A shock to the (health) system: experiences of adults with rare disorders during the first COVID-19 wave.

BACKGROUND: Before COVID-19, people with rare diseases (RD) experienced numerous disparities in quality of life and healthcare access and quality, yet little is known about the experiences of this underserved group during the pandemic. RESULTS: During the first wave of the COVID-19 pandemic in the United States, spring and summer of 2020, 759 participants representing 231 unique RDs responded to open-ended questions about the impact of the pandemic on life with a RD, healthcare access, and coping. Qualitative conventional content analysis was used to analyze responses. Identified themes represented positive and negative dimensions of change, including a shock to the (health) system, coping with uncertainty, and the value of social support while isolated. CONCLUSIONS: Limitations in healthcare access and quality were the most frequently described as impacts of COVID-19. Other major negative impacts included exacerbation of symptoms, psychological distress, and a lack of usual social support and reliable information. However, participants also noted silver linings, especially in healthcare. For some, expanded telehealth enhanced their ability to access medical and mental health providers and RD specialists. Finally, many participants hoped that, by highlighting social and health inequities faced by people with RDs and other minorities, the pandemic would prompt greater understanding and policies that could improve the quality of life of the RD community.

Barriers to mental health service use among people with disabilities during the COVID-19 pandemic.

PURPOSE/OBJECTIVE: The COVID-19 pandemic has exacerbated existing health inequities for people with disabilities (PWD), including disparities in mental health needs and service use. The present study investigated prospective predisposing, enabling, and illness-related correlates of mental health service need and use among PWD during the COVID-19 pandemic. RESEARCH METHOD/DESIGN: Data were collected online at two time points: October-December 2020 and October-December 2021. U.S. adults with disabilities completed self-report measures on demographic and disability characteristics, pandemic-related stressors (e.g., worries about COVID-19), depression, anxiety, barriers to service use, and perceived mental health needs and service use. Two logistic regressions were used to examine the effect of predisposing, enabling, psychosocial barriers, and illness-related factors on perceived mental health service need and service use. RESULTS: Perceptions of mental health service needs were significantly predicted by gender (female-identified, transgender and gender diverse [TGD]), younger age, increased depressive symptoms, and presence of a prepandemic mental health condition. Among those who reported a perceived need, mental health service use was predicted by gender (female-identified and TGD), greater income, lower frequency of anticipated provider disability bias, and presence of a prepandemic mental health condition. CONCLUSIONS/IMPLICATIONS: This study provides vital descriptive data on the pattern of mental health service utilization among PWD during the COVID-19 pandemic, a uniquely disruptive, challenging time. Findings further underscore the necessity of providing disability competency training and bias reduction interventions to mental health professionals, as anticipated provider disability bias was a key factor in nonservice use of PWD. (PsycInfo Database Record (c) 2023 APA, all rights reserved).

Disclosing the obvious: Psychosocial implications of (not) explaining facial differences.

Although the disclosure of invisible stigmatized identities has been frequently researched, little work has examined disclosure of facial differences (FD), in which stigma is often highly visible. People with FD are frequently expected by others to disclose or explain their condition. Qualitative work indicates that people with FD have a range of disclosure approaches from agentic disclosure (feeling obliged to disclose), autonomous nondisclosure (choosing not to disclose or to disclose only to certain people), and autonomous disclosure (choosing to openly disclose). The purpose of the present study was to validate these disclosure approaches in a large international sample, examine their frequency of use, and test their relationship to psychosocial outcomes using validated measures. English-speaking participants (n = 288) with 33 different types of FD completed an online survey of disclosure approaches and psychosocial outcomes (i.e. anxiety, depression, self-esteem, stigma, job satisfaction, and relationship self-concept). Participants disclosed to 59 % of the people they knew. Selective and indiscriminate disclosure were the most frequently used and recommended approaches. As predicted, autonomous disclosure was associated with more positive psychosocial outcomes than agentic (non)disclosure and autonomous nondisclosure. Findings from this study can support disclosure autonomy, and thus positive psychosocial outcomes, among people with FD.

Enacted Stigma Experiences and Identity Noticeability of LGBQ+ Women with Rare Diseases.

