Siblings Of Childhood Cancer Survivors Research Articles

PHYSICAL DEVELOPMENT CHARACTERISTICS OF CHILDREN TREATED FOR BRAIN TUMOR AND HEALTHY SIBLINGS OF CANCER PATIENTS (A PILOT STUDY)

Introduction. In families with such a child his/her siblings often live, whose development will be affected by the current stressful situation for all family members. Siblings of cancer patients appear to be a psychological vulnerable group, as they have low self-esteem, fear of death and loneliness, anxiety, anger. The study aimed to assess the level of physical development (PD) of children, treated for brain tumors, and siblings of childhood cancer survivors, in relation to stressful circumstances of living in a family with an oncological relative. Material and methods. The study involved children aged 6 to 17 years who had completed treatment for a brain tumor (BT), as well as siblings of children with various cancer types. We measured 27 boys and 36 girls with BT, the sample of siblings included 26 boys and 31 girls. To assess the compliance of the level of PD of children of both groups with age standards, for each child of a given gender and age a centile corridor was determined by body length (BL) and BMI, and individual values of signs were ranked in Z-points, using WHO standards. A detailed questionnaire was conducted to assess some characteristics of a child's family environment. Statistics analysis was performed with Statistica 12 software package. Results. For a group of children with BT, a significant shift in the distribution of PD scores along body length and BMI towards low values was found, for a group of siblings, the opposite picture was obtained – the distribution of body length and BMI scores shifted to the higher values. It has been shown that higher developmental scores in body length are significantly more common in children who are older siblings in relation to patients with BT. Conclusion. Thus, brain tumor treatment affects not only the physical development of a child, but is a factor contributing to the growth processes of the body of siblings of children with BT. Siblings of cancer patients represent a special group of children, which indicates the need to take into account the special needs of all their family members in the process of treatment and rehabilitation, as well as the incorrectness of using siblings as a control group in scientific research.

Financial hardship among siblings of long-term survivors of childhood cancer: A Childhood Cancer Survivor Study report.

Siblings of children with cancer may experience adverse household economic consequences, but their financial outcomes in adulthood are unknown. A total of 880 siblings (aged 18-64years) of adult-aged childhood cancer survivors were surveyed to estimate the prevalence of financial hardship by three established domains (behavioral, material, and psychological). For individual financial hardship items matching the contemporaneous National Health Interview Survey or Behavioral Risk Factor Surveillance System, siblings were compared with the general population by calculating adjusted prevalence odds ratios (ORs) to sample-weighted responses. Multivariable logistic regression models examined associations between sibling characteristics and each hardship domain and between sibling hardship and survivors' cancer/treatment characteristics. Behavioral, material, and psychological hardship was reported by 24%, 35%, and 28%, respectively. Compared with national survey respondents, siblings were more likely to report worries about medical bills (OR, 1.14; 95% confidence interval [CI], 1.06-1.22), difficulty affording nutritious foods (OR, 1.79; 95% CI, 1.54-2.07), and forgoing needed medical care (OR, 1.38; 95% CI, 1.10-1.73), prescription medications (OR, 2.52; 95% CI, 1.99-3.20), and dental care (OR, 1.34; 95% CI, 1.15-1.57) because of cost. Sibling characteristics associated with reporting financial hardship in one or more domains included female sex, older age, chronic health conditions, lower income, not having health insurance, high out-of-pocket medical expenditures, and nonmedical/nonhome debt. No survivor cancer/treatment characteristics were associated with sibling financial hardship. Adult siblings of childhood cancer survivors were more likely to experience financial hardship compared with the general population. Childhood cancer may adversely affect entire households, with potentially lasting implications.

Financial hardship among siblings of long-term survivors of childhood cancer: A Childhood Cancer Survivor Study (CCSS) report.

