Family adaptation in families of individuals with Down syndrome from 12 countries.

Our current understanding of adaptation in families of individuals with Down syndrome (DS) is based primarily on findings from studies focused on participants from a single country. Guided by the Resiliency Model of Family Stress, Adjustment, and Adaptation, the purpose of this cross-country investigation, which is part of a larger, mixed methods study, was twofold: (1) to compare family adaptation in 12 countries, and (2) to examine the relationships between family variables and family adaptation. The focus of this study is data collected in the 12 countries where at least 30 parents completed the survey. Descriptive statistics were generated, and mean family adaptation was modeled in terms of each predictor independently, controlling for an effect on covariates. A parsimonious composite model for mean family adaptation was adaptively generated. While there were cross-country differences, standardized family adaptation mean scores fell within the average range for all 12 countries. Key components of the guiding framework (i.e., family demands, family appraisal, family resources, and family problem-solving communication) were important predictors of family adaptation. More cross-country studies, as well as longitudinal studies, are needed to fully understand how culture and social determinants of health influence family adaptation in families of individuals with DS.

Participants' perceptions support the coexistence of benefits and burdens of cancer clinical trial participation.

BACKGROUND: To advance oncology treatment for adults, comprehensive understanding of how and why people decide to enroll in, remain in, and withdraw from cancer clinical trials is needed. While quantitative findings provide insights into these benefits and burdens, they provide limited understanding of how adults with cancer appraise their situation and approach decisions to undertake a clinical trial. The goal of this mixed methods analysis was to conceptualize participants' assessment of benefits and burdens related to cancer clinical trial participation. MATERIALS AND METHODS: This sub-group analysis of 21 participants was part of a larger sequential, explanatory mixed methods study. We used Creamer's integrated approach to linking quantitative and qualitative data to assess convergence, with qualitative data explaining quantitative results. Participants were grouped into four categories based on quantitative benefit/burden scores and thematic analysis of their qualitative data was used to describe these categories. RESULTS: Across groups participants varied in descriptions of benefits and burdens of cancer clinical trial participation and reasons for participating. Those reporting high benefit/low burden described "seizing the opportunity to participate;" those reporting low benefit/low burden described "taking responsibility" through trial participation; those reporting low benefit/high burden described how they were "willing to endure," and those with high benefit/high burden emphasized "deciding to act." CONCLUSIONS: Participants' qualitative descriptions of benefits and burdens were more nuanced and dynamic than reflected in their quantitative ratings. Thus, current measures may be missing important concepts, such as logistic challenges of trial participation. Our results have implications for consenting procedures and decisional support guidance offered to patients and their caregivers.

Building Family Interventions for Scalability and Impact.

Family nursing researchers are charged with addressing the conceptual and methodological underpinnings of family research when developing family-focused interventions. Step-by-step guidance is needed that integrates current science of intervention development with family science and helps researchers progress from foundational work to experimental work with policy integration. The purpose of this manuscript is to provide pragmatic, evidence-based guidance for advancing family intervention research from foundational work through efficacy testing. Guidance regarding the development of family interventions is presented using the first three of Sidani's five-stage method: (a) foundational work to understand the problem targeted for change; (b) intervention development and assessment of acceptability and feasibility; and (c) efficacy testing. Each stage of family intervention development is described in terms of process, design considerations, and policy and practice implications. Examples are included to emphasize the family lens. This manuscript provides guidance to family scientists for intervention development and implementation to advance family nursing science and inform policy.

Advancing family science and health equity through the 2022-2026 National Institute of Nursing Research strategic plan.

BACKGROUND: Consistent with the National Institute of Nursing Research's mission of leading nursing research to address current health challenges, the new Strategic Plan identifies five research lenses: health equity, social determinants of health, population and community health, prevention and health promotion, and systems and models of care. Family research, central to nursing research and practice, is the cornerstone of social ecology and represents a critical intersection of social and structural determinants of health. PURPOSE: We argue why family health is essential to the 2022-2026 Strategic Plan and how the lenses can strengthen family research. METHODS: Drawing from collective expertise and existing literature in family research, sociology, psychology, and nursing science, the authors present a new conceptual model that integrates structural racism and heteropatriarchy to examine the salience of family structure statuses for family outcomes and discuss approaches to research design, empirical measurement, and interpretation in order to bring this new model into practice. DISCUSSION: The NINR Strategic Plan has the potential to dismantle structures that perpetuate racism and health inequity within and across family structures. An underaddressed research area under the new Strategic Plan relates to how social determinants of health influence and are influenced by families. CONCLUSION: We challenge all investigators, not just family scientists, to expand the scope of their research to conceptualize the role of family on health inequities.

