Mortality and morbidity of patients with neuroblastoma who survived for more than 10 years after treatment—Niigata Tumor Board Study

Kubota, Masayuki; Okuyama, Naoki; Hirayama, Yutaka; Asami, Keiko; Ogawa, Atsushi; Watanabe, Akihiro

doi:10.1016/j.jpedsurg.2009.09.002

Cited by 7 publications

(6 citation statements)

References 14 publications

(19 reference statements)

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“…Late effects in the cohort were consistent with the clinical presentation of neuroblastoma and therapeutic approaches used over the years . Radiation therapy provided for local control of paraspinal tumors contributed to the increased risk of endocrine and musculoskeletal late effects.…”

Section: Discussionsupporting

confidence: 59%

“…This is in contrast to previous studies among children who had been diagnosed with advanced disease and/or had received autologous transplantation, which reported late effects in the majority (>90%) of survivors, but not among survivors with lower stage disease. For example, a summary of outcomes among neuroblastoma survivor participants (all stages of disease) in the CCSS reported a 20‐year cumulative incidence of a chronic condition of 41%; a smaller study of 43 Japanese survivors identified health conditions in only 28% of the participants . The higher prevalence of chronic conditions observed in our study likely occurred because outcomes were ascertained uniformly and clinically and because SJLIFE participants were older (median, 31.9 years) than survivors in previous studies.…”

Section: Discussioncontrasting

confidence: 45%

“…For example, a summary of outcomes among neuroblastoma survivor participants (all stages of disease) in the CCSS reported a 20-year cumulative incidence of a chronic condition of 41% 9 ; a smaller study of 43 Japanese survivors identified health conditions in only 28% of the participants. 4 The higher prevalence of chronic conditions observed in our study likely occurred because outcomes were ascertained uniformly and clinically and because SJLIFE participants were older (median, 31.9 years) than survivors in previous studies. Interestingly, we did not find the cumulative burden of chronic conditions to vary significantly by disease stage at diagnosis.…”

Section: Discussionmentioning

confidence: 99%

“…Neuroblastoma is the most common extracranial solid tumor among children and accounts for approximately 6% of all cancers in children . Previous reports of neuroblastoma survivors have documented multimorbidity affecting a spectrum of organ systems . In the largest study to date, which consisted of 954 neuroblastoma survivors, the Childhood Cancer Survivor Study (CCSS) found increased risks of musculoskeletal, neurological, endocrine, and sensory conditions in comparison with siblings .…”

Section: Introductionmentioning

confidence: 99%

“…1 Previous reports of neuroblastoma survivors have documented multimorbidity affecting a spectrum of organ systems. [2][3][4][5][6][7][8] In the largest study to date, which consisted of 954 neuroblastoma survivors, the Childhood Cancer Survivor Study (CCSS) found increased risks of musculoskeletal, neurological, endocrine, and sensory conditions in comparison with siblings. 9 However, a limitation of these data was that health outcomes were based on self-report and thus were subject to both ascertainment and recall bias.…”

Section: Introductionmentioning

confidence: 99%

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Clinically ascertained health outcomes, quality of life, and social attainment among adult survivors of neuroblastoma: A report from the St. Jude Lifetime Cohort

Wilson

Brinkman

Cook

et al. 2020

Cancer

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Background The objective of this study was to characterize chronic disease, health‐related quality of life (HRQOL), emotional distress, and social attainment among long‐term survivors of neuroblastoma. Methods Chronic health conditions among 136 ≥10‐year neuroblastoma survivors (median age, 31.9 years; range, 20.2‐54.6 years) and 272 community controls (median age, 34.7 years; range, 18.3‐59.6 years) were graded with a modified version of the Common Terminology Criteria for Adverse Events (version 4.03). HRQOL and emotional distress were assessed with the Medical Outcomes Study 36‐Item Short Form Health Survey and the Brief Symptom Inventory‐18. Log‐binomial regression and logistic regression were used to compare the prevalence of chronic conditions and the frequency of reduced HRQOL, distress, and social attainment between survivors and controls. The cumulative burden approach was used to estimate multimorbidity. Results By the age of 35 years, survivors had experienced, on average, 8.5 grade 1 to 5 conditions (95% confidence interval [CI], 7.6‐9.3), which was higher than the average for controls (3.3; 95% CI, 2.9‐3.7). Compared with controls, survivors had a higher prevalence of any pulmonary (P = .003), auditory (P < .001), gastrointestinal (P < .001), neurological (P = .003), or renal condition (P < .001); were more likely to report poor physical HRQOL (P = .01) and symptoms of anxiety (P = .01) and somatization (P = .01); and were less likely to live independently (P = .01) or marry (P = .01). In analyses limited to survivors, those with 1 or more grade 3 to 5 conditions were more likely to report reduced general health (odds ratio [OR], 6.6; 95% CI, 1.6‐26.9), greater bodily pain (OR, 4.2; 95% CI, 1.0‐17.0), and unemployment (OR, 3.2; 95% CI, 1.2‐8.5). Conclusions Because of the high burden of chronic diseases and the associations of these morbidities with reduced HRQOL and social attainment, screening and interventions that provide opportunities to optimize health are important among neuroblastoma survivors.

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Section: Discussionsupporting

confidence: 59%

Section: Discussioncontrasting

confidence: 45%

Section: Discussionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

Section: Introductionmentioning

confidence: 99%

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Clinically ascertained health outcomes, quality of life, and social attainment among adult survivors of neuroblastoma: A report from the St. Jude Lifetime Cohort

Wilson

Brinkman

Cook

et al. 2020

Cancer

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RLIP76 Inhibition: A Promising Developmental Therapy for Neuroblastoma

Singhal

Nagaprashantha

Singhal³

et al. 2017

Pharm Res

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Refractory and relapsed neuroblastoma (NB) present with significant challenges in clinical management. Though primary NBs largely with wild-type p53 respond well to interventions, dysfunctional signaling in the p53 pathways in a MYCN oncogene driven background is found in a number of children with NB. The p53-mutant NB is largely unresponsive to available therapies and p53-independent targeted therapeutics represents a vital need in pediatric oncology. We analyzed the findings on mercapturic acid pathway (MAP) transporter RLIP76, which has broad and critical effects on multiple pathways as essential for carcinogenesis, oxidative stress and drug-resistance, is over-expressed in NB. RLIP76 inhibition by antibodies or depletion by antisense causes apoptosis and sensitization to chemo-radiotherapy in many cancers. In addition, recent studies indicate that the interactions between p53, MYCN, and WNT regulate apoptosis resistance and protein ubiquitination. RLIP76 and p53 interact with each other and colocalize in NB cells. Targeted depletion/inhibition of RLIP76 causes apoptosis and tumor regression in NB irrespective of p53 status. In the present review, we discuss the mechanisms and the role of RLIP76 in oxidative stress, drug-resistance and clathrin-dependent endocytosis (CDE), and analyze the molecular basis for the role of RLIP76 targeted approaches in the context principal drivers of NB pathogenesis, progression and drug-resistance. The evidence from RLIP76 studies in other cancers, when taken in the context of our recent RLIP76 focused mechanistic studies in NB, provides strong basis for further characterization and development of RLIP76 targeted therapies for NB.

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