Parental Understanding of Their Child's Germline Genomic Testing: Intent of Disclosure to Their Child and Family.

Genomic testing is becoming increasingly common in the care of pediatric patients with cancer. Parental understanding of germline results and their intent and timing of results disclosure to their child and family may have significant implications on the family unit. The purpose of this study was to examine parental understanding of germline genomic results and plans for disclosure to their child and other relatives. Semi-structured interviews were conducted with 64 parents of children with cancer, approximately eight weeks after parents had received their child's results. Parents of children with negative results (n = 20), positive results (n = 15), or variants of uncertain significance (n = 29), were interviewed. Fifty-three parents (83%) correctly identified their child's results as negative, uncertain, or positive. Most parents had disclosed results to family members; however, only 11 parents (17%) acknowledged discussing results with their child. Most parents delayed disclosure due to the young age of their child at the time of testing. In summary, most parents appropriately described their child's germline genomic results, yet few discussed the results with their child due to age. Families should be followed with supportive counseling to assist parents in the timing and content of result disclosure to their children.

Parental Preferences Surrounding Timing and Content of Consent Conversations for Clinical Germline Genetic Testing Following a Child's New Cancer Diagnosis.

PURPOSE: Clinical genomic testing is increasingly being used to direct pediatric cancer care. Many centers are interested in offering testing of tumors and paired germline tissues at or near the time of cancer diagnosis. We conducted this study to better understand parent preferences surrounding timing and content of consent conversations for clinical germline genetic testing of their children with cancer as a part of real-time cancer care. PATIENTS AND METHODS: A seven-question survey developed by the Division of Cancer Predisposition and collaborators at St Jude Children's Research Hospital (St Jude) was distributed to members of the St Jude Patient Family Advisory Council, which included parents of childhood cancer survivors and bereaved parents whose children with cancer had died. Parents were asked to provide free text comments after each question. Qualitative methods were used to derive codes from parent comments, and survey results were depicted using descriptive statistics. RESULTS: The survey was completed by 172 parents. Ninety-three (54%) endorsed an approach for consent conversations ≥ 1 month after cancer diagnosis, whereas 58 (34%) endorsed an approach at 1-2 weeks and 21 (12%) at 1-2 days. Needing time to adjust to a new or relapsed cancer diagnosis and feeling overwhelmed were frequent themes; however, parents acknowledged the urgency and importance of testing. Parents desired testing of as many cancer-related genes as possible, with clinical utility the most important factor for proceeding with testing. Most parents (75%) desired germline results to be disclosed in person, preferably by a genetic counselor. CONCLUSION: Parents described urgency and benefits associated with germline testing, but desired flexibility in timing to allow for initial adjustment after their child's cancer diagnosis.

Managing Pandora's Box: Familial Expectations around the Return of (Future) Germline Results.

BACKGROUND: Pediatric oncology patients are increasingly being offered germline testing to diagnose underlying cancer predispositions. Meanwhile, as understanding of variant pathogenicity evolves, planned reanalysis of genomic results has been suggested. Little is known regarding the types of genomic information that parents and their adolescent children with cancer prefer to receive at the time of testing or their expectations around the future return of genomic results. METHODS: Parents and adolescent children with cancer eligible for genomic testing for cancer predisposition were surveyed regarding their attitudes and expectations for receiving current and future germline results (ClinicalTrials.gov Identifier: NCT02530658). RESULTS: All parents (100%) desired to learn about results for treatable or preventable conditions, with 92.4% wanting results even when there is no treatment or prevention. Parents expressed less interest in receiving uncertain results for themselves (88.3%) than for their children (95.3%). Most parents (95.9%) and adolescents (87.9%) believed that providers have a responsibility to share new or updated germline results indefinitely or at any point during follow-up care. Fewer parents (67.5%) indicated that they would want results if their child was deceased: 10.3% would not want to be contacted, 19.3% were uncertain. CONCLUSIONS: Expectations for return of new or updated genomic results are high among pediatric oncology families, although up to one third of parents have reservations about receiving such information in the event of their child's death. These results underscore the importance of high-quality pre-and post-test counseling, conducted by individuals trained in consenting around genomic testing to elicit family preferences and align expectations around the return of germline results.

Coping Trajectories and the Health-Related Quality of Life of Childhood Cancer Survivors.

