4CHILD - Four Counties Database of Cerebral Palsy, Vision Loss and Hearing Loss in Children (Berkshire, Buckinghamshire, Northamptonshire, Oxfordshire)

The University of Oxford received data from ONS and subsequently NHS Digital from 2003 onwards for the purpose of 4Child. 4Child is a database which was established in 1984 to collect information about children with cerebral palsy and/or severe vision loss and/or hearing loss born to residents of Berkshire, Buckinghamshire, Northampton and Oxfordshire. It was used as a resource to carry out surveillance, research and service evaluation on the causes and consequences of the three potentially disabling conditions of cerebral play, vision loss and hearing loss; to access the effectiveness of interventions during pregnancy and soon after births; as well as to assess the need for services to support affected children.

Formerly called the Oxford Register of Early Childhood Impairments (ORECI) 4Child has been collecting information about children with cerebral palsy (CP), sensorineural deafness or severe vision loss born to residents of Berkshire, Buckinghamshire, Northamptonshire and Oxfordshire since 1984 and has information about 2,700 children with one or more of these impairments.

The register was set up against a background of uncertainty of the contribution of increased numbers of low birth weight survivors on the numbers of disabled children in the population. At the time, there were no routinely collected and easily accessible data on early childhood morbidity and so the register was set up as a framework to examine clinical associations of disabling conditions, to assess services, and to assess the effectiveness of perinatal intervention.

The aims of the database when it was established were to:

i. Monitor the prevalence of cerebral palsy, vision loss and hearing loss in children from 1984 onwards in the four counties of Berkshire, Buckinghamshire, Northamptonshire and Oxfordshire.

ii. To provide a research platform and support research and service audit initiatives using data from 4Child, in order to contribute to knowledge and understand of the causes and consequences of the impairments and how they might be prevented and better managed.

iii. To develop links with other researchers and collaborate in research within the UK, Europe and other centre around the world.

The 4Child database was run by the 4Child research team within the National Perinatal Epidemiology Unit (NPEU) at the University of Oxford.

The operation of the 4Child database involved receiving information about children who were born or lived during in early childhood (up to age 5 years) in one of the four counties, who were suspected and then diagnosed as having one of the three impairments: cerebral palsy, vision loss or hearing loss; of note some children have more than one of these impairments.

The information about the affected children was provided by any health professional who came into contact with the child during early childhood – so called multi-source notification. Multi-source notification is the mechanism used to ensure that no children were missed. The 4Child team at the University of Oxford also received information from the Office for National Statistics about any children who died. This enabled the team to carry out research into death rates for children with these impairments and to understand the reasons why some children and adults with these impairments die earlier than would normally be expected.

The information received about the children was identifiable personal data and included the names, addresses and dates of birth of the children. Receiving identifiable information was necessary to enable the team to identify when a child was notified to the team more than once so that that any duplicate notifications could be removed. The identifiable information was also used to enable collection of follow-up information about the children to obtain details about the extent and impact of their impairment. Using the information, with relevant regulatory permissions, the team were able to contact some families to invite them to participate in research.

Funding was provided to support the work of 4Child from a number of sources over the years. Latterly it was funded by the Department of Health but this final grant ended in 2010. The 4Child research team was disbanded once the funding ran out. Only one member of the research team remains in the NPEU.

As it was not possible to obtain further funds the database closed to registrations of newly diagnosed children in 2010. Nevertheless, the existing database is an invaluable and unique source of whole population data about children with these three important impairments as currently there is no other similar information collected in England in this systematic way.

In view of the fact that was not possible to obtain further funding to continue the active work of the database a decision was recently made to remove all the identifiable personal information on the database and this process was completed in August 2019. This means that all names, addresses, postcodes, dates of birth, dates of notification and diagnosis, where applicable dates of death, and all other date-related information held in the database have been deleted. All relevant date information was replaced with age at the event. The information held includes the cause of death.

The database will be securely archived to comply with good practice and also to preserve the resource for potential future research subject to the necessary approvals including an application to NHS Digital.

