Exploratory factor analysis of the Illness Intrusiveness Rating Scale for parents of children with atypical genital appearance due to differences of sex development (DSD).

OBJECTIVE: Illness intrusiveness refers to the subjective cognitive appraisal of a chronic health condition interfering in daily, valued activities and may be highly relevant for parents of children with atypical genital appearance due to differences of sex development (DSD). However, a measure of illness intrusiveness has not been validated for this population. The current study aimed to evaluate the factor structure of the Illness Intrusiveness Scale for Parents (IIS-P) and examine convergent validity. METHODS: Participants included 102 parents (Mage = 33.39 years, SD = 6.48; 58% mothers) of 65 children (<2 years old) diagnosed with DSD participating in a larger, longitudinal study. Parents completed the IIS-P as well as self-report measures of stigma, and anxious and depressive symptoms. An exploratory factor analysis (EFA) was conducted. RESULTS: EFA results supported a 1-factor intrusiveness solution (α = .93), as well as a 2-factor solution measuring intrusiveness on daily living (α = .92) and community connectedness (α = .85). The 1-factor solution and both factors of the 2-factor solution demonstrated significant convergent validity with stigma as well as anxious and depressive symptoms. CONCLUSIONS: Support emerged for both 1- and 2-factor solutions of the IIS-P in parents of children with DSD. The decision to evaluate illness intrusiveness as a total score or to examine the subscales of daily living and community connectedness should be tailored to the unique aims of researchers and clinicians. Future research should conduct a confirmatory factor analysis with both 1- and 2-factor models with larger, more diverse samples of caregivers.

The study of psychosocial outcomes of parents bereaved by pediatric illness: a scoping review of methodology and sample composition.

OBJECTIVE: Parents of children who died of a medical condition experience a range of psychosocial outcomes. The current scoping review aims to summarize the outcomes assessed, methodology, and sample characteristics of recent psychosocial research conducted with this population. METHODS: Included studies were limited to peer-reviewed, psychosocial outcomes research published between August 2011 and August 2022, written in English, and including caregiver study participants of children who died of a medical condition. Data sources were scholarly journal articles from 9 electronic databases, including Scopus, Web of Science, Academic Search Primer, ProQuest Research Library, PubMed, Embase, PsycINFO, Psychology & Behavioral Sciences Collection, and Health Source: Nursing/Academic Edition. The Mixed Methods Appraisal Tool-2018 evaluated methodological quality. RESULTS: The study sample included 106 studies, most of which were either qualitative (60%) or quantitative (29%). Mixed-methods studies (8%) and randomized clinical trials (2%) were also identified. Study quality was variable, but most studies met all quality criteria (73%). Studies primarily represented cancer populations (58%), White participants (71%), and mothers (66%). Risk-based psychosocial outcomes (e.g., grief) were more commonly assessed than resilience-based outcomes. CONCLUSIONS: The current scoping review revealed that recent research assessing the psychosocial outcomes of bereaved parents is limited in the representation of diverse populations, primarily qualitative, of broadly strong methodological quality, and oriented to psychosocial risk. To enhance the state of the science and inform evidence-based psychosocial services, future research should consider varied methodologies to comprehensively assess processes of risk and resilience with demographically and medically diverse populations.

The contributions of clinical disease activity, functional disability, and illness intrusiveness to depressive symptoms in pediatric inflammatory bowel disease.

BACKGROUND: Clinical disease activity associated with inflammatory bowel disease (IBD) can place physical limitations on youths' activities of daily living. In turn, functional limitations potentially contribute to youths' heightened experience of IBD-induced intrusions on a wide range of routine and valued activities (i.e., illness intrusiveness), which can increase their risk for depressive symptoms. The present study examined the contributions of clinical disease activity, functional disability, and illness intrusiveness to depressive symptoms in youth with IBD. METHODS: Youth (N = 180) completed the Functional Disability Inventory (FDI), Illness Intrusiveness Scale-Child (IIS-C), and Children's Depression Inventory-2 (CDI-2). Physicians completed the Physicians Global Assessment of disease activity (PGA). RESULTS: Results revealed a mediating effect for functional disability in the association between disease activity and depressive symptoms (PGA → FDI → CDI-2); illness intrusiveness mediated the association between functional disability and depressive symptoms (i.e., FDI → IIS-C → CDI-2). Serial mediation revealed that clinical disease activity conferred an indirect effect on youth depressive symptoms through the sequential effects of functional disability and illness intrusiveness (i.e., PGA → FDI → IIS-C → CDI-2). CONCLUSIONS: Taken together, these findings indicate that youth who encounter more physical limitations as a function of clinical disease activity are more likely to experience an amplified sense of IBD-related intrusions on their ability to participate in meaningful activities. In turn, heightened illness intrusiveness increases the likelihood of depressive symptoms. Clinical interventions that help youth maintain adequate functional ability in the face of IBD disease activity and encourage involvement in positively valued activities could decrease the negative impact of IBD on youths' emotional adjustment.

