Background. Clinical management of families with autosomal recessive genetic disorders focuses almost exclusively on the affected family members. However, clinically unaffected members of such families may also be severely troubled by the serious illness in a family member. The purpose of this study was to explore the experiences of healthy siblings of patients with a chronic genetic disease, Fanconi Anemia (FA). Procedure. We used a qualitative, descriptive design, which consisted of in-depth, semi-structured interviews. A convenience sample of nine siblings of patients with FA was recruited from a National Cancer Institute clinical research protocol, which targets families with inherited bone marrow failure syndromes. NVivo 2.0 software facilitated qualitative content analysis of the data. Results. Siblings' rich descriptions provided novel insights into the intricate hardships of living within a family in which a rare, life-threatening, chronic genetic illness in one member is the focus of daily life. Four major themes of the sibling experience emerged from the interview data: (1) containment, (2) invisibility, (3) worry, and (4) despair. Conclusions. Our data suggest that unrecognized psychosocial issues exist for the apparently healthy siblings of patients with FA. This study explores the psychosocial consequences of living in a family with FA and one of only a few studies to explore the sibling experience of chronic illness using a contemporaneous approach. These findings support the need for an increased awareness among health care providers; future hypothesis driven investigation, and improved assessment of problems with potential psychological morbidity.

Hip fracture in older adults may be the sentinel event leading to functional decline, long-term disability, and death. For the substantial number of older persons who fracture a hip each year, the chances of full functional recovery remains relatively low. The purpose of this study was to examine the differences between older persons with hip fracture who recover fully and those individuals who do not fully recover. A chronic disease trajectory framework guided the theoretical design of the research. Data were collected from the medical records of 102 persons aged 60 years and older who sustained a proximal hip fracture from 1993 to 1998 at a medical center in North Carolina. Data included personal characteristics, prefracture health status, hospital factors, rehabilitation features, and functional outcomes. The analysis consisted of a two-step hierarchical logistic regression model with the control variables of age, prefracture ambulation status, and prefracture cognitive status entered first and the variables of type of therapy program, frequency of therapy, number of therapy provider organizations, and location of therapy at 4 weeks entered second. Significance of the final model was observed, Chi-Square(7, n = 99)=43.55, p < .05. Significant individual predictors (p < .05) in the model were prefracture ambulation status (43.56 odds ratio) and cognitive status (6.44 odds ratio). Post-hoc analysis of the cases revealed a substantial lack of stability in ambulation status from three months post-fracture to the six-month and one-year follow-up. Findings support other research studies that indicate prefracture personal characteristics of older persons who fracture a hip are the most influential factors in predicting successful recovery. Linkage of the findings to the chronic disease trajectory model suggests that intervention efforts should focus on prevention, health promotion, and policies that extend the ability of health care providers to assist older persons with hip fracture in managing their conditions. / Ph. D.

Chronic obstructive pulmonary disease (COPD) signifies a significant public health challenge that is both avoidable and treatable. There was no standardized education offered to the COPD population at the practice location. The scope of the project encompassed standardizing education by developing a self-management packet for the COPD patients. The goal of this project was to examine how the development of a standardized COPD self-management packet enhances the quality of care and strategizes reducing 30-day readmissions compared to nonstandardized delivery of education. Orem's self-care theory and Bandura's self-efficacy concept were used to explain the principle of self-management, while Rosswurm and Larrabee's evidence-based practice model was used to guide practice change. The U.S. Prevention Service Task Force's level of evidence hierarchy was chosen to categorize the strengths and weaknesses of the evidence referenced for this project. Postdevelopment surveys using the Likert scale were distributed to the facility's COPD committee, and a 70% response rate of strongly agreed to all questions was achieved. There were no adverse responses, and the packet was approved unanimously. Based on the positive responses, the packet will be easily adapted and beneficial in practice. The recommendation is to pilot the packet on the medical-surgical unit and follow-up postdischarge with phone calls to ascertain patients' perspective of the packet. Utilization of the education packet will lead to positive social change by affording the stakeholders self-management awareness and positive outcome measures including reducing the COPD 30-day readmission rate, curtailing economic strains, and promoting positive patient-centered relationships.

