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The Survivorship Passport (SurPass) for childhood cancer survivors provides a personalized treatment summary together with a care plan for long-term screening of possible late effects. HL7 FHIR connectivity of Electronic Health Record (EHR) systems with the SurPass has been proposed to reduce the burden of collecting and organizing the relevant information. We present the results of testing and validation efforts conducted across six clinics in Austria, Belgium, Germany, Italy, Lithuania, and Spain. We also discuss ways in which this experience can be used to reduce efforts for the SurPass integration in other clinics across Europe.
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Supervivientes de Cáncer , Registros Electrónicos de Salud , Humanos , Niño , Europa (Continente) , Estándar HL7 , Neoplasias/terapia , Interoperabilidad de la Información en SaludRESUMEN
BACKGROUND: To overcome knowledge gaps and optimize long-term follow-up (LTFU) care for childhood cancer survivors, the concept of the Survivorship Passport (SurPass) has been invented. Within the European PanCareSurPass project, the semiautomated and interoperable SurPass (version 2.0) will be optimized, implemented, and evaluated at 6 LTFU care centers representing 6 European countries and 3 distinct health system scenarios: (1) national electronic health information systems (EHISs) in Austria and Lithuania, (2) regional or local EHISs in Italy and Spain, and (3) cancer registries or hospital-based EHISs in Belgium and Germany. OBJECTIVE: We aimed to identify and describe barriers and facilitators for SurPass (version 2.0) implementation concerning semiautomation of data input, interoperability, data protection, privacy, and cybersecurity. METHODS: IT specialists from the 6 LTFU care centers participated in a semistructured digital survey focusing on IT-related barriers and facilitators to SurPass (version 2.0) implementation. We used the fit-viability model to assess the compatibility and feasibility of integrating SurPass into existing EHISs. RESULTS: In total, 13/20 (65%) invited IT specialists participated. The main barriers and facilitators in all 3 health system scenarios related to semiautomated data input and interoperability included unaligned EHIS infrastructure and the use of interoperability frameworks and international coding systems. The main barriers and facilitators related to data protection or privacy and cybersecurity included pseudonymization of personal health data and data retention. According to the fit-viability model, the first health system scenario provides the best fit for SurPass implementation, followed by the second and third scenarios. CONCLUSIONS: This study provides essential insights into the information and IT-related influencing factors that need to be considered when implementing the SurPass (version 2.0) in clinical practice. We recommend the adoption of Health Level Seven Fast Healthcare Interoperability Resources and data security measures such as encryption, pseudonymization, and multifactor authentication to protect personal health data where applicable. In sum, this study offers practical insights into integrating digital health solutions into existing EHISs.
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Telemedicina , Humanos , Telemedicina/métodos , Europa (Continente) , Encuestas y Cuestionarios , Registros Electrónicos de Salud , Supervivientes de Cáncer , Seguridad Computacional , SupervivenciaRESUMEN
PURPOSE: Long-term follow-up (LTFU) care for childhood cancer survivors (CCSs) is essential to improve and maintain their quality of life. The Survivorship Passport (SurPass) is a digital tool which can aid in the delivery of adequate LTFU care. During the European PanCareSurPass (PCSP) project, the SurPass v2.0 will be implemented and evaluated at six LTFU care clinics in Austria, Belgium, Germany, Italy, Lithuania and Spain. We aimed to identify barriers and facilitators to the implementation of the SurPass v2.0 with regard to the care process as well as ethical, legal, social and economical aspects. METHODS: An online, semi-structured survey was distributed to 75 stakeholders (LTFU care providers, LTFU care program managers and CCSs) affiliated with one of the six centres. Barriers and facilitators identified in four centres or more were defined as main contextual factors influencing implementation of SurPass v2.0. RESULTS: Fifty-four barriers and 50 facilitators were identified. Among the main barriers were a lack of time and (financial) resources, gaps in knowledge concerning ethical and legal issues and a potential increase in health-related anxiety in CCSs upon receiving a SurPass. Main facilitators included institutions' access to electronic medical records, as well as previous experience with SurPass or similar tools. CONCLUSIONS: We provided an overview of contextual factors that may influence SurPass implementation. Solutions should be found to overcome barriers and ensure effective implementation of SurPass v2.0 into routine clinical care. IMPLICATIONS FOR CANCER SURVIVORS: These findings will be used to inform on an implementation strategy tailored for the six centres.
