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Purpose: To evaluate the prevalence of and risk factors for hypertension in childhood cancer survivors (CCSs) who were treated with potentially nephrotoxic therapies.Methods: In the Dutch Childhood Cancer Survivor Study LATER cohort part 2 renal study, 1024 CCS ≥5 years after diagnosis, aged ≥18 years at study participation, treated between 1963 and 2001 with nephrectomy, abdominal radiotherapy, total body irradiation (TBI), cisplatin, carboplatin, ifosfamide, high-dose cyclophosphamide (≥1 g/m2 per single dose or ≥10 g/m2 total) or haematopoietic stem cell transplantation participated and 500 controls from Lifelines. Hypertension was defined as blood pressure (BP) (mmHg) systolic ≥140 and/or diastolic ≥90 or receiving medication for diagnosed hypertension. At the study visit, the CKD-EPI 2012 equation including creatinine and cystatin C was used to estimate the glomerular filtration rate (GFR). Multivariable regression analyses were used.For ambulatory BP monitoring (ABPM), hypertension was defined as BP daytime: systolic ≥135 and/or diastolic ≥85, night time: systolic ≥120 and/or diastolic ≥70, 24-h: systolic ≥130 and/or diastolic ≥80. Outcomes were masked hypertension (MH), white coat hypertension and abnormal nocturnal dipping (aND).Results: Median age at cancer diagnosis was 4.7 years (interquartile range, IQR 2.4–9.2), at study 32.5 years (IQR 27.7–38.0) and follow-up 25.5 years (IQR 21.4–30.3). The prevalence of hypertension was comparable in CCS (16.3%) and controls (18.2%). In 12% of CCS and 17.8% of controls, hypertension was undiagnosed. A decreased GFR (2) was associated with hypertension in CCS (OR 3.4, 95% CI 1.4–8.5). Risk factors were abdominal radiotherapy ≥20 Gy and TBI. The ABPM-pilot study (n = 77) showed 7.8% MH, 2.6% white coat hypertension and 20.8% aND.Conclusion: The prevalence of hypertension was comparable among CCS who were treated with potentially nephrotoxic therapies compared to controls, some of which were undiagnosed. Risk factors were abdominal radiotherapy ≥20 Gy and TBI. Hypertension and decreased GFR were associated with CCS. ABPM identified MH and a ND.

Simple Summary We studied survivors of childhood cancer who received cancer treatment that might affect the kidneys and compared them to controls from the general population. We investigated if there was a difference in the occurrence of tubular dysfunction. The tubules are the part of the kidney responsible for reabsorption of needed substances to the blood and the removal of wastes. After around 25 years since their cancer diagnosis, we found that in general there were no differences between survivors and controls, but survivors more often had losses of small proteins in the urine. Yet, some survivors of childhood cancer were found to have an increased risk of tubular dysfunction. Namely, survivors treated with the chemotherapeutic agents ifosfamide, cisplatin or carboplatin. Therefore, these patients should be monitored during their follow-up. The aim of this nationwide cross-sectional cohort study was to determine the prevalence of and risk factors for tubular dysfunction in childhood cancer survivors (CCS). In the DCCSS-LATER 2 Renal study, 1024 CCS (>= 5 years after diagnosis), aged >= 18 years at study, treated between 1963 and 2001 with potentially nephrotoxic therapy (i.e., nephrectomy, abdominal radiotherapy, total body irradiation, cisplatin, carboplatin, ifosfamide, high-dose cyclophosphamide, or hematopoietic stem cell transplantation) participated, and 500 age- and sex-matched participants from Lifelines acted as controls. Tubular electrolyte loss was defined as low serum levels (magnesium < 0.7 mmol/L, phosphate < 0.7 mmol/L and potassium < 3.6 mmol/L) with increased renal excretion or supplementation. A alpha 1-microglobulin:creatinine ratio > 1.7 mg/mmol was considered as low-molecular weight proteinuria (LMWP). Multivariable risk analyses were performed. After median 25.5 years follow-up, overall prevalence of electrolyte losses in CCS (magnesium 5.6%, potassium 4.5%, phosphate 5.5%) was not higher compared to controls. LMWP was more prevalent (CCS 20.1% versus controls 0.4%). LMWP and magnesium loss were associated with glomerular dysfunction. Ifosfamide was associated with potassium loss, phosphate loss (with cumulative dose > 42 g/m(2)) and LMWP. Cisplatin was associated with magnesium loss and a cumulative dose > 500 mg/m(2) with potassium and phosphate loss. Carboplatin cumulative dose > 2800 mg/m(2) was associated with potassium loss. In conclusion, long-term tubular dysfunction is infrequent. Yet, ifosfamide, cisplatin and carboplatin are risk factors.

