Comprehensive investigation of congenital anomalies in cerebral palsy: Protocol for a European-Australian population-based data linkage study (The Comprehensive CA-CP Study)

Shona Goldsmith, Guiomar Garcia Jalon, Nadia Badawi, Eve Blair, Ester Garne, Catherine Gibson, Sarah McIntyre, Heather Scott, Hayley Smithers-Sheedy, Guro L. Andersen

Research output: Contribution to journalArticle

Abstract

Introduction Cerebral palsy (CP), an umbrella term for non-progressive conditions of cerebral origin resulting in motor impairments, is collectively the most common cause of physical disability in childhood. Cerebral and/or non-cerebral congenital anomalies are present in 15%-40% of children with CP. In order to identify effective prevention strategies for this substantial proportion of CP, a comprehensive understanding of the epidemiology of these congenital anomalies is required. International collaboration is needed, as previous attempts have fallen short due to a lack of power, since the anomalies are individually rare and CP comprises many clinical descriptions. The aim of this study is to generate new knowledge about the aetiologies of CP through a focused investigation into the role of congenital anomalies. Methods and analysis This collaborative, population-based data linkage study includes nine geographic regions (six in Europe, three in Australia) served by both congenital anomaly and CP registers. Register data for children with CP (both with and without congenital anomalies) and children with specific congenital anomalies (without CP) born between 1991 and 2009 will be linked and de-identified within each region. The resulting linked data sets will be quality assured, recoded, harmonised and then pooled into one data set. Analysis of the combined data set will include: frequencies/proportions of congenital anomalies and outcomes (type of CP, severity, impairments); descriptive analyses comparing timing of congenital anomaly development and brain injury/abnormality responsible for CP; ORs to calculate the odds of CP following a specific congenital anomaly; and identification of anomalies on causal pathways to CP. Ethics and dissemination Ethics approval for this collaborative study, The Comprehensive CA-CP Study, has been obtained from the Cerebral Palsy Alliance Human Research Ethics Committee (EC00402). Study findings will be disseminated at conferences and published in peer-reviewed journals, and recommendations will be made regarding the collection and classification of congenital anomaly data by CP registers.

Original languageEnglish
Article numbere022190
JournalBMJ Open
Volume8
Issue number7
DOIs
Publication statusPublished - 1 Jul 2018

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Cerebral Palsy
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Research Ethics Committees
Brain Injuries

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Goldsmith, Shona ; Garcia Jalon, Guiomar ; Badawi, Nadia ; Blair, Eve ; Garne, Ester ; Gibson, Catherine ; McIntyre, Sarah ; Scott, Heather ; Smithers-Sheedy, Hayley ; Andersen, Guro L. / Comprehensive investigation of congenital anomalies in cerebral palsy : Protocol for a European-Australian population-based data linkage study (The Comprehensive CA-CP Study). In: BMJ Open. 2018 ; Vol. 8, No. 7.
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abstract = "Introduction Cerebral palsy (CP), an umbrella term for non-progressive conditions of cerebral origin resulting in motor impairments, is collectively the most common cause of physical disability in childhood. Cerebral and/or non-cerebral congenital anomalies are present in 15{\%}-40{\%} of children with CP. In order to identify effective prevention strategies for this substantial proportion of CP, a comprehensive understanding of the epidemiology of these congenital anomalies is required. International collaboration is needed, as previous attempts have fallen short due to a lack of power, since the anomalies are individually rare and CP comprises many clinical descriptions. The aim of this study is to generate new knowledge about the aetiologies of CP through a focused investigation into the role of congenital anomalies. Methods and analysis This collaborative, population-based data linkage study includes nine geographic regions (six in Europe, three in Australia) served by both congenital anomaly and CP registers. Register data for children with CP (both with and without congenital anomalies) and children with specific congenital anomalies (without CP) born between 1991 and 2009 will be linked and de-identified within each region. The resulting linked data sets will be quality assured, recoded, harmonised and then pooled into one data set. Analysis of the combined data set will include: frequencies/proportions of congenital anomalies and outcomes (type of CP, severity, impairments); descriptive analyses comparing timing of congenital anomaly development and brain injury/abnormality responsible for CP; ORs to calculate the odds of CP following a specific congenital anomaly; and identification of anomalies on causal pathways to CP. Ethics and dissemination Ethics approval for this collaborative study, The Comprehensive CA-CP Study, has been obtained from the Cerebral Palsy Alliance Human Research Ethics Committee (EC00402). Study findings will be disseminated at conferences and published in peer-reviewed journals, and recommendations will be made regarding the collection and classification of congenital anomaly data by CP registers.",
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Goldsmith, S, Garcia Jalon, G, Badawi, N, Blair, E, Garne, E, Gibson, C, McIntyre, S, Scott, H, Smithers-Sheedy, H & Andersen, GL 2018, 'Comprehensive investigation of congenital anomalies in cerebral palsy: Protocol for a European-Australian population-based data linkage study (The Comprehensive CA-CP Study)' BMJ Open, vol. 8, no. 7, e022190. https://doi.org/10.1136/bmjopen-2018-022190

Comprehensive investigation of congenital anomalies in cerebral palsy : Protocol for a European-Australian population-based data linkage study (The Comprehensive CA-CP Study). / Goldsmith, Shona; Garcia Jalon, Guiomar; Badawi, Nadia; Blair, Eve; Garne, Ester; Gibson, Catherine; McIntyre, Sarah; Scott, Heather; Smithers-Sheedy, Hayley; Andersen, Guro L.

