Stigma, Intrusiveness, and Distress in Parents of Children with a Disorder/Difference of Sex Development.

OBJECTIVE: The impact of parent-reported stigma due to their child's disorder/difference of sex development (DSD) on parent psychosocial adjustment is poorly understood. In other pediatric populations, perceived interference of medical conditions into daily activities (i.e., illness intrusiveness ) mediates the relationship of stigma to adjustment. This study assessed relationships between parent-focused and child-focused stigma → illness intrusiveness → depressive and anxious symptoms . Exploratory analyses sought to identify patient characteristics associated with stigma. METHOD: Caregivers (59 women and 43 men) of 63 children diagnosed with a DSD up to age 4 years completed measures of demographics, parent-focused and child-focused stigma, illness intrusiveness, and depressive and anxious symptoms. RESULTS: Increased parent-focused and child-focused stigma were associated with increased illness intrusiveness, which, in turn, was associated with increased depressive and anxious symptoms for parents nested within dyads. Among children with DSD family histories, parents reported greater child-focused stigma. CONCLUSION: Parents who experience DSD-related stigma report greater interference of their child's DSD into their daily activities, which is associated with poorer psychosocial adjustment. Findings support developing clinical interventions related to parents' perceptions of stigma and illness intrusiveness to improve parent adjustment.

Health Disparities in Transgender and Gender Expansive Adolescents: A Topical Review From a Minority Stress Framework.

OBJECTIVE: To present a topical review of minority stressors contributing to psychosocial and physical health disparities in transgender and gender expansive (TGE) adolescents. METHODS: We conducted a topical review of original research studies focused on distal stressors (e.g., discrimination; victimization; rejection; nonaffirmation), proximal stressors (e.g., expected rejection; identity concealment; internalized transphobia), and resilience factors (e.g., community connectedness; pride; parental support) and mental and physical health outcomes. RESULTS: Extant literature suggests that TGE adolescents experience a host of gender minority stressors and are at heightened risk for negative health outcomes; however, limited research has directly applied the gender minority stress framework to the experiences of TGE adolescents. Most research to date has focused on distal minority stressors and single path models to negative health outcomes, which do not account for the complex interplay between chronic minority stress, individual resilience factors, and health outcomes. Research examining proximal stressors and resilience factors is particularly scarce. CONCLUSIONS: The gender minority stress model is a helpful framework for understanding how minority stressors contribute to health disparities and poor health outcomes among TGE adolescents. Future research should include multiple path models that examine relations between gender minority stressors, resilience factors, and health outcomes in large, nationally representative samples of TGE adolescents. Clinically, adaptations of evidence-based interventions to account for gender minority stressors may increase effectiveness of interventions for TGE adolescents and reduce health disparities in this population of vulnerable youth.

The Relationship Between Parent Distress and Child Quality of Life in Pediatric Cancer: A Meta-Analysis.

PROBLEM: Pediatric cancer places both parents and children at risk for psychosocial difficulties, including a specific risk for diminished quality of life. Previous research has identified relationships between parent and child psychosocial adjustment outcomes (e.g., depression, anxiety), yet the relationships between parent adjustment and child quality of life have yet to be comprehensively evaluated via meta-analysis. ELIGIBILITY CRITERIA: A systematic review and meta-analysis were conducted using EBSCO, with PsychINFO, MEDLINE, Academic Search Premiere, and Health Source: Nursing/Academic Edition. SAMPLE: Fourteen studies met inclusion criteria. RESULTS: Fourteen correlations from 1646 parents of children with cancer were evaluated, resulting in a medium-magnitude correlation between parent psychosocial adjustment and child quality of life (r = 0.23, p < .001). Additional analyses evaluating the relationship between parent psychosocial adjustment and child social/emotional quality of life resulted in a medium-magnitude correlation (r = 0.24, p < .001). CONCLUSIONS: A significant relationship exists between parent psychosocial adjustment and child quality of life. However, this relationship appears slightly less strong than those found in meta-analyses evaluating other child psychosocial adjustment outcomes. IMPLICATIONS: Parent distress is an important factor to evaluate in the context of pediatric cancer, as it appears to have implications for child quality of life, in addition to other child psychosocial adjustment outcomes.