Purpose: Augmented survival of childhood nephroblastoma and neuroblastoma has increased long-term side effects such as metabolic syndrome (MetS). Risk stratification is difficult after abdominal radiation because waist circumference underestimates adiposity. We aimed to develop a strategy for determining MetS in irradiated survivors using an integrated biomarker profile and vascular ultrasonography. Methods: The NCEP-ATPIII MetS-components, 14 additional serum biomarkers and 9 vascular measurements were assessed in a single-centre cohort of childhood nephroblastoma (n = 67) and neuroblastoma (n = 36) survivors and controls (n = 61). Multivariable regression models were used to study treatment effects. Principal component analysis (PCA) was used to study all biomarkers in a combined analysis, to identify patterns and correlations. Results: After 27.5 years of follow-up, MetS occurred more often in survivors (14%) than controls (3%). Abdominal radiotherapy and nephrectomy, to a lesser extent, were associated with MetS and separate components and with several biomarker abnormalities. PCA of biomarkers revealed a pattern on PC1 from favourable lipid markers (HDL-cholesterol, adiponectin) towards unfavourable markers (triglycerides, LDL-cholesterol, apoB, uric acid). Abdominal radiotherapy was associated with the unfavourable biomarker profile (β = 1.45, P = 0.001). Vascular measurements were not of added diagnostic value. Conclusions: Long-term childhood nephro- and neuroblastoma survivors frequently develop MetS. Additional assessment of biomarkers identified in PCA – adiponectin, LDL, apoB, and uric acid – may be used especially in abdominally irradiated survivors, to classify MetS as alternative for waist circumference. Vascular ultrasonography was not of added value.

There is limited evidence that supports the use of the global longitudinal strain (GLS) in long-term cardiac monitoring of childhood acute lymphoblastic leukemia survivors (CLSs). Our aim was to assess the utility of automated GLS to detect left ventricular systolic dysfunction (LVSD) in long-term CLSs. Asymptomatic and subclinical LVSD were defined as LVEF < 50% and GLS < 18.5%, respectively. Echocardiographic measurements and biomarkers were compared with a control group. Inverse probability weighting was used to reduce confounding. Regression models were used to identify factors associated with LVEF and GLS in the survivors. Ninety survivors with a median follow-up of 18 (11–26) years were included. The prevalence of LVSD was higher using GLS than with LVEF (26.6% vs. 12.2%). The measurements were both reduced as compared with the controls (p < 0.001). There were no differences in diastolic parameters and NT-ProBNP. Survivors were more likely to have Hs-cTnI levels above the detection limit (40% vs. 17.2%, p = 0.006). The dose of anthracycline was associated with LVEF but not with GLS in the survivors. Biomarkers were not associated with GLS or LVEF. In conclusion, LVSD detection using automated GLS was higher than with LVEF in long-term CLSs. Its incorporation into clinical routine practice may improve the surveillance of these patients.

Contains fulltext : 248815.pdf (Publisher’s version ) (Open Access) BACKGROUND: Long-term follow-up (LTFU) care, although endorsed, is not available for the majority of adult survivors of childhood, adolescence and young adult (CAYA) cancer. Barriers to implementation include lack of time, knowledge, personnel and funding. Sustainable solutions are urgently needed to address the needs of CAYA cancer survivors to improve the quality of life and reduce the burden of late effects on survivors, health care systems and society. The European Union-funded PanCareFollowUp project, initiated by the Pan-European Network for Care of Survivors after Childhood and Adolescent Cancer, was established to facilitate the implementation of person-centred survivorship care across Europe. PATIENTS AND METHODS: The PanCareFollowUp Care Intervention was co-developed with survivors as part of the PanCareFollowUp project. It is a person-centred approach to survivorship care, supported by guidelines and with flexibility to adapt to local health care settings. The Care Intervention consists of three steps: (1) previsit completion of a Survivor Questionnaire (by the survivor) and Treatment Summary (by the health care provider [HCP]), (2) a clinic visit including shared decision-making, and (3) a follow-up call to finalise the individualised Survivorship Care Plan. RESULTS: We developed the key components of the PanCareFollowUp Care Intervention: a PanCareFollowUp Survivor Questionnaire, Treatment Summary template, Survivorship Care Plan template, and educational materials for HCPs and survivors. Wide implementation of the PanCareFollowUp Care Intervention will be supported with a freely distributed Replication Manual on completion of the PanCareFollowUp project. CONCLUSIONS: The PanCareFollowUp Care Intervention will support the implementation of person-centred, guideline-based LTFU care in different health care settings across Europe to improve survivors' health and well-being.

