Linking a European cohort of children born with congenital anomalies to vital statistics and mortality records: A EUROlinkCAT study.
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SUBMITTER: Loane M
PROVIDER: S-EPMC8396745 | biostudies-literature |
REPOSITORIES: biostudies-literature
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Project description:IntroductionCongenital 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 analysisEUROCAT 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.EthicsThe 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.DisseminationThe 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.
| S-EPMC8240574 | biostudies-literature
Project description:BackgroundParents 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.MethodA 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.ResultsOne 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.ConclusionsFindings 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.
| S-EPMC9652126 | biostudies-literature
Project description:BackgroundCongenital anomalies are a major cause of co-morbidity in children. Diagnostic code lists are increasingly used to identify congenital anomalies in administrative health records. Evidence is lacking on comparability of these code lists.ObjectivesTo compare prevalence of congenital anomalies and prognostic outcomes for children with congenital anomalies identified in administrative health records using three different code lists.MethodsWe developed national cohorts of singleton livebirths in England (n = 7 354 363, 2003-2014) and Scotland (n = 493 556, 2003-2011). Children with congenital anomalies were identified if congenital anomaly diagnosis was recorded at birth, during subsequent hospital admission or as cause of death before 2 years old. We used three code lists: the EUROCAT list for congenital anomaly surveillance in Europe; the Hardelid list developed to identify children with chronic conditions (including congenital anomalies) admitted to hospital in England; and the Feudtner list developed to indicate children with complex chronic conditions (including congenital anomalies) admitted to hospitals in the United States. We compared prevalence, and risks of postnatal hospital readmission and death according to each code list in England and Scotland.ResultsPrevalence of congenital anomalies was highest using the EUROCAT list (4.1% of livebirths in England, 3.7% in Scotland), followed by Hardelid (3.1% and 3.0% of livebirths, respectively) and Feudtner (1.8% and 1.5% of livebirths, respectively). 67.2%-73.3% of children with congenital anomalies in England and 65.2%-77.0% in Scotland had at least one postnatal hospital admission across the three code lists; mortality ranged between 42.6-75.4 and 41.5-88.7 deaths per 1000 births in England Scotland, respectively. The risk of these adverse outcomes was highest using Feudtner and lowest using EUROCAT code lists.ConclusionsThe prevalence of congenital anomalies varied by congenital anomaly code list, over time and between countries, reflecting in part differences in hospital coding practices and admission thresholds. As a minimum, researchers using administrative health data to study congenital anomalies should report sensitivity analyses using different code lists.
| S-EPMC7003968 | biostudies-literature
Project description:BackgroundMortality data at the population level are often aggregated in age classes, for example 5-year age groups with an open-ended interval for the elderly aged 85+. Capturing detailed age-specific mortality patterns and mortality time trends from such coarsely grouped data can be problematic at older ages, especially where open-ended intervals are used.MethodsWe illustrate the penalized composite link model (PCLM) for ungrouping to model cancer mortality surfaces. Smooth age-specific distributions from data grouped in age classes of adjacent calendar years were estimated by constructing a two-dimensional regression, based on B-splines, and maximizing a penalized likelihood. We show the applicability of the proposed model, analysing age-at-death distributions from cancers of all sites in Denmark from 1980 to 2014. Data were retrieved from the Danish Cancer Society and the Human Mortality Database.ResultsThe main trends captured by PCLM are: (i) a decrease in cancer mortality rates after the 1990s for ages 50-75; (ii) a decrease in cancer mortality in later cohorts for young ages, especially, and very advanced ages. Comparing the raw data by single year of age, with the PCLM-ungrouped distributions, we clearly illustrate that the model fits the data with a high level of accuracy.ConclusionsThe PCLM produces detailed smooth mortality surfaces from death counts observed in coarse age groups with modest assumptions, that is Poisson distributed counts and smoothness of the estimated distribution. Hence, the method has great potential for use within epidemiological research when information is to be gained from aggregated data, because it avoids strict assumptions about the actual distributional shape.
