Health-related quality of life experiences in children and adolescents born with esophageal atresia: A Swedish-German focus group study.

BACKGROUND: Esophageal atresia (EA) is a rare malformation of the esophagus, which needs surgical treatment. Survival rates have reached 95%, but esophageal and respiratory morbidity during childhood is frequent. Child and parent perspectives and cultural and age-specific approaches are fundamental in understanding children's health-related quality of life (HRQoL) and when developing a pediatric HRQoL questionnaire. We aimed to increase the conceptual and cross-cultural understanding of condition-specific HRQoL experiences among EA children from Sweden and Germany and investigate content validity for an EA-specific HRQoL questionnaire. METHODS: Eighteen standardized focus groups (FGs) with 51 families of EA children aged 2-17 years in Sweden (n = 30 families) and Germany (n = 21 families) were used to explore HRQoL experiences, which were content analyzed into HRQoL domains. The Swedish HRQoL domains were analyzed first and used as framework to evaluate HRQoL content reported in the German FGs. HRQoL experiences were then categorized as physical, social, and emotional HRQoL burden or resource. RESULTS: One thousand nine hundred eight HRQoL statements were recorded. All nine EA-specific HRQoL domains identified in the Swedish FGs (eating, social relationships, general life issues, communication, body issues, bothersome symptoms, confidence, impact of medical treatment, and additional difficulties due to concomitant anomalies) were recognized in the FGs held in Germany, and no additional EA-specific HRQoL domain was found. The HRQoL dimensions referenced physical burden (n = 655, 34.5%), social burden (n = 497, 26.0%), social resources (n = 303, 15.9%), emotional burden (n = 210, 11.0%), physical resources (n = 158, 8.3%), and emotional resources (n = 85, 4.5%). CONCLUSION: This first international FG study to obtain the EA child and his or her parents' perspective on HRQoL suggests Swedish-German qualitative comparability of the HRQoL domains and content validity for a cross-cultural EA-specific HRQoL questionnaire. EA children make positive and negative HRQoL experiences, but prominently related to physical and social burden, which underlines appropriate follow-up care and future research.

Health-related quality of life among children, young people and adults with esophageal atresia: a review of the literature and recommendations for future research.

PURPOSE: Esophageal atresia (EA) is a rare congenital malformation requisite of surgical treatment. Survival rates have significantly increased in recent decades, but treated children are at risk of chronic morbidity. This study aims to review the literature on health-related quality of life (HRQOL) in patients with EA and describe the questionnaires used. METHODS: A literature search was conducted in Pubmed, Cinahl and PsycINFO, from inception to January 2015. A meta-analysis of studies investigating HRQOL in patients with EA compared to healthy references was performed. The effect size was calculated as Cohen's d. RESULTS: Twelve articles (published 1995-2014) describing HRQOL among children, young people and adults were identified. Only European studies were found, and these had variable sample sizes (8-128). Overall HRQOL was reported to be reduced in five of the seven studies that compared overall HRQOL with a healthy reference population. Impaired physical or general health was described in eight articles. In the meta-analysis, eight eligible studies provided 16 estimates of the effect of EA, six of which reached statistical significance for worse HRQOL (p < 0.05). Using Cohen's criteria, the pooled estimate of the effect of EA was small for overall and physical HRQOL, and <0.2 for the mental and social HRQOL components. Altogether, 15 different questionnaires were used, and none were condition specific for EA. CONCLUSIONS: Different findings are reported; however, this study suggests that patients with EA may have a reduced HRQOL. Moreover, HRQOL is not adequately measured in this group. Additional research is required.

Exploring the limitations of language interpretation: A qualitative study on clinicians' experiences at French Office of Immigration and Integration.

