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Abstract

Unpaid caregiving is associated with significant burdens, including financial strain, time constraints, diminished quality of life, and elevated stress levels. Despite these challenges, existing literature on disease burdens devotes limited attention to caregiver experiences. The aim of this scoping literature review was to identify instruments used to measure caregiver burden to better inform future studies of caregiver costs. This study included articles that estimated the costs or burdens associated with unpaid caregiving to patients in the United States and used a survey or cohort study design to conduct primary or secondary quantitative data analysis. Across the 46 articles abstracted, 27 unique survey instruments were identified; 23 (89%) instruments were validated, 12 (46%) were publicly available, and 14 (54%) were designed for or validated among caregivers. Among studies included in this review, 18 (39%) studies designed their own questionnaires to assess caregiver burden. This review additionally identified six nonsurvey data sources, such as medical claims data, used to estimate caregiver costs. The heterogeneity across measurement tools limits comparability across studies. Standardized, validated, and accessible instruments are essential for understanding caregiver burdens and advancing research to improve outcomes for patients and their caregivers.

Keywords

burdens caregiver costs survey instruments unpaid

Unpaid care plays a crucial and indispensable role in the health care, economic, and social landscape of the United States. An estimated 21% of US adults provide unpaid care to family members or friends, often stepping in as an extension of the health care system.¹ These unpaid caregivers—typically family members, partners, or close friends—offer nonprofessional, unpaid support to individuals with chronic illnesses, disabilities, or age-related conditions to help them maintain daily function and quality of life.^2,3 Their responsibilities span a wide range of tasks, including personal hygiene assistance, managing medications, providing meals, helping with mobility, and facilitating transportation to health care appointments.

Reliance on unpaid caregivers is becoming increasingly critical as the US health care system confronts an aging population with complex medical needs, including multiple chronic conditions and cognitive impairments such as Alzheimer’s disease. A report by the Assistant Secretary for Planning and Evaluation found that by 2040, the number of adults who need regular caregiving services will grow by 30%.⁴ However, the United States is facing significant shortages in the direct care workforce.⁵ Direct care workers—including home health aides, personal care aides, and nursing assistants—are in short supply due to low wages, high turnover, limited training opportunities, and challenging work conditions.^6,7 Many long-term care facilities and home care agencies are unable to recruit and retain sufficient staff to meet demand, particularly in rural or underserved areas.^8–10 This structural inadequacy in the formal care sector has placed an even greater burden on families to fill in the gaps through unpaid care.

Despite the critical value of unpaid caregiving, caregivers often face substantial personal and financial burdens. Specifically, caregivers frequently incur work-related opportunity costs such as missed workdays, reduced work hours, career disruptions, or early retirement.¹¹ Many caregivers also need to modify their living arrangements or household routines to accommodate the care recipients’ needs, leading to feelings of stress and anxiety for the caregiver.^12,13 Caregivers may also expend as much as 26% of their annual income on caregiving-related activities, including out-of-pocket medical expenses, medication costs, and assisted living facilities, contributing to feelings of financial strain and diminished household wealth.¹⁴

Current literature on disease burdens patients and their families experience focuses on the patient perspectives, often neglecting the unique and additional challenges caregivers face.^15–17 Two recent systematic reviews of out-of-pocket costs and indirect burdens borne by patients and families or caregivers found that only 20% of studies (n = 100) incorporated the caregiver perspective.^16,17 This omission is significant, as the burdens caregivers experience may be different from and additional to those patients experience, potentially leading to an underestimation of the overall impact of a disease. Furthermore, among the limited studies that do address caregiver burdens, definitions and methodologies are often inconsistent.¹⁵ These discrepancies hinder meaningful comparisons of burdens across studies and complicate efforts to monitor changes over time.

The goal of this review was to assess the methods and instruments used to collect caregiver burdens and identify the instruments that have been validated to better inform future studies of caregiver costs. This work builds on prior systematic reviews that identified patient and caregiver burdens.

Materials and Methods

Search and data sources

The authors of the current study identified a recent systematic review by Schmidt et al.¹⁷ that focused on burdens accrued by patients or unpaid caregivers, including direct medical costs, direct nonmedical costs, and indirect impacts. As Schmidt et al.¹⁷ included multiple categories of caregiver costs, the authors obtained and screened the list of citations included in Schmidt et al.¹⁷ as a starting point for this review.

Schmidt et al.¹⁷ searched PubMed, Embase, and Cochrane for English-language articles published between April 2017 and March 2022. The review included quantitative and qualitative studies of patient or caregiver burdens but excluded studies without patient or caregiver burdens; studies for which reported burdens could not be differentiated into categories; estimates for economic analyses, such as cost-of-illness studies and cost-effectiveness analyses; and policy analysis predicting future impacts.

In addition to screening the studies in Schmidt et al.,¹⁷ the authors searched PubMed for articles published between April 2022 and August 2024 using the search terms in Schmidt et al.¹⁷ The goal of this additional search was to expand the timeline of this scoping review and capture more recent publications. However, in this expanded PubMed search, the authors excluded terms relating to patients, including patient, beneficiary, and enrollee, because caregivers are the focus of this review. The authors also did not use the same list of exclusion terms from Schmidt et al.¹⁷ and instead opted to manually screen these exclusions out given that the search time frame of this study was shorter, and the inclusion criteria was expected to yield a manageable number of citations. See Appendix A for the detailed search strategy for Schmidt et al.¹⁷ and this review.

Study selection and data abstraction

The Preferred Reporting Items for Systematic reviews and Meta-Analyses and Joanna Briggs Institute guidance for scoping reviews was used to guide the screening and abstraction processes.^18,19 This study used a two-stage screening process to identify relevant studies. For the title/abstract and full-text review stages, the authors conducted a dual independent review of each article. A third reviewer was consulted to resolve disagreements at each stage. Studies were excluded if the full-text article was unavailable.

This review was limited to peer-reviewed research that (1) estimated the costs or burdens associated with unpaid caregiving (eg, care provided by family or friends) to patients, (2) focused on a US population, (3) was published in English, and (4) used a survey or cohort study design to conduct primary or secondary quantitative data analysis. This reviewed included studies whose data collection included the United States but also spanned other countries. Outcomes included any burden experienced by caregivers while providing care related to a medical need to a patient, such as work-related absenteeism or lost wages, presenteeism, transportation or travel, reductions in quality of life, and stress or anxiety resulting from caregiving. The authors excluded studies whose focus was on caregiving for nonmedical conditions, such as parental caregiving in absence of a medical need. Appendix B details the study inclusion criteria.

