Caregiver Priorities Research Articles

Caregiver Health Beliefs Associated with Use of Pediatric Therapy Services Among Children with Special Health Care Needs.

(1) Explore the relationship between various sociodemographic factors and caregiver health beliefs (CHBs), and (2) examine whether these relationships were associated with the use of pediatric therapy services. We conducted a cross-sectional, secondary data analysis using the 2011 Survey of Pathways Diagnosis and Services dataset. 4,032 children ages 6-17 years had complete data on caregiver health beliefs and pediatric therapy use. Select CHBs reflected whether caregivers believe (1) problems related to their child's condition can be prevented or decreased with treatment, (2) they have the power to change their child's condition, and (3) their child's condition is a mystery. Pediatric therapy included physical therapy, occupational therapy, and speech therapy. We performed multivariate logistic regression to test the associations between CHBs and select child and family characteristics (Aim 1) and use of pediatric therapy services (Aim 2). Caregivers with lower educational attainment were more likely to believe their child's condition was a mystery and less likely to believe they had the power to change their child's condition. Use of pediatric therapy services tended to be lower among children whose caregivers believed they had the power to change their child's condition and higher among children whose caregivers believed their child's condition was a mystery. Our findings add to existing research that suggests CHBs differ across groups of caregivers. This, combined with the recognition that CHBs also likely differ from providers, underscores the importance of eliciting caregiver beliefs, values, and priorities to help ensure the provision of truly family-centered care. Pediatric therapy services support and optimize child development. Significant service use disparities exist, however, with children representing various marginalized groups being less likely to use pediatric therapy services. Little is known about the mechanisms underlying such disparities. We sought to explore the relationship between family sociodemographic factors, caregiver health beliefs, and pediatric therapy use. Our findings suggest that caregivers with fewer socioeconomic resources expressed beliefs that are consistent with limited knowledge and agency regarding their child's condition. Thus, providers must work to engage families in conversations about their child's development, elicit their knowledge and beliefs, and identify social or financial barriers to care.

Informal Family Caregiving of Patients with Diabetic Extremity Wounds: An Integrative Review.

Annually, 26 million people worldwide develop diabetic extremity wounds, adversely impacting the lives of patients and their family caregivers, who provide vital health assistance to these patients. This integrative review synthesized scientific literature of informal caregiving experiences for patients with diabetic extremity wounds. Five databases were searched for relevant English-language quantitative or qualitative research; ten studies were included in the final analysis. The caregiving experience included disrupted routines, frustration, guilt, poor health care communication, helplessness, and anxiety. Increased caregiver burden and decreased quality of life were associated with higher patient amputation level, worsening wound appearance and patient pain, poor health care communication, lack of social support and/or caregiving help, and lack of caregiver employment outside the home. Future research opportunities include further exploration of caregiver tasks and priorities, caregiving experiences in settings lacking family support structures and equitable health system access, and relationships between the caregiver experience and patient health system utilization.

Putting patient value first: Using a modified nominal group technique for the implementation of enhanced recovery after cardiac surgery recommendations

ObjectiveIn 2019, the Society for Enhanced Recovery After Cardiac Surgery (ERAS-CS) published perioperative guidelines to optimize the care of patients undergoing cardiac surgery. For centers with limited capacity, a sequential approach to the implementation of the full guidelines may be more feasible. Therefore, we aimed to explore the priority of implementation of the ERAS-CS guideline recommendations from a patient and caregiver perspective. MethodsUsing a modified nominal group technique, individuals who previously underwent cardiac surgery and their caregivers ranked ERAS-CS recommendations within 3 time points (ie, preoperative, intraoperative, and postoperative) and across 2 to 3 voting rounds. Final round rankings (median, mean and first quartile) were used to determine relative priorities. ResultsSeven individuals (5 patients and 2 caregivers) participated in the study. Patient engagement tools (2, 2.29, and 1.50), surgical site infection reduction (2, 1.67, and 1.25), and postoperative systematic delirium screening (1, 2.43, and 1.00) were the top-ranked ERAS-CS recommendations in the preoperative, intraoperative, and postoperative time points, respectively. ConclusionsExploration of patient and caregiver priorities may provide important insights to guide the healthcare team with clinical pathway development and implementation. Further study is needed to understand the impact of the integration of patient and caregiver values on effective and sustainable clinical pathway implementation.