Lesbian, gay, bisexual, and queer (LGBQ+) people and those with rare diseases (RDs) experience considerable enacted stigma due to their sexual identity and disability/RD status. The frequency, severity, and type of enacted stigma is often influenced by identity noticeability. Although research has shown the challenging impacts of compounded oppression on multiply marginalized individuals, there has been no empirical research to date on the experiences of LGBQ+ women with RDs. This mixed methods study used survey and interview methods to explore enacted stigma experiences and identity noticeability of 29 LGBQ+ women with RDs. We found that RD visibility had a significant positive correlation to RD stigma. Additionally, age had a significant negative correlation to RD stigma and RD visibility. Common concerns for participants included healthcare stigma, sexual identity erasure, RD and/or LGBQ+ community exclusion, and heteropatriarchal expectations and norms. Increased research and support are needed for LGBQ+ women with RDs.

"If not me, who?": Awareness- and Self-Advocacy-Related Experiences of Adults With Diverse Rare Disorders.

In a two-study project, researchers used qualitative methods and inductive thematic analyses to investigate the lived awareness- and advocacy-related experiences of 27 adults with over 35 different rare diseases, disorders, or disabilities (RDs). In Study 1, participants in two focus groups described how a lack of RD awareness led to experiences with several types of stigma, complicated their expressions and disclosures of disability, and spurred them to work towards awareness. Participant priorities identified in Study 1 motivated researchers to design and conduct Study 2. In Study 2, researchers interviewed 18 RD self-advocates about their lived experiences with and ideas regarding advocacy. Their recommendations included increasing social and systemic support, education, and media and professional representation. Advocates in Study 2 also warned of potential roadblocks to self-advocacy and change, including systemic invalidation and bias, lack of access to activist spaces, and limited time and energy for advocacy. Overall, analyses exposed the complex and interwoven influences of RD awareness and advocacy.

Narrative identity among people with disabilities in the United States during the Covid-19 pandemic: The interdependent self.

This study examines narrative identity among a large, diverse sample of people with disabilities (PWDs) in the United States during the "second wave" of the Covid-19 pandemic (October-December 2020). The study relied on abductive analyses, combining a purely inductive phase of inquiry followed by two rounds of investigation that filtered inductive insights through three theoretical lenses: social-ecological theory, the theory of narrative identity, and perspectives from the interdisciplinary field of disability studies. The central result was the identification of a particular configuration of self, one that was demonstrably interdependent with both immediate interpersonal contexts and with broader cultural contexts. This interdependent self was interpreted in both positive and negative ways by PWDs. These findings invite future inquiry into commonplace conceptualizations of an independent self at the center of personality research and suggest that dominant conceptualizations of "the good life" may overly emphasize independence.

Relationship between number of disability-related conditions and stigma among U.S. adults.

Purpose/Objective Research: Stigma is a common experience for people with disabilities. Stigmatization may lead to disparities in education, employment, and health care, quality of life, and psychological distress. This study's purpose was to (a) determine the relationship between number of disability conditions and stigma for adults with disabilities; and (b) examine whether identifying as a person with a disability, noticeability of a disability or gender moderate relationship between number of disability conditions and stigma. RESEARCH METHOD/DESIGN: We conducted secondary analyses of data from the Disability Health Identity survey, originally collected in 2017 (n = 711). RESULTS: Multivariable linear regression analysis indicated the association between number of disability conditions and stigma was not significant (.567). However, the association between number of disability conditions and stigma was modified by noticeability (p = .007). The relationship between number of disability conditions and stigma was stronger for people with more noticeable disabilities. CONCLUSION/IMPLICATIONS: This research is important because it increases understanding of factors that may play a role in stigma experiences for people with disabilities. (PsycInfo Database Record (c) 2022 APA, all rights reserved).

Predicting depression and anxiety among adults with disabilities during the COVID-19 pandemic.

PURPOSE/OBJECTIVE: Emerging research has highlighted sources of magnified stress and trauma for people with disabilities during the COVID-19 pandemic, as compared to others in the general population. However, little research has examined the mental health impact of the pandemic on people with disabilities in relation to disability-related stigma, social isolation, and demographic characteristics. The present study therefore sought to identify predictors of depression and anxiety symptoms among U.S. adults with disabilities during the COVID-19 pandemic. RESEARCH METHOD/DESIGN: Data were collected online between October and December 2020. U.S. adults with disabilities (n = 441) completed self-report measures of depression, anxiety, psychosocial processes, and a range of demographic and disability characteristics. RESULTS: In our sample, 61.0% and 50.0% of participants met criteria for a probable diagnosis of major depressive disorder and generalized anxiety disorder, respectively. Participants also experienced significantly higher levels of disability-related stigma and social isolation compared to prepandemic norms. Hierarchical regression analyses identified higher social isolation, presence of chronic pain, younger age, higher disability-related stigma, and higher worries about contracting COVID-19 as significant predictors of both depression and anxiety symptoms. CONCLUSION/IMPLICATIONS: This study highlights important demographic and psychosocial predictors of mental health risks for people with disabilities in the context of COVID-19. Findings further underscore the need to attend to those at elevated risk within the disability community as rehabilitation professionals, disability organizations, and policy makers work to support people with disabilities in postpandemic recovery and create a more equitable response to ongoing and future public health crises. (PsycInfo Database Record (c) 2022 APA, all rights reserved).