10048 Background: Siblings of children with cancer may experience adverse household economic and psychosocial impacts during and after treatment. While many long-term survivors of childhood cancer experience financial hardship, financial outcomes of siblings in later adulthood are unknown. Methods: We surveyed randomly selected nearest age siblings of survivors (aged 18-64y) enrolled in the CCSS to estimate the prevalence of financial hardship, using 20 items adapted from validated national surveys grouped into 3 domains (material, psychological, behavioral). We calculated the prevalence of reporting any hardship within each domain. Multivariable logistic regression estimated associations between sibling sociodemographic characteristics and each domain of hardship. For individual financial hardship items with a matching item in the contemporaneous National Health Interview Survey (n = 21,271) or Behavioral Risk Factor Surveillance System (n = 259,901), we compared siblings with national survey respondents aged 18-64, calculating adjusted prevalence ratios to sample-weighted responses, adjusted for sex, race, ethnicity, household income, education, and marital status. Results: 1,008 siblings participated (of 1,590 approached; 63%) with median age of 46y (IQR 39–53y). Siblings were 57% female, 89% non-Hispanic White, 69% college-educated, and 74% married. The prevalence of reporting any material, psychological, and behavioral hardship among siblings was 34%, 28%, and 23%, respectively. Sibling factors associated with reporting material financial hardship included: female sex (OR 1.7, 95% CI 1.2–2.4), age (30–39y vs 21–29y: OR 2.61, 95% CI 1.1–6.5), household income ($50,000–$74,999 vs ≥$75,000: OR 2.3, 95% CI 1.4–3.6), lack of health insurance (OR 2.4, 95% CI 1.1–5.1), presence of severe/disabling chronic medical conditions (OR 1.7, 95% CI 1.1–2.8), out-of-pocket medical expenses ≥10% of income (vs < 10%: OR 4.9, 95% CI 1.9–12.3), and increased non-medical/non-home debt (for example, $25,000–$49,999 vs none: OR 5.2, 95% CI 3.2–8.6). Associations with psychological and behavioral hardship were similar. Compared with national survey respondents, siblings were more likely to report financial hardship on several individual items: difficulty paying medical bills (PR 1.2, 95% CI 1.0–1.4), worries about bills (PR 1.1, 95% CI 1.1–1.2), foregoing medical care (PR 1.3, 95% CI 1.0–1.6) and dental care (PR 1.4, 95% CI 1.2–1.6) due to cost, and worries about affording nutritious foods (PR 1.7, 95% CI 1.5–2.0). Conclusions: Adult siblings of childhood cancer survivors are more likely to experience multiple aspects of financial hardship than US adults in the general population, suggesting that childhood cancer may have life-long impact on siblings. Increased support for families across the trajectory of cancer treatment may help prevent or reduce financial hardship.

Health-related and cancer risk concerns among siblings of childhood cancer survivors: a report from the Childhood Cancer Survivor Study (CCSS).

To characterize the prevalence and predictors of concerns regarding future health and cancer risk among siblings of childhood cancer survivors. This study reports longitudinal data (baseline and follow-up) from 3969 adult siblings (median age = 29 [range 18-56] years) of long-term survivors of childhood cancer (median time since diagnosis 19.6 [9.6-33.8] years). Self-reported future health and cancer risk concerns (concerned vs not concerned) were assessed. Demographics and health data reported by both the siblings and their matched cancer survivors were examined as risk factors for health concerns using multivariable logistic regression. Percentage of siblings reporting future health and cancer risk concerns, respectively, decreased across decade of survivors' diagnosis: 1970s (73.3%; 63.9%), 1980s (67.2%; 62.6%), and 1990s (45.7%; 52.3%). Risk factors associated with future health concerns included sibling chronic health conditions (grade 2 Odds Ratio [OR]=1.57, 95% CI: 1.12-2.20; grades 3-4 OR=1.86, 95% CI: 1.18-2.94; compared to less than grade 2). Risk factors associated with future cancer concerns included sibling chronic health conditions (grade 2 OR=1.43, 95% CI: 1.05-1.94; grades 3-4 OR=1.64, 95% CI: 1.09-2.47; compared to less than grade 2). Sibling concerns regarding future health and cancer have diminished in recent decades. There are subgroups of siblings that are at-risk for future health and cancer risk concerns. Routine screening of concerns in at-risk siblings of survivors of childhood cancer may benefit the siblings of cancer survivors. These individuals may benefit from early interventions during diagnosis and treatment of their siblings.

Health and cancer concerns among siblings of childhood cancer survivors: A report from the Childhood Cancer Survivor Study (CCSS).

