Health-related quality-of-life among patients with premature ovarian insufficiency: a systematic review and meta-analysis.

PURPOSE: To systematically review studies investigating health-related quality-of-life (HrQoL) in patients with premature ovarian insufficiency (POI), to examine questionnaires used and to conduct a meta-analysis of control studies with normal ovarian function. METHODS: Data sources: PubMed, Embase, Web of science, CNKI, and CQVIP, searched from inception until June 2018. The search strategy was a combination of medical (e.g. POI), subjective (e.g. well-being) and methodological (e.g. questionnaires) keywords. PRISMA guidelines were used to assess outcome data quality/validity by one reviewer, verified by a second reviewer. Risk of bias within studies was evaluated. A meta-analysis compared HrQoL in patients and non-patients. Due to measurement differences in the studies, the effect size was calculated as standard mean difference. RESULTS: We identified 6869 HrQoL studies. Nineteen geographically diverse studies met inclusion criteria, dated from 2006, using 23 questionnaires. The meta-analysis included six studies with 645 POI participants (age 33.3 ± 5.47) and 492 normal-ovarian control subjects (age 32.87 ± 5.61). Medium effect sizes were found for lower overall HrQoL (pooled SMD = - 0.73, 95% CI - 0.94, - 0.51; I2 = 54%) and physical function (pooled SMD = - 0.54, 95% CI - 0.69, - 0.39; I2 = 55%). Heterogeneity was investigated. Effect sizes varied for sexual function depending on the measure (SMD = - 0.27 to - 0.74), overall HrQoL (SF-36) had the largest effect size (- 0.93) in one study. The effect sizes for psychological and social HrQoL were small. CONCLUSION: POI is associated with low-to-medium effect size on HrQoL compared to normal ovarian controls. The greatest effects are found in general HrQoL and most sexual function areas. Condition-specific questionnaires and RCTs are recommended for further investigation.

The prevalence and role of coping strategies in the nutritional intake of children born with esophageal atresia: a condition-specific approach.

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.

Development and pilot-testing of a condition-specific instrument to assess the quality-of-life in children and adolescents born with esophageal atresia.

The survival rate of children with esophageal atresia has today reached 95%. However, children are at risk of chronic morbidity related to esophageal and respiratory dysfunction, and associated anomalies. This study describes the pilot testing of a condition-specific health-related quality-of-life instrument for children with esophageal atresia in Sweden and Germany, using a patient-derived development approach consistent with international guidelines. Following a literature review, standardized focus groups were conducted with 30 Swedish families of children with esophageal atresia aged 2-17 years. The results were used for item generation of two age-specific pilot questionnaire versions. These were then translated from Swedish into German with considerations of linguistic and semantical perspectives. The 30-item pilot questionnaire for children aged 2-7 years was completed by 34 families (parent report), and the 50-item pilot questionnaire for children aged 8-17 years was completed by 52 families (51 child report, 52 parent report), with an overall response rate of 96% in the total sample. Based on predefined psychometric criteria, poorly performing items were removed, resulting in an 18-item version with three domains (Eating, Physical health and treatment, Social isolation and stress,) for children aged 2-7 years and a 26-item version with four domains (Eating, Social relationships, Body perception, and Health and well-being) for children aged 8-17 years. Both versions demonstrated good internal consistency reliability and acceptable convergent and known-groups validity for the total scores. The study identified specific health-related quality-of-life domains for pediatric patients with esophageal atresia, highlighting issues that are important for follow-up care. After field testing in a larger patient sample, this instrument can be used to enhance the evaluation of pediatric surgical care.

Coping strategies used by children and adolescents born with esophageal atresia - a focus group study obtaining the child and parent perspective.

