| 1. |
- Rosén, Anna, 1975-
(författare)
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Mass screening for celiac disease in 12-year-olds : Finding them and then what?
- 2012
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Doktorsavhandling (övrigt vetenskapligt/konstnärligt)abstract
- Background Mass screening for celiac disease (CD) as a public health intervention is controversial. Before implementation, a suitable screening strategy should be outlined, and the acceptability of the screening scrutinized. Also, the benefits of early detection and possible negative consequences should be explored and compared. The overall aim of this thesis was to evaluate different strategies for finding 12-year-olds with undiagnosed CD in the general population, and to explore the experiences of those receiving the diagnosis in a mass screening.Methods A school-based CD screening of 12-year-olds was conducted in five study sites across Sweden. Out of 10041 children who were invited, 7208 had a blood sample analyzed for CD-marker tissue transglutaminase of isotype IgA (tTG-IgA) and 7161 for total serum IgA (s-IgA). If the s-IgA value was low, tTG-IgG was also measured. Additional analysis of endomysial antibodies (EMA) was performed if borderline values of tTG were found. In total, 192 had elevated CD-markers, 184 underwent a small intestinal biopsy and 153 eventually had CD diagnosed. Before receiving knowledge about their CD status, children and their parents filled in questionnaires regarding symptoms and CD-associated conditions. Questionnaires were returned by 7054 children (98%) and 6294 parents (88%). Later, all adolescents who had been diagnosed with CD more than one year ago (n=145), and their parents, were invited to a mixed-method follow-up study in which they shared their experiences in questionnaires, written narratives and focus group discussions. In total, we have information on 117 (81%) of these adolescents, either from the adolescents themselves (n=101) and/or from their parent/s (n=125). Data were analyzed using a combination of descriptive and analytical quantitative and qualitative methodologies.Results We found that information on symptoms and CD-associated conditions were poor predictors for finding undiagnosed CD in the study population. Questionnaire-based case-finding by asking for CD-associated symptoms and conditions would have identified 52 cases (38% of all cases) at a cost of blood-sampling 2282 children (37% of the study population). The tTG-IgA test had an excellent diagnostic accuracy with the area under the receiver operating characteristic curve of 0.988. If using the recommended cut-off for tTG-IgA (>5 U/mL) 151 had fulfilled biopsy criteria and 134 CD cases had been identified. The strategy of lowering the cut-off to tTG-IgA>4 U/mL, and adding the EMA analysis in those with tTG-IgA between 2-4 U/mL, identified another 17 cases (a 12% increase) at the cost of performing 32 additional biopsies. Measuring total s-IgA in 7161 children discovered only two additional cases at the cost of performing 5 additional biopsies. The positive predictive value of our screening strategy was 80%. Results from the follow-up study of the screening-detected CD cases illustrated that 54% reported health improvement after initiated treatment, but also that these health benefits had to be balanced against social sacrifices. We also found that although the screening-detected diagnosis was met with surprise and anxiety, the adolescents and their parents were grateful for being made aware of the diagnosis. A majority of parents (92%) welcomed a future screening, but both adolescents and parents suggested that it should be conducted earlier in life.Conclusion Obtaining information on symptoms and CD-associated conditions was not a useful step in finding undiagnosed CD cases in a general population. The serological marker tTG-IgA, however, had excellent diagnostic accuracy also when lowering the cut-off. The diagnosis had varying impact on adolescents’ quality of life, and their perceived change in health had to be balanced against the social sacrifices resulting from the diagnosis. Overall, CD mass screening seemed acceptable to most of those who were diagnosed and their parents.
