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The incidence of different forms of ileus following surgery for abdominal birth defects in infants: a systematic review with a meta-analysis method

  • Laurens D. Eeftinck Schattenkerk ORCID logo EMAIL logo , Gijsbert D. Musters , David J. Nijssen , Wouter J. de Jonge , Ralph de Vries , L.W. Ernest van Heurn and Joep P.M. Derikx
Published/Copyright: August 17, 2021
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Innovative Surgical Sciences
From the journal Innovative Surgical Sciences Volume 6 Issue 4

Abstract

Objectives

Ileus following surgery can arise in different forms namely as paralytic ileus, adhesive small bowel obstruction or as anastomotic stenosis. The incidences of these different forms of ileus are not well known after abdominal birth defect surgery in infants. Therefore, this review aims to estimate the incidence in general between abdominal birth defects.

Content

Studies reporting on paralytic ileus, adhesive small bowel obstruction or anastomotic stenosis were considered eligible. PubMed and Embase were searched and risk of bias was assessed. Primary outcome was the incidence of complications. A meta-analysis was performed to pool the reported incidences in total and per birth defect separately.

Summary

This study represents a total of 11,617 patients described in 152 studies of which 86 (56%) had a follow-up of at least half a year. Pooled proportions were calculated as follows; paralytic ileus: 0.07 (95%-CI, 0.05–0.11; I 2=71%, p≤0.01) ranging from 0.14 (95% CI: 0.08–0.23) in gastroschisis to 0.05 (95%-CI: 0.02–0.13) in omphalocele. Adhesive small bowel obstruction: 0.06 (95%-CI: 0.05–0.07; I 2=74%, p≤0.01) ranging from 0.11 (95% CI: 0.06–0.19) in malrotation to 0.03 (95% CI: 0.02–0.06) in anorectal malformations. Anastomotic stenosis after a month 0.04 (95%-CI: 0.03–0.06; I 2=59%, p=0.30) ranging from 0.08 (95% CI: 0.04–0.14) in gastroschisis to 0.02 (95% CI: 0.01–0.04) in duodenal obstruction. Anastomotic stenosis within a month 0.03 (95%-CI 0.01–0.10; I 2=81%, p=0.02) was reviewed without separate analysis per birth defect.

Outlook

This review is the first to aggregate the known literature in order approximate the incidence of different forms of ileus for different abdominal birth defects. We showed these complications are common and the distribution varies between birth defects. Knowing which birth defects are most at risk can aid clinicians in taking prompt action, such as nasogastric tube placement, when an ileus is suspected. Future research should focus on the identification of risk factors and preventative measures. The incidences provided by this review can be used in those studies as a starting point for sample size calculations.

Keywords: abdominal birth defect; adhesive small-bowel obstruction; anastomotic stenosis; ileus; infant; paediatric surgery

Introduction

Ileus following surgery, consisting of both paralytic and mechanical causes, is a frequent complication after abdominal surgery, leading to increased morbidity, mortality, medical costs, and increased length of hospital stay [1], [2], [3], [4]. It is a clinical diagnosis which is characterized by intolerance to oral feeds, vomiting, abdominal distention, and the absence of flatus or stool.

Ileus following surgery can arise in different forms which depend on the definition used. Common causes are paralytic ileus, adhesive small bowel obstruction (SBO) and anastomotic stenosis. Paralytic ileus is a transient form of ileus which arises shortly after the operation in a response to surgical stress and is based on temporarily intestinal paralysis [5]. Anastomotic stenosis and SBO are mechanical forms of ileus that present later after surgery and could lead to reoperation.

Since abdominal birth defects are rare, the incidence of these forms of ileus are not well known. Yet, knowing the incidences would provide context for clinical decision making as well as a starting point for future research into preventative measures. Therefore, the objective of this review is to systematically aggregate the available data on the incidence of different forms of ileus following surgery for birth defects in infants.

Methods

Studies were selected according to the criteria outlined below based on the PRISMA Guidelines [6]. Our protocol has been registered with the International Prospective Register of Systematic Reviews (PROSPERO) on 7 March 2019 (registration number: CRD42019119268).

Participants

All studies reporting on any form of ileus following surgery for birth defects as primary of secondary endpoint were considered eligible. Only articles that described infants (≤three years) and specifically named the different forms of ileus were included. Animal studies, in vitro studies, non-English or non-Dutch articles, congress abstracts and studies with less than 10 cases were excluded.

Search strategy

The electronic databases of the National Institutes of Health PubMed and EMBASE were systematically searched in February 2020 using both simple search terms as well as hierarchical family forms (e.g. MESH). The search strategy was designed together with a medical information specialist (RV). It combined four groups of search terms and their equivalents [1]: terms related to the age group of the patients at the moment of surgery (e.g. infantile patients) [2]; terms related to the location of surgery (e.g. abdominal surgery) [3]; terms related to congenital abdominal anomalies (e.g. gastroschisis) [4]; terms related to post-operative complications (e.g. adhesive ileus). Mesh and search terms used in Pubmed are included in Appendix 1.

Primary and secondary outcomes

The primary endpoint was the pooled percentage of the three forms of ileus. We separately reviewed anastomotic stenosis within a month and after one month. Secondary endpoint was the pooled percentage per birth defect.

Terms included in paralytic ileus are: ileus (not related to anastomotic stricture) and post-operative ileus. Terms that only implied feeding problems without specifying the reason were not included. Terms included in adhesive small bowel obstruction are: intestinal obstruction, small bowel obstruction, adhesive ileus and stricture (not related to anastomosis). No additional terms or definitions were used for anastomotic stenosis; each article that specifically stated anastomotic stenosis was included. The early anastomotic stenosis, occurring within one month, were excluded for the analysis into late onset anastomotic stenosis.

For each complication separately, a Forest plot was created containing the estimated overall pooled proportion and the corresponding 95%-CIs. In each Forest plot, we also reported the pooled proportion and CIs per disease if [1]; at least three studies reported the specific complication in the disease or [2] if the total number of patients with the disease was ≥100 combined [3]; there was at least one event of a complication present in all studies on a specific disease combined. Birth defects that did not meet these criteria were present in the overall pooled proportion and reported as residuals. Follow-up was reported when relevant.

For all studies with multiple arms, data of both trial-arms were combined. If only one arm matched the inclusion criteria, the appropriate arm was used. Additionally extracted parameters were: author, country of conduct, year of publication, journal, study design, duration of follow-up, number of participants and type of birth defect.

Data extraction

Titles and abstracts were screened to exclude nonrelated publications. Screening was done by two independent authors (LES, DN) using Rayyan. Disagreements were resolved by discussion between the two reviewers. If no consensus could be reached, a third specialist author was consulted (JD, GM). Then, the full texts of the remaining articles were read to determine eligibility for inclusion (LES, DN). If the full text was not found the authors were contacted. The reference lists of the included articles were cross checked to find additional articles.