The authors used DistillerSR to abstract the study’s purpose, country, disease or treatment, sample size, caregiver burden categories, study survey instrument or secondary data sources, and frequency of data collection. The authors abstracted caregiver burden and study survey instrument information for direct medical costs, direct nonmedical costs, and indirect impacts because Schmidt et al.¹⁷ collected these categories. Although Schmidt et al.¹⁷ did not explicitly search for intangible burdens, the authors of this study also collected these data points for any intangible burden studies meeting the inclusion criteria that reported them. A single reviewer abstracted each study, whereas a second reviewer verified the abstracted elements.

Data synthesis

For each survey instrument identified in the data abstraction, the authors reviewed the instrument’s website (if available), the peer-reviewed literature, and the gray literature to identify burdens that are collectible in the instrument, the number of items included in the instrument, and whether the instrument was publicly available. The authors also conducted a search of the peer-reviewed literature to determine whether the instrument had been validated.

Instruments were categorized by the overarching type of burden collected, including direct medical, direct nonmedical, indirect, and intangible burden. This categorization is consistent with prior literature reviews on patient and caregiver burdens.^16,17,20 Direct medical costs include costs caregivers spend for health care services, such as copays, home health care costs, and medication costs. These may include costs for health care services for the patient or costs for health care services for the caregiver for conditions resulting from caregiving. Direct nonmedical costs include all other out-of-pocket spending attributable to managing or treating the patient (but not directly for medical services). Indirect impacts include measurable nonmonetary outlays that affect caregiver well-being and can be quantified, including caregiver productivity, financial impacts, and time impacts. Intangible burdens are also impacts to caregiver well-being, but intangible burdens represent qualitative experiences and feelings that are challenging to quantify, including psychological burdens and coping behaviors.²⁰ The authors identified burden categories using the reported elements in the instrument. The authors categorized instruments that collected elements from only one type of burden as one-dimensional survey instruments and instruments that collected elements across multiple burden categories (eg, direct medical and indirect elements) as multidimensional.

Results

Overview

The search strategy returned 952 records; 815 of these records were identified from Schmidt et al.,¹⁷ and the expanded search in PubMed identified 137 records (Fig. 1). Among these citations, 85 of these records were included for full-text review; however, the full texts for four records were unable to be located and these four records were excluded from further review. Of the 81 records screened during the full-text review, 46 studies were abstracted in this review. Most records were excluded for focusing on a non-US caregiver population (n = 860, 94%). A subset of records used only qualitative methods and were ineligible for this review (n = 42, 4%).

FIG. 1.

PRISMA diagram. A search of PubMed and a prior systematic review identified 952 articles. After abstract and full-text review, 46 articles met the inclusion criteria and were included for further analysis. PRISMA, Preferred Reporting Items for Systematic reviews and Meta-Analyses.

Table 1 describes the studies included in this review. Slightly more than half of studies (n = 24, 52%) focused on caregiver and patient burdens, whereas the remaining half focused on caregiver burdens alone (n = 22, 48%). More than half of all studies (n = 24, 52%) examined data on a national level, whereas fewer studies looked at multiple sites (n = 9, 20%) and single institutions (n = 10, 22%). Three studies (7%) examined data across multiple countries, including the United States. Surveys were the most common instrument for quantitative data collection (n = 41, 89%). Two studies used medical records, and five studies used medical claims. Only two studies used qualitative interviews in addition to their quantitative data collection. Other data sources included telemedicine appointment records and employee absenteeism data from integrated employer databases. Nine studies used a combination of data sources, including medical claims and employee absenteeism data (n = 4, 8%), survey and medical records (n = 1, 2%), survey and medical claims (n = 1, 2%), survey and telemedicine appointment records (n = 1, 2%), and surveys and interviews (n = 2, 4%). The majority of studies in this review were cross-sectional studies (n = 32, 70%), followed by retrospective cohort studies (n = 7, 15%), prospective cohort studies (n = 5, 11%), case–control (n = 1, 2%), and randomized controlled trials (n = 1, 2%).

Table 1.

Caregiver-Only and Caregiver/Patient-Focused Studies

Study characteristic	Studies (N = 46)	References
Study population
Caregiver only	22	^21–42
Caregiver and patient	24	^43–66
Geographical setting
National	24	^{23,25,29,31–33,36,37,40,45–49,51,52,54,55,57–59,61,65,66}
Multiple sites/cities/states	9	^{21,24,28,30,35,42,44,50,64}
Single institution/city/state	10	^{22,26,27,38,39,41,43,53,60,63}
Multiple countries, including United States	3	^34,56,62
Data source
Survey	41	^{21–24,26–32,34–48,50,51,53–64,66}
Medical records	2	^52,53
Medical claims	5	^{25,33,43,49,65}
Interviews	2	^42,51
Other	5	^{25,33,47,49,65}
Study design
Cross-sectional	32	^{21,22,24,26,28–32,37–40,42–46,48,50,51,53–58,60–63,66}
Retrospective cohort	7	^{25,33,34,49,52,59,65}
Prospective cohort	5	^{27,35,36,41,47}
Case–control	1	²³
Randomized controlled trial	1	⁶⁴

Note: Studies can be reported across multiple rows if they used more than one data source.

Nearly half of the studies in this review used an existing survey instrument (n = 21, 46%), whereas 18 studies (39%) designed one or more of their own questionnaires, and 8 studies (17%) did not report any information on their survey tool. In addition, 13 studies (28%) used other, nonsurvey data sources as their sole data source or in combination with a survey.

Caregiver burdens

Table 2 lists the four caregiver burden categories: direct medical, direct nonmedical, indirect and intangible, and corresponding subcategories identified by studies in this review. Among studies reporting direct medical costs, most studies did not indicate a subcategory (n = 19). The next most common direct medical cost subcategories were diagnosis/treatment/management costs (n = 7) and medical supply costs (n = 5). Only one study each estimated dental costs, hospice care costs, and long-term care facility costs.

Table 2.

Caregiver Burden Categories Estimated by Studies Included in this Review (N = 46)