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| 2. |
- Norström, Fredrik, 1974-
(författare)
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The burden of celiac disease and the value of having it diagnosed
- 2012
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Doktorsavhandling (övrigt vetenskapligt/konstnärligt)abstract
- Background: Celiac disease is a chronic disease characterized by intolerance to gluten. It is considered a public health problem affecting about 1% of Western populations, but, with most cases still undiagnosed. A glutenfree diet is the only effective treatment for the disease. Objectives: To investigate the burden of celiac disease and the value of having it diagnosed. Additionally, the implications for a potential future celiac disease mass screening are discussed. Methods: A questionnaire was sent during 2009 to 1,560 randomly selected adult members of the Swedish Society for Coeliacs, in equal-sized age- and sex strata, and 1,031 (66%) responded. Members were asked about symptoms, health-related quality of life as measured by EQ-5D, and health care consumption during the year prior to diagnosis and during the past year. They were also asked about the delay in having their celiac disease diagnosed and the appearance of other immune-mediated diseases. A school-based celiac disease screening of 12-year-olds was performed during 2005-2006. After blood sampling the 7,567 participating children and their parents received a questionnaire including the EQ-5D instrument in order to measure the child’s health-related quality of life. Comparisons were made between children with screening-detected celiac disease, those with previously diagnosed disease and those without the disease. Parents were asked about their willingness to pay for a celiac disease screening of their child, which was compared with the actual cost of a screening. Results: Adult celiac disease patients had a poorer health-related quality of life than the general population, and a high prevalence of symptoms before celiac disease diagnosis. The mean delay from symptoms to diagnosis was 9.7 years. After initiated treatment with a gluten-free diet, health-related quality of life was improved to the level of the general population, and symptom relief and reduction in health care consumption were also reported. For children, health-related quality of life was similar across groups. The average cost per child for a screening was 47 EUR. Parents’ mean willingness to pay for a screening was 79 EUR, median 10 EUR. Conclusion: The delay in celiac disease diagnosis is long, and treatment with a gluten-free diet substantially improved health for clinically detected cases. For screening-detected celiac disease the health benefits are still uncertain. A mass screening might nevertheless be justified to avoid the burden of living with undiagnosed disease, and parents’ willingness to pay indicates that performing it in childhood might be economically motivated. However, as both the cost-effectiveness of a screening and the short- and long term health benefits for screening-detected cases have not yet been sufficiently investigated, it is too early to make a recommendation for a celiac disease mass screening.
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| 3. |
- Edvardsson, Kristina, 1973-
(författare)
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Health promotion in pregnancy and early parenthood : the challenge of innovation, implementation and change within the Salut Programme
- 2013
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Doktorsavhandling (övrigt vetenskapligt/konstnärligt)abstract
- Background: In 2005, the Västerbotten County Council launched a child health promotion programme, “the Salut Programme”, in response to an alarming prevalence of overweight and obesity, and trends of increased dental caries, among young county citizens. The programme, initially developed in four pilot areas, is built on multidisciplinary and cross-sectoral collaboration and aims to support and strengthen health promotion activities in health care, social services and school settings. It targets children and adolescents (0-18 years of age) and their parents, and starts during pregnancy. This thesis focuses on interventions provided by antenatal care, child health care, dental services, and open pre-schools, directed to expectant parents and families with children aged 0-1 ½ years. Within the programme context, the aim was to explore socio-demographic patterns of overweight and obesity in expectant parents (Paper I), firsttime parents’ experiences of health promotion and lifestyle change during pregnancy and early parenthood (Paper II), professionals’ experiences of factors influencing programme implementation and sustainability (Paper III and IV), and early programme outcomes on professionals’ health promotion practices and collaboration following countywide dissemination and implementation (Paper IV). Methods and results: A population based cross-sectional study among expectant parents showed overweight and obesity in 29% of women (pre-pregnancy) and in 