Cancer-related stress in childhood cancer survivorship: Prevalence and associations with perceptions of health risks and quality of life.

OBJECTIVE: Limited research has characterized cancer-related stress (CRS) among families of childhood cancer survivors. We examined the prevalence of CRS among survivors and caregivers, as well as its association with health risk perceptions (i.e., prognosis, risk for diminished quality of life) and views of survivor quality of life (QoL). METHODS: At five years post-diagnosis or relapse, survivors (n = 100; Mage  = 15.84 years; 89% White), mothers (n = 127), and fathers (n = 59) reported their CRS. Perceived prognosis and risk for diminished QoL were rated on a 0%-100% visual analogue scale, while the PedsQL assessed QoL. RESULTS: CRS was low (M = 1.6-1.8, scale: 1-4); mothers reported greater stress than survivors, p = 0.038, d = 0.25. There was an indirect effect of survivors' perceived prognosis on their QoL through CRS, CI = 0.04 to 0.25, R2  = 0.32. Among mothers, there was an indirect effect of perceived prognosis/risk for diminished QoL on their reports of survivor QoL through CRS, CI = 0.03 to 0.23 and -0.15 to -0.03, R2  = 0.28 and 0.32, respectively. There were no indirect effects among fathers. CONCLUSIONS: CRS may be an important, modifiable factor that could improve survivors' QoL. Research is needed to examine how CRS changes over time to assess the utility of interventions among female survivors, mothers, and those with lower prognosis estimates.

Impact of End-of-Life Circumstances on the Adjustment of Bereaved Siblings of Children Who Died from Cancer.

The aim of this study was to examine the impact of end-of-life (EoL) circumstances on grief and internalizing symptoms among bereaved siblings. Bereaved families (N = 88) were recruited from three sites 3-12 months (M = 11.57, SD = 3.48) after their child's death from cancer. One sibling per family aged 8-17 years (M = 12.41, SD = 2.64) was randomly selected to participate. Families completed measures of siblings' grief and internalizing symptoms, as well as a structured interview about circumstances surrounding the death. Mother and sibling reports of EoL circumstances were generally concordant, except there was a discrepancy between mothers and children about whether or not children expected their sibling's death (t(75) = 1.52, p = .018). Mother reports of sibling internalizing symptoms were above the normative mean (t(83) = 4.44, p ≤ .001 (M = 56.01 ± 12.48), with 39% (n = 33) in the borderline/clinical range. Sibling opportunity to say goodbye was associated with greater grief-related growth (t(79) = - 1.95, p = .05). Presence at the death and wishing they had done something differently were both associated with greater grief (t(80) = - 2.08, p = .04 and t(80) = - 2.24, p = .028, respectively) and grief-related growth (t(80) = - 2.01, p = .048 and t(80) = - 2.31, p = .024, respectively). However, findings were primarily unique to sibling report, with few mother-reported effects. The adjustment of bereaved siblings may be affected by certain modifiable circumstances surrounding the death of their brother or sister. A proportion of bereaved siblings had elevated internalizing symptoms irrespective of circumstances at EoL. Further work is needed to understand predictors of adjustment among bereaved siblings to provide better support and optimize their outcomes.

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.

Parent-Adolescent Concordance Regarding Fertility Perspectives and Sperm Banking Attempts in Adolescent Males With Cancer.

OBJECTIVE: Approximately half of male childhood cancer survivors experience impaired fertility, yet fertility preservation (FP) remains underutilized. Although parent recommendation influences adolescents' decision-making, parents may be uncertain and/or underrate their sons' parenthood goals. This study assessed parent-adolescent and family-level concordance regarding adolescent fertility perspectives (i.e., values, goals) and associations with FP attempts. METHODS: A prospective pilot study examined the impact of a family-centered values clarification tool (FAST) on banking attempts among adolescent males newly diagnosed with cancer at risk for infertility. The FAST assessed adolescent and parent perceptions of adolescents' fertility values and goals (i.e., perceived threat of infertility, perceived benefits/barriers to banking). Parent-adolescent concordance and family-level concordance on fertility perspectives were examined, along with associations with banking attempts and salient demographic factors. RESULTS: Ninety-eight participants (32 adolescents aged 12-20, 37 mothers, 29 fathers) from 32 families completed the FAST before treatment initiation. Parent-adolescent dyads were concordant on approximately one-half of responses. Banking attempts were associated with higher family-level concordance regarding perceived benefits, r(32) = .40, p = .02. Older adolescent age was associated with higher family-level concordance regarding perceived threat, r(31) = .37, p = .04, and benefits, r(32) = .40, p = .03. Fathers' education was associated with higher family-level concordance regarding barriers, r(21) = .53, p = .01. CONCLUSIONS: When parents were concordant with their son's fertility values and goals, particularly perceived benefits, adolescents were more likely to attempt FP. Clinicians should facilitate sharing of fertility perspectives within families before cancer treatment, especially with younger adolescents. Psychosocial support for families facing FP decisions is recommended at diagnosis and across the care continuum.