12117 Background: Siblings of long-term survivors of childhood cancer can be at risk for persistent concerns regarding their future health and risk for cancer. We examined self-perceived future health and cancer risk concerns among such siblings. Methods: 3,969 siblings (median age 29 [range 18–56] years) of 5+ year matched pair cancer survivors (n= 3,969; age 25 [6–48] years; time since diagnosis 19.6 [9.6-33.8] years) in the CCSS self-reported physical/psychosocial problems, including concerns regarding future health and cancer risk (dichotomized as concerned vs not concerned). Chronic health conditions (CHC) were graded using the Common Terminology Criteria for Adverse Events system: mild (grade 1), moderate (grade 2), severe/disabling (grade 3),or life-threatening (grade 4). Sibling demographics, their matched survivor’s diagnosis, era and treatment components, complications (death, relapse, disfigurement) as well as self-reported health status and CHCs for siblings and survivors were examined as potential risk factors for concern using multivariable logistic regression. Adjusted odds ratios (OR) and 95% confidence intervals (CI) are reported. Results: The prevalence of siblings reporting concerns regarding health and cancer risk decreased based on decades of matched survivor diagnosis: 1970-79 (73.3%; 63.9%), 1980-89 (67.2%; 62.6%), 1990-99 (45.7%; 52.3%). Risk factors for concerns included sibling poor/fair current health (future health OR 3.65, 95% CI 2.37-5.62; cancer risk OR 1.54, 1.12-2.13) compared to good/very good/excellent health. Sibling grade 2 (future health OR 1.46, 1.23-1.74; cancer risk OR 1.20, 1.01-1.42) or grade 3-4 CHCs (future health OR 1.37, 1.09-1.71; cancer risk OR 1.28, 1.03-1.58) were associated with greater concerns compared to those with less than grade 2 CHCs. Survivor treatment with chemotherapy/radiation was associated with elevated cancer risk concerns (OR 1.51, 1.13-2.02) compared to surgery/no therapy. Siblings of survivors with grade 3-4 CHCs (OR 1.35, 1.12-1.63) had greater future health concerns compared to those with less than grade 2 CHCs. Sibling bereavement was a risk factor for future health (OR 1.45, 1.04-2.03) and cancer risk (OR 1.44, 1.05-1.99) concerns. Conclusions: The prevalence of sibling concerns regarding future health and cancer have diminished in more recent decades. Subgroups of siblings are at-risk for concerns over future health and cancer risk, partially determined by medical characteristics of their survivor and their own health status.

Long-term psychological and educational outcomes for survivors of neuroblastoma: A report from the Childhood Cancer Survivor Study.

Neuroblastoma survivors may be at elevated risk for psychological impairments because of their young age at diagnosis and neurotoxic treatment, but this is not well described. A total of 859 ≥5-year survivors of neuroblastoma younger than 18 years (diagnosed in 1970-1999), who had a median age at diagnosis of 0.8 years (range: 0.0-7.3 years) and a median follow-up of 13.3 years (range: 8.0-17.9 years), were compared with 872 siblings of childhood cancer survivors who were younger than 18 years with the parent-reported Behavior Problem Index (BPI) for psychological functioning. Age- and sex-adjusted multivariate log-binomial models were used to identify factors associated with impairment in BPI domains (scores worse than the sibling 10th percentile). The impact of psychological impairment on educational outcomes was examined among survivors. Compared with siblings, neuroblastoma survivors had an increased prevalence of impairment in the domains of anxiety/depression (19% vs 14%; P = .003), headstrong behavior (19% vs 13%; P < .001), attention deficits (21% vs 13%; P < .001), peer conflict/social withdrawal (26% vs 17%; P < .001), and antisocial behavior (16% vs 12%; P = .01). Common treatment exposures (vincristine, cisplatin, and retinoic acid) were not associated with impairment. Having 2 or more chronic health conditions predicted impairment in 4 domains (P < .001). Specifically, pulmonary disease predicted impairment in all 5 domains (P ≤ .004). Endocrine disease (P ≤ .004) and peripheral neuropathy (P ≤ .02) each predicted impairment in 3 domains. Psychological impairment was associated with special education service usage and educational attainment less than college. Neuroblastoma survivors are at elevated risk for psychological impairment, which is associated with special education service usage and lower adult educational attainment. Those with chronic health conditions may represent a high-risk group for targeted screening and intervention. Cancer 2018. © 2018 American Cancer Society.

Health-Related Quality of Life of Young Adults Treated with Recombinant Human Growth Hormone during Childhood.

BackgroundSince recombinant human growth hormone (rhGH) became available in 1985, the spectrum of indications has broadened and the number of treated patients increased. However, long-term health-related quality of life (HRQoL) after childhood rhGH treatment has rarely been documented. We assessed HRQoL and its determinants in young adults treated with rhGH during childhood.Methodology/Principal FindingsFor this study, we retrospectively identified former rhGH patients in 11 centers of paediatric endocrinology, including university hospitals and private practices. We sent a questionnaire to all patients treated with rhGH for any diagnosis, who were older than 18 years, and who resided in Switzerland at time of the survey. Three hundred participants (58% of 514 eligible) returned the questionnaire. Mean age was 23 years; 56% were women; 43% had isolated growth hormone deficiency, or idiopathic short stature; 43% had associated diseases or syndromes, and 14% had growth hormone deficiency after childhood cancer. Swiss siblings of childhood cancer survivors and the German norm population served as comparison groups. HRQoL was assessed using the Short Form-36. We found that the Physical Component Summary of healthy patients with isolated growth hormone deficiency or idiopathic short stature resembled that of the control group (53.8 vs. 54.9). Patients with associated diseases or syndromes scored slightly lower (52.5), and former cancer patients scored lowest (42.6). The Mental Component Summary was similar for all groups. Lower Physical Component Summary was associated with lower educational level (coeff. -1.9). Final height was not associated with HRQoL.Conclusions/SignificanceIn conclusion, HRQoL after treatment with rhGH in childhood depended mainly on the underlying indication for rhGH treatment. Patients with isolated growth hormone deficiency/idiopathic short stature or patients with associated diseases or syndromes had HRQoL comparable to peers. Patients with growth hormone deficiency after childhood cancer were at high risk for lower HRQoL. This reflects the general impaired health of this vulnerable group, which needs long-term follow-up.