Item development, internal consistency, and known-groups validity of the Self-Directed Mobility Scale.

PURPOSE: The aims of the current study include to: (1) describe the item development; and (2) begin the process of establishing the internal consistency and known-groups validity of the Self-Directed Mobility Scale. The purpose of the scale is to assess paediatric physical and occupational therapists' views towards self-directed mobility and their perceived ability and intentions to advocate for children's access to self-directed mobility. METHODS: Three individuals with expertise in kinesiology, psychology, paediatric rehabilitation, and disability studies served as the expert panel for item development. Four samples were included to determine internal consistency and known-groups validity: 350 physical therapists, 89 occupational therapists, 279 kinesiology undergraduate students, and 528 health and wellness undergraduate students. RESULTS: The internal consistency was above the acceptable level of 0.70 (range = 0.72-0.77) for all samples when two items regarding promoting other motor skills prior to powered mobility use and the temporary use of a mobility device were removed. Known-groups validity was established between all samples. CONCLUSIONS: The Self-Directed Mobility Scale appears to be a valid tool for assessing views of self-directed mobility and mobility advocacy intentions in paediatric physical and occupational therapists, as well as undergraduate students. Future work should examine the internal consistency based on study sample to ensure the (> 0.70) acceptable Cronbach's alpha level is met.Implications for rehabilitationThe Self-Directed Mobility Scale is a viable measurement tool to assess views of self-directed mobility and mobility advocacy intentions of pediatric physical and occupational therapists.In combination with other measures, the Self-Directed Mobility Scale may be used in future rehabilitation research to evaluate factors associated with provision of mobility technology to children with disabilities.

An inductive qualitative content analysis of stigma experienced by people with rare diseases.

OBJECTIVE: There are more than 6,000 known rare diseases (RDs), which are often serious, chronic, and progressive conditions. Cumulatively, having a RD is actually common, impacting an estimated 300 million people worldwide. While the stigmatization of some specific RDs has been studied, examining stigma in a large sample of many RDs allows for a broader understanding of patterns. DESIGN: We used inductive qualitative content analysis to analyze survey responses to an open-ended question about challenges of living with a RD among 384 people with 178 distinct RDs. RESULTS: We identified eight codes which were organized under the following three themes: structurally enacted, interpersonally enacted, and felt stigma. People with RDs experience structurally enacted stigma in the forms of healthcare stigma, education/workplace stigma, and an overall lack of accessibility. They also face interpersonally enacted stigma, including insufficient social support, a lack of understanding from others, and capitalist norms of productivity and self-sufficiency. Additionally, they experience felt stigma related to shame and the pressure to pass as able-bodied. CONCLUSION: Possible solutions to RD stigma include increased education about RDs for healthcare professionals, a societal shift towards prioritizing accessibility, strengthened legal protections for disabled people, and expanded disability justice-focused community organizing.

Exploring the Interaction of Disability Status and Childhood Predictors of Physical Activity and Sport Participation: An Exploratory Decision-Tree Analysis.

A secondary data analysis of 33,093 children and adolescents age 6-17 years (12% with disabilities) from a 2016-2017 National Survey of Children's Health nonrepresentative sample aimed to identify (a) unique clusters of sociodemographic characteristics and (b) the relative importance of disability status in predicting participation in daily physical activity (PA) and sports. Exploratory classification tree analyses identified hierarchical predictors of daily PA and sport participation separately. Disability status was not a primary predictor of daily PA. Instead, it emerged in the fifth level after age, sex, body mass index, and income, highlighting the dynamic intersection of disability with sociodemographic factors influencing PA levels. In comparison, disability status was a second-level predictor for sport participation, suggesting that unique factors influencing PA level are likely experienced by disabled children and adolescents. The authors employ an intersectionality lens to critically discuss implications for research in adapted PA.

