Patient-reported neurocognitive function in adult survivors of childhood and adolescent osteosarcoma and Ewing sarcoma.

PURPOSE: Little is known regarding long-term neurocognitive outcomes in osteosarcoma and Ewing sarcoma (EWS) survivors despite potential risk factors. We evaluated associations among treatment exposures, chronic health conditions, and patient-reported neurocognitive outcomes in adult survivors of childhood osteosarcoma and EWS. METHODS: Five-year survivors of osteosarcoma (N = 604; median age 37.0 years) and EWS (N = 356; median age 35.0 years) diagnosed at < 21 years from 1970 to 1999, and 697 siblings completed the Childhood Cancer Survivor Study Neurocognitive Questionnaire and reported chronic health conditions, education, and employment. Prevalence of reported neurocognitive difficulties were compared between diagnostic groups and siblings. Modified Poisson regression identified factors associated with neurocognitive difficulties. RESULTS: Osteosarcoma and EWS survivors, vs. siblings, reported higher prevalences of difficulties with task efficiency (15.4% [P = 0.03] and 14.0% [P = 0.04] vs. 9.6%, respectively) and emotional regulation (18.0% [P < 0.0001] and 15.2% [P = 0.03] vs. 11.3%, respectively), adjusted for age, sex, and ethnicity/race. Osteosarcoma survivors reported greater memory difficulties vs. siblings (23.5% vs. 16.4% [P = 0.01]). Comorbid impairment (i.e., ≥ 2 neurocognitive domains) was more prevalent in osteosarcoma (20.0% [P < 0.001]) and EWS survivors (16.3% [P = 0.02]) vs. siblings (10.9%). Neurological conditions were associated with worse task efficiency (RR = 2.17; 95% CI = 1.21-3.88) and emotional regulation (RR = 1.88; 95% CI = 1.01-3.52), and respiratory conditions were associated with worse organization (RR = 2.60; 95% CI = 1.05-6.39) for EWS. Hearing impairment was associated with emotional regulation difficulties for osteosarcoma (RR = 1.98; 95% CI = 1.22-3.20). Patient report of cognitive difficulties was associated with employment but not educational attainment. CONCLUSIONS: Survivors of childhood osteosarcoma and EWS are at increased risk for reporting neurocognitive difficulties, which are associated with employment status and appear related to chronic health conditions that develop over time. IMPLICATIONS FOR CANCER SURVIVORS: Early screening, prevention, and treatment of chronic health conditions may improve/prevent long-term neurocognitive outcomes.

Newly identified chemotherapy-induced peripheral neuropathy in a childhood cancer survivorship clinic.

Chemotherapy-induced peripheral neuropathy (CIPN), a debilitating side effect of pediatric cancer therapy, can be challenging to diagnose. We estimated the prevalence of newly identified and previously diagnosed CIPN in the regional HEROS Childhood Cancer Survivorship Clinic. From 2016 to 2018, 148 survivors (45.3% female, age 17.1 [SD 7.7] years, 81.8% in ongoing routine oncology follow-up) had their initial survivorship evaluation at an average of 7.4 (SD 6.6) years from diagnosis. Fifty-six survivors (37.8%) had CIPN, of these 46 (82.1%) were newly identified. Our findings demonstrate CIPN may be missed in routine oncology care, and new methods are needed to screen for CIPN.

The association of health status and cancer history of young adult survivors of childhood cancer with parental accompaniment to survivorship clinic visits.

