Home-Based Palliative Care for Children With Incurable Cancer: Long-term Perspectives of and Impact on General Practitioners.

CONTEXT: Although a large percentage of children with advanced-stage cancer die at home, remarkably little information is available regarding the experience of general practitioners (GPs) with respect to providing home-based palliative care to children with incurable cancer. OBJECTIVES: The objective of this study was to explore the perspectives of GPs who care for children with advanced-stage cancer in a home-based setting. METHODS: In this cross-sectional study, 144 GPs who provided home-based palliative care to 150 children with incurable cancer from 2001 through 2010 were invited to complete a questionnaire addressing their perspectives regarding: 1) symptom management, 2) collaboration with other health care professionals, 3) the child's death and care after death, and 4) impact of having provided palliative care, scored on distress thermometer (range 0-10). RESULTS: A total of 112 GPs (78%) responded, and 91 GPs completed the questionnaire for 93 patients. The median interval between the child's death and completing the questionnaire was seven years. The most prevalent symptoms reported in the patients were fatigue (67%) and pain (61%). Difficulties with communicating with (14%), coordinating with (11%), collaborating with (11%), and contacting (2%) fellow members of the multidisciplinary treatment team were rare. Hectic (7%) and shocking (5%) situations and panic (2%) around the child's death were rare. GPs reported feelings of sadness (61%) and/or powerlessness (43%) around the time of the patient's death, and they rated their own distress level as relatively high during the terminal phase (median score 6, range 0-9.5). The majority of GPs (94%) reported that they ultimately came to terms with the child's death. CONCLUSION: In general, GPs appear to be satisfied with the quality of home-based palliative care that they provide pediatric patients with incurable cancer. Communication among health care professionals is generally positive and is considered important. Finally, although the death of a pediatric patient has a profound impact on the GP, the majority of GPs eventually come to terms with the child's death.

Participation in a clinical trial for a child with cancer is burdensome for a minority of children.

AIM: This study explored how parents who had lost a child to cancer felt about them taking part in a clinical trial. METHODS: A retrospective questionnaire was sent to parents who had lost a child to cancer. They were asked whether their child took part in a clinical trial during their palliative phase, their motives for their child's participation, how they perceived their child's burden and whether they would, hypothetically speaking, enrol again. RESULTS: The 24 parents of 16 deceased children who had participated in a clinical trial explained their motives for their child's participation. The most common answers, with multiple responses, were treatment for future patients (n = 16), hope for a cure (n = 9) and prolonging their child's life (n = 6). Eight parents said that participating was not burdensome for their child and four said it was very burdensome, with others answering in between. None of the parents would decline participation if they would be in the same situation again. CONCLUSION: Performing clinical trials, even in a vulnerable population, such as children with cancer at the end of life, may not always lead to increased burden. None of the parents would in future, given the same circumstances, decline participation in a clinical trial.

Talking about Death with Children with Incurable Cancer: Perspectives from Parents.

OBJECTIVE: To investigate the rationale and consequences associated with a parent's decision to discuss death with a child with incurable cancer. STUDY DESIGN: We present data from a larger retrospective study involving bereaved parents of a child who died of cancer. Parents were asked whether they had discussed the impending death with their child, whether they reflected on this discussion positively, their reasons for not discussing death with their child, and the manner in which the conversation regarding death occurred. The data were analyzed qualitatively using a framework approach. RESULTS: Of the 86 parents of 56 children who answered the questions regarding discussing death with their child, 55 parents of 35 children did not discuss the impending death with their child. The following themes were identified: the parents' inability to discuss the impending death; the parents' desire to protect their child; views regarding talking with children; parents' views of child characteristics; the child's unwillingness to discuss the subject; lack of opportunity to talk; and the child's disability. The parents who did discuss death with their child generally used symbolic and/or religious narratives, or they had brief, direct conversations regarding death. The majority of parents felt positive regarding their decision about whether to talk with their child about his/her impending death. CONCLUSION: Most parents in this study cited several reasons for not discussing death with their child. Our findings highlight the sensitive and complex issues surrounding these conversations, indicating that there may be a role for clinicians in supporting parents.

End-of-Life Decisions in Pediatric Cancer Patients.

