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Research Project
EUROlinkCAT: Establishing a linked European Cohort of Children with Congenital Anomalies
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Exploring Research Priorities of Parents Who Have Children With Down Syndrome, Cleft Lip With or Without Cleft Palate, Congenital Heart Defects, or Spina Bifida Using ConnectEpeople: A Social Media Coproduction Research Study
Publication . Sinclair, Marlene; McCullough, Julie E.M.; Elliott, David; Latos-Bielenska, Anna; Braz, Paula; Cavero-Carbonell, Clara; Jamry-Dziurla, Anna; João Santos, Ana; Páramo-Rodríguez, Lucía
Background: Using social media for research purposes is novel and challenging in terms of recruitment, participant knowledge about the research process, and ethical issues. This paper provides insight into the recruitment of European parents of children with specific congenital anomalies to engage in coproduction research by using social media. Secret Facebook groups, providing optimal security, were set up for newly recruited research-aware parents (RAPs) to communicate privately and confidentially with each other and for the research team to generate questions and to interpret findings.
Objective: This study aimed to use social media for the recruitment and engagement of parents in research and to determine the research priorities of parents who have children with Down syndrome, cleft lip with or without cleft palate, congenital heart defects, and spina bifida.
Methods: The design was exploratory and descriptive with 3 phases. Phase 1 included the recruitment of RAPs and generation of research questions important to them; phase 2 was a Web-based survey, designed using Qualtrics software, and phase 3 included analysis and ranking of the top 10 research questions using an adapted James Lind Alliance approach. Simple descriptive statistics were used for analysis, and ethical approval was obtained from the Ethics Filter Committee of the Institute of Nursing and Health Research, Ulster University.
Results: The recruitment of 32 RAPs was a sensitive process, varying in the time taken to consent (mean 51 days). However, parents valued the screening approach using the State-Trait Anxiety Inventory as a measure to ensure their well-being (mean 32.5). In phase 1, RAPs generated 98 research questions. In phase 2, 251 respondents accessed the Web-based survey, 248 consented, and 80 completed the survey, giving a completeness rate of 32.3% (80/248). Most parents used social media (74/80, 92%). Social media, online forums, and meeting in person were ranked the most preferable methods for communication with support groups networks and charities. Most respondents stated that they had a good understanding of research reports (71/80, 89%) and statistics (68/80, 85%) and could differentiate among the different types of research methodologies (62/80, 78%). Phase 3 demonstrated consensus among RAPs and survey respondents, with a need to know the facts about their child's condition, future health, and psychosocial and educational outcomes for children with similar issues.
Conclusions: Social media is a valuable facilitator in the coproduction of research between parents and researchers. From a theoretical perspective, ocularcentrism can be an applicable frame of reference for understanding how people favor visual contact.
Congenital anomalies: parents' concerns and opinions towards public health policies and improved effectiveness of health services
Publication . Santos, Ana João; Braz, Paula; Machado, Ausenda; Dias, Carlos Matias
BACKGROUND: The changes deriving from the birth of a child with a Congenital anomaly (CA) or Cerebral Palsy (CP) implies not only new emotional demands resulting from the child's condition, but in many cases an increase of interactions with health services and professionals. Health services and health policies must acknowledge and offer the opportunity for the family to define its own problems.
METHODS: The cross-sectional descriptive qualitative study was conducted with a convenience sample of parents of children diagnosed with four groups of CA (severe heart anomalies; spina bifida; orofacial clefts and Down syndrome) and/or CP. A semi-structured online questionnaire to be answered by parents was sent by web link to focal points of five parents associations and professional institutions in Portugal. Data was analysed through thematic content analysis (open-ended questions) and descriptive analysis (closed-ended questions).
RESULTS: Families feel professionals should recognise the importance of obtaining more complete and accurate information about the child's diagnosis. Health services were perceived as displaced and unordered, since parents indicate a lack of multidisciplinary interventions and lack of coordination or communication between professionals. Nonetheless, parents felt overall confident on the health care provided and on the “training” they get to care for their children deriving from the interactions with health professionals.
CONCLUSION: Health policies should ensure that families and children with CA and/or CP have access to reliable information. Health services and professionals should take into account the unique needs of each family and child and develop integrated interventions.
Information needs of parents of children with congenital anomalies across Europe: a EUROlinkCAT survey
Publication . Marcus, Elena; Latos-Bielenska, Anna; Jamry-Dziurla, Anna; Barišić, Ingeborg; Cavero-Carbonell, Clara; Den Hond, Elly; Garne, Ester; Genard, Lucas; Santos, Ana João; Lutke, LRenée; Matias Dias, Carlos; Neergaard Pedersen, Christina; Neville, Amanda J.; Niemann, Annika; Odak, Ljubica; Pierini, Anna; Rico, Juan; Rissmann, Anke; Rankin, Judith; Morris, Joan K.
Background: Parents of children who have a congenital anomaly can experience significant worry about their child's health. Access to clear, helpful, and trustworthy information can provide a valuable source of support. In this study the aim was to explore the information needs of parents/carers of children with congenital anomalies across Europe.
Method: A cross-sectional online survey was developed in nine languages to measure parents' information needs, including: (1) the 'helpfulness'/'trustworthiness' of information received from eight relevant sources, and (2) overall satisfaction with information received. Parents/carers of children (0-10 years) with cleft lip, spina bifida, congenital heart defect [CHD] requiring surgery, and/or Down syndrome were recruited online via relevant organisations in 10 European countries from March-July 2021. Quantitative analyses using multivariable logistic regressions were performed.