Burden category	Elements	References
Direct medical (n = 23)	Diagnosis/treatment/management costs (beyond cost sharing) (n = 7)	^{22,38,42,45,47,57,64}
	Dental care costs (n = 1)	⁵¹
	Home health/home care costs (n = 4)	^25,38,42,64
	Hospice care costs (n = 1)	²⁵
	Insurance premiums (n = 3)	^50,54,61
	Long-term care facility costs (n = 1)	²⁵
	Medical supply costs (n = 5)	^{28,33,38,39,64}
	Over-the-counter product costs (n = 2)	^26,64
	Medical transportation (n = 3)	^38,46,50
	Medical costs, subcategories not stated (n = 19)	^{22,25,26,28–30,33,35,38,39,43,45,46,49,54,57,61,62,64}
Direct nonmedical (n = 24)	Accommodation costs (n = 7)	^{27,37,42,47,56,57,64}
	Child/dependent care costs (n = 8)	^{22,26–28,30,47,57,62}
	Clothing costs (n = 2)	^32,39
	Household care costs or bills (n = 5)	^{27,28,32,42,62}
	Home/vehicle modifications (n = 3)	^23,39,55
	Legal costs (n = 2)	^22,32
	Meal costs (n = 6)	^{27,32,37,39,43,64}
	Personal care costs (n = 3)	^32,39,62
	Transportation/travel/parking-related costs (n = 19)	^{27,28,30,35,37,39,42,43,46–48,50,52,53,56,57,60,62,64}
Indirect (n = 33)	Absenteeism/lost wages/loss of income (n = 27)	^{21,22,26–28,31,32,34–38,40,44,47,48,51,53–59,62,63,65}
	Informal care time (n = 13)	^{26,28,30,32,36–38,47,49,54,60,62,66}
	Presenteeism/productivity (n = 11)	^{22,23,31,32,34,38,40,41,51,57,62}
	Loss of leisure time (n = 1)	³⁸
Intangible (n = 22)	Anxiety, stress, depression (n = 10)	^{22–24,26,28,36,41,49–51}
	Medical debt, bankruptcy, depletion of assets, borrowing of money (n = 3)	^23,26,42
	Food insecurity, housing insecurity (n = 4)	^26,42,46,49
	Diminished ability to build savings and acquire assets (lower net worth, lower asset ownership) (n = 2)	^23,42
	Long-term career implications (unwanted changes in jobs and responsibilities, prolonged period away from work) (n = 6)	^{21–23,36,38,46}
	Financial stress/burden (n = 1)	³⁹
	Quality-of-life impacts (n = 12)	^{23,24,26,29,33,34,46,50,55,56,63,66–68}
	Family/social life impacts (n = 3)	^26,36,41
	Reduced spending on basic goods, groceries, and leisure activities (n = 5)	^{23,26,28,34,42}

Among studies reporting direct nonmedical costs, transportation/parking costs were most frequently estimated (n = 19), followed by child/dependent costs (n = 8) and accommodation costs (n = 7). Among studies reporting indirect impacts, studies most commonly estimated absenteeism/work loss (n = 26), time spent caregiving (n = 12), and presenteeism (n = 10). One study also collected data on lost leisure time among caregivers.

Among studies estimating intangible burdens, quality of life was the most commonly estimated subcategory (n = 12), followed by long-term career implications (n = 6) and reduced spending on basic goods, groceries, and leisure activities (n = 5).

One-dimensional survey instruments, by burden category

This study identified 19 unique one-dimensional survey instruments; these instruments collected direct medical, indirect, and intangible burdens among unpaid caregivers across 20 studies (Table 3).

Only one study used a one-dimensional existing survey instrument for direct medical costs, the CommonWealth Fund Biennial Health Insurance Survey (Table 3). This questionnaire is publicly available and includes questions on health insurance coverage and out-of-pocket medical costs. The study that employed the CommonWealth Fund questionnaire used a subset of questions from the survey on unexpected costs, increased out-of-pocket expenses, and financial worry.

Table 3.

Instruments Used to Collect Direct Medical Costs

Instruments	Validated	Publicly available	Specific to caregivers	Description
Direct Medical
CommonWealth Fund Biennial Health Insurance Questionnaire (n = 1)	Not located	Yes	No	Instrument that assesses unexpected costs, including increased out-of-pocket costs and financial worry
Indirect
Work Productivity and Activity Impairment (WPAI) (n = 12)	Yes	Yes	Yes	Six-item instrument that measures the number of work hours missed over the past 7 days in paid and unpaid work due to physical or emotional health problems. WPAI has been adapted for informal caregivers and measures caregiver depression, strain, number of activities of daily living tasks, and hours spent caregiving
Caregiver Work Limitations Questionnaire (WLQ) (n = 2)	Yes	Yes	Yes	25-item instrument to assess the impact of physical and emotional health problems associated with caregiving on work tasks
Intangible
Quality of life
DEMQOL-Proxy (Dementia Quality of Life) (n = 1)	Yes	Yes	Yes	31-item instrument that measures health-related quality of life in caregivers of people with dementia
CarerQol 7 Dimensions (CarerQol-7D) (n = 2)	Yes	Yes	Yes	Seven-dimensional instrument that measures the impact of providing informal care on caregivers, including impacts to caregiver relationships, mental health, ability to conduct daily activities alongside care responsibilities, financial well-being, physical health, personal fulfillment associated with caregiving, and caregiver support
Traumatic Brain Injury Caregiver Quality of Life (TBI-CareQoL) (n = 1)	Yes	Not located	Yes	15-item instrument to measure general and caregiving-specific aspects of health-related quality of life among caregivers of people with traumatic brain injury. Caregiving-specific dimensions include feelings of loss of self, anxiety, feelings of being trapped, and strain
Patient-Reported Outcomes Measurement Information System (PROMIS) 10 General Health (n = 2)	Yes	Yes	No	10-item instrument that evaluates physical, mental, emotional, and social health in adults and children
Healthy Days Module (n = 2)	Yes	Yes	No	Four-item instrument that measures health-related quality of life through questions on participant overall health, physical health, and mental health in the past 30 days
Visual Analog Scale (VAS) (n = 2)	Yes	Yes	No	Single-item subjective measure for acute and chronic pain where scores are recorded by making a handwritten mark on a 10-cm line that represents a continuum between “no pain” and “worst pain
EuroQol-5 Dimension (EQ-5D) (n = 1)	Yes	Fees may apply	No	Five-dimension instrument that elicits self-reported health status using measures of mobility, self-care, usual activities, pain or discomfort, and anxiety or depression. EQ-5D has multiple instruments ranging from 15 to 25 questions
Short Form Health Survey, 12 or 36 Questions (SF-12 or SF-36) (n = 2)	Yes	Fees may apply	No	Multi-item measure addressing eight concepts of health, including daily limitations, vitality, and pain due to health, physical, or emotional problems. SF-12 is a shortened version of the SF-36, created to reduce the burden of response
Quality of Life Neurological Disorders (NeuroQoL) (n = 1)	Yes	Fees may apply	No	Multi-item measure of quality of life, including physical, mental, and social health among patients with neurological conditions. This tool has been validated among caregivers of patients with neurological conditions
Financial toxicity
Comprehensive Score for Financial Toxicity (n = 1)	Yes	Not located	No	110-item instrument that measures financial distress associated with cancer diagnosis and treatment
HelpSteps (n = 1)	Not located	Not located	No	130-item instrument that assesses health-related social problems, including food insecurity, housing insecurity, and income
Caregiver stress, depression, or other emotional impacts
Caregiver Appraisal Scale (n = 1)	Yes	Yes	Yes	27-item instrument to measure general stress and benefits of caregiving, including caregiving tasks, relationships and interpersonal support, as well as the impact of caregiving on lifestyle, emotional health, and physical health
Caregiver Guilt Questionnaire (CGQ) (n = 1)	Yes	Not located	Yes	22-item instrument that measures guilt experienced by caregivers over five subdomains, including guilt about doing wrong by the care recipient, failing to meet the challenges of caregiving, experience of negative emotion related to caregiving, self-care, and neglecting other relatives
Urban Life Stress Inventory (n = 1)	Not located	Not located	No	21-item instrument to measure community-level stressors across economic stability, social and community context, and neighborhood/physical environment
Ryff Scales of Psychological Well-Being (n = 1)	Yes	Permission may be required	No	Multi-item instrument that measures psychological well-being, including autonomy, personal growth, environmental mastery, relationships with others, and purpose in life. This scale is available in a long form (84 items) and medium form (54 items)
Patient Health Questionnaire (PHQ) (n = 1)	Yes	Yes	No	Nine-item instrument used to screen, diagnose, monitor, and measure the severity of depression

