Growth of condition-related knowledge among youth with spina bifida: associations with neurocognitive functioning and self-management skills.

OBJECTIVE: This study aims to characterize the growth in condition-related knowledge in youth with spina bifida (SB), identify neurocognitive predictors of growth, and examine associations between growth in knowledge and subsequent levels of medical self-management skills. METHODS: Participants were recruited from a larger longitudinal study involving 140 youth with SB and caregivers, who completed questionnaires and interviews every 2 years over 8 years. The current study included the youth report of condition-related knowledge and medical self-management skills. Youth attention and executive functioning were assessed via parent and teacher reports and performance-based assessment. Latent growth curves were conducted in Mplus Version 8 (Múthen, L. K., & Múthen, B. O. [1998]. Mplus User's Guide. [Eighth]. Muthén & Muthén) to examine change over time in youth-reported condition-related knowledge. Neurocognitive variables were included as predictors of growth in knowledge and regression analyses were used to predict medical self-management skills from growth in condition-related knowledge. RESULTS: Youth condition-related knowledge increased linearly. Better youth performance on working memory and attention performance-based tasks predicted a higher intercept for condition-related knowledge at T1, but not slope. Teacher and parent reports of inattention and executive dysfunction were not consistent predictors of intercept and growth. Slope of condition-related knowledge was not predictive of subsequent youth self-management skills. CONCLUSIONS: Youth with SB gain condition-related knowledge over time. However, executive dysfunction and inattention may impede gains in condition-related knowledge. Thus, executive functioning supports, attention-related interventions, and psychoeducation may support condition-related knowledge gains and later medical self-management skills, but further research assessing family and cultural factors is needed.

Comparing Cognitive Disengagement Syndrome Growth in Youth With and Without Spina Bifida.

OBJECTIVE: Cognitive disengagement syndrome (CDS; formally known as sluggish cognitive tempo), difficulties with social engagement, and lower levels of autonomy have been identified as maladaptive comorbidities in youth with spina bifida (SB). This study compared growth curves of CDS for youth with and without SB and examined whether these trajectories were associated with later functioning. METHODS: Longitudinal data spanning 8 years included youth with SB (n = 68, Mage = 8.34) and a demographically matched sample of typically developing (TD) peers (n = 68, Mage = 8.49). Adolescents, along with their caregivers and teachers, reported on youth social skills, behavioral functioning, and CDS. Growth curve models were examined by comparing CDS trajectories by SB status. RESULTS: Growth curves indicated that youth with SB had higher levels of teacher-reported CDS at ages 8 and 9, but growth curves were relatively stable for both groups. When predicting social skills, higher levels of teacher-reported (but not mother-reported) CDS at baseline predicted worse social functioning for both youth with and without SB in adolescence. For the slope findings, higher rates of mother-reported CDS over time predicted worse social skills (ß = -0.43) and lower levels of youth decision-making (ß = -0.43) for the SB group, while higher rates of teacher-reported CDS predicted worse social skills for the TD group. CONCLUSION: Next steps include understanding the impact that impaired social functioning and restricted autonomy have on youth with and without SB due to CDS to inform interventions. Additionally, advocacy for increased awareness of CDS-related impairment is needed, particularly for youth with chronic health conditions.

Problem solving and collaborative involvement among adolescents with spinal cord injury and their caregivers.

Objective: To determine the relationship between constructive adolescent problem solving (positive problem-solving orientation and rational problem-solving style) and caregiver problem solving and collaborative involvement with primary caregiver among adolescents with spinal cord injuries (SCIs). Positive constructive adolescent problem solving was hypothesized to be predicted by more effective caregiver problem solving and higher collaborative involvement. Methods: Participants in this cross-sectional study were 79 adolescent and primary caregiver dyads recruited from a pediatric rehabilitation care system in North America. All participants completed a standardized problem-solving instrument and adolescent participants completed an adapted measure of collaborative parent involvement. Results: More effective caregiver problem solving and adolescent perceptions of more collaboration with caregivers around SCI care were significantly associated with higher positive problem-solving orientation and higher rational problem-solving style among adolescents. Conclusions: Results underscore the importance of caregiver problem-solving skills and their collaboration with adolescents with SCI when addressing care needs. Clinically, findings highlight opportunities for parent involvement and skill-building as an important factor of rehabilitation for adolescents with SCI.