General Problem Cattlemen of south central Utah in Sevier and Wayne County areas have been plagued for the past twenty-five or thirty years by a malady known to the cattleman as "Brisket Disease." It is a chronic disease affecting both male and female of all ages. It is characterized in advance stages by enlargement of the brisket and throat regions, loss of appetite, a general unthrifty condition and marked ascites. The seriousness of the disease is indicated by a morbidity rate varying from one to five percent of the cattle grazing these areas with almost one hundred percent mortality rate. Many of the people of these southern Utah counties depend largely on livestock for their livelihood. In order for the livestock industry of these counties to operate as an economical and profitable enterprise, a better performance of the animals on summer ranges and a decrease in the death loss must be obtained. Scope The information contained in this thesis is the result of observation and data collected from the cattle grazing on 7-mile, U. M, and Sheep Valley Ranges, during the summers of 1948 through 1953. The work reported consists of forage analyses, blood analyses, liver biopsies, gross and histopathological studies of the tissues of affected animals. Controlled experiments were carried out to note if mineral supplements would prevent the disease. The sick animals were treated on the range at the same elevation to see if they could be cured. Purpose The purpose for undertaking the study was to determine the etiology and nature of the disease as it is manifested by symptoms and pathology; and to develop methods of treatment and control.

Indiana University-Purdue University Indianapolis (IUPUI) / With an estimated 30.2 million diagnosed and undiagnosed cases among adults (and another 84 million at risk), diabetes mellitus is one of the most prevalent chronic conditions and a leading cause of mortality in the US. Diabetes self-management education and training (DSME/T) is a recognized standard of care and provides patients with the requisite knowledge and skills to properly manage the condition, improve long-term health outcomes, and reduce health care expenditures. Yet, DSME/T is greatly underutilized. Health insurance coverage that does not include benefits for DSME/T effectively imposes barriers to access for patients in need of the service. Many states have adopted laws and regulations requiring public and/or private insurers in their market to provide benefits for DSME/T; however, these requirements vary by state. It is unclear if these policies effectively improve access to DSME/T services. This dissertation seeks to rigorously assess the impact of state-mandated benefits and reimbursement provisions on access to DSME/T among adult patients with diabetes. The first analysis utilizes a unique combination of legal and programmatic data to quantify changes in the supply of DSME/T resources after the adoption of state-mandated benefits (potential access). The second analysis merges legal data with the Medical Expenditure Panel Survey (MEPS) from 2008 to 2016 to examine the impact of state mandates and reimbursement provisions on patient utilization of DSME/T (realized access). Lastly, the final analysis utilizes electronic health record data (2010-2016) from a safety net population to determine if patients’ evaluated need for DSME/T predicts the likelihood of receiving a DSME/T referral during a provider encounter (equitable access). Using novel data sources and a sophisticated policy analysis technique, this study provides a rigorous assessment of the impact of decades of state policies designed to improve access to care. / 2020-04-02

Indiana University-Purdue University Indianapolis (IUPUI) / Due to frequent utilization, high costs, high prevalence, and negative health outcomes, the care of patients managing type 2 diabetes mellitus (T2DM) remains an important focus for providers, payers, and policymakers. The challenges of care delivery, including care fragmentation, reliance on patient self-management behaviors, adherence to care management plans, and frequent medical visits are well-documented in the literature. T2DM management produces numerous clinical data points in the electronic health record (EHR) including laboratory test values and self-reported behaviors. Recency or absence of these data may limit providers’ ability to make effective treatment decisions for care management. Increasingly, the context in which these data are being generated is changing. Specifically, telehealth usage is increasing. Adoption and use of telehealth for outpatient care is part of a broader trend to provide care at-a-distance, which was further accelerated by the COVID-19 pandemic. Despite unknown implications for patients managing T2DM, providers are increasingly using telehealth tools to complement traditional disease management programs and have adapted documentation practices for virtual care settings. Evidence suggests the quality of data documented during telehealth visits differs from that which is documented during traditional in-person visits. EHR data of differential quality could have cascading negative effects on patient healthcare outcomes. The purpose of this dissertation is to examine whether and to what extent levels of EHR data quality are associated with healthcare outcomes and if EHR data quality is improved by using health information technologies. This dissertation includes three studies: 1) a cross-sectional analysis that quantifies the extent to which EHR data are timely, complete, and uniform among patients managing T2DM with and without a history of telehealth use; 2) a panel analysis to examine associations between primary care laboratory test ages (timeliness) and subsequent inpatient hospitalizations and emergency department admissions; and 3) a panel analysis to examine associations between patient portal use and EHR data timeliness.

