Caregiver burden and risk of epithelial ovarian cancer in the Nurses' Health Studies.

Psychosocial stress may increase ovarian cancer risk and accelerate disease progression. We examined the association between caregiver burden, a common stressor, and risk of epithelial ovarian cancer. We prospectively followed 67,724 women in the Nurses' Health Study (NHS; 1992-2012) and 70,720 women in the NHSII (2001-2009) who answered questions on informal caregiving (i.e., caregiving outside of work). Women who reported no informal caregiving were considered non-caregivers while, among women who provided care outside of work, caregiver burden was categorized by time spent caregiving and perceived stress from caregiving. For the 34% of women who provided informal care for ≥15 hours per week, 42% described caregiving as moderately to extremely stressful. Pooled multivariate analyses indicated no difference in ovarian cancer risk for women providing ≥15 hours of care per week compared to non-caregivers (hazard ratio (HR)=0.96; 95% confidence interval (CI): 0.79-1.18), and no association was evident for women who reported moderate or extreme stress from caregiving compared to non-caregivers (HR=0.96; 95% CI: 0.75-1.22). Together with prior work evaluating job strain and ovarian cancer risk, our findings suggest that, when evaluating a stressor's role in cancer risk, it is critical to consider how the stressor contributes to the overall experience of distress.

Health care satisfaction and medical literacy habits among caregivers of individuals with Down syndrome.

Patients with Down syndrome have significant specialized health care needs. Our objective was to understand the needs, satisfaction, and online habits of caregivers as they care for persons with Down syndrome. A mixed-method survey was distributed through REDCap from April 2022 to June 2022 in the United States; a Spanish-translated version was distributed through SurveyMonkey from August 2022 to March 2023 in Mexico. We received 290 completed responses from the United States and 58 from caregivers in Mexico. We found that current health care options are not meeting the needs of many individuals with DS in both the United States (39.7%) and Mexico (46.6%). Caregivers expressed frustrations with the inaccessibility and inapplicability of health care information. In particular, they often found the volume of information overwhelming, given their limited medical background. Additionally, health care recommendations were not customized and lacked practical recommendations. Most caregivers in both the United States (72.1%) and Mexico (82.8%) believe it is not easy to find answers to medical questions about their loved ones with DS. Online platforms with customized, specific health information related to DS could offer innovative solutions to these unmet needs for families and primary care providers.

Impact of an automated, remote monitoring and coaching intervention in reducing hospice cancer family caregiving burden: A multisite randomized controlled trial.

BACKGROUND: Care for those with life-limiting cancer heavily involves family caregivers who may experience significant physical and emotional burden. The purpose of this study was to test the impact of Symptom Care at Home (SCH), an automated digital family caregiver coaching intervention, during home hospice, when compared to usual hospice care (UC) on the primary outcome of overall caregiver burden. Secondary outcomes included Caregiver Burden at weeks 1 and 8, Mood and Vitality subscales, overall moderate-to-severe caregiving symptoms, and sixth month spouse/partner bereavement outcomes. METHODS: Using a randomized, multisite, nonblinded controlled trial, 332 cancer family caregivers were enrolled and analyzed (159 SCH vs. 173 UC). Caregivers were primarily White (92%), female (69%), and spouse caregivers (53%). Caregivers provided daily reports on severity levels (0-10 scale) for their anxiety, depressed mood, fatigue, disturbed sleep, and caregiving interference with normal activities. These scores combined constituted the Caregiver Burden primary outcome. Based on reported symptoms, SCH caregivers received automated, tailored coaching about improving their well-being. Reports of moderate-to-severe caregiving symptoms also triggered hospice nurse notification. Secondary outcomes of Mood and Vitality were subcomponents of the Caregiver Burden score. A combined bereavement adjustment tool captured sixth month bereavement. RESULTS: The SCH intervention reduced overall Caregiver Burden compared to UC (p < .001), with a 38% reduction at 8 weeks and a medium-to-large effect size (d = .61). SCH caregivers experienced less (p < .001) disruption in both Mood and Vitality. There were higher levels of moderate-to-severe caregiving symptoms overtime in UC (OR, 2.722). All SCH caregivers benefited regardless of caregiver: sex, caregiver relationship, age, patient diagnosis and family income. SCH spouse/partner caregivers achieved better sixth month bereavement adjustment than UC (p < .007). CONCLUSIONS: The SCH intervention significantly decreased caregiving burden over UC and supports the maintenance of family caregiver mood and vitality throughout caregiving with extended benefit into bereavement.