OBJECTIVE: To identify coping trajectories from diagnosis through survivorship and test whether particular trajectories exhibit better health-related quality of life (HRQOL) at 5 years post-diagnosis. METHODS: Families of children with cancer (ages 5-17; M = 10.48, SD = 4.03) were recruited following a new diagnosis of cancer (N = 248). Three follow-up assessments occurred at 1-year (N = 185), 3-years (N = 101), and 5-years (N = 110). Mothers reported on children's coping using the Responses to Stress Questionnaire for Pediatric Cancer. Survivor HRQOL was measured at 5-year follow-up using self-report on the PedsQL 4.0. Longitudinal patterns of coping were derived using Latent Class Growth Analysis and mean-levels of survivor-report HRQOL were compared across classes. RESULTS: Two primary control coping trajectories emerged, "Moderate and Stable" (50%) and "Low-moderate and Decreasing" (50%), with no significant differences in HRQOL across trajectories. Three secondary control coping trajectories emerged, "Moderate-high and Increasing" (54%), "Moderate and Stable" (40%), and "High and Increasing" (6%), with survivors in the last trajectory showing better HRQOL. Two disengagement coping trajectories emerged, "Low and Stable" (85%) and "Low and Variable" (15%), with no significant differences in HRQOL across trajectories. CONCLUSIONS: Coping trajectories were relatively stable from diagnosis to 5 years. A small group of survivors with high and increasing secondary control coping over time, per mother-report, reported better HRQOL. Future research should consider tailoring coping interventions to children with cancer to improve survivors' HRQOL.

Temporal relationship between daily pain and actigraphy sleep patterns in pediatric sickle cell disease.

Limited research is available on the relationship between objective sleep patterns and pain in children with SCD. Research in other chronic pain populations suggests that the effect of sleep disruption on pain may be stronger than the effect of pain on sleep that night. To examine the bi-directional relationship between objective sleep patterns and daily pain in a pediatric SCD sample. Participants were 30 African American children with SCD 8-18 years (13 ± 2.8 years; 66.7% female) with frequent pain. Children and parents completed questionnaires to assess pain, medications, and depression/anxiety. Over a 14-day period, children completed a pain diary and ambulatory actigraphy monitoring to assess nighttime sleep (duration, efficiency and WASO). Greater pain severity was associated with worse sleep efficiency and greater WASO that night, controlling for age, sex, opioid medication, and depression/anxiety symptoms. Worse sleep efficiency was associated with the occurrence of pain and more severe pain the next day. There was no relationship between WASO and pain. Similarly, sleep duration did not influence pain. Results lend support for a bi-directional relationship between sleep parameters and daily pain in pediatric SCD, and identify sleep as a potential target for future research and intervention.

Cancer as a stressful life event: Perceptions of children with cancer and their peers.

BACKGROUND: The medical traumatic stress model is commonly applied to childhood cancer, assuming that the diagnosis of cancer is a traumatic event. However, to the authors' knowledge, little is known regarding what specifically children perceive as stressful about cancer or how it compares with other stressful events more often experienced by children. METHODS: Children with cancer (254 children) and demographically similar peers without a history of serious illness (202 children) identified their most stressful life event as part of a diagnostic interview assessing for symptoms of posttraumatic stress disorder (PTSD). The events identified as most stressful were categorized thematically, with categories established separately for cancer-related and non-cancer-related events. Events also were examined to assess whether they met Diagnostic and Statistical Manual of Mental Disorders (DSM) A criteria for PTSD. RESULTS: In the group of children with cancer, 54% described a cancer-related event as the most stressful event they had experienced. Six distinct categories of cancer-related events and 10 categories of non-cancer-related events were identified. The same noncancer events were identified by children in both groups, and occurred at similar frequencies. The percentage of cancer-related events that met DSM A criteria for PTSD differed dramatically depending on which version of the DSM was applied. CONCLUSIONS: Children do not necessarily view their cancer experience as their most stressful life event. The findings of the current study suggest that the diagnosis of cancer might be better viewed as a manageable stressor rather than a major trauma, and are consistent with the change in the fifth edition of the DSM to eliminate the diagnosis of a life-threatening illness as a qualifying trauma for PTSD. Cancer 2017;123:3385-93. © 2017 American Cancer Society.