Development of the Huntington Disease Family Concerns and Strategies Survey From Focus Group Data

Williams, Janet K.; Barnette, J. Jackson; Reed, David; Sousa, Valmi D.; Schutte, Debra L.; McGonigal-Kenney, Meghan; Jarmon, Lori; Phillips, Emily; Tripp–Reimer, Toni; Paulsen, Jane S.

doi:10.1891/1061-3749.18.2.83

Cited by 15 publications

(17 citation statements)

References 41 publications

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“…The design of the larger study is reported in more detail elsewhere (Sparbel et al, 2008; Williams & Ayres, 2007; Williams et al, 2007; Williams et al, 2008). In brief, the larger study was a two phase mixed methods study in which the conduct of focus groups and development of a family concerns and strategies survey were completed in the first phase, and the survey was distributed and analyzed in the second phase.…”

Section: Methodsmentioning

confidence: 99%

“…Groups extended from 1 to 2 hours, length did not appear to be related to the number of participants in the groups. Teen and adult participants were asked to describe concerns regarding themselves, concerns regarding the health of the family member with HD, strategies used to manage these concerns, and what could help them (Williams et al, 2008). …”

Section: Methodsmentioning

confidence: 99%

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Caregiving by Teens for Family Members With Huntington Disease

et al. 2009

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The purpose of this report is to describe caregiving by teens for family members with Huntington disease (HD). Thirty-two teens in HD families in the United States and Canada participated in focus groups from 2002 to 2005 in a study to identify concerns and strategies to manage concerns. An unexpected finding was 24 (77%) described caregiving activities. Descriptive analysis of caregiving statements identified themes of Tasks and Responsibilities, Subjective Burden, Caregiving in Context of Personal Risk for HD, and Decisional Responsibility. Teens took an active part in nearly all aspects of care with the exception of contacting health care providers and attending doctors’ appointments. Some described emotional distress, and many provided care knowing they had the potential to develop HD. Teens recognized the need for decisions but lacked the authority to make these decisions. Findings may be relevant for other teens who strive to meet caregiver and student roles and developmental tasks.

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Section: Methodsmentioning

confidence: 99%

Section: Methodsmentioning

confidence: 99%

Caregiving by Teens for Family Members With Huntington Disease

et al. 2009

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“…However, no measures of family members' perception of health care services for persons with Huntington Disease were found. Thus, a new measure of Community Health Care Services (CHCS) for family of persons with HD was developed and fully described elsewhere 18. (unpubl.…”

Section: Introductionmentioning

confidence: 99%

“…This instrument was developed from a larger parent study whose purpose was to identify concerns of family members of persons with HD and strategies used to manage these concerns 19. The parent study used a mixed methods design in which common themes were identified from focus groups with 91 adult family members at six sites in the US and Canada, and a survey was constructed from the themes and focus group statements 18. (unpubl.…”

Section: Introductionmentioning

confidence: 99%

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A new scale to measure family members' perception of community health care services for persons with Huntington disease

Sousa

Williams

Barnette

et al. 2010

Evaluation Clinical Practice

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Rationale, Aims, and Objectives Huntington Disease (HD) is a progressive genetic brain disease leading to disruptive cognitive, behavioral, and physical impairments. Persons with the condition and their caregivers need appropriate and accessible health care services to help them manage the disease adequately. The purpose of this study was to evaluate the psychometric properties of a new scale that measures family members' perception of community health care services (CHCS) for persons with HD. Methods A methodological design was used to examine the initial reliability and dimensionality of the CHCS scale among 245 family members of persons with a diagnosis of HD. Data analysis consisted of computing Cronbach's alpha coefficients, calculating the 95% confidence interval for alphas, and performing item-analysis and exploratory factor analysis. Results Reliability of the scale based on Cronbach's alpha (α) was .83. Factor analysis using Principal Component Analysis and Varimax Rotation suggested that three interpretable factors underlie the scale. Factor 1: HD Knowledge, had α =.82, eigenvalue of 4.67, and explained 33.42% of the variance; Factor 2: HD Community Resources, had α = .62, eigenvalue of 1.68, and explained 12.02% of the variance; and, Factor 3: Individualized HD Management, had α =.77, eigenvalue of 1.45, and explained 10.39% of the variance. Conclusions Findings from this study provide evidence of both construct validity and internal consistency reliability of the CHCS scale. Further psychometric testing of the scale in other samples of family caregivers of persons with HD is warranted.

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Mutation‐related magnetization‐transfer, not axon density, drives white matter differences in premanifest Huntington disease: Evidence from in vivo ultra‐strong gradient MRI

Casella

Chamberland

Laguna

et al. 2022

Human Brain Mapping

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White matter (WM) alterations have been observed in Huntington disease (HD) but their role in the disease‐pathophysiology remains unknown. We assessed WM changes in premanifest HD by exploiting ultra‐strong‐gradient magnetic resonance imaging (MRI). This allowed to separately quantify magnetization transfer ratio (MTR) and hindered and restricted diffusion‐weighted signal fractions, and assess how they drove WM microstructure differences between patients and controls. We used tractometry to investigate region‐specific alterations across callosal segments with well‐characterized early‐ and late‐myelinating axon populations, while brain‐wise differences were explored with tract‐based cluster analysis (TBCA). Behavioral measures were included to explore disease‐associated brain‐function relationships. We detected lower MTR in patients' callosal rostrum (tractometry: p = .03; TBCA: p = .03), but higher MTR in their splenium (tractometry: p = .02). Importantly, patients' mutation‐size and MTR were positively correlated (all p ‐values < .01), indicating that MTR alterations may directly result from the mutation. Further, MTR was higher in younger, but lower in older patients relative to controls ( p = .003), suggesting that MTR increases are detrimental later in the disease. Finally, patients showed higher restricted diffusion signal fraction (FR) from the composite hindered and restricted model of diffusion (CHARMED) in the cortico‐spinal tract ( p = .03), which correlated positively with MTR in the posterior callosum ( p = .033), potentially reflecting compensatory mechanisms. In summary, this first comprehensive, ultra‐strong gradient MRI study in HD provides novel evidence of mutation‐driven MTR alterations at the premanifest disease stage which may reflect neurodevelopmental changes in iron, myelin, or a combination of these.

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Development of the Huntington Disease Family Concerns and Strategies Survey From Focus Group Data

Cited by 15 publications

References 41 publications

Caregiving by Teens for Family Members With Huntington Disease

Caregiving by Teens for Family Members With Huntington Disease

A new scale to measure family members' perception of community health care services for persons with Huntington disease

Mutation‐related magnetization‐transfer, not axon density, drives white matter differences in premanifest Huntington disease: Evidence from in vivo ultra‐strong gradient MRI

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