Validity and eligibility assessment

All included articles were assessed for the methodological quality and risk of bias. For cohort studies the Newcastle Ottawa quality assessment scale was used [7]. In randomized controlled trials this was done using the Jadad scoring system [8]. The assessment was done by LES and DN separately.

Data synthesis

For each complication and each disease in a study, a weighted average of the logit proportions was determined by the use of the generic inverse variance method. The logit proportions were back transformed to the summary estimate and 95%-CIs were obtained in a summary proportion representing the pooled proportion of the form of ileus. Heterogeneity was assessed using the I 2 and χ 2 statistics. Analyses were performed with the use of R-studio version 3.6.1 (package “meta” (Schwarzer, 2007) and “metaprop” (Viachtbauer, 2010)). The random-effects model was used for interpretation. Heterogeneity was deemed significant if the pooled data’s p value was <0.05 or χ 2 statistics were ≥75. Heterogeneity was interpreted as small (I 2≤0.25), medium (I 2=0.25–0.50) or strong (I 2≥0.50), according to Higgins [9].

Results

Study characteristics

In total, 5,784 records were identified. After automated removal of duplicates, 3,909 records were left for title and abstract screening. Of the 3,909 records, 722 were included and assessed for full text. Following full text evaluation 152 studies were included for quantitative analysis (Figure 1). Of the 152 studies, 118 were retrospective cohort studies, 25 were prospective cohort studies, four were retrospective multicentre cohort studies, two were retrospective matched case-control studies, two were randomized controlled trials and one was a combined study of a prospective and retrospective cohort. Studies were conducted in 31 different countries. Asian countries were most prominent with 58 studies, European studies represented 41, North- & South-America represented 38, Africa represented 9, The Middle-East 4 and Oceania represented 2. Of the 152 studies, 86 (57%) reported a follow-up of at least half a year.

Figure 1: Flow-chart article selection.
Figure 1:

Flow-chart article selection.

This systematic review and meta-analysis represent 11,617 patients described in 152 studies presented in Table 1 []. Among these patients, the congenital conditions were divided as follows: Hirschsprung’s disease (n=4,341, 37%); gastroschisis (n=1,558, 13%); duodenal obstruction (n=1,068, 9%); anorectal malformations (n=1,047, 9%); small intestinal atresia (n=794, 7%); congenital diaphragmatic hernia (n=778, 7%); biliary atresia (n=681, 6%); malrotation (n=608, 5%); omphalocele (n=464, 4%); choledochal cyst (n=148, 1%); meconium ileus (n=54, >1%); Meckel’s diverticula (n=46, >1%); colonic atresia (n=30, >1%).

Table 1:

Study characteristics.