Caregiver-reported quality of communication in pancreatic and periampullary cancer.

BACKGROUND: Communication between caregivers and clinical team members is critical for transitional care, but its quality and potential impact on outcomes are not well understood. This study reports on caregiver-reported quality of communication with clinical team members in the postpancreatectomy period and examines associations of these reports with patient and caregiver outcomes. METHODS: Caregivers of patients with pancreatic and periampullary malignancies who had undergone pancreatectomy were surveyed. Instrument measures assessed care experiences using the Caregiver Perceptions About Communication with Clinical Team Members (CAPACITY) instrument. The instrument has two main subscales: communication, assessing the extent to which providers helped caregivers comprehend details of clinical visits, and capacity, defined as the extent to which providers assessed whether caregivers were able to care for patients. RESULTS: Of 265 caregivers who were approached, 240 (90.6%) enrolled in the study. The mean communication and capacity subscale scores were 2.7 ± 0.6 and 1.5 ± 0.6, respectively (range, 0-4 [higher = better]). Communication subscale scores were lower among caregivers of patients who experienced (vs. those who did not experience) a 30-day readmission (2.6 ± 0.5 vs. 2.8 ± 0.6, respectively; p = .047). Capacity subscale scores were inversely associated with restriction in patient daily activities (a 0.04 decrement in the capacity score for every 1 point in daily activity restriction; p = .008). CONCLUSIONS: After pancreatectomy, patients with pancreatic and periampullary cancer whose caregivers reported worse communication with care providers were more likely to experience readmission. Caregivers of patients with greater daily activity restrictions were less likely to report being asked about the caregiver's skill and capacity by clinicians. PLAIN LANGUAGE SUMMARY: This prospective study used a validated survey instrument and reports on the quality of communication between health care providers and caregivers as reported by caregivers of patients with pancreatic and periampullary cancer after pancreatectomy. In an analysis of 240 caregivers enrolled in the study, lower communication scores (the extent to which providers helped caregivers understand clinical details) were associated with higher odds of 30-day patient readmission to the hospital. In addition, lower capacity scores (the extent to which providers assessed caregivers' ability to care for patients) were associated with greater impairment in caregivers. The strikingly low communication quality and capacity assessment scores suggest substantial room for improvement, with the potential to improve both caregiver and patient outcomes.

The cancer caregiving burden trajectory over time: varying experiences of perceived versus objectively measured burden.

INTRODUCTION: The objective of this study was to comprehensively understand the burden experienced by caregivers (CGs) providing home-based, end-of-life care to patients with cancer. We examined the relationship between objective and subjective burden including whether and how burden changes over time. METHODS: A case series of terminal cancer patient-caregiver dyads (n = 223) were recruited from oncology clinics and followed for 12 months or until patient death. Data were collected every other week and in-person from CGs in their homes using quantitative surveys, diaries, and monthly structured observations. RESULTS: Bivariate correlations revealed a significant association between subjective burden and activities of daily living (ADLs), instrumental activities of daily living (IADL), high-intensity tasks, and time spent on ADLs; these correlations varied over time. Models examining the slope of subjective burden revealed little systematic change; spouse caregiver and patient functional limitations were positively, and Black caregiver was negatively associated with subjective burden. Generally, the slopes for measures of objective burden were significant and positive. Models showed subjective burden was positively associated with most measures of objective burden both within caregiver (concurrent measures were positively associated) and between CGs (those with higher subjective also had higher objective). CONCLUSIONS: Cancer caregiving is dynamic; CGs must adjust to the progression of the patient's disease. We found an association between subjective and objective burden both within and between CGs. Black CGs were more likely to report lower subjective burden compared to their White counterparts. More detailed investigation of the sociocultural components that affect caregiver experience of burden is needed to better understand how and where to best intervene with targeted supportive care services.

