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- Dellenmark-Blom, Michaela, 1983, et al.
(författare)
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Establishment of a condition-specific quality-of-life questionnaire for children born with esophageal atresia aged 2-7 across 14 countries
- 2023
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Ingår i: Frontiers in Pediatrics. - 2296-2360. ; 11
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Tidskriftsartikel (refereegranskat)abstract
- BackgroundEsophageal atresia (EA) is a rare congenital anomaly characterized by a discontinuity of the esophagus. Following surgical repair, survival rates have improved dramatically the past decenniums and today exceed 90%, but the children commonly present with esophageal and respiratory morbidity. In 2018, a condition-specific quality-of-life questionnaire for children with esophageal atresia (EA) aged 2-7 in Sweden-Germany was finalized (The EA-QOL questionnaire). The study aim was to describe the evaluation of the new translations across 12 new countries in Europe, Asia, Africa, Central-and North America.MethodsFollowing forward-backward translation into the new languages, the 17-item EA-QOL questionnaire was tested in cognitive debriefing interviews with parents of children with EA aged 2-7. Parents rated if each item was easy to understand (clarity) and sensitive to answer (interference with personal integrity). They could skip responding to a non-applicable/problematic item and give open comments. Predefined psychometric criteria were used; item clarity >= 80%/item sensitive to answer <= 20%/item feasibility <= 5% missing item responses. The decision to modify the translation was based on native expert, patient stakeholder, and instrument developer review, and the need for harmonization between translations.ResultsSimilar to findings in the Swedish-German cognitive debriefing, the cross-cultural analysis of input from 116 parents from 12 new countries (4-14 parents, median 9 parents/country) showed that all items in the EA-QOL questionnaire fulfilled the criteria for item clarity >= 80% and sensitive to answer (ranging from 1%-4.5%), although results varied between countries. Four items had missing responses between 5.2% and 13.4%, three within the same domain and were in line with parents' explanations. Poor translations and feasibility were improved.ConclusionsBased on parent input, the collaboration between native experts, patient stakeholders, and instrument developers, a linguistic version of the EA-QOL questionnaire for children aged 2-7 for use in and across 14 countries has been established. These efforts have set the conditions for a cross-cultural field test of the EA-QOL questionnaire and will open the doors for a new chapter in outcome research, registries, and clinical practice concerning children with EA. In the long-term, this will help increase knowledge of the disease's burden, promote patient-centeredness, exchange of information between nations, and strengthen evidence-based treatments for children born with EA.
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| 2. |
- Dellenmark-Blom, Maria, 1983, et al.
(författare)
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Factors of family impact in a Swedish-German cohort of children born with esophageal atresia
- 2022
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Ingår i: Orphanet Journal of Rare Diseases. - : Springer Science and Business Media LLC. - 1750-1172. ; 17:1
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Tidskriftsartikel (refereegranskat)abstract
- Background After repair of esophageal atresia (EA), childhood survivors commonly present with digestive and respiratory morbidity, and around 55% have associated anomalies. Although it is known that these problems can reduce health-related quality of life in children with EA, less is understood about the impact on the family. We aimed to identify factors related to family impact in children with EA. Methods One parent each of a child with EA (2-18 years) in 180 families from Sweden and Germany answered the PedsQL (TM) Family Impact Module as the dependent variable. The independent variables were the child's parent-reported health-related quality of life as measured by PedsQL (TM) 4.0, current symptoms, school situation, and parent/family characteristics together with child clinical data from the medical records. Results Stepwise multivariable regression analysis showed a multifactorial model of the total family impact scores (R-2 = 0.60), with independent factors being the child's overall generic health-related quality of life, school-absence >= 1/month, severe tracheomalacia, a family receiving carer's allowance, and a parent with no university/college education, p < 0.05. Logistic regression analysis showed that an increased number of symptoms in the child the preceding 4 weeks lowered the family impact scores; however, the child's feeding (R-2 = 0.35) and digestive symptoms (R-2 = 0.25) explained more in the variation of scores than the child's respiratory symptoms (R-2 = 0.09), p < 0.0001. Conclusions Family functioning may be a contributing factor to the maintenance of child health. The study findings suggest multifactorial explanations to family impact in children with EA, which are essential when optimizing the support to these families in clinical and psychosocial practice. Future research should explore experiences of family impact from all family members' perspectives and multicenter studies are warranted to understand better the effectiveness of psychosocial-educational interventions to families of children with EA.