Tobacco Use Among Siblings of Childhood Cancer Survivors: A Report From the Childhood Cancer Survivor Study.

Having a brother or sister with childhood cancer may influence health behaviors during adulthood. The aim of this study was to compare tobacco use in siblings of survivors with peers and to identify factors associated with sibling tobacco use. A retrospective cohort study was conducted using adult siblings (N = 1,974) of 5+ year cancer survivors in the Childhood Cancer Survivor Study (CCSS) and participants (N = 24,105, weighted to match CCSS) in the 2007 National Health Interview Survey. Self-reported tobacco use, sociodemographic, and cancer-related risk factors were analyzed. Siblings were equally likely to have ever smoked compared to their peers (odds ratio [OR] 1.02, 95% confidence interval [CI] 0.93-1.12). Siblings were less likely to be current smokers (OR 0.83, 95%CI 0.73-0.94), but more likely to be former smokers (OR 1.21, 95%CI 1.08-1.35). Siblings with low education were more likely to ever smoke (OR 1.51, 95%CI 1.15-2.00) and be current smokers (OR 1.67, 95%CI 1.24-2.26) compared to their peers. Among siblings, risk factors for current tobacco use included the following: low income <$20,000 (OR 1.66, 95%CI 1.09-2.54), low education (OR 6.68, 95%CI 4.07-10.97), psychological distress (OR 5.36, 95%CI 2.21-13.02), and heavy alcohol use (OR 3.68, 95%CI 2.50-5.41). Siblings of survivors take up smoking at similar rates to their peers, but are more likely to quit. Efforts are needed to address disparities by providing greater psychosocial support and education for the lowest socioeconomic status families facing childhood cancer.

School Attendance in Childhood Cancer Survivors and Their Siblings

To investigate school absenteeism among childhood cancer survivors and their siblings and examine factors related to absenteeism in survivors. A cross-sectional study was conducted among consecutive cancer survivors attending a large pediatric cancer survivor clinic. Absenteeism rates were obtained for survivors and their closest in age sibling from school report cards. Absenteeism was compared with a population control group of 167752 students using 1-sample t tests. The Child Vulnerability Scale, Pediatric Quality of Life Inventory, and Behavior Assessment System for Children were administered to survivors. Univariate and multiple regression analyses assessed variables associated with days absent. One hundred thirty-one survivors (median age at assessment: 13.4 years, range 8.0-19.2; median age at diagnosis: 9.4 years, range 4.3-17.3) and 77 siblings (median age at assessment: 13 years, age range 7-18) participated. Survivors and siblings missed significantly more school days than the population control group (mean ± SD: 9.6 ± 9.2 and 9.9 ± 9.8 vs 5.0 ± 5.6 days, respectively, P < .0001). Among matched survivor-sibling pairs (N = 77), there was no difference in absenteeism (9.6 ± 9.2 vs 9.9 ± 9.8 days, P = .85). Absenteeism in survivors was significantly associated with a low Pediatric Quality of Life Inventory Physical Health Summary Score (P = .01). Parents' perception of their child's vulnerability and emotional and social functioning were not associated with absenteeism. Childhood cancer survivors and siblings miss more school than the general population. The only predictor of absenteeism in survivors is poor physical quality of health. More research should be devoted to school attendance and other outcomes in siblings of childhood cancer survivors.

Cancer Prevention and Screening Practices of Siblings of Childhood Cancer Survivors: A Report from the Childhood Cancer Survivor Study

To compare the skin and breast/cervical cancer prevention/screening practices of adult siblings of childhood cancer survivors with controls and to identify modifying factors for these practices. Cross-sectional, self-report data from 2,588 adult siblings of 5+ year survivors of childhood cancer were analyzed to assess cancer prevention/screening practices. Two age, sex, and race/ethnicity-matched samples (N = 5,915 and N = 37,789) of the Behavioral Risk Factor Surveillance System participants served as the comparison populations. Sociodemographic and cancer-related data were explored as modifying factors for sibling cancer prevention/screening practices through multivariable logistic regression. Compared with controls, siblings were more likely to practice skin cancer prevention behaviors: use of protective clothing [OR, 2.85; 95% confidence interval (CI), 2.39-3.39], use of shade (OR, 2.11; 95% CI, 1.88-2.36), use of sunscreen (OR, 1.27; 95% CI, 1.14-1.40), and wearing a hat (OR, 1.77; 95% CI, 1.58-1.98). No differences were noted for breast/cervical cancer screening including mammography and Pap testing. Having less than a high school education and lack of health insurance were associated with diminished cancer prevention/screening behaviors. Survivor diagnosis, treatment intensity, adverse health, chronic health conditions, and second cancers were not associated with sibling cancer prevention/screening behaviors. Siblings of cancer survivors report greater skin cancer prevention practices when compared with controls; however, no differences were noted for breast/cervical cancer screening practices. Access to care and lack of education may be associated with decreased cancer prevention/screening behaviors. Interventions are needed to address these barriers. Research should be directed at understanding the impact of the cancer experience on sibling health behaviors.

