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Abstract

Objective. Birth prevalence estimates for major congenital gastrointestinal anomalies are sparse. This paper serves as an in-depth, a priori systematic review protocol for our systematic review identifying and assessing studies reporting population-level birth prevalence estimates for gastrointestinal anomalies. Methods. We will use the Condition, Context, Population framework and will include studies reporting the population-level birth prevalence of gastroschisis, omphalocele, intestinal and esophageal atresia, Hirschsprung’s disease, congenital diaphragmatic hernia, and anorectal malformation in infants. We will assess bias using the Joanna Briggs Institute Critical Appraisal Checklist for Studies Reporting Prevalence Data. Results. Results will be published in a separate paper, and will be stratified by World Bank income classification. Conclusion. Knowledge of the current prevalence of major congenital gastrointestinal anomalies is essential to efficient surgical resource planning. This review will compile existing global estimates of prevalence and incidence of common gastrointestinal congenital anomalies to support policy development.

Keywords

congenital anomaly GI anomaly gastrointestinal anomaly global surgery prevalence incidence systematic review gastroschisis omphalocele exomphalos intestinal atresia esophageal atresia congenital diaphragmatic hernia anorectal malformation

Introduction

Globally, gastrointestinal (GI) anomalies contribute to a substantial number of neonatal and infant deaths each year, but the burden of disease (eg, birth prevalence) of these conditions is poorly understood in most settings and under-researched in low resource settings in particular. Approximately 0.98% of neonatal deaths and 1.11% of infant deaths are attributable to these anomalies (collectively) globally each year and mortality is disproportionately high in low- and middle-income countries (LMICs).^1,2 These anomalies include a range of structural and/or functional malformations affecting the digestive tract and/or abdominal wall, and include gastroschisis, omphalocele (exomphalos), intestinal and esophageal atresias, congenital diaphragmatic hernia, Hirschsprung’s disease, and anorectal malformations.^3-5 To date, there have been few systematic reviews or meta-analyses compiling existing population-level birth prevalence estimates for these gastrointestinal anomalies that can be used to inform pediatric surgical interventions, research, and policy. Further, existing surveillance systems predominately represent high-income countries (HICs) and/or report aggregated birth prevalence estimates (rather than GI anomaly specific birth prevalence).^6-12 The current lack of understanding of common GI congenital anomaly prevalence limits effective clinical interventions, policy development, and resource allocation.

Knowledge of these prevalence estimates is even more limited in LMICs.^13-16 Although the World Health Organization estimates that the majority (90%-94%) of the overall congenital anomaly disease burden is concentrated in LMICs, it is unclear if the prevalence of GI anomalies is also higher in LMICs than in HICs.^1,17-19 If prevalence of these conditions is higher in LMICs than HICs or if existing prevalence estimates are underestimates of true burden of disease, current surgical resource planning for these conditions may not address the true burden of disease.²⁰

Many of the existing GI anomaly birth prevalence estimates are not representative of the total prevalence of these conditions across all births at risk for these conditions, and few are from LMICs. Paoletti et al.¹⁴ conducted a systematic review of global CDH prevalence and found a pooled CDH prevalence of 2.3 per 10 000 live births. In their review, Paoletti et al.¹⁴ report 2.1 cases per 10 000 births in Colombia,²¹ 2.4 cases per 10 000 births in Central and South America (all participating countries combined),⁸ 1.1 cases per 10 000 births in Iran,²² and 3.4 per 10 000 births in Argentina.^14,23 However, the majority of estimates in this review are from HICs.¹⁴ Tiruneh et al.¹³ conducted a systematic review of omphalocele and gastroschisis prevalence among children with congenital anomalies in sub-Saharan Africa, reporting omphalocele in 4.47% of infants with at least one congenital birth defect and gastroschisis in 3.22% of infants with at least one congenital birth defect.¹³ These data indicate that omphalocele and gastroschisis contribute to a substantial proportion of anomaly cases, but do not tell us the prevalence of these conditions among all births (with and without anomalies). Chen et al.²⁴ and Duess et al.¹⁶ each conducted a systematic review of Hirschsprung’s disease prevalence among preterm births (finding that 7% and 6% of preterm births had Hirschsprung’s disease, respectively, with substantial variation in estimates over study years). Although each of these studies reports prevalence estimates for GI anomalies, the study populations do not generally examine the overall populations at risk for these conditions. Further, many prevalence estimates are from single hospital center studies where the population denominator is the number of GI anomaly cases among the number of admitted preterm births or surgical cases rather than the total number of births.^13,25-28 Prevalence estimates drawn from hospital admissions data may not reflect those in the general population if the group that presents to the hospital is not representative of the general population, and may not account for lack of access to care.