Cross-Cultural Disparities in Psychosocial Research with Individuals with Classical Congenital Adrenal Hyperplasia: A Scoping Review.

INTRODUCTION: There are increased calls to address psychosocial needs among individuals with classical congenital adrenal hyperplasia (CAH). However, cross-cultural disparities exist in treatment practices and psychosocial outcomes that impact the generalizability of evidence-based recommendations. To date, this disparity has not been quantified. The present scoping review uses a dual approach to contrast rates of CAH diagnosis with CAH psychosocial research rates across countries. METHODS: Six electronic database searches were conducted for: (1) CAH incidence/birth/prevalence rates; and (2) psychosocial research with affected individuals and their families. Two authors reviewed each abstract for inclusion criteria. RESULTS: Sixty-eight and 93 full-text articles, respectively, were evaluated for incidence and country. The countries/regions with the highest reported CAH rates are Thailand, Ghana, and India. Those with the greatest portion of psychosocial publications are the USA, Germany, and the UK. CONCLUSION: A discrepancy exists between those countries with the highest CAH rates and those publishing psychosocial research. Specifically, increased rates of CAH are seen in non-Western countries/regions, whereas most psychosocial research arises out of Western Europe and the USA. Due to cultural differences between these regions, increased global collaboration is needed to both inform psychosocial research and translate findings in ways that are representative worldwide.

Adverse Birth Experiences and Parent Adjustment Associated With Atypical Genital Appearance Due to Differences of Sex Development.

OBJECTIVE: Differences/disorders of sex development (DSDs) are rare, congenital conditions involving discordance between chromosomes, gonads, and phenotypic sex and are often diagnosed in infancy. A key subset of parents of children newly diagnosed with a DSD experience clinically elevated distress. The present study examines the relationship between perinatal factors (i.e., gestational age, delivery method) and trajectories of parental adjustment. METHODS: Parent participants (mothers = 37; fathers = 27) completed measures at baseline, 6- and 12-month follow-up. Multilevel linear regression controlled for clustering of the data at three levels (i.e., time point, parent, and family) and examined the relationship between perinatal factors and trajectories of depressive and anxious symptoms. Two-way interactions between perinatal factors and parent type were evaluated. RESULTS: Overall depressive and anxious symptoms decreased over time. There were significant interactions between gestational age and parent type for depressive and anxious symptoms, with younger gestational age having a stronger negative effect on mothers vs. fathers. There was a significant interaction between time and gestational age for depressive symptoms, with 36 weeks' gestational age demonstrating a higher overall trajectory of depressive symptoms across time compared to 38 and 40 weeks. Findings for the delivery method were not significant. CONCLUSIONS: Findings uniquely demonstrated younger gestational age was associated with increased depressive symptoms, particularly for mothers compared to fathers. Thus, a more premature birth may predispose parents of infants with DSD to distress. Psychosocial providers should contextualize early diagnosis-related discussions within stressful birth experiences when providing support.

Parental Uncertainty Scale Factor Structure in Pediatric DSD With Ambiguous Genitalia.

OBJECTIVE: Illness uncertainty is a salient experience for caregivers of children with disorders/differences of sex development (DSD) presenting with ambiguous genitalia; however, no validated measure of illness uncertainty exists for this unique population. Thus, the current study aimed to preliminarily identify the factor structure of the Parental Perception of Uncertainty Scale (PPUS) in caregivers of children with DSD presenting with ambiguous genitalia and examine the convergent validity of the PPUS. METHODS: Participants included 115 caregivers (Mage = 32.12 years, SD = 6.54; 57% mothers) of children (<2-year-olds) diagnosed with DSD participating in a larger, longitudinal study. Caregivers completed the PPUS as well as self-report measures of anxious, depressive, and posttraumatic stress symptoms. An exploratory factor analysis was conducted. RESULTS: Exploratory factor analysis results indicated that a 23-item 1-factor solution was the most parsimonious and theoretically sound factor structure (α = 0.92). Convergent validity analyses demonstrated further support for the use of the 23-item 1-factor solution over the original PPUS factor structure. CONCLUSION: These results demonstrate the preliminary clinical and research utility of the PPUS with caregivers of children with DSD presenting with ambiguous genitalia. The PPUS may benefit from further refinement through qualitative research and item adaptation to capture uncertainties unique to DSD presenting with ambiguous genitalia. In addition, future research should replicate the proposed factor structure using confirmatory factor analysis with a separate, larger sample of caregivers of children with DSD to confirm the factor structure.