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Background: Inpatient pediatric obesity treatments are highly effective, although dropouts and weight regain threaten long-term results. Preliminary data indicate that leptin, adiponectin, and cardiometabolic comorbidities might predict treatment outcomes. Previous studies have mainly focused on the individual role of adipokines and comorbidities, which is counterintuitive, as these risk factors tend to cluster. This study aimed to predict the dropouts and treatment outcomes by pre-treatment patient characteristics extended with cardiometabolic comorbidities (individually and in total), leptin, and adiponectin. Methods: Children aged 8-18 years were assessed before, immediately after and 6 months after a 12-month inpatient obesity treatment. Anthropometric data were collected at each visit. Pre-treatment lipid profiles; glucose, insulin, leptin, and adiponectin levels; and blood pressure were measured. The treatment outcome was evaluated by the change in body mass index (BMI) standard deviation score (SDS) corrected for age and sex. Results: We recruited 144 children with a mean age of 14.3 ± 2.2 years and a mean BMI of 36.7 ± 6.2 kg/m2 corresponding to 2.7 ± 0.4 BMI SDS. The 57 patients who dropped out during treatment and the 44 patients who dropped out during aftercare had a higher pre-treatment BMI compared to the patients who completed the treatment (mean BMI, 38.3 ± 6.8 kg/m2 vs 35.7 ± 5.5 kg/m2) and those who completed aftercare (mean BMI, 34.6 ± 5.3 kg/m2 vs 37.7 ± 6.3 kg/m2) (all p<0.05). Additionally, aftercare attenders were younger than non-attenders (mean age, 13.4 ± 2.3 years vs 14.9 ± 2.0, p<0.05).Patients lost on average 1.0 ± 0.4 SDS during treatment and regained 0.4 ± 0.3 SDS post-treatment corresponding to regain of 43 ± 27% (calculated as the increase in BMI SDS post-treatment over the BMI SDS lost during treatment). A higher BMI and more comorbidities inversely predicted BMI SDS reduction in linear regression (all p<0.05).The absolute BMI SDS increase after returning home was predicted by pre-treatment leptin and systolic blood pressure, whereas the post-treatment BMI SDS regain was predicted by pre-treatment age, leptin, and adiponectin levels (all p<0.05) in multivariate linear regressions. Conclusion: Patients who need treatment the most are at increased risk for dropouts and weight regain, emphasizing the urgent need for interventions to reduce dropout and support inpatients after discharge. Furthermore, this study is the first to report that pre-treatment leptin and adiponectin levels predict post-treatment BMI SDS regain, requiring further research.
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Enfermedades Cardiovasculares , Obesidad Infantil , Adipoquinas , Adiponectina , Adolescente , Niño , Humanos , Leptina , Obesidad Infantil/terapia , Centros de Rehabilitación , Factores de Riesgo , Resultado del Tratamiento , Aumento de Peso , Pérdida de PesoRESUMEN
Background: Currently available treatment programs for children with obesity only have modest long-term results, which is (at least partially) due to the poorer self-control observed within this population. The present trial aimed to determine whether an online self-control training, training inhibition, and redirecting attentional bias, can improve the short- and long-term treatment outcome of (in- or outpatient) child obesity treatment programs. Methods: In this double-blind multi-center randomized controlled trial (RCT), participants aged 8-18 years with obesity were allocated in a 1:1 ratio to receive an online self-control or sham training added to their in- or outpatient multidisciplinary obesity treatment (MOT) program. The primary endpoint was BMI SDS. Data were analyzed by linear mixed models and the main interactions of interest were randomization by time and randomization by number of sessions, as the latter was cumulatively expressed and therefore represents the effect of increasing dose over time. Results: One hundred forty-four inpatient (mean age 14.3 ± 2.2 years, BMI 2.7 ± 0.4 SDS, 42% male) and 115 outpatient children (mean age 11.9 ± 2.1 years, BMI 2.4 ± 0.4 SDS, 45% male) were included. Children's BMI lowered significantly during treatment in both the in- and outpatient treatment centers, p < 0.001. In a mixed model with BMI as dependent variable, randomization by time was non-significant, but the number of self-control trainings (randomization * number of sessions) interacted significantly with setting and with age (p = 0.002 and p = 0.047), indicating a potential effect in younger inpatient residents. Indeed, a subgroup analysis on 22 inpatient children of 8-12 years found a benefit of the number of self-control trainings on BMI (p = 0.026). Conclusions: The present trial found no benefit of the self-control training in the entire study population, however a subgroup of young, inpatient participants potentially benefited.