53% of men (n=4,352♀, 3,949♂). The likelihood for obesity was higher in expectant parents with lower levels of education, among those unemployed or on sick leave, and those living in rural areas. In 62% of couples, at least one of the partners was overweight or obese; a positive partner correlation was also found for BMI (I). An interview study with 24 first-time parents (n=12♀, 12♂) revealed that they primarily undertook lifestyle changes to secure the health of the fetus in pregnancy, and to provide a healthy environment in childhood. Parents described themselves as highly receptive to information about how their lifestyle could influence fetal health, and they frequently discussed pregnancy risks related to tobacco and alcohol, as well as toxins and infectious agents in foods. However, parents did not seem inclined to make lifestyle changes primarily to promote their own health. The antenatal and child health care services were perceived as being mainly directed towards women, and parents described a lack of a holistic view of the family which included experiences of fathers being treated as less important (II). An interview study undertaken with professionals (n=23) in the Salut Programme pilot areas indicated programme sustainability at most sites, two years after implementation, although less adherence was described within child health care. Factors influencing programme sustainability, as described by professionals, were identified at multiple organisational levels (III). A before-and-after survey among professionals (n=144) measured outcomes of the county-wide implementation of the Salut Programme in 13 out of 15 county municipalities. Results showed significant improvements in professionals’ health promotion practices and collaboration across sectors. A number of important implementation facilitators and barriers, acting at different organizational levels, were also identified via a survey comprised of open-ended questions (IV). Conclusion: The Salut Programme, developed with high involvement of professionals, and strongly integrated in existing organisational structures and practices, shows potential for improving health promotion practices and cross-sectoral collaboration. The findings can inform further development of the Salut Programme.as well as new health promotion initiatives, and inform policy practice and future research. These aspects include approaches in health promotion and prevention, father involvement during pregnancy and early parenthood, and factors influencing implementation and sustainability of cross-sectoral health promotion programmes.
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| 4. |
- Häggström, Jenny, et al.
(författare)
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Is the Salut Programme an effective and cost-effective universal health promotion intervention for parents and their children? : a register-based retrospective observational study
- 2017
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Ingår i: BMJ Open. - : BMJ Publishing Group Ltd. - 2044-6055. ; 7:9
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Tidskriftsartikel (refereegranskat)abstract
- OBJECTIVES: This study investigates the effectiveness and cost-effectiveness of the Salut Programme, a universal health promotion intervention, compared with care-as-usual, over the periods of pregnancy, delivery and the child's first 2 years of life.METHOD: We adopted a register-based retrospective observational design using existing data sources with respect to both exposures and outcomes. Health outcomes and costs were compared between geographical areas that received care-as-usual (non-Salut area) and areas where the programme was implemented (Salut area). We included mothers and their children from both the Salut and non-Salut areas if: (1) the child was born 2002-2004 (premeasure period) or (2) the child was born 2006-2008 (postmeasure period). The effectiveness study adopted two strategies: (1) a matched difference-in-difference analysis using data from all participants and (2) a longitudinal analysis restricted to mothers who had given birth twice, that is, both in the premeasure and postmeasure periods. The economic evaluation was performed from a healthcare and a limited societal perspective. Outcomes were clustered during pregnancy, delivery and birth and the child's first 2 years.RESULTS: Difference-in-difference analyses did not yield any significant effect on the outcomes. Longitudinal analyses resulted in significant positive improvement in Apgar scores, reflecting the newborn's physical condition, with more children having a normal Apgar score (1 min +3%, 5 min +1%). The cost of the programme was international dollar (INT$)308/child. From both costing perspectives, the programme yielded higher effects and lower costs than care-as-usual, being thus cost-saving (probability of around 50%).CONCLUSIONS: Our findings suggest that the Salut Programme is an effective universal intervention to improve maternal and child health, and it may be good value for money; however, there is large uncertainty around the cost estimates.