A Pragmatic Guide to Qualitative Analysis for Pediatric Researchers.

OBJECTIVE: To describe four approaches to qualitative analysis in order to provide a pragmatic, application-oriented guide to their use in research focused on children and their families. METHODS: Four commonly used approaches to qualitative analysis-framework analysis, rapid qualitative analysis, content analysis, and reflexive thematic analysis-are described and compared, including their analytic strategies, tips for use, terminology, and application to a hypothetical example. RESULTS: A pragmatic guide to each method is provided as well as examples of how each analytic approach could be utilized to analyze the same dataset. CONCLUSIONS: A variety of approaches to qualitative analysis are available to both novice and experienced qualitative researchers. The approach selected from the options presented in this article will depend on numerous factors, such as the clinical problem being explored, research context, aims, objectives, research questions, and resources available such as time and funds, and the qualitative expertise of the team.

Family members' experience of well-being as racial/ethnic minorities raising a child with a neurodevelopmental disorder: A qualitative meta-synthesis.

Raising a child with a neurodevelopmental disorder has often been associated with poorer quality of life and family functioning. Yet, many family members describe themselves as resilient and capable of achieving well-being. Whether and how this occurs in racial/ethnic minority families remains largely unexplored. The aim of this study was to systematically synthesize qualitative studies exploring how families from a racial/ethnic minority background in the United States (1) experienced well-being and (2) responded to challenges they faced while caring for a child diagnosed with three selected neurodevelopmental disorders: autism spectrum disorder, attention deficit hyperactivity disorder, and intellectual disability. A systematic literature search was conducted in November and December of 2019 and updated in October 2021. Three themes were developed based on included studies: "moving toward well-being as a caregiver," "family and culture: impact on well-being," and "community and culture: impact on well-being." The findings in this review indicate that to develop well-being, racial/ethnic minority families faced additional barriers, including racial/ethnic discrimination and stigma within their family and cultural community. The knowledge generated has the potential to identify areas of intervention to promote resilience and well-being in racial/ethnic minority families raising a child with a neurodevelopmental disorder.

Tracing the Use of the Family Management Framework and Measure: A Scoping Review.

This article reports the results of a scoping review of research applications of the Family Management Style Framework (FMSF) and the Family Management Measure (FaMM). We identified 32 studies based on the FMSF and 41 studies in which the FaMM was used, 17 of which were based on the FMSF. Both the framework and measure have been used by investigators in multiple countries, with most applications of the FaMM outside the United States. Although the FMSF and FaMM were originally developed for use with families in which there was a child with a chronic physical condition, both have been applied to a broader range of health conditions and to studies focusing on families with an adult member facing a health challenge. Based on our findings, we make recommendations for how researchers can more fully address all aspects of the FMSF.

Measurement of Family Management in Families of Individuals With Down Syndrome: A Cross-Cultural Investigation.

Down syndrome (DS) is the most common genetic cause of intellectual disability worldwide. The purpose of this analysis was to determine the internal consistency reliability of eight language versions of the Family Management Measure (FaMM) and compare family management of DS across cultures. A total of 2,740 parents of individuals with DS from 11 countries completed the FaMM. The analysis provided evidence of internal consistency reliability exceeding .70 for four of six FaMM scales for the entire sample. Across countries, there was a pattern of positive family management. Cross-cultural comparisons revealed parents from Brazil, Spain, and the United States had the most positive family management and respondents from Ireland, Italy, Japan, and Korea had the least positive. The rankings were mixed for the four remaining countries. These findings provide evidence of overall strong internal consistency reliability of the FaMM. More cross-cultural research is needed to understand how social determinants of health influence family management in families of individuals with DS.

The experience of adults with sickle cell disease and their HLA-matched adult sibling donors after allogeneic hematopoietic stem cell transplantation.