Physical Activity, Medical Home, and Health Behavior Counseling Among Adolescents with Special Health Care Needs: NSCH 2016-2017.

OBJECTIVES: Aim 1 was to establish updated prevalence estimates for meeting national physical activity (PA) guidelines among adolescents with and without special healthcare needs (SHCN), 12-17 years old. To identify at-risk subgroups, our sub-aim was to compare the distribution of prevalence estimates across PA levels by SHCN subtypes, and in reference to peers without SHCN. Aim 2 was to examine the association between meeting PA guidelines, having a medical home, and receiving positive health behavior counseling in this population. METHODS: Weighted prevalence estimates for meeting the 2018 National PA Guidelines (inactive, insufficiently active, sufficiently active: guidelines met) were calculated from a secondary analysis of the National Survey of Children's Health 2016-2017 ( n = 16,171, 27% SHCN). Adjusted odds ratios and 95% CIs were estimated from logistic regression models to measure the association between PA, medical home, and postive health behavior counseling. RESULTS: Of adolescents with SHCN, 15% were reported to be meeting PA guidelines compared to 19% of peers without SHCN peers. Among adolescents with a medical home, regardless of SHCN status, those receiving positive health behavior counseling had 1.70 times the adjusted odds of meeting PA guidelines compared to peers without counseling. CONCLUSIONS FOR PRACTICE: Adolescents with and without SHCN were more likely to meet PA guidelines if they had a medical home and received positive health behavior counseling, highlighting the value of comprehensive healthcare practices for PA promotion.

Perceived Barriers Before and After a 3-Month Period of Modified Ride-On Car Use.

PURPOSE: The purpose of the study is to examine how perceived barriers change before and after a 3-month period of modified ride-on car use. METHODS: This study used a qualitative content analysis of perceived barriers. Fourteen caregivers (13 mothers; 1 grandmother) responded to a single-question, free-response survey before and after a 3-month period of modified ride-on car use. RESULTS: A total of 11 and 20 perceived barriers were reported before and after the 3-month period. Environmental barriers were the most frequently reported before and after the 3-month period. CONCLUSIONS: Pediatric physical therapists need to be aware of the potential perceived barriers that families may experience in regard to young children with disabilities using modified ride-on cars and determine strategies to support families on an individual basis.

Social support, stress, and life satisfaction among adults with rare diseases.

OBJECTIVE: The goal of this study was to examine the role of stress and four different types of social support in satisfaction with life (SWL) among adults with rare diseases (RDs). We examined whether support is directly related to SWL (main effects model) or related through moderating the inverse relationship between stress and SWL (stress-buffering hypothesis). METHOD: Data came from a cross-sectional survey of adults living in the United States diagnosed with any RD. Participants (n = 1,203) filled out measures of stress, four short-form scales of social support (emotional, informational, tangible, companionship), and SWL. Hierarchical multiple regression was used to test both the main effects model and stress-buffering hypothesis. RESULTS: Controlling for age, gender, years since diagnosis, and symptom severity, emotional support, but not informational or tangible support, positively predicted SWL. Companionship support was related to SWL over and above other factors. Stress negatively predicted SWL, but none of the types of support moderated this relationship. CONCLUSION: Results suggest companionship and emotional support are beneficial to individuals with RDs regardless of their stress levels. Intervention research should focus on ways to foster more companionship and emotional support to improve SWL in this underserved population. (PsycInfo Database Record (c) 2020 APA, all rights reserved).

Perceived Barriers of Modified Ride-On Car Use of Young Children With Disabilities: A Content Analysis.

PURPOSE: Modified ride-on cars have emerged as an early powered mobility option for young children with disabilities. The purpose of this study was to identify, extract, and synthesize perceived barriers of modified ride-on car use reported in previous studies. METHODS: This study was descriptive using a qualitative content analysis of previously published studies identified from a systematic literature search. RESULTS: Categories of perceived barriers were identified: device, environmental, child-related perceived barriers regarding health, tolerance, and abilities, and caregiver-related perceived barriers regarding physical requirements, time, and motivation. Device and environmental perceived barriers were the most reported. CONCLUSIONS: Pediatric physical therapists play a critical role in working with families to promote their self-efficacy for using the modified ride-on car and their capacity for overcoming the inherent difficulties associated with use. Most of the reported perceived barriers are modifiable, at least to some degree, with likely effects on modified ride-on car use.

Socioemotional functioning with facial paralysis: Is there a congenital or acquired advantage?