The concordance of communication between patients and health professionals is essential to promoting positive health outcomes. However, concordance may be broken where language barriers exist therefore creating a need to use interpretation services. This is the case when rapid diagnostic testing (RDT) of HIV, HBV, and HCV is offered to migrants. The use of interpreters to establish communication with patients having limited French proficiency (LFP) however, is often not used and can be problematic. Despite being offered, interpretation services are frequently underutilised, which makes communication challenging. This problem has not received enough attention in the literature, particularly in a technologically advanced setting where solutions may be found. Our objective was to explore how interpreters are used within the context of medical consultations when RDT for HIV, HBV, and HCV is offered to legal migrants with LFP. A cross-sectional qualitative study was used with a purposive sample that included doctors and nurses who had conducted rapid screening tests with migrants in four centers in France and who had access to interpretive services. Semi-structured interviews explored healthcare providers' (HP) use of interpreters at the OFII. The use of professional or ad hoc interpreters, telephone interpreters, and the equivalence of concepts such as health literacy between the HP and the interpreter were explored. The utility of a new tool to promote communication concordance was evaluated. Twenty interviews were conducted with eleven doctors and nine nurses with a median age of 58 years (25-67 years). All participants had access to interpretive services although many did not solicit them because of 1) unawareness on how to use the services, 2) preconceived notions of the length of time to involve an interpreter and how this would add to consultation times, or 3) the proximity of an ad hoc interpreter. Not using interpreter services could result in RDTs not being offered to immigrants. Subjects such as confidentiality, the embarrassment of a third party's presence, the lack of appropriate training and differing levels of health literacy were also discussed by participants. Insight from HPs allows us to better understand how both telephone and in-person interpretation are used, viewed, and why they are underused to communicate with limited French language skills patients. Our findings will help us develop a conceptual model for a digital communication tool to overcome barriers with migrant patients with limited French language skills.

Body size and lifestyle in an urban population entering adulthood: the 'Grow up Gothenburg' study.

AIM: To present data on body size, lifestyle and health status in students in their final year in high schools in Gothenburg area, Sweden, with specific attention to origin and gender. METHODS: Weight, height and waist circumference were measured by standardized procedures. Self-administered questionnaires described dietary habits, sleep, physical activity, body image, country of origin and general health. RESULTS: Eighty-six percent of participants, (2600 girls, 2714 boys, mean age 18.6 years) were of Nordic origin, 86% reported no chronic health problems and 14%/19% of girls/boys were overweight or obese. Girls consumed more vegetables and fruits and fewer sweet drinks than boys, while breakfast consumption was most common in Nordic groups. Boys reported more positive answers than girls regarding body image. Nordic girls had more negative body image and higher morbidity compared with other groups. CONCLUSION: Within this generally healthy cohort, boys were more likely to be overweight/obese than girls, although paradoxically boys were more satisfied with their appearance. Nordic girls constitute a group with particularly high risk of reporting low body self-esteem and chronic morbidity. In the longer term, the current cross-sectional data on body size, lifestyle and health will provide important baseline information for future follow-up studies of health outcomes in later life.

The Development of an mHealth Tool for Children With Long-term Illness to Enable Person-Centered Communication: User-Centered Design Approach.