There is a growing focus on describing both negative and positive outcomes in the wake of childhood cancer. The purpose of this study was to describe and explore the relationships between posttraumatic stress symptoms (PTSS) and posttraumatic growth (PTG) among children living beyond cancer and one of their parents. As part of a larger online survey, 113 children (Mage at time of study = 15.82 (SD = 4.81); Mage at diagnosis = 5.86 (SD = 4.66)) and one of their parents completed questionnaires assessing PTSS and PTG. Descriptive statistics were used to describe the sample and levels of PTSS and PTG. Data were z-transformed and analyzed using bivariate correlations and t-tests. An actor–partner interdependence model (APIM) was used to test whether children’s and their parents’ PTSS was associated with their own PTG (actor effect) and the others’ PTG (partner effect). PTSS was low and PTG was moderate in this sample relative to scale ranges. There were no significant differences between the children’s and their parents’ PTSS (p = 0.535) or PTG (p = 0.534). Results from the APIM showed no significant actor effects (p = 0.185). A significant overall partner effect (p = 0.020) emerged. Lower PTSS for children was associated with greater PTG for their parents (b = −0.29, p = 0.018), but parent’s PTSS was not associated with children’s PTG (p = 0.434). This sample reported similar levels of PTSS and PTG to that which has been reported in the literature. Children and their parents’ scores on PTSS and PTG measures were not significantly different from one another. Children’s PTSS was negatively associated with their parents PTG, illuminating the ways in which PTSS and PTG may be related in the context of childhood cancer. Exploring family-based strategies to reduce PTSS and enhance PTG may be warranted, though further studies are required.

Simple Summary Over the last twenty years, innovations in the treatment of childhood cancer have increased survival rates. However, female childhood cancer survivors (CCS) are prone to late reproductive aftereffects, including premature ovarian insufficiency (POI). Nonetheless, patients might experience different side effects on fertility according to the type of diagnosed cancer and subsequent treatment. Anti-Müllerian hormone (AMH) is currently used in reproductive medicine to screen for impaired ovarian reserves. However, it does not represent the gold standard in oncofertility. In this systematic review and network meta-analysis of age-matched case–control studies, we evaluate the role of AMH for ovarian reserve screening according to the type of childhood cancer and determined which group of survivors are more prone to POI by means of direct and indirect comparisons among the CCS cohorts. Abstract Background: Female childhood cancer survivors (CCS) might have impaired ovarian reserves, especially after alkylating agents or radiotherapy. The purpose of this systematic review and network meta-analysis is to evaluate the role of serum anti-Müllerian hormone (AMH) for ovarian reserve screening and the risk of premature ovarian insufficiency (POI) according to the subtype of childhood cancer. (2) Methods: PRISMA-NMA guidelines were followed. We carried out a network meta-analysis based on a random effects model for mixed multiple treatment comparisons to rank childhood cancers effects on fertility by surface under the cumulative ranking curve (SUCRA). Studies were selected only if they had an age-matched control group. Quality assessment was performed using Newcastle–Ottawa Scale. The co-primary outcomes were mean AMH levels and the incidence of POI. (3) Results: A total of 8 studies (1303 participants) were included. Women treated for a neuroblastoma during infancy were more likely to be ranked first for impaired AMH levels (SUCRA = 65.4%), followed by mixed CCS (SUCRA = 29.6%). The greatest rates of POI were found in neuroblastoma survivors (SUCRA = 42.5%), followed by acute lymphoid leukemia (SUCRA = 26.3%) or any other neoplasia (SUCR A = 20.5%). (4) Conclusions: AMH represents a trustworthy approach for ovarian reserve screening. Direct and indirect comparisons found no differences in mean AMH levels and POI risk between subtypes of CCS and healthy controls. SUCRA analysis showed that female neuroblastoma survivors were more at risk for reduced serum AMH levels and increased risk of POI.

In the past decades, new cancer treatment approaches for children and adolescents have led to a decrease in recurrence rates and an increase in long-term survival. Recent studies have focused on the evaluation of the late effects on bone of pediatric cancer-related treatments, such as chemotherapy, radiation and surgery. Treatment of childhood cancer can impair the attainment of peak bone mass, predisposing to premature onset of low bone mineral density, or causing other bone side-effects, such as bone quality impairment or avascular necrosis of bone. Lower bone mineral density and microarchitectural deterioration can persist during adulthood, thereby increasing fracture risk. Overall, long-term follow-up of childhood cancer survivors is essential to define specific groups at higher risk of long-term bone complications, identify unrecognized long-term adverse effects, and improve patient care. Children and adolescents with a cancer history should be carefully monitored, and patients should be informed of possible late complications of their previous medical treatment. The International Osteoporosis Foundation convened a working group to review the bone complications of pediatric cancer survivors, outlining recommendations for the management of bone health, in order to prevent and treat these complications.