Studies measuring indirect impacts from providing caregiving services (n = 14) used two existing instruments: the Work Productivity and Activity Impairment (WPAI) survey and Caregiver Work Limitations Questionnaire (WLQ; Table 3). WPAI was the most frequently used instrument (n = 12 studies) across studies measuring indirect impacts in this review; only two studies used WLQ. Both the WPAI and WLQ, which measure the impact of caregiving on productivity and impacts to work, are publicly available and validated. Notably, WPAI is a shorter measure of work impairment with 6 items, whereas WLQ is a 25-item instrument, which may have contributed to WPAI’s popularity.

Studies measuring intangible burdens (n = 21) employed 16 unique instruments that focused on 3 unique categories of intangible burdens: quality-of-life, emotional impacts, and financial toxicity (Table 3). Nine instruments measured caregiver quality of life. While three of the quality-of-life instruments specifically address caregiving populations, six were designed for general or patient populations but were administered to the caregiver populations. All quality-of-life instruments were validated in the peer-reviewed literature, and the majority (n = 5, 56%) are publicly available. Five instruments measured emotional impacts to caregivers, including caregiver depression, stress, and guilt. Only two of these instruments, the Caregiver Appraisal Scale and the Caregiver Guilt Questionnaire, were specifically designed to estimate emotional impacts of caregiving, whereas the remaining three instruments were designed for general or patient populations. An additional two instruments were used to measure financial toxicity among caregivers; however, neither instrument was specifically designed for caregivers.

Multidimensional survey instruments

Eight studies (17%) used an instrument that assessed a combination of burden categories (Table 4). Among these, this study identified seven unique instruments that investigated multiple burden categories associated with the costs of caregiving, including emotional, physical, financial, and quality-of-life impacts. The Hemophilia Caregiver Impact instrument was the only instrument used by two studies to assess caregiving burden, whereas the rest were employed by one study each. Dimensions assessed by the various instruments include emotional, physical, financial, social, and cognitive burdens. Although all instruments were validated, only two of them were confirmed to be available publicly. The length of these instruments varied substantially, from 4-item measures (the Zarit Burden Interview score) to the 180-item Family Experience Interview Schedule.

Table 4.

Instruments Measuring More Than One Burden Category

Instruments	Validated	Publicly available	Specific to caregivers	Description
Hemophilia Caregiver Impact (n = 2)	Yes	Unknown	Yes	36-item instrument that measures caregiver burden and emotions associated with caring for people with hemophilia, including practical impact, positive emotions, financial impact, emotional impact, physical impact, social impact, symptom impact, and lifestyle impact
Assessment of Caregiver Experience with Neuromuscular Disease (ACEND) (n = 1)	Yes	Fees may apply	Yes	41-item scale that measures caregiver burden and quality of life, including physical impacts, time impacts, emotional impacts, and financial impacts
Family Experience Interview Schedule (n = 1)	Yes	Not located	Yes	180-item interview that measures the caregiver and family experience, including assistance in daily living, impact on daily routines, financial expenditures, negative experiences, and positive experiences
Impact on Family Scale (n = 1)	Yes	Not located	Yes	27-item instrument that measures parents’ perceptions of the effects of their child’s health condition on family life, including financial impact, familial–social impact, personal strain, and mastery
Zarif Burden Interview Score (n = 1)	Yes	Yes	Yes	Four-item instrument that measures caregiver burden across three dimensions: demographics, mood toward caregiving, and impact of caregiving on caregiver quality of life
Mayo-Portland Adaptability Inventory, 4th Edition (n = 1)	Yes	Yes	Yes	Three-item subscale, 35-item instrument that assesses problems caregivers may encounter while treating individuals with brain injury, including physical, cognitive, emotional, behavioral, and social
Family Expense Diary (n = 1)	Yes	Not located	Yes	Instrument in which caregivers report nonmedical out-of-pocket expenses incurred by families or caregivers, including transportation, lodging, food, dependent care, housekeeping, lost work hours, and incidental expenses

Other, not stated instruments

Over 30% of studies used researcher-designed questionnaires (n = 18) to explore burdens associated with caregiving; most of these studies used researcher-designed questions in collaboration with an established survey instrument (n = 12, 67%). Although some studies indicated that they conducted internal validation on the researcher-designed questions, many did not specify validation efforts for the researcher-designed questions. Example questions included free responses such as “costs of hepatocyte transplantation,”⁴³ as well as yes/no questions, such as “has your child ever been evaluated or re-evaluated for special education services.”²¹

Nonsurvey data sources

Thirteen studies (28%) collected data from nonsurvey data sources (see Table 5). The nonsurvey data sources captured direct medical, direct nonmedical, and indirect impacts but did not include data for intangible burdens. The most commonly used nonsurvey data source was medical claims databases (n = 5). All five studies using claims data accessed the data from employer-sponsored repositories, including Merative MarketScan data and OptumHealth Care Solutions. In four of the studies, researchers linked claims data to integrated employer databases with information on employee absences, income, and disability payments. These studies identified patient–caregiver pairs using the primary plan holder identifier in the data and assumed the primary plan holder was a caregiver to the patient. Researchers then used information on the primary plan holders’ workplace absences and disability pay to estimate caregiver absences. One study also used historical income data to estimate the lost income progression of caregivers associated with their caregiving duties.

Table 5.