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| 3. |
- Bloemeke, J., et al.
(författare)
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Health-Related Quality of Life Assessment in Children and their Families: Aspects of Importance to the Pediatric Surgeon
- 2020
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Ingår i: European Journal of Pediatric Surgery. - : Georg Thieme Verlag KG. - 0939-7248 .- 1439-359X. ; 30:03, s. 232-238
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Tidskriftsartikel (refereegranskat)abstract
- With the growing amount of chronic and rare diseases in childhood that often require lifelong treatment, improvement of health-related quality of life is a major goal in therapy. Therefore, the assessment of health-related quality of life from the patient's perspective as a relevant outcome parameter in clinical practice gained increased recognition. Health-related quality of life measures are still rarely applied in the pediatric practice context, although progress has been made in the development of instruments that are now ready for implementation. Inclusion of measures in research and practice is needed to accumulate and critically appraise knowledge about health-related quality of life to broaden the understanding of the child's health status, impacting on the whole family. From the perspective of a pediatric surgeon, knowledge about health-related quality of life of the children and their parents is important since it might influence treatment decisions and facilitates patient-physician communication. This article will review the concept of health-related quality of life, its methodological challenges and the application, and the challenges of health-related quality of life instruments in pediatric practice and health services research.
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| 4. |
- Dellenmark-Blom, Michaela, 1983, et al.
(författare)
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Clinical Factors Affecting Condition-Specific Quality-of-Life Domains in Pediatric Patients after Repair of Esophageal Atresia: The Swedish-German EA-QOL Study
- 2020
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Ingår i: European Journal of Pediatric Surgery. - : Georg Thieme Verlag KG. - 0939-7248 .- 1439-359X. ; 30:1, s. 96-103
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Tidskriftsartikel (refereegranskat)abstract
- Introduction We aimed to identify clinical factors affecting condition-specific health related quality of life (HRQOL) domains in children born with esophageal atresia (EA). This can facilitate preventive care to risk groups of HRQOL impairments. Materials and Methods A total of 124 Swedish and German families of EA children answered the validated EA-QOL questionnaires (response rate 68%), for evaluation of three HRQOL domains in children 2 to 7 years old (53 parents) and four HRQOL domains in children 8 to 17 years old (62 children/71 parents). Clinical data were collected through medical records and a questionnaire. Statistics included between-group analysis, univariable and stepwise multivariable regression analysis, p < 0.05. Results Between 2 to 7 years, no primary anastomosis ( p = 0.022) and female gender ( p = 0.026) predicted worse scores related to "physical health and treatment," and gastrostomy insertion related to "eating" ( p = 0.0001), and "social isolation and stress" ( p = 0.001). Between 8 to 17 years, no primary anastomosis (child report), prematurity, esophageal dilatation (parent report) predicted poor HRQOL related to "eating" ( p < 0.05), associated anomalies to "body perception" ( p = 0.031, parent report), female gender ( p = 0.018, child report) and severe EA ( p = 0.011 child report, p = 0.004 parent report) to "social relationships," and severe EA predicted worse "health and well-being" scores ( p = 0.004, parent report). An increased number of digestive symptoms (difficulty swallowing food, heartburn, and vomiting), lowered all EA-QOL domain scores in both age groups ( p < 0.001). An increased number of respiratory problems (cough, wheezing, airway infections. breathlessness, and chest tightness), lowered scores in two HRQOL domains among children 2 to 7 years ( p < 0.05). Conclusion Impairments within condition-specific HRQOL domains in EA children are found in congenital and surgical subgroups, and notably related to digestive symptoms throughout childhood.
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| 8. |
- Dellenmark-Blom, Michaela, 1983, et al.