Childhood cancersurvivors may be differentially impacted by coronavirus disease 2019 (COVID‐19). From April to June of 2020, we examined psychosocial/health concerns in 4148 adult survivors and 571 siblings. Although more survivors reported concerns about getting sick (p = .002) and needing hospitalization (p = .003) in general, survivors and siblings were comparably concerned about being infected with and the consequences of COVID‐19. Cranial radiation was associated with social isolation (relative risk [RR] = 1.3, CI = 1.1–1.7), and central nervous system (CNS) tumors were associated with unemployment due to COVID‐19 (RR = 1.7, CI = 1.2–2.2). Some survivors appear more vulnerable and may require more support to meet health care and vocational needs during COVID‐19, though siblings also perceive substantial risk.

Individuals surviving cancer and brain tumors may experience growth hormone (GH) deficiency as a result of tumor growth, surgical resection and/or radiotherapy involving the hypothalamic-pituitary region. Given the pro-mitogenic and anti-apoptotic properties of GH and insulin-like growth factor-I, the safety of GH replacement in this population has raised hypothetical safety concerns that have been debated for decades. Data from multicenter studies with extended follow-up have generally not found significant associations between GH replacement and cancer recurrence or mortality from cancer among childhood cancer survivors. Potential associations with secondary neoplasms, especially solid tumors, have been reported, although this risk appears to decline with longer follow-up. Data from survivors of pediatric or adult cancers who are treated with GH during adulthood are scarce, and the risk versus benefit profile of GH replacement of this population remains unclear. Studies pertaining to the safety of GH replacement in individuals treated for nonmalignant brain tumors, including craniopharyngioma and non-functioning pituitary adenoma, have generally been reassuring with regards to the risk of tumor recurrence. The present review offers a summary of the most current medical literature regarding GH treatment of patients who have survived cancer and brain tumors, with the emphasis on areas where active research is required and where consensus on clinical practice is lacking.

The population of childhood cancer survivors (CCS) has grown rapidly in recent decades. Although cured of their original malignancy, these individuals are at increased risk of serious late effects, including age-associated complications. An impaired immune system has been linked to the emergence of these conditions in the elderly and CCS, likely due to senescent immune cell phenotypes accompanied by low-grade inflammation, which in the elderly is known as “inflammaging.” Whether these observations in the elderly and CCS are underpinned by similar mechanisms is unclear. If so, existing knowledge on immunosenescent phenotypes and inflammaging might potentially serve to benefit CCS. We summarize recent findings on the immune changes in CCS and the elderly, and highlight the similarities and identify areas for future research. Improving our understanding of the underlying mechanisms and immunosenescent markers of accelerated immune aging might help us to identify individuals at increased risk of serious health complications.

BackgroundAs survivors of childhood cancer age, development of cancer treatment-related chronic health conditions often occur. This study aimed to describe the pattern of chronic prescription medication use and identify factors associated with polypharmacy among survivors of childhood cancer.MethodsThis was a retrospective study conducted at the pediatric oncology long-term follow-up clinic in Hong Kong. Eligible subjects included survivors who were (1) diagnosed with cancer before 18 years old, (2) were at least 3 years post-cancer diagnosis and had completed treatment for at least 30 days, and (3) receiving long-term follow-up care at the study site between 2015 and 2018. Dispensing records of eligible survivors were reviewed to identify medications taken daily for ≥30 days or used on an “as needed” basis for ≥6 months cumulatively within the past 12-month period. Polypharmacy was defined as the concurrent use of ≥5 chronic medications. Multivariable log-binomial modeling was conducted to identify treatment and clinical factors associated with medication use pattern and polypharmacy.ResultsThis study included 625 survivors (mean current age = 17.9 years, standard deviation [SD] = 7.2 years) who were 9.2 [5.2] years post-treatment. Approximately one-third (n = 219, 35.0%) of survivors were prescribed at least one chronic medication. Frequently prescribed medication classes include systemic antihistamines (26.5%), sex hormones (19.2%), and thyroid replacement therapy (16.0%). Overall prevalence of polypharmacy was 5.3% (n = 33). A higher rate of polypharmacy was found in survivors of CNS tumors (13.6%) than in survivors of hematological malignancies (4.3%) and other solid tumors (5.3%) (P = .0051). Higher medication burden was also observed in survivors who had undergone cranial radiation (RR = 6.31; 95% CI = 2.75–14.49) or hematopoietic stem-cell transplantation (HSCT) (RR = 3.53; 95% CI = 1.59–7.83).ConclusionAlthough polypharmacy was observed in a minority of included survivors of childhood cancer, chronic medication use was common. Special attention should be paid to survivors of CNS tumors and survivors who have undergone HSCT or cranial radiation. These individuals should be monitored closely for drug–drug interactions and adverse health outcomes that may result from multiple chronic medications, particularly during hospitalization in an acute care setting.