Physical Activity and Pain in Youth With Sickle Cell Disease.

Study objectives were to examine the relationships between physical activity, pain, and psychological distress in youth 8 to 17 years of age with sickle cell disease. Participants were 206 youth with sickle cell disease (M = 11.73 years, 54.9% female, 99.5% African American). Caregivers and youth completed a clinical psychosocial screening battery. Results revealed frequent pain (37.6%), moderate median pain intensity, and elevated median pain interference in youth. Lower caregiver-reported physical activity was associated with worse pain outcomes. Increased anxiety was also associated with worse pain outcomes. A better understanding of the relationship between physical activity/inactivity and pain will guide multifactorial treatment interventions.

Barriers to care in pediatric cancer: The role of illness uncertainty in relation to parent psychological distress.

OBJECTIVE: The current study evaluated perceived barriers to care for parents of children with cancer and the mediating effect of illness uncertainty (IU; uncertainty from the ambiguity or unpredictability of the illness) on the relationship between barriers and parental psychological distress. We hypothesized that greater barriers to care would be related to higher levels of IU and, in turn, higher anxiety, depression, and posttraumatic stress symptom (PTSS) ratings. METHODS: As part of an ongoing study of family adjustment to pediatric cancer, 145 caregivers of children diagnosed with cancer completed questionnaires assessing barriers to care, parent IU, and anxious symptoms, depressive symptoms, and PTSS. Time since cancer diagnosis ranged from 1 to 12 months. RESULTS: Three mediation models assessed IU as a mediator between barriers to care and anxious symptoms, depressive symptoms, and PTSS, controlling for annual income. IU significantly mediated the relationship between barriers to care and depressive symptoms (B = -.03, SE = .02; 95% CI [-.08, -.01]) and to PTSS (B = -.15, SE = .10; 95% CI [-.38, -.03]). The mediation model was not significant for anxious symptoms. CONCLUSION: Experiencing barriers to obtaining treatment for their child with cancer is a significant risk factor for symptoms of depression and PTSS among parents. Specifically, greater barriers to care is significantly associated with IU, a well-established precursor to distress in this population. Interventions targeting IU may help ameliorate distress within the context of unmodifiable barriers to care.

Featured Article: The Relationship Between Parent and Child Distress in Pediatric Cancer: A Meta-Analysis.

OBJECTIVE: Parents and children affected by pediatric cancer are at risk for psychological distress, including depression, anxiety, and post-traumatic stress. A link is believed to exist between parent and child distress; however, no systematic analysis of this relationship has occurred. A meta-analysis was conducted to assess the relationship between parent and child distress among families affected by pediatric cancer. METHODS: A systematic review and meta-analysis was conducted using EBSCO (searching PsycINFO, MEDLINE, Academic search Premiere, and Health Source: Nursing/Academic Edition) and PubMed. The initial search yielded a total of 29,118 articles. Inclusion criteria were that studies assessed the relation between parent and child distress in the context of pediatric cancer, were written in English, and were published in peer-reviewed journals. 28 articles met inclusion criteria. RESULTS: A statistically significant association was found between overall parent and child distress (r = .32, p < .001), such that increased parent-reported distress was associated with increased distress in their children. Significant relationships were also present among each type of parental distress (i.e., depression, anxiety, post-traumatic stress, and global distress; rs = .31-.51, ps < .001) and overall child distress. Moderation analyses via meta-regression indicated that parent proxy-report of child symptoms was associated with a stronger relationship between parent and child distress than child self-report of their own distress. CONCLUSIONS: Aligned with the social-ecological framework, familial factors appear to be highly relevant in understanding distress following pediatric cancer diagnosis. Indeed, greater parent distress was associated with greater child distress.

Predictors of Psychosocial Distress in Parents of Young Children with Disorders of Sex Development.