Author Year Journal Country Design FU>0.5y Study duration Anomaly POI SBO AS < month ÀS > month
Stollman 2008 Journal of Paediatric Surgery Netherlands Retrospective cohort study No 1971–2004 Small intestinal atresia 2/110 12/110 1/110 6/110
Guo 2010 Transplantation Proceedings China Retrospective cohort study No 2006–2009 Biliary atresia 2/22 X X X
Walter-Nicolet 2009 Journal of Paediatric Gastroenterology and Nutrition France Prospective cohort study Yes 2004–2006 Gastroschisis X 2/73 X X
Wang 2013 Hepatobiliary Pancreat Dis Int China Retrospective cohort study Yes 2008–2011 Biliary atresia X 2/73 X X
Lee 2012 Paediatric Surgery International South Korea Retrospective cohort study No 2001–2010 Small intestinal atresia X 3/11 X X
Cox 2005 Paediatric Surgery International South Africa Retrospective cohort study Yes 1966–2004 Colonic atresia X 0/14 2/14 0/14
Festen 2002 Journal of Paediatric Surgery Netherlands Multi-centre (5) retrospective cohort Yes 1980–1992 Small intestinal atresia 1/15 X 4/15 1/14
Escobar 2005 Journal of Paediatric Surgery USA Retrospective cohort study Yes 1972–2004 Hirschsprung’s disease X 5/33 X 5/33
Yan 2017 Biomedical Research China Prospective cohort study Yes 2011–2014 Hirschsprung’s disease 2/38 2/38 X 2/38
Mirshemirani 2007 Acta Medica Iranica Iran Prospective cohort study Yes 1993–2003 ARM X X 0/30 0/30
Dariel 2015 European Journal of Paediatric Surgery Canada Retrospective cohort study Yes 2006–2010 Gastroschisis X 6/63 X X
Mendez-Martinez 2016 European Journal of General Medicine Mexico Prospective cohort study No 2008–2013 Gastroschisis 6/42 7/42 X X
Fredriksson 2015 British Journal of Surgery Sweden Retrospective cohort study Yes 1976–2011 Hirschsprung’s disease X 19/65 X X
Fredriksson 2015 British Journal of Surgery Sweden Retrospective cohort study Yes 1976–2011 Malrotation X 13/45 X X
Fredriksson 2015 British Journal of Surgery Sweden Retrospective cohort study Yes 1976–2011 Small intestinal atresia X 11/40 X X
Fredriksson 2015 British Journal of Surgery Sweden Retrospective cohort study Yes 1976–2011 Gastroschisis X 9/85 X X
Fredriksson 2015 British Journal of Surgery Sweden Retrospective cohort study Yes 1976–2011 Duodenal obstruction X 10/93 X X
Fredriksson 2015 British Journal of Surgery Sweden Retrospective cohort study Yes 1976–2011 Omphalocele X 4/44 X X
Fredriksson 2015 British Journal of Surgery Sweden Retrospective cohort study Yes 1976–2011 Congenital diaphragmatic hernia X 6/75 X X
Fredriksson 2015 British Journal of Surgery Sweden Retrospective cohort study Yes 1976–2011 ARM X 4/58 X X
Fredriksson 2015 British Journal of Surgery Sweden Retrospective cohort study Yes 1976–2011 Biliary atresia X 0/28 X X
Werbeck 2010 Journal of Paediatric Surgery USA Retrospective cohort study Yes 1991–2010 Gastroschisis X 2/13 X X
Demirogullari 2011 Paediatric Surgery International Turkey Retrospective cohort study No 1998–2011 ARM X 2/112 X X
Rouzrokh 2010 Paediatric Surgery International Iran Retrospective cohort study Yes 2006–2009 Hirschsprung’s disease X X X 12/86
S. Li 2017 European Review for Medical and Pharmacological Sciences China Retrospective cohort study Yes 2006–2013 Hirschsprung’s disease X X 0/15 X
Ghosh 2016 ANZ Journal of Surgery Australia Retrospective cohort study Yes 2005–2012 Hirschsprung’s disease X 3/50 X 2/50
Chen 2014 World J. Paediatric Surgery China Retrospective cohort study Yes 2003–2012 Duodenal obstruction X 4/287 X X
Bianchi 1998 Seminars in Paediatric Surgery England Retrospective cohort study Yes 1984–1997 Hirschsprung’s disease X 2/13 X X
Mattioli 1998 Journal of Paediatric Surgery Italy Retrospective cohort study No 1993–1996 Hirschsprung’s disease X X 0/8 0/8
Teitelbaum 1998 Seminars in Paediatric Surgery USA Retrospective cohort study Yes X Hirschsprung’s disease X 4/24 X 0/24
Demirbilek 1999 Paediatric Surgery International Turkey Retrospective cohort study Yes 1987–1997 ARM X X X 1/31
Santos 1999 Journal of Paediatric Surgery USA Retrospective cohort study No 1988–1999 Hirschsprung’s disease X 4/65 X X
de la Torre 2000 Journal of Paediatric Surgery Mexico Retrospective cohort study Yes 1994–2000 Hirschsprung’s disease X 0/10 X X
Fleet 2000 Journal of Paediatric Surgery England Retrospective cohort study Yes 1991–1997 Gastroschisis X X X 1/10
Fleet 2000 Journal of Paediatric Surgery England Retrospective cohort study Yes 1991–1997 Small intestinal atresia X X X 1/6
Fleet 2000 Journal of Paediatric Surgery England Retrospective cohort study Yes 1991–1997 Colonic atresia X X X 0/3
Hay 2000 Journal of Paediatric Surgery Egypt Retrospective cohort study No X Biliary atresia X 1/21 X X
Langer 2000 Journal of Paediatric Surgery USA Prospective cohort study No X Hirschsprung’s disease X 2/22 10/22 X
Patwardhan 2001 Journal of Paediatric Surgery England Retrospective cohort study Yes 1994–1999 ARM X 5/49 X X
Snyder 2001 Journal of Paediatric Surgery USA Retrospective cohort study No 1969–2000 Gastroschisis X 21/199 X X
Snyder 2001 Journal of Paediatric Surgery USA Retrospective cohort study No 1969–2000 Small intestinal atresia X 9/25 X X
Höllwarth 2002 Journal of Paediatric Surgery Austria Retrospective cohort study Yes 1988–2000 Hirschsprung’s disease X X X X
Saxena 2001 The world journal of Hernia Germany Prospective cohort study Yes 1984–1998 Omphalocele 1/50 2/50 X X
Saxena 2001 Paediatric Surgery International Germany Prospective cohort study Yes 1984–1998 Gastroschisis X 10/70 X X
Önen 2003 Paediatric Surgery International Turkey Prospective/retrospective cohort No 1990–2000 Meckel’s diverticulum X 2/34 X X
Shah 2003 Journal of Paediatric Surgery India Prospective cohort study Yes X ARM X X X 0/12
Weidner 2003 Journal of Paediatric Surgery USA Retrospective cohort study No 1998–2001 Hirschsprung’s disease X 0/15 X X
Escobar 2004 Journal of Paediatric Surgery USA Retrospective cohort study Yes 1972–2001 Duodenal obstruction X 5/169 X 1/169
Kubota 2004 Journal of Paediatric Surgery Japan Prospective cohort study Yes 1990–2001 Hirschsprung’s disease X 1/41 X X
Wester 2004 Journal of Paediatric Surgery Finland Retrospective cohort study No 2000–2003 Hirschsprung’s disease X X 0/15 0/15
Majid 2015 Pakistan Paediatric Journal Pakistan Prospective cohort study Yes X Duodenal obstruction X 3/27 X X
Sauer 2005 Journal of Paediatric Surgery Canada Retrospective cohort study No 1999–2003 Hirschsprung’s disease X 2/24 X 0/24
Thepcharoennirund 2005 Journal of Medical Association of Thailand Thailand Retrospective cohort study No 1987–2004 Gastroschisis 12/129 X X X
Chiu 2006 Journal of Perinatal Medicine USA Retrospective cohort study No 1994–2004 Gastroschisis X 1/43 X X
Choudhry 2006 Paediatric Surgery International England Retrospective cohort study Yes 1998–2003 Gastroschisis X 2/32 X X
Choudhry 2006 Paediatric Surgery International England Retrospective cohort study Yes 1999–2003 ARM X 0/46 X X
Choudhry 2006 Paediatric Surgery International England Retrospective cohort study Yes 2000–2003 Omphalocele X 0/25 X X
Choudhry 2006 Paediatric Surgery International England Retrospective cohort study Yes 2001–2003 Meconium ileus X 5/20 X X