Improved Sitting Balance in Children with Cerebral Palsy: Body Functions to Activity and Participation

AbstractThe aim of this study is to evaluate the changes in activity and participation domains of International Classification of Functioning (ICF), Disability and Health, in children with cerebral palsy (CP) who received integrated botulinum toxin A (BoNT-A) treatment with intensive rehabilitation with a treatment goal of improved sitting balance. In this prospective observational study, 29 patients with CP (mean age: 6.7 ± 3.8 years) and the Gross Motor Function Classification System (GMFCS) levels of IV and V were included. The primary outcome measures were the mean changes from baseline to posttreatment weeks 10 to 12 of the Child and Adolescent Scale of Participation (CASP) and Caregiver Priorities and Child Health Index of Life with Disabilities (CPCHILD). The modified Ashworth scale (MAS) and Tardieu scale (TS) results at 6 to 8 weeks of posttreatment were the secondary outcome measures. Statistically significant improvements in home and community participation of CASP (p < 0.001 and 0.001), CPCHILD (p < 0.01), MAS (p < 0.001), and all parameters of TS (p < 0.05) were recorded after treatment. The result of this prospective, observational study showed that the functional improvements in sitting balance by the integrated BoNT-A treatment provides positive effects on activity and participation levels of the patient and quality of life of patients and their families.

Psychometric Evaluation of the Polish Version of the Caregiver Priorities and Child Health Index of Life with Disabilities (CPCHILD).

PurposeThe assessment of the quality of life is an important element of the clinical examination of the patient. The aim of this study was translation and cross-cultural adaptation of the “Caregiver Priorities and Child Health Index of Life with Disabilities” (CPCHILD) questionnaire into Polish language, and testing of reliability and validity of the CPCHILD-PL for children with cerebral palsy (CP).Material and MethodsA Polish version of CPCHILD was created according to internationally accepted guidelines. Parents (n=77) of 51 boys/26 girls between 3 and 17 years with CP with Gross Motor Function Classification System I–V (GMFCS I–V) participated. To assess the reliability each domain and the total measure was tested for internal consistency and test-retest reliability. Convergent validity was evaluated by correlating the CPCHILD-PL with the CHAQ (Childhood Health Assessment Questionnaire) questionnaire.ResultsTest-retest reliability assessed by Spearman correlation coefficient for the final result of CPCHILD-PL and for most of domains were above 0.90. The values of Cronbach’s-α coefficient (measuring internal consistency) were high for all domains (except for domain 5: Health) and the entire CPCHILD-PL, with the range 0.88–0.96. The comparison between CPCHILD-PL and the Disability Index (DI) of the CHAQ showed a negative correlation. The higher the DI, the lower the CPCHILD result. The Spearman’s rank coefficient was −0.75.ConclusionThe Polish version for the CPCHILD for children with CP seems to be reliable and valid tool for assessing health-related quality of life from the caregiver perspective. It can be used in research and clinical practice for evaluation and comparison of health-related quality of life in children with CP in different countries.

Last-mile Delivery of Early Childhood Development Services

Background: India has the dual challenge of a high prevalence of developmental delay and disabilities in early childhood and a paradoxical underutilization of available intervention services due to limited accessibility and acceptability by their families. Early Childhood Development (ECD) services delivered by community health workers (CHWs) ensure its last-mile delivery to every household. IIts acceptability is improved by including evidence-based, culturally, and contextually sensitive approaches as is done in the International Guide for Monitoring Child Development (IGMCD). The IGMCD is a tool that monitors and supports the development of children under 3 years of age and also enables provision of early intervention services when required. The IGMCD, recognizes caregiver strengths and priorities and helps to build a rapport between caregivers and providers. Clinical Description: We describe six children and their families from Velugam, Dadra-Nagar Haveli district, who received ECD services from CHWs who used the IGMCD package. These cases highlight how the CHWs used the IGMCD package to identify developmental delays, health and psychosocial risk factors to development and provide strategies to caregivers to support their children's development. Management: The CHW used individualized strategies to promote responsive caregiving and enhance opportunities for early learning. In addition, the IGMCD package reinforces health, nutrition, and ECD-directed messages that are provided at the Anganwadi centers. Conclusion: Children and families in underserved communities can receive comprehensive ECD services through CHWs who are trained to deliver the IGMCD.

