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Lived Experiences of Fragile X Syndrome Caregivers: A Scoping Review of Qualitative Studies.

ABSTRACT: Fragile X Syndrome (FXS) is the most common x-linked monogenic cause of Intellectual Disability (ID) and Autism Spectrum Disorder (ASD). Taking care of children with ID is challenging and overwhelming due to the multiple facets of caregiving. This scoping review aimed at summarizing the qualitative literature on the experiences of families living with FXS, identify key themes and determine the gaps in the extant literature. We conducted a literature search in May 2019 using four databases; PubMed, Web of Science, African-Wide-Information, and Scopus. The keywords used in our search strategy were associated with caregivers, lived experiences, FXS, and qualitative research. All English language articles with full-text reporting were included. Studies associated with other neurodevelopmental conditions and quantitative studies were excluded. We identified 12 out of 203 articles that described the lived experiences of families with FXS. Most articles originated from the United States of America and mothers were the main caregivers. We summarized our findings into four major themes which are; grief experiences, challenges of living with FXS, coping mechanisms and the need to plan for future outcomes. This scoping review highlights the scarcity of qualitative FXS literature in the African population and frustrations endured by families with FXS due to the low knowledge of FXS by healthcare workers. More research is needed to evaluate the impact of living with FXS in males and fathers.

SUBMITTER: Kamga KK

PROVIDER: S-EPMC7056838 | biostudies-literature | 2020

REPOSITORIES: biostudies-literature

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Lived Experiences of Fragile X Syndrome Caregivers: A Scoping Review of Qualitative Studies.

Kamga Karen Kengne KK De Vries Jantina J Nguefack Seraphin S Munung Syntia Nchangwi SN Wonkam Ambroise A

Frontiers in neurology 20200227

Fragile X Syndrome (FXS) is the most common x-linked monogenic cause of Intellectual Disability (ID) and Autism Spectrum Disorder (ASD). Taking care of children with ID is challenging and overwhelming due to the multiple facets of caregiving. This scoping review aimed at summarizing the qualitative literature on the experiences of families living with FXS, identify key themes and determine the gaps in the extant literature. We conducted a literature search in May 2019 using four databases; PubMe ...[more]

PMID: 32174884

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Project description:Mothers, caregivers, and healthcare providers in 163 countries have used paper and electronic home-based records (HBRs) to facilitate primary care visit. These standardized records have the potential to empower women, improve the quality of care for mothers and children and reduce health inequities. This review examines experiences of women, caregivers and providers with home-based records for maternal and child health and seeks to explore the feasibility, acceptability, affordability and equity of these interventions. We systematically searched MEDLINE, MEDLINE In-Process, MEDLINE Ahead of Print, Embase, CINAHL, ERIC, and PsycINFO for articles that were published between January 1992 and December 2017. We used the CASP checklist to assess study quality, a framework analysis to support synthesis, and GRADE-CERQual to assess the confidence in the key findings. Of 7,904 citations, 19 studies met our inclusion criteria. In these studies, mothers, caregivers and children shared HBR experiences in relation to maternal and child health which facilitated the monitoring of immunisations and child growth and development. Participants' reports of HBRs acting as a point of commonality between patient and provider offer an explanation for their perceptions of improved communication and patient-centered care, and enhanced engagement and empowerment during pregnancy and childcare. Healthcare providers and nurses reported that the home-based record increased their feeling of connection with their patients. Although there were concerns around electronic records and confidentiality, there were no specific concerns reported for paper records. Mothers and other caregivers see home based records as having a pivotal role in facilitating primary care visits and enhancing healthcare for their families. The records' potential could be limited by users concerns over confidentiality of electronic home-based records, or shortcomings in their design. Health systems should seize the opportunity HBRs provide in empowering women, especially in the contexts of lower literacy levels and weak health care delivery systems.

Project description:OBJECTIVES:To provide a comprehensive synthesis of informal caregivers' experiences of caring for a significant other following discharge from cardiac surgery. DESIGN:Systematic integrated review without meta-analysis. DATA SOURCES:A bibliographic search for publications indexed in six databases (Cochrane Library, CINAHL, MEDLINE, EMBASE, AMED and PsycINFO), including a scan of grey literature sources (GreyNet International, Google Scholar, Web of Science, WorldCat and the Clinical Trials Registry) was conducted in October 2018. ELIGIBILITY CRITERIA FOR SELECTING STUDIES:Studies were included if they described views and perspectives of informal caregivers of cardiac surgery patients (non-intervention studies (qualitative and quantitative)), and the effectiveness of interventions to evaluate support programme for informal caregivers of cardiac surgery patients (intervention studies). RESULTS:Of the 4912 articles identified in searches, 42 primary research studies were included in a narrative synthesis with 5292 participants, including 3231 (62%) caregivers of whom 2557 (79%) were women. The median sample size across studies was 96 (range 6-734). Three major themes emerged from the qualitative study data: (1) caregiver information needs; (2) caregiver work challenges and (3) caregivers adaption to recovery. Across the observational studies (n=22), similar themes were found. The trend across seven intervention studies focused on caregiver information needs related to patient disease management and symptom monitoring, and support for caregivers to reduce symptoms of emotional distress. CONCLUSION:Informal caregivers want to assist in the care of their significant others after hospital discharge postcardiac surgery. However, caregivers feel insecure and overwhelmed and they lack clear/concise discharge information and follow-up support during the early at-home recovery period. The burden of caregiving has been recognised and reported since the early 1990s, but there remains a limited number of studies that assesses the effectiveness of caregiver interventions. PROSPERO REGISTRATION NUMBER:CRD42018096590.