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| 5. |
- Myléus, Anna, 1978-
(författare)
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Towards explaining the Swedish epidemic of celiac disease : an epidemiological approach
- 2012
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Doktorsavhandling (övrigt vetenskapligt/konstnärligt)abstract
- Background: Celiac disease occurs worldwide in approximately 1% of the population, whereof the majority of cases are undiagnosed. Sweden experienced an epidemic (1984-1996) of clinically detected celiac disease in children below 2 years of age, partly attributed to changes in infant feeding. Whether the epidemic constituted a change in disease occurrence and/or a shift in the proportion of diagnosed cases remains unknown. Moreover, the cause of the epidemic is not fully understood.Objective: To increase the knowledge regarding the occurrence of celiac disease in Sweden, with focus on the epidemic period and thereafter, as well as the etiology of celiac disease in general, by investigating the Swedish epidemic and its potential causes.Methods: We performed a two-phased cross-sectional multicenter screening study investigating the total prevalence, including both clinically- and screening-detected cases, of celiac disease in 2 birth cohorts of 12-year-olds (n=13 279): 1 of the epidemic period (1993) and 1 of the post-epidemic period (1997). The screening strategy entailed serological markers analyses, with subsequent small intestinal biopsy when values were positive. Diagnosis was ascertained in clinical cases detected prior to screening. Infant feeding practices in the cohorts were ascertained via questionnaires. An ecological approach combined with an incident case-referent study (475 cases, 950 referents) performed during the epidemic were used for investigating environmental- and lifestyle factors other than infant feeding. Exposure information was obtained via register data, a questionnaire, and child health clinic records. All studies utilized the National Swedish Childhood Celiac Disease Register.Results: The total prevalences of celiac disease were 2.9% and 2.2% for the 1993 and 1997 cohorts, respectively, with 2/3 cases unrecognized prior to screening. Children born in 1997 had a significantly lower celiac disease prevalence compared to those born in 1993 (prevalence ratio, 0.75; 95% confidence interval [CI], 0.60-0.93). The cohorts differed in infant feeding; more specifically in the proportion of infants introduced to dietary gluten in small amounts during ongoing breastfeeding. Of the environmental and lifestyle factors investigated, no additional changes over time coincided with the epidemic. Early vaccinations within the Swedish program were not risk factors for celiac disease. Early infections (≥3 parental-reported episodes) were associated with increased risk for celiac disease (adjusted odds ratio [OR] 1.5; 95% CI, 1.1-2.0), a risk that increased synergistically if, in addition to having ≥3 infectious episodes, the child was introduced to gluten in large amounts, compared to small or medium amounts, after breastfeeding was discontinued (OR 5.6; 95% CI, 3.1-10). Early infections probably made a minor contribution to the Swedish epidemic through the synergistic effect with gluten, which changed concurrently. In total, approximately 48% of the epidemic could be explained by infant feeding and early infections.Conclusion: Celiac disease is both unexpectedly prevalent and mainly undiagnosed in Swedish children. Although the cause of the epidemic is still not fully understood, the significant difference in prevalence between the 2 cohorts indicates that the epidemic constituted a change in disease occurrence, and importantly, corroborates that celiac disease can be avoided in some children, at least up to 12 years of age. Our findings suggest that infant feeding and early infections, but not early vaccinations, have a causal role in the celiac disease etiology and that the infant feeding practice – gradually introducing gluten-containing foods from 4 months of age, preferably during ongoing breastfeeding – is favorable.
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| 6. |
- Pulkki-Brännström, Anni-Maria, et al.
(författare)
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The equity impact of a universal child health promotion programme
- 2020
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Ingår i: Journal of Epidemiology and Community Health. - : BMJ Publishing Group Ltd. - 0143-005X .- 1470-2738. ; 74, s. 605-611
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Tidskriftsartikel (refereegranskat)abstract
- BACKGROUND: Real-world evaluations of complex interventions are scarce. We evaluated the effect of the Salut Programme, a universal child health promotion intervention in northern Sweden, on income-related inequalities in positive birth outcomes and healthcare utilisation up to 2 years after delivery.METHODS: Using the mother's place of residence at delivery, the child and the mother were classified as belonging to either the control area (received care-as-usual) or the intervention area (where the intervention was implemented from 2006) and either the premeasure (children born between 2002 and 2004) or the postmeasure (children born between 2006 and 2008) period. Parents' earned income was used as the socioeconomic ranking variable. The Relative Concentration Index was computed for six binary birth outcome indicators and for inpatient and day patient care for children and their mothers. Changes in inequality over time were compared using a difference-in-difference approach.RESULTS: Income-related inequalities in birth outcomes and child healthcare utilisation were absent, except that full-term pregnancies were concentrated among the poor at premeasure in the intervention area. In contrast, mothers' healthcare utilisation was significantly pro-poor in the control area. The extent of inequality changed differentially between premeasure and postmeasure for two birth outcomes: full-term pregnancies and infants with normal birth weight. Inequalities in healthcare utilisation did not change significantly in either area over time.CONCLUSION: In northern Sweden, income-related inequalities in birth outcomes and child healthcare utilisation are largely absent. However, relative inequalities in mothers' healthcare utilisation are large. We found no evidence that the Salut Programme affected changes in inequality over time.