Results: One thousand seventy parents/carers of children with a cleft lip (n = 247), spina bifida (n = 118), CHD (n = 366), Down syndrome (n = 281), and Down syndrome with CHD (n = 58) were recruited in Poland (n = 476), the UK (n = 120), Germany (n = 97), the Netherlands/Belgium (n = 74), Croatia (n = 68), Italy (n = 59), other European countries (n = 92), and not specified/non-European countries (n = 84). Most participants were mothers (92%) and aged 31-40 years (71%). Participants were most likely to rate support groups (63%), patient organisations (60%), specialist doctors/nurses (58%), and social media (57%) as 'very helpful' information sources. 'Very trustworthy' ratings remained high for specialist doctors/nurses (61%), however, they declined for support groups (47%), patient organisations (48%), and social media (35%). Germany had the highest proportion of participants who were 'very satisfied' (44%, 95% CI = 34%-54%) with information, whereas this percentage was lowest in Croatia (11%, 95% CI = 3%-19%) and Poland (15%, 95% CI = 11%-18%). Parents of children with Down syndrome had significantly lower satisfaction ratings than parents of children with CHD; 13% (95% CI = 8%-18%) reported being 'very satisfied' compared to 28% (95% CI = 23%-33%) in the CHD group.
Conclusions: Findings suggest that informal sources of information (e.g. support groups) are of value to parents, however, they are not deemed as trustworthy as specialist medical sources. Satisfaction ratings differed across countries and by anomaly, and were particularly low in Croatia and Poland, as well as for parents of children with Down syndrome, which warrants further investigation.
EUROlinkCAT protocol for a European population-based data linkage study investigating the survival, morbidity and education of children with congenital anomalies
Publication . Morris, Joan K; Garne, Ester; Loane, Maria; Barisic, Ingeborg; Densem, James; Latos-Bieleńska, Anna; Neville, Amanda; Pierini, Anna; Rankin, Judith; Rissmann, Anke; de Walle, Hermien; Tan, Joachim; Given, Joanne Emma; Claridge, Hugh; EUROlinkCAT Consortium
Introduction: Congenital anomalies (CAs) are a major cause of infant mortality, childhood morbidity and long-term disability. Over 130 000 children born in Europe every year will have a CA. This paper describes the EUROlinkCAT study, which is investigating the health and educational outcomes of children with CAs for the first 10 years of their lives.
Methods and analysis: EUROCAT is a European network of population-based registries for the epidemiological surveillance of CAs. EUROlinkCAT is using the EUROCAT infrastructure to support 22 EUROCAT registries in 14 countries to link their data on births with CAs to mortality, hospital discharge, prescription and educational databases. Once linked, each registry transforms their case data into a common data model (CDM) format and they are then supplied with common STATA syntax scripts to analyse their data. The resulting aggregate tables and analysis results are submitted to a central results repository (CRR) and meta-analyses are performed to summarise the results across all registries. The CRR currently contains data on 155 594 children with a CA followed up to age 10 from a population of 6 million births from 1995 to 2014.
Ethics: The CA registries have the required ethics permissions for routine surveillance and transmission of anonymised data to the EUROCAT central database. Each registry is responsible for applying for and obtaining additional ethics and other permissions required for their participation in EUROlinkCAT.
Dissemination: The CDM and associated documentation, including linkage and standardisation procedures, will be available post-EUROlinkCAT thus facilitating future local, national and European-level analyses to improve healthcare. Recommendations to improve the accuracy of routinely collected data will be made.Findings will provide evidence to inform parents, health professionals, public health authorities and national treatment guidelines to optimise diagnosis, prevention and treatment for these children with a view to reducing health inequalities in Europe.
EUROlinkCAT - Establishing a Linked European Cohort of Children with Congenital Anomalies: relatório final de projeto
Publication . Santos, Ana João; Braz, Paula; Machado, Ausenda; Matias Dias, Carlos
Cerca de 2% de todos os nascidos vivos na Europa (cerca de 50.000 nascimentos por ano) apresentam uma anomalia congénita (AC) e 80% destas são doenças raras. As AC são uma das principais causas de mortalidade e morbidade infantil assim como de incapacidade de longa duração. Em Portugal o Registo Nacional de Anomalias Congénitas (RENAC) é um instrumento estratégico na vigilância epidemiológica das AC que ocorrem em Portugal. É um registo nosológico de base populacional que colabora com o European Surveillance of Congenital Anomalies (EUROCAT) na vigilancia das AC a nível europeu. 21 registos de 14 países, participaram no projeto EUROlinkCAT que procurou utilizar a experiência e a infraestrutura do EUROCAT para estabelecer um conjunto de bases de dados harmonizadas, com informação a nível europeu, sobre as necessidades de crianças nascidas com AC e, sempre que possível, de crianças nascidas sem AC (controlos) através da ligação da bases de dados das AC às bases de dados da mortalidade, morbilidade e educação. Este projeto teve duas grandes finalidades: a) investigar a evolução do estado de saúde de crianças nascidas com AC durante os primeiros 10 anos de vida, e avaliar os custos de hospitalizações durante os primeiros cinco anos de vida, assim como o acesso à educação; b) facilitar o desenvolvimento de uma relação mais recíproca entre famílias com crianças com AC, profissionais de saúde e investigadores, através da plataforma online ConnectEpeople.
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Funding agency
European Commission
Funding programme
H2020
Funding Award Number
733001