Parkinson’s disease (PD) is one of the most common neurodegenerative diseases in the United States (Kowal, Dall, Chakrabarti, Storm, & Jain, 2013), affecting approximately 1 million Americans (American Parkinson’s Disease Association, 2020). However, there is limited research on the employment experiences of individuals with Parkinson’s disease (PD) (Koerts, Konig, Tucha, & Tucha, 2016). Informed by the capability approach (Sen, 2009) and social cognitive career theory (Lent & Hackett, 1994), this dissertation study addressed gaps in the extant literature, examining what employment options individuals with Parkinson's disease perceive as feasible after disease onset. This research explored the effects of contextual factors on employment options, including how understanding of workplace accommodations influences employment options, the role of discrimination and stigma in shaping employment options, and the extent to which individuals with Parkinson’s disease are receiving work-related decision support from health care providers. The author conducted individual semi-structured interviews with 23 adults under the age of 65 with Parkinson’s disease. Interviews were audio-recorded and transcribed. The author utilized an integrated approach to analysis, primarily employing a thematic analysis approach, as outlined by Braun and Clarke (2006). Additionally, a more interpretive analysis strategy (the Listening Guide, as outlined by Gilligan, Spencer, Weinberg & Bertsch, 2003) was integrated into the broader thematic analysis in order to deepen analysis of discrimination and stigma. Findings highlight specific facilitators and barriers to sustaining employment after the onset of PD, exploring how individuals with PD identify and gain crucial access to accommodations. This research demonstrates how a participant’s history of access to workplace accommodations influences participants’ perceived employment options, as well as the perceived effects of work on health. Participants’ narratives demonstrate how stigma and discrimination affect employment experiences and outcomes, influencing participants’ outcome expectations regarding current and future employment options. Participants reported that work-related conversations with health care providers were limited, with the data offering several explanations as to why these conversations were limited in nature. This study provides meaningful insights into the employment experiences of adults with Parkinson’s disease, with potential relevance to the experiences of adults with chronic and/or progressive diseases more broadly. These findings have important implications for social work and health care practice and education, disability policy, early intervention strategies after the onset of PD and priorities for future research. / 2024-05-31T00:00:00Z

Individuals with chronic medical conditions are at an increased risk for adverse reproductive and perinatal outcomes. However, there is limited condition-specific information on the reproductive and perinatal implications of chronic conditions and their associated treatments. In this dissertation, we focus on thyroid disorders and migraines, two relatively common chronic conditions among females of reproductive age, and three distinct reproductive outcomes, spanning from preconception to delivery. In study one, we analyzed the association between thyroid disorders and fecundability, the per-cycle probability of conception among non-contracepting couples, using data from Pregnancy Study Online (PRESTO), a preconception cohort study of pregnancy planners in the United States and Canada. We did not find an association between a diagnosis of hypothyroid, hyperthyroid, thyroid autoimmunity, or thyroid nodules/thyroid cancer and fecundability. We also found no association between thyroid medication use or non-use among individuals with a thyroid disorder and fecundability. In the second study, we focused on migraines and spontaneous abortion (SAB), which is pregnancy loss before twenty weeks gestation, also using PRESTO data. We found that a history of migraines is not independently associated with SAB risk in females, but that routine use of medication for migraines during the preconception period may be associated with an increased risk of SAB. In the third study, we focused on the association between migraines and hypertensive disorders of pregnancy (HDOP), which include gestational hypertension and preeclampsia. We used data from the Clinical Practice Research Datalink Gold, a longitudinal database of de-identified patient records from hundreds of primary care practices in the United Kingdom. We found that while migraines overall are associated with a small increase in risk of HDOP, this increase is most substantial among those with pre-pregnancy migraines that persist in the first trimester of pregnancy. Overall, we observed that while a history of diagnosed thyroid disorder or migraines ascertained via self-report during the preconception period may not be associated with the reproductive and perinatal outcomes we studied, certain subgroups of individuals with migraines may be at increased risk of SAB or HDOP. Migraine severity and persistence in the first trimester are likely important factors in determining the magnitude of this increased risk. Nuanced research into chronic conditions with a disproportionate disease burden among females of reproductive age will guide and improve reproductive health care for individuals with chronic conditions. This dissertation aims to address these gaps in the literature by exploring the relationship between two chronic conditions and three reproductive and perinatal outcomes: thyroid disorders and fecundability, migraines and spontaneous abortion, and migraines and hypertensive disorders of pregnancy. / 2023-04-20T00:00:00Z

