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Comprehensive investigation of congenital anomalies in cerebral palsy: protocol for a European-Australian population-based data linkage study (The Comprehensive CA-CP Study)
  1. Shona Goldsmith1,
  2. Guiomar Garcia Jalon2,
  3. Nadia Badawi1,3,
  4. Eve Blair4,
  5. Ester Garne5,
  6. Catherine Gibson6,
  7. Sarah McIntyre1,
  8. Heather Scott6,
  9. Hayley Smithers-Sheedy1,
  10. Guro L Andersen7
  1. 1 Cerebral Palsy Alliance Research Institute, Discipline of Child and Adolescent Health, The University of Sydney, Sydney, New South Wales, Australia
  2. 2 Northern Ireland Cerebral Palsy Register, School of Nursing and Midwifery, Queen’s University Belfast, Royal Group of Hospitals, Belfast, UK
  3. 3 Grace Centre for Newborn Care, The Children’s Hospital at Westmead, Sydney, New South Wales, Australia
  4. 4 Telethon Kids Institute, University of Western Australia, Perth, Australia
  5. 5 Paediatric Department, Hospital Lillebaelt Kolding, Kolding, Denmark
  6. 6 South Australian Birth Defects Register, Women’s and Children’s Hospital, Women’s and Children’s Health Network, Adelaide, South Australia, Australia
  7. 7 The Cerebral Palsy Register of Norway, Vestfold Hospital Trust, Tønsberg, Norway
  1. Correspondence to Ms Shona Goldsmith; sgoldsmith{at}cerebralpalsy.org.au

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.

  • epidemiology
  • paediatric neurology

This is an open access article distributed in accordance with the Creative Commons Attribution Non Commercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited, appropriate credit is given, any changes made indicated, and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/.

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Footnotes

  • Contributors SM, GA, EG, EB, SG, GGJ, NB, CG, HS and HS-S contributed to the design of the study. SG is a PhD scholar on the study, supervised by SM and NB. SG, SM and NB are investigators on the NHMRC grants and Cerebral Palsy Alliance Research Foundation PG1215 grants, and SG, SM, GA, EG, GGJ, EB, CG, HS and HS-S are investigators on the Cerebral Palsy Alliance Research Foundation PG2816 grant. SG developed and conducted the original survey to identify potential participating regions, with important input from SM, GGJ, NB, EB, EG, CG, HS, HS-S and GA. SG wrote the first draft of the ethics application, study protocol and this manuscript, with supervision from SM and critical review and contributions from GGJ, NB, EB, EG, CG, HS, HS-S and GA. All authors (SG, GGJ, NB, EB, EG, CG, SM, HS, HS-S and GA) read and approved the final manuscript.

  • Funding This work is supported by the National Health and Medical Research Council of Australia (NHMRC) (Early Career Fellowship 1111270-SM) (Postgraduate research scholarship 1113806-SG) and the Cerebral Palsy Alliance Research Foundation (Project grants PG1215 and PG2816). SM, NB, SG and HS-S receive salary support from the Cerebral Palsy Alliance.

  • Disclaimer The NHMRC and Cerebral Palsy Alliance have no role in the design of the study or data collection, analysis, interpretation of data or manuscript writing.

  • Competing interests None declared.

  • Patient consent Not required.

  • Ethics approval Cerebral Palsy Alliance Human Research Ethics Committee (EC00402), a National Health and Medical Research Council approved committee in Australia.

  • Provenance and peer review Not commissioned; externally peer reviewed.

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