| S-EPMC6469310 | biostudies-literature
Project description:ObjectiveTo report and compare the proportion of children with and without congenital anomalies undergoing gastrostomy for tube feeding in their first 5 years.MethodsA European, population-based data-linkage cohort study (EUROlinkCAT). Children up to 5 years of age registered in nine EUROCAT registries (national and regional) in six countries and children without congenital anomalies (reference children) living in the same geographical areas were included. Data on hospitalisation and surgical procedures for all children were obtained by electronic linkage to hospital databases.ResultsThe study included 91 504 EUROCAT children and 1 960 272 reference children. Overall, 1200 (1.3%, 95% CI 1.2% to 1.6%) EUROCAT children and 374 (0.016%, 95% CI 0.009% to 0.026%) reference children had a surgical code for gastrostomy within the first 5 years of life. There were geographical variations across Europe with higher rates in Northern Europe compared with Southern Europe. Around one in four children with Cornelia de Lange syndrome and Wolf-Hirschhorn syndrome had a gastrostomy. Among children with structural anomalies, those with oesophageal atresia had the highest proportion of gastrostomy (15.9%).ConclusionsThis study including almost 2 million reference children in Europe found that only 0.016% of these children had a surgery code for gastrostomy before age 5 years. The children with congenital anomalies were on average 80 times more likely to need a gastrostomy before age 5 years than children without congenital anomalies. More than two-thirds of gastrostomy procedures performed within the first 5 years of life were in children with congenital anomalies.
| S-EPMC9234789 | biostudies-literature
Project description:Electronic health care databases are increasingly being used to investigate the epidemiology of congenital anomalies (CAs) although there are concerns about their accuracy. The EUROlinkCAT project linked data from eleven EUROCAT registries to electronic hospital databases. The coding of CAs in electronic hospital databases was compared to the (gold standard) codes in the EUROCAT registries. For birth years 2010-2014 all linked live birth CA cases and all children identified in the hospital databases with a CA code were analysed. Registries calculated sensitivity and Positive Predictive Value (PPV) for 17 selected CAs. Pooled estimates for sensitivity and PPV were then calculated for each anomaly using random effects meta-analyses. Most registries linked more than 85% of their cases to hospital data. Gastroschisis, cleft lip with or without cleft palate and Down syndrome were recorded in hospital databases with high accuracy (sensitivity and PPV ≥ 85%). Hypoplastic left heart syndrome, spina bifida, Hirschsprung's disease, omphalocele and cleft palate showed high sensitivity (≥ 85%), but low or heterogeneous PPV, indicating that hospital data was complete but may contain false positives. The remaining anomaly subgroups in our study, showed low or heterogeneous sensitivity and PPV, indicating that the information in the hospital database was incomplete and of variable validity. Electronic health care databases cannot replace CA registries, although they can be used as an additional ascertainment source for CA registries. CA registries are still the most appropriate data source to study the epidemiology of CAs.
| S-EPMC10033551 | biostudies-literature
Project description:Social surveys prospectively linked with death records provide invaluable opportunities for the study of the relationship between social and economic circumstances and mortality. Although survey-linked mortality files play a prominent role in U.S. health disparities research, it is unclear how well mortality estimates from these datasets align with one another and whether they are comparable with U.S. vital statistics data. We conduct the first study that systematically compares mortality estimates from several widely-used survey-linked mortality files and U.S. vital statistics data. Our results show that mortality rates and life expectancies from the National Health Interview Survey Linked Mortality Files, Health and Retirement Study, Americans' Changing Lives study, and U.S. vital statistics data are similar. Mortality rates are slightly lower and life expectancies are slightly higher in these linked datasets relative to vital statistics data. Compared with vital statistics and other survey-linked datasets, General Social Survey-National Death Index life expectancy estimates are much lower at younger adult ages and much higher at older adult ages. Cox proportional hazard models regressing all-cause mortality risk on age, gender, race, educational attainment, and marital status conceal the issues with the General Social Survey-National Death Index that are observed in our comparison of absolute measures of mortality risk. We provide recommendations for researchers who use survey-linked mortality files.