The Canadian healthcare system, by design, has been historically oriented to delivering acute and symptom-driven care; however, the current cost of treating chronic disease has risen to an average of nearly 45% of direct costs of the national health budget. As a consequence more attention is being directed to the diagnosis and treatment of chronically ill patients who also may suffer from disabilities, illiteracy, impairment in judgment, depression, or multiple co-morbidities. This has also resulted in a new emphasis on health and disease self-management, to help patients to mitigate and manage the impacts of chronic diseases. This approach affects and involves the patient’s entire circle of care including the patient, healthcare providers, and the patient’s family and friends. This study discusses how support elements (i.e. decision support, education and training, family and community support) and sustainability elements (i.e. recreation and entertainment, rewards systems, online social networks) combined with online technological support can help to support and provide motivation for chronically ill patients to adopt self-management in a sustainable manner. The PLS (Partial Least Squares) statistical approach was used to validate a proposed SEM (Structural Equation Model) research model with data collected from 198 participants across North America without any prior exposure to our proposed system. The research model hypothesized that support and sustainability constructs have a strong positive influence on the willingness of users to adopt and use the proposed system. The model results in a very good fit for Behavioural Intention to Adopt for patients with no caregiver support (R2=0.71), and for patients with such support (R2=0.65). The results clearly validate our proposed model including a high predictive relevance for endogenous variables. This research provides useful theoretical and practical insights and understanding for design, development and promotion of chronic care self-management systems as well as the perceptions of users regarding the adoption and use of such systems. / Dissertation / Doctor of Philosophy (PhD)