BACKGROUND: Children with long-term illnesses frequently experience symptoms that could negatively affect their daily lives. These symptoms are often underreported in health care. Despite a large number of mobile health (mHealth) tools, few are based on a theoretical framework or supported by scientific knowledge. Incorporating universal design when developing a product can promote accessibility and facilitate person-centered communication. OBJECTIVE: The aim of this study is to identify the symptom-reporting needs of children with cancer and congenital heart defects that could be satisfied by using a mobile app. Another aim is to evaluate how the child might interact with the app by considering universal design principles and to identify parents' views and health care professionals' expectations and requirements for an mHealth tool. METHODS: User-centered design is an iterative process that focuses on an understanding of the users. The adapted user-centered design process includes 2 phases with 4 stages. Phase 1 involved interviews with 7 children with long-term illnesses, 8 parents, and 19 health care professionals to determine their needs and wishes for support; a workshop with 19 researchers to deepen our understanding of the needs; and a workshop with developers to establish a preliminary tool to further investigate needs and behaviors. Phase 2 involved interviews with 10 children with long-term illnesses, 9 parents, and 21 health care professionals to evaluate the mock-up (prototype) of the mHealth tool. Data were synthesized using the interpretive description technique. RESULTS: A total of 4 aspects of needs emerged from the synthesis of the data, as follows: different perspectives on provided and perceived support; the need for an easy-to-use, non-clinic-based tool to self-report symptoms and to facilitate communication; the need for safety by being in control and reaching the child's voice; and a way of mapping the illness journey to facilitate recall and improve diagnostics. The children with long-term illnesses expressed a need to not only communicate about pain but also communicate about anxiety, fatigue, fear, and nausea. CONCLUSIONS: The findings of this study indicated that the PicPecc (Pictorial Support in Person-Centered Care for Children) app is a potential solution for providing communicative support to children with long-term illnesses dealing with multiple symptoms and conditions. The interview data also highlighted symptoms that are at risk of being overlooked if they are not included in the mobile app. Further studies are needed to include usability testing and evaluation in hospitals and home care settings.

Swedish translation and cross-cultural adaptation of eight pediatric item banks from the Patient-Reported Outcomes Measurement Information System (PROMIS)®.

BACKGROUND: This study is part of the Swedish initiative for the establishment of standardized, modern patient-reported measures for national use in Swedish healthcare. The goal was to translate and culturally adapt eight pediatric Patient-Reported Outcomes Measurement Information System (PROMIS®) item banks (anger, anxiety, depressive symptoms, family relationships, fatigue, pain interference, peer relationships and physical activity) into Swedish. METHODS: Authorization to translate all currently available pediatric PROMIS item banks (autumn, 2016) into Swedish was obtained from the PROMIS Health Organization. The translation followed the Functional Assessment of Chronic Illness Therapy translation recommendations with one major modification, which was the use of a bilingual multi-professional review workshop. The following steps were applied: translation, reconciliation, a two-day multi professional reviewer workshop, back translation, and cognitive debriefing with eleven children (8-17 years) before final review. The bilingual multi-professional review workshop provided a simultaneous, in-depth assessment from different professionals. The group consisted of questionnaire design experts, researchers experienced in using patient-reported measures in healthcare, linguists, and pediatric healthcare professionals. RESULTS: All item banks had translation issues that needed to be resolved. Twenty-four items (20.7%) needed resolution at the final review stage after cognitive debriefing. The issues with translations included 1. Lack of matching definitions with items across languages (6 items); 2. Problems related to language, vocabulary, and cultural differences (6 items); and 3. Difficulties in adaptation to age-appropriate language (12 items). CONCLUSIONS: The translated and adapted versions of the eight Swedish pediatric PROMIS item banks are linguistically acceptable. The next stage will be cross-cultural validation studies in Sweden. Despite the fact that there are cultural differences between Sweden and the United States, our translation processes have successfully managed to address all issues. Expert review groups from already-established networks and processes regarding pediatric healthcare throughout the country will facilitate the future implementation of pediatric PROMIS item banks in Sweden.

Psychometrics of three Swedish physical pediatric item banks from the Patient-Reported Outcomes Measurement Information System (PROMIS)®: pain interference, fatigue, and physical activity.