Real-life Wrist Movement Patterns Capture Motor Impairment in Individuals with Ataxia-Telangiectasia.

Sensitive motor outcome measures are needed to efficiently evaluate novel therapies for neurodegenerative diseases. Devices that can passively collect movement data in the home setting can provide continuous and ecologically valid measures of motor function. We tested the hypothesis that movement patterns extracted from continuous wrist accelerometer data capture motor impairment and disease progression in ataxia-telangiectasia. One week of continuous wrist accelerometer data were collected from 31 individuals with ataxia-telangiectasia and 27 controls aged 2–20 years old. Longitudinal wrist sensor data were collected in 14 ataxia-telangiectasia participants and 13 controls. A novel algorithm was developed to extract wrist submovements from the velocity time series. Wrist sensor features were compared with caregiver-reported motor function on the Caregiver Priorities and Child Health Index of Life with Disabilities survey and ataxia severity on the neurologist-performed Brief Ataxia Rating Scale. Submovements became smaller, slower, and less variable in ataxia-telangiectasia compared to controls. High-frequency oscillations in submovements were increased, and more variable and low-frequency oscillations were decreased and less variable in ataxia-telangiectasia. Wrist movement features correlated strongly with ataxia severity and caregiver-reported function, demonstrated high reliability, and showed significant progression over a 1-year interval. These results show that passive wrist sensor data produces interpretable and reliable measures that are sensitive to disease change, supporting their potential as ecologically valid motor biomarkers. The ability to obtain these measures from a low-cost sensor that is ubiquitous in smartwatches could help facilitate neurological care and participation in research regardless of geography and socioeconomic status.Supplementary InformationThe online version contains supplementary material available at 10.1007/s12311-022-01385-5.

Foam Splint versus Spica Cast-Early Mobilization after Hip Reconstructive Surgery in Children-Preliminary Data from a Prospective Randomized Clinical Trial.

Background: Surgical hip joint reconstruction may be the method of choice for children and adolescents with developmental dysplasia of the hip (DDH), as well as neurogenic dislocation of the hip (NDH) and Legg–Calvé–Perthes disease (LCPD). Following pelvic surgery, immobilization using a spica cast is considered to be the gold standard, despite the fact that casting may cause complications, such as hygienic problems, skin lesions, neurological deficits, and rigidity of the adjacent joints. An alternative for postoperative immobilization is a foam splint. The purpose of this randomized controlled trial was to compare spica cast and foam splint immobilization after hip reconstruction in children and adolescents with DDH, NDH, and LCPD. Methods: In a prospective randomized clinical trial, children and adolescents (age: 4–14 years), who received hip reconstructive surgery (osteotomy of the ilium and proximal femur, open reduction, soft tissue techniques) for DDH, NDH, and LCPD were included. Patient recruitment, group allocation, surgery, and aftercare were carried out in a department for orthopaedic surgery in Central Europe. Standardized questionnaires SF-36 (Short Form-36), EQ-5D (Euro Quality of Life 5D and CPCHILD (Caregiver Priorities and Child Health Index of Life with Disabilities) were gathered before, six, and twelve weeks after surgery from each patient. Group one received a spica cast and group two a foam splint for a period of six weeks postoperatively. There was no difference in surgical treatment. Results: Twenty-one out of thirty planned patients were enrolled in the study. One patient had to be excluded because of a lack of compliance. All quality of life (QOL) scores showed a significant reduction at the 6-week follow-up compared to the preoperative assessment. After twelve weeks, the scores came back close to the preoperative values. A significant reduction was seen in the spica cast group pre- vs. postoperatively for the variables CPCHILD (81% vs. 64%, p = 0.001), EQ-5d (65% vs. 45%, p = 0.014), and SF-36 (85% vs. 74%, p = 0.004). The corresponding values for the foam splint group also presented a reduction for all scores, but without statistical significance. Complications occurred in five cases. Conclusions: Recent retrospective studies suggest that foam splint immobilization after hip reconstruction surgery is a safe and feasible method, promising fewer complications compared to spica casting. The preliminary results of this prospective randomized clinical trial show an improvement of the scores when using a foam splint compared to the conventionally used spica cast. Benefits for the patients may be fewer adverse events and no need to undergo a second round of anaesthesia for recasting. Data suggest higher patient and caretaker satisfaction in the foam splint group.