PURPOSE: We evaluated demographic, financial and support predictors of distress for parents of young children with disorders of sex development including atypical genital development, and characterized early parental experiences. This work extends our previous findings to identify those parents at risk for distress. MATERIALS AND METHODS: Participants included mothers (76) and fathers (63) of a child (78) diagnosed with disorders of sex development characterized by moderate to severe genital atypia. Parents completed a demographic questionnaire, measures of anxious and depressive symptoms, quality of life, illness uncertainty and posttraumatic stress symptoms, and rated their satisfaction with the appearance of their child's genitalia. RESULTS: Depressive and posttraumatic stress symptoms of caregivers were comparable to standardized norms while levels of anxious symptoms were below norms. A subset of parents reported clinically elevated symptoms. Overall 26% of parents reported anxious symptoms, 24% reported depressive symptoms and 17% reported posttraumatic stress symptoms. Levels of illness uncertainty were lower than those of parents of children with other chronic illnesses. Differences by parent sex emerged, with mothers reporting greater distress. Lower income, increased medical care and travel expenses, and having no other children were related to increased psychosocial distress. CONCLUSIONS: Early psychosocial screening is recommended for parents of children with disorders of sex development. Clinicians should be aware that financial burden and lack of previous parenting experience are risk factors for distress.

Uncertainty and Posttraumatic Stress: Differences Between Mothers and Fathers of Infants with Disorders of Sex Development.

Parents of children with disorders of sex development (DSD) report significant psychological distress, including posttraumatic stress symptoms (PTSS), with mothers consistently reporting higher rates of psychological distress than fathers. However, psychological factors contributing to PTSS in both parents are not well understood. The present study sought to fill this gap in knowledge by examining PTSS and illness uncertainty, a known predictor of psychological distress, in parents of children recently diagnosed with DSD. Participants were 52 mothers (Mage = 32.55 years, SD = 5.08) and 41 fathers (Mage = 35.53 years, SD = 6.78) of 53 infants (Mage = 9.09 months, SD = 6.19) with DSD and associated atypical genital development. Participants were recruited as part of a larger, multisite study assessing parents' psychosocial response to their child's diagnosis of DSD. Parents completed measures of illness uncertainty and PTSS. Mothers reported significantly greater levels of PTSS, but not illness uncertainty, than fathers, and were more likely than fathers to report clinical levels of PTSS (21.2% compared to 7.3%). Hierarchical regression revealed that parent sex, undiagnosed or unclassified DSD status, and illness uncertainty were each associated with PTSS. The overall model accounted for 23.5% of the variance associated with PTSS. Interventions targeting illness uncertainty may be beneficial for parents of children with newly diagnosed DSD.

Baseline Characteristics of Infants With Atypical Genital Development: Phenotypes, Diagnoses, and Sex of Rearing.

PURPOSE: Little is known about the phenotypes, diagnoses, and sex of rearing of infants with atypical genital development in the United States. As part of a multicenter study of these infants, we have provided a baseline report from US difference/disorder of sex development clinics describing the diagnoses, anatomic features, and sex of rearing. We also determined whether consensus guidelines are followed for sex designation in the United States. METHODS: Eligible participants had moderate-to-severe genital atypia, were aged <3 years, and had not undergone previous genitoplasty. Karyotype, genetic diagnosis, difference/disorder of sex development etiology, family history, and sex of rearing were collected. Standardized examinations were performed. RESULTS: Of 92 subjects, the karyotypes were 46,XX for 57%, 46,XY for 34%, and sex chromosome abnormality for 9%. The median age at the baseline evaluation was 8.8 months. Most 46,XX subjects (91%) had congenital adrenal hyperplasia (CAH) and most 46,XY subjects (65%) did not have a known diagnosis. Two individuals with CAH underwent a change in sex of rearing from male to female within 2 weeks of birth. The presence of a uterus and shorter phallic length were associated with female sex of rearing. The most common karyotype and diagnosis was 46,XX with CAH, followed by 46,XY with an unknown diagnosis. Phenotypically, atypical genitalia have been most commonly characterized by abnormal labioscrotal tissue, phallic length, and urethral meatus location. CONCLUSIONS: An increased phallic length was positively associated with rearing male. Among the US centers studied, sex designation followed the Consensus Statement recommendations. Further study is needed to determine whether this results in patient satisfaction.

Transition readiness: The linkage between adverse childhood experiences (ACEs) and health-related quality of life.