In: BMJ Open, Vol. 8, No. 7, e022190, 01.07.2018.

Research output: Contribution to journalArticle

TY - JOUR

T1 - Comprehensive investigation of congenital anomalies in cerebral palsy

T2 - Protocol for a European-Australian population-based data linkage study (The Comprehensive CA-CP Study)

AU - Goldsmith, Shona

AU - Garcia Jalon, Guiomar

AU - Badawi, Nadia

AU - Blair, Eve

AU - Garne, Ester

AU - Gibson, Catherine

AU - McIntyre, Sarah

AU - Scott, Heather

AU - Smithers-Sheedy, Hayley

AU - Andersen, Guro L.

PY - 2018/7/1

Y1 - 2018/7/1

N2 - Introduction Cerebral palsy (CP), an umbrella term for non-progressive conditions of cerebral origin resulting in motor impairments, is collectively the most common cause of physical disability in childhood. Cerebral and/or non-cerebral congenital anomalies are present in 15%-40% of children with CP. In order to identify effective prevention strategies for this substantial proportion of CP, a comprehensive understanding of the epidemiology of these congenital anomalies is required. International collaboration is needed, as previous attempts have fallen short due to a lack of power, since the anomalies are individually rare and CP comprises many clinical descriptions. The aim of this study is to generate new knowledge about the aetiologies of CP through a focused investigation into the role of congenital anomalies. Methods and analysis This collaborative, population-based data linkage study includes nine geographic regions (six in Europe, three in Australia) served by both congenital anomaly and CP registers. Register data for children with CP (both with and without congenital anomalies) and children with specific congenital anomalies (without CP) born between 1991 and 2009 will be linked and de-identified within each region. The resulting linked data sets will be quality assured, recoded, harmonised and then pooled into one data set. Analysis of the combined data set will include: frequencies/proportions of congenital anomalies and outcomes (type of CP, severity, impairments); descriptive analyses comparing timing of congenital anomaly development and brain injury/abnormality responsible for CP; ORs to calculate the odds of CP following a specific congenital anomaly; and identification of anomalies on causal pathways to CP. Ethics and dissemination Ethics approval for this collaborative study, The Comprehensive CA-CP Study, has been obtained from the Cerebral Palsy Alliance Human Research Ethics Committee (EC00402). Study findings will be disseminated at conferences and published in peer-reviewed journals, and recommendations will be made regarding the collection and classification of congenital anomaly data by CP registers.

AB - Introduction Cerebral palsy (CP), an umbrella term for non-progressive conditions of cerebral origin resulting in motor impairments, is collectively the most common cause of physical disability in childhood. Cerebral and/or non-cerebral congenital anomalies are present in 15%-40% of children with CP. In order to identify effective prevention strategies for this substantial proportion of CP, a comprehensive understanding of the epidemiology of these congenital anomalies is required. International collaboration is needed, as previous attempts have fallen short due to a lack of power, since the anomalies are individually rare and CP comprises many clinical descriptions. The aim of this study is to generate new knowledge about the aetiologies of CP through a focused investigation into the role of congenital anomalies. Methods and analysis This collaborative, population-based data linkage study includes nine geographic regions (six in Europe, three in Australia) served by both congenital anomaly and CP registers. Register data for children with CP (both with and without congenital anomalies) and children with specific congenital anomalies (without CP) born between 1991 and 2009 will be linked and de-identified within each region. The resulting linked data sets will be quality assured, recoded, harmonised and then pooled into one data set. Analysis of the combined data set will include: frequencies/proportions of congenital anomalies and outcomes (type of CP, severity, impairments); descriptive analyses comparing timing of congenital anomaly development and brain injury/abnormality responsible for CP; ORs to calculate the odds of CP following a specific congenital anomaly; and identification of anomalies on causal pathways to CP. Ethics and dissemination Ethics approval for this collaborative study, The Comprehensive CA-CP Study, has been obtained from the Cerebral Palsy Alliance Human Research Ethics Committee (EC00402). Study findings will be disseminated at conferences and published in peer-reviewed journals, and recommendations will be made regarding the collection and classification of congenital anomaly data by CP registers.

KW - epidemiology

KW - paediatric neurology

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DO - 10.1136/bmjopen-2018-022190

M3 - Article

VL - 8

JO - BMJ (Open)

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