Differences in trajectories of quality of life according to type of dementia: 6-year longitudinal findings from the IDEAL programme.

BACKGROUND: People with different types of dementia may have distinct symptoms and experiences that affect their quality of life. This study investigated whether quality of life varied across types of dementia and over time. METHODS: The participants were 1555 people with mild-to-moderate dementia and 1327 carers from the IDEAL longitudinal cohort study, recruited from clinical services. As many as possible were followed for up to 6 years. Diagnoses included were Alzheimer's disease, vascular dementia, mixed Alzheimer's and vascular dementia, Parkinson's disease dementia, dementia with Lewy bodies, and frontotemporal dementia. Self- and informant-rated versions of the Quality of Life in Alzheimer's Disease scale were used. A joint model, incorporating a mixed effects model with random effects and a survival model to account for dropout, was used to examine whether quality of life varied by dementia type at the time of diagnosis and how trajectories changed over time. RESULTS: The strongest associations between dementia type and quality of life were seen around the time of diagnosis. For both self-ratings and informant ratings, people with Parkinson's disease dementia or dementia with Lewy bodies had lower quality of life scores. Over time there was little change in self-rated scores across all dementia types (- 0.15 points per year). Informant-rated scores declined over time (- 1.63 points per year), with the greatest decline seen in ratings by informants for people with dementia with Lewy bodies (- 2.18 points per year). CONCLUSIONS: Self-rated quality of life scores were relatively stable over time whilst informant ratings showed a steeper decline. People with Parkinson's disease dementia or dementia with Lewy bodies report particularly low levels of quality of life, indicating the importance of greater attention to the needs of these groups.

Caregiver greeting to infants under 6 months already reflects emerging differences in those later diagnosed with autism.

As infants develop, caregivers adjust their behaviour to scaffold their infant's emerging skills, such that changes in infants' social abilities are expected to elicit changes in caregiver behaviour. We examined whether changes in the probability of infant-directed caregiving behaviour-specifically, greeting, a ubiquitous signal used by caregivers to initiate reciprocal interactions-differ between infant-caregiver dyads with an infant later diagnosed with autism and dyads with a neurotypically developing infant during infants' first 6 months. Using longitudinal data from 163 dyads, we found that caregivers in autism dyads (n = 40) used greeting less and at later infant ages than caregivers with a neurotypically developing infant (neurotypical dyads, n = 83). Caregivers in dyads with infants at elevated familial genetic likelihood for autism who did not receive an autism diagnosis (EL-non-autism dyads, n = 40) showed no differences in greeting compared with neurotypical dyads. Socioeconomic status partially mediated the difference between autism and neurotypical dyads. These findings show that autism and socioeconomic status were associated with the mutually adapted dynamics of dyadic interaction beginning in the first postnatal weeks. Importantly, differences in caregiver greeting observed in autism dyads are not interpreted as suboptimal behaviour from caregivers but rather indicate how early emerging social differences related to autism, years before overt features are present, may alter social learning opportunities elicited by the infant.

Post-stroke Cognition is Associated with Stroke Survivor Quality of Life and Caregiver Outcomes: A Systematic Review and Meta-analysis.