Caregivers\u2019 experience of sleep management in Smith\u2013Magenis syndrome: a mixed-methods study

BackgroundSmith–Magenis syndrome (SMS) is a rare genetic syndrome associated with a unique profile of early morning waking and daytime sleepiness. Children with SMS evidence high rates of self-injury and aggression and have a preference for adult over peer attention, with strong motivation to interact with a particular caregiver. In addition, people with SMS have lower adaptive functioning skills relative to cognitive abilities and demonstrate high levels of impulsivity. Taken together, these factors may result in individuals being awake overnight requiring vigilant caregiver supervision. Despite these complexities, no study has described the strategies caregivers take to keep their children with SMS safe overnight or considered the impact of these experiences on caregivers or the wider family.MethodsThe current study used a mixed-methods approach to consider sleep management strategies and challenges for caregivers of people with SMS at different ages. Caregivers completed an international online survey about sleep management and related difficulties, use of interventions and access to services and support. Semi-structured interviews were conducted with 14 caregivers in the UK to increase understanding of caregiver experiences and priorities for change in the UK context. Interviews were transcribed verbatim and coded using thematic analysis.ResultsEvidence from the online survey (n = 40) revealed wide-ranging impacts of poor sleep on the person with SMS and the wider family. Only 5% of caregivers reported that the sleep problems had no impact on their child, and 76% reported a moderately or extremely significant impact on themselves. For some individual caregivers, sleep management difficulties improved over time whereas for others no change was reported. Weekly respite emerged as the ideal provision for 49% of caregivers, although only 14% had access to this. The majority of caregivers (54%) received no respite. Thematic analysis of qualitative interviews revealed interactions between aspects of the behavioural phenotype of SMS which may contribute to complex and unusual presentations in relation to sleep management and safety.ConclusionsCaregivers’ priorities for sleep management and support were delineated, with key implications for services in terms of the use of SMS-sensitive strategies and respite provision.

Dynamic Standing Exercise in a Novel Assistive Device Compared with Standard Care for Children with Cerebral Palsy Who Are Non-Ambulant, with Regard to Quality of Life and Cost-Effectiveness

Children with cerebral palsy who are non-ambulant cannot stand or walk independently. In Sweden, daily static standing is part of standard care for these children even though it is time-consuming and costly, and while it may benefit the well-being of many children, it is inconvenient for others. A novel stander enables dynamic standing through walking movements. The aim of this study was to compare dynamic and static standing regarding quality of life and cost-effectiveness. Twenty children with cerebral palsy who were non-ambulant were included in this randomized controlled study with a cross-over design. Mean age of the participants was 11.6 ± 3.6 years, nine were female. They participated in four months of static and four months of dynamic standing. Quality of life was assessed with the Caregiver Priorities and Child Health Index of Life with Disabilities (CPCHILD), which has been shown to be valid and reliable, and is tailored to children with severe cerebral palsy. Information about other aspects of quality of life and about cost-effectiveness was collected by study-specific questionnaires. Dynamic standing was the preferred alternative from the family perspective, as it cost less (€64, p < 0.01) and had greater benefits than standard (static standing) care. From societal and healthcare provider perspectives, dynamic standing had benefits but higher costs than static standing (€290 and €354 respectively, p < 0.01). These findings may contribute to the development of individualized standing recommendations.

Quality of Life After Deep Brain Stimulation of Pediatric Patients with Dyskinetic Cerebral Palsy: A Prospective, Single-Arm, Multicenter Study with a Subsequent Randomized Double-Blind Crossover (STIM-CP).