Additional Data Sources Used for Assessing Caregiver Burdens

Data source	Burden categories	Description	Example uses
Medical claims databases (n = 5)	Direct medical, indirect	Contains records submitted to health insurers for reimbursement, including diagnoses and medical procedures	Estimate total medical costs for a caregiver’s familySum number of medical appointments for patient to identify caregiver time away from work
Integrated employer databases (n = 4)	Indirect	Contains work absences, short-term disability leave, long-term disability leave, and employee income	Estimate caregiver time away from work using absencesEstimate total disability payoutsEstimate lost income progression due to caregiving responsibilities
Medical expenditure panel survey dataset (n = 3)	Direct medical, indirect	Nationally representative survey that collects data on the cost and use of health care along with access and use of health insurance	Estimate workdays missedEstimate total medical costs for a caregiver’s family
Medical records (n = 2)	Direct medical, direct nonmedical, indirect	Contains records on health care encounters, including patient demographics and address, medical diagnoses, and care received	Estimate caregivers’ travel time to clinic using patient addresses within geomapping toolsEstimate round-trip mileage costsSum hospital charges
National Survey of Children’s Health (n = 2)	Direct medical, direct nonmedical, indirect	Nationally representative survey data of the health and development of children and their families	Estimate workdays missedEstimate total medical costs for a caregiver’s family
Telemedicine appointment records (n = 1)	Indirect	Contains number of telemedicine visits, wait times, and visit durations	Estimate caregiver time away from work using visit duration and wait time

Five studies (11%) used data from two nationally representative survey: Medical Expenditure Survey (MEPS) and National Survey of Children’s Health (NSCH) to estimate direct medical, direct nonmedical, and indirect burdens to caregivers. Three studies analyzed data from the MEPS, a nationally representative, publicly available dataset sponsored by the Agency for Healthcare Research and Quality. MEPS collects data on families’ health care utilization and costs. Two studies analyzed data from NSCH, sponsored by the Health Resources and Services Administration. NSCH is publicly available and collects information related to the health and well-being of children, including access to and use of health care, family interactions, parental health, school and after-school experiences, and neighborhood characteristics.

Three studies used medical records and telemedicine appointment records to estimate indirect impacts to caregivers. Two studies extracted patient home addresses from medical records and used these addresses to estimate caregiver travel time for clinic visits using geomapping tools. One study used telemedicine appointment records to identify the amount of time a caregiver may have spent away from work for the appointment.

Discussion

This study identified 46 studies that estimated direct medical, direct nonmedical, indirect, and intangible burdens among caregivers. To the authors’ knowledge, this study is the first review to identify survey instruments that measure unpaid caregiver burden. This study identified 27 survey instruments and 6 nonsurvey data sources that were used in the literature to quantitatively estimate caregiver burden.

Most of the survey instruments in this review were validated in the peer-reviewed literature. However, only half of the instruments were publicly available and approximately one third of the studies in this review developed their own questionnaires. The cost and application process barriers to accessing validated instruments may lead researchers to design their own research questions. Use of researcher-designed questionnaires can hinder comparability across studies, as each set of questions may be unique. The validation of the researcher-designed questionnaires also varied, with several studies not reporting their validation approaches. Validation ensures that the instruments have face and content validity for their target populations and adequately capture what the instrument is intending to measure. Without validation, instruments may produce unreliable data that limit the generalizability of results.

Use of existing instruments varied by burden category, with direct medical cost studies being least likely to use existing survey instruments. Of the 23 identified studies estimating direct medical costs, most (n = 22, 96%) did not report using a survey instrument, opting to design their own questionnaire (44%) or use secondary data sources (26%) or did not report the instrument (26%). Although secondary data sources, such as medical claims and medical records, were used in some studies to approximate caregiver direct medical costs, these data sources may not be accessible to all researchers due to their high cost, restricted access, and use requirements. Further, these claims data covered only a subset of individuals with employer-sponsored private health insurance and thereby may not be representative of the caregiving population. For example, these databases would be unable to detect caregiving for elderly patients covered by Medicare. These studies also assumed that the policy holder was a caregiver to the patient and were unable to account for other paid or unpaid caregivers on the policy. Given that claims databases were more frequently used to estimate direct medical costs than survey instruments, these limitations could lead to misestimation of caregiver direct medical costs. Studies also used publicly available MEPS and NSCH data to assess direct medical and direct nonmedical costs; however, these datasets may not provide sufficient sample sizes for certain patient populations.

The variation in questions across survey instruments and infrequent use of each survey instrument may also limit comparability across studies. Only one instrument, the WPAI, was used in more than two studies. Further, burden definitions may vary across instruments, which complicate any summarization or meta-analyses of caregiver burdens. For example, nine different instruments measured health-related quality of life. In addition, only half of studies used an instrument designed specifically for caregivers. The burdens caregivers experience may differ from patient experiences, and instruments used in the caregiver population should also be designed to capture their needs and experiences. Using a patient-centered instrument could omit key caregiver-only categories, such as funeral costs and respite care.

Further, although studies estimated indirect impacts more frequently than other burden categories, the instruments used had a narrow scope. For example, WPAI estimates only missed workdays, so this instrument does not capture other indirect subcategories, such as travel time and lost leisure time. Although some of the indirect burdens may have been captured in multidimensional instruments, these multidimensional instruments were used by only eight studies.

Implications for future research

This study identified the instruments and other data sources used to collect caregiver burdens, along with gaps that could provide a starting point for further investigation. The authors recommend several areas for future research.

First, given that only half of studies used an instrument designed for caregivers, future work should (1) explore whether existing instruments for patients can be validated within a caregiver population and (2) identify any barriers to using these instruments among caregivers. This review found several instances where an instrument specifically designed for one medical condition was adapted to another condition or an instrument designed for a patient population was adapted to a caregiver population. For example, Schwartz et al. adapted the Hemophilia Caregiver Impact instrument to measure quality of life among caregivers of patients with Duchenne muscular dystrophy.²³ Future work should also explore whether existing patient instruments or disease-specific instruments could be adapted to a caregiver population.

Second, prior meta-analyses of caregiver burdens highlight wide variation in the instruments researchers use, within and across diseases.¹⁵ Most studies in this review employed unique instruments used by only one study, which complicates the interpretation and comparison of caregiver burdens across studies. Further, approximately 30% (n = 18) of the studies in this review used questions designed by researchers rather than existing questionnaires. The range of instruments used to measure caregiver burdens may be one of the major drivers of high variation in estimates of caregiver costs; each study is capturing a unique variant of the caregiver experience. To facilitate comparisons across measures of caregiver burdens, future research should consider an in-depth analysis of existing caregiver instruments to assess comparability of question domains across instruments and evaluate the appropriateness of each tool for evaluating caregiver burdens. Further, evaluations of caregiver support initiatives often use different instruments to measure caregiver burden, which also hinders comparability of results across evaluation efforts.^69,70 Understanding which instruments are most appropriate for measuring the caregiver burdens is important for identifying support types that most meaningfully alleviate caregiver burden and for tailoring these programs to the diverse needs of caregivers.

In addition to assessing comparability of tools, future work should also consider developing standards for collecting caregiver burdens. Standards have been established for other areas of patient-centered and health economics research, such as the Panel on Cost-Effectiveness in Health and Medicine.⁷¹ However, work to establish standards for caregiver burdens would first need to harmonize the definition of each burden, as prior research has found substantial variation in how caregiver burdens are defined.^15,20

Third, it was unclear if the use of researcher-developed questions was driven by need for flexible questions, barriers to access and use of existing instruments (eg, cost or length), unawareness of existing instruments, or another reason. Future research should consider identifying the rationale for use of researcher-developed questions because this could identify avenues to improve uptake of existing instruments.