(författare)
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The prevalence and role of coping strategies in the nutritional intake of children born with esophageal atresia: a condition-specific approach
- 2019
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Ingår i: Diseases of the esophagus : official journal of the International Society for Diseases of the Esophagus. - : Oxford University Press (OUP). - 1442-2050. ; 32:7
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Tidskriftsartikel (refereegranskat)abstract
- This study describes results of a condition-specific approach to the assessment of coping strategies in nutritional intake situations used by children with esophageal atresia. One hundred three families of children 2-17 years old with esophageal atresia participated (94% response rate). Following standardized focus groups with 30 families, nine coping items were developed, reflecting nine different coping strategies in nutritional intake situations. The coping items were pilot tested by 73 new families and evaluated for feasibility, validity, and reliability. The families also completed a validated condition-specific quality-of-life questionnaire for children with esophageal atresia, which included the scale Eating-Quality-of-life. Data were analyzed using descriptives, between-group analysis, and Spearman's rho (P<0.05). Altogether, the coping items were feasible, valid, and reliable. Items reflecting problem-focused strategies revealed that 89% of 2-17 years old 'recognized their responsibility' and managed nutritional intake problems on their own, 79% 'tried to solve their feeding problems' testing different solutions, 79% took a 'confronting approach' to do what peers did in eating situations, and 54% 'sought other people's support'. Items reflecting emotion-focused strategies showed that 86% of the children 'accepted' their feeding difficulties, 68% 'reappraised feeding difficulties into positive outcomes' such as to eat only when food tasted good. Moreover, 63% of the children 'avoided' nutritional intake situations, 29% 'expressed worry or fear' when faced with these situations, while 25% 'distanced' themselves from eating problems by hiding or throwing away food. The children's use of coping strategies were mostly related to the existence of digestive symptoms (P<0.05). Positive and negative coping strategies were identified. Of particular note was a correlation cluster of the so-called disengagement strategies 'avoidance', 'expression of emotional concerns' and 'distancing'. These strategies were negatively correlated with Eating-Quality-of-Life. Conversely, taking a 'confronting approach' correlated positively with Eating-Quality-of-life (P<0.05). Hence, most children with esophageal atresia employ various coping strategies in nutritional intake situations. A good Eating-Quality-of-life may be positively affected by treating digestive morbidity and encouraging children to take an active approach to their eating problems rather than using disengagement coping. © The Author(s) 2019. Published by Oxford University Press on behalf of International Society for Diseases of the Esophagus.
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| 9. |
- Soyer, T., et al.
(författare)
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Feasibility, Reliability, and Validity of the Turkish Version of the Esophageal-Atresia-Quality-of-Life Questionnaires to Assess Condition-Specific Quality of Life in Children and Adolescents Born with Esophageal Atresia
- 2021
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Ingår i: Turkish Journal of Gastroenterology. - : AVES Publishing Co.. ; 32:8, s. 640-650
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Tidskriftsartikel (refereegranskat)abstract
- Background: This study reports the feasibility, validity, and reliability of the Turkish versions of the Esophageal-Atresia-Quality-of-Life (EA-QOL) questionnaires, which were originally developed in Sweden and Germany. Methods: After translation from Swedish to Turkish and cognitive debriefings, 51 families of children aged 2-7 years (parent-reported, 17 items) and 54 families of children 8-17 years (child-reported and parent-reported, 24 items) responded to the EA-QOL questionnaires and a validated generic HRQOL instrument (PedsQL4.0). The medical records of the patients and the questionnaires were used to obtain clinical data. The Turkish version of the EA-QOL questionnaires were evaluated for feasibility (<5% missing item responses), reliability (internal consistency/retest reliability for 3 weeks), and validity (known groups/concurrent/convergent). The level of significance was P <.05. Results: The feasibility of the Turkish version of the EA-QOL questionnaires was good. The internal consistency of all scales was satisfactory, as were the levels of agreement of EA-QOL scores between the field study and the retest study. Known-group validity and concurrent validity were achieved, since the EA-QOL questionnaires showed that esophageal symptoms and feeding difficulties were negatively associated with EA-QOL total scores, both in the age-specific versions (child-reported and parent- reported), and with respect to respiratory symptoms in the version for EA children 2-7 years (parent-reported). A higher number of respiratory symptoms decreased the EA-QOL total scores in both age groups (parent-reported, P < .05). Correlations between the EA-QOL total scores and PedsQL-4.0 total scores supported convergent validity. Conclusion: The Turkish version of the EA-QOL questionnaires is feasible, valid, and reliable to assess condition-specific HRQOL in EA children.
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