BACKGROUND: Esophageal atresia (EA) is a rare malformation, which requires surgical treatment. Survival rates today reach 95%, but EA remains a significant cause of chronic morbidity with increased risk of psychosocial problems and impaired health-related quality of life (HRQOL). No study of coping strategies of children with EA has been reported in the literature to date, but increased knowledge could lead to improved outcomes and better HRQOL. METHODS: Standardized focus groups with children with EA and their parents were conducted to identify issues related to health care needs and HRQOL, with group members relating their coping experiences. Identified coping statements were content analysed using a card sorting procedure and descriptive statistics. RESULTS: Thirty families (18 children 8-17 years; 32 parents of children with EA 2-17 years) participated in 10 focus groups. A total of 590 coping statements were recorded. Nine coping strategies were identified: problem solving (n = 116), avoidance (n = 95), recognizing responsibility (n = 71), confronting (n = 70), seeking social support (n = 63), positive reappraisal (n = 58), emotional expression (n = 46), acceptance (n = 40) and distancing (n = 31). Nine situational contexts were identified: nutritional intake (n = 227), communication of one's health condition (n = 78), self-perception when experiencing troublesome symptoms (n = 59), appearance of body or scar(s) (n = 57), physical activities like sport and play (n = 43), sleep (n = 34), hospital care (n = 33), stigmatization and social exclusion (n = 30) and medication intake (n = 29). CONCLUSIONS: Focus group methodology contributed to an increased understanding of disease-specific coping processes among children and adolescence with EA. Findings illustrate that they use several coping strategies, some of which they seem to adopt at early age and use in disease-related contexts of physical, social and emotional character. Such coping may influence health and HRQOL in children with EA. In view of the importance of establishing good coping strategies early in life, health care professionals should integrate coping aspects into care management. Future studies are warranted.

Epidemiology and patient-reported measurement outcome of pelvic fractures in children and adolescents - A population-based cohort study from the Swedish fracture register.

BACKGROUND AND PURPOSE: Pediatric pelvic fractures are uncommon, representing 0.2-3% of total pediatric fractures. The long-term patient-reported outcome in the pediatric population has not been evaluated yet. The purpose of the study was to describe the epidemiology of pelvic and acetabular fractures in pediatric patients including long-term patient-reported outcomes. PATIENTS AND METHODS: The Swedish Fracture Register (SFR) was used to identify all patients aged 6-17 years at injury with a pelvic fracture between 2015 and 2021. All patients were invited to answer Patient-Reported measurement instruments in 2021. RESULTS: The study cohort consisted of 223 patients with a median age at fracture of 15 years and with 62 % boys. 201 sustained a pelvic and 22 acetabular fractures. Falls were the leading cause of fracture, followed by transport accidents. Most fractures (both pelvis and acetabulum) were type A (73 %), and 21 fractures (9 %) could not be classified according to AO. 85 % of fractures were treated non-surgically. All Type C fractures were treated surgically. Seven PROMIS® profile domains were completed by 31 % of the sample at a mean follow-up time of 3.5 years after pelvic/acetabular fracture. Most patients had "no concern" or "mild concern" but those who had surgery had an inferior t-score in most domains. CONCLUSION: Most fractures occurred in older individuals, with falls during sports activities being the most common cause. This raises important questions about prevention strategies. The PROMIS-Pain-Interference scale indicated that the younger the age at fracture, the more pain was reported at follow-up.

The reliability of the disabled children's quality-of-life questionnaire in Swedish children with diabetes.

AIM: To determine the reliability of the disabled children's quality-of-life measure (DISABKIDS) chronic generic questionnaire and diabetes module in children. The questionnaire is being evaluated for repeated routine health-related quality-of-life (HrQoL) assessment and in association with the Swedish national paediatric diabetes registry (Swediabkids), which is a tool for regular clinical use. METHODS: Children and parents completed the questionnaire during a routine visit to the diabetes clinic. In total, 120 families completed the test and retest. Split-half reliability correlation and intraclass correlation (ICC) coefficients were calculated. Bland & Altman plots were calculated on the generic HRQoL domain. RESULTS: Both child and parent versions showed good internal consistency. Test-retest ICC coefficients for the generic HrQoL module were 0.913 for the children and 0.820 for the parent version. All generic domains independently showed good reliability. The diabetes module had a score of 0.855 for children and 0.823 for parents. Split-half correlation for generic and diabetes modules was 0.930 and 0.848 for children, 0.953 and 0.903 for parents. Bland and Altman plots showed substantial agreement between the two administrations for both children and parents. CONCLUSION: The DISABKIDS questionnaire is a reliable instrument for the repeated measurements of HrQoL in children with diabetes.

Assessment of childhood diabetes-related quality-of-life in West Sweden.