AIM: To provide a rich description and in-depth understanding of the recipient-donor allogeneic hematopoietic stem cell transplantation experience. BACKGROUND: A stem cell transplant has a high likelihood of improving symptoms of sickle cell disease in adults. While studies have reported the transplant experiences of recipients and donors with haematological malignancies, no published reports have examined the experience of both adult recipients with sickle cell disease and their donors. DESIGN: Exploratory qualitative descriptive analysis. METHODS: We conducted individual interviews with 13 recipients and donors (eight males, five females) representing five recipient-donor dyads and one recipient-donor triad from one Midwest transplant centre between August 2017-February 2018. Interviews were digitally audio-recorded, transcribed verbatim and analysed using conventional content analysis. FINDINGS: Five themes were identified: the downward spiral and a second chance; getting the monster off my back; difficult and manageable; it was worth it; and relating to the healthcare team. CONCLUSIONS: The results provide a description and insights into the complex nature of the stem cell transplant experience in sickle cell disease from the perspectives of both recipients and donors. IMPACT: Health provider awareness of recipient-donor experiences can contribute to family-centred care that supports the health and quality of life for both recipients and donors. This understanding promotes high quality clinical care and improved communications by taking into account the knowledge, values and informed preferences of recipients and donors and contributes to improved decision-making and clinical care. Future research can assess family experiences that support informed choice for potential transplant candidates.

Understanding familial response to parental advanced cancer using the family management style framework.

Purpose: A parental advanced cancer diagnosis can have profound and lasting impact on family functioning. We used an adapted version of the Family Management Style Framework (FMSF) to identify and describe patterns of family management in parental advanced cancer. Design: This is a secondary analysis of data from a cross-sectional, mixed-methods study. Forty-two adults participated in semi-structured, in-depth interviews about their experiences as a parent with advanced cancer and completed measures of psychosocial functioning. We analyzed interviews using codes derived from the FMSF and used directed content analysis to identify themes reflected in the coded data. Findings: We identified five distinct patterns of family management in parental advanced cancer: (1) Equipped and Optimistic; (2) Equipped and Pragmatic; (3) Discouraged and Struggling; (4) Apprehensive and Passive; and (5) Discouraged and Conflicted. Conclusions: The FMSF was a useful framework for differentiating and understanding underlying patterns of family response to parental advanced cancer.

Family management of childhood chronic conditions: Does it make a difference if the child has an intellectual disability?

The purpose of this analysis was to assess the applicability of the Family Management Measure (FaMM) to families in which there was a child with an intellectual disability versus a chronic condition. Drawing on data from 571 parents of children with a chronic physical condition and 539 parents of children with Down syndrome, we compared the two groups across the six FaMM scales. After accounting for the covariate effects of race, ethnicity, family income, and child age, we found significant differences in four of the six FaMM scales, with parents of children with Down syndrome reporting a significantly more positive view on the Condition Management Effort and View of Condition Impact scales and a significantly less positive view on the Child's Daily Life and Condition Management Ability scales than parents of children with a chronic physical condition. There were no significant differences between groups on the Family Life Difficulty and the Parental Mutuality scales. The analysis provided evidence of the applicability of the FaMM for studying families in which there is a child with Down syndrome and its utility in identifying the common and unique challenges of family management between the groups.

Concept Analysis and the Advance of Nursing Knowledge: State of the Science.

PURPOSE: Despite an overwhelming increase in the number of concept analyses published since the early 1970s, there are significant limitations to the impact of this work in promoting progress in nursing science. DESIGN: We conducted an extensive review of concept analyses published between 1972 and 2017 to identify patterns in analysis and followed this with exploration of an exemplar related to the concept of normalization to demonstrate the capabilities of analysis for promoting concept development and progress. METHODS: Scoping review of peer-reviewed literature published in the Cumulative Index to Nursing and Allied Health Literature (CINAHL) in which the terms "concept analysis," "concept clarification," and "concept derivation" appeared in any part of the reference. The original search returned 3,489 articles. This initial pool was refined to a final sample of 958 articles published in 223 journals and addressing 604 concepts. A review of citations of the original analysis of the concept of normalization resulted in 75 articles selected for closer examination of the process of concept development. FINDINGS: Review showed a clear pattern of repetition of analysis of the same concept, growth in number of published analyses, preponderance of first authors with master's degrees, and 43 distinct descriptions of methods. Review of the 75 citations to the normalization analysis identified multiple ways concept analysis can inform subsequent research and theory development. CONCLUSIONS: Conceptual work needs to move beyond the level of "concept analysis" involving clear linkage to the resolution of problems in the discipline. Conceptual work is an important component of progress in the knowledge base of a discipline, and more effective use of concept development activities are needed to maximize the potential of this important work. It is important to the discipline that we facilitate progress in nursing science on a theoretical and conceptual level as a part of cohesive and systematic development of the discipline. CLINICAL RELEVANCE: The absence of effective concepts impedes the ability to recognize, discuss, define, and conduct studies important to clinical practice and research. This article reflects the pressing need as well as the potential for concept analysis work to be approached in a way that promotes nursing science and enables conceptually sound research to improve clinical care.