Limitations

This study had several limitations. The authors based the search strategy on Schmidt et al.¹⁷ which did not include search terms for intangible burdens. This may have led to incomplete assessment of intangible survey instruments and data sources. Schmidt et al.¹⁷ also excluded cost estimates for economic analyses, and this review thereby does not include instruments and data sources potentially used in cost-of-illness studies estimating costs of unpaid caregiving.

Among the included studies, not all studies directly stated the survey instruments used and the populations taking the surveys. For example, the authors were unable to distinguish between studies that administered the patient version of WPAI versus those that administered the caregiver version of WPAI. In addition, for studies that surveyed caregivers and patients, the population on which the survey instruments were used was not always clear. This study cautiously included the instrument when the surveyed population was unclear, and the results may include some instruments focused on only patient burdens.

Consistent with guidelines for scoping reviews, this study did not conduct a formal assessment of quality for the studies or survey instruments identified in the review. Thus, the results cannot draw conclusions about the strength of the body of evidence or quality of the survey instruments. To increase the applicability of findings, this study limited the review to studies published in English from the United States.

Conclusions

Unpaid caregivers incur significant financial and nonfinancial burdens attributable to their caregiving duties. Given the increasing need for unpaid and family caregivers, it is imperative to understand the daily burdens these individuals face. However, the large number of instruments used to measure caregiver burdens may limit the ability to compare estimates of caregiver burden across studies. Continued assessments of existing instruments and identifying avenues to harmonize burden measures are critical to improving the outcomes and experiences of those patients and their caregivers.

Authors’ Contributions

D.P.: Writing—original draft (lead), writing—review and editing (equal), conceptualization (equal), formal analysis (lead), and methodology (lead). K.G.: Writing—review and editing (equal), funding acquisition (equal), conceptualization (equal), and writing (supporting). E.S.: Writing—original draft (supporting), formal analysis (supporting), investigation (supporting), and writing—review and editing (supporting). Z.T.: Formal analysis (supporting) and investigation (supporting). M.D.: Formal analysis (supporting) and investigation (supporting). O.K.: Writing—review and editing (equal), conceptualization (equal), and funding acquisition (equal).

Footnotes

Acknowledgments

The authors would like to acknowledge the editorial support for this article by Margaret Johnson from RTI International and acknowledge the help of Theresa Schmidt, who provided the review team with a list of citations from her review, Expanding the Catalog of Out-of-Pocket Costs.

Author Disclosure Statement

The authors did not have any conflicts of interest.

Funding Information

This literature review was supported by Patient-Centered Outcomes Research Institute (PCORI).

Disclaimer

All statements, findings, and conclusions presented in this publication are solely those of the authors and do not necessarily represent the views of the Patient-Centered Outcomes Research Institute® (PCORI®). This publication was developed from materials originally created through a PCORI® contract [IDIQ-TO#05-RTI-ENG-AOSEPP-09-20-2021] to support PCORI’s work. Questions or comments may be sent to PCORI at info@pcori.org.

Appendix

Appendix B.

Selection Criteria

	Inclusion	Exclusion
Population	Unpaid or informal caregivers of patients	Individuals other than families/caregivers of patients (eg, doctors, nurses, hospitals)
Intervention	Medical condition causing impact to caregiver	Parental caregiving unless related to medical condition
Outcomes	Caregiver burdens (direct costs, indirect impacts, intangible costs)	None
Setting/publication type	Quantitative data collection Peer-reviewed publications Published in 2017 or later Published in English Published in the United States	Nonquantitative studies (eg, qualitative studies, study protocols, policy analysis, hypothetical results, literature reviews) Book sections, comments, letters, editorials Published before 2017 Published in a language other than English Study population is outside the United States Dissertations, commentaries, working papers, news articles, trade publications

References

AARP. National Alliance for Caregiving. Caregiving in the United States, 2020. 2020. [Last accessed December 4, 2025]. Available from: https://www.aarp.org/pri/topics/ltss/family-caregiving/caregiving-in-the-united-states/

Colombo

, Llena-Nozal

, Mercier

, Tjadens

. Help wanted? providing and paying for long-term care. Organisation for Economic Co-operation and Development Publishing, 2011, Paris; doi: 10.1787/9789264097759-en

Meulenbroeks

, Schroeder

, Epp

. Bridging the gap: A mixed methods study investigating caregiver integration for people with geriatric syndrome. Int J Integr Care, 2021; 21(1):14.

Favreault

, Dey

, Anderson

, Lamont

, Marton

. Future change in caregiving networks: How family caregivers and direct care workers support older adults now and in the future. Office of the Assistant Secretary for Planning and Evaluation, 2023. Available from: https://aspe.hhs.gov/sites/default/files/documents/a449863a8c93838d37f78ccf29e9231f/future-change-caregiving-networks.pdf. [Last accessed December 4, 2025].

PHI National. Direct Care Workers in the United States: Key Facts. 2022. Available from: https://www.phinational.org/wp-content/uploads/2022/08/DCW-in-the-United-States-2022-PHI.pdf. [Last accessed December 4, 2025].

Harootunian

, Perry

, Buffett

, O’Gara

, Hoagland

. Addressing the direct care worker shortage. Bipartisan Policy Center, 2023. Available from: https://bipartisanpolicy.org/report/addressing-the-direct-care-workforce-shortage/. [Last accessed December 4, 2025].

Khavjou

, Suarez

, Tyler

, Squillace

, Dey

, Oliveira

. Wages of direct care workers lower than other entry-level jobs in most states: Issue brief. Office of the Assistant Secretary for Planning and Evaluation,. 2023. Available from: https://aspe.hhs.gov/sites/default/files/documents/7a611d901c615e5611ea095b1dcf8d08/wages-dcw-lower-ib.pdf. [Last accessed December 4, 2025].

Falvey

, Hade

, Friedman

, et al. Severe neighborhood deprivation and nursing home staffing in the United States. J Am Geriatr Soc, 2023; 71(3):711–719.

Leland

, Prusynski

, Shore

, et al. Skilled nursing facility staffing shortages: Sources, strategies, and impacts on staff who stayed. Health Serv Res, 2024; 59(6):e14355; doi: 10.1111/1475-6773.14355

10.

Sharma

, Bin Abdul Baten

, Ullrich

, MacKinney

, Mueller

. Nursing home closures and access to post-acute care and long-term care services in rural areas. J Rural Health, 2024; 40(3):557–564.

11.

Chari

, Engberg

, Ray

, Mehrotra

. The opportunity costs of informal elder-care in the United States: New estimates from the American Time Use Survey. Health Serv Res, 2015; 50(3):871–882.

12.

Liu

, Heffernan

, Tan

. Caregiver burden: A concept analysis. Int J Nurs Sci, 2020; 7(4):438–445.