Choudhry 2006 Paediatric Surgery International England Retrospective cohort study Yes 2001–2003 Malrotation X 3/23 X X
Choudhry 2006 Paediatric Surgery International England Retrospective cohort study Yes 2001–2003 Diaphragmatic hernia X 1/25 X X
Choudhry 2006 Paediatric Surgery International England Retrospective cohort study Yes 2001–2003 Hirschsprung’s disease X 2/33 X X
Choudhry 2006 Paediatric Surgery International England Retrospective cohort study Yes 2001–2003 Small intestinal atresia X 4/36 X X
Lee 2006 Journal of Paediatric Surgery USA Retrospective cohort study Yes 1981–2002 Omphalocele 1/20 X X X
A. Li 2006 Chinese Medical Journal China Retrospective cohort study Yes 1999–2004 Hirschsprung’s disease X 1/252 X 16/252
Liem 2006 Asian Journal of Surgery Vietnam Prospective cohort study Yes 2002–2004 Hirschsprung’s disease 0/53 X X X
Maksoud-Filho 2006 Paediatric Surgery International Brazil Retrospective cohort study No 1998–2005 Gastroschisis X 1/43 X X
Owen 2006 Journal of Paediatric Surgery England Retrospective cohort study No 1990–2004 Gastroschisis X 0/48 X X
Banieghbal 2007 Journal of Paediatric Surgery South Africa Prospective cohort study Yes 2002–2005 Small intestinal atresia X X 2/16 X
Dutta 2007 Journal of laparoendoscopic USA Retrospective cohort study No 2002–2005 Biliary atresia X 2/10 X X
Menon 2007 Journal of Paediatric Surgery India Prospective cohort study Yes 1997–2005 ARM X X X 0/46
Riehle 2007 Journal of Paediatric Surgery USA Retrospective cohort study No 1993–2004 Congenital diaphragmatic hernia X 7/125 X X
Stringer 2007 Journal of Paediatric Surgery United Kingdom Prospective cohort study Yes 1994–2006 Biliary atresia X 2/60 X X
Baglaj 2007 European Journal of Paediatric Surgery United Kingdom Retrospective cohort study No 1986–2006 Small intestinal atresia X X X 2/26
Henrich 2007 Paediatric Surgery International Germany Prospective cohort study No 1994–2004 Gastroschisis 11/40 X X X
Henrich 2007 Paediatric Surgery International Germany Prospective cohort study No 1994–2004 Omphalocele 3/26 X X X
Ishikawa 2008 Paediatric Surgery International Japan Retrospective cohort study No 1990–2001 Hirschsprung’s disease X 1/49 X X
Shinall 2008 Journal of Paediatric Surgery USA Retrospective cohort study No 1997–2001 Hirschsprung’s disease X X X 3/60
Spilde 2008 Journal of Paediatric Surgery USA Retrospective cohort study No 2003–2007 Duodenal obstruction X X 2/29 X
Tongsin 2008 Journal of Medical Association of Thailand Thailand Retrospective cohort study No 1988–2007 Small intestinal atresia 9/142 3/142 X 8/142
van Eijck 2008 Journal of Paediatric Surgery Netherlands Retrospective cohort study No 1971–2004 Gastroschisis X 14/55 X X
van Eijck 2008 Journal of Paediatric Surgery Netherlands Retrospective cohort study No 1971–2004 Omphalocele X 12/92 X X
Zheng 2008 Paediatric Surgery International China Retrospective cohort study No 2004–2007 ARM X 1/38 X X
Dassinger 2009 Paediatric Surgery International USA Retrospective cohort study No 1993–2008 Colonic atresia X X 0/12 X
Ferreira 2009 Surgical Endoscopy France Prospective cohort study No X Congenital diaphragmatic hernia 1/30 X X X
Gourlay 2008 Journal of Paediatric Surgery USA Retrospective cohort study Yes 1993–2003 Congenital diaphragmatic hernia X 4/38 X X
Hua 2009 Ghang Gung Medical Journal Taiwan Retrospective cohort study Yes 1991–2006 Choledochal cyst X 1/30 X X
Liu 2009 Journal of Laparoendoscopic China Retrospective cohort study Yes 2003–2007 Biliary atresia X 0/10 X X
Obermayr 2008 European Journal of Paediatric Surgery Germany Retrospective cohort study Yes 2002–2007 Hirschsprung’s disease X X X 1/22
Takahashi 2009 Journal of Paediatric Surgery Japan Retrospective cohort study Yes 1963–2008 Biliary atresia 1/12 X X X
Gunnarsdottir 2009 European Journal of paediatric Surgery Sweden Prospective cohort study Yes 2000–2007 Hirschsprung’s disease X 1/29 X X
Hong 2010 European journal of Obstetrics & Gynaecology China Prospective cohort study Yes 2004–2008 Gastroschisis 1/17 1/17 X X
Payne 2010 Journal of Neonatal-Perinatal Medicine USA Matched case-control study Yes 1999–2007 Gastroschisis X 9/127 X X
Vu 2010 Paediatric surgery International Vietnam Prospective cohort study Yes 2004–2009 Hirschsprung’s disease X X X 4/51
de Vos 2011 South African Journal of Science South Africa Retrospective cohort study Yes 2000–2009 ARM X X X 4/39
Hill 2011 Journal of Laparoendoscopic USA Retrospective cohort study No 2001–2010 Duodenal obstruction 14/58 X X 0/58
Karimi 2011 Paediatric Surgery International Netherlands Retrospective cohort study No 1984–2007 Meconium ileus X 4/34 X X
Kozlov 2010 European Journal of Surgery Russia Retrospective cohort study No 2005–2009 Duodenal obstruction X X 0/27 X
Travassos 2011 Journal of Paediatric Surgery Netherlands Retrospective cohort study Yes 1988–2010 Hirschsprung’s disease X 1/15 X 0/15
van der Zee 2011 World J. Surgery Netherlands Retrospective cohort study Yes 2000–2010 Duodenal obstruction X X X 1/28
Li 2012 Paediatric surgery International China Retrospective cohort study No 2009–2012 Small intestinal atresia X 3/35 X X
Liem 2012 Journal of Paediatric Surgery Vietnam Prospective cohort study Yes 2008–2010 ARM X X X 0/10
Romao 2012 Journal of Paediatric Surgery Canada Retrospective cohort study Yes 2000–2009 Congenital diaphragmatic hernia X 3/22 X X
Sato 2012 Paediatric Surgery International Japan Retrospective cohort study No 2005–2011 Small intestinal atresia 1/25 X X X
Sato 2012 Paediatric Surgery International Japan Retrospective cohort study No 2005–2011 ARM 1/13 X X X
Weil 2011 Journal of Paediatric Surgery USA Retrospective cohort study No 2000–2009 Gastroschisis X X X X
Ghaffarpour 2013 Journal of Paediatric Surgery Sweden Retrospective cohort study No X Duodenal obstruction X 0/28 X X
Ferreira 2013 Journal of Paediatric Surgery France Retrospective cohort study Yes 2006–2010 Congenital diaphragmatic hernia X 9/37 X X
Jensen 2013 Journal of Laparoendoscopic USA Retrospective cohort study No 2005–2011 Duodenal obstruction X X X 1/66
Nam 2013 World Journal of Surgery South Korea Retrospective cohort study Yes 2008–2011 Congenital diaphragmatic hernia X 5/50 X X
Nio 2013 Paediatric Surgery International Japan Prospective RCT Yes 2006–2011 Biliary atresia 2/69 X X X
van der Zee 2013 World Journal of Surgery Netherlands Retrospective cohort study Yes 2000–2011 ARM X 1/19 X 1/19
Diao 2014 International Journal of Surgery China Retrospective cohort study Yes 2011–2013 Choledochal cyst X 0/27 X X
Diao 2014 Journal of Paediatric Surgery China Retrospective cohort study Yes 2011–2012 ARM X X X 0/31
Elder 2014 Journal of Paediatric Surgery USA Retrospective cohort study Yes 2000–2011 Malrotation X 13/102 X X
Friedmacher 2014 Paediatric surgery International Austria Retrospective cohort study Yes 1975–2008 Gastroschisis X 27/108 X 8/108
Ming 2014 Journal of Paediatric Surgery China Retrospective cohort study Yes 1992–2012 ARM X X X 3/66