A systematic review compiling evidence from peer-reviewed birth prevalence studies is essential to estimate the burden of GI anomaly disease globally, regionally, and nationally. Appraisal of these estimates can help to efficiently design health care policy, financing, resources, and research to address the burden of GI anomalies and gaps in knowledge about these conditions. As such, the aim of our systematic review is to identify, compile, and critically appraise national and regional population-based GI anomaly birth prevalence estimates in both LMIC and HIC settings. This manuscript establishes a priori our systematic review aims, methodology, and primary outcomes. Publication of this protocol will support transparency in methodology and results reporting and allow for greater peer and subject matter expert input of the review.²⁹

Methods

Aims

The systematic review will address the following research question: What is the population-level birth prevalence of GI anomalies (gastroschisis, omphalocele, intestinal atresia, esophageal atresia, Hirschsprung’s disease, CDH, and ARM globally and across World Bank (WB) income classifications (HICs and LMICs)? Below, we outline our methodology and protocol for the review.

Inclusion and Exclusion Criteria

Inclusion criteria include all clinical studies reporting the population-level birth prevalence of gastroschisis, omphalocele (exomphalos), intestinal atresia, esophageal atresia, Hirschsprung’s disease, CDH, and/or ARM. While it would be ideal to include only studies reporting these conditions according to standardized International Classification of Diseases and Related Health Problems (ICD) 9 and 10 codes, this is often infeasible and unavailable in low resource settings and has the potential to severely restrict studies included in this review.^30,31 Population-level prevalence/incidence estimates will be defined as those estimates calculated at sub-national, national, regional, or global levels and in which the target and study populations are representative of all births or children at risk of the disease, both treated and untreated. We include studies referring to birth prevalence as “incidence,” as this terminology (though interchangeable by definition) is used inconsistently in the literature. Surveillance, prospective cohort, retrospective cohort, case-control, and cross-sectional population-based studies will be included. Studies from all publication years will be assessed for inclusion given anticipated sparsity in relevant studies. If a study is a nationally representative survey of households (eg, using the Surgeons OverSeas Assessment of Surgical Need tool) to ascertain an estimated number of recent cases (resulting in death, disability, or survival), the study will be included.³²

Exclusion criteria include not being a population-based study (eg, hospital registry based or single study site-based where the study population is not representative of general population at risk of these conditions), being a systematic review or meta-analysis, being published in a language other than English, Spanish, or Portuguese, and being a study or report that is not published in peer-reviewed journals. Inclusion of additional languages is precluded by coauthor linguistic knowledge. If a study is conducted in a national hospital setting and in a country in which we anticipate that the majority births are registered and we can assume that most (if not all) cases reach care, the study will be included (eg, a national hospital surveillance study for gastroschisis in Swedish national hospitals with the population denominator being the country’s number of registered births).³³ No exclusion criteria will be applied to year of publication given the anticipated sparsity of studies addressing this research question. The last date of extraction for each database will be September 8, 2023.

Population, Setting, and Participants

Based on the nature of our research question, we will use the Condition, Context, Population (CoCoPop) framework for prevalence and incidence systematic review (Table 1).^34,35 This framework will help to ensure that each study included in the review addresses the correct conditions in the population and context of our research question. The condition will be having at least one of the following GI congenital anomalies: gastroschisis, omphalocele, intestinal atresia, esophageal atresia, Hirschsprung’s disease, congenital diaphragmatic hernia, and/or anorectal malformation. The context includes all countries around the world.³⁶ We will use the 2023 World Bank income classifications to identify each country as a low-income country (LIC), middle-income country (MIC), or HIC, and will stratify our results by income classification (LIC, MIC, and HIC).³⁷ The population of interest will be infants of any sex assigned at birth who have at least one of these conditions.

Table 1.

CoCoPop (Condition, Context, Population) Framework^34,35 and Search Strategy.