Preliminary Validation of the Psychosocial Assessment Tool in the Neonatal Intensive Care Unit.

OBJECTIVE: Research suggests families whose infants are admitted to the Neonatal Intensive Care Unit (NICU) experience elevated distress and may have pre-existing risk factors for maladjustment. This study sought to validate the newly developed Psychosocial Assessment Tool (PAT-NICU/Cardiac Intensive Care Unit [CICU]), a comprehensive screening measure for family psychosocial risk in the NICU. METHODS: The sample included 171 mothers, who completed the PAT-NICU/CICU and other related measures within 2 weeks of their infant's NICU admission at a level 4 unit within a large pediatric hospital. PAT-NICU/CICU scores were compared to a companion risk survey completed by NICU social workers. Test-retest reliability was assessed through repeated measures at 2-month follow-up. RESULTS: Analyses suggest the PAT-NICU/CICU is effective in classifying psychosocial risk. This is supported by statistically significant correlations between the PAT-NICU/CICU and validated measures, in addition to elevated scores on concurrent measures by risk classification. Internal consistency, test-retest reliability, and acceptability for the PAT-NICU/CICU were satisfactory. CONCLUSIONS: This preliminary study demonstrates the validity, reliability, and acceptability of the PAT-NICU/CICU as a psychosocial screening tool to aid identification of families who may benefit from supportive services during NICU admission. This new measure is a more comprehensive tool that assesses a wide variety of risk factors and stress responses. However, future studies of this measure are needed with more diverse samples. Prompt screening of NICU parents may facilitate earlier linkage with appropriate levels of resources or intervention. This research is crucial in improving risk assessment and psychosocial care for families in the NICU.

Working memory and cognitive flexibility predict growth trajectories of sluggish cognitive tempo in youth with spina bifida.

As sluggish cognitive tempo (SCT) shows similar inattention and neuropsychological dysfunction as youth with spina bifida (SB), it is important to examine whether neuropsychological functioning may affect the development of SCT in this population. Participants were 140 youth with SB and their parents who participated in five waves of a longitudinal study across eight years (ages 8-15 years at Time 1). At Time 1, teacher-, mother-, and father-report of SCT showed 9%, 8.3%, and 5.3% impairment in SCT respectively compared to other youth in the sample. Growth curves were used to examine changes over time in mother-, father-, and teacher-reported SCT. Four neurocognitive variables were included (attention, working memory, cognitive flexibility, plan/organize) as predictor variables of SCT growth and intercepts. Mother, father, and teacher-report of SCT were included in separate models. Age and shunt status were included as covariates in the growth models. Inattentive symptoms were also included as a covariate in working memory, shift, and plan/organize models. Worse working memory at Time 1 predicted an increase in mother-reported SCT symptoms over eight years (ß = -.28) and poorer cognitive shifting skills predicted higher rates of teacher-reported SCT over time (ß = -.61). All other neurocognitive variables predicted the intercept, but not the slope of SCT symptoms. Results indicate that worse neurocognitive functioning is associated with higher levels of SCT symptoms and that worse working memory and cognitive flexibility at baseline predicted an increase in SCT symptoms as youth with SB age. It may be important for clinicians to monitor and assess levels of SCT in youth with SB.

The impact of CNS-directed treatment on quality of life in childhood cancer survivors.