Family-Centered Care From the Perspective of Parents of Children Cared for in a Pediatric Intensive Care Unit: An Integrative Review.

PROBLEM: The Institute for Patient- and Family-Centered Care's (IPFCC) definition of family-centered care (FCC) includes the following four core concepts: respect and dignity, information sharing, participation, and collaboration. To date, research has focused on the provider experience of FCC in the PICU; little is known about how parents of children hospitalized in the pediatric intensive care unit (PICU) experience FCC. ELIGIBILITY CRITERIA: Articles were included if they were published between 2006 and 2016, included qualitative, quantitative, or mixed methods results, related to care received in a PICU, and included results that were from a parent perspective. SAMPLE: 49 articles from 44 studies were included in this review; 32 used qualitative/mixed methods and 17 used quantitative designs. RESULTS: The concepts of respect and dignity, information sharing, and participation were well represented in the literature, as parents reported having both met and unmet needs in relation to FCC. While not explicitly defined in the IPFCC core concepts, parents frequently reported on the environment of care and its impact on their FCC experience. CONCLUSIONS: As evidenced by this synthesis, parents of critically ill children report both positive and negative FCC experiences relating to the core concepts outlined by the IPFCC. IMPLICATIONS: There is a need for better understanding of how parents perceive their involvement in the care of their critically ill child, additionally; the IPFCC core concepts should be refined to explicitly include the importance of the environment of care.

Incorporating nonlinearity into mediation analyses.

BACKGROUND: Mediation is an important issue considered in the behavioral, medical, and social sciences. It addresses situations where the effect of a predictor variable X on an outcome variable Y is explained to some extent by an intervening, mediator variable M. Methods for addressing mediation have been available for some time. While these methods continue to undergo refinement, the relationships underlying mediation are commonly treated as linear in the outcome Y, the predictor X, and the mediator M. These relationships, however, can be nonlinear. Methods are needed for assessing when mediation relationships can be treated as linear and for estimating them when they are nonlinear. METHODS: Existing adaptive regression methods based on fractional polynomials are extended here to address nonlinearity in mediation relationships, but assuming those relationships are monotonic as would be consistent with theories about directionality of such relationships. RESULTS: Example monotonic mediation analyses are provided assessing linear and monotonic mediation of the effect of family functioning (X) on a child's adaptation (Y) to a chronic condition by the difficulty (M) for the family in managing the child's condition. Example moderated monotonic mediation and simulation analyses are also presented. CONCLUSIONS: Adaptive methods provide an effective way to incorporate possibly nonlinear monotonicity into mediation relationships.

Pilot Randomized Trial of Active Music Engagement Intervention Parent Delivery for Young Children With Cancer.

Objectives: To examine the feasibility/acceptability of a parent-delivered Active Music Engagement (AME + P) intervention for young children with cancer and their parents. Secondary aim to explore changes in AME + P child emotional distress (facial affect) and parent emotional distress (mood; traumatic stress symptoms) relative to controls. Methods: A pilot two-group randomized trial was conducted with parents/children (ages 3-8 years) receiving AME + P ( n = 9) or attention control ( n = 7). Feasibility of parent delivery was assessed using a delivery checklist and child engagement; acceptability through parent interviews; preliminary outcomes at baseline, postintervention, 30 days postintervention. Results: Parent delivery was feasible, as they successfully delivered AME activities, but interviews indicated parent delivery was not acceptable to parents. Emotional distress was lower for AME + P children, but parents derived no benefit. Conclusions: Despite child benefit, findings do not support parent delivery of AME + P.

Feeding problems in infancy and early childhood: evolutionary concept analysis.

