Family and Caregivers

Abstract

People of African descent and those identifying as Black and/or Latino experience a disproportionate burden of sickle cell disease (SCD), a chronic, serious blood condition. Caregivers of children with chronic medical conditions report worse mental health than others. Disease associated stressors can affect caregivers of children with SCD. We conducted a systematic review to summarize the prevalence of mental health symptoms in caregivers of children with SCD and if symptoms were associated with the child’s SCD. This review is reported according to the Preferred Reporting Items for Systematic Reviews and Meta-Analysis (PRISMA) guidelines. We searched PubMed, PsycINFO and Embase identifying 1,322 records of which 40 met criteria for inclusion in this review. Findings suggest caregivers experience mental health problems and poorer mental health was associated with worse child SCD-related outcomes and treatment adherence. Efforts should be made to routinely screen SCD caregiver mental health and to refer accordingly.

Keywords: Anxiety, caregivers, depression, mental health, parents, sickle cell disease, stress, systematic review

Sickle cell disease (SCD) is a chronic, inherited blood condition characterized by physical symptoms of fatigue, severe pain episodes, and susceptibility to infection starting in early childhood with consequent risk of hospitalization and reduced lifespan. 1 People of African descent have the highest frequency of SCD and sickle cell trait among racial/ethnic groups. In the United States (U.S.) and elsewhere, most people with SCD self-identify as Black and/or Latino. 2,3 Those communities are disproportionately subject to under-resourced conditions4 and higher social vulnerability. 5–8 Individuals with SCD and their families may be subject to stressors related to the condition and/or socioeconomic challenges, including health-related stigma and discriminatory practices9,10 in social or medical contexts.

For children and adolescents living with SCD, chronic and acute health threats and adverse social factors increase their risks of adverse mental health, especially depression and anxiety. 11 Adolescents with chronic health conditions, including SCD, commonly report depressive symptoms and anxiety. 12–19Being the parent of a child with a serious chronic medical condition can be taxing due to challenges from logistical, financial, and psychological stressors. 20,21 A recent systematic review and meta-analysis examining behavioral and mental health outcomes of parents of children with a variety of chronic medical conditions reported worse parental mental health and more frequent and severe symptoms of anxiety and depression compared with parents of healthy children. 22 In that review, only one report assessed mothers of children with SCD and it focused exclusively on depressive mood. 23

To our knowledge, no systematic review has focused on mental health symptoms among caregivers of children with SCD. Here we asked about mental health symptoms of caregivers of children with SCD. Specifically we set out to synthesize the literature regarding: (1) the symptoms caregivers report and how they were assessed; (2) the prevalence of the symptoms among caregivers of children with SCD was; (3) how those symptoms compared with those of caregivers of healthy children or children with a different chronic condition; (4) the associations of caregiver mental health symptoms with severity of clinical symptoms of the children with SCD in their care; and (5) the gaps exist in the literature.

Methods

The reporting of this systematic review adheres to the Preferred Reporting Items for Systematic Reviews and Meta-Analysis (PRISMA) guidelines. 24 A review protocol was developed in advance and registered in the PROSPERO (CRD42020178326), to which the study team adhered.

Search strategy and selection criteria.

A comprehensive literature search was conducted on April 1, 2020 and updated on August 24, 2022 in the PubMed (pubmed.gov), PsycINFO (EBSCO) and Embase (ELSEVIER) databases. The search compromised the following components: mental health, caregivers, and SCD. The full search strategies are available from the authors upon request. Search results were imported into an EndNote Citation Manager library, which was used to remove duplicate records. Unique records were imported into Covidence, (https://www.covidence.org), which was used to facilitate screening and data extraction.

Studies were included in this review if they met the following criteria: original research; caregiver (i.e., parent or primary caregiver) of a youth with SCD were study participants; caregiver mental health symptoms (e.g., anxiety, depressive symptoms) and emotional health (e.g., parenting stress) were assessed; a cross-sectional study design was used; and written in the English language. Studies that used data from larger longitudinal studies were included in this review if the secondary data analyses were performed at study baseline and were cross-sectional. All authors participated in the selection of studies with two reviewers independently assessing each study’s title and abstract with discrepancies discussed by the full team until agreement was reached regarding study inclusion or exclusion. Next, two authors independently reviewed the full text of remaining studies with discrepancies regarding inclusion rectified by the team until consensus was reached.

Data extraction.

Using an iterative process, the researchers developed a comprehensive data extraction form. The tool included the following fields: (1) study characteristics—first and last authors’ names, publication year, location, funding source; (2) sample characteristics—number and demographics of youth with SCD (i.e., sex, age, percent of the sample with hemoglobin SS [HbSS]), as well as any other control samples, number and demographics of caregivers (i.e., sex, age, marital status, highest education level, socioeconomic status [SES]); (3) mental and emotional health symptoms/constructs assessed and the measures used; (4) descriptive statistics for caregiver mental health; and (5) results of analyses testing associations between caregiver mental health and youth SCD-related outcomes. Two authors independently extracted the data for each study; the team resolved differences in the data extracted by consensus.

Quality appraisal.

Two authors independently appraised each study using the Newcastle-Ottawa Quality Assessment scale, a validated tool, 25 adapted for cross-sectional studies. 26 The scale contains eight items within three domains: selection (four criteria addressing sample representativeness, sample size, recruitment, and ascertainment of the exposure); comparability (one criterion addressing steps undertaken to statistically control for confounding variables); and outcome (three criteria addressing assessment of the outcome and appropriateness of the statistical test). The study team met weekly to review appraisals; discrepancies in ratings were discussed until consensus was achieved. Following consensus, the study team categorized overall study quality as good, fair, or poor consistent with recommendations of the Agency for Healthcare Research and Quality. 27 Details on scoring criteria within each domain and categorization of overall study quality are available from the authors upon request.

Data synthesis.

The broad study question addressed in this review as well as the heterogeneity in the included studies preclude a meta-analysis. Therefore, summary tables of evidence were created and examined. When creating the table summarizing participant characteristics (Table 1), caregiver demographic variables were collapsed for ease of interpretation as follows: (1) marital status = married or had a domestic partner; (2) highest education level = high school graduate or less, and at least some college or trade school; and (3) SES = lower income (U.S. settings: less than or equal to $30,000 [U.S. dollars not adjusted for inflation] or caregiver receives public assistance/insurance; non-U.S. settings in low/middle-income countries: proportion in lower half of the range of SES variable presented). Participant characteristics, when reported, were summarized across studies.

Full Article: https://pmc.ncbi.nlm.nih.gov/articles/PMC10683928/