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.

Trajectory of Infant Problematic Feeding Symptoms: Study Protocol.

BACKGROUND: Infants with developmental risk factors are more likely to have feeding problems and develop chronic feeding disorders. Early detection and understanding of the progression of problematic feeding and its relationship with a child's biological functioning and the family feeding environment will enhance effective symptom management and development of interventions to prevent pediatric feeding disorders. OBJECTIVES: The New Through Two (NewThru2) feeding study protocol is described. Study aims are to (a) characterize symptoms of problematic feeding and trajectories of symptoms from pre-discharge from neonatal intensive care through age 24 months; (b) determine the relationship of child biological function at discharge with symptom characteristics and trajectories; (c) describe the child's feeding environment and its relationship to symptoms from discharge through 24 months; and (d) determine the relationship between problematic feeding symptoms and growth and developmental outcomes. METHODS: NewThru2 is a prospective, longitudinal, mixed method study following over 200 infants who received care in a neonatal intensive care unit and were identified as at risk for compromised development. The study follows enrolled infants through 24 months of age. Symptoms of problematic feeding are measured pre-discharge by clinical observation and medical record review and post-discharge by parent report. Biological function is measured by medical history and cardiorespiratory and autonomic nervous system function during feeding prior to discharge. Child feeding environment is measured by strategies parents use to manage feeding, the effect of feeding on the parent and family, and the use of feeding services. A subset of parents is interviewed to achieve a contextual understanding of the family feeding environment. Child outcome measures include parent-reported feeding skills and clinician-reported growth and neurodevelopment. DISCUSSION: The results of this study will improve understanding of pediatric feeding disorders during a time of development sensitive to adequate nutrition and with infants at risk for developmental delays or impairments.

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.

Patterns of Parenting Confidence Among Infants With Medical Complexity: A Mixed-Methods Analysis.

BACKGROUND: Parenting confidence is an important factor in fostering optimal health and development of infants with medical complexity. However, our understanding of how parents of medically complex infants describe development of confidence is limited. The purpose of this mixed-methods study was to describe the nature and development of parenting confidence. METHODS: A mixed-method design was used to examine how parents described their level of confidence. Ten parents of infants with medical complexity. Quantitative measures provided patterns of confidence and qualitative data focused on parent descriptions of confidence. Parents completed online surveys at 3 time points: (1) study enrollment, (2) infant discharge from hospital, and (3) 3 months after discharge. Parents were purposively sampled, using their confidence patterns, for qualitative phone interviews. RESULTS: Our analysis of quantitative findings revealed 3 confidence patterns: (1) increasing, (2) stable, and (3) varying. Parents described their confidence as either (1) a state of being confident or (2) how they behaved in the parenting role. Parents felt both certain and uncertain in their level of confidence and described confidence as being situationally dependent. IMPLICATIONS FOR PRACTICE: Parenting confidence needs to be cultivated through encouragement and repeated exposure to parenting behaviors. Nurses are well-suited to help identify parents with low confidence to support parents so that they can develop confidence. IMPLICATIONS FOR RESEARCH: Because there is variability in parent confidence during this critical early period of life, future research should consider a larger cohort of parents that compares confidence in diverse parent groups (ie, married vs living together couples, same-sex couples, and single parents). Research should also examine effective strategies to promote confidence and associated long-term health and developmental outcomes.

Inquiry into women's pathways to diagnosis of endometriosis: A qualitative study protocol.

AIMS: This protocol describes a study aiming to: (1) describe pathways and experiences of women's symptom recognition, appraisal and management of endometriosis; and (2) identify differences in pathways and experiences among a socioeconomically and racially diverse group of women. DESIGN: Descriptive qualitative study with stratified purposeful sampling. METHODS: Data will be collected from a minimum of 24 women with provider-presumed or surgically confirmed diagnoses across two time points. The study will recruit across socioeconomic status (SES) and race/ethnicity (non-Hispanic White, non-Hispanic Black, and Hispanic/Latina) to ensure diversity of the sample. Recruitment will occur at a large public hospital in the southeastern United States (US). Participants will be interviewed using semi-structured interview guides informed by Elder and Giele's Life Course perspective approaching women as active beings in dynamic systems shaped by: 1) their location in time and space; 2) linked lives; and 3) human agency, and 4) the time of their lives. Each woman's experiences, symptoms, and contacts with health-care systems will be mapped to trace their diagnostic pathways. Coded interviews and data will undergo within- and across-case analysis to identify similarities and differences in their experiences. Institutional review board approval was obtained June 2019. DISCUSSION: The participants' diagnostic maps will enable us to distinguish the differences in pathways and experiences between and across groups. Findings will inform the development of interventions aimed at shortening the time to diagnosis. IMPACT: This will be the first study to compare pathways to diagnosis of endometriosis in a socioeconomically and racially diverse sample of US women using the life course perspective. The results from this research stand to inform future interventions aimed at helping women achieve more timely diagnoses.

