Establishing risk factors and outcomes for congenital hypothyroidism with gland in situ using population-based data linkage methods: study protocol.
Authors
Knowles, Rachel
Peters, Catherine
Aitkenhead, Helen
Ifederu, Adeboye
Schoenmakers, Nadia
Sebire, Neil J
Walker, Erin
Hardelid, Pia
Publication Date
2022-03Journal Title
BMJ Paediatr Open
ISSN
2399-9772
Publisher
BMJ
Volume
6
Issue
1
Language
en
Type
Article
This Version
VoR
Metadata
Show full item recordCitation
Ruiz Nishiki, M., Cabecinha, M., Knowles, R., Peters, C., Aitkenhead, H., Ifederu, A., Schoenmakers, N., et al. (2022). Establishing risk factors and outcomes for congenital hypothyroidism with gland in situ using population-based data linkage methods: study protocol.. BMJ Paediatr Open, 6 (1) https://doi.org/10.1136/bmjpo-2021-001341
Abstract
INTRODUCTION: There has been an increase in the birth prevalence of congenital hypothyroidism (CH) since the introduction of newborn screening, both globally and in the UK. This increase can be accounted for by an increase in CH with gland in situ (CH-GIS). It is not known why CH-GIS is becoming more common, nor how it affects the health, development and learning of children over the long term. Our study will use linked administrative health, education and clinical data to determine risk factors for CH-GIS and describe long-term health and education outcomes for affected children. METHODS AND ANALYSIS: We will construct a birth cohort study based on linked, administrative data to determine what factors have contributed to the increase in the birth prevalence of CH-GIS in the UK. We will also set up a follow-up study of cases and controls to determine the health and education outcomes of children with and without CH-GIS. We will use logistic/multinomial regression models to establish risk factors for CH-GIS. Changes in the prevalence of risk factors over time will help to explain the increase in birth prevalence of CH-GIS. Multivariable generalised linear models or Cox proportional hazards regression models will be used to assess the association between type of CH and school performance or health outcomes. ETHICS AND DISSEMINATION: This study has been approved by the London Queen Square Research Ethics Committee and the Health Research Authority's Confidentiality Advisory Group CAG. Approvals are also being sought from each data provider. Obtaining approvals from CAG, data providers and information governance bodies have caused considerable delays to the project. Our methods and findings will be published in peer-reviewed journals and presented at academic conferences.
Keywords
Protocol, 1506, epidemiology, ethics, neonatology
Sponsorship
Wellcome Trust (219496/Z/19/Z)
NIHR Great Ormond Street Hospital BRC (19PE26)
Identifiers
bmjpo-2021-001341
External DOI: https://doi.org/10.1136/bmjpo-2021-001341
This record's URL: https://www.repository.cam.ac.uk/handle/1810/335669
Rights
Licence:
http://creativecommons.org/licenses/by-nc/4.0/
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