Telemedicine can potentially meet objectives of long‐term follow‐up care (LTFU) for childhood cancer survivors (CCS) while reducing barriers. We surveyed providers at our institution about their satisfaction with video‐conference virtual visits (VV) with 81 CCS during COVID‐19 restrictions. The same 81 CCS (or parent proxies) were surveyed about their experience, of which 47% responded. Providers and CCS were highly satisfied with VV (86% and 95% “completely/very satisfied,” respectively). CCS rated VV “as/nearly as” helpful as in‐person visits (66%) and 82% prefer VV remain an option postpandemic. High levels of survivor and provider satisfaction with VV support ongoing investigation into implementation for LTFU.

In women with premature ovarian insufficiency (POI), hormonal therapy (HT) is indicated to decrease the risk of morbidity and to treat symptoms related to prolonged hypoestrogenism. While general recommendations for the management of HT in adults with POI have been published, no systematic suggestions focused on girls, adolescents and young women with POI following gonadotoxic treatments (chemotherapy, radiotherapy, stem cell transplantation) administered for pediatric cancer are available. In order to highlight the challenging issues specifically involving this cohort of patients and to provide clinicians with the proposal of practical therapeutic protocol, we revised the available literature in the light of the shared experience of a multidisciplinary team of pediatric oncologists, gynecologists and endocrinologists. We hereby present the proposals of a practical scheme to induce puberty in prepubertal girls and a decisional algorithm that should guide the clinician in approaching HT in post-pubertal adolescents and young women with iatrogenic POI.

Background Emerging research suggests that survivors of childhood and adolescent cancers are at risk for morbidity and mortality associated with air pollutants. However, caregiver perceptions of the effects of air pollution are unknown. Thus, to address this gap we described caregivers’ perceptions of air pollution’s impact on general population health and specifically on childhood cancer survivors, and caregivers’ air pollution information-seeking and exposure reduction behaviors. Methods Participants were Utah residents, ≥18 years, and caregiver of a childhood cancer survivor who had completed treatment. Semi-structured interviews were conducted with caregivers to describe their perspectives on air quality, how air pollution impacts health (general population and survivor health), and their information seeking and exposure reduction behaviors. Interviews were recorded, transcribed, and analyzed through two rounds of structured coding. Results Caregivers (N = 13) were non-Hispanic white and primarily females (92.3%) between 30 and 49 years old (46.2%). Most families lived within the Wasatch Front (69.2%), the main metropolitan of Utah. Two categories emerged pertaining to caregiver’s perceptions of air pollution: 1) Limited awareness about the health effects of air pollution, and 2) Unsuccessful information seeking and minimal exposure reduction behaviors. All caregivers held negative perceptions of air pollution in Utah, but most were unaware of how pollution affects health. While some families limited air pollution exposure by avoiding outdoor activity or physically leaving the region, few practiced survivor-specific exposure reduction. Nearly half of caregivers worried about potential effects of air pollution on survivor health and wanted more information. Conclusions Despite negative perceptions of air pollution, caregivers were divided on whether air pollution could impact survivor health. Few caregivers engaged in exposure reduction for their cancer survivor. As air pollution levels increase in the U.S., continued research on this topic is essential to managing cancer survivor respiratory and cardiovascular health.