Disability arising from post-stroke cognitive impairment is a likely contributor to the poor quality of life (QoL) stroke survivors and their carers frequently experience, but this has not been summarily quantified. A systematic literature review and meta-analysis was completed examining the association between general and domain-specific post-stroke cognitive functioning and adult stroke survivor QoL, caregiver QoL, and caregiver burden. Five databases were systematically searched, and eligibility for inclusion, data extraction, and study quality were evaluated by two reviewers using a standardised protocol. Effects sizes (r) were estimated using a random effects model. Thirty-eight studies were identified, generating a sample of 7365 stroke survivors (median age 63.02 years, range 25-93) followed for 3 to 132 months post-stroke. Overall cognition (all domains combined) demonstrated a significant small to medium association with QoL, r = 0.23 (95% CI 0.18-0.28), p < 0.001. The cognitive domains of speed, attention, visuospatial, memory, and executive skills, but not language, also demonstrated a significant relationship with QoL. Regarding caregiver outcomes, 15 studies were identified resulting in a sample of 2421 caregivers (median age 58.12 years, range 18-82) followed for 3 to 84 months post-stroke. Stroke survivor overall cognitive ability again demonstrated a significant small to medium association with caregiver outcomes (QoL and burden combined), r = 0.17 (95% CI 0.10-0.24), p < 0.001. In conclusion, lower post-stroke cognitive performance is associated with significant reductions in stroke survivor QoL and poorer caregiver outcomes. Cognitive assessment is recommended early to identify those at risk and implement timely interventions to support both stroke survivors and their caregivers.

Global availability and uptake of psychological services for adults, caregivers and children with food allergy.

BACKGROUND: Food allergy (FA) is associated with poor health-related quality of life and high levels of psychological distress. Psychological support is extremely important but not always available. As part of the Global Access to Psychological Services for Food Allergy (GAPS) study, we aimed to assess psychological distress and service use among adults, caregivers and children with FA in a global survey. METHODS: Participants (n = 1329 adults with FA; n = 1907 caregivers of children with FA) from >20 countries were recruited through patient organisations, social media advertisements and online survey panels to complete an online survey. Surveys were available in six languages. RESULTS: A total of 67.7% of adults and 77.2% of caregivers reported direct experience, and 51.6% of caregivers said their child had experienced FA-related psychological distress. The most commonly reported issue was anxiety about having an allergic reaction. Less than 20% had been assessed for FA-related psychological distress. There were significant differences across countries for levels of distress, screening for distress, seeing a mental health professional and being diagnosed with a FA-related mental health disorder (all p < .001). The United Kingdom, Australia and Brazil had the highest number of participants reporting distress. The most commonly reported barrier to seeing a mental health professional was cost. CONCLUSIONS: FA-related distress is common across countries, but with substantial country-to-country variability. Allergy providers are encouraged to routinely assess families for psychological distress and provide access to appropriate mental health resources. Development and implementation of evidence-based, patient-informed accessible, affordable FA interventions in multiple languages is urgently needed.

Embedding Authorship Identity into a Portal-Based Agenda Setting Intervention to Support Older Adults and Care Partners.

BACKGROUND: Patients, families, and clinicians increasingly communicate through patient portals. Due to potential for multiple authors, clinicians need to know who is communicating with them. OurNotes is a portal-based pre-visit agenda setting questionnaire. This study adapted OurNotes to include a self-identification question to help clinicians interpret information authored by nonpatients. OBJECTIVES: To describe adapted OurNotes use and clinician feedback to inform broader implementation. DESIGN: Evaluation of adapted OurNotes in a geriatric practice. PARTICIPANTS: Older adults with a portal account and a clinic visit; eight clinicians were interviewed. INTERVENTION: OurNotes adaptation to clarify whether the author is the patient, the patient with help, or a nonpatient. APPROACH: Cross-sectional chart review of OurNotes completion, patient characteristics, and visit topics by author type. Clinician interviews explored experiences with OurNotes. RESULTS: Out of 503 visits, 134 (26%) OurNotes questionnaires were completed. Most respondents (n = 92; 69%) identified as the patient, 18 (14%) identified as the patient with help, and 24 (17%) identified as someone other than the patient. On average, patients who authored their own OurNotes were younger (80.9 years) compared to patients who received assistance (85.8 years), or patients for whom someone else authored OurNotes (87.8 years) (p < 0.001). A diagnosis of cognitive impairment was present among 20% of patients who self-authored OurNotes vs. 79% of patients where someone else authored OurNotes (p < 0.001). Topics differed when OurNotes was authored by patients vs. nonpatients. Symptoms (52% patient vs. 83% nonpatient, p = 0.004), community resources (6% vs. 42%, p < 0.001), dementia (5% vs. 21%, p = 0.009), and care partner concerns (1% vs. 12%, p = 0.002) were more often mentioned by nonpatients. Clinicians valued the self-identification question for increasing transparency about who provided information. CONCLUSIONS: A self-identification question can identify nonpatient authors of OurNotes. Future steps include evaluating whether transparency improves care quality, especially when care partners are involved.