Prevalence and predictors of risky and heavy alcohol consumption among adult siblings of childhood cancer survivors

To describe alcohol consumption patterns and risk factors for risky and heavy alcohol use among siblings of childhood cancer survivors compared with survivors and national controls. Secondary analysis of prospectively collected data from two national surveys was performed including a cohort of 3034 adult siblings of childhood cancer survivors (age 18-56 years) and 10,398 adult childhood cancer survivors, both from the Childhood Cancer Survivor Study, plus 5712 adult participants from the population-based National Alcohol Survey. Cancer-related experiences, self-reported current health, and mental health were examined in relation to alcohol consumption patterns including heavy and risky drinking. Adult siblings of childhood cancer survivors were more likely to be heavy drinkers (OR adj = 1.3; 1.0-1.6) and risky drinkers (OR adj = 1.3; 1.1-1.6) compared with controls from a national sample. Siblings were also more likely to drink at these two levels compared with survivors. Factors associated with heavy drinking among siblings included being 18-21 years old (OR adj = 2.9; 2.0-4.4), male (OR adj = 2.3; 1.7-3.0), having a high school education or less (OR adj = 2.4; 1.7-3.5), and drinking initiation at a young age (OR adj = 5.1; 2.5-10.3). Symptoms of depression, (OR adj = 2.1; 1.3-3.2), anxiety (OR adj = 1.9; 1.1-3.3), and global psychiatric distress (OR adj = 2.5; 1.5-4.3) were significantly associated with heavy alcohol use. Siblings of children with cancer are more likely to be risky and heavy drinkers as adults compared with childhood cancer survivors or national controls. Early initiation of drinking and symptoms of psychological distress should be identified during early adolescence and effective sibling-specific interventions should be developed and made available for siblings of children with cancer.

Characteristics of responders to a request for a buccal cell specimen among survivors of childhood cancer and their siblings

Analysis of biological samples in large cohort studies may provide insight into the mechanism of, and risk factors for, disease onset and progression. This study describes the methods used to collect biological samples from a large multi-center cohort of childhood cancer survivors and siblings of childhood cancer survivors and evaluates the predictors of a positive response among these individuals. Among survivors, female sex, white race/ethnicity, college graduation, never smoking, accessing the healthcare system in the past 2 years, and having a second malignant neoplasm were the strongest predictors of returning a sample. Among siblings, a similar demographic profile defined those likely to submit the requested sample. To reduce selection bias and increase the value of these samples for future analysis, additional phone calls and reminders targeting non-responders are needed to improve response rates among those least likely to respond to a single mailed request.

Longitudinal Changes in Obesity and Body Mass Index Among Adult Survivors of Childhood Acute Lymphoblastic Leukemia: A Report From the Childhood Cancer Survivor Study

We examined the rate of increase in the body mass index (BMI; kg/m(2)) after final height attainment in survivors of acute lymphoblastic leukemia (ALL) and a noncancer comparison group. Childhood Cancer Survivor Study (CCSS) is a retrospectively ascertained cohort study that prospectively tracks the health status of adults who were diagnosed with childhood cancer between 1970 and 1986 and a comparison group of siblings. Changes in BMI from baseline enrollment to time of completion of follow-up (mean interval, 7.8 years) were calculated for 1,451 ALL survivors (mean age, 32.3 years at follow-up) and 2,167 siblings of childhood cancer survivors (mean age, 35.9 years). The mean BMI of the CCSS sibling comparison group increased with age (women, 0.25 units/yr, 95% CI, 0.22 to 0.28 units; men, 0.23 units/yr, 95% CI, 0.20 to 0.25 units). Compared with CCSS siblings, ALL survivors who were treated with cranial radiation therapy (CRT) had a significantly greater increase in BMI (women, 0.41 units/yr, 95% CI, 0.37 to 0.45 units; men, 0.29 units/yr; 95% CI, 0.26 to 0.32 units). The rate of BMI increase was not significantly increased for ALL survivors who were treated with chemotherapy alone. Younger age at CRT exposure significantly modified risk. CRT used in the treatment of childhood ALL is associated with a greater rate of increasing BMI, particularly among women treated with CRT during the first decade of life. Health care professionals should be aware of this risk and interventions to reduce or manage weight gain are essential in this high-risk population.