Contains fulltext : 193305.pdf (Publisher’s version ) (Open Access) INTRODUCTION: The variability in late toxicities among childhood cancer survivors (CCS) is only partially explained by treatment and baseline patient characteristics. Inter-individual variability in the association between treatment exposure and risk of late toxicity suggests that genetic variation possibly modifies this association. We reviewed the available literature on genetic susceptibility of late toxicity after childhood cancer treatment related to components of metabolic syndrome, bone mineral density, gonadal impairment and hearing impairment. METHODS: A systematic literature search was performed, using Embase, Cochrane Library, Google Scholar, MEDLINE, and Web of Science databases. Eligible publications included all English language reports of candidate gene studies and genome wide association studies (GWAS) that aimed to identify genetic risk factors associated with the four late toxicities, defined as toxicity present after end of treatment. RESULTS: Twenty-seven articles were identified, including 26 candidate gene studies: metabolic syndrome (n=6); BMD (n=6); gonadal impairment (n=2); hearing impairment (n=12) and one GWAS (metabolic syndrome). Eighty percent of the genetic studies on late toxicity after childhood cancer had relatively small sample sizes (n

Simple Summary Around 80% of children treated for childhood cancer become long-term survivors. Although chemotherapy and radiotherapy improve survival of these patients, they cause a low-grade chronic inflammation (inflamm-aging) which induces premature aging processes and vital organ failure, a condition known as frailty. Understanding frailty’s biological and molecular mechanisms and identifying inflamm-aging key biomarkers in childhood cancer survivors could be useful to facilitate the screening of comorbidities and to understand whether treatments, used to counteract inflamm-aging, may prevent side effects. Abstract Anti-cancer treatments improve survival in children with cancer. A total of 80% of children treated for childhood cancer achieve 5-year survival, becoming long-term survivors. However, they undergo several chronic late effects related to treatments. In childhood cancer survivors a chronic low-grade inflammation, known as inflamm-aging, is responsible for frailty, a condition characterized by vital organ failure and by premature aging processes. Inflamm-aging is closely related to chemotherapy and radiotherapy, which induce inflammation, accumulation of senescent cells, DNA mutations, and the production of reactive oxygen species. All these conditions are responsible for the onset of secondary diseases, such as osteoporosis, cardiovascular diseases, obesity, and infertility. Considering that the pathobiology of frailty among childhood cancer survivors is still unknown, investigations are needed to better understand frailty’s biological and molecular processes and to identify inflamm-aging key biomarkers in order to facilitate the screening of comorbidities and to clarify whether treatments, normally used to modulate inflamm-aging, may be beneficial. This review offers an overview of the possible biological mechanisms involved in the development of inflamm-aging, focusing our attention on immune system alteration, oxidative stress, cellular senescence, and therapeutic strategies.

Purpose: Sun-exposure can cause health problems, including melanoma and nonmelanoma skin cancer, especially in Australia where the incidence of skin cancer is particularly high. Childhood cancer survivors (CCSs) have an augmented risk due to previous cancer history and treatment. Despite recommendations advising sun protection, CCSs may be placing themselves at risk. We considered daily summer sun-exposure in an Australian cohort of CCSs and in community reference groups, and identified factors associated with sun-exposure in these populations. Methods: Summer sun-exposure data were collected on 471 CCSs (119 parents of survivors aged < 16, and 352 survivors aged ≥16) and a reference group of 470 participants from the community (155 parents of children aged < 16, and 355 adults aged ≥16). Survivors completed paper questionnaires and the reference groups completed an online survey. Medical records confirmed survivors’ clinical information. Ordinal logistic regressions identified factors associated with daily summer sun-exposure. Results: More daily summer sun-exposure was reported by both parents reporting for the young survivors (U = 5,522.5, P < .001; U = 31,412, P < .001) and by older survivors (U = 5,039.5, P < .001; U = 29,913, P < .001). Among younger participants greater sun-exposure was associated with being a CCS, while in older participants, greater sun-exposure was associated with being a CCS, a male, smoker/ex- smoker and was also increased in those with more sensitive skin reaction to sunlight. Focusing only on the CCSs, when treatments were considered, none statistically predicted sun-exposure in young CCSs while older CCSs who received radiotherapy were less likely to spend excess time in the sun. Conclusion: CCSs have sun-exposure at times of day when sun-related skin damage is highest, despite advice to avoid highest risk times. This data can inform sun-protection programs and lifestyle advice aimed at ameliorating the potential increased risk of skin cancer in CCSs.