PURPOSE: Adult childhood cancer survivors are frequently accompanied by a parent to survivorship clinic. From clinical evaluations among young adult survivors of childhood cancer we aimed to (1) investigate the association between accompaniment and the survivors' health complexity; and (2) determine whether accompaniment is associated with adherence to recommended surveillance tests and follow-up in clinic. METHODS: This was a cross-sectional study of all patients ≥ 18 years old at their first visit to the regional Yale Childhood Cancer Survivorship Clinic from 2003 to 2018. Patients underwent standardized evaluations for medical, neurocognitive, and emotional late effects of therapy; individuals accompanying patients were documented. RESULTS: The 168 patients were a median of 12.0 (range: 0-17.9) years at diagnosis and 22.7 (range: 18.1-39.9) years at evaluation, and 45.8% were accompanied by a parent. In multivariable analyses, 18.0-24.99 years vs. 25.0-39.99 years at visit (OR = 3.43, p = 0.022) and central nervous system (CNS) tumor diagnosis (OR = 6.09 vs. leukemia/lymphoma diagnosis, p = 0.010) were significantly associated with parental accompaniment. Accompaniment was not associated with number and severity of medical late effects, neurocognitive impairment, or emotional distress. Accompaniment was not associated with completed surveillance tests or a clinic follow-up within 2 years. CONCLUSION: Forty-six percent of survivors were accompanied by a parent, and accompaniment was not associated with survivor health status. Accompaniment was not associated with adherence to recommended surveillance tests or clinic follow-up.

Severity of reduced bone mineral density and risk of fractures in long-term survivors of childhood leukemia and lymphoma undergoing guideline-recommended surveillance for bone health.

BACKGROUND: Survivors of childhood leukemia/lymphoma are at increased risk for reduced bone mineral density (BMD). The authors sought to determine the frequency of reduced BMD detected by off-therapy surveillance, factors associated with reduced BMD, and the association of reduced BMD with fractures. METHODS: This cross-sectional study included childhood leukemia/lymphoma survivors attending 2 survivorship clinics who received guideline-recommended BMD surveillance ≥2 years post-therapy with dual-energy x-ray absorptiometry (from January 1, 2004 to August 31, 2016). Lumbar spine BMD z-scores were height-for-age-adjusted. Low and very low BMD were >1 SD and >2 SDs below norms, respectively. Treatment, chronic conditions, and fractures were abstracted from medical records. Logistic regression was used to examine the association of low BMD with patient/treatment factors and fractures. RESULTS: In total, 542 patients (51.5% female) with a mean age of 15.5 years (range, 4.4-52.2 years) who were 6 years post-therapy (range, 2.0-35.1 years) were evaluated, including 116 who reported post-therapy fractures. Lumbar spine low BMD was identified in 17.2% of survivors, and very low BMD was identified in 3.5% of survivors, but frequencies varied considerably between subgroups; 10.8% of survivors aged 15 to 19 years at diagnosis had very low BMD. In multivariable analyses, older age at diagnosis, white race, and being underweight were significantly associated with low BMD. Survivors with low BMD had greater odds of nondigit fractures (odds ratio, 2.2; 95% CI, 1.3-3.7) and specifically long-bone fractures (odds ratio, 2.7; 95% CI, 1.5-4.7). CONCLUSIONS: In this study of childhood leukemia/lymphoma survivors undergoing guideline-recommended dual-energy x-ray absorptiometry surveillance, patients who were older at diagnosis, white, and underweight were at the highest risk for lumbar spine low BMD. Low BMD was associated with a greater risk of fractures, emphasizing the clinical importance of surveillance.

Impact of Survivorship Care on Young Adult Survivors of Childhood Cancer With Post-Traumatic Stress Symptoms.

OBJECTIVES: To assess post-traumatic stress symptoms (PTSSs) in young adult survivors of childhood cancer not receiving survivorship care and to determine whether attending a survivorship-focused healthcare visit was associated with changes in PTSSs. SAMPLE & SETTING: 44 young adult survivors from the Yale Cancer Center in Connecticut without prior survivorship clinic attendance. METHODS & VARIABLES: As part of a larger trial, participants were randomized to a model of survivorship-focused health care. The University of California at Los Angeles Post-Traumatic Stress Disorder (PTSD) Reaction Index assessed PTSS severity and frequency before and after the visit. RESULTS: At baseline, almost half of the participants were classified as partial PTSD likely or PTSD likely. Many met criteria for elevated levels of individual symptoms, particularly avoidance or numbing. At follow-up, PTSSs did not differ significantly from baseline. IMPLICATIONS FOR NURSING: Survivorship care should be encouraged by nurses in healthcare settings that do not specialize in caring for long-term survivors. Nurses should facilitate screening for PTSSs and promote interventions among survivors completing cancer therapy to help them transition to survivorship care.