The need to keep the data in the long term will be subject to review. Initially, the mortality data will be retained for two years. Before the end of this period the University of Oxford will decide whether there is a continuing purpose for the mortality data which would justify keeping the information for longer. In the event a decision is made not to continue holding the data from death certificates, this information will be deleted from the database. At that stage the University of Oxford will also review the value of continuing to hold the rest of the data in the 4Child database.

This Data Sharing Agreement will permit the retention of the data previously supplied by NHS Digital and predecessor organisations. No other processing activities will be permitted. No new data will be supplied and/or linked to the dataset and no uses of the data (e.g. for research) are permitted.

This 4Child programme is now completed and closed.

The 4Child register was originally a disease register to enable surveillance, research and service evaluation to be carried out. The register is no long active.

This Data Sharing Agreement permits the retention of the data previously supplied by NHS Digital and predecessor organisations, which has now been pseudonymised.

This agreement permits processing of the data for the purpose of secure storage and back up.

This agreement does not permit any further processing that involves analysis, linkage, onward sharing. If further data processing is required the applicant must submit an amendment request to NHS Digital before data is accessed.

The data originally requested from NHS Digital was for use in the 4Child Four Counties Database of Cerebral Palsy, Vision Loss and Hearing Loss (PIAG 4-09 b)/2003).

The intention is to archive the pseudonymised dataset for the immediate future to preserve it as a resource to be used in future research subject to funding and the necessary approvals.

Any further analysis will only take place following an amendment to this Agreement that would allow further processing of the data.

The data will be stored in the National Perinatal Epidemiology Unit (where it has been stored since inception). The NPEU is part of the Nuffield Department of Population Health at the University of Oxford. The data will be stored on NDPH servers since this is the department in which the NPEU sits.

The 4Child programme is now completed and closed.

The following description of outputs is for information only and the processing of the data occurred before this current Agreement.

No new outputs will be produced using the data under this agreement.

The 4Child data is one of the few remaining complete existing population-based dataset of cerebral palsy, vision loss and hearing loss in England from which the impact of these impairments on mortality and long-term survival can be investigated. Other such data (including the longstanding Liverpool cerebral palsy register) were deleted in their totality some time ago. The 4Child database therefore is a valuable data source of information.

At present there is no plan (or funding) to process these data.

Future analysis on survival may be required, at which point an amendment application to process the data will be submitted to NHS Digital

The information on the register has been used to monitor trends and variations in birthweight specific prevalence rates of these three conditions.

4Child was one of only three active registers in the UK that carried out surveillance and monitoring of cerebral palsy which had a continuous unbroken data collection since the 1980s. Until closure of the register provided annual surveillance reports of rates of cerebral palsy overall and in particular high risk subgroups including low birth weight infants and children born as one of a multiple pregnancy. The study also provided surveillance reports of rates of vision loss and hearing loss in children.

In addition to surveillance and monitoring activities the data from 4Child were used in various research projects including to examine the relationship between the target time for decision to delivery interval for emergency caesarean section and outcomes for the neonate and at three years, This paper and other published outputs have made an important contribution to the understanding of the causes and consequence of cerebral palsy, vision loss and hearing loss.

Examples of publications are as follows:

Greenwood C, Yudkin PL, Sellers S, Impey L, Doyle P, Johnson A. Why is there a modifying effect of gestational age on risk factors for cerebral palsy? Arch Dis Child. 2005;90:F141-F6.

Morris C, Kurinczuk JJ, Fitzpatrick R. Child or family assessed measures of activity performance and participation for children with cerebral palsy: a structured review. Child: Care Health & Development. 2005;31(4):397-407.

Jarvis S, Glinianaia SV, Arnaud C, Fauconnier J, Johnson A, McManus V, Topp M, Uvebrant P, Cans C, Krageloh-Mann I, SCPE Collaboration of European Cerebral Palsy Registers. Case gender and severity in cerebral palsy varies with intrauterine growth. Archives of Diseases in Childhood. 2005;90:474-9.

Morris C, Kurinczuk JJ, Fitzpatrick R, Rosenbaum PL. Who best to make the assessment? Professionals' and families' classifications of gross motor function in cerebral palsy are highly consistent. Arch Dis Child. 2006;91:675-9.

