Positive emotions despite substantial burden: The alchemy of haemophilia caregiving

Stark

Borowiec

et al. 2022

Self Cite

Background Providing caregiving support to people with Duchenne muscular dystrophy (DMD) is challenging, beginning in early childhood, and continuing through the progression of multidimensional disability. This study addressed the interplay between caregiver impact, out-of-pocket expenditures, and DMD disability. To examine these interconnections, we investigated the association between caregiver impact domains and out-of-pocket expenditures; and the presence of clusters in caregivers on the basis of DMD-related disability domains in the patients for whom they provided caregiving support. Methods This web-based study recruited 566 DMD caregivers (140 males, 426 females; mean age 41.6 years, SD 8.8, range 21–72), examining caregiver impact using the DMD Caregiver Impact Measure, PROMIS-derived parent-proxy (PPP) measures of their child’s disability, and items tapping out-of-pocket expenditures related to home and vehicle accommodations and assistive devices. T-tests compared caregiver impact scores by out-of-pocket expenditures incurred. Latent Profile Analyses (LPA) were conducted to generate impact profiles related to child’s disability as reported by caregiver proxies. Results Higher out-of-pocket expenditures were generally associated with worse impact on the subscales, but several expenditures (e.g., kitchen, bathroom, scooter) were associated with lower impact. LPA indicated that the four-group solution provided the best relative fit and yielded good profile separation (entropy = 0.91). Caregivers with lowest impact reported the highest mobility, cognitive, and upper extremity functioning of their DMD care recipients, whereas the highest caregiver impact was driven by their care recipient’s negative affect and fatigue. The upper-middle impact group showed great variability in proxy-disability domains, whereas the lower-middle group had similar levels of disability across domains. Profiles were represented across all child ages. Conclusion Out-of-pocket expenditures were often associated with worse caregiver impact, but some associated with milder impact (i.e., bathroom or kitchen modification, investing in a ceiling lift or medical scooter). While their son’s level of disability and age were related to impact on the DMD caregiver, the domains giving rise to highest caregiver impact were not the most visible aspects of disability, such as mobility, but rather negative affect and fatigue. Other contextual attributes are likely implicated, and will be addressed in the companion paper.

Section: Discussionsupporting

confidence: 88%

Interplay of disability, caregiver impact, and out-of-pocket expenditures in Duchenne muscular dystrophy: a cohort study

Stark

Borowiec

et al. 2022

Self Cite

“…Their prominent focus on their brother's health and family welfare suggest core intrinsic values, such as those centered on relationships, personal well-being, and community [34]. These findings show some similarity with research on hemophilia caregivers that suggested that adopting positive ways of thinking about one's life limitations may transform the negatives of caregiving into something positive [35]. This study focused on description, and explicitly avoided interpreting the valence of such differences.…”

Section: Discussionmentioning

confidence: 68%

Siblings’ life aspirations in the context of Duchenne muscular dystrophy: a mixed-methods case-control study

Biletch

Stuart

et al. 2022

Self Cite

Background The inevitable and progressive loss of independence in Duchenne Muscular Dystrophy (DMD) patients may have an impact on their siblings’ life aspirations. The present cross-sectional case-control study investigated how aspirations differed between brothers and sisters of people with DMD and a stratified comparison group of nationally representative children/adults. Methods A web-based survey was administered October through December 2020. Aspirations were measured using qualitative (open-ended) and quantitative (closed-ended) questions. Qualitative prompts asked participants about wishes, goals, and how they define quality of life (QOL) and were coded by six trained raters. Quantitative questions included 29 closed-ended goal-delineation items from the QOL Appraisal Profilev2. These data were analyzed using multivariate models adjusting for propensity scores (demographic differences) and testing for the effects of role (sibling or comparison), age, and role-by-age interactions. Results The study sample of DMD sibling (n = 349) and comparison (n = 619) participants provided open-ended data on 968 wishes statements, 390 QOL-definition statements, and 328 goals statements. Inter-rater reliability (kappa = 0.77) reflected good agreement between raters. Results of both open-ended and closed-ended data, and of both unadjusted and adjusted analyses suggested that DMD siblings, with age, were more focused on DMD-related, family/community, intimacy, and work concerns than their peers. They were less focused on improving mood, independence, pragmatics, or subtle fine-tuning of problem-solving in life. In contrast, the comparison group was more focused on goals related to growth, purpose, and reflection. Some group differences were amplified amongst older siblings. Conclusion This is the first study to evaluate DMD siblings’ aspirations in comparison to their peers. While there were many similarities between groups, the differences in aspirations between DMD siblings and their peers encompassed not just DMD, family/community, and intimacy, but also more work concerns. Directions for future quantitative and qualitative research are discussed.

“…The BAI is an idiometric measure, meaning it assesses context- and circumstance-specific patient experience and meaning [ 35 ]. While data reduction techniques like principal components analysis have been used in some past research with the BAI (e.g., [ 44 , 45 ]), the component structure varies across participant population [ 35 ]. This variability is to be expected for an idiometric measure [ 35 ], but also makes it difficult to compare results across studies.…”

Section: Methodsmentioning

confidence: 99%

Appraisal and patient-reported outcomes following total hip arthroplasty: a longitudinal cohort study

Rapkin

Sniderman

et al. 2022

Self Cite

Background Total hip arthroplasty (THA) is a successful procedure that provides pain relief, restores function, and improves quality of life (QOL) for patients with advanced arthritis in their hip joint. To date, little research has examined the role of cognitive appraisal processes in THA outcomes. This study examined the role of cognitive appraisal processes in THA outcomes in the first year post-surgery. Methods This longitudinal cohort study collected data at pre-surgery, 6 weeks post-surgery, 3 months post-surgery, and 12 months post-surgery. Adults (n = 189) with a primary diagnosis of osteoarthritis were consecutively recruited from an active THA practice at a Canadian academic teaching hospital. Measures included the Hip Disability and Osteoarthritis Outcome Score (HOOS), the Mental Component Score (MCS) of the Rand-36, and the Brief Appraisal Inventory (BAI). Analysis of Variance examined the association between BAI items and the HOOS or MCS scores. Random effects models investigated appraisal main effects and appraisal-by-time interactions for selected BAI items. Results HOOS showed great improvement over the first 12 months after THA, and was mitigated by three appraisal processes in particular: focusing on problems with healthcare or living situation, and preparing one’s family for health changes. MCS was stable and low over time, and the following appraisal processes were implicated by very large effect sizes: not comparing themselves to healthier people, focusing on money problems, preparing their family for their health changes, or trying to shed responsibilities. Conclusions Appraisal processes are relevant to health outcomes after THA, with different processes coming into play at different points in the recovery trajectory.