Obesity in adult survivors of childhood acute lymphoblastic leukemia: A follow-up report from the Childhood Cancer Survivor Study

9518 Background: We previously reported an association between ≥ 20 Gy cranial radiotherapy (CRT) and obesity in adult survivors of childhood ALL. This follow-up study aimed to determine if, with time, (1) the prevalence of obesity and (2) the rate of BMI change were increased in ALL survivors in comparison with sibling controls. Methods: A longitudinal cohort comparing 1,451 adult survivors of childhood ALL (mean age 32.3 years) to 2,167 siblings of childhood cancer survivors (mean age 35.9 years). BMI was calculated from self- reported heights and weights and used to determine the prevalence of being overweight (25.0 ≤ BMI &lt; 30.0) or obese (BMI ≥ 30.0). Polytomous logistic regression was used to estimate odds ratios (ORs) and 95% confidence intervals (CIs) for being overweight or obese among ALL survivors compared to sibling controls. BMIs at baseline and follow-up (with a mean interval of 7.8 years) were used to calculate mean BMI increases per year occurring between these timepoints. Results: Age- and race-adjusted ORs for obesity among survivors compared with siblings are shown in the table . Notably, 10–19 Gy CRT was associated with an increased likelihood of being obese in comparison with siblings. In longitudinal analysis, survivors who were treated with CRT had a significantly greater net increase in BMI/year in comparison with siblings. At follow-up, 69.7% of females treated with ≥ 20 Gy CRT prior to the age of 10 were overweight (31.1%) or obese (38.6%). Conclusion: Cranial radiotherapy ≥ 10 Gy employed in the treatment of childhood ALL is strongly associated among both male and female survivors with increases in the prevalence of obesity and average BMI gains over time. Female survivors who received CRT ≥ 20 Gy, particularly those treated at a younger age, were most affected. [Table: see text] No significant financial relationships to disclose.

Visual, auditory, sensory, and motor impairments in long‐term survivors of hematopoietic stem cell transplantation performed in childhood

Because of treatment-related toxicity, research is increasingly being focused on long-term sequelae secondary to hematopoietic stem cell transplantation (HSCT) in survivor populations. This study describes the incidence of auditory, sensory, motor, and visual impairments, including cataracts, among 235 individuals who were treated with HSCT during childhood or adolescence. Outcomes were compared with 705 siblings of childhood cancer survivors. Participants completed a survey with questions on posttransplant organ system impairments. Approximately half of survivors were transplanted when younger than 10 years of age. The median length of followup was 11 years. The cumulative incidence of cataracts was 36% at 15 years post-HSCT, although cataracts occurred only in those who received total body irradiation as an HSCT conditioning agent or head irradiation before transplant. Persistent pain was reported by 21% of survivors. Loss of hearing in one or both ears, and legal blindness in one or both eyes, each occurred after transplant in 2% of survivors. Occurrences were uncommon, but survivors were 4.3 times (95% confidence interval [CI]: 2.0-9.4) more likely to report coordination problems, 7.7 times (95% CI: 3.2-18.5) more likely to report chewing or swallowing problems, and 3.5 times (3.5; 95% CI: 1.6-7.9) more likely to report muscle weakness than those in the comparison group. Muscle weakness was strongly associated with positive history of chronic graft-versus-host disease. Increased risks were found for motor impairments, hearing loss, vision loss, and persistent pain among study participants. Cataracts were a frequent adverse effect, suggesting that close monitoring with appropriate intervention for preservation of vision, particularly among those who received total body irradiation, should be a primary goal in survivors of HSCT performed in childhood.

Cataracts and Other Neurosensory and Neuromotor Late Effects in Childhood HSCT Survivors.