PURPOSE: Pediatric cancer survivors may have lower quality of life (QoL), but most research has assessed outcomes either in treatment or long-term survivorship. We focused on early survivorship (i.e., 3 and 5 years post-diagnosis), examining the impact of CNS-directed treatment on child QoL, as well as sex and age at diagnosis as potential moderators. METHODS: Families of children with cancer (ages 5-17) were recruited at diagnosis or relapse (N = 336). Survivors completed the PedsQL at 3 (n = 96) and 5 years (n = 108), along with mothers (101 and 105, respectively) and fathers (45 and 53, respectively). The impact of CNS treatment, sex, and age at diagnosis on child QoL was examined over both time since diagnosis and time since last treatment using mixed model analyses. RESULTS: Parent-report of the child's total QoL was in the normative range and stable between 3 and 5 years when examining time since diagnosis, while child reported QoL improved over time (p = 0.04). In terms of time since last treatment, mother and child both reported the child's QoL improved over time (p = 0.0002 and p = 0.0006, respectively). Based on parent-report, males with CNS-directed treatment had lower total QoL than females and males who did not receive CNS-directed treatment. Age at diagnosis did not moderate the impact of treatment type on total QoL. CONCLUSIONS: Quality of life (QoL) in early survivorship may be low among males who received CNS-directed treatment. However, this was only evident on parent-report. Interventions to improve child QoL should focus on male survivors who received CNS-directed treatment, as well as females regardless of treatment type.

Stress and Coping in Youth With Spina Bifida: A Brief Longitudinal Study in a Summer Camp Setting.

INTRODUCTION: It is well established that youth with chronic conditions experience elevated levels of stress; the manner in which they respond to or cope with this stress is likely to impact both health and psychosocial outcomes. The current study examined stress and coping in youth and young adults with spina bifida (SB) using the response to stress questionnaire-SB version (RSQ-SB; Connor-Smith et al., 2000). METHODS: Data were collected as part of a camp-based psychosocial intervention for children (ages 7-13), adolescents (ages 14-19), and young adults (ages 20-38) with SB. Participants completed the RSQ-SB as well as questionnaires assessing demographics and condition severity. Data were collected prior to camp (T1) and 1 month (T2) after camp ended. Self-report data were collected from adolescents and young adults; parents of children and adolescents reported on their child's stress and coping. Ratios of primary control coping, secondary control coping, disengagement coping, involuntary engagement, and involuntary disengagement coping were calculated. Descriptive statistics and t-tests were utilized to describe coping and stress responses and to determine potential change over time. T-tests were also used to compare youth and parent reported coping styles with those of youth with type 1 diabetes (T1D) and sickle cell disease (SCD). Associations between demographic/disease factors and coping styles were also examined. RESULTS: Parent and youth report indicated that youth with SB tend to use primary control coping. Youth with SB use more primary control coping and less disengagement coping compared to youth with SCD and youth with T1D. Few significant changes in coping were found between T1 and T2. IQ and socioeconomic status were significantly associated with coping styles. CONCLUSION: Youth with SB use more primary control coping compared to other coping methods and as compared to other pediatric populations. Future studies should examine mechanisms by which primary control coping is advantageous for youth with SB. Future interventions should be more focused on promoting adaptive coping behaviors and be tailored to developmental age and access to resources.

A Systematic Review of Benefit-Finding and Growth in Pediatric Medical Populations.

OBJECTIVE: This review synthesizes the literature on benefit-finding and growth (BFG) among youth with medical illnesses and disabilities and their parents. Specifically, we summarized: (a) methods for assessing BFG; (b) personal characteristics, personal, and environmental resources, as well as positive outcomes, associated with BFG; (c) interventions that have enhanced BFG; and (d) the quality of the literature. METHODS: A medical research librarian conducted the search across PubMed, Scopus, PsycInfo, Google Scholar, and Cochrane Library. Studies on BFG among children ages 0-18 with chronic illnesses and disabilities, or the parents of these youth were eligible for inclusion. Articles were uploaded into Covidence; all articles were screened by two reviewers, who then extracted data (e.g., study characteristics and findings related to BFG) independently and in duplicate for each eligible study. The review was based on a systematic narrative synthesis framework and adhered to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines (PROSPERO registration number: CRD42020189339). RESULTS: In total, 110 articles were included in this review. Generally, BFG capabilities were present across a range of pediatric health conditions and disabilities. Correlates of both youth and parent BFG are presented, including personal and environmental resources, coping resources, and positive outcomes. In addition, studies describing interventions aimed at enhancing BFG are discussed, and a quality assessment of the included studies is provided. CONCLUSIONS: Recommendations are provided regarding how to assess BFG and with whom to study BFG to diversify and extend our current literature.

Parent Perceptions of Infant Symptoms and Suffering and Associations With Distress Among Bereaved Parents in the NICU.