Patients with dyskinetic cerebral palsy are often severely impaired with limited treatment options. The effects of deep brain stimulation (DBS) are less pronounced than those in inherited dystonia but can be associated with favorable quality of life outcomes even in patients without changes in dystonia severity. The aim is to assess DBS effects in pediatric patients with pharmacorefractory dyskinetic cerebral palsy with focus on quality of life. The method used is a prospective, single-arm, multicenter study. The primary endpoint is improvement in quality of life (CPCHILD [Caregiver Priorities & Child Health Index of Life with Disabilities]) from baseline to 12months under therapeutic stimulation. The main key secondary outcomes are changes in Burke-Fahn-Marsden Dystonia Rating Scale, Dyskinesia Impairment Scale, Gross Motor Function Measure-66, Canadian Occupational Performance Measure (COPM), and Short-Form (SF)-36. After 12months, patients were randomly assigned to a blinded crossover to receive active or sham stimulation for 24 hours each. Severity of dystonia and chorea were blindly rated. Safety was assessed throughout. The trial was registered at ClinicalTrials.gov, number NCT02097693. Sixteen patients (age: 13.4 ± 2.9 years) were recruited by seven clinical sites. Primary outcome at 12-month follow-up is as follows: mean CPCHILD increased by 4.2 ± 10.4 points (95% CI [confidence interval] -1.3 to 9.7; P=0.125); among secondary outcomes: improvement in COPM performance measure of 1.1 ± 1.5 points (95% CI 0.2 to 1.9; P=0.02) and in the SF-36 physical health component by 5.1 ± 6.2 points (95% CI 0.7 to 9.6; P=0.028). Otherwise, there are no significant changes. Evidence to recommend DBS as routine treatment to improve quality of life in pediatric patients with dyskinetic cerebral palsy is not yet sufficient. Extended follow-up in larger cohorts will determine the impact of DBS further to guide treatment decisions in these often severely disabled patients. © 2021 The Authors. Movement Disorders published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society.

Policy Series: Family Caregiving Policies: Where We Are Now

This session reviews prospects for advancing family caregiving policy under the Biden Administration, by reporting on the RAISE (Recognize, Assist, Include, Support, and Engage) Family Caregivers Act, enacted in January 2018. The Act directs the Secretary of HHS to develop a national family caregiving strategy, and supports research and consensus-building activities, in collaboration with The John A. Hartford Foundation. It aims to identify actions that communities, providers, government, and others may take to recognize and support family caregivers. To this end, the Administration for Community Living (ACL) has convened an Advisory Council, comprising 15 voting members from various stakeholder groups, to guide the effort; the project also commissioned primary data collection on caregiver priorities and recommendations, using a Request for Information (RFI) in the Federal Register garnering roughly 1600 responses, 12 focus groups with diverse family caregivers, and listening sessions with stakeholder groups. Wendy Fox-Grage, of the National Academy on State Health Policy, which supports RAISE Act activities, will describe the project’s scope of work and activities to date. Pamela Nadash from the LeadingAge LTSS Center @UMass Boston, who leads the data analysis component, will present findings from the commissioned research, while Molly Evans, (MA Executive Office of Elder Affairs) will review the current state of state-level policies aimed at supporting family caregivers. The symposium will conclude with Grace Whiting, CEO of the National Alliance for Caregiving, who will present an advocate’s perspective on the status of family caregiving policy. Eileen Tell, of ET Consultants, will act as discussant.

Findings From RAISE Act Research: Family Caregiver Priorities

To understand the needs and policy priorities of family caregivers, the Advisory Council commissioned research: first, through a request for information (RFI) in the Federal Register, which garnered roughly 1600 responses. Qualitative analysis revealed that family caregivers have diverse needs spanning their financial security as well as their needs for caregiver-focused supports; recommendations were similarly diverse, including requests for caregiver pay, improved access to respite, and other major policy changes. These findings fed into 12 focus groups focusing on diverse populations of caregivers, yielding more depth around caregiver priorities. Six stakeholder listening sessions built on these results, aiming to develop concrete suggestions for a national caregiver strategy – a key outcome of the Advisory Council. Such strategies ranged from a major publicity campaign creating awareness of family caregivers, to suggestions on implementing caregiver assessments, to more ambitious goals such as improved financing for long term services and supports more broadly.