Nasr 2014 Journal of Paediatric Surgery Canada Retrospective matched case control cohort study No 2000–2010 Hirschsprung’s disease 15/54 1/54 X 4/54
Shrestha 2014 Journal Nepal Paediatric Society Nepal Prospective cohort study Yes 2008–2013 Hirschsprung’s disease X 0/12 X 0/12
Sulkowski 2014 Journal of Paediatric Surgery USA Retrospective multi-centre database research Yes 1999–2009 Hirschsprung’s disease X 82/1,555 X 83/1,555
Yang 2014 Journal of Paediatric Surgery China Retrospective cohort study No 2011–2013 ARM X X X 0/20
Madadi-Sanjani 2015 Biomedical Research International Germany Retrospective cohort study No 1975–2008 Biliary atresia X 5/153 X X
Martinez-Criado 2015 Cirugia Espanola Spain Retrospective cohort study No 2003–2012 Hirschsprung’s disease X 6/73 X 4/73
Miyano 2015 Journal of Laparoendoscopic Japan Retrospective cohort study Yes 2007–2012 Malrotation X 1/14 X X
Shangjie 2014 Cell Biochemical Biophysiology China Prospective cohort study No 2009–2014 Hirschsprung’s disease 28/281 27/281 X X
Almosallam 2016 Ann Saudi Medicine Saudi Arabia Retrospective cohort study No 2000–2014 ARM X 4/104 X X
Diao 2016 Surgical Endoscopy China Retrospective cohort study No 2013–2014 ARM X X X X
Guerra 2016 Journal of Paediatric Surgery Canada Retrospective cohort study No 1995–2014 Hirschsprung’s disease X X X 5/36
Inoue 2016 Surgical endoscopy Japan Retrospective cohort study Yes 2000–2014 Congenital diaphragmatic hernia X 4/24 X X
Matsumoto 2016 Surgical Today Japan Retrospective cohort study No 1997–2015 Choledochal cyst X 2/13 X X
Raitio 2016 European Journal of Paediatric Surgery England Retrospective cohort study Yes 2002–2014 Malrotation X X X X
Diao 2017 Surgical endoscopy China Retrospective cohort study No 2013–2016 ARM X X X 0/15
Dingemann 2017 European Journal of Paediatric Surgery Germany Multicentre retrospective cohort No 2007–2012 Omphalocele X 1/54 X X
C. Lu 2017 Journal of Paediatric Surgery China Multicentre retrospective cohort Yes 2005–2012 Hirschsprung’s disease X X X 21/650
Y. Lu 2017 Transplantation Proceedings China Retrospective cohort study No 2009–2014 Biliary atresia X X X X
Oh 2017 Surgical Endoscopy South Korea Retrospective cohort study No 2005–2015 Duodenal obstruction X X X 0/22
Risby 2017 Journal of Paediatric Surgery Denmark Retrospective cohort study Yes 1997–2009 Gastroschisis X 12/47 X X
Son 2017 Journal of Paediatric Surgery Vietnam Retrospective cohort study No 2009–2015 Duodenal obstruction X X X 2/112
Tyson 2017 Journal of Laparoendoscopic USA Retrospective cohort study No 2007–2015 Congenital diaphragmatic hernia X 5/54 X X
van den Eijnden 2017 World Journal of Surgery Netherlands Retrospective cohort study No 1989–2014 Choledochal cyst X 1/30 0/30 X
Zani 2017 Paediatric Surgery International Canada Retrospective cohort study Yes 2004–2014 Duodenal obstruction X 4/92 X 6/92
Zmora 2016 American Journal of Surgery USA Retrospective cohort study No 2007–2015 Gastroschisis X 1/11 X X
Zmora 2016 American Journal of Surgery USA Retrospective cohort study No 2007–2015 Omphalocele X 0/6 X X
Avci 2018 Eastern Journal of Medicine Turkey Retrospective cohort study No 2008–2017 Duodenal obstruction X 2/32 X X
Peng 2018 Journal of Paediatric Surgery China Retrospective cohort study No 2011–2015 Small intestinal atresia X 2/41 X X
Xiao 2018 Medicine China Prospective cohort study Yes 2011–2014 ARM X X X 0/56
Xiao 2018 Journal of Surgical Research China Prospective RCT Yes 2011–2015 Biliary atresia X 5/166 X X
Zhang 2018 Journal of Paediatric Surgery China Retrospective cohort study Yes 2011–2014 Hirschsprung’s disease X X X 0/23
England 2012 Journal of Paediatric Surgery South Africa Retrospective cohort study Yes 2005–2009 ARM X X X 12/42
Wakhlu 2000 Journal of Paediatric Surgery India Retrospective cohort study Yes 1972–1998 Omphalocele X 2/64 X X
Pratap 2007 Journal of Paediatric Surgery India Retrospective cohort study Yes 2002–2006 Hirschsprung’s disease X X X x
Abbas 2016 Journal of Paediatric Surgery USA Retrospective cohort study Yes 2002–2015 Malrotation X 6/56 X X
Chan 2019 Journal of laparoendoscopic Hong Kong Retrospective database Yes 1993–2007 Biliary atresia X 2/22 X X
de Bie 2019 Journal of Paediatric Surgery Belgium Retrospective multicentre database Yes 2000–2016 Congenital diaphragmatic hernia X 1/62 X X
Dewberry 2019 Journal of Paediatric Surgery USA Retrospective cohort No 2008–2018 Congenital diaphragmatic hernia X 5/70 X X
Dewberry 2019 Journal of surgical research USA Retrospective cohort Yes 2007–2017 Small intestinal atresia X 2/47 X 2/47
Dübbers 2002 European Journal of Paediatric Surgery Germany Retrospective cohort Yes 1990–2000 Hirschsprung’s disease X 2/35 X X
Gabler 2018 South African Medical Journal South Africa Retrospective cohort No X ARM X 0/50 X X
Gao 2019 Journal of International Medical Research China Retrospective cohort No 2018 Meckel’s diverticulum X 0/12 X X
Gao 2019 Journal of International Medical Research China Retrospective cohort No 2018 Hirschsprung’s disease X 0/35 X X
He 2016 Journal of Laparoendoscopic China Retrospective cohort Yes 2011–2016 Congenital diaphragmatic hernia X 1/14 X X
Joda 2019 Updates in Surgery Iraq Prospective cohort Yes 2010–2017 Small intestinal atresia X 3/34 X 4/34
Jona 2001 Paediatric Endo-Surgery & Innovative Techniques USA Retrospective cohort No 1993–2000 Hirschsprung’s disease X 0/44 X X
Long 2019 Arch Dis Child Fetal Neonatal Ed England Retrospective national database cohort Yes 2009–2010 Congenital diaphragmatic hernia X 9/140 X X
Marei 2019 Egyptian Paediatric Association Gazette Egypt Retrospective cohort Yes 2014–2017 Small intestinal atresia 3/22 X 1/22 X
Narang 2019 Journal of Obstetrics & Gynaecology New Zealand Retrospective cohort study Yes 2011–2016 Gastroschisis X 6/71 X X
Narang 2019 Journal of Obstetrics & Gynaecology New Zealand Retrospective cohort study Yes 2011–2016 Omphalocele X 0/22 X X
Jung 1995 Journal of Paediatric Surgery South Korea Retrospective cohort study No 1980–1991 Hirschsprung’s disease X 2/77 X X
Ren 2018 Journal of Laparoendoscopic China Retrospective cohort study Yes 2005–2016 ARM 1/25 X X X
Ryu 2019 Annals of Surgery Treatment and Research South Korea Retrospective cohort study No 2001–2018 Choledochal cyst X 1/43 X X
Sakaguchi 2019 World journal of Paediatric Surgery Japan Retrospective cohort study No 1995–2004 ARM 3/39 X X X
Sato 1998 Journal of Paediatric Surgery Japan Retrospective cohort study No 1970–1997 Small intestinal atresia X 2/88 X 2/88
Sola 2018 Paediatric Surgery International USA Retrospective cohort study No 1999–2016 Hirschsprung’s disease X 4/100 X X
Yang 2019 Medicine China Retrospective cohort study Yes 2013–2016 Small intestinal atresia X 2/42 X 3/42
H. Zhu 2019 Journal of Paediatric Surgery China Retrospective cohort study Yes 2008–2017 Small intestinal atresia X 4/39 X 3/39
H. Zhu 2019 Paediatric Surgery International China Retrospective cohort study Yes 2003–2017 Malrotation X 10/252 X X
T. Zhu 2019 International Journal of Colorectal Disease China Retrospective cohort study Yes 2010–2015 Hirschsprung’s disease X X X 0/157