BACKGROUND: End-of-life decisions (ELDs) have been investigated in several care settings, but rarely in pediatric oncology. OBJECTIVE: The aims of this study were to characterize the practice of end-of-life decision making in a Dutch academic medical center and to explore pediatric oncologists' perspectives on decision making. METHODS: Between 2001 and 2010, in a specified period of 2 years, 57 children died of cancer. The attending pediatric oncologists of 48 deceased children were eligible for this study. They were requested to complete a retrospective questionnaire on characteristics of ELDs that may have preceded a child's death. ELDs were defined as decisions concerning administering or forgoing treatment that may unintentionally or intentionally hasten death. RESULTS: In 31 of 48 cases (65%) one or more ELDs were made. In 20 of 31 cases potentially life-prolonging treatments were discontinued or withheld, and in 22 of 31 cases drugs were administered to alleviate pain or other symptoms in potentially life-shortening dosages. Frequently mentioned considerations for making ELDs were no prospects of improvement (n=21;68%) and unbearable suffering without a curative perspective (n=13;42%). ELDs were discussed with parents in all cases, and with the child in 9 of 31 cases. After the child's death, the pediatric oncologist met the parents in all ELD cases and in 11 of 17 non-ELD cases. Pediatric oncologists were satisfied with care around the child's death in 90% of the ELD cases versus 59% of the non-ELD cases. CONCLUSIONS: In two-thirds of cases, ELDs preceded the death of a child with cancer. This is the first study providing insights into the characteristics of ELDs from a pediatric oncologist's point of view.

Parents' Faith and Hope during the Pediatric Palliative Phase and the Association with Long-Term Parental Adjustment.

BACKGROUND: The loss of a child is associated with an increased risk for developing psychological problems. However, studies investigating the impact of parents' faith and hope for a cure during the palliative phase on long-term parental psychological functioning are limited. OBJECTIVE: The study's objective was to explore the role of faith and hope as a source of coping and indicator of long-term parental adjustment. METHODS: Eighty-nine parents of 57 children who died of cancer completed questionnaires retrospectively, exploring faith, hope, and sources of coping, and measuring parents' current level of grief and depression. RESULTS: For 19 parents (21%) faith was very important during the palliative phase. The majority of parents remained hopeful for a meaningful time with their child (n=68, 76%); a pain-free death (n=58, 65%); and a cure (n=30, 34%). Their child (n=70, 79%) was parents' main source of coping. Twelve parents (14%) suffered from traumatic grief, and 22 parents (25%) showed symptoms of depression. Parents' faith was not associated with less long-term traumatic grief (OR=0.86, p=0.51) or symptoms of depression (OR=0.95, p=0.74), and parents' hope for a cure was not related to more long-term traumatic grief (OR=1.07, p=0.71) or symptoms of depression (OR=1.12, p=0.47). CONCLUSIONS: Faith was important for a minority of parents and was not associated with less long-term traumatic grief or symptoms of depression. The majority of parents remained hopeful. Hope for a cure was not associated with more long-term traumatic grief or symptoms of depression.

Parents' experiences of pediatric palliative care and the impact on long-term parental grief.

CONTEXT: Approximately 25% of children diagnosed with cancer eventually die. Losing a child puts parents at increased risk for developing psychological problems. OBJECTIVES: To explore parents' perceptions of the interaction with health care professionals (communication, continuity of care, and parental involvement) and symptom management during the pediatric palliative phase, and to investigate the influence on long-term grief in parents who lost a child to cancer. METHODS: A total of 89 parents of 57 children who died of cancer between 2000 and 2004 participated in this retrospective cross-sectional study by completing a set of questionnaires measuring grief (Inventory of Traumatic Grief), parents' perceptions of the interaction with health care professionals (communication, continuity of care, and parental involvement), and symptom management during the palliative phase. Care was assessed on a five point Likert scale (1=disagree and 5=agree). RESULTS: Parents highly rated communication (4.6±0.6), continuity of care (4.3±0.6), and parental involvement (4.6±0.7) during the palliative phase. Parents' most often reported physical and psychological symptoms of their child during the palliative phase were fatigue (75%), pain (74%), anxiety to be alone (52%), and anger (48%). Higher ratings of parents on communication (ß=-9.08, P=0.03) and continuity of care (ß=-11.74, P=0.01) were associated with lower levels of long-term parental grief. The severity of the child's dyspnea (ß=2.96, P=0.05), anxiety to be alone (ß=4.52, P<0.01), anxiety about the future (ß=5.02, P<0.01), anger (ß=4.90, P<0.01), and uncontrolled pain (ß=6.60, P<0.01) were associated with higher levels of long-term parental grief. Multivariate models combining the interaction with health care professionals and symptom management showed a significant influence of both aspects on long-term parental grief. CONCLUSION: Both interaction with health care professionals, especially communication and continuity of care, and symptom management in children dying of cancer are associated with long-term parental grief levels.

Emotional distress in 652 Dutch very long-term survivors of childhood cancer, using the hospital anxiety and depression scale (HADS).