CONTEXT: Healthcare providers and parents face many challenges caring for infants at the end of life (EOL). Symptom assessment and management in critically ill infants can be especially difficult. However, the impact of the infant's EOL experience on bereaved parents is largely unknown. OBJECTIVE: Explore associations between parental perceptions of infant symptoms and suffering at EOL in the neonatal intensive care unit (NICU) and parent adjustment following the death. METHODS: Retrospective, cross-sectional pilot study involving parents of infants who died within the previous five years in a large, Midwestern, level IV NICU. Parents were recruited through mailed invitations, and 40 mothers and 27 fathers participated from 40 families. Parents retrospectively reported on infant symptom burden and suffering during the last week of life and the Impact of Events Scale-Revised (IES-R), and Prolonged Grief-13 (PG-13). Hierarchical regressions examined demographic/medical factors and parent perceptions at EOL in relation to post-traumatic stress symptoms (PTSS) and prolonged grief (PG). RESULTS: Clinical levels of PTSS (Mothers = 18%; Fathers = 11%) and PG (Mothers and Fathers = 3%) were low. Maternal perception of higher symptom burden was associated with greater PTSS, R2 = 0.46, P= 0.001, and PG, R2 = 0.47, P < 0.01. Paternal perception of greater infant suffering was associated with greater PTSS, R2 = 0.48, P= 0.001, and PG, R2 = .38, P < 0.01. CONCLUSION: Perceptions of symptoms and suffering were associated differently with mother and father adjustment after bereavement. While not necessarily causal, better symptom management at EOL could minimize distress for both infants and their parents.

Parent-Child Communication and Reproductive Considerations in Families with Genetic Cancer Predisposition Syndromes: A Systematic Review.

Background: Uptake of genetic testing for heritable conditions is increasingly common. In families with known autosomal dominant genetic cancer predisposition syndromes (CPS), testing youth may reduce uncertainty and provide guidance for future lifestyle, medical, and family building considerations. The goals of this systematic review were to examine: (1) how parents and their children, adolescents, and young adults (CAYAs) communicate and make decisions regarding testing for CPS and (2) how they communicate and make decisions about reproductive health/family building in the context of risk for CPS. Methods: Searches of MEDLINE/Pubmed, CINAHL, Web of Science, and PsycINFO yielded 4161 articles since January 1, 2000, which contained terms related to youth, pediatrics, decision-making, genetic cancer predispositions, communication, and family building. Results: Articles retained (N = 15) included five qualitative, six quantitative, and four mixed-method designs. Parents generally agreed testing results should be disclosed to CAYAs at risk or affected by genetic conditions in a developmentally appropriate manner. Older child age and child desire for information were associated with disclosure. Greater knowledge about risk prompted adolescents and young adults to consider the potential impact on future relationships and family building. Conclusions: Most parents believed it was their responsibility to inform their CAYAs about genetic testing results, particularly to optimize engagement in recommended preventative screening/lifestyle behaviors. Disclosing test results may be challenging due to concerns such as young age, developmental appropriateness, and emotional burden. Additional research is needed on how CPS risk affects CAYAs' decisions about reproductive health and family building over time.

Myoendothelial Junctions of Mature Coronary Vessels Express Notch Signaling Proteins.

RATIONALE: Myoendothelial junctions (MEJs) within the fenestrae of the internal elastic lamina (IEL) are critical sites that allow for endothelial cell (EC) - vascular smooth muscle cell (VSMC) contact and communication. Vascular Notch signaling is a critical determinant of normal vasculogenesis and remodeling, and it regulates cell phenotype via contact between ECs and VSMCs. To date, no studies have linked Notch signaling to the MEJ despite it requiring cell-cell contact. Furthermore, very little is known about Notch in the adult coronary circulation or the localization of Notch signaling and activity within the mature intact blood vessel. OBJECTIVE: We tested the hypothesis that vascular Notch signaling between ECs and VSMCs occurs at MEJs. METHODS AND RESULTS: Notch receptor and ligand immunofluorescence was performed in human coronary EC and VSMC co-cultures across transwell inserts (in vitro MEJs) and in the intact mouse coronary circulation. Human coronary VSMC Notch activity induced by human coronary ECs at the in vitro MEJ was assessed using a CBF-luciferase construct. We observed Jagged1, Notch1, Notch2, and Notch3 expression within the in vitro and in vivo MEJs. We also demonstrated a 3-fold induction (p < 0.001) of human coronary VSMC Notch signaling by ECs at the in vitro MEJ, which was completely blocked by the Notch inhibitor, DAPT (p < 0.01). CONCLUSION: We demonstrate for the first time in mature blood vessels that Notch receptors and ligands are expressed within and are active at coronary MEJs, demonstrating a previously unrecognized mode of Notch signaling regulation between the endothelium and smooth muscle.

