Building a Minimal Long-Term-Follow-Up Data Set for Newborn Screening

Version 1 : Received: 27 February 2024 / Approved: 28 February 2024 / Online: 28 February 2024 (08:08:13 CET)
Version 2 : Received: 12 March 2024 / Approved: 12 March 2024 / Online: 12 March 2024 (08:14:19 CET)
Version 3 : Received: 22 April 2024 / Approved: 23 April 2024 / Online: 24 April 2024 (03:49:20 CEST)

How to cite: Kellar-Guenther, Y.; Barringer, L.; Raboin, K.; Nichols, G.; Chou, K.Y.; Nguyen, K.; Burke, A.R.; Fawbush, S.; Meyer, J.B.; Dorsey, M.; Brower, A.; Chan, K.; Lietsch, M.; Taylor, J.; Caggana, M.; Sontag, M.K. Building a Minimal Long-Term-Follow-Up Data Set for Newborn Screening. Preprints 2024, 2024021612. https://doi.org/10.20944/preprints202402.1612.v2 Kellar-Guenther, Y.; Barringer, L.; Raboin, K.; Nichols, G.; Chou, K.Y.; Nguyen, K.; Burke, A.R.; Fawbush, S.; Meyer, J.B.; Dorsey, M.; Brower, A.; Chan, K.; Lietsch, M.; Taylor, J.; Caggana, M.; Sontag, M.K. Building a Minimal Long-Term-Follow-Up Data Set for Newborn Screening. Preprints 2024, 2024021612. https://doi.org/10.20944/preprints202402.1612.v2

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MDPI and ACS Style

Kellar-Guenther, Y.; Barringer, L.; Raboin, K.; Nichols, G.; Chou, K.Y.; Nguyen, K.; Burke, A.R.; Fawbush, S.; Meyer, J.B.; Dorsey, M.; Brower, A.; Chan, K.; Lietsch, M.; Taylor, J.; Caggana, M.; Sontag, M.K. Building a Minimal Long-Term-Follow-Up Data Set for Newborn Screening. Preprints 2024, 2024021612. https://doi.org/10.20944/preprints202402.1612.v2

APA Style

Kellar-Guenther, Y., Barringer, L., Raboin, K., Nichols, G., Chou, K.Y., Nguyen, K., Burke, A.R., Fawbush, S., Meyer, J.B., Dorsey, M., Brower, A., Chan, K., Lietsch, M., Taylor, J., Caggana, M., & Sontag, M.K. (2024). Building a Minimal Long-Term-Follow-Up Data Set for Newborn Screening. Preprints. https://doi.org/10.20944/preprints202402.1612.v2

Chicago/Turabian Style

Kellar-Guenther, Y., Michele Caggana and Marci K. Sontag. 2024 "Building a Minimal Long-Term-Follow-Up Data Set for Newborn Screening" Preprints. https://doi.org/10.20944/preprints202402.1612.v2

Abstract

Newborn screening (NBS) is hailed as a public health success, but little is known about what happens to these children after diagnosis. There has been difficulty gathering long-term follow-up (LTFU) data consistently, reliably, and with minimal effort by state NBS programs from clinicians. Six programs have been working towards a core minimal LTFU dataset, starting with data elements proposed by the Advisory Committee on Heritable Disorders in Newborns and Children’s Follow-up and Treatment workgroup (ACHDNC FUTR). This minimal data set could begin to address the impact of early diagnosis by NBS. After three rounds of data collection and revision to a data collection tool that defines the minimal LTFU data elements, the group agreed that it was most important for the dataset to capture two items for the denominator--diagnosis and if the child moved or died—with three outcomes: if the child was still alive, if the child had contact with a specialist, and if they received appropriate care specific to their diagnosis within the year. All six programs were able to provide these data. In 2022, about 83.8% (563/672) of the children in these LTFU programs were alive, 92.0% saw a specialist, and 87.7% received appropriate care.

Keywords

newborn screening; public health; equity; long-term follow-up data

Subject

Public Health and Healthcare, Public Health and Health Services

Copyright: This is an open access article distributed under the Creative Commons Attribution License which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

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