The Combined Effects of Youth and Parent Illness Intrusiveness on Depressive Symptoms in Adolescents with Inflammatory Bowel Disease.

Inflammatory bowel disease (IBD) presents physical and emotional challenges for families and imposes significant lifestyle intrusions on both youth and parents. The present study examined the effects of IBD disease activity and youth illness intrusiveness on depressive symptoms in adolescents, and the moderating influence of parent illness intrusiveness on these associations. Adolescents and parents completed measures of illness intrusiveness; youth completed a measure of depressive symptoms. Physicians provided estimates of IBD disease activity. Mediation analysis revealed an IBD disease activity → youth intrusiveness → youth depressive symptoms indirect effect. Moderated mediation analyses revealed this indirect effect to be greater among youth whose parents endorsed more IBD-related intrusions. Youth encountering greater activity disruptions related to IBD are vulnerable to depressive symptoms. When parents also experience IBD-induced intrusions, youth are at even greater risk for depressive symptoms. Clinical implications are discussed within the context of youths' and parents' experiences of IBD.

Psychosocial, neurodevelopmental, and transition of care practices provided to children with CHD across North American cardiac clinics.

Children with CHD are at risk for psychosocial and neurodevelopmental difficulties, as well as lapses in care during their transition from paediatric to adult CHD providers. The American Heart Association and American Academy of Pediatrics released guidelines for best practices in the neurodevelopmental and transitional care for children with CHD in 2012 and 2011, respectively. CHD providers from 48 (42.1% response rate) geographically diverse cardiac clinics completed a 31-item electronic survey designed to assess the cardiac teams' consistency with neurodevelopmental evaluation and management recommendations, consultation/liaison patterns for psychosocial services, and procedures regarding transitional services for emerging adults. Responses suggest most cardiac teams refer patients to psychosocial services as needed, and 39.6% of teams screen for psychosocial distress. CHD providers at 66.7% of cardiac clinics reported a formal neurodevelopmental programme/clinic. Nearly half of cardiac teams conduct routine neurodevelopmental evaluations, most frequently occurring at 9 months of age. Less than 10% of cardiac clinics have resources to meet the American Heart Association and American Academy of Pediatrics 2012 neurodevelopmental evaluation and management guidelines. Formal paediatric to adult CHD transition programmes were reported at 70.8% of cardiac clinics and were associated with younger ages of transition to adult CHD care. Care practices varied across the 48 represented cardiac clinics, indicating inconsistent practices for patients with CHD. Barriers and facilitators to the provision of care for children in these areas were reported and are presented. More support is needed for cardiac clinics to continue improvements in psychosocial, neurodevelopmental, and transitional care services.

Transition readiness and quality of life in emerging adult college students.

OBJECTIVE: To examine the relative contribution of transition readiness (i.e., healthcare self-management) to health-related quality of life (HRQoL) among emerging adult (EA) college students without a chronic medical condition (CMC).Participants: College students (n = 2372; Mage = 19.32, SD = 1.26) from a Midwestern university.Methods: Participants completed online measures of demographics, HRQoL, and transition readiness.Results: Hierarchical regression analyses found transition readiness accounted an additional 3-4% of the variability in mental and physical HRQoL (p < .001), beyond demographic factors. 11.3% of EAs reported overall mastery of transition readiness, with navigating health insurance being the weakest area.Conclusions: Findings support the consensus that transition readiness is relevant to HRQoL for all EAs, including those without a CMC. EAs without a CMC demonstrate relatively weak transition readiness skills. Primary and university-based healthcare might consider programs supporting transition readiness and HRQoL among underresourced EAs.

Symptoms of Survivors of Pediatric Hematopoietic Stem Cell Transplant by Age, Sex, and Transplant Type.

