Being the Pillar for Children with Rare Diseases-A Systematic Review on Parental Quality of Life.
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SUBMITTER: Boettcher J
PROVIDER: S-EPMC8125857 | biostudies-literature |
REPOSITORIES: biostudies-literature
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Project description:Objective(1) To identify instruments used to assess quality of life (QoL) in children and adolescents with neuromuscular diseases; (2) To identify the psychometric properties contained in these instruments.MethodsThis is a scoping review in which the electronic databases Embase, Scielo, Scopus, Pubmed and Lilacs were used as well as grey literature. The following terms were used in the search for articles published in the last 10 years: children, adolescents, neuromuscular disease, and quality of life.ResultsIn total, 15 articles were included and evaluated, indicating 7 instruments used to assess QoL (PedsQL™ Inventory 3.0 Neuromuscular Module, the PedsQL™ 4.0, the PedsQL DMD Module, the PedsQL ™ MFS, the SOLE, the KIDSCREEN and the LSI-A). The number of items ranged from 17 to 45. In addition, 6 instruments showed psychometric properties, but only 2 showed good and high quality, either in internal reliability or reproducibility.ConclusionOur results were able to map the main QoL assessment instruments of children and adolescents with neuromuscular disease and the most cited instrument was the PedsQL™ Inventory 3.0 Neuromuscular Module. Larger studies that assess psychometric properties and that are validated for most diseases are needed.
| S-EPMC10870459 | biostudies-literature
Project description:Previous research has examined several parental, child-related, and contextual factors associated with parental quality of life (QoL) among parents with a child or an adolescent with autism spectrum disorders (ASD); however, no systematic review has examined the relationship between parental QoL and parental involvement in intervention. To fill this gap, a systematic review was conducted using four electronic databases and checked reference lists of retrieved studies. Records were included in the systematic review if they presented original data, assessed parental QoL, and involvement in intervention for children or adolescents with ASD, were published in peer-reviewed journals between 2000 and 2020, and were written in English. Among the 96 screened full-texts, 17 articles met the eligibility criteria. The selected studies included over 2000 parents of children or adolescents with ASD. Three categories of parental involvement (i.e., none, indirect, direct) were identified, which varied across studies, although most had direct parental involvement. The results from this review show that increased parental involvement in the intervention for children or adolescents with ASD may be one way to promote their QoL. However, further research specifically focused on parental involvement during the intervention for children and adolescents with ASD is warranted.
| S-EPMC8469292 | biostudies-literature
Project description:Maintaining or improving quality of life (QoL) and well-being is a universal goal across the lifespan. Being physically active has been suggested as one way to enhance QoL and well-being. In this systematic review, conducted in part for the 2018 U.S. Health and Human Services Physical Activity Guidelines for Americans Scientific Advisory Committee Report, we examined the relationship between physical activity (PA) and QoL and well-being experienced by the general population across the lifespan and by persons with psychiatric and neurologic conditions. Systematic reviews, meta-analyses, and pooled analyses from 2006 to 2018 were used for the evidence base. Strong evidence (predominantly from randomized controlled trials [RCTs]) demonstrated that, for adults aged 18-65 years and older adults (primarily 65 years and older), PA improves QoL and well-being when compared with minimal or no-treatment controls. Moderate evidence indicated that PA improves QoL and well-being in individuals with schizophrenia and Parkinson's disease, and limited evidence indicated that PA improves QoL and well-being for youth and for adults with major clinical depression or bipolar disorder. Insufficient evidence existed for individuals with dementia because of a small number of studies with mixed results. Future high-quality research designs should include RCTs involving longer interventions testing different modes and intensities of PA in diverse populations of healthy people and individuals with cognitive (e.g., dementia) and mental health conditions (e.g., schizophrenia) to precisely characterize the effects of different forms of PA on aspects of QoL and well-being.
| S-EPMC7752999 | biostudies-literature
Project description:BackgroundResearch in a variety of countries indicates that healthcare access and health-related quality of life are challenged among people with a variety of rare diseases (RDs). However, there has been little systematic research on the experiences of children and adults with RDs in the American healthcare system that identifies commonalities across RDs. This research aimed to: (1) Describe demographics, disease characteristics, diagnostic experiences, access to healthcare, knowledge about RDs, support from healthcare professionals, and patient satisfaction among people with RDs and their caregivers; (2) examine predictors of patient satisfaction among adults with RDs; (3) compare health-related quality of life and stigma to US population norms; 4) examine predictors of anxiety and depression among adults and children with RDs.ResultsThis large-scale survey included (n = 1128) adults with RD or parents or caregivers of children with RDs representing 344 different RDs. About one third of participants waited four or more years for a diagnosis and misdiagnosis was common. A subset of participants reported experiencing insurance-related delays or denials for tests, treatments, specialists, or services. Approximately half of participants felt their medical and social support was sufficient, yet less than a third had sufficient dental and psychological support. Patients were generally neither satisfied or dissatisfied with their healthcare providers. Major predictors of satisfaction were lower stigma, lower anxiety, shorter diagnostic odyssey, greater physical function, and less pain interference. Adults and children with RDs had significantly poorer health-related quality of life and stigma in all domains compared to US norms. Predictors of both anxiety and depression were greater stigma/poor peer relationships, fatigue, sleep disturbance, limited ability to participate in social roles, and unstable disease course.ConclusionsPeople in the U.S. with RDs have poor health-related quality of life and high stigma. These factors are related to patient satisfaction and healthcare access, including diagnostic delays and misdiagnosis. Advocacy work is needed in order to improve healthcare access and ultimately health-related quality of life for children and adults with RDs.