Quality of life in individuals with Down syndrome aged 4 to 21 years.

BACKGROUND: To date, investigations of quality of life (QoL) in children with Down syndrome (DS) are rather limited. The purpose of this study was twofold: to examine QoL in children with DS and to explore possible differences in the QoL by age and gender. METHODS: A cross-sectional study of 211 parents of children with DS was conducted using an online survey that included a consent form, a demographic questionnaire and the English version of KidsLife, which is a measure of children's QoL. RESULTS: Our results demonstrated moderate or favourable levels of QoL except for the emotional well-being domain among children with DS. The children's QoL showed no variance by gender. However, emotional and material well-being, interpersonal relations and social inclusion varied by age. CONCLUSIONS: The current findings provide important evidence that will help healthcare, educational and social services professionals to understand the multiple aspects of QoL in children with DS and support parents in their efforts to ensure the child's QoL. The results also point to the need to develop interventions aimed at improving QoL in those areas where children with DS are at increased risk for poorer QoL.

Mothers' and fathers' views of family management and health-related quality of life for young adult survivors of childhood brain tumors.

PURPOSE: To examine associations between fathers' and mothers' appraisals of family management and physical and emotional health-related quality of life (QOL) for young adult survivors of childhood brain tumors. DESIGN: Cross-sectional. SAMPLE: 47 mothers and 39 fathers (39-67 years old); 47 survivors (18-33 years old). METHODS: Analyses evaluated relationships among family management (Survivor's Daily Life, Condition Management Ability, Condition Management Effort, Family Life Difficulty, View of Condition Impact, Parental Mutuality), quality of life, and parental role. FINDINGS: Except for Parental Mutuality, family management ratings were not significantly different for mothers and fathers, and parental views of survivors' physical and emotional QOL improved with better family management. Parental role moderated associations between physical and emotional QOL and Survivors' Daily Life and between emotional QOL and Condition Management Ability, Condition Management Effort, and View of Condition Impact. IMPLICATIONS FOR PSYCHOSOCIAL PROVIDERS: Assess and address survivor QOL through family management from multiple perspectives.

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.

Parent-Reported Contribution of Family Variables to the Quality of Life in Children with Down Syndrome: Report from an International Study.

PURPOSE: The intent of this study was to determine parents' views of the contribution of family variables to the quality of life (QoL) of children with Down Syndrome (DS). Based on prior research, we hypothesized that parents would report that family variables reflecting positive aspects of family functioning contributed to better QoL; and family variables reflecting the negative aspects of family functioning contributed to poorer QoL. DESIGN AND METHODS: A cross-sectional study was carried out. Invitation letters were sent to DS support groups. Upon agreement, the parents of the children were sent a link to an online survey that included a consent form, demographic questionnaire, and child and family measures. RESULTS: Results demonstrated the contribution of family demands to children's interpersonal relations. Family appraisals concerning the child and the family's ability predicted the children's physical and material well-being, personal development, self-determination, social inclusion, interpersonal relations, and rights. Family problem-solving served as a prominent predictor for children's physical and emotional well-being, personal development, interpersonal relations, and rights. Also, family resources predicted various aspects of children's QoL including physical, emotional, and material well-being, self-determination, social inclusion, interpersonal relations, and rights. CONCLUSION: Results confirmed the significant relationship between family and children's QoL variables. Family appraisal and family problem-solving were especially identified as significant predictors of children's QoL that can be targeted for family interventions, since the family variables are modifiable aspects of family life. PRACTICE IMPLICATIONS: Nurses can use current findings to develop interventions to enhance QoL of children with DS and families.

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.

Well siblings of children with chronic illness: A synthesis research study.

Well siblings of chronically ill children experience family disruptions that profoundly affect them. Previous research focusing on well siblings' experiences has often produced inconsistent findings, likely the result of varying study designs and samples. The purposes of this synthesis research study were twofold: (a) to assess the applicability of existing grounded theory of sibling response to a child's cancer to a wider range of childhood conditions; and (b) to refine the existing theory to reflect the experiences of the broader sample. Data for the synthesis came from a National Institute of Nursing Research-funded mixed-methods synthesis examining the intersection of childhood chronic illness and family life. The current analysis was based on well sibling results extracted from 78 research reports published between 2000 and 2014. An existing grounded theory, Creating a Tenuous Balance (CTB), was discovered at the outset of analysis and used as the primary framework for coding and synthesizing results. The focus of most studies was siblings' responses to 14 chronic conditions, with cancer being the most often studied. Results reflected siblings' perspectives of their experiences as well as perspectives of parents and the ill child. The analysis substantiated all four patterns of sibling behavior included in CTB, with the patterns of adapting to changes in personal and family life, and handling strong emotions being especially challenging aspects of the sibling experience. Moreover, the results expanded several aspects of CTB. The analysis provided evidence of the applicability of the theory to a varied group of chronic conditions and enabled us to identify important areas for developing interventions to support siblings.

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.