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BACKGROUND: Obesity and age influence the reliability of dual energy X-ray absorptiometry scanning (DEXA) and bioimpedance spectroscopy (BIS). Both are used in clinical settings, but have not been compared for measurements in obese children. We compared DEXA and BIS for evaluating body composition and inherent changes in obese children before and after a 10-month weight loss programme. METHODS: DEXA and BIS were used to evaluate 130 patients at baseline and 75 at follow-up. We tested agreement between the two techniques using Bland-Altman plots and proportional bias using Passing-Bablok regressions. RESULTS: The Bland-Altman plots showed wide agreement limits before and after weight loss and when monitoring longitudinal changes. At baseline, the Passing-Bablok regressions revealed a proportional bias for all body compartments. After significant weight loss no proportional bias was found for fat mass and percentage, although BIS systematically underestimated fat mass by 2.9 kg. Longitudinally, no proportional bias was found in the measured changes of absolute fat, fat-free mass and fat-free percentage between both methods, although BIS systematically underestimated fat and fat-free mass by 2.6 and 0.7 kg, respectively. CONCLUSION: While BIS and DEXA are not interchangeable at baseline, the agreement between the two improved after significant weight loss. Proportional changes in fat mass, fat-free mass and fat-free percentage were similar for both techniques. BIS is a viable alternative to DEXA for future paediatric obesity studies measuring treatment effect at group levels, but is not superior to DEXA and cannot be used for monitoring individual changes due to wide limits of agreement.
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Composición Corporal , Pérdida de Peso , Absorciometría de Fotón , Tejido Adiposo , Índice de Masa Corporal , Niño , Impedancia Eléctrica , Humanos , Obesidad , Reproducibilidad de los Resultados , Análisis EspectralRESUMEN
BACKGROUND AND OBJECTIVES: Endothelial dysfunction is the first, although reversible, sign of atherosclerosis and is present in obese adolescents. The primary end point of this study was to investigate the influence of a multicomponent treatment on microvascular function. Additional objectives and end points were a reduced BMI SD score, improvements in body composition, exercise capacity, and cardiovascular risk factors, an increase in endothelial progenitor cells (EPCs), and a decrease in endothelial microparticles (EMPs). METHODS: We used a quasi-randomized study with 2 cohorts of obese adolescents: an intervention group (n = 33; 15.4 ± 1.5 years, 24 girls and 9 boys) treated residentially with supervised diet and exercise and a usual care group (n = 28; 15.1 ± 1.2 years, 22 girls and 6 boys), treated ambulantly. Changes in body mass, body composition, cardiorespiratory fitness, microvascular endothelial function, and circulating EPCs and EMPs were evaluated after 5 months and at the end of the 10-month program. RESULTS: Residential intervention decreased BMI and body fat percentage, whereas it increased exercise capacity (P < .001 after 5 and 10 months). Microvascular endothelial function also improved in the intervention group (P = .04 at 10 months; + 0.59 ± 0.20 compared with + 0.01 ± 0.12 arbitrary units). Furthermore, intervention produced a significant reduction in traditional cardiovascular risk factors, including high-sensitivity C-reactive protein (P = .012 at 10 months). EPCs were increased after 5 months (P = .01), and EMPs decreased after 10 months (P = .004). CONCLUSIONS: A treatment regimen consisting of supervised diet and exercise training was effective in improving multiple adolescent obesity-related end points.