BACKGROUND: After a more successful treatment of pediatric cancer, the number of childhood cancer survivors is progressively increasing. Consequently, awareness of psychological late sequelae is important. PROCEDURE: The Hospital Anxiety and Depression Scale (HADS) was used as a screening tool for emotional distress in a single center cohort of 652 childhood cancer survivors (median age 23 y [range, 15 to 46 y], median follow-up time 15 y [range, 5 to 42 y]). Results were compared with a control group of 440 Dutch subjects. A higher HADS score linearly reflect a higher level of emotional distress, and a score ≥15 is indicative of clinically significant emotional distress. RESULTS: Mean HADS score of the childhood cancer survivors was not different from the control group (P=0.38). Survivors exposed to global central nervous system (CNS) irradiation had a significantly higher HADS score than the control group (8.3±6.6; P=0.05) as well as other survivors (P=0.01). Forty-three survivors (7%) had a HADS score ≥15. Survivors with a HADS score ≥15 were variously spread over the diagnostic-related and treatment-related subgroups. Linear regression analysis showed that high educational achievement (ß=-1.28; P<0.01) and age at the time of the study (ß=0.08; P=0.03) were both significantly associated with the HADS score. CONCLUSIONS: Emotional distress does not occur more often in childhood cancer survivors than in the normal population. No disease-related or treatment-related variable was independently associated with emotional distress.

Gonadal function recovery in very long-term male survivors of childhood cancer.

BACKGROUND: Although gonadal toxicity has been reported, no data are available on recovery of gonadal function in very long-term survivors of childhood cancer. Inhibin B is a novel reliable serum marker which has been shown to be of value in childhood cancer survivor studies to identify risk groups for impaired gonadal function, but consecutive long-term follow-up studies using serum inhibin B as a marker are not available. OBJECTIVE: To evaluate possible recovery of gonadal dysfunction over time in adult male survivors of childhood cancer. METHODS: In this retrospective study, adult male long-term childhood cancer survivors (n=201) who visited our outpatient late effects clinic were included and we used inhibin B as a surrogate marker for gonadal function. RESULTS: Median age at diagnosis was 5.9 years (range 0.0-17.5) and discontinuation of treatment was reached at a median age of 8.2 years (range 0.0-20.8). Inhibin B levels were first measured after a median follow-up time of 15.7 years (range 3.0-37.0). Median interval between the first (T1) and second measurement (T2) was 3.3 years (range 0.7-11.3). Median inhibin B level was 127 ng/L (range 5-366) at T1 and 155 ng/L (range 10-507) at T2. The prediction model suggests that inhibin B levels do not normalise in survivors with a very low Inhibin B level at T1. CONCLUSIONS: Our results suggest that recovery of gonadal function is possible even long after discontinuation of treatment. However, this recovery does not seem to occur in survivors who already reached critically low inhibin B levels after discontinuation of treatment.

Treatment factors rather than genetic variation determine metabolic syndrome in childhood cancer survivors.

BACKGROUND: Genetic variation that regulates insulin resistance, blood pressure and adiposity in the normal population might determine differential vulnerability for metabolic syndrome after treatment for childhood cancer. OBJECTIVE: To evaluate the contribution of candidate single nucleotide polymorphisms (SNPs) relevant for metabolic syndrome in our single centre cohort of adult long-term childhood cancer survivors. METHODS: In this retrospective study 532 survivors were analysed. Median age at diagnosis was 5.7 years (range 0.0-17.8 years), median follow-up time was 17.9 years (range 5.0-48.8) and median age at follow-up was 25.6 years (range 18.0-50.8). JAZF1 gene rs864745, THADA gene rs7578597, IRS1 gene rs2943641, TFAP2B gene rs987237, MSRA gene rs7826222, ATP2B1 gene rs2681472 and rs2681492 were genotyped. The association of genotypes with total cholesterol levels, blood pressure, body mass index, waist circumference and frequency of diabetes were assessed. RESULTS: Metabolic syndrome was more frequent in cranially (23.3%, P=0.002) and abdominally (23.4%, P=0.009) irradiated survivors as compared with non-irradiated survivors (10.0%). Association of allelic variants in rs2681472 and rs2681492 with hypertension, rs987237 and rs7826222 with waist circumference and rs864745, rs7578597 and rs2943641 with diabetes were not significant. None of the SNPs was associated with the metabolic syndrome. Adjusting for age, sex, follow-up time, cranial irradiation and abdominal irradiation did not change these results. CONCLUSIONS: Treatment factors and not genetic variation determine hypertension, waist circumference, diabetes and metabolic syndrome in adult long-term survivors of childhood cancer.