A variety of life threatening childhood conditions are now successfully treated with hematopoietic stem cell transplantation (HSCT), as either a first line or rescue therapy. Improved survival rates have sharpened the need for research focused on adverse medical effects that may emerge or persist many years after treatment. Few studies on late effects related to neurological functioning have been conducted among survivors treated with HSCT as children or adolescents. This analysis from the Bone Marrow Transplant Survivor Study quantified the frequency of neuromotor and neurosensory outcomes, including cataracts, using a self- or parent-reported questionnaire. Eligibility included HSCT survivors of 2-years or longer who were transplanted before age 21 years between 1974 and 1998 at the University of Minnesota or City of Hope Cancer Center. Outcomes on sensory, visual, auditory, or motor impairments among 235 HSCT survivors were compared to 705 siblings of childhood cancer survivors with similar age, sex, and race distributions (1:3 frequency matching). Risk ratios (RR) and confidence intervals were modeled using generalized estimating equations with a Poisson distribution and a log link. Cumulative incidence of cataracts by time since transplant was calculated in a competing risk analysis using the Markov Chain Approach. Wilcoxon signed rank tests were used to test differences in cataract cumulative incidence between allogeneic and autologous transplant, and by those with versus without a history of chronic graft versus host disease (cGVHD) among allogeneic transplant recipients. Of survivors successfully contacted, 60% participated in the study. Non-participants did not differ statistically from participants by transplant type, conditioning regimen, cGVHD (past or present), or age at transplant. Among participants, median age at HSCT was 10 years and 82% received their transplant at least 11 years before interview. Diagnoses included 57% with acute leukemia and 13% with aplastic anemia. Persistent pain was reported by 21% of survivors (RR 2.1, p<.001) and cataracts by 38% (RR 270, p<.001). The cumulative incidence of cataracts was 21% at year 5 post-transplant, 32% at year 10, and 36% at year 15. Although cumulative incidence curves for cataracts were significantly higher in allogeneic versus autologous recipients (p=.009), 86 of the 90 survivors with cataracts received total body irradiation (TBI). When the analysis was limited to those who received TBI, no differences in cataract incidence by transplant type (p=.47) or by cGVHD history (p=.43) were observed. Other than cataracts and persistent pain, motor impairments, including problems with swallowing (7.7%; RR 7.7, p<.001); coordination (6.8%; RR 4.3, p<.001) and muscle weakness (5.5%; RR 5.3, p=.002) constituted the most frequent late effects considered. Overall, with the exception of cataracts, relatively few neuromotor and neurosensory problems were reported by childhood HSCT recipients. However, one-fifth of childhood HCST survivors reported persistent pain, suggesting the need for further investigation to understand the causes. As in adults, cataracts are a frequent adverse effect of childhood HSCT, occurring in nearly 40% of these survivors within 15 years post-transplant. Close follow-up with preservation of vision, particularly among those who received TBI, should be a primary goal in this patient population.

Stroke following therapy for Hodgkin's Disease (HD): A report from the Childhood Cancer Survivor Study (CCSS)

8523 Background: An increased risk for strokes has not been previously reported among HD survivors. This report examines the rate and risk factors for stroke in long-term survivors of HD. Methods: The CCSS is a multi-institutional cohort study of long-term (≥ 5 years) survivors who were diagnosed between 1970–1986. The rate of first occurrence of self-reported strokes was determined for HD survivors (N=1665) and compared with a random sample of siblings of childhood cancer survivors (N=3846). The mean age at interview of the HD survivors was 31.9 years of age, with a mean age at diagnosis of 14.6 years. The incidence rates of HD survivors at ≥ 5 years post diagnosis and siblings were calculated separately. Cox proportional hazards models were used to estimate rate ratio (RR) of developing stroke between HD survivals and siblings. Results: Fifteen HD survivors reported a stroke: one during therapy, 13 occurring ≥ 5 years after HD diagnosis, and one without an age at stroke reported. The rate of stroke in HD survivors ≥ 5 years post diagnosis was 0.68 per 1000 person-years (95% CI, 0.40–1.18). Of the 13 late occurring strokes, all had chest/mantle radiation (median dose 40 Gy), with a median interval of 15 years from HD diagnosis to stroke. The median age of stroke was 31 years (range 21–40). Seven siblings reported a previous stroke, with a rate of 0.069 per 1000 person-years (95% CI, 0.03–0.15). The RR of stroke for all HD survivors, in comparison with siblings, was 3.88 (95% CI, 1.46–10.3; p = 0.0065). The rate of stroke for HD survivors who were treated with chest radiation therapy (RT) was 0.82 per 1000 person-years (95% CI, 0.48–1.41). Compared to siblings, the RR of HD survivors treated with chest RT was 4.28 (95% CI, 1.6–11.2; p=0.0031). Anthracycline exposure, treatment with chemotherapy only, hypertension and diabetes mellitus were not associated with an increased risk of stroke. Conclusions: Though an infrequent outcome, survivors of childhood HD may be at an increased risk of stroke. In this study, previous exposure to chest RT was strongly associated with risk of stroke. Because the risk may be modified by prevention, it is imperative to further study this process. No significant financial relationships to disclose.