Parental perception of child vulnerability in childhood cancer survivors.

BACKGROUND: Parents' perception of their children's vulnerability to illness following cancer treatment is largely unknown, but is important to understand given known challenges of transitioning survivors to postcancer care. We investigated the frequency of and factors associated with perceived vulnerability by parents of childhood cancer survivors attending a regional survivorship clinic. PROCEDURE: This cohort study was offered to all parents of pediatric patients (currently ≤18 years) attending the Yale childhood cancer survivorship clinic January 2010 to October 2016 who were ≥1 year postcurative cancer therapy. Participating parents (one per patient) completed the standardized Child Vulnerability Scale at the beginning of the clinic visit (cutoff score ≥10 for perceived vulnerability). Patient sociodemographics, cancer history, and posttherapy complications were abstracted from medical records. RESULTS: Overall, 116 parents participated (98% participation rate) consisting of 89% mothers; survivors were 46% female, had a current mean age of 12.7 ± 3.9 years, and were a mean of 6.4 ± 3.8 years posttherapy. Twenty-eight percent (n = 33) of parents perceived their children as vulnerable. Survivor sociodemographics (age, sex, race/ethnicity, family income, insurance, parental marital status, number of siblings), cancer diagnosis, years off-therapy, survivorship visit number, treatment intensity, and late complications (number, type, severity) were not associated with perception of vulnerability. CONCLUSIONS: A sizeable proportion of parents continue to perceive their children as vulnerable even years after cancer therapy completion independent of current health status or past cancer history. Our data suggest the need to educate all parents of childhood cancer survivors regarding health risk, including those at lower risk for late complications.

Randomized Trial of the Impact of Empowering Childhood Cancer Survivors With Survivorship Care Plans.

Background: A survivorship care plan (SCP), that is, individualized treatment summary and schedule of off-therapy surveillance, will be mandated by January 2019. It is unclear whether SCPs improve adherence to recommended follow-up care in the community. In this trial, we evaluated the impact of randomly assigning childhood cancer survivors to 1) SCPs to be taken to their primary care physician (PCP) to implement or 2) survivorship clinic (SC) on health care quality measures. Methods: Eligibility included cancer diagnosis younger than age 18 years (2000-2012), cancer free, one or more years off therapy, and no prior survivorship clinic attendance. At 12 months, the random assignment groups were compared (SCP+PCP vs SC) by intent-to-treat analysis with two-sided statistical tests in terms of patient adherence to guideline-recommended surveillance tests (eg, echocardiogram) and number of newly identified late complications of therapy. Results: From 2011 to 2013, 96 participants (46.9% female, mean age = 15.9 ± 6.1 years) were randomly assigned. Adherence to 14 evaluated guideline-recommended surveillance tests ranged from 0% to 46.9% in the SCP+PCP group (n = 47) and from 50.0% to 86.4% in the SC group (n = 47). Adherence to 10 tests was statistically significantly different between the groups (all P < .05). One mild new late complication was identified in the SCP+PCP group compared with 21 late complications, ranging from mild to severe, identified in 11 patients in the SC group (2.1% vs 23.4% of patients, respectively, P = .003). Conclusions: Our randomized trial suggests that empowering childhood cancer survivors with SCPs to be implemented by their PCPs is not sufficient to meet consensus follow-up recommendations.

Physical activity barriers, preferences, and beliefs in childhood cancer patients.