Exploring Informal Caregivers’ Priorities of Nursing Home Dementia Care From Communities of Color

Informal caregivers are critical stakeholders in nursing home (NH) care for individuals with dementia. Given racial and ethnic disparities in United States NHs, there is a need to understand informal caregivers’ perspectives, particularly among those that identify as members of a community of color. We conducted a scoping review of informal caregiver priorities of nursing home dementia care. Included studies exclusively examined priorities of informal caregivers identifying as Black, Indigenous, or people of color. The final sample (n=12) included two United States studies representing African American and Korean informal caregivers. The remaining studies were conducted in other countries. Informal caregivers expressed a desire for professional support during the nursing home transition, increased staff knowledge of dementia, and improved resident engagement. These findings highlight the paucity of informal caregivers identifying as Black, Indigenous, or people of color represented in US nursing home dementia research. Future efforts must include communities of color.

Patient, Caregiver, and Healthcare Provider Priorities for Future Multiple Myeloma Research: Results of a James Lind Alliance Priority Setting Partnership

Multiple myeloma, also referred to simply as myeloma, is an incurable cancer of the plasma cells. Every year, more than 3,000 Canadians are diagnosed with myeloma and more than 1,000 Canadians die from this disease. As a result of recent advances in treatment, people are living longer with myeloma. However, as the disease has yet no cure, further research is needed to continuously improve the diagnosis, treatment, and management of myeloma, and to provide a better quality of life for those living with this disease.The first project of its kind for myeloma, the Canadian Myeloma Priority Setting Partnership (PSP) was established in 2019 to identify research priorities shared by people with myeloma, caregivers and healthcare providers. The Canadian Myeloma PSP followed the rigorous methodology developed by the James Lind Alliance (JLA), a UK-based, non-profit initiative that seeks to reduce the gap that exists between funded research and the needs of the community. The project was generously funded and supported by Myeloma Canada, a national charitable organization exclusively devoted to improving the lives of Canadians impacted by myeloma.Following the JLA guidelines, we convened a national steering committee whose members consisted of people with myeloma, caregivers and healthcare providers; together, they guided the iterative priority setting process. The first phase of the process involved the development of a survey used to elicit unanswered research questions, also known as “uncertainties,” relating to four wide-ranging topics: diagnosis, treatment, management, and living well with myeloma. The survey generated 3,042 uncertainties from 594 participants from across Canada.In the second phase of the process, the uncertainties were processed and verified according to the JLA guidelines to produce 76 indicative questions. These questions were subsequently reviewed to determine whether they fell within the project scope, and then compared against all available systematic reviews and clinical practice guidelines on myeloma published within the previous ten years (2010-2020). This process yielded 45 indicative questions that were both within the scope of the project and unanswered by previous research.The third phase involved an interim prioritization survey which asked participants to select and rank their top 10 questions for future myeloma research from among the list of 45 indicative questions. A total of 651 participants from across the country completed the second survey and the results were used to create a shortlist of 18 questions. The shortlist was then submitted for discussion at a consensus-building or prioritization workshop.The prioritization workshop, the final phase of the PSP process, was held in April 2021. The online event was attended by 23 participants (people living with myeloma, caregivers and healthcare providers), while four JLA Advisors facilitated the discussions using nominal group techniques.This event produced a ranked list of the top 10 research priorities for future research on myeloma (see Table 1).These priorities will be disseminated as widely as possible among researchers, clinicians, research funders and the broader myeloma community. The goal is not only to inform on, but also to solicit funding for future research directions that are meaningful to those directly affected by the disease. [Display omitted] DisclosuresReiman: Myeloma Canada, Canadian Institutes of Health Research, New Brunswick Health Research Foundation, Canadian Cancer Society, Terry Fox Research Institute, AstraZeneca, Roche, Pfizer, Amgen, BristolMyersSquibb,: Research Funding; Myeloma Canada: Membership on an entity's Board of Directors or advisory committees; Compositions and Methods for Inhibiting Blood Cancer Cell Growth. Canadian Patent Pending 176000 (2017-10-20) Peptides for the Treatment of Resorptive Bone Disease. Murugesan A and Reiman T. United States Provisional Patent; 62/249,471 (2015-11-02). Cance: Patents & Royalties. Reece: Amgen: Consultancy, Honoraria; Janssen: Consultancy, Honoraria, Research Funding; Takeda: Consultancy, Honoraria, Research Funding; Celgene: Consultancy, Honoraria, Research Funding; Millennium: Research Funding; Karyopharm: Consultancy, Research Funding; BMS: Honoraria, Research Funding; GSK: Honoraria; Sanofi: Honoraria. Song: Celgene: Research Funding; Celgene, Janssen, Amgen, Takeda: Honoraria.