OBJECTIVE: Facial paralysis (FP) may impact emotional clarity, attachment, stigma, anxiety, and depression. The distinction between being born with it or acquiring it later may prompt developmental differences with consequences into adulthood, yet this has not been systematically studied. Two competing hypotheses were tested. First, the acquired advantage hypothesis states facial expression early in development plays a crucial role in socioemotional functioning, perhaps via facial feedback, and people with acquired FP would function better. Second, the congenital advantage hypothesis states impaired facial expression during early development promotes adaptation for socioemotional functioning, and people with congenital FP would function better. METHOD: Adults with congenital (n = 112) or acquired (n = 434) FP completed survey measures of emotional clarity, attachment security, stigma, anxiety symptoms, and depression symptoms. RESULTS: One-sample t tests showed that congenital and acquired participants had greater anxiety, depression, and stigma compared with the measures' norm data, and only acquired participants had more problems with emotional clarity and attachment than norms. ANCOVAs indicated that acquired, compared with congenital, participants had greater problems with emotional clarity, anxiety, and depression, controlling for age, gender, and FP severity. Hierarchical multiple regressions found that stigma, emotional clarity problems, younger age, insecure attachment, and acquired onset predicted anxiety and depression. CONCLUSION: People with FP, especially acquired FP, are at greater risk of socioemotional problems, yet no interventions exist. Contrary to work suggesting that facial feedback is crucial for socioemotional development, results suggest that those born with FP are better adapted than those who acquire it. (PsycINFO Database Record (c) 2020 APA, all rights reserved).

Relationship of rare disorder latent clusters to anxiety and depression symptoms.

OBJECTIVE: A single rare disorder or disease (RD) affects fewer than 200,000 Americans. Combined, RDs affect 30 million Americans. Collectively, people with RDs have higher levels of anxiety and depression compared with those with prevalent diseases. Previous attempts to study psychological distress within individual RDs or disease classification systems are limited by low power, generalizability, and predictive utility. The present study used mixture modeling to identify meaningful RD clusters associated with anxiety and depression. METHOD: One-thousand and 218 participants with 232 RDs were surveyed on RD characteristics and anxiety and depression symptoms. Mixture modeling identified symptom clusters based on RD characteristics (i.e., age of symptom onset, disease course, fatigue, pain, physical function, duration of symptoms, and visibility). RESULTS: The following six clusters were identified (most frequently represented RD in parentheses): stable (cutaneous T cell lymphoma), late onset and visible (spinocerebellar ataxia), moderate symptoms (spinocerebellar ataxia), invisible and tiring (idiopathic hypersomnia), severe symptoms (spinocerebellar ataxia), and early onset with severe symptoms (Ehlers-Danlos syndrome). There was a significant relationship between cluster membership and anxiety and depression symptoms. Clusters in order from least to most distress were: stable; late onset and visible; moderate symptoms; invisible and tiring; severe symptoms; and early onset with very severe symptoms. The last two clusters had clinically significantly more distress than the stable cluster. CONCLUSIONS: Researchers, clinicians, RD organizations, and policymakers could make wide-reaching impacts by prioritizing funding, research, and interventions for people likely to fall in a cluster at high risk for distress. (PsycINFO Database Record (c) 2020 APA, all rights reserved).

Real World Tracking of Modified Ride-On Car Usage in Young Children With Disabilities.

BACKGROUND: Go Baby Go is a community program that provides modified ride-on cars to young children with disabilities. AIMS: (1) To describe the real world modified ride-on car usage of young children with disabilities; (2) To compare subjectively reported modified ride-on car usage recorded by parents with objectively reported usage based on electronic tracking data. METHODS: 14 young children (1-3 years old) with disabilities used a modified ride-on car for three months. RESULTS: On average, parent-reported activity log data indicated that children used the modified ride-on car for 17.8 minutes per session (SD = 9.9) and 195.1 total minutes (SD = 234.8) over three months. Objective tracking data indicated 16.5 minutes per session (SD = 8.6) and 171.4 total minutes (SD = 206.1) over three months. No significant difference of modified ride-on car usage was found between parent-reported activity log data and objective tracking; yet, the mean absolute difference between tracking methods was 96 minutes (SD = 8.6) and suggests over- or under-reporting of families. Children used the modified ride-on car more in the first half compared to the second half of the three-month period (p < .05). CONCLUSIONS: This study may inform future research studies and local chapters of the Go Baby Go community program.