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| 7. |
- Sampaio, Filipa, PhD, 1985-, et al.
(författare)
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Health and economic outcomes of a universal early intervention for parents and children from birth to age five : evaluation of the Salut Programme using a natural experiment
- 2023
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Ingår i: Cost Effectiveness and Resource Allocation. - : BioMed Central (BMC). - 1478-7547. ; 21:1
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Tidskriftsartikel (refereegranskat)abstract
- Background: The aim of this study was to investigate the health and economic outcomes of a universal early intervention for parents and children, the Salut Programme, from birth to when the child completed five years of age.Methods: This study adopted a retrospective observational design using routinely collected linked register data with respect to both exposures and outcomes from Västerbotten county, in northern Sweden. Making use of a natural experiment, areas that received care-as-usual (non-Salut area) were compared to areas where the Programme was implemented after 2006 (Salut area) in terms of: (i) health outcomes, healthcare resource use and costs around pregnancy, delivery and birth, and (ii) healthcare resource use and related costs, as well as costs of care of sick child. We estimated total cumulative costs related to inpatient and specialised outpatient care for mothers and children, and financial benefits paid to mothers to stay home from work to care for a sick child. Two analyses were conducted: a matched difference-in difference analysis using the total sample and an analysis including a longitudinal subsample.Results: The longitudinal analysis on mothers who gave birth in both pre- and post-measure periods showed that mothers exposed to the Programme had on average 6% (95% CI 3–9%) more full-term pregnancies and 2% (95% CI 0.03-3%) more babies with a birth weight ≥ 2500 g, compared to mothers who had care-as-usual. Savings were incurred in terms of outpatient care costs for children of mothers in the Salut area ($826). The difference-in-difference analysis using the total sample did not result in any significant differences in health outcomes or cumulative resource use over time.Conclusions: The Salut Programme achieved health gains, as a health promotion early intervention for children and parents, in terms of more full-term pregnancies and more babies with a birth weight ≥ 2500 g, at reasonable cost, and may lead to lower usage of outpatient care. Other indicators point towards positive effects, but the small sample size may have led to underestimation of true differences.
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| 8. |
- Vaezghasemi, Masoud, et al.
(författare)
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Multifaceted determinants of social-emotional problems in preschool children in Sweden : An ecological systems theory approach
- 2023
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Ingår i: SSM - Population Health. - : Elsevier. - 2352-8273. ; 21
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Tidskriftsartikel (refereegranskat)abstract
- Social-emotional problems occurring early in life can place children at future risk of adverse health, social and economic outcomes. Determinants of social-emotional problems are multi-layered and originate from different contexts surrounding children, though few studies consider them simultaneously. We adopted a holistic approach by using Bronfenbrenner's process-person-context-time model as a structuring device. We aimed to assess what characteristics of families and children from pregnancy, over birth, and up to 3 years of age are associated with social-emotional problems in boys and girls. This study used regional data from the Salut Programme, a universal health promotion programme implemented in Antenatal and Child Health Care, and data from national Swedish registers. The study population included 6033 3-year-olds and their parents during the period 2010-2018. Distinct logistic regression models for boys and girls were used to assess associations between the family social context, parents' lifestyle, parent's mental health, children's birth characteristics, and indicators of proximal processes (the independent variables); and children's social-emotional problems as measured by the parentcompleted Ages and Stages Questionnaire: Social-Emotional between 33 and 41 months of age (the outcome). Overall, a less favourable family social context, detrimental lifestyle of the parents during pregnancy, and parents' mental illness from pregnancy onwards were associated with higher odds of social-emotional problems in 3 -year-olds. Higher screentime and infrequent shared book-reading were associated with higher odds of socialemotional problems. The multifaceted determinants of children's social-emotional problems imply that many diverse targets for intervention exist. Additionally, this study suggests that Bronfenbrenner's process-person-context-time theoretical framework could be relevant for public health research and policy.