BACKGROUND: The Patient-Reported Outcomes Measurement Information System (PROMIS®) aims to provide self-reported item banks for several dimensions of physical, mental and social health. Here we investigate the psychometric properties of the Swedish pediatric versions of the Physical Health item banks for pain interference, fatigue and physical activity which can be used in school health care and other clinical pediatric settings. Physical health has been shown to be more important for teenagers' well-being than ever because of the link to several somatic and mental conditions. The item banks are not yet available in Sweden. METHODS: 12- to 19-year-old participants (n = 681) were recruited in public school settings, and at a child- and psychiatric outpatient clinic. Three one-factor models using CFA were performed to evaluate scale dimensionality. We analyzed monotonicity and local independence. The items were calibrated by fitting the graded response model. Differential Item analyses (DIF) for age, gender and language were calculated. RESULTS: As part of the three one-factor models, we found support that each item bank measures a unidimensional construct. No monotonicity or local dependence were found. We found that 11 items had significant lack of fit in the item response theory (IRT) analyses. The result also showed DIF for age (seven items) and language (nine items). However, the differences on item fits and effect sizes of McFadden were negligible. After considering the analytic results, graphical illustration, item content and clinical relevance we decided to keep all items in the item banks. CONCLUSIONS: We translated and validated the U.S. PROMIS item banks pain interference, fatigue and physical activity into Swedish by applying CFA, IRT and DIF analyses. The results suggest adequacy of the translations in terms of their psychometrics. The questionnaires can be used in school health and other pediatric care. Future studies can be to use Computerized Adaptive Testing (CAT), which provide fewer but reliable items to the test person compared to classical testing.

Parent-child-agreement on health-related quality of life and its determinants in patients born with Esophageal Atresia: a Swedish-German cross-sectional study.

BACKGROUND: The aim was to compare parent and child-reported health-related quality of life (HRQOL) of children born with esophageal atresia (EA) and determine factors that affect the level of parent-child agreement. METHODS: We included 63 parent-child dyads of children born with EA aged 8-18 from Germany and Sweden. The generic PedsQL 4.0™ questionnaire and the condition-specific EA QOL questionnaire were used to assess children's HRQOL from parents' and children's perspectives. The PedsQL™ Family Impact Module was used to assess parental HRQOL and Family Functioning. RESULTS: On an individual level, intra-class correlation coefficients indicated strong levels of parent-child agreement (.61-.97). At the group level, the analyses showed no significant differences between the responses of parents and children. When a disagreement occurred, parents were more likely to rate generic HRQOL lower than the children (19-35%) and condition-specific HRQOL higher than the children (17-33%). Findings of the binary logistic regression analyzes showed that the child's age, gender, and country (Germany vs. Sweden) were significant predictors of parent-child agreement in condition-specific HRQOL. We did not identify any significant variables that explain agreement for the generic HRQOL. CONCLUSION: The parent-child agreement is mostly good, suggesting that parent-reports are a reliable source of information. However, discrepancies may occur and can be explained by the child's age, gender, and country (Sweden vs. Germany). Both perspectives are essential sources for treating EA patients and should not be considered right or wrong. Instead, this information broadens the perspective on pediatric EA patients.

Health literacy in a complex digital media landscape: Pediatric obesity patients' experiences with online weight, food, and health information.

This study aimed to explore experiences with online information regarding food, weight management, and health in a group of adolescents in treatment for obesity. Individual semi-structured interviews with 20 adolescents were conducted. Participants used a screen-recorded laptop to demonstrate their search procedures and online information sources. The transcribed interviews were categorized using qualitative content analysis. The adolescents described both encouraging and discouraging experiences. On one hand, they said that online forums could provide nutritious meal ideas and inspiration as well as social support for behavior change. On the other hand, they mentioned that there was a confusing amount of misleading commercial content online and also experiences of peer-facilitated food marketing in online networks. An overarching theme was generated: social media might be a resource for health inspiration, health information, and social support, but requires awareness and competencies. Implications for clinical practice are discussed in light of these findings.

A population-based and case-controlled study of children and adolescents with narcolepsy: Health-related quality of life, adaptive behavior and parental stress.