The effect of internet-administered support (carer eSupport) on preparedness for caregiving in informal caregivers of patients with head and neck cancer compared with support as usual: a study protocol for a randomized controlled trial.

BACKGROUND: Informal caregivers (ICs) of patients with cancer provide essential and mainly uncompensated care. A self-perceived preparedness to care for the patient is associated with a lower caregiver burden, described as the extent to which caregiving is perceived as having adverse effects on IC functioning and well-being. ICs' well-being is associated with patient-perceived quality of care, suggesting that interventions to optimize ICs' health are essential in order to improve patient care. Head and neck cancer (HNC) is the seventh most common malignant disease in the world. The disease and its treatment have a significant negative impact on the patient's health and quality of life. Symptoms usually interfere with swallowing, food and fluid intake, breathing, speaking, and communication. ICs frequently manage patients' symptoms and side effects, especially problems related to nutrition and oral pain, without being properly prepared. Carer eSupport is an Internet-administered intervention, based on focus group discussions with ICs, developed in collaboration with ICs and healthcare professionals, tested for feasibility, and deemed feasible. This study protocol outlines the methods of investigating the effects of Carer eSupport plus support as usual (SAU) on self-reported preparedness for caregiving, caregiver burden, and well-being in the ICs of patients with HNC, compared with ICs receiving SAU only. METHODS AND ANALYSIS: In this randomized controlled trial, 110 ICs of patients with HNC, undergoing radiotherapy combined with surgery and/or medical oncological treatment, will be randomized (1:1) to Carer eSupport plus SAU or SAU only. Data will be collected at baseline (before randomization), post-intervention (after 18 weeks), and 3 months after post-intervention. The primary outcome is self-reported preparedness for caregiving. Secondary outcomes are self-reported caregiver burden, anxiety, depression, and health-related quality of life. The effect of Carer eSupport plus SAU on preparedness for caregiving and secondary outcomes, compared with SAU only, will be evaluated by intention to treat analyses using linear regression models, mixed-model regression, or analysis of covariance. DISCUSSION: If proven effective, Carer eSupport has the potential to significantly improve ICs' preparedness for caregiving and their wellbeing, thereby improving patient-perceived quality of care and patient wellbeing. TRIAL REGISTRATION: ClinicalTrials.gov; NCT06307418, registered 12.03.2024 (https://clinicaltrials.gov/search? term=NCT06307418).

Perception of unfamiliar caregivers during sickness - Using the new Caregiver Perception Task (CgPT) during experimental endotoxemia.

Social withdrawal is a well-established part of sickness behavior, but in some contexts sick animals might gain from keeping close instead of keeping away. For instance, sick individuals are more willing to be near known individuals who can provide care and safety (close others) compared to when healthy. Yet, interactions with some strangers might also be beneficial (i.e., healthcare professionals), but it is not known how sickness interplay with social behavior towards such individuals. Here, we assessed if sickness affects perception of caregivers, and developed a new task, the Caregiver Perception Task (CgPT). Twenty-six participants performed the CgPT, once after an injection of lipopolysaccharide (LPS, 0.8 ng/kg body weight, n = 24), and once after an injection of saline (n = 25), one hour and forty-five minutes post-injection. During the task, participants watched short video clips of three types of caregivers: a healthcare professional taking care of a sick individual, a healthcare professional not taking care of a sick individual, and a non-healthcare professional taking care of their sick adult child or partner. After each video clip, the likability, trustworthiness, professionalism, and willingness to interact with and receive care from the caregiver were rated on visual analogue scales. Results showed that participants injected with saline rated healthcare professionals who did not take care of a sick individual less positively on all aspects compared to healthcare professionals who took care of a sick individual. Moreover, compared to saline, LPS increased the participants' willingness to receive care from healthcare professionals and non-healthcare professionals providing care, but not from healthcare professionals not providing care. Thus, our results indicate that sick individuals may approach unknown individuals with potential to provide care and support.