CoCoPop statement	Search number	Search builder	Search string
Condition: gastroschisis, omphalocele, intestinal atresia, esophageal atresia, Hirschsprung’s Disease, congenital diaphragmatic hernia, and/or anorectal malformation	1		“Gastroschisis” OR “major abdominal wall defects” OR “major abdominal wall defect” OR “congenital abdominal wall defect” OR “congenital abdominal wall defects” OR “abdominal wall defect” OR “abdominal wall defects”
	2		Omphalocele OR exomphalos OR major abdominal wall defects OR congenital abdominal wall defect
	3		Intestinal atresia OR small intestinal atresia OR duodenal atresia, duodenal stenosis, small bowel atresia
	4		Esophageal atresia OR Oesophageal atresia OR long gap EA
	5		Congenital diaphragmatic hernia OR CDH OR Bochdalek hernia OR Morgagni hernia
	6		Hirschsprung’s Disease OR congenital megacolon OR congenital aganglionosis
	7		Anorectal malformation OR ARM OR imperforate anus
Context: LIC, MIC, and/or HIC			We will not apply LIC/MIC/HIC search terms, as we are interested in all settings. We will, however, flag which articles are in LICs, MICs, HICs, and the following countries during screening for ease of analysis: Afghanistan, Burundi, Ethiopia, Gambia, Rwanda, Tanzania, Algeria, Angola, Argentina, Bangladesh, Belarus, Bolivia, Bosnia and Herzegovina, Brazil, China, Colombia, Dominican Republic, Ecuador, Egypt, Gabon, Ghana, India, Indonesia, Iran, Jordan, Kenya, Libya, Macedonia, Malaysia, México, Nigeria, Pakistan, Palestine, Gaza, Palestine Gaza, Gaza Strip, Gaza Strip, Palestine, Gaza, Palestine, Palestine, Palestine, West Bank, Palestine West Bank, West Bank, West Bank, Palestine, Papua New Guinea, Peru, Philippines, Romania, Serbia, South Africa, Sri Lanka, Sudan, Syria, Thailand, Tunisia, Turkey, Uzbekistan, Zambia, Zimbabwe, Australia, Austria, Belgium, Canada, Chile, Czech Republic, England, UK, United Kingdom, France, Germany, Italy, Hungary, Lithuania, New Zealand, Poland, Qatar, Saudi Arabia, Scotland, Singapore, South Korea, Spain, Sweden, Uruguay, United States of America, US, USA, Latin America, Africa, sub-Saharan, East Asia, Pacific, East Asia and Pacific, Europe, Central Asia, Europe and Central Asia, Caribbean, Latin America and Caribbean, Middle East, North Africa, Middle East and North Africa, South Asia
Population: Infants of any sex assigned at birth and of treatment status living in LICs, MICs, and HICs	8		“Child” OR “children” OR “pediatric” OR “pediatrics” OR “infant” OR “infants” OR “at birth” OR “minors” OR “adolescent” OR “adolescents” OR “children” OR “minor” OR “minors” OR “neonate” OR “neonatal” OR “neonates”
Study outcome: prevalence (point or period), incidence (proportion, rate)	9		“prevalence” OR “point prevalence” OR “period prevalence” OR “incidence” OR “incidence rate” OR “incidence proportion” OR “risk” OR “live births” OR “cases” OR “number of cases” OR “per live births” OR “per 10 000 live births” OR “per 10 000 live births” OR “per 5000 live births” OR “per 5000 live births”
Condition + Context, + Population (Gastroschisis)	10	1 AND 8 AND 9
Condition + Context, + Population (Omphalocele/Exomphalos)	11	2 AND 8 AND 9
Condition + Context, + Population (Intestinal Atresia)	12	3 AND 8 AND 9
Condition + Context, + Population (Esophageal Atresia)	13	4 AND 8 AND 9
Condition + Context, + Population (Congenital diaphragmatic hernia)	14	5 AND 8 AND 9
Condition + Context, + Population (Hirschsprung’s Disease)	15	6 AND 10 AND 11
Condition + Context, + Population (Anorectal malformation)	16	7 AND 8 AND 9
Databases to be searched
PubMed
EBSCO (Academic Search Complete, CINAHL, Global Health)
SCOPUS (including EMBASE)

Our outcome is birth prevalence. We are interested in the total number of cases in the population during a defined time period (treated and untreated) and in measures of birth prevalence, though terminology for reporting this metric varies across the literature.

Given the variation in the denominators of these estimates and the variations in reporting practices, we will categorize and report estimates in three mutually exclusive categories based on the terminology and denominator each given study uses: (1) number of cases per 10 000 live births, (2) number of prevalent cases per 10 000 total births (live births and stillbirths, with or without terminations), and (3) other measures of incidence and prevalence (eg, number of prevalent cases per 10 000 pregnancies). If a single study reports multiple measures, we will report all measures in our review. If a study reports estimates for multiple countries, we will report each estimate in our review. If a study erroneously reports birth prevalence using incidence terminology, we will report results as birth prevalence. For estimates using a denominator other than 10 000, we will convert the denominator to 10 000.