Validation of the Gait Outcomes Assessment List questionnaire and caregiver priorities for individuals with cerebral palsy.

To expand upon previous validation of the Gait Outcomes Assessment List (GOAL) questionnaire in individuals with cerebral palsy (CP), to rank items by importance, and to summarize written-in (free text) goals. For this cross-sectional study, the parent-version 5.0 of the GOAL was completed by 310 consecutive caregivers of 310 individuals aged 3 to 25 years with CP (189 males, 121 females; mean [SD] age: 10y [4y 2mo]; Gross Motor Function Classification System [GMFCS] levels I-IV) concurrent with a gait analysis. Distribution properties and validity were quantified using questionnaires, kinematics, and oxygen consumption. Items classified as at least 'difficult' to perform and 'very important' to improve were considered caregiver priorities and rank ordered. Free text goals were categorized. Results were summarized for everyone and by GMFCS level. Most scores were normally distributed. Validity was acceptable, with concurrent greater than construct validity. Among all 310 caregivers, fatigue was the highest priority, followed by gait pattern and appearance items. The rank of priorities varied by GMFCS level. Common free text goals included toileting independently as well as improved fine motor and ball sport skills. The GOAL is a valid tool that can help prioritize goals across GMFCS levels I to IV. Identifying the top goals may improve shared decision-making and prioritize research for this sample.

Patient and Caregiver Prioritization of Palliative and End-of-Life Cancer Care Quality Measures.

Development and prioritization of quality measures typically relies on experts in clinical medicine, but patients and their caregivers may have different perspectives on quality measurement priorities. To inform priorities for health system implementation of palliative cancer and end-of-life care quality measures by eliciting perspectives of patients and caregivers. Using modified RAND-UCLA Appropriateness Panel methods and materials tailored for knowledgeable lay participants, we convened a panel to rate cancer palliative care process quality measure concepts before and after a 1-day, in-person meeting. Nine patients and caregivers with experience living with or caring for patients with cancer. Panelists rated each concept on importance for providing patient- and family-centered care on a nine-point scale and each panelist nominated five highest priority measure concepts ("top 5"). Cancer patient and caregiver panelists rated all measure concepts presented as highly important to patient- and family- centered care (median rating ≥ 7) in pre-panel (mean rating range, 6.9-8.8) and post-panel ratings (mean rating range, 7.2-8.9). Forced choice nominations of the "top 5" helped distinguish similarly rated measure concepts. Measure concepts nominated into the "top 5" by three or more panelists included two measure concepts of communication (goals of care discussions and discussion of prognosis), one measure concept on providing comprehensive assessments of patients, and three on symptoms including pain management plans, improvement in pain, and depression management plans. Patients and caregivers nominated one additional measure concept (pain screening) back into consideration, bringing the total number of measure concepts under consideration to 21. Input from cancer patients and caregivers helped identify quality measurement priorities for health system implementation. Forced choice nominations were useful to discriminate concepts with the highest perceived importance. Our approach serves as a model for incorporating patient and caregiver priorities in quality measure development and implementation.

Health-related quality of life and caregiver burden after hip reconstruction and spinal fusion in children with spastic cerebral palsy.

To evaluate the effect of hip reconstruction or spinal fusion on health-related quality of life (HRQoL) in non-ambulatory children with spastic cerebral palsy (CP) and caregiver burden. This was a prospective, longitudinal study of changes in HRQoL and caregiver burden over the 5 years after surgical correction of hip instability or scoliosis in children with bilateral spastic CP classified in Gross Motor Function Classification levels IV or V. Serial parent proxy measures of HRQoL and caregiver burden were obtained before and 6 weeks, and 3, 6, 9, 12, 24, and 60 months after surgery using the Caregiver Priorities and Child Health Index of Life with Disabilities and the Assessment of Caregiver Experience with Neuromuscular Disease. Scores 5 years or more after surgery were compared to pre-surgery scores using paired Student's t-tests. Serial outcome trajectories were estimated by linear mixed modeling. Of 69 participants (40 males, 29 females; mean age 11y 6mo, SD 4y 1mo, range 3y 10mo-20y 7mo), 43 had hip reconstruction and 26 had spinal fusion. Clinically significant improvements in HRQoL were detected (average increase 7.6 points) 5 years or more postoperatively, with hip reconstruction providing greater benefit. Domains improved by surgery included positioning, transferring/mobility, comfort/emotions, and health. Caregiver burden did not change as these children remain maximally dependent. Surgical treatment of hip and spine deformity improves HRQoL, especially for painful hip instability, but does not change caregiver burden.