Impact of central nervous system-directed treatment on competence and adjustment among children in early cancer survivorship.

BACKGROUND: Central nervous system (CNS)-directed treatments can cause long-term academic, social, and emotional difficulties for children with cancer. However, limited research has examined the emergence of problems longitudinally and has often stratified risk by diagnosis alone. Therefore, this study compared competence and adjustment in children, who did and did not receive CNS-directed treatment, over the first 3 years following a cancer diagnosis. PROCEDURE: Mothers, fathers, and children (ages 5-18 years at diagnosis) from 217 families reported on the child's competence (academic, social) and adjustment (anxious/depressed, withdrawn/depressed) near a new cancer diagnosis or relapse and 3 years later. Children were categorized into CNS-directed treatment (n = 112; including cranial radiation, intrathecal chemotherapy, and/or neurosurgery) and non-CNS-directed treatment (n = 105) groups. RESULTS: At enrollment, there were few differences in competence and emotional adjustment among children based on treatment or diagnostic group. At 3 years, mothers and fathers reported poorer social competence for the CNS-directed treatment group, and fathers reported poorer school competence for the CNS-directed treatment group. Over time, father ratings of social competence increased for the non-CNS-directed treatment group, but not the CNS-directed treatment group. While father ratings of academic competence declined for the CNS-directed treatment group, mother ratings declined the most for children diagnosed with a brain tumor. All children demonstrated higher anxious/depressed scores over time. CONCLUSIONS: CNS-directed treatment may be a valuable indicator to identify childhood cancer survivors at risk for poor competence during early survivorship. Follow-up screening and supportive services are recommended, as well as additional longitudinal research.

Impact of a novel family-centered values clarification tool on adolescent sperm banking attempts at the time of a new cancer diagnosis.

PURPOSE: Over half of males experience fertility impairment after childhood cancer therapy, which often causes psychosocial distress. Yet, fertility preservation (FP) remains underutilized. The goals of this study were to determine the feasibility and impact of implementing a family-centered FP values clarification tool on sperm banking attempts among adolescent males newly diagnosed with cancer, and identify key determinants of banking attempts. METHODS: A prospective pilot study was conducted among families of males (12-25 years old), prior to cancer therapy. Thirty-nine of 41 families agreed to participate (95%); 98 participants (32 adolescents, 37 mothers, 29 fathers) completed the Family-centered Adolescent Sperm banking values clarification Tool (FAST). Analyses assessed the impact of the FAST on banking attempts and examined associations between demographic/medical characteristics, FAST subscales (perceived threat, benefits, barriers), and banking attempts. RESULTS: Twenty-three (59%) adolescents attempted to bank, compared to 8 adolescents (33%) during baseline assessment (p=.04). Significant associations were identified between banking attempts and adolescents' report of perceived threat (rpb=.45, p=.01) and benefits (rpb=.57, p=.01). Only mothers' proxy reports of adolescent perceived threat (rpb=.42, p=.01) and benefits (rpb=.47, p=.003) were associated with banking attempts, while fathers' self-reported perceived benefits (rpb=.43, p=.03), self-reported barriers (rpb=.49, p=.01), and proxy reports of adolescent perceived threat (rpb=.38, p=.04) and benefits (rpb=.59, p=.02) were associated with banking attempts. CONCLUSION: Adolescent sperm banking attempt rates significantly increased after implementation of a family-centered FP values clarification tool prior to cancer treatment. Findings underscore the importance of targeting both adolescents and their parents, particularly fathers, in FP efforts.

Concerns of Parents With Children Receiving Home-Based Pediatric Palliative Care.