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Aterosclerosis/prevención & control , Dieta Reductora/métodos , Endotelio Vascular/fisiopatología , Terapia por Ejercicio/métodos , Obesidad/terapia , Vasodilatación/fisiología , Pérdida de Peso/fisiología , Adolescente , Aterosclerosis/etiología , Aterosclerosis/fisiopatología , Índice de Masa Corporal , Niño , Ejercicio Físico , Femenino , Humanos , Estilo de Vida , Masculino , Obesidad/complicaciones , Obesidad/fisiopatología , Pronóstico , Factores de RiesgoRESUMEN
The aim of this randomized, placebo-controlled study was to explore the effect of metformin in children with a neurogenic or myogenic motor deficit, who are therefore prone to develop overweight, adiposity, and insulin resistance. Study participants (n = 42) had a mean age of 15.5 yr, a short stature (height -2.4 SD), a relatively high BMI (+1.7 SD), and a high body fat fraction (41.9% or +2.8 SD). Abdominal CT confirmed the high fat mass and disclosed a high fraction of visceral fat. As expected, insulin resistance was increased. As compared to placebo, metformin intake for 6 months exerted an insulin sensitizing effect and lowered weight (mean difference of 2 kg within 6 months, p = 0.007) and BMI (p = 0.016). Weight loss appeared to be primarily due to loss of visceral fat ( approximately 20% vs. placebo; p < 0.0001). Results were similar across diagnostic subgroups. In conclusion, metformin treatment for 6 months was associated with a rise in insulin sensitivity and with a reduction of visceral adiposity in children and adolescents with a primary muscle disorder or with a neural tube defect. These findings suggest that insulin resistance underpins, at least partly, the overweight and visceral adiposity of these patients, who are not necessarily obese.
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Hipoglucemiantes/uso terapéutico , Grasa Intraabdominal/efectos de los fármacos , Metformina/uso terapéutico , Distrofia Muscular de Duchenne/tratamiento farmacológico , Disrafia Espinal/tratamiento farmacológico , Aumento de Peso/efectos de los fármacos , Adolescente , Glucemia/efectos de los fármacos , Índice de Masa Corporal , Niño , Femenino , Humanos , Insulina/sangre , Resistencia a la Insulina/fisiología , Masculino , Obesidad/tratamiento farmacológicoRESUMEN
We report on 3 term newborns with the association of patent ductus arteriosus (PDA) and severe hypothyroidism attributed to thyroid agenesis (n = 2) or feto-maternal pit-1 deficiency (n = 1). This association suggests that sufficient thyroid hormone availability is among the prerequisites for normal postnatal ductal closure. Unravelling the maturational effects of thyroid hormone on the ductus arteriosus might also be relevant in preterm infants as further studies of the perinatal thyroid axis may clarify the potential role of thyroid hormone in PDA closure in preterm infants. Based on our observation, hypothyroidism should be considered in term infants with PDA.
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Conducto Arterioso Permeable/complicaciones , Hipotiroidismo/complicaciones , Glándula Tiroides/anomalías , Hormonas Tiroideas/metabolismo , Conducto Arterioso Permeable/metabolismo , Conducto Arterioso Permeable/patología , Femenino , Humanos , Hipotiroidismo/metabolismo , Hipotiroidismo/patología , Recién NacidoRESUMEN
To determine whether hyperinsulinemia and reduced insulin sensitivity in individuals born small for gestational age (SGA) could be related to persisting abnormalities of the GH/IGF-I axis, we assessed overnight GH secretory profiles and measured fasting glucose, insulin, intact and 32,33 split proinsulin, and IGF-I levels in 16 short SGA children (age range 2.3-8.0 y) and in controls. Insulin sensitivity was calculated using the homeostasis model. Compared with short normal-birthweight controls (n = 7, age range 2.3-5.0 y), short SGA children had higher fasting insulin levels (means: 26.8 vs 20.6 pmol/L, p = 0.02), lower insulin sensitivity [means: 204 vs 284 %homeostasis model assessment (HOMA), p = 0.01], and higher beta cell function (112 vs 89 %HOMA, p = 0.04). SGA children also had lower levels of IGFBP-1 (87.0 vs 133.8, p = 0.04), but similar IGF-I levels (IGF-I SDS: -1.1 vs -1.7, p = 0.4). Compared with normal-height controls (n = 15, age range 5.6-12.1 y), SGA children had higher overnight GH secretion (GH maximum: 55.9 vs 39.6 mU/L, p = 0.01; mean: 13.1 vs 8.9, p = 0.004; minimum: 1.2 vs 0.6, p = 0.02). Interestingly, among SGA children, fasting insulin levels and insulin sensitivity were significantly related to overnight GH secretion (insulin sensitivity vs maximum GH: r = -0.68, p = 0.01; vs GH pulse amplitude r = -0.71, p = 0.007). The only hormone level significantly related to current height velocity was C-peptide (r = 0.75, p = 0.008). In conclusion, elevated fasting insulin levels and reduced insulin sensitivity in short SGA children was related to elevated levels of overnight GH secretion. We hypothesize that resistance to the somatotropic actions of GH and IGF-I in short SGA children may contribute directly to reduced insulin sensitivity.