Disease Severity and Motor Impairment Correlate With Health-Related Quality of Life in AP-4-Associated Hereditary Spastic Paraplegia.

ObjectiveAP-4-associated hereditary spastic paraplegia (AP-4-HSP) is a childhood-onset neurogenetic disease and mimic of cerebral palsy. Data on health-related quality of life (HRQoL) are lacking. To establish a metric for HRQoL and caregiver priorities, we used the Caregiver Priorities and Child Health Index of Life with Disabilities (CPCHILD) questionnaire to assess HRQoL in correlation with disease severity in 64 patients with AP-4-HSP.MethodsA cross-sectional analysis of caregiver-reported HRQoL was performed using the CPCHILD questionnaire in combination with a detailed clinical characterization.ResultsHRQoL was impaired in all domains in patients with AP-4-HSP (mean score: 59.6 ± 12.6 [SD]), with no significant difference between the 4 subtypes. Age, as a surrogate for disease duration, and Spastic Paraplegia Rating Scale scores, as an indicator for corticospinal tract dysfunction and motor impairment, correlated with lower CPCHILD scores (Pearson r = −0.31, p = 0.01 and r = −0.52, p < 0.0001, respectively). Patients with tetraplegia showed lower CPCHILD scores compared with individuals with diplegia or no spasticity. Wheelchair dependence reduced HRQoL in all domains. The presence of seizures, including medically refractory epilepsy, was not associated with lower CPCHILD scores. Standardized assessment of caregiver priorities identified several areas of high importance to HRQoL.ConclusionsWe show that the CPCHILD questionnaire, developed for use in children with cerebral palsy, can be used to assess HRQoL in patients with childhood-onset complex hereditary spastic paraplegia. HRQoL is reduced in patients with AP-4-HSP and correlates with the degree of motor impairment. These results provide a framework for medical decision making and a baseline for the future development of treatment guidelines and interventional trials.

Correlation between hand functioning, cognition and quality of life of CP children: 2-year prospective randomized study

IntroductionFor treatment of CP patients multidisciplinary approach is often promoted. But it is not cost-efficient to involve all medical staff and variety of diagnostic/intervention tools for every patient.ObjectivesHaving knowledge on pivotal manifestations of CP and what are they related to would ease multidirectional approach in practice. It will shorten a search of pathologies only to those related to main manifestations related to quality of life.Methods611 children aged 2 to 18 years (mean age–6y5m) with CP(G80.0-G80.9): examined by 3 independent doctors. 56%males, 44%females. Randomized blinded assessment. Fine hand function assessed by “9-hole peg”,“Box and Blocks” tests, dynamometry. Intellectual functioning assessment was done using Raven Matrices. Quality of Life(QoL) assessment according to Caregiver Priorities and Child Health Index of Life with Disabilities (CPCHILD). The intraclass correlation coefficient(ICC) was used for finding out discrepancies between observers. Inferencial statistics including 95%CI and P-value.ResultsICC coefficient between observers was highly reliable–0.93(95%CI:0.89–0.95). The mean QoL score for children GMFCS levels I and II was 58.5(SD 16.6), for GMFCS III, IV and V children–22.4(SD13.4). QoL was more related to fine hand functioning(r=0,344) than to cognition(r=0,295). There was a strong correlation bond between fine hand functioning and cognition (r=0,663). In case fine hand function improved positive changes in cognition were observed in 74% of subjects(p&lt;0,05). Correlation between grasp power and IQ was weak(r=0,184). Grasp power improvement was slightly related to QoL(r=0,102).ConclusionsFine hand functioning improves QoL even more that cognition.So training of fine motor skills should be given a priority in case of limited rehabilitation resources.DisclosureNo significant relationships.