PURPOSE: Childhood cancer patients report low physical activity levels despite the risk for long-term complications that may benefit from exercise. Research is lacking regarding exercise barriers, preferences, and beliefs among patients (1) on- and off-therapy and (2) across the age spectrum. METHODS: Cross-sectional study in the Yale Pediatric Hematology-Oncology Clinic (October 2013-October 2014). Participants were ≥ 4 years old, > 1 month after cancer diagnosis at < 20 years, not acutely ill, expected to live > 6 months, and received chemotherapy and/or radiation. Participants (or parents if < 13 years) completed a survey. RESULTS: The 162 patients (99% participated) were 34% children (4.0-12.9 years), 31% adolescents (13.0-17.9 years), and 35% adults (≥ 18 years). Most had leukemia/lymphoma (66%); 32% were on-therapy. On-therapy patients were more likely than off-therapy patients (73 vs. 48%; p = 0.003) to report ≥ 1 barrier related to physical complaints, such as "just too tired" (46 vs. 28%; p = 0.021) or "afraid" of injury (22 vs. 9%; p = 0.027). The majority preferred walking (73%), exercising at home (91%), exercising in the afternoon (79%), and a maximum travel time of 10-20 min (54%); preferences did not vary significantly by therapy status or age. Most respondents (94%) recognized the benefits of exercise after cancer, but 50% of on- vs. 12% of off-therapy patients believed "their cancer diagnosis made it unsafe to exercise regularly" (p < 0.001). CONCLUSIONS: Physical activity barriers pertaining to physical complaints and safety concerns were more pronounced in on-therapy childhood cancer patients but persisted off-therapy. Preferences and beliefs were relatively consistent. Our data can inform interventions in different patient subgroups.

Experiences with the survivorship care plan in primary care providers of childhood cancer survivors: a mixed methods approach.

PURPOSE: Focusing on primary care providers (PCPs) of known childhood cancer survivors, we used a mixed methods approach to understand providers' experiences with the survivorship care plan (SCP), as well as preferences and barriers regarding survivorship care. METHODS: The SCP was sent to 202 eligible PCPs of patients attending a regional survivorship clinic. The PCPs then completed quantitative postal surveys. Sixteen PCPs underwent an additional qualitative interview. Provider characteristics were examined as potential predictors of experiences, preferences, and barriers regarding survivorship care. Grounded theory was utilized to analyze the qualitative data. RESULTS: Respondents (n=134) included 55% pediatricians, 23% internists, 20% family practitioners, and 2% obstetricians/gynecologists. Only 8% of PCPs preferred to have primary responsibility for survivorship care; 46% favored a specialized clinic. A minority of PCPs reported that they felt "very comfortable" using the SCP to guide surveillance (31%) or deliver all survivorship care (3%). Endorsed barriers included lack of knowledge/training (74%), not recalling or understanding the contents of the SCP (69%), and uncertainty about who is responsible for providing care (58%). Two distinct themes emerged from the qualitative interviews: a preference not to be responsible for survivorship care, and uncertain utility of the SCP. CONCLUSIONS: Our study suggests that PCPs of childhood cancer survivors generally felt uncomfortable using the SCP and providing survivorship care. Even with the availability of the SCP, the biggest barrier was lack of knowledge. Our data suggests that including PCPs in survivorship care will require additional educational opportunities and evaluation of more collaborative models of care delivery.

Dietary patterns in Liberian refugees in Buduburam, Ghana.

Previous research suggests that acculturation (i.e., exposure and assimilation to local culture) is associated with changes in dietary patterns among immigrants. This study investigates this association in a refugee population using time in refugee settlement as a proxy for acculturation. A cross-sectional survey was conducted among a systematic sample to (a) identify dietary patterns in Liberian refugees and Ghanaians living in or near a refugee settlement, (b) compare adherence to these dietary patterns between groups, and (c) investigate the association between acculturation and dietary patterns in Liberian refugees. Participants were Liberian and Ghanaian women with young children living in the Buduburam refugee settlement or Awutu in Ghana (n = 480; 50% Liberian; mean age 28, SD 6.3, range 16-48 years). Time in settlement was assessed by self-report; food consumption was assessed by food frequency questionnaire. Principal component analysis was used to identify dietary patterns; a generalized linear model was used to test the association of interest. Three distinct dietary patterns emerged: Healthy, Sweets, and Fats. Ghanaians were more adherent to the Healthy pattern than Liberians (p < 0.05). Liberians were more adherent to the Sweets and Fats patterns than Ghanaians (p < 0.05). There were no significant differences in dietary pattern adherence among the Liberians based on time in settlement. Ghanaians living in Awutu were more adherent to the Healthy pattern than Ghanaians who lived in settlement (p < 0.05). Differences in dietary patterns were observed between Liberian refugees and Ghanaians. These differences were not associated with acculturation and may be related to the food environment in the settlement.