Risk of bias was assessed and is shown in Table 2. Most studies included reported fair quality on the NOS which was also the case for the two RCTs using the Jadad score. The mean scores on the NOS of articles describing paralytic ileus and early anastomotic stenosis was slightly lower (5, 5) compared to articles describing adhesive small bowel obstruction and late onset anastomotic stenosis [6].

Table 2:

Assessment risk of bias.

Author Year Jadad
 New Ottawa scale (NOS)
Score Selection Comparability Outcome Total
(0–5) (0–4*) (0–2*) (0–3*) (0–9)
Stollman 2008 *** * *** 7
Guo 2010 *** * ** 6
Walter-Nicolet 2009 *** ** *** 8
Wang 2013 *** * *** 7
Lee 2012 *** * ** 6
Cox 2005 *** – *** 6
Festen 2002 ** ** ** 6
Escobar 2005 *** – *** 6
Yan 2017 *** ** *** 8
Mirshemirani 2007 *** – *** 6
Dariel 2015 *** * *** 7
Mendez-Martinez 2016 *** * ** 6
Fredriksson 2015 *** – *** 6
Werbeck 2010 ** – ** 4
Demirogullari 2011 ** – ** 4
Rouzrokh 2010 *** * *** 7
S. Li 2017 ** * *** 6
Ghosh 2016 ** * *** 6
Chen 2014 *** ** *** 8
Bianchi 1998 *** – *** 6
Mattioli 1998 ** – ** 4
Teitelbaum 1998 ** – *** 5
Demirbilek 1999 *** – *** 6
Santos 1999 *** – *** 6
de la Torre 2000 *** – *** 6
Fleet 2000 ** * *** 6
Hay 2000 *** – *** 6
Langer 2000 *** * ** 6
Patwardhan 2001 *** * ** 6
Snyder 2001 *** ** ** 7
Höllwarth 2002 *** – *** 6
Saxena 2001 *** * *** 7
Önen 2003 *** – ** 5
Shah 2003 ** – *** 5
Weidner 2003 ** ** ** 6
Escobar 2004 *** * ** 6
Kubota 2004 ** * ** 5
Wester 2004 *** – ** 5
Majid 2015 *** ** *** 8
Sauer 2005 ** ** ** 6
Thepcharoennirund 2005 ** – ** 4
Chiu 2006 *** * ** 6
Choudhry 2006 *** – *** 6
Lee 2006 ** * *** 6
A. Li 2006 *** * *** 7
Liem 2006 ** – ** 4
Maksoud-Filho 2006 *** – ** 5
Owen 2006 ** ** ** 6
Banieghbal 2007 ** – *** 5
Dutta 2007 ** – ** 4
Menon 2007 ** – *** 5
Riehle 2007 *** – ** 5
Stringer 2007 *** * ** 6
Baglaj 2007 ** * ** 5
Henrich 2007 *** – * 4
Ishikawa 2008 *** – ** 5
Shinall 2008 *** ** ** 7
Spilde 2008 *** * ** 6
Tongsin 2008 *** * ** 6
van Eijck 2008 *** – ** 5
Zheng 2008 *** – *** 6
Dassinger 2009 *** – ** 5
Ferreira 2009 ** – ** 4
Gourlay 2008 *** – *** 6
Hua 2009 *** * *** 7
Liu 2009 *** * *** 7
Obermayr 2008 *** * *** 7
Takahashi 2009 ** – *** 5
Gunnarsdottir 2009 *** ** *** 8
Hong 2010 ** * ** 5
Payne 2010 **** * *** 8
Vu 2010 *** * *** 7
de Vos 2011 *** – *** 6
Hill 2011 *** – ** 5
Karimi 2011 *** – ** 5
Kozlov 2010 *** ** ** 7
Travassos 2011 *** ** *** 8
van der Zee 2011 *** – *** 6
Li 2012 *** – ** 5
Liem 2012 ** * ** 5
Romao 2012 ** * *** 6
Sato 2012 *** * ** 6
Weil 2011 *** * *** 7
Ghaffarpour 2013 *** * ** 6
Ferreira 2013 *** – *** 6
Jensen 2013 *** ** ** 7
Nam 2013 *** ** *** 8
Nio 2013 2 – – – –
van der Zee 2013 ** – *** 5
Diao 2014 ** * *** 6
Elder 2014 *** – *** 6
Friedmacher 2014 *** ** *** 8
Ming 2014 *** * *** 7
Nasr 2014 *** * *** 7
Shrestha 2014 *** – *** 6
Sulkowski 2014 *** ** *** 8
Yang 2014 ** – ** 4
Madadi-Sanjani 2015 *** ** ** 7
Martinez-Criado 2015 *** ** ** 7
Miyano 2015 *** – *** 6
Shangjie 2014 *** * ** 6
Almosallam 2016 *** – ** 5
Diao 2016 ** – *** 5
Guerra 2016 *** * ** 6
Inoue 2016 ** – *** 5
Matsumoto 2016 *** * ** 6
Raitio 2016 *** – *** 6
Diao 2017 ** – ** 4
Dingemann 2017 *** * *** 7
C. Lu 2017 *** – *** 6
Y. Lu 2017 *** * *** 7
Oh 2017 *** ** ** 7
Risby 2017 *** – ** 5
Son 2017 *** ** ** 7
Tyson 2017 *** * ** 6
van den Eijnden 2017 *** * *** 7
Zani 2017 *** * *** 7
Zmora 2016 *** – ** 5
Avci 2018 *** * ** 6
Peng 2018 *** * ** 6
Xiao 2018 3 – – – –
Zhang 2018 ** * *** 6
England 2012 *** – *** 6
Wakhlu 2000 *** – ** 5
Pratap 2007 *** – *** 6
Abbas 2016 *** * *** 7
Chan 2019 ** * ** 5
de Bie 2019 ** * *** 6
Dewberry 2019 *** ** ** 7
Dübbers 2002 *** – ** 5
Gabler 2018 *** – ** 5
Gao 2019 ** – ** 4
He 2016 **** * *** 8
Joda 2019 *** ** *** 8
Jona 2001 ** – ** 4
Long 2019 *** ** *** 8
Marei 2019 ** * *** 6
Narang 2019 ** * *** 6
Jung 1995 ** – * 3
Ren 2018 ** * ** 5
Ryu 2019 *** * ** 6
Sakaguchi 2019 *** * ** 6
Sato 1998 *** – ** 5
Sola 2018 *** * ** 6
Yang 2019 *** * ** 6
H. Zhu 2019 ** ** *** 7
T. Zhu 2019 *** * ** 6

Paralytic ileus

In total, 22 studies reported on paralytic ileus and entailed 1,332 patients and 120 events of paralytic ileus [35, 42, 46, 48, 49, 51, 61, 65, 81, 82, 96, 98, 100, 103, 109, 112, 114, 115, 132, 151, 153, 155]. The pooled proportion of total paralytic ileus was 0.07 (95%-CI: 0.05–0.11; I 2=71%, p≤0.01).