Study Sources and Search Strategy

We will apply search terms (Table 1) for all GI anomalies using seven databases: PubMed, JSTOR, Academic Search Complete (EBSCO), CINAHL (EBSO), EMBASE, Global Health (EBSCO), and SCOPUS, noting that Academic Search Complete, CINAHL, and Global Health can be searched through EBSCO and EMBASE can be searched through SCOPUS. Our full search strategy, including all Medical Subject Heading (MeSH) terms specific to each database, is outlined in Supplemental File 1. All study methods have been published in PROSPERO (CRD42023460268) and follow the Preferred Reporting Items for Systematic reviews and Meta-Analyses for Protocols 2015 (PRISMA-P 2015) guidelines (Supplemental File 2).³⁰ Any changes to the protocol will be updated in the PROSPERO registration and explicitly described and justified in the results manuscript.

Study Screening and Selection

We will screen all studies using Covidence systematic review software.³⁸ We will first screen studies based on abstracts. Studies that pass the first screening phase will be screened based on the full text, during which bias assessment (described below) will be conducted. The number of studies included and excluded at each step will be summarized using a PRISMA-P diagram.³⁹ The first author (KRL) will be the only abstract screener. KRL and PEG will conduct full-text review. KRL will double code the bias assessment for all articles screened by PEG to assess inter-reviewer reliability. If inter-reviewer reliability is less than 80% agreement across the two reviewers, discrepancies will be adjudicated amongst KRL, PEG, and ERS.

Bias Assessment and Quality Appraisal

All bias assessment scoring and data extraction will be conducted using the Covidence software. To assess the risk of bias, we will use the JBI Critical Appraisal Checklist for Studies Reporting Prevalence Data (Supplemental File 3), with risk of bias conducted at the study-level and for the prevalence estimate in the study.^40-42,34 Any issues in study quality and bias assessment will be discussed and decided as a research group. We will report results from all studies that meet study inclusion criteria, regardless of their JBI checklist score.

The JBI checklist is based on a total score that is the sum of nine questions (a higher score is indicative of lower risk of bias): (1) Was the sample frame appropriate to address the target population?, (2) Were study participants sampled in an appropriate way?, (3) Was the sample size adequate?, (4) Were the study subjects and the setting described in detail?, (5) Was the data analysis conducted with sufficient coverage of the identified sample?, (6) Were valid methods used for the identification of the condition?, (7) Was the condition measured in a standard, reliable way for all participants?, (8) Was there appropriate statistical analysis?, (9) Was the response rate adequate, and if not, was the low response rate managed appropriately?⁴² Mean, standard deviation (SD), median, minimum, and maximum JBI score values will be reported for all conditions together and for each condition separately. Overall quality appraisal will be assessed using this bias assessment separately for each study condition.

Data Extraction and Synthesis

Data will be exported from Covidence to SAS 9.4 for data cleaning, coding, and summarizing.⁴³ We will collect the following variables from all studies included in the final screening: title, author list, year of publication, journal of publication, study design, country (or countries), World Bank income region(s) and income classification(s), measure of occurrence type (prevalence, incidence), measure of occurrence value (including upper and lower confidence intervals (CIs) and P-values if reported), exposure condition(s) (gastroschisis, omphalocele, intestinal atresia, esophageal atresia, congenital diaphragmatic hernia, Hirschsprung’s disease, and/or anorectal malformation). We will not conduct any meta-analyses, including the pooling of all estimates.

Ethical Approval and Informed Consent

This study will not collect or include patient health information (PHI) and involves no data collection from human subjects. As such, informed consent is not needed for this systematic review and procedures for this study were determined exempt from Institutional Review Board (IRB) approval (23-3083; Exemption Category 4: secondary data collection/specimens). No authors received specific funding for this study. No authors have conflicts of interests to disclose. All results will be made publicly available, with the goal of publishing in an open-access journal. Data in the results paper for this systematic review are able to be ascertained via the tables in the manuscript and the original articles included in the review. Raw data tables may also be requested from the corresponding author.

Results

Results from this paper will be published in a separate paper. The purpose of this paper is to publish an a priori protocol for this systematic review.