CONTEXT: Caring for a child who will die from a life-limiting illness is one of the most difficult experiences a parent may face. Pediatric palliative care (PPC) has grown as a specialty service to address the unique needs of children and families with serious illness. However, gaps remain between the needs of families in PPC and the support received. OBJECTIVES: The objective of this study was to explore the concerns of parents who have a child in home-based PPC. METHODS: Semistructured interviews were conducted with 25 mothers and 10 fathers from 25 families shortly after their child's referral to home-based PPC. Children (57% male, Mage = 10.5 years, SD = 3.95, range = 4-18 years) had a range of diagnoses. Data were analyzed using inductive content analysis. RESULTS: Parents' concerns clustered into four main themes: 1) ensuring that their child's remaining days were spent living well physically, emotionally, and socially; 2) uncertainty regarding their child's diagnosis, prognosis, and treatments; 3) their child's death (e.g., the process of dying and when it will occur); and 4) the family, including the impact of the child's illness and death on siblings and wanting to cherish as much time together with family as possible. CONCLUSION: Parents of children receiving home-based PPC expressed concerns across a range of domains, both about their seriously ill child and the broader family. These results highlight salient worries among parents of children in PPC and point to critical areas for intervention for seriously ill children and the broader family.

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.

"We Can Always Adopt": Perspectives of Adolescent and Young Adult Males with Cancer and Their Family on Alternatives to Biological Parenthood.

Purpose: Approximately half of male childhood cancer survivors experience impaired fertility, which is known to cause psychological distress. Yet, less than 50% of at-risk adolescent and young adult (AYA) males pursue fertility preservation (FP) at diagnosis. Alternatives to biological parenthood (e.g., adoption/sperm donation) may be considered, but little is known about perspectives regarding these alternatives among AYA males and their families. Methods: Families of AYAs were recruited for a mixed-method study examining FP decisions at cancer diagnosis. One month later, 48 participants from 20 families (18 male AYAs, 12-22 years of age, 19 mothers, 11 fathers) completed semistructured interviews, including two questions about: (a) alternative routes to biological parenthood, and (b) their knowledge about the processes involved and/or challenges associated with such alternatives. Verbatim transcripts were coded for thematic content using the constant comparison method. Results: Three main themes were identified, of which two represent both ends of considering alternative parenthood: (a) Willingness to consider alternatives to biological parenthood, primarily adoption; (b) No consideration/discussion of alternative family building options; and (c) Variable knowledge of alternatives and/or associated challenges. Notably, more AYAs than parents mentioned a specific preference for biological children. Conclusions: One-month postcancer diagnosis, most parents reported willingness to consider alternatives to biological parenthood for their sons, while AYA males were less knowledgeable or open to these options. Future research should prospectively examine how these attitudes affect FP decisions before treatment. Medical and psychosocial providers should counsel patients and survivors accordingly to optimize reproductive outcomes and prevent psychosocial distress if parenthood goals are unfulfilled.

Romantic Relationships and Physical Intimacy Among Survivors of Childhood Cancer.

Purpose: Childhood cancer survivors are at risk for impaired psychosocial functioning, but limited research has focused on psychosexual outcomes in young adulthood. This qualitative study examined the perceived impact of childhood cancer on adult survivors' romantic relationships and sexual/physical intimacy. Methods: Phone interviews were completed with adult survivors of childhood cancer, exploring the impact of cancer on (1) romantic relationships and (2) sexual/physical intimacy. Verbatim transcripts were coded using thematic content analysis until saturation was confirmed (n = 40). Results: Survivors in this study (n = 40) were 23-42 years old (M = 29.8; 63% female) and 10-37 years postdiagnosis (M = 18.4). Regarding romantic relationships, 60% of participants reported a negative impact, while 55% of participants reported positive effects; ∼25% of participants reported no impact of childhood cancer on adult romantic relationships. Negative themes included fertility-related concerns, physical effects (e.g., self-consciousness), feeling emotionally guarded, and delayed dating. Positive themes were creating new perspectives, increased maturity, and stronger bonds with partners. Forty percent of survivors in this study perceived having fewer partners than peers. Regarding sexual/physical intimacy, 68% of participants reported a negative impact (themes: body image, fertility-related concerns, sexual/physical dysfunction), while 33% of participants reported no effects. Conclusions: This study demonstrates both positive and negative effects of childhood cancer on adult survivors' romantic relationships, whereas effects on physical intimacy were predominantly negative. Further research is needed to inform effective psychosexual interventions, and health care providers should routinely address these topics in survivorship care.

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.