13.

Martsolf

, Kandrack

, Rodakowski

, et al. Work performance among informal caregivers: A review of the literature. J Aging Health, 2020; 32(9):1017–1028.

14.

Skufca

, Rainville

. Caregiving Out-of-Pocket Costs Study 2021. AARP Research,. 2021. Available from: https://www.aarp.org/pri/topics/ltss/family-caregiving/family-caregivers-cost-survey/. [Last accessed December 4, 2025].

15.

Das

, Nguyen

, Ho

, Lee

, Robinson

, Gao

. Methods for measuring and valuing informal care: A systematic review and meta-analysis in stroke. Value Health, 2024; 27(12):1789–1804; doi: 10.1016/j.jval.2024.06.007

16.

Poehler

, Buell

, Bingaman

, Daniel

, Hayes

. Out-of-pocket and indirect patient and caregiver cost taxonomy: A scoping review. 2023. Prepared by RTI International. Available from: https://www.pcori.org/sites/default/files/PCORI-Out-of-Pocket-Cost-Taxonomy-Scoping-Review-Sept-2023.pdf. [Last accessed December 5, 2025].

17.

Schmidt

, Juday

, Patel

, Karmarkar

, Smith-Howell

, Fendrick

. Expanding the catalog of patient and caregiver out-of-pocket costs: A systematic literature review. Popul Health Manag, 2024; 27(1):70–83.

18.

Peters

, Godfrey

, McInerney

, Munn

, Tricco

, Khalil

. Scoping reviews. In: Joanna Briggs Institute reviewer’s manual. 2017. Available from: https://jbi.global/scoping-review-network/resources. [Last accessed December 5, 2025].

19.

Tricco

, Lillie

, Zarin

, et al. PRISMA extension for scoping reviews (PRISMA-ScR): Checklist and explanation. Ann Intern Med, 2018; 169(7):467–473.

20.

Giombi

, D’Angelo

, Kirsch

, Shenkar

. Intangible burdens incurred from patient and caregiver out-of-pocket costs for medical care: A scoping review. BMJ Open, 2025; 15(5):e095832; doi: 10.1136/bmjopen-2024-095832

21.

Gordon

, Welkie

, Quaye

, et al. Burden of employment loss and absenteeism in adults and caregivers of children with sickle cell disease. Blood Adv, 2024; 8(5):1143–1150.

22.

Zhao

, Page

, Altszuler

, et al. Family burden of raising a child with ADHD. J Abnorm Child Psychol, 2019; 47(8):1327–1338.

23.

Schwartz

, Stark

, Audhya

, Gooch

. Characterizing the quality-of-life impact of Duchenne muscular dystrophy on caregivers: A case-control investigation. J Patient Rep Outcomes, 2021; 5(1):124.

24.

Brickell

, Wright

, Lippa

, et al. Resilience is associated with health-related quality of life in caregivers of service members and veterans following traumatic brain injury. Qual Life Res, 2020; 29(10):2781–2792; doi: 10.1007/s11136-020-02529-y 1879–1889.

25.

Brook

, Rajagopalan

, Smeeding

. Healthcare costs and absenteeism among caregivers of adults with partial-onset seizures: Analysis of claims from an employer database. Am Health Drug Benefits, 2018; 11(8):396–403.

26.

Chang

, Shah

, Hoefgen

, et al.; H2O Study Group. Lost earnings and nonmedical expenses of pediatric hospitalizations. Pediatrics, 2018; 142(3):e20180195.

27.

Clark

, Cummings

, Kuhlthau

, Frassica

, Noviski

. Impact of pediatric intensive care unit admission on family financial status and productivity: A pilot study. J Intensive Care Med, 2019; 34(11–12):973–977.

28.

Edwards

, Panitch

, Constantinescu

, Miller

, Stone

. Survey of financial burden of families in the U.S. with children using home mechanical ventilation. Pediatr Pulmonol, 2018; 53(1):108–116.

29.

Galvin

, Howard

, Denny

, Dickinson

, Tatton

. The social and economic burden of frontotemporal degeneration. Neurology, 2017; 89(20):2049–2056.

30.

Lakshmanan

, Song

, Belfort

, et al. The financial burden experienced by families of preterm infants after NICU discharge. J Perinatol, 2022; 42(2):223–230.

31.

Lerner

, Benson

, Chang

, et al. Measuring the work impact of caregiving for individuals with schizophrenia and/or schizoaffective disorder with the Caregiver Work Limitations Questionnaire (WLQ). J Occup Environ Med, 2017; 59(10):1007–1016.

32.

Lerner

, Lavelle

, Adler

, et al. A population-based survey of the workplace costs for caregivers of persons with treatment-resistant depression compared with other health conditions. J Occup Environ Med, 2020; 62(9):746–756.

33.

Martínez-Martín

, Macaulay

, Jalundhwala

, et al. The long-term direct and indirect economic burden among Parkinson’s disease patients in 5 Western European countries. Movement Disorders, 2019; 34(3):362–371.

34.

Mighiu

, O’Hara

, Ferri Grazzi

, et al. Impact of progressive familial intrahepatic cholestasis on caregivers: Caregiver-reported outcomes from the multinational PICTURE study. Orphanet J Rare Dis, 2022; 17(1):32.

35.

Nelson

, Ma

, Cheng

, Ewing-Cobbs

, Clark

, Keenan

. Healthcare utilization and missed workdays for parents of children with traumatic brain injury. J Head Trauma Rehabil, 2019; 34(4):257–267.

36.

Suthoff

, Mainz

, Cox

, et al. Caregiver burden due to pulmonary exacerbations in patients with cystic fibrosis. J Pediatr, 2019; 215:164–171.e2; doi: 10.1016/j.jpeds.2019.08.038

37.

Vessey

, DiFazio

, Strout

, Snyder

. Impact of non-medical out-of-pocket expenses on families of children with cerebral palsy following orthopaedic surgery. J Pediatr Nurs, 2017; 37:101–107.

38.

Whittington

, Knupp

, Vanderveen

, Kim

, Gammaitoni

, Campbell

. The direct and indirect costs of Dravet Syndrome. Epilepsy Behav, 2018; 80:109–113.

39.

Hastert

, Kyko

, Ruterbusch

, et al. Caregiver costs and financial burden in caregivers of African American cancer survivors. J Cancer Surviv, 2024; 18(2):565–574.

40.

Keita Fakeye

, Samuel

, Drabo

, Bandeen-Roche

, Wolff

. Caregiving-related work productivity loss among employed family and other unpaid caregivers of older adults. Value Health, 2023; 26(5):712–720.

41.

Ozluk

, Cobb

, Hoots

, Sylwestrzak

. Association between mobile app use and caregivers’ support system, time spent on caregiving, and perceived well-being: Survey study from a large employer. J Med Internet Res, 2022; 24(4):e28504; doi: 10.2196/28504

42.