Discussion

Few studies have systematically summarized the literature for the birth prevalence of GI anomalies, and none have done so applying the same methodology to a large set of major GI anomalies together.^13,14,16,24 Anomalies of the GI tract are common, require timely care, and have higher mortality in LMICs compared to HICs.² Yet, we do not know the birth prevalence of these conditions in most settings, particularly in LMICs. This research gap warrants a large, critical, systematic search and appraisal of literature for measures of occurrence for these diseases.

Many current estimates of congenital anomalies are drawn from hospital-based and/or HIC-based studies of prevalence of these conditions among preterm infants or infants with multiple congenital anomalies. However, knowing how many infants and children have these conditions is essential to efficiently and equitably allocate pediatric surgical resources to address the burden of these diseases. Further, critical appraisal of the existing literature is essential to making recommendations for epidemiologic and clinical practice on this topic.

As in any systematic review, publication bias is a risk. However, we will apply no exclusion criteria regarding statistical significance or nullity of findings (where modelling is required to estimate prevalence). We mitigate the risk of selection bias by including articles in multiple languages, but cannot include all languages (eg, French). We do not have exclusion criteria regarding treatment status or publication year, which also helps mitigate risk of selection bias. We do not include “grey” literature, such as government reports. These reports may or may not contain government estimates of burden of disease. However, a recent study found limited grey literature on pediatric surgical condition policy so this is unlikely.⁴⁴

We will search seven databases to lessen the risk of not identifying all relevant articles. We use the CoCoPop framework to support equal application of inclusion and exclusion criteria across potential articles. To increase transparency in methodologies and in reporting results, we will use the PROSPERO registered protocol, this protocol paper, and the PRISMA-P (2015) guidelines to guide our review methodology and reporting of results. Finally, we critically assess each study to be included in the review for bias on the same nine indicators using the JBI Critical Appraisal Checklist for Studies reporting Prevalence Data.^40,42

Conclusion

This systematic review of major GI anomaly prevalence will be the largest systematic review, inclusive of the most GI anomalies, to date. These results are essential to informing surgical resource allocation, policy, intervention planning, and research for children globally. The difference in mortality between LMICs and HICs for these conditions is stark, and investigation of these disparities also requires investigation of baseline prevalence and prevalence of potential exposures.² Further, compilation of knowledge on current and historical birth prevalence of these conditions will support researchers, policy makers, clinicians, and public health personnel in surgical epidemiology and clinical practice.

Supplemental Material

sj-docx-1-gph-10.1177_30502225251283177 – Supplemental material for Systematic Review of the Prevalence of Gastrointestinal Congenital Anomalies: A Global and Regional Review Protocol

Supplemental material, sj-docx-1-gph-10.1177_30502225251283177 for Systematic Review of the Prevalence of Gastrointestinal Congenital Anomalies: A Global and Regional Review Protocol by Kelsey R. Landrum, Pamela Espinoza, Cesia Cotache-Condor, Henry E. Rice, Brian W. Pence, Mark G. Shrime, Chris Agala, Jessie K. Edwards and Emily R. Smith in Sage Open Pediatrics

Footnotes

Acknowledgements

The authors would like to acknowledge those researchers already publishing on these topics and the participants of these studies, which make this review possible.

Author Contributions

KRL conceptualized the search strategy, with support from HER and ERS. KRL will conduct all abstract screenings. KRL and PEG will conduct full-text screenings and bias assessments. KRL will compile results and lead preparation of the manuscript reporting results with significant support from all coauthors. All coauthors reviewed this manuscript and provided substantial recommendations and support to develop the protocol, develop the protocol manuscript, and revise the protocol manuscript. ERS, HER, and BWP will supervise this systematic review.

Data Availability Statement

All results will be made publicly available, with the goal of publishing in an open-access journal. Data in the results paper for this systematic review are able to be ascertained via the tables in the manuscript and the original articles included in the review. Raw data tables may also be requested from the corresponding author.

Declaration of Conflicting Interests

The author(s) declared no potential conflicts of interest with respect to the research, authorship, and/or publication of this article.

Funding

The author(s) received no financial support for the research, authorship, and/or publication of this article.

Ethical Statement

This study will not collect or include patient health information (PHI). No data collection from human subjects is involved in this study. As such, IRB approval is not needed.

Informed Consent/Patient Consent

This study will not collect or include any patient health information (PHI). No data collection from human subjects is involved in this study. No analysis, subgroup analysis, or reporting of results will contain identifying information. As, such informed, patient consent is not needed.

Trial Registration Number/Date

PROSPERO CRD42023460268.

ORCID iD

Kelsey R. Landrum

Supplemental Material

Supplemental material for this article is available online.

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