AIM: The aim of this study was to report an analysis of the concept of pediatric feeding problems. BACKGROUND: Reviews of the literature on pediatric feeding problems and disorders repeatedly reference the lack of a shared conceptualization of feeding problems. It is difficult to track aetiology, prevalence and incidence of a phenomenon when available definitions and diagnoses lack practical utility. DESIGN: An evolutionary concept analysis. DATA SOURCES: A search was conducted in October 2014 of Google Scholar, CINAHL, PubMed and Web of Science databases, with MeSH terms and key words including: failure to thrive, feeding disorder/difficulty/problems, infantile anorexia, oral aversion, mealtime behaviour and dysphagia. Inclusion criteria were: subject of feeding problems, index children 0-10 years of age, English language and full text. METHODS: The articles (n = 266) were sorted into disciplines of authorship, including Psychology, Medicine, Nursing, Nutrition, Occupational Therapy, Speech Language Pathology or Other. The sample was divided into a historical sample (n = 42) for pre-2000 articles and current for those published post-2000. The current sample was later reduced to 100 and coded for surrogate terms, related concepts, attributes, antecedents and consequences. RESULTS: The historical view of pediatric feeding problems shows a tradition of mother blame or parental culpability, both direct and indirect. Currently, there exist many different definitions and typologies, but none have sound validity or generalizability. Areas of attribute consensus across disciplines are problematic feeding behaviours and selective or restrictive intake. CONCLUSION: A spectrum conceptualization of feeding problems is suggested for further development, with attributes that would be critical to have a feeding problem.

Adaptive Modeling: An Approach for Incorporating Nonlinearity in Regression Analyses.

Although regression relationships commonly are treated as linear, this often is not the case. An adaptive approach is described for identifying nonlinear relationships based on power transforms of predictor (or independent) variables and for assessing whether or not relationships are distinctly nonlinear. It is also possible to model adaptively both means and variances of continuous outcome (or dependent) variables and to adaptively power transform positive-valued continuous outcomes, along with their predictors. Example analyses are provided of data from parents in a nursing study on emotional-health-related quality of life for childhood brain tumor survivors as a function of the effort to manage the survivors' condition. These analyses demonstrate that relationships, including moderation relationships, can be distinctly nonlinear, that conclusions about means can be affected by accounting for non-constant variances, and that outcome transformation along with predictor transformation can provide distinct improvements and can resolve skewness problems.© 2017 Wiley Periodicals, Inc.

A Case Study Approach to Nursing Home Placement: Smooth and Difficult Cases and Implications for Nursing.

Nursing home placement is one of the most challenging aspects of the caregiving journey. A case study approach was used to understand the experiences of caregivers during the first few months following nursing home placement. Two caregivers were selected from a larger qualitative descriptive study because their experiences exemplified smooth and difficult transitions for both themselves and their older family member. The caregivers were interviewed shortly after placement and 3 months post-placement. Four major contextual issues were identified that indicated the similarities and differences between the two cases, including (a) the caregiver's relationship with the older adult during the home caregiving time and post nursing home placement, (b) the circumstances surrounding placement, (c) support systems, and (d) continued involvement in care post-placement. Nursing home staff who understand these issues and address concerns through family-centered care can ease the transition and promote successful collaborations between staff and families.

Family Caregivers Define and Manage the Nursing Home Placement Process.

The nursing home placement process is complex and difficult for family caregivers. This qualitative descriptive study examines the experiences of caregivers involved in the management of care and placement of an older family member using the Family Management Style Framework. Ten caregivers were recruited from four nursing homes in the Midwest. The caregivers were interviewed shortly after placement and again 3 months post-placement. Results provide a unique understanding of care management and the nursing home placement process from the perspective of the primary family caregiver. Overall, there were similarities across the same types of caregiving dyads, for example, spousal and adult-children caregivers. Caregivers expressed the need to maintain the identity of their older family member, a familial responsibility for caregiving, and change in their family relationship over time. Appreciating caregivers' challenges and needs gives health care professionals a better understanding for how to provide assistance for a smoother nursing home transition.

Investigators' successful strategies for working with Institutional Review Boards.

This study was designed to identify successful strategies used by investigators for working with their Institutional Review Boards (IRBs) in conducting human subjects research. Telephone interviews were conducted with 46 investigators representing nursing, medicine, and social work. Interview transcripts were analyzed using qualitative descriptive methods. Investigators emphasized the importance of intentionally cultivating positive relationships with IRB staff and members, and managing bureaucracy. A few used evasive measures to avoid conflict with IRBs. Few successful strategies were identified for working with multiple IRBs. Although most investigators developed successful methods for working with IRBs, further research is needed on how differences in IRB culture affect human subjects protection, and on best approaches for obtaining IRB approval of multi-site studies.