Perceived Infant Well-Being and Self-Reported Distress in Neonatal Nurses.

BACKGROUND: Infants who are admitted to a neonatal intensive care unit (NICU) may experience significant symptom burden. Parents are often distressed by these symptoms, which can affect their long-term coping and distress. There is limited research examining nurse perceptions of infant well-being (symptoms, suffering, and quality of life [QOL]) and associations with nurse distress. OBJECTIVE: The objective of this descriptive study was to explore associations between nurse perceptions of infant well-being and self-reported distress. METHODS: Nurses caring for infants with potentially life-threatening/life-limiting conditions were recruited from a Level IV NICU in the Midwestern United States as a part of a study on infant symptom burden. Nurses reported their perceptions of infant well-being and their own distress on a 5-point Likert scale. Surveys were administered at the bedside weekly for up to 12 weeks, depending on length of stay. Infant suffering and QOL were examined in relation to nurse distress. A cross-classified multilevel model was used to account for dependence within nurse and within patient. RESULTS: A total of 593 surveys were collected from nurses. Using a cross-classified multilevel model with variables entered simultaneously, nurse perceptions of greater infant suffering and lower infant QOL were significantly associated with greater nurse distress. DISCUSSION: Preliminary evidence shows that greater perceived infant suffering and lower perceived infant QOL may be associated with greater levels of self-reported distress in NICU nurses. Further work is needed to better understand factors related to symptom management in the NICU and the potential role of caregiver distress and compassion fatigue in NICU nurses.

Long-term trajectories of depression symptoms in mothers of children with cancer.

OBJECTIVE: To identify trajectories of depression symptoms in mothers of children with cancer from diagnosis/relapse through 5 years and examine maternal factors at diagnosis/relapse predicting membership in these trajectories. METHOD: Mothers (n = 327; Mage = 37.6 years, SD = 7.7 years; 85.9% White) reported depression symptoms near the time of their child's diagnosis/relapse and then again at 1-, 3-, and 5-years postdiagnosis/relapse. Mothers also reported perceived stress, coping (primary control, secondary control, and disengagement coping), and spirituality near the time of diagnosis. Latent class growth analysis was used to identify latent trajectories of depression symptoms, and a 3-step multinomial logistic regression tested covariate predictors of membership in the trajectories. RESULTS: Three trajectories were identified: "low depression symptoms" (63.3%), "moderate depression symptoms" (31.5%), and "high depression symptoms" (5.2%). Mothers who used more primary and secondary control coping were more likely to be in the low depression symptom trajectory as compared with the moderate (OR = 1.64, p = .024 and OR = 1.38, p = .013, respectively) or high trajectories (OR = 1.99, p = .008 and OR = 1.81, p = .001, respectively). CONCLUSIONS: Although mothers of children with cancer generally displayed improved mental health further from diagnosis, mothers with more depression symptoms after diagnosis/relapse displayed substantial stability in depression symptoms over the 5 years. Mothers of children with cancer may benefit from early screening of mental health and coping strategies, as well as interventions to bolster effective coping for those with elevated depression symptoms. (PsycINFO Database Record (c) 2020 APA, all rights reserved).

Perceptions of participating in family-centered fertility research among adolescent and young adult males newly diagnosed with cancer: A qualitative study.