Waters

, Biddell

, Killela

, et al. Financial burden and recommended multilevel solutions among caregivers of pediatric hematopoietic stem cell transplant recipients. Pediatr Blood Cancer, 2023; 70(12):e30700; doi: 10.1002/pbc.30700

43.

Rose

, Grosse

, Garcia

, et al. The financial and time burden associated with phenylketonuria treatment in the United States. Mol Genet Metab Rep, 2019; 21:100523.

44.

Chang

, Renehan

, Taylor

, et al. Societal costs of localized renal cancer surgery. Can J Urol, 2018; 25(4):9401–9406.

45.

Chovatiya

, Begolka

, Thibau

, Silverberg

. Impact and associations of atopic dermatitis out-of-pocket health care expenses in the United States. Dermatitis, 2022; 33(6S):S43–S51.

46.

Datz

, Tumin

, Miller

, Smith

, Bhalla

, Tobias

. Pediatric chronic pain and caregiver burden in a national survey. Scand J Pain, 2019; 19(1):109–116.

47.

Finkelstein

, Cahill

, Young

, et al. Telemedicine for pediatric urological postoperative care is safe, convenient and economical. J Urol, 2020; 204(1):144–148; doi: 204(3):569–573

48.

Gauthier-Loiselle

, Michalopoulos

, Cloutier

, et al. Costs associated with the administration of erythropoiesis-stimulating agents for the treatment of anemia in patients with non-dialysis-dependent chronic kidney disease: A US societal perspective. J Manag Care Spec Pharm, 2021; 27(12):1703–1713.

49.

Kahn

, Lin

, Ozbay

, Wang

, Chao

, Skup

. Indirect costs and family burden of pediatric Crohn’s disease in the United States. Inflamm Bowel Dis, 2017; 23(12):2089–2096.

50.

Kaye

, Lybrand

, Chew

. Assessment and management of psychosocial needs: Social work utilization in comprehensive cleft team care. Cleft Palate Craniofac J, 2018; 55(8):1081–1091.

51.

King-Stephens

, Wheless

, Krogh

, et al. Burden of disease in patients with a history of status epilepticus and their caregivers. Epilepsy Behav, 2020; 112:107374.

52.

Liberman

, McCarthy

. The cost of callbacks: Return visits for diagnostic imaging discrepancies in a pediatric emergency department. Emerg Radiol, 2019; 26(4):381–389.

53.

Meirick

, Shah

, Dolan

, Weinstein

. Determining the prevalence and costs of unnecessary referrals in adolescent idiopathic scoliosis. Iowa Orthop J, 2019; 39(1):57–61.

54.

Schaible

, Colquit

, Caciula

, Carnes

, Li

, Moreau

. Comparing impact on the family and insurance coverage in children with cerebral palsy and children with another special healthcare need. Child, 2018; 44(3):370–377.

55.

Schwartz

, Stark

, Borowiec

, Audhya

, Gooch

. Interplay of disability, caregiver impact, and out-of-pocket expenditures in Duchenne muscular dystrophy: A cohort study. J Patient Rep Outcomes, 2022; 6(1):21.

56.

Shenoi

, Horneff

, Cidon

, et al. The burden of systemic juvenile idiopathic arthritis for patients and caregivers: An international survey and retrospective chart review. Clin Exp Rheumatol, 2018; 36(5):920–928.

57.

Skalicky

, Rentz

, Liu

, et al. Economic burden, work, and school productivity in individuals with tuberous sclerosis and their families. J Med Econ, 2018; 21(10):953–959.

58.

Sullivan

, Ghushchyan

, Navaratnam

, et al. School absence and productivity outcomes associated with childhood asthma in the USA. J Asthma, 2018; 55(2):161–168.

59.

Sullivan

, Ghushchyan

, Navaratnam

, et al. The national burden of poorly controlled asthma, school absence and parental work loss among school-aged children in the United States. J Asthma, 2018; 55(6):659–667.

60.

Tankersley

, Winders

, Aagren

, et al. Subcutaneous immunotherapy takes more than the time in the clinic. Curr Med Res Opin, 2021; 37(11):1925–1931.

61.

Wang

, Valero-Elizondo

, Ali

, et al. Out-of-pocket annual health expenditures and financial toxicity from healthcare costs in patients with heart failure in the United States. J Am Heart Assoc, 2021; 10(14):e022164; doi: 10.1161/JAHA.121.022164

62.

Xiao

, Bertwistle

, Khela

, Middleton-Dalby

, Hall

. Patient and caregiver socioeconomic burden of first-line systemic therapy for advanced gastroesophageal adenocarcinoma. Future Oncol, 2022; 18(10):1199–1210.

63.

Zand

, Kim

, van Deen

, et al. The effects of inflammatory bowel disease on caregivers: Significant burden and loss of productivity. BMC Health Serv Res, 2020; 20(1):556.

64.

Kaufman

, Huang

, Holland

, et al. Healthcare use and out-of-pocket costs for rural family caregivers and care recipients in a randomized controlled trial. J Am Geriatr Soc, 2024; 72(8):2523–2531.

65.

Pandya

, Young

, Packnett

, et al. Work absenteeism, disability, and lost wages among patients with acute myeloid leukemia and their caregivers: A cohort study using US administrative claims and productivity data. Expert Rev Pharmacoecon Outcomes Res, 2024; 24(4):521–532.

66.

Tahami Monfared

, Khachatryan

, Hummel

, et al. Assessing quality of life, economic burden, and independence across the Alzheimer’s disease continuum using patient-caregiver dyad surveys. J Alzheimers Dis, 2024; 99(1):191–206.

67.

Burns

, Abby

, Watts

, Mohamed

. (2025. How do Medicaid Home Care Programs Support Family Caregivers? Retrieved from. Available from: https://www.kff.org/medicaid/how-do-medicaid-home-care-programs-support-family-caregivers/

68.

Swartzell

, Fulton

, Crowder

. State-level Medicaid 1915(c) home and community-based services waiver support for caregivers. Nurs Outlook, 2022; 70(5):749–757; doi: 10.1016/j.outlook.2022.06.005

69.

Lopez-Hartmann

, Wens

, Verhoeven

, Remmen

. The effect of caregiver support interventions for informal caregivers of community-dwelling frail elderly: A systematic review. Int J Integr Care, 2012; 12:e133; doi: 10.5334/ijic.845

70.

Zhou

, Zhang

, Pan

, et al. Instruments for Measuring the Burden of Family Caregivers of Cancer Patients: A Systematic Review of Measurement Properties. J Clin Nurs, 2025; 34(7):2479–2492; doi: 10.1111/jocn.17548

71.

Neumann

, Sanders

, Russell

, Siegel

, Ganiats

. Cost-effectiveness in health and medicine. Oxford University Press, 2016.