Separate pooled proportions were calculated for the following conditions: Hirschsprung’s disease 0.07 (95%-CI: 0.02–0.24; n=45/426; I 2=91%; p≤0.01); small intestinal atresia 0.05 (95%-CI: 0.03–0.09; n=16/314; I 2=18%; p=0.25); gastroschisis 0.14 (95%-CI: 0.08–0.23; n=30/228; I 2=52%; p=0.03); biliary atresia 0.05 (95%-CI: 0.02–0.11; n=5/103; I 2=0%; p=0.45); omphalocele 0.05 (95%-CI: 0.02–0.13; n=5/96; I 2=8%; p=0.27); anorectal malformations 0.06 (95%-CI: 0.03–0.15; n=5/77; I 2=0%; p=0.83). Duodenal obstruction (n=58) and congenital diaphragmatic hernia (n=30) are included in the overall proportion but did not meet the criteria for separate statistical analysis (Figure 2).

Figure 2: Pooled proportion of paralytic ileus.
Figure 2:

Pooled proportion of paralytic ileus.

Adhesive small bowel obstruction (SBO)

In total, 99 studies reported on small bowel obstruction entailing 8,470 patients and 572 events of SBO all anomalies combined [10, 11, 14], [15], [16], [17], [18], [19, 21, 24, 26, 29], [30], [31, 33, 34, 37], [38], [39], [40], [41, 43, 45, 47, 49, 51], [52], [53], [54, 56], [57], [58], [59], [60, 62, 63, 67, 70], [71], [72], [73], [74, 79], [80], [81, 85], [86], [87], [88], [89, 92], [93], [94, 96, 97, 99], [100], [101, 103, 105, 106, 109], [110], [111, 113, 115], [116], [117], [118], [119], [120], [121, 123], [124], [125], [126, 128, 131, 132, 134, 136], [137], [138], [139], [140], [141], [142], [143], [144], [145], [146], [147], [148], [149], [150, 152, 154, 156], [157], [158], [159], [160]. Length of follow up was at least half a year in 56 (57%) of the studies.

The pooled proportion of total SBO was 0.06 (95%-CI: 0.05–0.07; I 2=74%, p≤0.01). Separate proportions were calculated for the following conditions: Hirschsprung’s disease 0.05 (95%-CI: 0.03–0.07; n=174/3,044; I 2=77%; p≤0.01); gastroschisis 0.09 (95%-CI: 0.06–0.14; n=130/1,147; I 2=75%; p≤0.01); congenital diaphragmatic hernia 0.08 (95%-CI: 0.06–0.11; n=60/736; I 2=35%; p=0.09); duodenal obstruction 0.04 (95%-CI: 0.02–0.08; n=28/728; I 2=61%; p=0.01); small intestinal atresia 0.09 (95%-CI: 0.05–0.14; n=60/690; I 2=74%; p≤0.01); biliary atresia 0.03 (95%-CI: 0.02–0.05; n=19/543; I 2=0%; p=0.48); malrotation 0.11 (95%-CI: 0.06–0.19; n=46/492; I 2=73%; p≤0.01); anorectal malformations 0.03 (95%-CI: 0.02–0.06; n=17/476; I 2=37%; p=0.54); omphalocele 0.04 (95%-CI: 0.02–0.24; n=21/357; I 2=51%; p=0.31); choledochal cyst 0.03 (95%-CI: 0.01–0.08; n=5/143; I 2=0%; p=0.48). Meconium ileus (n=54), Meckel’s diverticula (n=46) and colonic atresia (n=14) are included in the overall proportion but did not meet the criteria for separate statistical analysis (Figure 3).

Figure 3: Pooled proportion of small bowel obstruction.
Figure 3:

Pooled proportion of small bowel obstruction.

Anastomotic stenosis

Of the 14 studies reporting on anastomotic stenosis within one month of follow-up, 365 patients were included and 22 events of anastomotic stenosis occurred [13, 18, 20, 35, 57, 59, 64, 75, 78, 108, 109, 118, 129, 151].

The pooled proportion of total anastomotic stenosis within a month was 0.03 (95%-CI: 0.01–0.10; I 2=81%, p=0.02). Diseases reported on were: small intestinal atresia (n=8/163), Hirschsprung’s disease (n=10/60), duodenal obstruction (n=2/56), anorectal malformations (n=0/30), choledochal cyst (n=0/30) and colonic atresia (n=2/26).

In total, 40 studies reported on anastomotic stenosis after one month of follow-up entailing 4,468 patients and 214 events of anastomotic stenosis occurred [12, 18, 22, 23, 25, 32], [33], [34], [35], [36, 38, 40, 44, 49, 55, 62, 66, 68, 73, 75], [76], [77], [78, 81, 83, 84, 95, 99, 102, 104, 105, 107, 109, 111, 113, 115, 116, 119, 120, 122, 129, 130, 132], [133], [134], [135, 142, 156, 158, 159, 161]. Length of follow up was at least half a year in 29 (73%) of the studies.

The pooled proportion of anastomotic stenosis was 0.04 (95%-CI: 0.03–0.06; I 2=59%, p=0.30). Separate proportions were calculated for the following conditions: Hirschsprung’s disease 0.04 (95%-CI: 0.03–0.07; n=162/3,238; I 2=70%; p=0.11); small intestinal atresia 0.06 (95%-CI: 0.04–0.08; n=32/548; I 2=0%; p=0.77); duodenal obstruction 0.02 (95%-CI: 0.01–0.04; n=11/547; I 2=42%; p=0.29); gastroschisis 0.08 (95%-CI: 0.04–0.14; n=9/118; I 2=0%; p=0.77). Colonic atresia (n=17) is included in the overall proportion but did not meet the criteria for separate statistical analysis (Figure 4).

Figure 4: Pooled proportion of anastomotic stenosis after 1 month.
Figure 4:

Pooled proportion of anastomotic stenosis after 1 month.