Feasibility of caregiver diabetes distress screening in routine clinical care of youth with type 1 diabetes.

AIMS: Caregiver diabetes distress (DD) consists of feeling overwhelmed, sad, and/or concerned; one-third of parents of youth with type 1 diabetes (T1D) report severe distress up to 4 years after T1D diagnosis. PAID-PR (Problem Areas in Diabetes Survey-Parent Revised) assesses DD primarily in research settings; however, less is known about its clinical utility. We aimed to identify the feasibility of implementing PAID-PR screening at a diverse, academic US paediatric diabetes center during routine clinic follow-up visits through quality improvement methodologies. METHODS: The PAID-PR was intended to be offered in English to caregivers at all paediatric T1D appointments, by the front desk during appointment check-in, or through REDCap prior to telehealth appointments or at local sites to all eligible caregivers. Adult psychosocial resources were provided to all, regardless of score. Forms were scored after appointments; scores ≥80 were referred to Diabetes Psychology providers for follow-up. RESULTS: A total of 391 caregivers completed the PAID-PR, though only half of eligible caregivers received it in person. Response rates were highest in person (90%), compared to REDCap (25%). In total, 27% (n = 107) scored ≥56 (DD). Of those with DD, 21% (n = 23) scored ≥80 and were referred to psychology. Demographics are reported in Table 1. PAID-PR score was positively correlated to A1c (p = 0.038) and inversely to child age (p = 0.014). CONCLUSION: Clinic caregiver DD screening was implemented with higher response rates in person; however, expanding in-person screening to all eligible caregivers is necessary. Furthermore, since the PAID-PR was in English, some caregivers with DD were likely missed. Future directions include screening in additional languages.

Examining the impact of Native American myopathy on the quality of life and healthcare accessibility of patients and caregivers.

Native American myopathy (NAM, also known as STAC3 disorder) (OMIM 255995) is an ultra-rare genetic disease impacting multiple body systems. The quality of life and caregiver burden associated with this condition remain poorly characterized. In this study, the Pediatric Quality of Life Inventory and a survey comprised of de novo questions concerning genetic testing, counseling, and caregiver burden were employed to investigate the health-related quality of life (HRQoL) in patients and caregivers with NAM. Study findings uncovered a concerning trend: patients with NAM experienced a notable decline in HRQoL, with reasons that warrant further investigation. Particularly striking was the downturn observed during the transition from adolescence to adulthood-across Physical, Social, and Emotional Functioning domains. Taken together, this study has elucidated novel insights into the impact of NAM, and areas of concern to improve HRQoL have subsequently been highlighted.

Patient and caregiver preferences for haemophilia treatments: A discrete-choice experiment.

INTRODUCTION: An evolving haemophilia treatment landscape provides new possibilities for previously unattainable lifestyles. AIM: We sought to understand how people with haemophilia (PwH) and their caregivers value the potential benefits of novel prophylactic treatments. We conducted a discrete-choice experiment (DCE) to quantify preferences for features of haemophilia treatments among adults and caregivers of children with haemophilia. A best-worst scaling (BWS) exercise measured the perceived burden of treatment administration features. METHODS: A cross-sectional, web-based survey was administered to male adults (≥18 years) and caregivers of male children (≤17 years) living with haemophilia in the United States. Respondents evaluated eight pairs of hypothetical haemophilia treatment profiles defined by six attributes in the DCE and 15 features in the BWS. RESULTS: In the DCE, both adults with haemophilia (n = 151) and caregivers (n = 151) prioritised avoiding the risk of developing inhibitor/ anti-drug antibodies and treatments that allowed for a more active life. They placed a lower priority on reducing the number of spontaneous bleeding episodes, route and frequency of administration, and avoiding the risk of hospitalisation due to adverse events. The BWS documented the burdensomeness of IV infusions and medications that require mixing and refrigeration. CONCLUSION: PwH and caregivers prefer treatments that enable a more active lifestyle with a lower risk of inhibitor development. Both groups valued the ability to lead an active life over reducing spontaneous bleeding, with caregivers placing the most weight on this attribute. As new treatments expand possibilities, healthcare professionals and PwH should continue to share decision-making, incorporating clinical judgment and individual preferences.