| S-EPMC9096775 | biostudies-literature
Project description:BackgroundHeadaches are the most common complaints among pediatric populations. Determining the cause and appropriate treatment for headaches may be challenging and costly, and the impact of headaches on the lives of patients and their families is not well understood.ObjectiveA systematic literature review was conducted to examine what PROMs are currently used, and to identify quality of life (QoL) concepts important to children suffering from headaches and any known determinants of QoL.MethodsEmbase, Medline, Web of Science, CINAHL, EBSCOhost, PsychINFO, Cochrane CENTRAL and Google Scholar were searched from their inception through to June 2021. Studies investigating QoL, using a validated outcome measure in pediatric patients with headaches, were included. Relevant studies were identified through title and abstract screening and full text review by two independent reviewers. A citation review of included studies was performed. QoL concepts were extracted from the outcome measures that were used in each study to develop a preliminary conceptual model of QoL in children suffering from headaches. Determinants of QoL were also identified and categorized.ResultsA total of 5421 studies were identified in the search. Title and abstract screening resulted in the exclusion of 5006 studies. Among the 415 studies included for full text review, 56 were eligible for final analysis. A citation review resulted in the addition of five studies. Most studies were conducted in high-income countries and included a patient-sample accordingly (n = 45 studies). Sixteen different PROMs were identified in the included studies, of which the PedsQL was used the most often (n = 38 studies). The most common health concepts reported were physical functioning (n = 113 items), social and psychological wellbeing (N = 117, n = 91 resp.). Twenty-five unique determinants of QoL were extracted from the included studies.ConclusionThere is a need for a condition-specific PROM to facilitate the measurement of QoL outcomes in the pediatric headache population. A conceptual model was developed based on the findings from the health concepts. Findings from this review could be used for future qualitative interviews with pediatric patients with headaches to elicit and refine important QoL concepts.
| S-EPMC10506203 | biostudies-literature
Project description:ObjectiveIt is important to know the psychological variables that are related to quality of life and well-being in healthy elderly people. The main objective of the present review is to analyse which factors, through psychological variables, are determinant on the adaptive processes that acquire relevance in the last stage of life.Data sourcesAn electronic search was conducted in WOS, Science Direct, PsycARTICLES, Psychology Database and Psycinfo.Study selectionThe search terms used were derived from the combination of the following search string: (("Emotional Factors" OR "Emotional Effects") AND ("Emotional Intelligence" OR "Emotional Regulation") AND ("Quality of Life" OR "Personal Satisfaction") AND ("Healthy Old People" OR "Healthy Old Adults") AND ("Healthy Aging" OR "Successful Aging")).Data extraction11th April 2021.Data synthesisIn total, 13 articles were selected.ConclusionsThe articles showed the importance of social support, proactive coping strategies (emotional regulation) and emotional intelligence as key factors in the elderly population for their positive influence on variables such as quality of life and well-being.Prospero id crd42021224789
| S-EPMC8841035 | biostudies-literature
Project description:IntroductionCerebral palsy is the most common cause of physical disability in children and adolescents and is associated with impairments that may reduce the quality of life (QOL) of this population. Patient-reported outcome measures (PROMs) can facilitate the assessment of the effect of disease and treatment on QOL, from a patient viewpoint. The purpose of this systematic review is to identify PROMs that are used to measure QOL and subjective well-being (SWB) outcomes in young people with cerebral palsy and to evaluate the suitability of these PROMs for application in economic evaluations within this population.Methods and analysisMEDLINE, Scopus, the Cochrane Library, Web of Science Core Collection, EconLit, PsycINFO, CINAHL, EMBASE and Informit will be systematically searched from inception to date of search. Published peer-reviewed, English-language articles reporting PROMs measuring QOL or SWB outcomes in children and adolescents with cerebral palsy will be included. One reviewer will conduct the initial search and screen titles and abstracts for potentially eligible studies. The search will be performed in November 2017. To reduce the likelihood of reviewer selection bias, two other reviewers will independently screen a randomly selected subsample (10%) of the citations. Two reviewers will then retrieve full texts of potentially eligible studies and assess them against predefined inclusion criteria. The suitability of selected PROMs for use in economic evaluations of young people with cerebral palsy will be assessed using the International Society of Quality of Life Research recommended Minimum Standards and the Patient-Centered Outcomes and Comparative Effectiveness Research checklist. A narrative synthesis of extracted data will be presented including study descriptive data, PROMs measurement properties, settings in which they were applied and the valuation methods. Recommendations for practice on the selection of PROMs for use in economic evaluations of children and adolescents with cerebral palsy will be presented.Ethics and disseminationEthical approval is not required as the proposed systematic review will not use primary data. The results of this study will be widely disseminated through publication in a peer-reviewed journal and conference presentation(s).Systematic review registration numberInternational Prospective Register of Systematic Reviews number: CRD42016049746.
