the patients after stoma creation (n = 92/336).
      Table 2Major stoma related morbidity following stoma formation, N = 336.
      CharacteristicCreation (total = 336)Clavien Dindo 3Clavien Dindo 4Clavien Dindo 5
      Morbidity within 30 days
      Stoma related morbidity, n (%)49 (15)35131
      Type of morbidity, n (%)
      High output19 (6)145
      Stoma necrosis9 (3)9
      Stoma prolapse8 (2)71
      Stoma stenosis7 (2)331
      Central line sepsis6 (2)24
      Morbidity after 30 daysTotal = 307
      Stoma related morbidity, n (%)58 (19)517
      Type of morbidity, n (%)
      High output19 (6)163
      Stoma prolapse14 (5)14
      Stoma stenosis6 (2)6
      Parastomal hernia5 (2)5
      Adhesive obstruction3 (1)3
      Revisions stoma for persistent leakage2 (1)2
      Fistula to stoma1 (1)1
      Other
      E.coli meningitits (n = 2), removal broviac van cava (n = 1), platzbauch (n = 1), stoma revision for bowel torsion (n = 1), diversion colitis (n = 1), perforation (n = 1), conversion colostomy to ileostomy for functional problems (n = 1).
      8 (3)44
      Overall stoma related morbidity, n (%)92 (27)
      ≠Patients that died to non-stoma related cause within 30 days after stoma closure not included in the analysis (n = 29).
      low asterisk E.coli meningitits (n = 2), removal broviac van cava (n = 1), platzbauch (n = 1), stoma revision for bowel torsion (n = 1), diversion colitis (n = 1), perforation (n = 1), conversion colostomy to ileostomy for functional problems (n = 1).
      Of all young children, 87% (n = 292/336) underwent stoma closure. Stoma closure was not performed in 38 patients since they died before stoma closure could be performed. In four patients a permanent stoma was already intended at initial surgery. The remaining two young children are awaiting their stoma closure. Following stoma closure, 5% (n = 16/292) of the young children received a new stoma due to post-operative morbidity or functional problems, of which four underwent construction of a permanent colostomy.
      Following stoma closure, major stoma related morbidity occurred in 8% (n = 24/292) of the patients within 30 days (Table 3). The most commonly occurring complications were anastomotic leakage (2%, n = 6/292), anastomotic stenosis (2%, n = 5/292), and central line sepsis (1%, n = 3/292). After 30 days, a complication was registered in 16% (n = 47/288) of patients, mostly comprising incisional hernia (6%, n = 17/288), anastomotic stenosis (5%, n = 13/288), and adhesive obstruction (4%, n = 12/288). Overall stoma related morbidity rate either within or after 30 days was 23% (n = 66/292).
      Table 3Major stoma related morbidity after stoma closure, N = 292.
      CharacteristicCount (total = 292)Clavien Dindo 3Clavien Dindo 4Clavien Dindo 5
      Stoma related morbidity, n (%)24 (8)195
      Type of morbidity, n (%)
      Anastomotic leak6 (2)51
      Anastomotic stenosis5 (2)5
      Central line sepsis3 (1)3
      Wound abscess1 (1)1
      Other
      Relaparotomy without additional resection (n = 2), septic arthritis (n = 1), perforation after dilatation (n = 1), anastomosis during stoma closure, removal NG tube sigmoid (n = 1), 1x additional resection sigmoid, dehydration (n = 1), torsion after Duhamel (n = 1), bleeding (n = 1) ≠Patients that died within 30 days after stoma closure not included in the analysis (n = 4).
      9 (3)54
      Morbidity after 30 daysTotal = 288
      Stoma related morbidity, n (%)47 (16)35111
      Type of morbidity, n (%)
      Incisional hernia17 (6)152
      Anastomotic stenosis13 (5)931
      Adhesive obstruction12 (4)75
      Correction scar tissue3 (1)3
      Enterocutaneous fistula (from old stoma)2 (1)11
      Overall stoma related morbidity, n (%)66 (23)
      low asterisk Relaparotomy without additional resection (n = 2), septic arthritis (n = 1), perforation after dilatation (n = 1), anastomosis during stoma closure, removal NG tube sigmoid (n = 1), 1x additional resection sigmoid, dehydration (n = 1), torsion after Duhamel (n = 1), bleeding (n = 1)≠Patients that died within 30 days after stoma closure not included in the analysis (n = 4).
      Overall, major stoma related morbidity, either after stoma formation and/or after closure, occurred in 39% (n = 130/336) of the young children. Of the patients that underwent stoma closure 10% (n = 28/288) had a high grade complication after stoma formation, as well as after stoma closure. Over time, splitting the cohort into four groups of five consecutive years, there was no significant change in overall major stoma related morbidity rates (p = 0.52).
      Young children treated with an ileostomy were more at risk of major stoma related morbidity after stoma formation compared to young children treated with a colostomy (OR 2.5; 95%-CI 1.3–4.7, ≤0.01). Gender (p = 0.35), having had a surgical procedure before stoma formation (p = 0.76), inflammatory disease (p = 0.44), prematurity (p = 0.47), weight at stoma formation (p = 0.14) and the need for vasopressin after stoma formation (p = 0.31) were not related to the risk of major stoma related morbidity (Supplementary Table 2).
      Following stoma closure, patients with an ileostomy were also more at risk of major stoma related morbidity (OR:2.7; 95%-CI:1.3–5.8, ≤0.01). Inflammatory disease (p = 0.22), weight at stoma closure (p = 0.18), time to stoma closure in days (p = 0.82), and suture resorption speed (p = 0.84) did not significantly affect the risk of stoma related morbidity (Supplementary Table 3).