Preference of treatment characteristics among people with haemophilia or their caregivers, and physicians in the Japanese healthcare environment.

INTRODUCTION: Studies of treatment preferences in haemophilia have been conducted in many countries. This study is the first to examine treatment characteristic preferences among people with haemophilia (PWH) and their caregivers, and physicians in Japan. AIM: To examine current treatment preferences of PWH and their caregivers, plus those of physicians at haemophilia treatment centres (HTCs) and non-HTCs for different treatment characteristics in Japan. METHODS: Physicians listed on a survey panel were invited to participate in the survey and to refer PWH and caregivers to participate in the survey. Web-based surveys were conducted to examine physician and PWH/caregiver background, prophylaxis background, prophylaxis goals, understanding of haemophilia treatment products, important information sources, preferences while choosing prophylaxis products, understanding of the patient's condition, and potential product switching. A discrete choice experiment exercise was included in the survey. RESULTS: A total of 107 physicians and 44 PWH/caregivers participated in the study. Key treatment goals of physicians included optimisation of haemophilia management. PWH/caregivers were focused on quality of life and reduced treatment burden. Consistent differences in haemophilia treatment strategies at HTCs and non-HTCs were observed for prescribed treatments, preferences in choosing prophylaxis products, understanding of patients' condition, and reasons for potential product switch. CONCLUSION: Our study utilises real-world survey data and presents preferences for haemophilia treatment characteristics among physicians, PWH and their caregivers in Japan, which could encourage improvements in individualised treatment and disease management. Alignment between treatment approaches at HTCs and non-HTCs could facilitate improvements in the quality of care for PWH across Japan.

A comprehensive systematic literature review of the burden of illness of Lennox-Gastaut syndrome on patients, caregivers, and society.

Fully elucidating the burden that Lennox-Gastaut syndrome (LGS) places on individuals with the disease and their caregivers is critical to improving outcomes and quality of life (QoL). This systematic literature review evaluated the global burden of illness of LGS, including clinical symptom burden, care requirements, QoL, comorbidities, caregiver burden, economic burden, and treatment burden (PROSPERO ID: CRD42022317413). MEDLINE, Embase, and the Cochrane Library were searched for articles that met predetermined criteria. After screening 1442 deduplicated articles and supplementary manual searches, 113 articles were included for review. A high clinical symptom burden of LGS was identified, with high seizure frequency and nonseizure symptoms (including developmental delay and intellectual disability) leading to low QoL and substantial care requirements for individuals with LGS, with the latter including daily function assistance for mobility, eating, and toileting. Multiple comorbidities were identified, with intellectual disorders having the highest prevalence. Although based on few studies, a high caregiver burden was also identified, which was associated with physical problems (including fatigue and sleep disturbances), social isolation, poor mental health, and financial difficulties. Most economic analyses focused on the high direct costs of LGS, which arose predominantly from medically treated seizure events, inpatient costs, and medication requirements. Pharmacoresistance was common, and many individuals required polytherapy and treatment changes over time. Few studies focused on the humanistic burden. Quality concerns were noted for sample representativeness, disease and outcome measures, and reporting clarity. In summary, a high burden of LGS on individuals, caregivers, and health care systems was identified, which may be alleviated by reducing the clinical symptom burden. These findings highlight the need for a greater understanding of and better definitions for the broad spectrum of LGS symptoms and development of treatments to alleviate nonseizure symptoms.