Purpose: The purpose of this study was to describe symptoms experienced by survivors of pediatric hematopoietic stem cell transplant (HSCT), and demographic and treatment-factors associated with ongoing symptomology. Methods: Fifty pediatric survivors completed a cross-sectional pilot study. Questionnaires were administered online via REDCap to assess symptoms experienced in the last week. Survivors also consented to a medical record chart review. Results: Survivors were on average 5.4 years post-HSCT (range 1.1 to 9 years), male (58%), and Caucasian (80%) who received an allogeneic HSCT (92%). The most commonly reported symptoms were difficulty concentrating (42.5%), pain (38%), worry (38%), nervousness (37.5%), and lack of energy/fatigue (34%). Survivors reported up to 14 symptoms, with 90% of the sample experiencing at least one symptom in the previous week. Average number of symptoms varied by age group between 2.1 (8-9 years) and 6.8 (18 and older). Age and female gender were associated with higher levels of fatigue. Conclusions: The majority of survivors experienced at least one symptom in the previous week. Neuropsychological symptoms and pain endure well into survivorship that can influence outcomes such as function and health-related quality of life (HRQOL). Research is needed on biological mechanisms of ongoing symptomology, effective interventions to prevent or mitigate symptoms, and the impact of symptoms on patient outcomes including daily functioning and HRQOL. Implications Survivors of pediatric HSCT continued to experience symptoms for up to nine years. Survivors should be frequently screened for symptoms, as symptoms may affect function, learning/employment outcomes, and HRQOL.

Relationship Between Caregiver Uncertainty, Problem-Solving, and Psychological Adjustment in Pediatric Cancer.

OBJECTIVE: The current study examined the roles of constructive and dysfunctional problem-solving strategies in the relationships between illness uncertainty and adjustment outcomes (i.e., anxious, depressive, and posttraumatic stress symptoms) in caregivers of children newly diagnosed with cancer. METHODS: Two hundred thirty-eight caregivers of children (0-19 years of age) newly diagnosed with cancer (2-14 weeks since diagnosis) completed measures of illness uncertainty, problem-solving strategies, and symptoms of anxiety, depression, and posttraumatic stress. RESULTS: A mediation model path analysis assessed constructive and dysfunctional problem-solving strategies as mediators between illness uncertainty and caregiver anxious, depressive, and posttraumatic stress symptoms. Dysfunctional problem-solving scores partially mediated the relationships between illness uncertainty and anxious, depressive, and posttraumatic stress symptoms. Constructive problem-solving scores did not mediate these relationships. CONCLUSIONS: The current findings suggest that illness uncertainty and dysfunctional problem-solving strategies, but not constructive problem-solving strategies, may play a key role in the adjustment of caregivers of children newly diagnosed with cancer. Interventions aimed at managing illness uncertainty and mitigating the impact of dysfunctional problem-solving strategies may promote psychological adjustment.

Factor Analysis of the Stigma Scale-Parent Version in Pediatric Disorders/Differences of Sex Development.

Stigma is a salient experience for both caregivers/parents and individuals with Disorders/Differences of Sex Development (DSD) as evidenced through qualitative and preliminary quantitative reports. However, few validated measures of associative stigma (i.e., vicarious stigma experienced through close association with someone who is socially stigmatized) for parents of children with DSD exist. The present study aims to (1) determine the factor structure of the adapted Stigma Scale - Parent, and (2) examine convergent validity of the factor structure with measures of parent psychosocial adjustment. Confirmatory factor analytic results revealed two factors: a parent-focused factor and a child-focused factor. The parent-focused factor demonstrated convergent validity with parent adjustment measures, but the child-focused factor did not. Together, these results indicate that parent-focused and child-focused stigma are distinct factors, with parent-focused associative stigma being related to parent adjustment following DSD diagnosis. Future research should further refine this measure to determine predictive validity and clinical value.

Parental Efficacy and Control Questionnaire in Hematopoietic Stem Cell Transplant: Preliminary Validation.

OBJECTIVE: Develop and evaluate the preliminary validity of a self-report measure of parents' treatment-related efficacy and control, Parental Efficacy and Control Questionnaire-Hematopoietic Stem Cell Transplant (PECQ-HCT), in a pediatric HCT sample. METHODS: Participants included 185 parents of children (≤12 years old) receiving HCT participating in a larger, longitudinal study. Parents completed the PECQ-HCT as well as measures of social problem-solving skills, collective family efficacy, family beliefs, and parental distress. RESULTS: Exploratory factor analysis results indicated that a 37-item four-factor model was the best fitting and most theoretically sound, χ2(df = 1,596) = 14,089.95, p < .01, comparative fit index = 0.92, Tucker-Lewis Index = 0.90, and root mean square error of approximation = 0.07. Preliminary subscale scores demonstrated adequate internal consistency as well as good content and criterion-related validity. CONCLUSIONS: If replicated using a confirmatory factor analysis in a separate sample, these findings suggest that the four-factor PECQ-HCT measure may be useful for measuring HCT-related parental efficacy and perceived control.