| S-EPMC5640099 | biostudies-literature
Project description:Amblyopia is a common condition, which can affect up to 5% of the general population. Health-related quality-of-life (HRQoL) implications of amblyopia and/or its treatment have been explored in the literature. A systematic literature search was undertaken during the period of 7-14 May 2010 to identify the HRQoL implications of amblyopia and/or its treatment. A total of 35 papers were included in the literature review. The HRQoL implications of amblyopia related specifically to amblyopia treatment, rather than to the condition itself. These included impact on family life, social interactions, difficulties in undertaking daily activities, as well as feelings and behaviour. The identified studies adopted a number of methodologies. The study populations included children with the condition, parents of children with amblyopia, and adults who had undertaken amblyopia treatment as a child. Some studies developed their own measures of HRQoL, and others determined HRQoL through proxy measures. The reported findings of the HRQoL implications are of importance when considering the management of cases of amblyopia. The issues identified in the literature review are discussed with respect to how HRQoL is measured (treatment compliance vs proxy measures), and whether HRQoL is taken from a child's or a parent's perspective. Changing societal views over glasses and occlusion therapy are also discussed. Further research is required to assess the immediate and long-term effects of amblyopia and/or its treatment on HRQoL using a more standardised approach.
| S-EPMC3078103 | biostudies-other
Project description:BackgroundMeasurement of health utilities is required for economic evaluations. Few studies have evaluated health utilities for rare conditions; even fewer have incorporated disutility that may be experienced by caregivers. This study aimed to (1) estimate health utilities for three rare conditions currently recommended for newborn screening at the state or federal level, and (2) estimate the disutility, or spillover, experienced by parents of patients diagnosed with a rare, heritable disorder.MethodsA stated-preference survey using a time trade-off approach elicited health utilities for Krabbe disease, phenylketonuria, and Pompe disease at varying stages (mild, moderate, severe) and onset of disease symptoms (infancy, childhood, and adulthood). We recruited respondents from a nationally representative community sample (n = 862). Respondents valued disease specific health states in three consecutive question frames: (1) adult health state (> = 18 years of age), (2) child health state (< 18 years of age), and (3) as a parent of a child with a condition (parent spillover state). Corresponding mean utilities were calculated for plausible disease states in adulthood and childhood. Mean disutility was estimated for parental spillover. Predictors of utilities were evaluated using a negative binomial regression model.ResultsMore severe conditions and infant health states received lower estimated utility and greater estimated disutility among parents. Conditions with the lowest estimated health utilities were severe infantile Pompe disease (0.40, CI: 0.34-0.46) and infantile Krabbe disease (0.37, CI: 0.32-0.43). Disutility was evident for all conditions evaluated (range: 0.07-0.19).ConclusionsRare childhood conditions are associated with substantial estimated losses in quality of life. Evidence of disutility among parents further warrants the inclusion of spillover effects in cost-effectiveness analyses. Continued research is needed to assess and measure the effects of childhood disease from a family perspective.
| S-EPMC6342747 | biostudies-literature
Project description:OBJECTIVES:The aim of this systematic review was to assess the oral-health-related quality of life (OHRQoL) of adult patients with rheumatic diseases. MATERIAL AND METHODS:A systematic literature search was performed, including clinical studies on adults (aged at least 18 years) with a verified diagnosis of rheumatic disease. RESULTS:26 out of 41 clinical studies including rheumatoid arthritis (RA, seven studies), systemic sclerosis (SSc, five), Sjögren syndrome (SS, eight), Behcet disease (BD, four), systemic lupus erythematosus (SLE, one) and ankylosing spondylitis (AS, one) were found. In 15 studies, a healthy control group was recruited. The short form of the Oral Health Impact Profile (OHIP 14) was most frequently applied. The majority of studies (14/15) reported worse OHRQoL in patients with rheumatic disease compared to healthy individuals. In particular, patients with SS (salivary flow and composition) or BD (oral ulcers) showed a relation between OHRQoL and disease-specific oral manifestations. Most studies investigating subscales of OHRQoL (5/6) found the subscale physical disability to be predominantly affected in patients with rheumatic diseases. About half of the studies reported impaired psychosocial aspects. CONCLUSION:Patients with rheumatic diseases exhibit reduced OHRQoL, especially in diseases with oral manifestations like SS and BD. Physical affections due to oral diseases and psychosocial impairments caused by disease-related parameters must be recognized within patient-centered dental care.
| S-EPMC7231140 | biostudies-literature