Gonadal dysfunction and infertility are major endocrinological late effects among childhood cancer survivors. Chemotherapy and radiation have gonadotoxic effects and diminish the ovarian reserve. The serum concentration of anti-Müllerian hormone (AMH) is a useful marker of ovarian reserve in survivors. We conducted a longitudinal study to investigate the variations of AMH in evaluating the acute and chronic effects of cancer therapy on the ovary. Three young female patients with different hematological diseases were registered, and their medical records were reviewed. Patient 1 with myelodysplastic syndrome received reduced-intensity hematopoietic stem cell transplantation (HSCT) at 10 yr of age. Breast development and menarche occurred spontaneously after HSCT; however, AMH level became undetectable and gonadotropin did not increase. Patient 2 with acute lymphoblastic leukemia had been receiving chemotherapy since 11 yr of age. AMH level became undetectable but increased after chemotherapy and was associated with regular menstruation. Patient 3 with acute myeloid leukemia received chemotherapy at 13 yr of age and myeloablative HSCT at 14 yr of age. AMH level became undetectable after HSCT, and the patient developed amenorrhea. These different patterns in the recovery phase demonstrated that the AMH level immediately after the end of cancer therapy is inappropriate for the evaluation of the ovarian reserve.

Objective To extend the limited research on psychosexual development among childhood cancer survivors, by not only focusing on the prevalence and age of milestone attainment, but also survivors' attitudes toward the timing of reaching such milestones. Methods Adult survivors of childhood cancer (N = 90; Mage = 29.8, SD = 5.2), recruited from a US pediatric institution, completed online surveys indicating whether they had reached 5 milestones of psychosexual development (ie, first kiss, first boy-/girlfriend, first physical intimacy, sexual debut, first time in love), age at attainment, and perceptions about the timing (ie, right time, wished it had happened earlier, wished they had waited). Results Almost all survivors had reached each milestone (≥90%), except for sexual debut (83.3%). Survivors reported their first kiss as the earliest milestone at age 14.6 (N = 82, 92%) and falling in love as the latest milestone at age 18.8 (N = 80; 90%). This timing did not differ by sex/cancer-specific factors. Most survivors (~60%) felt they reached each milestone at the right time. Compared with US normative data, both male and female survivors were less likely to have experienced their sexual debut and were approximately 1.5 years older at sexual debut. Nevertheless, 59% of survivors felt that this timing was right and 31% wished they had waited longer. Conclusions This is the first study to demonstrate that although childhood cancer survivors may delay some aspects of psychosexual development, most are satisfied with this timing. Research and clinical practice should emphasize survivors' perceptions/satisfaction toward psychosexual development rather than focusing only on normative milestone attainment.

Background: Improved risk stratification, more effective therapy and better supportive care have resulted in survival rates after childhood cancer of around 80% in developed countries. Treatment however can be harsh, and three in every four childhood cancer survivors (CCS) develop at least one late effect, such as gonadal impairment. Gonadal impairment can cause involuntary childlessness, with serious consequences for the well-being of CCS. In addition, early menopause increases the risk of comorbidities such as cardiovascular disease and osteoporosis. Inter-individual variability in susceptibility to therapy related gonadal impairment suggests a role for genetic variation.Currently, only one candidate gene study investigated genetic determinants in relation to gonadal impairment in female CCS; it yielded one single nucleotide polymorphism (SNP) that was previously linked with the predicted age at menopause in the general population of women, now associated with gonadal impairment in CCS. Additionally, one genome wide association study (GWAS) evaluated an association with premature menopause, but no GWAS has been performed using endocrine measurements for gonadal impairment as the primary outcome in CCS.Methods: As part of the PanCareLIFE study, the genetic variability of chemotherapy induced gonadal impairment among CCS will be addressed. Gonadal impairment will be determined by anti-Müllerian hormone (AMH) levels or alternatively by fertility and reproductive medical history retrieved by questionnaire. Clinical and genetic data from 837 non-brain or non-bilateral gonadal irradiated long-term CCS will result in the largest clinical European cohort assembled for this late-effect study to date. A candidate gene study will examine SNPs that have already been associated with age at natural menopause and DNA maintenance in the general population. In addition, a GWAS will be performed to identify novel allelic variants. The results will be validated in an independent CCS cohort.Discussion: This international collaboration aims to enhance knowledge of genetic variation which may be included in risk prediction models for gonadal impairment in CCS.

Estimates of the number of childhood cancers with a genetic basis range from 5–8.5% found in germline samples to 29% based on clinical criteria. Family history-taking practice is a fundamental first step in detecting at risk individuals and families. This study focused on Li-Fraumeni Syndrome (LFS), a highly penetrant cancer syndrome. Reported family history in a cohort of 648 of cancer survivor cohort (CCS) was examined. Eligible CCS were: (i) aged up to 14 years at diagnosis; (ii) more than 5 years postdiagnosis; (iii) treated for a childhood cancer at the study hospitals in NSW, Australia; (iv) in remission for more than 3 years. CCS completed self-administered questionnaires. Medical records confirmed diagnosis and treatment-related information. Our findings reveal an increased cancer risk among sibling and relatives of CCS. 91% of siblings diagnosed with cancer were diagnosed under the age of 40 and about 30% diagnosed under the aged of 15 revealing a 5- (RR = 5.1; 95% CI, 3.3–7.9) and 44-fold (RR = 44.6; 95% CI, 18.4–108.3) increased risked of cancer compared with the Australian population, respectively. About 2% of CCS reported that they had been diagnosed with a genetic cancer syndrome. However, 11% of survivors described a family history pattern which met Chompret criteria for screening for TP53 mutations associated with LFS. Our data suggests that familial cancer predispositions may be initially overlooked. Aperiodic and accurate ascertainment of family cancer history of childhood cancer patients and survivors is therefore recommended.