Adverse childhood experiences (ACEs) are consistently associated with deleterious health outcomes in adolescence and adulthood. Although research has implicated health-risk behaviors as a possible link in this relationship, the role of health care management skills has yet to be examined in this context. Transition readiness, defined as health care management skills during the transition from adolescence to adulthood, may be relevant in understanding health risk among individuals with ACEs. The present study evaluated health care management skills in adolescents and young adults (AYAs) transitioning to adult services, as AYA skills during this period set the stage for long-term health care management skills. The present study assessed transition readiness as a mediator between ACEs and health-related quality of life (HRQoL). College undergraduates (N = 1,011) between the ages of 18-23 years completed an online battery of self-report measures, including total ACEs (ACES-SF), transition readiness (TRAQ), and physical and emotional HRQoL (SF-36). Total ACE scores were negatively associated with physical and emotional HRQoL, and transition readiness was positively correlated with physical and emotional HRQoL. Transition readiness mediated the relationship between cumulative ACEs and both physical HRQoL (B = -0.079, 95% confidence interval [-0.17, -0.01]) and emotional HRQoL (B = -0.14, 95% confidence interval [-0.28, -0.015]). When examined by sex, this relationship remained significant for females only. Results suggest a need to promote health care management skills to improve transition readiness as means of enhancing health-related outcomes among AYAs with a history of ACEs.

Parent-Rated Severity of Illness and Anxiety among Caregivers of Children Born with a Disorder of Sex Development Including Ambiguous Genitalia.

BACKGROUND/AIMS: Parents of children born with disorders of sex development (DSD) often experience anxiety, but risk factors, including parental perception of the severity of their child's DSD, have not been examined. We hypothesized that severity of illness (SOI) ratings would relate to parental anxiety, and would be higher for parents of children with a potentially life-threatening DSD (e.g., 21-hydroxylase deficiency). METHODS: Eighty-nine parents (Mage = 33.0, 56.2% mothers) of 51 children (Mage in months = 8.7) with a DSD including ambiguous genitalia were recruited from 12 specialized DSD clinics. Parents completed questionnaires prior to genitoplasty, 6 months post-genitoplasty, and 12 months post-genitoplasty (if completed). Data were analyzed with linear mixed modeling. RESULTS: Parental anxiety decreased over time, χ2(1) = 10.14, p < 0.01. A positive relationship between SOI and anxiety was found, with SOI being a strong predictor of anxiety (b = 0.53, p < 0.01; χ2[1] = 5.33, p < 0.05). An SOI by time interaction indicated SOI had an increasing effect on anxiety over time, b = 0.06, p < 0.05; χ2(1) = 6.30, p < 0.05. There was no diagnosis by SOI interaction. CONCLUSION: Parental anxiety decreased over time, but those with higher SOI ratings reported greater initial anxiety followed by slower resolution over time. Underlying etiology of DSD had no effect on the relationship between SOI and anxiety.

Post traumatic stress symptoms in parents of children with cancer: A mediation model.

OBJECTIVE: Caregivers of pediatric cancer patients are at risk for posttraumatic stress symptoms (PTSS). Previous literature has identified caregiver illness uncertainty as a predictor of PTSS, yet little is known about the mechanism by which illness uncertainty may affect PTSS. Rumination, or perseverations about the cause and consequences of an event, has been related to posttraumtic stress disorder in other populations. However, limited research of this relationship exists for pediatric cancer caregivers. Further, no studies have evaluated rumination in relation to illness uncertainty. The current study examined rumination in relation to illness uncertainty and PTSS in caregivers with children actively receiving treatment for cancer. It was hypothesized that rumination would be related to illness uncertainty and would mediate the relationship between illness uncertainty and PTSS. METHODS: Caregivers (N = 59) completed the Parent Perception of Uncertainty Scale, Ruminative Responses Scale, and the Impact of Event Scale-Revised. RESULTS: illness uncertainty was identified as a significant predictor of rumination (B = 0.148, p = .015), and rumination was a significant predictor of PTSS (B = 1.83, p < .001). Biased-corrected bootstrap regression analysis revealed that rumination mediated the relationship between illness uncertainty and PTSS (R2 = 0.53, p < .001). CONCLUSIONS: Rumination was identified as a mediator between the relationship of illness uncertainty and PTSS. These findings underscore the importance of rumination in caregivers of children with cancer, as a potential construct for identifying caregivers at risk of PTSS, and as a possible mechanism for targeted intervention.