AIM: To investigate health-related quality-of-life (HrQoL) in childhood diabetes and the level of agreement between West Sweden and European reference data for the new multi-cultural European questionnaire - DISABKIDS. METHOD: Twenty percent of the Swedish paediatric diabetes population was included in the survey. Child-parent pairs completed the DISABKIDS chronic generic (37 questions) and diabetes modules (10 questions) during their routine clinic visit. A one-page results summary, based on positive domains, was used to provide feedback to clinicians. RESULTS: Three hundred and sixty-one child-parent pairs were included in the analysis. In Sweden, diabetes was perceived by the children as having less impact than the European average. Swedish parents rated the HrQoL of their children lower than did the European parents. Swedish girls had a lower HrQoL than boys and greater difficulty accepting their diabetes; adolescents had greater difficulty accepting the diagnosis than younger children. Parents reported greater impact of diabetes on their children than the children themselves but reported no difference between boys and girls. Parents reported better acceptance of treatment in boys. The child's reported quality-of-life (QoL) is related to age and gender. CONCLUSION: Our results confirm the applicability of DISABKIDS to the Swedish paediatric diabetes population.

Improved treatment satisfaction but no difference in metabolic control when using continuous subcutaneous insulin infusion vs. multiple daily injections in children at onset of type 1 diabetes mellitus.

OBJECTIVE: The aim of this study was to compare safety, metabolic control, and treatment satisfaction in children/adolescents at onset of type 1 diabetes mellitus who were treated with either continuous subcutaneous insulin infusion (CSII) or multiple daily injections (MDI). RESEARCH DESIGN AND METHODS: Seventy-two children/adolescents (7-17 yr of age) were enrolled in this open, randomized, parallel, multicenter study. Approximately half of the patients were treated with MDI (natural protamine hagedorn [NPH] insulin twice daily and rapid-acting insulin three to -four times daily, n = 38) by pen, and the other half received CSII (n = 34). The patients were followed for 24 months with clinical visits at the entry of the study and after 1, 6, 12, and 24 months. During these visits, hemoglobin A1c, insulin doses, weight, and height were registered. Severe episodes of hypoglycemia and ketoacidosis as well as technical problems were recorded. In addition, the patients/parents answered the Diabetes Treatment Satisfaction Questionnaire. RESULTS: There was no significant difference in metabolic control between the treatment groups. Treatment satisfaction was significantly higher in the group treated with CSII compared with the MDI group (p

Factors associated with the employment problems of people with established epilepsy.

The employment experiences of 245 respondents with epilepsy as their main diagnosis were examined as part of a study into the rehabilitation needs of an epilepsy outpatient clinic. It was found that 9% of the sample was unemployed and a further 16% were in receipt of a disability pension. Patients with seizures in remission were more likely to be employed and less likely to have experienced job problems, to feel limited by the epilepsy or to experience stigma. Job problems per se were experienced by 35% of the population. Of those with uncontrolled seizures, 50% had had job problems; however, 22% thought that their current employment situation had not been unduly influenced by epilepsy. It was those respondents who were younger or who were diagnosed early with epilepsy who were most likely to perceive their current situation as a result of having epilepsy. The survey suggests that unemployment is not the major problem it was once thought to be but that discrimination at work is a more serious problem which could lead to under-employment and restricted career development. Career planning should be instigated early and employment services should include information, practical advice and emotional support.

The perceived rehabilitation needs of a hospital-based outpatient sample of people with epilepsy.

A postal survey was carried out to identify the perceived epilepsy rehabilitation needs of a hospital-based outpatient population. A response rate of 70% resulted in 245 patients being surveyed. Data showed that 65% of the total sample wanted more rehabilitation assistance and that 27% required substantial contact with the rehabilitation services. The most common request was for more medical information both via written material and telephone contact with a specially trained epilepsy nurse. Approximately one in six patients would like to attend a course on how to live with epilepsy. Access to a psychologist was most often requested during the first year following diagnosis and demand for courses on how to live with epilepsy was highest in the second to fourth year following diagnosis. Requests for social worker assistance were associated with employment issues. Frequency of seizures, duration of epilepsy and age were significant variables in relation to demands for rehabilitation resources. The general conclusions are that (1) the minimum standards of a rehabilitation service should include greater access to medical information via a variety of authoritative sources; and (2) that team-based resources are wanted by a substantial proportion of the population in relation to specific and definable problems, which would involve intensive input from psychologists and social workers.