      4. Discussion

      Major stoma related morbidity, leading to redo surgery, intensive care admission or death, was high in this retrospective study including 336 young children. Following stoma formation, 27% (n = 92/336) of the young children experienced major stoma related morbidity, mainly consisting of high output stoma, prolapse and stoma stenosis. The major morbidity rate was 23% (n = 66/292) following stoma closure, most commonly comprising anastomotic leakage/stenosis, incisional hernia and adhesive obstructions. For combined stoma interventions, major stoma related morbidity was 39% (n = 130/336). The rate of this morbidity was stable over the years. Ileostomy was the only factor that was independently associated with a higher risk of developing major morbidity following stoma formation (OR:2.5; 95%-CI:1.3–4.7) as well as following closure (OR:2.7; 95%-CI:1.3–5.8). Duration of stoma, underlying inflammatory disease, weight at closure and suture resorption speed were non-significantly related to major stoma related morbidity during stoma closure.
      The most common complications following stoma formation was a high output stoma, which can result in malnutrition. Prolonged malnutrition, due to nutrient and fluid loss via a stoma, can negatively impact growth and adverse cognitive development [
      • Homan G.J.
      Failure to Thrive: A Practical Guide.
      ,
      • Corbett S.S.
      • Drewett R.F.
      To what extent is failure to thrive in infancy associated with poorer cognitive development? A review and meta-analysis.
      ]. It is reported that in the presence of a stoma, up to 90% of the young children are declining on the growth chart, which is why some advocate early stoma closure [
      • Bethell G.
      • Kenny S.
      • Corbett H.
      Enterostomy-related complications and growth following reversal in young children.
      ,
      • Chong C.
      • et al.
      Neonates living with enterostomy following necrotising enterocolitis are at high risk of becoming severely underweight.
      ,
      • Crealey M.
      • et al.
      Managing newborn ileostomies.
      ,
      • Honoré K.D.
      • et al.
      Stoma Closure Improves Head Circumference Growth in Very Preterm Young children after Necrotizing Enterocolitis.
      ]. This opinion is supported by the finding that most young children thrive after stoma closure, irrespective of morbidity, weight, or underlying pathology [
      • Bethell G.
      • Kenny S.
      • Corbett H.
      Enterostomy-related complications and growth following reversal in young children.
      ].
      Considering the increased risk of malnutrition due to stoma morbidity and its impact on growth, it is interesting to see that weight at stoma closure and days until stoma closure were not significantly associated with morbidity following closure in our cohort. This contradicts the accepted preference of many surgeons to wait for a safe weight (e.g. >2.5 kg) before considering stoma closure as to reduce the risk of surgery in a fragile patient [
      • Lee J.
      • et al.
      Enterostomy closure timing for minimizing postoperative complications in premature young children.
      ]. Due to the negative effects of a stoma, some surgeons advocate for early closure within 6 to 8 weeks after creation [
      • Zani A.
      • et al.
      The Timing of Stoma Closure in Young children with Necrotizing Enterocolitis: A Systematic Review and Meta-Analysis.
      ,
      • Veenstra M.
      • et al.
      Timing of ostomy reversal in neonates with necrotizing enterocolitis.
      ]. Until now there is no consensus on the optimal timing of stoma closure in young children. It seems that early closure or closure at a lower weight does not increase the risk of morbidity. Combining this with the finding of recent reports that early closure might result in early catch-up growth might imply the feasibility of early closure [
      • Zani A.
      • et al.
      The Timing of Stoma Closure in Young children with Necrotizing Enterocolitis: A Systematic Review and Meta-Analysis.
      ,
      • Veenstra M.
      • et al.
      Timing of ostomy reversal in neonates with necrotizing enterocolitis.
      ,
      • Talbot L.J.
      • et al.
      Influence of weight at enterostomy reversal on surgical outcomes in young children after emergent neonatal stoma creation.
      ]. An argument against early closure is the presence of adhesions, potentially resulting in difficulties during stoma closure. Part of the adhesions might resolve in the first few weeks to months following index surgery, but this highly depends on the extensiveness of surgery and degree of inflammation with high inter-individual variability. Therefore, the perfect timing of early closure demands future research.
      Following stoma closure anastomotic leakage and stenosis were amongst the most common major complications within 30 days. With an overall incidence of 2% for anastomotic leakage and 6% for anastomotic stenosis, the occurrence in our cohort is similar to previous studies [