It is well established that engaging in a physically active lifestyle benefits the long-term development and well-being of all children and youth. For children living with chronic medical conditions (CMCs), the benefits of participating in physical activity are critically important for achieving positive health outcomes and mitigating the risk of secondary chronic disease and more significant morbidity. Unfortunately, most children with CMCs are insufficiently active to achieve its associated benefits, often citing disease-related barriers to participation. To better understand how children with CMCs navigate their participation in physical activity, the concept of physical literacy can be applied as a helpful lens. In brief, physical literacy describes one’s physical, cognitive, and affective capacities to engage in an active lifestyle. Despite facing unique risks and barriers in active contexts, some evidence suggests that children with CMCs can still achieve or excel in their physical literacy. This suggests that those who are struggling to navigate their participation in an active lifestyle would benefit from potential support in their physical literacy development. However, efforts to intervene could be misplaced or misguided without adequate contextual knowledge about how the lived experiences of children with CMCs inform their physical literacy development. The overall purpose of this research was to comprehensively examine how physical literacy is developed among children with CMCs. Collectively, this is accomplished throughout the dissertation by contextualising, describing, and reflecting on how children with CMCs develop physical literacy. Margaret Whitehead’s conceptualisation of physical literacy was the guiding framework for this work, built on a philosophical foundation combining monism, existentialism, and phenomenology. A pragmatic approach was applied to guide the research process, employing mixed and multiple methods. This research was conducted with school-aged children, 8 to 12 years old, living with CMCs and recruited from outpatient Cardiology, Endocrinology, Hematology, Cystic Fibrosis/Respirology, and Neurology clinics at a local children’s hospital in Ottawa, Ontario. Where applicable, physical literacy was measured using the second edition of the Canadian Assessment of Physical Literacy. Qualitative methods were strategically applied and combined throughout the research program, including interviews, focus groups, open-ended questionnaires and field notes. This research was conducted during the COVID-19 pandemic, which dramatically influenced physical activity behaviours worldwide. Thus, the physical literacy development of children with CMCs could only be understood by first acknowledging the relative impact of the pandemic. The purpose of Article 1 was to evaluate and understand how the COVID-19 pandemic influenced the physical literacy development of children with CMCs during the first two years (2020-22). Compared to pre-pandemic matched pairs, the overall physical literacy levels of children with CMCs were significantly diminished during the pandemic, driven primarily by decreases in physical competence and daily behaviour. It was challenging for children with CMCs to play active games in the context of the pandemic, where they had to learn how to navigate additional constraints to prevent the spread of COVID-19. These precautions also constrained how children with CMCs developed social connections, which was cited as a reason that participants felt less motivated to engage in physical activities during the pandemic. Children with CMCs also spoke hesitantly about returning to pre-pandemic activities, as they perceived changes in their physical competence and the participation contexts would make it difficult to be engaged (i.e., mask-wearing requirements, uncertain safety rules, potentially sudden closures). Nonetheless, participants with CMCs maintained high levels of measured intrinsic motivation and confidence in physical activity. Once the pandemic context was better understood, the purpose of Article 2 was to describe the overall physical literacy development of children with CMCs by examining measured outcomes and comparing the experiences and perspectives of those with high and low levels of physical literacy. Most children with CMCs (~80%) were beginning (< 17th percentile) or progressing (<65th percentile) in their physical literacy journey when compared to normative classifications. Total physical literacy seemingly contributed to a sense of self and how participants with CMCs approached new experiences. In the affective domain, attitudes towards physical activity were generally informed by prior experiences, and the meaningfulness of physical activity was more salient than enjoyment. In their physical competence, participants with CMCs found it particularly difficult to navigate bodily self-regulation alongside complex movement patterns and understood the implications of practice for sustained participation in physical activity. Cognitively, it is promising that children with CMCs adopted general definitions of physical activity, recognising the benefits of an active lifestyle for their health, and understood the relative importance of evaluating risks in active contexts. Participants with higher scores spoke more about physical activity experiences where the affective, cognitive, physical, and behavioural domains seemingly overlapped (i.e., curiosity in active settings, valuing practice experiences, performing complex movement patterns in game contexts, etc.). Overall, findings identified multiple areas where children with CMCs may be supported in their physical literacy development, including opportunities to practice bodily self-regulation strategies and develop confidence in risk evaluation. Finally, the purpose of Article 3 was to reflect upon the acquired knowledge of physical literacy among children with CMCs by observing how it may be applied within a community-based setting. The “I Can Play Anything” multi-sport program was developed for the RA Centre, a community-based recreational facility in Ottawa, Ontario. The program was designed based on the concept of physical literacy and teaching principles drawn from various behaviour change theories, including self-determination theory. Descriptive, reflective and reflexive data were collected using a combination of interviews, focus groups, field notes and open-ended questionnaires. The developed themes represent the combined expectations, applications, and potential implications of the program for children with CMCs, which are: 1) Learning to play, 2) Inclusion supported by individualised variations, and 3) Understanding one’s need for rest. Reported perceptions and experiences within the community-based multi-sport program reflect how children with CMCs may be effectively supported in similar, active contexts. The final chapter of this dissertation integrates the results from all three articles, emphasising areas of physical literacy development which are particularly relevant to the lived experiences of children with CMCs. Salient ideas discussed throughout this dissertation were recognising and valuing skill progressions developed through practice, the influence of meaningful experiences on active behaviours, self-regulating activity intensities in active contexts, and applying a general definition of embodied participation to physical literacy. My research contributes valuable knowledge to the methodological, theoretical, and practical advancement of physical literacy research, particularly among children with CMCs. This dissertation emphasises and encourages the unique potential for collaboration between the health and recreation sectors to facilitate the ongoing physical literacy development of children living with CMCs.