Project description:BackgroundPrevious studies have not synthesized existing literature on the lived experiences of pregnant and parenting adolescents (aged 10-19) in Africa. Such evidence synthesis is needed to inform policies, programs, and future research to improve the well-being of the millions of pregnant or parenting adolescents in the region. Our study fills this gap by reviewing the literature on pregnant and parenting adolescents in Africa. We mapped existing research in terms of their substantive focus, and geographical distribution. We synthesized these studies based on thematic focus and identified gaps for future research.MethodsWe used a three-step search strategy to find articles, theses, and technical reports reporting primary research published in English between January 2000 and June 2021 in PubMed, Jstor, AJOL, EBSCO Host, and Google Scholar. Three researchers screened all articles, including titles, abstracts, and full text, for eligibility. Relevant data were extracted using a template designed for the study. Overall, 116 studies met the inclusion criteria and were included in the study. Data were analyzed using descriptive and thematic analyses.ResultsResearch on pregnant and parenting adolescents is limited in volume and skewed to a few countries, with two-fifths of papers focusing on South Africa (41.4%). Most of the studies were African-led (81.9%), received no funding (60.3%), adopted qualitative designs (58.6%), and were published between 2016 and 2021 (48.3%). The studies highlighted how pregnancy initiates a cycle of social exclusion of girls with grave implications for their physical and mental health and social and economic well-being. Only 4.3% of the studies described an intervention. None of these studies employed a robust research design (e.g., randomized controlled trial) to assess the intervention's effectiveness. Adolescent mothers' experiences (26.7%) and their education (36.2%) were the most studied topics, while repeat pregnancy received the least research attention.ConclusionResearch on issues affecting pregnant and parenting adolescents is still limited in scope and skewed geographically despite the large burden of adolescent childbearing in many African countries. While studies have documented how early pregnancy could result in girls' social and educational exclusion, few interventions to support pregnant and parenting adolescents exist. Further research to address these gaps is warranted.

Project description:BackgroundConsequences of distal renal tubular acidosis (dRTA) on growth, bone and kidney, sometimes associated with hearing loss, may significantly affect quality of life (QoL). This descriptive qualitative study explores QoL linked to dRTA and gathers the impressions of patients with this rare disease (and caregivers) 5 years after enrolment in a clinical study, during which patients were treated with ADV7103, a prolonged-release granule formulation combining potassium citrate and potassium bicarbonate. Semi-structured, one-hour interviews with 6 adult and 13 paediatric patients with a confirmed diagnosis of dRTA and with parents of paediatric patients were performed using an interview guide. Qualitative analysis of anonymized interview transcripts based on grounded theory was conducted.ResultsThe main QoL domains impacted by dRTA and its treatment were education/work, social/family life, and emotional and physical well-being. ADV7103 (administered twice daily) was compared with the standard of care (SoC) taken before study entry (more than twice daily). Patients/parents reported that switching from previous SoC to ADV7103 had changed their lives: Difficulties at school due to burdensome administrative issues and need to explain disease and treatment affecting all families of paediatric patients (n = 13) disappeared, facilitating parents who had stopped working (to deal with their child's treatment) to return to work, Family functioning was improved (n = 18), as travel and holidays became easier to organise and patients/parents stopped thinking about managing treatment daily/nightly, reducing tension in the family or couple, The emotional burden of disease perceived was relieved (n = 12) in the absence of treatment-related invasive questions from others, Gastro-intestinal adverse events and taste problems improved with ADV7103 (n = 18) and better compliance led to milder physical impacts and less need to be hospitalised. The mean satisfaction score with ADV7103 compared to SoC was 9 out of 10 (10 = very satisfied). ADV7103 exceeded or met the expectations of 14 out of 17 patients that commented on that.ConclusionsQualitative interviews show that dRTA and its treatment have a significant impact on QoL of patients and parents and that ADV7103 helps improve daily-life and reduces treatment burden, resulting in greater overall satisfaction of the patients and their families. Trial registration EU Clinical Trials Register, EudraCT 2013-003828-36 on the 3rd of September 2013.

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Lived Experiences of Fragile X Syndrome Caregivers: A Scoping Review of Qualitative Studies.

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Lived Experiences of Fragile X Syndrome Caregivers: A Scoping Review of Qualitative Studies.

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