# Methodological and Procedural Considerations for Developing Decision Analytic Models to Assess the Health Economic Impacts of Newborn Bloodspot Screening: A Systematic Methodological Review

**Authors:** Jim Chilcott, Alice Bessey, James R. Bonham, Iván Castilla-Rodríguez, Sarah Davis, David Elliman, Sara Hunt, Chris Hyde, Silvia Lombardo, Jason Madan, John Marshall, Joan Morris, Katherine Payne, Oliver Rivero-Arias, Bethany Shinkins, Graham Shortland, Susan Spillane, Anthea Sutton, Sian Taylor-Phillips, Cristina Visintin

PMC · DOI: 10.3390/ijns11040096 · International Journal of Neonatal Screening · 2025-10-17

## TL;DR

This paper reviews methods for economic modeling in newborn screening to help policymakers make better decisions.

## Contribution

The study systematically identifies methodological challenges and provides recommendations for economic modeling in newborn bloodspot screening.

## Key findings

- Sixteen methodological studies were identified and analyzed for challenges in newborn screening economic modeling.
- Recommendations were developed for decision processes, model structure, data estimation, and policy considerations.
- Stakeholder workshops helped refine the identification of challenges and recommendations.

## Abstract

This methodological review identifies challenges in the development of health economic evaluations of newborn bloodspot screening (NBS) interventions and their consideration in NBS policy making. A systematic review of health economics methodological studies in NBS and stakeholder consultation was undertaken. The intervention under examination was defined as health economic decision analytic modelling used as decision support to NBS policy makers. An iterative search strategy was used to identify studies, and a data extraction framework was based upon a simple decision analytic model structure for the NBS decision problem. Synthesis was facilitated by two stakeholder workshops, which focused on ensuring the complete identification of challenges and developing recommendations. Sixteen methodological studies were identified. Data were extracted on challenges in decision criteria, decision variables, decision problem scope, defining model structure, selecting modelling method, the target condition, the screening test/protocol, outcome nodes, and other categories. Recommendations are made concerning supporting NBS decision making, NBS economic model structure and methods, data and estimation of model parameters, and overarching considerations. Recommendations for decision processes and methods research are put forward for the consideration of NBS policy makers and commissioners of research.

## Full-text entities

- **Diseases:** Congenital Anomaly (MESH:D000013), Leukodystrophy (MESH:D007966), Duchenne muscular dystrophy (MESH:D020388), SCID (MESH:D016511), Prostate Cancer (MESH:D011471), NBS (MESH:D006475), death (MESH:D003643), injury to (MESH:D014947), Cancer (MESH:D009369), inborn errors of the metabolism (MESH:D008661), medium chain acyl-CoA dehydrogenase deficiency (MESH:C536038)
- **Species:** Homo sapiens (human, species) [taxon 9606]

## Full text

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## Figures

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## References

57 references — full list in the complete paper: https://tomesphere.com/paper/PMC12550984/full.md

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Source: https://tomesphere.com/paper/PMC12550984