McManus V, Guillem P, Surman G, Cans C. SCPE work, standardisation and definition - An overview of the activities of SCPE a collaboration of European CP Registers. Chinese Journal of Contemporary Pediatrics. 2006;8(4):261-5.

Morris C, Kurinczuk JJ, Fitzpatrick R, Rosenbaum PL. Do the abilities of children with cerebral palsy explain their activities and participation? Dev Med Child Neurol. 2006;48:954-61.

Morris C, Kurinczuk JJ, Fitzpatrick R, Rosenbaum PL. Reliability of the Manual Ability Classification System for children with cerebral palsy. Dev Med Child Neurol. 2006;48:950-3.

Platt MJ, Cans C, Johnson A, Surman G, Topp M, Torrioli MG, Krageloh-Mann I. Trends in cerebral palsy among infants of very low birthweight (<1500 g) or born prematurely (<32 weeks) in 16 European centres: a database study. The Lancet. 2007;369(9555):43-50.

Morris C. Definition and classification of cerebral palsy: a historical perspective. Dev Med Child Neurol. 2007;49(s2):3-7.

MacKenzie IZ, Shah M, Lean K, Dutton S, Newdick H, Tucker D. Management of shoulder dystocia trends in incidence and maternal and neonatal morbidity. Obstet Gynecol. 2007;110:1059-68.

Kenyon S, Pike K, Jones DR, Brocklehurst P, Marlow N, Salt A, Taylor DJ. Childhood outcomes after prescription of antibiotics to pregnant women with spontaneous preterm labour: 7-year follow-up of the ORACLE II trial. The Lancet. 2008;372(9646):1319-27.

Sellier E, Surman G, Himmelmann K, Andersen G, Colver A, Krägeloh-Mann I, De-la-Cruz J, Cans C. Trends in prevalence of cerebral palsy in children born with a birthweight of 2,500 g or over in Europe from 1980 to 1998. Eur J Epidemiol. 2010;25(9):635-42.

Andersen GL, Romundstad P, De La Cruz J, Himmelmann K, Sellier E, Cans C, Kurinczuk JJ, Vik T. Cerebral palsy among children born moderately preterm or at moderately low birthweight between 1980 and 1998: a European register-based study. Dev Med Child Neurol. 2011;53(10):913-9.

Pearson GA, Kelly B, Russell R, Dutton S, Kurinczuk JJ, MacKenzie IZ. Target decision to delivery intervals for emergency caesarean section based on neonatal outcomes and three year follow-up. Eur J Obstet Gynecol Reprod Biol. 2011;159(2):276-81.

This data will be retained to comply with University of Oxford guidance and policy on good clinical practice. It also preserves a unique database of children with cerebral palsy, vision loss and hearing loss in England, which could still yield future benefits.

The 4Child register contributed vital data to enable important research into the trends, aetiology and consequences of cerebral palsy to be carried out in: the 4Child region, across the UK as part of the UKCP collaboration and across Europe as part of the SCPE collaboration.

The other key contribution that the 4Child data made was tracking the trends in the prevalence of cerebral palsy in the four counties. These were published in a series of annual reports and made available to clinical staff, policy makers and service planners. One of the important uses of the prevalence data was in the planning of service provision for children with cerebral palsy based on the number of affected children and the extent and severity of their impairments.

Over 90 peer-reviewed research papers using data from the 4Child register were published prior to the closure of the register.

Examples of the research conducted using 4Child data include:

1. A study to describe trends in the prevalence of cerebral palsy in preterm and low birthweight infants which demonstrated that over the period 1980 to 1996 the prevalence fell indicating the beneficial impacts of improved maternity and neonatal care over this period (Platt et al 2007).

2. A aetiological study to investigate the effects of gestational age at birth on the risk of cerebral palsy which identified and quantified the role of inflammatory factors which influenced the gestational-age specific risks, including, intrapartum hypoxia, neonatal sepsis, pre-eclampsia (Greenwood et al 2005).

3. A study to describe the long term consequences of cerebral palsy and the impact of cerebral palsy on subsequent risk of premature mortality and the predictors of mortality. This study demonstrated that the number and severity of impairments were the strongest predictors of risk of death (Hemming et al 2005).