Team-building in vocational rehabilitation: the Horizon Telecentre project.

This report concerns an ambitious project which created a functionally independent team of young people with physical disabilities who had no previous experience of employment. The aim of the project was to expand the training opportunities for people with disabilities through the provision of team-building. The team was brought together in order to run a self-financing office bureau business in Athens, providing services to the local community. This project intended to find an innovative alternative to sheltered workshops, which are not found in Greece. A systemic and developmental approach to team-building was explored. The approach consisted of work-hardening, education, experiential learning, and providing the physical structures necessary for self-employment. The paper describes the approach with examples, and includes an evaluation by the trainees. The paper also identifies the importance of including a team-building approach within vocational rehabilitation.

National general practice study of epilepsy: the social and psychological effects of a recent diagnosis of epilepsy.

OBJECTIVES: To determine the nature and extent of psychosocial problems in epilepsy and their associations. DESIGN: A postal survey was used drawing data from prospective consecutive cases. Data on demographic, medical, and social backgrounds were collected. A specially designed, validated attitude questionnaire examined 14 areas of psychosocial adjustment to epilepsy. SETTING: 124 primary care general practices. SUBJECTS: Adults (aged over 17) with a recent diagnosis of epilepsy (within previous 36 months). They were registered with the national general practice study of epilepsy and had a confirmed diagnosis according to the usual criteria. 216 subjects were approached for the survey by their general practitioners; 192 returned questionnaires. RESULTS: Problems in at least one area were experienced by 175 (91%) of the 192 subjects. Problems were generally mild, which contrasts strongly with findings in chronic cases. The areas of greatest concern were fear of seizures (80% of cases; 72% moderate or severe) and fear of stigma in employment (69% of cases; 40% moderate or severe). A highly significant relation was found between psychosocial effects and the frequency and recency of seizures. CONCLUSIONS: In the early stages of epilepsy psychosocial effects are closely related to the severity of the medical condition, suggesting that the argument for the stigmatising effect of the diagnosis by itself is less important than previously thought. The findings also suggest that problems may evolve as the condition becomes chronic.

Cross-cultural adaptation and use of the epilepsy psychosocial effects scale: comparison between the psychosocial effects of chronic epilepsy in Sweden and the United Kingdom.

PURPOSE: To establish Swedish weightings for the Epilepsy Psycho-Social Effects Scale (EPSES) and examine differences between Sweden and the U.K. in attitudes toward chronic epilepsy. METHODS: After translation and back-translation of the EPSES into Swedish, weightings were established through a paired comparison study in which 24 epilepsy professionals participated. The EPSES was then given to a hospital-based sample of 57 patients with chronic epilepsy and the results compared with a British sample matched for age, sex, and frequency of seizures. RESULTS: In the professional judgments of the EPSES statements, there were statistically significant differences in only four of 42 statements. Attitudes to employment, to medication, and to fear of seizures were considered more serious in the U.K., whereas social isolation was considered more a problem in Sweden. Patient attitudes to chronic epilepsy were found to be largely similar in the U.K. and Sweden; however, attitudes to employment, to the future, and to fear of seizures were significantly more a problem for patients in the U.K. CONCLUSIONS: The results indicate a great similarity in the interpretation of problems as measured by the EPSES in the U.K. and in Sweden. The differences in attitudes found are discussed in relation to economic and legislative differences between the two countries. Social as well as cultural differences should be taken into account when using standardized questionnaires. It is suggested that culturally specific weightings should be calculated and that these should be reevaluated at regular intervals.

A quantitative approach to measuring the social effects of epilepsy.

This paper describes the construction of a postal questionnaire which has been used to investigate the social affects of epilepsy on a wide range of patients. The social profile was designed following extensive patient interviews at the National Hospital for Nervous Diseases and the Chalfont Centre for Epilepsy. The initial research revealed 21 areas of concern for the individual with epilepsy, which are described. Reliability was assessed on a test/retest procedure. Validity was tested via a criterion-related study based on the behaviour of the subject. Following reliability and validity studies 14 areas were eventually selected for the final questionnaire. Instruments have been adapted for both children and adults.