      • Bethell G.
      • Kenny S.
      • Corbett H.
      Enterostomy-related complications and growth following reversal in young children.
      ]. It shows that anastomotic morbidity, which seems the main reason not to perform a primary anastomosis, is not completely prevented by stoma formation. Previous studies have recommended construction of primary anastomosis in infantile disease like necrotizing enterocolitis and intestinal atresias [
      • Eeftinck Schattenkerk L.D.
      • et al.
      Treatment of Jejunoileal Atresia by primary anastomosis or Enterostomy: Double the operations, double the risk of complications1,2,3.
      ,
      • Hall N.J.
      • et al.
      Resection and primary anastomosis is a valid surgical option for young children with necrotizing enterocolitis who weigh less than 1000 g.
      ]. Although comparing both treatment options is beyond the scope of this study, the high risk of major stoma related morbidity should not be undervalued when considering both options.
      Incisional hernia and obstructive adhesions were most frequently observed after more than 30 days following surgery. Although the mortality rates of these forms of stoma related morbidity are lower than in adults, they still demand redo surgery, sometimes in an emergency setting [
      • Eeftinck Schattenkerk L.D.
      • et al.
      Incisional hernia after abdominal surgery in young children: A retrospective analysis of incidence and risk factors.
      ,
      • Fredriksson F.
      • Christofferson R.H.
      • Lilja H.E.
      Adhesive small bowel obstruction after laparotomy during infancy.
      ]. The development of new treatment strategies aiming at prevention of stoma formation could lower the risk burden relate to long-term morbidity. This has already been shown by the gradual change in treatment from a two staged (including a stoma) Rehbein's procedure to a one staged pull through in Hirschsprung's disease patients, which started around the new millennium. This change in surgical approach has seemingly lowered the incidence of adhesive obstructions from 10 to 20% to 4–5% [
      • Fredriksson F.
      • Christofferson R.H.
      • Lilja H.E.
      Adhesive small bowel obstruction after laparotomy during infancy.
      ,
      • Lakshminarayanan B.
      • Hughes-Thomas A.O.
      • Grant H.W.
      Epidemiology of adhesions in young children and children following open surgery.
      ,
      • Langer J.C.
      • Seifert M.
      • Minkes R.K.
      One-stage Soave pull-through for Hirschsprung's disease: a comparison of the transanal and open approaches.
      ,
      • Young J.Y.
      • et al.
      High incidence of postoperative bowel obstruction in newborns and young children.
      ].
      The incidence of major stoma related morbidity did not differ between patients with an inflammatory and non-inflammatory disease, which underlines the high morbidity of stomas regardless of the underlying disease and inflammatory state of the patient which has been stated previously [
      • Bethell G.
      • Kenny S.
      • Corbett H.
      Enterostomy-related complications and growth following reversal in young children.
      ,
      • Wolf L.
      • et al.
      Complications of newborn enterostomies.
      ]. Although a higher non-stoma related mortality following stoma formation is to be expected from diseases such as necrotizing enterocolitis, the survival following stoma closure does not differ between these two groups [
      • Anderson J.G.
      • et al.
      Racial and Ethnic Disparities in Preterm Infant Mortality and Severe Morbidity: A Population-Based Study.
      ].
      Identification of risk factors for the development of morbidity following stoma formation and closure might lead to prevention and could aid in the selection which patients to treat with primary anastomosis instead of a stoma. In our cohort, only the location of the stoma was significantly related to the occurrence of morbidity, whilst all other factors were not.
      Limitations of this study are the retrospective design which might have led to underreporting of morbidity, although it is unlikely that serious morbidity was missed. To classify major morbidity related to a stoma, Clavien-Dindo grading was used. This reflects the most severe morbidity, but does not consider the minor morbidity that might have an impact on the overall stoma related morbidity as experienced by the young children. Moreover, this study attempted to identify risk factors for morbidity, but only location of the stoma was found to be an independent predictor. Potentially relevant patient or surgical risk factors might have been missing in the dataset.

      5. Conclusion

      Major stoma related morbidity occurs often in young children, both after stoma formation and closure. The risk of morbidity should be taken into account when considering treatment with the formation of a stoma.

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