Background: Gastroschisis has a great disparity in outcomes globally with less than 4%
mortality in high-income countries (HICs) and over 90% mortality in many tertiary paediatric
surgery centres across sub-Saharan Africa (SSA), including the study centres. Low-technology,
cost-effective, evidence-based interventions are available with the potential for improved
outcomes in low-resource settings.
Aim: To develop, implement and prospectively evaluate an interventional care bundle to reduce
mortality from gastroschisis in seven tertiary paediatric surgery centres across SSA.
Study Design: A hybrid type 2 effectiveness-implementation study design will be utilised,
which focusses equally on the effectiveness of the intervention through analysing clinical
outcomes and effectiveness of the implementation through analysis of service delivery and
implementation outcomes.
Methods: Using current literature an evidence-based, low-technology interventional care
bundle has been developed with both pre-hospital and in-hospital components. During 2018 a
systematic review, qualitative study and Delphi process will provide further evidence in
order to optimise the interventional bundle and implementation strategy for use in
low-resource environments. The interventional care bundle has core components, which will
remain consistent across the seven sites, and adaptable components, which will be determined
through in-country co-development meetings with all relevant members of the
multi-disciplinary team. Across the seven study sites it is estimated that 180-289 patients
will be included in the study, which has the potential to determine a difference in pre- and
post-intervention mortality between 10-15%.
Implementation will take place over a 4-week period at each of the study sites between April
- December 2019. Pre- and post-interventional data will be collected on clinical, service
delivery and implementation outcomes between September 2018 to October 2020 (2-years of
patient recruitment at each site with a 30-day post primary intervention follow-up period).
The primary clinical outcome will be all-cause, in-hospital mortality. Secondary outcomes
include the occurrence of a major complication, length of hospital stay and time to full
enteral feeds. Service delivery outcomes include time to hospital and primary intervention
and adherence to the pre-hospital and in-hospital management protocols. Implementation
outcomes will include acceptability, adoption, appropriateness, feasibility, fidelity,
coverage, cost and sustainability.
Expected Results: Pre- and post-intervention clinical outcomes will be compared using
Chi-squared analysis, unpaired t-test and/ or Mann-Whitney test. Time-series analysis will be
undertaken using Statistical Process Control to identify significant trends and shifts in
outcome overtime. Multivariate logistic regression analysis will be used to identify both
clinical and implementation factors affecting outcome with adjustment for confounders. P<0.05
will be deemed significant.
Outcome: This will be the first multi-centre interventional study to the investigator's
knowledge aimed at reducing mortality from gastroschisis in low-resource settings. If
successful, detailed evaluation of both the clinical and implementation components of the
study will allow sustainability in the study sites and further scale-up.