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Hormona del Crecimiento/fisiología , Recién Nacido Pequeño para la Edad Gestacional , Resistencia a la Insulina , Factor I del Crecimiento Similar a la Insulina/fisiología , Niño , Preescolar , Femenino , Humanos , Recién Nacido , MasculinoRESUMEN
GH therapy variably reduces the height deficit of short children born small for gestational age (SGA) but is associated with hyperinsulinemia. Intermittent, higher-dose GH regimens may be alternatives to continuous, lower-dose treatment. We examined whether the growth response to GH therapy is related to pre-treatment indices of endogenous somatotropic activity, and studied the reversibility of the insulin resistant state induced by GH. 13 non-GH deficient short SGA children were randomised to high-dose GH (100 mcg/kg/day) by daily sc injection (n = 9), or no GH treatment (n = 4) for 2 years (2/4 controls subsequently received GH treatment). Overnight GH profiles were performed at baseline; intravenous glucose tolerance tests were performed at baseline, yearly on GH treatment, and 3 months post-GH treatment. Fasting glucose, insulin and proinsulin levels were measured, insulin sensitivity estimated using Bergman's minimal model, and glucose tolerance calculated from rate of glucose disappearance. In all GH-treated children, gain in height SDS (mean gain yr 1 = +1.2 SDS) was inversely related to baseline peak overnight GH (r = -0.88, n = 10, p = 0.0008), IFG-I (r = -0.74, n = 11, p = 0.009), and fasting insulin levels (r = -0.71, n = 11, p = 0.014). GH treatment increased fasting glucose (means: baseline vs. yr 2: 3.7 vs. 4.4 mmol/l, p = 0.005), insulin (3.8 vs. 13.9 mU/l, p = 0.0002), and proinsulin levels (1.7 vs. 4.5 pmol/l, p = 0.004), and decreased insulin sensitivity (26.9 vs. 4.0 per min/mU/1 x 10(4), p = 0.002). Glucose tolerance initially decreased (baseline: 2.62 min(-1); yr 1: 2.18, p = 0.02; yr 2: 2.39, p = 0.12). However, by 3 months post-GH treatment significant improvements were seen in fasting insulin (post-GH: 5.2 mU/1, p = 0.0003 vs. yr 2), proinsulin (1.7 pmol/l, p = 0.002), and insulin sensitivity (17.6 per min/mU/1 x 10(4), p = 0.0001). Post-GH treatment, fasting glucose levels (4.1 mmol/l, p = 0.04) and glucose tolerance (2.49 min(-1), p = 0.4) were similar to baseline, and the slight increase in fasting insulin levels (5.2 mU/1, p = 0.04) was similar to that observed in non-GH treated children over the 2 yr study period (baseline vs. 2 years: 3.9 vs. 5.9 mU/1, n = 4). In conclusion, in this study of 13 short non-GH-deficient SGA children, high-dose GH therapy induced growth responses that were associated with reversible decreases in insulin sensitivity, and that were predicted by pretreatment markers of endogenous, but not stimulated, somatotropic activity.