OBJECTIVE: To investigate health-related quality of life (HrQoL) and adaptive behavior in young people with narcolepsy and stress among their parents. METHODS: In a cross-sectional exploratory quantitative study design, 37 young people with narcolepsy (8-20 years of age) and their parents were recruited. Thirty-one had post-H1N1 vaccination-related narcolepsy (PHV) and six had narcolepsy not related to PHV (nPHV). In addition, 40 age- and gender-matched controls (aged 5-20 years) were recruited. RESULTS: Thirty-one patients completed the generic HrQoL questionnaire KIDSCREEN and the disease-specific NARQoL-21. HrQoL was found to be significantly diminished in all domains in the PHV group (p = 0.001) and in the School/Concentration domain (p = 0.004) in the nPHV group compared to age- and gender-matched controls. The Adaptive Behavior Assessment System was completed by parents of 32 patients. They rated their children significantly lower in the General adaptive composite (p = 0.026) and the Conceptual (p = 0.050) and Social composite scores (p = 0.001) compared with reference data on healthy Swedish children's and young people's adaptive behavior. Parents of 36 patients filled in the 36-item short form of the Parenting Stress Index questionnaire. They rated significantly higher Total stress, Parent-child dysfunctional interaction, and Difficult child scores compared with parents of controls (p = 0.001, p = 0.005, p < 0.001). CONCLUSIONS: Children with narcolepsy have diminished HrQoL compared with controls. Parents of children with narcolepsy experience impaired adaptive behavior in their children and high levels of parenting stress. Identifying the contributory factors is necessary, and early intervention is crucial in order to improve the HrQoL of these children and their families.

Self-presentation in digital media among adolescent patients with obesity: Striving for integrity, risk-reduction, and social recognition.

BACKGROUND: Emerging research suggests that social media has the potential in clinical settings to enhance interaction with and between pediatric patients with various conditions. However, appearance norms and weight stigmatization can make adolescents with obesity uncomfortable about using these visual-based media. It is therefore important to explore these adolescents' perspectives to identify the implications and concerns regarding the use of social media in clinical settings. OBJECTIVE: To explore the experiences of adolescents in treatment for obesity in terms of how they present themselves on social media, their rationale behind their presentations, and their feelings related to self-presentation. METHODS: Interviews were conducted with 20 adolescents enrolled in a pediatric outpatient obesity clinic, then transcribed and categorized using qualitative content analysis and Goffman's dramaturgical model. Participants used a screen-recorded laptop to demonstrate their online self-presentation practices.Findings: Adolescent girls and boys undergoing treatment for obesity used visual-based social media, but girls in particular experienced weight stigma online and undertook self-presentation strategies to conceal weight-related content such as avoiding showing close-up photos of their bodies and not posting images of unhealthy "fattening" foods. Participants perceived the potential use of social media in clinical settings as being too risky and private. CONCLUSIONS: Given the complexity of general visual-based social media use by adolescents, and not wanting their patient status to be visible to peers, healthcare should primarily focus on working with more restricted instant messaging when engaging with adolescents with obesity.

Health-related quality of life experiences among children and adolescents born with esophageal atresia: Development of a condition-specific questionnaire for pediatric patients.

BACKGROUND/PURPOSE: The aims were to present the framework for the development a condition-specific health-related quality-of-life (HRQOL) questionnaire for children with esophageal atresia (EA) and to describe HRQOL experiences reported by children and by their parents. METHODS: Utilizing the well-established DISABKIDS methodology, standardized focus group discussions were held and transcribed. HRQOL experiences were identified, content analyzed and evaluated using descriptive statistics. RESULTS: 30 families (18 children 8-17years, 32 parents of children 2-17years) participated in ten focus group discussions. 1371 HRQOL experiences were identified referencing social, emotional and physical aspects of eating and drinking (n=368), relationships with other people (n=283), general life issues; physical activity, sleep and general health (n=202), communicative/interactive processes of one's health condition (n=161), body issues (n=109), bothersome symptoms (n=81), impact of health care use/medical treatment (n=78), confidence in oneself and in the future (n=65) and difficulties because of concomitant anomalies (n=24). A basis of two age-related HRQOL questionnaires for children with EA (2-7years, 8-17years) was subsequently constructed. CONCLUSIONS: EA interacts with various aspects of the child's life. In addition to HRQOL issues of eating and drinking, social dimensions like relationships and interactions with other people seem to be prominent condition-specific HRQOL parameters. The settings for the development of the first condition-specific HRQOL questionnaires for patients with EA are established.