Burden and preparedness of care partners of people living with amyotrophic lateral sclerosis at home in Korea: A care partner survey.

INTRODUCTION/AIMS: The care burden of people living with amyotrophic lateral sclerosis (pALS) increases with disease progression. This study aimed to investigate the home care status and preparedness of care partners of pALS (cALS) in Korea. METHODS: An online survey was conducted with family care partners of patients diagnosed with ALS for over 1 year in 2022. The data collected included care time, depression evaluated using the patient health questionnaire-9 (PHQ-9), preparedness for caregiving scale (PCS), and caregiver competence scale (CCS). Results were compared based on whether the pALS underwent a tracheostomy or not. RESULTS: Ninety-eight cALS of 98 pALS participated in the study, of whom 59 pALS had undergone tracheostomy. Among the cALS, 60.2% were spouses, and 34.7% were children. The cALS took care of the patients for 13 (8-20) hours/day (median, interquartile range [IQR]) on weekdays and 15 (10-24) h/day on weekends. Among the cALS, 91.8% were depressed, and 28.6% had severe depression. The median (IQR) PCS and CCS scores were low (11/32 (8-15) and 8/20 (8-11), respectively), and both were lower in those caring for patients without than with tracheostomy (p < .001 and p < .02, respectively). Most cALS (77.6%) wished to continue caring for their pALS at home. DISCUSSION: Family care partners of pALS spend more than half of each day caring for patients and are often depressed. Most cALS preferred providing care at home, but felt ill-prepared. Designing home-based medical care is necessary for pALS to thrive at home.

Examining variability in Naturalistic Developmental Behavioral Intervention strategy use in caregivers of children with autism spectrum disorders.

BACKGROUND: Naturalistic Developmental Behavioral Interventions (NDBIs) for young children with autism spectrum disorder commonly involve caregiver-mediated approaches. However, to date, there is limited research on how caregivers' skills change, and, in turn, impact child outcomes. METHODS: We evaluated the NDBI strategy use of 191 caregivers prior to participation in NDBIs (or control groups) across multiple randomized controlled trials, using the Measure of NDBI Strategy Implementation, Caregiver Change (MONSI-CC). Clustering analyses were used to examine caregiver variability in NDBI strategy use at intervention entry. Generalized Linear Mixed Models were used to examine changes in caregiver strategy use over the course of intervention and its impact on changes in children's social communication. RESULTS: Using clustering analysis, we found that caregivers' baseline skills fit four profiles: limited, emerging, variable, and consistent/high, with few demographic factors distinguishing these groups. Caregivers starting with limited or emerging skills improved in their strategy use with intervention. Caregivers starting with more skills (consistent/high or variable) maintained higher skills over intervention. Children of caregivers in these groups who received target NDBIs improved in their social communication skills. CONCLUSIONS: Results suggested that caregiver skills improve through participation in NDBIs and may directly contribute to their children's outcomes, although more research on mediating factors is needed. Individualized approaches for caregivers and their children starting with differing skill profiles at intervention entry may be warranted.

Psychometric properties of disease-specific health-related quality of life instruments for food allergy: A COnsensus-based Standards for the selection of health Measurement INstruments-based systematic review.

Food allergies severely impact the health-related quality of life (HRQoL) of patients and their caregivers (family or informal caregivers). Currently there is no comprehensive review to provide an overview and critical assessment of the instruments in the field. Six databases were searched from inception until 10 August 2023, and a combination of subject terms and free words was used to search the literature. We used the COnsensus-based Standards for the selection of health Measurement INstruments methodology (COSMIN) to evaluate the measurement properties of the instruments. Forty-one studies reported on ten eligible instruments. Based on COSMIN guidelines, one instrument was recommended for Grade A, and the remaining nine instruments were recommended for Grade B. The Grade A instrument identified, the Food Allergy Quality of Life Questionnaire-Parent Form (FAQLQ-PF), can help researchers assess the effectiveness of treatment for patients with food allergy and to understand the psychosocial impact of the disease on patients.