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| 9. |
- Westerlund, Anna, 1978-
(författare)
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The role of implementation science in healthcare improvement efforts : investigating three complex interventions
- 2018
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Doktorsavhandling (övrigt vetenskapligt/konstnärligt)abstract
- For decades, scholars have found significant gaps between the knowledge available and the knowledge applied in healthcare. Many potential benefits of adequate knowledge based interventions are therefore never achieved. A considerable body of knowledge has evolved on how to promote a better uptake of evidence-based knowledge into routine use. Even so, the actual impact and usefulness of implementation research findings among healthcare practitioners have not been extensively studied.Accordingly, the overall aim of this thesis is to contribute to the understanding of how the implementation of complex interventions into healthcare can be improved. This is done by investigating whether some of these efforts do correspond with available scientific knowledge on implementation.The thesis is based on three cases contributing to four studies. The cases studied are: the National Perinatal Patient Safety program (NPPS), the Dynamic and Viable Organisation initiative (DVO), and the International Child Development Program (ICDP). All studies focuses on the early stages of implementation.A mixed methods approach was adopted, involving both qualitative and quantitative methods. Data collection consisted of interviews, questionnaires, observations, and process diaries. Qualitative content analysis (conventional and directed), descriptive and non-parametric statistics were used. The focus was on implementation strategies used by healthcare actors in relation to factors influencing implementation processes and outcomes. More specifically, healthcare actors perspectives on such factors and whether they were addressed by the strategies used, was investigated. A process evaluation of implementation outcomes was also part of the thesis.The healthcare actors in focus were the adopters, i.e. practitioners expected to change their work practices, and implementation facilitators. The latter refer to actors with a more or less explicit responsibility to implement new practices or interventions aimed at improving the quality and effectiveness of the provided health services.Variation was found regarding how the implementation strategies used in the three cases corresponded with available scientific knowledge on implementation. In Case NPPS, the implementation facilitators planned, designed, and ensured that the core interventions of the implementation strategy were executed in a rational manner. Several important implementation factors were addressed by the strategy. The process evaluation of effects on readiness for change by the development of a team mental model among adopters showed positive results.In Case DVO a strategy was used that evolved over time, partly based on raised questions and feedback from staff and managers involved. The strategy can be described as an intuitive ‘socially accomplished activity’. This strategy involved addressing ‘Implementation Process-related factors’ in order to affect motivation and increase the tension for change among adopters.In Case ICDP, the results reflected a shortage of strategies during the early stage of implementation. The main intervention was the stepwise ICDP-education. A more comprehensive implementation strategy covering implementation factors highlighted as important among adopters was not developed. The process evaluation revealed vague directives on what was expected regarding the use and adaptation of ICDP to current practice versus preservation of fidelity to the original ICDP. This situation resulted in a rather large variation in how the changes in work practices were perceived among the health centres involved. No health centre practiced ICDP in its original form.A new knowledge-practice gap is discussed based on the findings in this thesis: a gap between the scientific knowledge on implementation and the actual implementation strategies used in practice during improvement efforts initiated by healthcare actors. The findings show that correspondence between scientific knowledge on implementation and what is actually done in order to accomplish change in practice might be more random (or implicit) than systematic. The question of how to transfer scientific knowledge on implementation into user-friendly resources for practitioners is discussed. A tentative model is suggested, which contributes to existing determinant frameworks by focusing on relations among factors. The model may be used in healthcare practice, to guide the design of an implementation strategy (or as a pathway for tailored implementation interventions) and aid the assignment of responsibilities in relation to factors that are known to affect implementation processes and outcomes.The question of how to transfer models and frameworks into user-friendly resources needs further attention. It is suggested that action oriented research aiming at further developing and establishing the concept of ‘practical implementation science’ should be conducted. This could be a way of bridging the knowledge-practice gap in healthcare.
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