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Cost-effectiveness of newborn screening for sickle cell disease: a systematic review protocol
Biswajit Mahapatra1, Nirmalya Mukherjee1, Sajda Khatoon1
1Evidence Synthesis and Implementation for Indigenous Health: A JBI Affiliated Group, Centre for Public Health Research, Manbhum Ananda Ashram Nityananda Trust (MANT), Kolkata, West Bengal, India.
Insights
This systematic review evaluates the cost-effectiveness of targeted versus universal newborn screening for sickle cell disease. Findings will inform global newborn screening strategies and resource allocation for this serious hematologic disorder.
Area of Science:
- Public Health
- Health Economics
- Genetics
Background:
- Sickle cell disease (SCD) is a prevalent, life-threatening hematologic disorder affecting newborns globally.
- Newborn screening (NBS) for SCD is crucial but presents economic challenges.
- The financial burden of NBS impacts households and national economies.
Purpose of the Study:
- To systematically review and assess the cost-effectiveness of targeted/selective newborn screening versus universal screening for sickle cell disease.
- To compare screening strategies across diverse international settings and healthcare systems.
Main Methods:
- Systematic review of economic evaluations comparing targeted/selective and universal NBS for SCD.
- Literature search using MeSH terms like "sickle cell disease," "newborn," and "economic evaluations."
- Data extraction and methodological quality assessment using JBI tools, with results summarized using dominance ranking and GRADE for certainty of evidence.
Main Results:
- Cost-effectiveness measured by cost per case detected, QALYs gained, or DALYs averted.
- Analysis will consider resource use and expenditures to inform decision-making.
- The review synthesizes evidence on the economic value of different SCD NBS approaches.
Conclusions:
- The findings will provide evidence-based recommendations on optimal newborn screening strategies for sickle cell disease.
- This research aims to guide policymakers in resource allocation for effective SCD newborn screening programs.
- Understanding cost-effectiveness is vital for sustainable and equitable global health initiatives for sickle cell disease.
Objective:
The purpose of this systematic review is to assess the cost-effectiveness of targeted/selective newborn screening compared with universal screening for sickle cell disease across various countries and settings.
Introduction:
The incidence of sickle cell disease is a widespread and potentially fatal hematologic disorder that affects thousands of newborns worldwide. The cost of newborn screening creates a burden on households and the economy.
Inclusion Criteria:
Studies will be eligible for inclusion in the review if they focus on the cost-effectiveness of newborn screening for sickle cell disease, comparing targeted/selective screening with universal screening.
Methods:
A preliminary search of MEDLINE (PubMed) was undertaken using MeSH terms, such as sickle cell disease, newborn , and economic evaluations . Two reviewers will screen the titles, abstracts, and full text independently against the inclusion criteria. Disagreements will be resolved by discussion or with a third reviewer. To assess methodological quality, the JBI checklist for economic evaluation will be used. Data will be extracted by 2 reviewers using a modified JBI data extraction form. The JBI dominance ranking matrix for economic evaluations will be used to summarize and compare the results. Cost-effectiveness will be measured on the basis of cost per test/case detected, quality-adjusted life years gained, or disability-adjusted life years averted. The Grading of Recommendations, Assessment, Development and Evaluation (GRADE) assessment will be conducted to evaluate the certainty of economic evidence, such as use of resources and expenditures, and to incorporate the results into the decision-making process.
Review Registration:
PROSPERO CRD42017057963.