Purpose Risk-stratified life-long follow-up care is recommended for adult childhood cancer survivors (CCS) to ensure appropriate prevention, screening, and management of late effects. The identification of barriers to long-term follow-up (LTFU), particularly in varying healthcare service contexts, is essential to develop and refine services that are responsive to survivor needs. We aimed to explore CCS and healthcare professionals (HCP) perspectives of healthcare system factors that function as barriers to LTFU in British Columbia, Canada. Methods We analyzed data from 43 in-depth interviews, 30 with CCS and 13 with HCP, using qualitative thematic analysis and constant comparative methods. Results Barriers to accessible, comprehensive, quality LTFU were associated with the following: (1) the difficult and abrupt transition from pediatric to adult health services, (2) inconvenient and under-resourced health services, (3) shifting patient-HCP relationships, (4) family doctor inadequate experience with late effects management, and (5) overdue and insufficient late effects communication with CCS. Conclusions Structural, informational, and interpersonal/relational healthcare system factors often prevent CCS from initially accessing LTFU after discharge from pediatric oncology programs as well as adversely affecting engagement in ongoing screening, surveillance, and management of late effects. Implications for Cancer Survivors Understanding the issues faced by adult CCS will provide insight necessary to developing patient-centered healthcare solutions that are key to accessible, acceptable, appropriate, and effective healthcare.

Purpose Long-term childhood cancer survivors may be at increased risk for poor social outcomes as a result of their cancer treatment, as well as physical and psychological health problems. Yet, important challenges, namely social isolation, are not well understood. Moreover, survivors' perspectives of social isolation as well as the ways in which this might evolve through young adulthood have yet to be investigated. The purpose of this research was to describe the trajectories of social isolation experienced by adult survivors of a childhood cancer. Methods Data from 30 in-depth interviews with survivors (9 to 38 years after diagnosis, currently 22 to 43 years of age, 60 % women) were analyzed using qualitative, constant comparative methods. Results Experiences of social isolation evolved over time as survivors grew through childhood, adolescence and young adulthood. Eleven survivors never experienced social isolation after their cancer treatment, nor to the present day. Social isolation among 19 survivors followed one of three trajectories; (1) diminishing social isolation: it got somewhat better, (2) persistent social isolation: it never got better or (3) delayed social isolation: it hit me later on. Conclusions Knowledge of when social isolation begins and how it evolves over time for different survivors is an important consideration for the development of interventions that prevent or mitigate this challenge. Implications for Cancer Survivors Assessing and addressing social outcomes, including isolation, might promote comprehensive long-term follow-up care for childhood cancer survivors.

Purpose Currently, 80 % of children diagnosed with cancer will be cured. However, many of these survivors go on to develop long-term health problems or late effects related to their previous cancer and therapy and require varying degrees of lifelong follow-up care. The purpose of this study was to identify the different ways that adult survivors of childhood cancer manage their medical and psychological challenges. Methods Data from in-depth interviews with 30 adult survivors of a childhood cancer (9 to 38 years after diagnosis, currently 22 to 43 years of age, 60 % women) were analyzed using qualitative, thematic narrative analysis methods. Results The survivors had not expected the medical, psychological, and social challenges that arose over time and that often remained unresolved. Five narrative themes revealed distinct ways that survivors managed their health challenges: (1) trying to forget cancer, (2) trusting the system to manage my follow-up care, (3) being proactive about my health, (4) stumbling from one problem to the next, and (5) struggling to find my way. Conclusions Variation exists in the ways in which childhood cancer survivors frame their health, their perceived significance of health challenges, strategies used to manage health, interactions with healthcare professionals and the health system, and parental involvement. Implications for Cancer Survivors This research provides novel insights that can be used to inform the development of patient-centered health services that promote the assessment and tailoring of care to the diverse ways survivors enact their agency, as well as their psychoeducational coping styles, therapeutic relationship needs, and information needs.