| S-EPMC6541424 | biostudies-literature
Project description:This study aimed to analyse population-based mortality attributed to rare congenital anomalies (CAs) and assess the associated time trends and geographical differences in Spain. Data on CA-related deaths were sourced from annual mortality databases kept by the National Statistics Institute of Spain (1999?2013). Based on the ICD-10, only CAs corresponding to rare diseases definition were included in this study. Annual age-adjusted mortality rates were calculated and time trends were evaluated by joinpoint regression analysis. Geographical differences were assessed using standardised mortality ratios and cluster detection. A total of 13,660 rare-CA-related deaths (53.4% males) were identified in the study period. Annual age-adjusted mortality rates decreased by an average of -5.2% (-5.5% males, -4.8% females, p < 0.001). Geographical analysis showed a higher risk of rare-CA-related mortality in regions largely located in the south of the country. Despite their limitations, mortality statistics are essential and useful tools for enhancing knowledge of rare disease epidemiology and, by extension, for designing and targeting public health actions. Monitoring rare-CA-related mortality in Spain has shown a 15-year decline and geographical differences in the risk of death, all of which might well be taken into account by the health authorities in order to ensure equality and equity, and to adopt appropriate preventive measures.
| S-EPMC6121521 | biostudies-literature
Project description:Background: In humans, the mortality rate dramatically decreases with age after birth, and the causes of death change significantly during childhood. In the present study, we attempted to explain age-associated decreases in mortality for congenital anomalies of the central nervous system (CACNS), as well as decreases in total mortality with age. We further investigated the age trajectory of mortality in the biologically related category "diseases of the nervous system" (DNS). Methods: The numbers of deaths were extracted from the mortality database of the World Health Organization (WHO) for the following nine countries: Denmark, Finland, Norway, Sweden, Austria, the Czech Republic, Hungary, Poland, and Slovakia. Because zero cases could be ascertained over the age of 30 years in a specific age category, the Halley method was used to calculate the mortality rates in all possible calendar years and in all countries combined. Results: Total mortality from the first day of life up to the age of 10 years and mortality due to CACNS within the age interval of [0, 90) years can be represented by an inverse proportion with a single parameter. High coefficients of determination were observed for both total mortality (R2 = 0.996) and CACNS mortality (R2 = 0.990). Our findings indicated that mortality rates for DNS slowly decrease with age during the first 2 years of life, following which they decrease in accordance with an inverse proportion up to the age of 10 years. The theory of congenital individual risk (TCIR) may explain these observations based on the extinction of individuals with more severe impairments, as well as the bent curve of DNS, which exhibited an adjusted coefficient of determination of R¯2 = 0.966. Conclusion: The coincidence between the age trajectories of all-cause and CACNS-related mortality may indicate that the overall decrease in mortality after birth is due to the extinction of individuals with more severe impairments. More deaths unrelated to congenital anomalies may be caused by the manifestation of latent congenital impairments during childhood.
| S-EPMC6067090 | biostudies-literature
Project description:Objectives: Both the National Vital Statistics System (NVSS) and the Fatality Analysis Reporting System (FARS) can be used to examine motor vehicle crash (MVC) deaths. These 2 data systems operate independently, using different methods to collect and code information about the type of vehicle (e.g., car, truck, bus) and road user (e.g., occupant, motorcyclist, pedestrian) involved in an MVC. A substantial proportion of MVC deaths in NVSS are coded as "unspecified" road user, which reduces the utility of the NVSS data for describing burden and identifying prevention measures. This study aimed to describe characteristics of unspecified road user deaths in NVSS to further our understanding of how these groups may be similar to occupant road user deaths. Methods: Using data from 1999 to 2015, we compared NVSS and FARS MVC death counts by road user type, overall and by age group, gender, and year. In addition, we examined factors associated with the categorization of an MVC death as unspecified road user such as state of residence of decedent, type of medical death investigation system, and place of death. Results: The number of MVC occupant deaths in NVSS was smaller than that in FARS in each year and the number of unspecified road user deaths in NVSS was greater than that in FARS. The sum of the number of occupant and unspecified road user deaths in NVSS, however, was approximately equal to the number of FARS occupant deaths. Age group and gender distributions were roughly equivalent for NVSS and FARS occupants and NVSS unspecified road users. Within NVSS, the number of MVC deaths listed as unspecified road user varied across states and over time. Other categories of road users (motorcyclists, pedal cyclists, and pedestrians) were consistent when comparing NVSS and FARS. Conclusions: Our findings suggest that the unspecified road user MVC deaths in NVSS look similar to those of MVC occupants according to selected characteristics. Additional study is needed to identify documentation and reporting challenges in individual states and over time and to identify opportunities for improvement in the coding of road user type in NVSS.
| S-EPMC6533142 | biostudies-literature