BACKGROUND: Over half of male childhood cancer survivors experience infertility after treatment, which is known to cause distress and impact future quality of life. Sperm banking rates remain low, and little is known about how adolescent and young adult (AYA) males and their families make fertility preservation (FP) decisions. This study examined AYA and parent perceptions of participating in a research study focused on testing a new FP decision tool at the time of cancer diagnosis. METHODS: Forty-four participants (19 mothers, 11 fathers, 14 male AYAs 12-25 years old) from 20 families completed brief assessments at diagnosis and approximately one month later, including a qualitative interview exploring the impact of study participation. Verbatim transcripts were coded through thematic content analysis using the constant comparison method. RESULTS: Two major themes emerged: (1) a positive effect of participating in the study and (2) a neutral effect (no positive/negative effect of participation). Subthemes that emerged for participants who noted a positive effect included (a) participation prompted deeper thinking, (b) participation influenced family conversations, and (c) participation resulted in altruism/helping others. No participant reported a negative effect. CONCLUSIONS: This study demonstrates that participation in family-centered research focused on FP among AYA males, before treatment begins, is perceived as beneficial or neutral at the time of a new cancer diagnosis. These findings provide support for future family-centered FP interventions for this population.

Conducting reproductive research during a new childhood cancer diagnosis: ethical considerations and impact on participants.

PURPOSE: Research among adults shows benefits and low perceived burden of engaging in behavioral research. However, questions remain regarding the ethics of conducting behavioral research in pediatric populations during sensitive situations, including during a new life-threatening diagnosis or at end-of-life. We examined reactions to participating in a behavioral reproductive research study among male adolescents newly diagnosed with cancer and their parents, as a step towards optimizing fertility preservation utilization in a population where future infertility is common. METHODS: Pediatric literature regarding the ethics of behavioral research was reviewed. In our pilot, forty-four participants (19 mothers, 11 fathers, 14 male adolescents newly diagnosed with cancer) from 20 families completed demographic questionnaires and a fertility preservation decision tool developed by the study team. Qualitative interviews exploring the impact of study participation were subsequently conducted. Verbatim transcripts were coded for thematic content using the constant comparison method. RESULTS: Literature review showed positive reactions to research participation among youth/caregivers. In our pilot study, 89% (n = 17) of mothers, 64% (n = 7) of fathers, and 71% (n = 10) of adolescents reported at least one benefit of participating. Eleven percent (n = 2) of mothers, 36% (n = 4) of fathers, and 29% (n = 4) of adolescents said they were not affected; none of the participants reported a negative effect. CONCLUSION: Consistent with prior literature, our study suggests behavioral reproductive research prior to cancer treatment can offer direct benefits to participants and society, without increasing burden. These findings will inform future interventions to improve long-term psychosocial and reproductive outcomes for youth with cancer.

Maternal high fructose diet and neonatal immune challenge alter offspring anxiety-like behavior and inflammation across the lifespan.

AIMS: This study examined the interaction between maternal high fructose diet and neonatal inflammation in neonates (P7), juveniles (P26-34) and adults on measures of anxiety-like behavior and cognition. The study aimed to assess the potential synergistic effects of these two forms of early-life inflammation. MAIN METHODS: We fed Sprague-Dawley dams with high fructose (60%) diet or normal chow. Each litter was treated with either saline or lipopolysaccharide (LPS) on postnatal day (P)3 and P5 and two pups were tested for USVs after maternal separation on P7. Post-weaning, juveniles were tested on the elevated zero maze (EZM) and in a context-object discrimination (COD) task prior to tissue harvest. Adults were tested on the EZM and the COD task as well. Immunohistochemistry and ELISA were used to assess molecular and cellular changes in the offspring. KEY FINDINGS: This study demonstrates that maternal diet and neonatal inflammation altered peripheral inflammation in neonates, altered anxiety-like behavior in juveniles, and altered anxiety-like behavior in adulthood. Maternal diet and sex increased juvenile peripheral inflammation and altered memory on the context-discrimination task. SIGNIFICANCE: Maternal diet has a profound impact on fetal and neonatal development, especially as obesity rates are on the rise worldwide. Together, these findings reveal enduring effects of maternal diet on offspring, support the findings on the effects of neonatal inflammation on anxiety-like behaviors in later-life periods, and add to the complex relationship between gestational and neonatal inflammation and anxiety.