Growth Hormone Treatment Improves Cognitive Function in Short Children with Growth Hormone Deficiency.

BACKGROUND/AIMS: We investigated the association between cognition and growth hormone (GH) status and GH treatment in short prepubertal children with broadly ranging GH secretion. METHODS: A total of 99 children (age 3-11 years), 41 with GH deficiency (GHD) and 58 with idiopathic short stature (ISS), were randomized to a fixed dose (43 µg/kg/day) or a prediction model-guided individualized dose (17-100 µg/kg/day) and followed up for 24 months. In a longitudinal and mixed within- and between-subjects study, we examined clinical effect size changes, measured by Cohen's d, in full-scale IQ (FSIQ) and secondary IQ indices. RESULTS: Significant increases giving medium effect size in FSIQ (p = 0.001, Cohen's d = 0.63), performance IQ (p = 0.001, Cohen's d = 0.65) and processing speed (p = 0.005, Cohen's d = 0.71) were found in the GH-deficient group. In contrast, perceptual organization only increased in the ISS group (p = 0.001, Cohen's d = 0.53). Baseline IQ was normally distributed with small but significant differences between the groups: GH-deficient children had lower FSIQ (p = 0.042) and lower performance IQ (p = 0.021). Using multiple regression analysis, 40% of the variance in delta processing speed scores (0-24 months) was explained by GHmax and IGF-ISDS at baseline. CONCLUSION: IQ, specifically fluid intelligence, increased in the GH-deficient children. The pretreatment status of the GH/IGF-I axis was significantly predictive for these changes. © 2015 S. Karger AG, Basel.

Growth-related quality of life.

Measuring a child's quality of life (QoL) is a complex dynamic, involving consideration of both present circumstances and future limitations. In relation to short stature, health-related QoL, with its emphasis on current health status, may be inadequate to describe QoL in children with treatable growth problems. Growth problems concern not only current physical health (being) and adaptation to the physical and social environment (belonging) but also what will happen in the future as a consequence of growth problems and the possibility of achieving the potential of the individual (becoming). These three aspects of QoL should be included in growth-related QoL in order to reflect the reduced QoL resulting from continued short stature and the potential benefit of growth enhancement. Future QoL instruments for growth-related problems should incorporate aspects of 'becoming' and the long-term consequences of growth improvement for the individual.

Improvements in behaviour and self-esteem following growth hormone treatment in short prepubertal children.

BACKGROUND/AIMS: To evaluate effects of growth hormone (GH) treatment on behaviour and psychosocial characteristics in short-stature children. METHODS: 99 referred prepubertal non-familiar short-stature children (32 GH deficiency; 67 idiopathic short stature) aged 3-11 years, randomized to fixed or individual GH doses and their parents completed questionnaires (Child Behaviour Checklist, Birleson Depression Self-Report Scale, Abbreviated Parent-Teacher Questionnaire, I Think I Am, Well-Being Visual-Analogue Scales for Short-Stature Children) at baseline (BL) and after 3, 12, and 24 months. RESULTS: At BL, children showed higher levels of internalizing behaviour (p < 0.001), lower levels of externalizing behaviour (p < 0.006) and self-esteem (p < 0.001) compared to reference values. During GH treatment, behavioural measures (p < 0.001) and depression (p < 0.01) changed towards the mean of the population within the first 3 months and remained improved to 24 months. Self-esteem improved at all time points (p < 0.001), and in all subgroups, as did well-being dimensions stability and mood (p < 0.05). Multiple regression analysis showed that greater improvements were related to lower BL value, height gain, higher maximal GH value, being older, and being male. CONCLUSION: On GH treatment, prepubertal short children significantly improved on behavioural, depression, and psychosocial evaluations over a 2-year period of GH treatment. Most change occurred within the first 3 months, which highlights this short period as important not only for growth and metabolic changes but also for behaviour and psychosocial improvements following GH treatment.