Protocol: A systematic review and meta-analysis of the role of fetal and infantile environmental exposure in etiopathogenesis of infantile hypertrophic pyloric stenosis.

Ozlem Boybeyi-Turer,Hasan Tolga Çelik,Sibel Kiran,Tutku Soyer,Umut Ece Arslan,Feridun Cahit Tanyel,Endi Lanza Galvão

doi:10.1371/journal.pone.0247003

Abstract

Infantile hypertrophic pyloric stenosis (IHPS) is one of the hallmark pediatric surgical diseases. However, its etiology remains incompletely understood. By systematically reviewing the literature, we aim to clarify the effect of the effect of occupational and environmental factors and role of nitric oxide (NO) metabolism in the etiopathogenesis of IHPS. The systematic review is drafted with the Preferred Reporting Items for Systematic Reviews and Meta-Analysis statement (PRISMA) and the Meta-analysis of Observational Studies in Epidemiology (MOOSE). Systematic literature search will be performed for the period 2000 (Jan) to 2020 (Dec) in the databases: MEDLINE, EMBASE, PubMed. The systematic search will cover the literature in English and Turkish language and will be limited to studies on human subjects. Four investigators will independently search the databases (MEDLINE, EMBASE, PubMed) according to the defined search strategy. The full-text of the selected articles will be screened independently by four reviewers, against the inclusion criteria. Descriptive data will be extracted from each study regarding: study details, methods, participants, outcomes and calculations of association for potential further statistical analysis. If meta-analysis could not be undertaken, systematic approach to analyzing the findings of included multiple studies will be described. Heterogeneity will be assessed by quantifying the inconsistency across studies using I2 statistic. Statistical analysis will be performed using Comprehensive Meta-Analysis Version 3.0 software. The p values lower than 0.05 will be considered statistically significant for all analyses.

Highlights

Infantile hypertrophic pyloric stenosis (IHPS) is one of the most frequent disorders requiring surgery in the first year of life [1, 2]
By systematically reviewing the literature, we aim to clarify the effect of environmental factors and the role of nitric oxide metabolism in IHPS origin
Once etiology and pathophysiology addressed accurately, the information will be helpful in identifying the causative risk factors and suggesting probable ways to prevent the disease

Summary

Introduction

Infantile hypertrophic pyloric stenosis (IHPS) is one of the most frequent disorders requiring surgery in the first year of life [1, 2]. The incidence of IHPS has been reported as 2–4 cases per 1,000 live births with wide variation between worldwide locations, ethnic origins and seasons [1,2,3] This variability in the incidence suggests possible environmental factors being involved in the etiology of IHPS. There are inconsistent results regarding possible effects of folic acid deficiency [12], maternal smoking [13], maternal hyperthyroidism, and nalidixic acid use [14], and paternal occupation [15] Since these reports about the possible exposures in IHPS etiopathogenesis have not been reviewed before, we aimed to systematically review the literature. By systematically reviewing the literature, we aim to clarify the effect of environmental factors (except previous systematic review topics; erythromycin, macrolides and birth related factors) and the role of nitric oxide metabolism in IHPS origin. Once etiology and pathophysiology addressed accurately, the information will be helpful in identifying the causative risk factors and suggesting probable ways to prevent the disease

Materials and methods

Objectives

Study design

Conclusion