Discussion

This systematic review pooled the reported proportions on different types of ileus following abdominal surgery for birth defects in infants. These proportions can be seen as an approximation of the incidences of these complications. According to our reported approximation, these incidences were 7% for paralytic ileus, 6% for adhesive small bowel obstruction, 3% for anastomotic stenosis within one month after surgery and 4% after one month. Within the different birth defects there is a large variation in the occurrence and the spread of these forms of ileus. Although risk factor identification is beyond the scope of this review, the available literature gives some suggestions why these diseases seem to be more at risk.

Out of all diseases paralytic ileus was most common in gastroschisis patients (14%). In these patients, a defect of the abdominal wall leads to extrusion of abdominal content antenatally. Postnatally, this content is reduced intra-abdominally either by primary closure or temporally use of silo and delayed closure. During both procedures the intestine is manipulated severely, which is known to increase the incidence and duration of paralytic ileus in adults [4].

Adhesions, which cause SBO, have long been accepted as a partly inevitable consequence of surgery. They occur as part of the natural healing process. It is hard to define the clinical significance of adhesions, since most are asymptomatic, but when they lead to small bowel obstruction, they can be fatal with mortality rates in children between 2 and 15% [121, 162, 163]. Recently duration of surgery and staged procedures have been identified as risk factors for SBO [37, 162, 164].

Our reported pooled incidence of 6% is comparable to most recent large (n≥100) individual cohort studies reporting on abdominal surgery in infants. These studies report an incidence of SBO between 6 and 10% [37, 165]. It is important to acknowledge that this review entails an aggregated incidence for birth defects only. Acquired diseases such as necrotizing enterocolitis, which seems to be at high risk with a reported incidence of SBO between 25 and 64%, are therefore not included [37, 162].

We found that patients with a malrotation, small intestinal atresia or gastroschisis were relatively most at risk of SBO. This is in concordance with previous studies [37, 121, 162], [163], [164], [165].

We divided anastomotic stenosis into two groups based on reported occurrence within or after one month of surgery since early onset is suggested to be caused by technical error or tissue oedema, whereas a delayed onset and stricture formation is related to chronic inflammation in time leading to anastomotic scarring [166].

Early onset of an anastomotic stenosis is not widely reported and might even be overlooked in the infantile cohort. This review shows that early stenosis does occur and should be considered when conducting research into post-operative complications in the infantile cohort. Technical factors, such as suture reportion speed or mode of suturing, of influence during anastomotic creation should be evaluated to identify risk factors.

Gastroschisis, and to a smaller extent intestinal atresias, were most at risk for late onset anastomotic stenosis. The process of anastomotic healing is to a great extent unclear. Most research has focussed on surgical innovations and techniques without the results leading to a conclusive resolution. Future research in the pathobiology at the cellular level might bring clarification on this matter [166].

This study has its limitations. Because of the variety in study designs and reported outcomes we were not able to look into risk factors which could have lowered heterogeneity. Although it must be noted that, by stratifying for birth defect, some outcomes had moderate to low heterogeneity. Another limitation was that because certain birth defects such as gastroschisis only occur in neonates, our stratification might have resulted in differences in mean age when comparing birth defects. This age difference could be an important reason why certain birth defects are more at risk of certain form of ileus. However, it is not the aim of this review to compare different birth defects but rather report an incidence for each individually. Thus, we believe that this age difference will not hinder the message of our review. If we had only included neonates in this review important birth defects, such as Hirschsprung’s disease, diagnosed beyond the neonatal period would have been excluded. Furthermore, it has to be stated that our results are based on retrospective cohorts available in the literature most of which did not have ileus as a primary outcome. This has undoubtedly increased the chances of occurrence of forms of bias such as selection, publication and reporting bias. Our risk of bias assessment showed most articles to have only fair quality mostly caused by the retrospective, observational nature of most included studies. Moreover, most studies did not have a strict definition of complications possibly resulting in observer bias. Lastly, only 57% of the included articles had a follow-up of at least half a year. Many other articles were unclear about the length of follow up. This lack of long-term follow-up might result in an underestimate of the real incidence of SBO and anastomotic stenosis. SBOs, for instance mostly arise within a year after surgery however episodes are reported 28 years after the initial laparotomy [17, 37, 121, 162], [163], [164], [165]. Although these limitations might have influenced the pooled analyses, at this moment the presented data is the best available approximation of these complications in this cohort.

Conclusion

This review is the first to aggregate the known literature in order to approximate the incidence of different forms of ileus for each abdominal birth defect. We showed these complications are common and the distribution differs between birth defects. Knowing which birth defects are most at risk might aid clinicians in taking prompt action when an ileus is suspected. Future research should focus on the identification of risk factors and preventative measures. The incidences provided by this review can be used as a starting point for sample size calculations.

Corresponding author: Laurens D. Eeftinck Schattenkerk (LES), MD, Department of Paediatric Surgery, Emma Children’s Hospital, Amsterdam University Medical Center, University of Amsterdam and Vrije Universiteit Amsterdam, Meibergdreef 9, 1005 AZ, Amsterdam, Netherlands; and Tytgat Institute for Liver and Intestinal Research, Amsterdam UMC, University of Amsterdam, Amsterdam, Netherlands, Phone: +31 (0)20 566 9111, E-mail:

Acknowledgments

We would like to thank Shaffy Roell for the support with the development of the illustrations.

  1. Research funding: None declared.

  2. Author contributions: All authors have accepted responsibility for the entire content of this manuscript and approved its submission.

  3. Competing interests: Authors state no conflict of interest.

  4. Informed consent: Not applicable.

  5. Ethical approval: Not applicable.

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Supplementary Material

The online version of this article offers reviewer assessments as supplementary material (https://doi.org/10.1515/iss-2020-0042).

Received: 2020-12-07
Accepted: 2021-04-06
Published Online: 2021-08-17

© 2021 Laurens D. Eeftinck Schattenkerk et al., published by De Gruyter, Berlin/Boston

This work is licensed under the Creative Commons Attribution 4.0 International License.

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  1. Frontmatter
  2. Editorial
  3. Editorial
  4. Reviews
  5. The incidence of different forms of ileus following surgery for abdominal birth defects in infants: a systematic review with a meta-analysis method
  6. Need for transition medicine in pediatric surgery – health related quality of life in adolescents and young adults with congenital malformations
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https://doi.org/10.1515/iss-2020-0042
Keywords for this article
abdominal birth defect; adhesive small-bowel obstruction; anastomotic stenosis; ileus; infant; paediatric surgery
Creative Commons
BY 4.0

Articles in the same Issue

  1. Frontmatter
  2. Editorial
  3. Editorial
  4. Reviews
  5. The incidence of different forms of ileus following surgery for abdominal birth defects in infants: a systematic review with a meta-analysis method
  6. Need for transition medicine in pediatric surgery – health related quality of life in adolescents and young adults with congenital malformations
  7. Image-guided surgery and novel intraoperative devices for enhanced visualisation in general and paediatric surgery: a review
  8. Original Article
  9. Ovarian lesions and tumors in infants and older children
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