Stroke following therapy for Hodgkin's Disease (HD): A report from the Childhood Cancer Survivor Study (CCSS)

8523 Background: An increased risk for strokes has not been previously reported among HD survivors. This report examines the rate and risk factors for stroke in long-term survivors of HD. Methods: The CCSS is a multi-institutional cohort study of long-term (≥ 5 years) survivors who were diagnosed between 1970–1986. The rate of first occurrence of self-reported strokes was determined for HD survivors (N=1665) and compared with a random sample of siblings of childhood cancer survivors (N=3846). The mean age at interview of the HD survivors was 31.9 years of age, with a mean age at diagnosis of 14.6 years. The incidence rates of HD survivors at ≥ 5 years post diagnosis and siblings were calculated separately. Cox proportional hazards models were used to estimate rate ratio (RR) of developing stroke between HD survivals and siblings. Results: Fifteen HD survivors reported a stroke: one during therapy, 13 occurring ≥ 5 years after HD diagnosis, and one without an age at stroke reported. The rate of stroke in HD survivors ≥ 5 years post diagnosis was 0.68 per 1000 person-years (95% CI, 0.40–1.18). Of the 13 late occurring strokes, all had chest/mantle radiation (median dose 40 Gy), with a median interval of 15 years from HD diagnosis to stroke. The median age of stroke was 31 years (range 21–40). Seven siblings reported a previous stroke, with a rate of 0.069 per 1000 person-years (95% CI, 0.03–0.15). The RR of stroke for all HD survivors, in comparison with siblings, was 3.88 (95% CI, 1.46–10.3; p = 0.0065). The rate of stroke for HD survivors who were treated with chest radiation therapy (RT) was 0.82 per 1000 person-years (95% CI, 0.48–1.41). Compared to siblings, the RR of HD survivors treated with chest RT was 4.28 (95% CI, 1.6–11.2; p=0.0031). Anthracycline exposure, treatment with chemotherapy only, hypertension and diabetes mellitus were not associated with an increased risk of stroke. Conclusions: Though an infrequent outcome, survivors of childhood HD may be at an increased risk of stroke. In this study, previous exposure to chest RT was strongly associated with risk of stroke. Because the risk may be modified by prevention, it is imperative to further study this process. No significant financial relationships to disclose.

Psychological outcomes in long-term survivors of childhood brain cancer: a report from the childhood cancer survivor study.

To evaluate and compare psychological outcomes in long-term survivors of pediatric brain cancer and siblings of childhood cancer survivors, and to identify significant correlates of psychological distress. One thousand one hundred one adult survivors of childhood brain cancer and 2,817 siblings completed a long-term follow-up questionnaire allowing assessment of symptoms associated with depression, somatization, and anxiety, as well as demographic, health, and medical information. A large majority of siblings and survivors report few, if any, symptoms of psychological distress. The prevalence of distress approximating clinically significant levels for both survivors (11%) and siblings (5%) reflects rates found in the general population. Yet when accounting for significant sociodemographic, socioeconomic, and health-status variables, survivors of childhood brain cancer, in the aggregate, appear to report significantly higher global distress and depression scores than do siblings. As in the general population, higher levels of distress among survivors and siblings were associated with female sex, low household income, lower educational attainment, being unmarried, not being employed in the past 12 months, and poor physical health status. No diagnostic or treatment-related variables were directly and significantly associated with increases in distress symptoms for survivors of childhood brain cancer. Cancer treatment does not appear to contribute directly to increased psychological distress. Instead, distress appears to be associated with diminished social functioning that may be related to cancer type or treatment. Implementation and evaluation of supportive interventions that enhance survivors' social and vocational skills should be considered.

Obesity in adult survivors of childhood acute lymphoblastic leukemia: a report from the Childhood Cancer Survivor Study.

To determine whether adult survivors (>or= 18 years of age) of childhood acute lymphoblastic leukemia (ALL) are at increased risk for obesity and to assess patient and treatment variables that influence risk. A retrospective cohort of participants of the Childhood Cancer Survivor Study was used to compare 1,765 adult survivors of childhood ALL to 2,565 adult siblings of childhood cancer survivors. Body-mass index (BMI; kilograms per square meter), calculated from self-reported heights and weights, was used to determine the prevalence of being overweight (BMI, 25-29.9) or obese (BMI >or= 30.0). Polytomous logistic regression was used to estimate odds ratios (ORs) and 95% confidence intervals (CIs) for being overweight or obese among ALL survivors relative to the sibling control group. The age- and race-adjusted OR for being obese in survivors treated with cranial radiation doses >or= 20 Gy in comparison with siblings was 2.59 for females (95% CI, 1.88 to 3.55; P <.001) and 1.86 for males (95% CI, 1.33 to 2.57; P <.001). The OR for obesity was greatest among females diagnosed at 0 to 4 years of age and treated with radiation doses >or= 20 Gy (OR, 3.81; 95% CI, 2.34 to 5.99; P <.001). Obesity was not associated with treatment consisting of chemotherapy only or with cranial radiation doses of 10 to 19 Gy. Cranial radiotherapy >or= 20 Gy is associated with an increased prevalence of obesity, especially in females treated at a young age. It is imperative that healthcare professionals recognize this risk and develop strategies to enhance weight control and encourage longitudinal follow-up.