Long-term risk of second malignant neoplasms after neuroblastoma in childhood: Role of treatment

2003 ◽  
Vol 107 (5) ◽  
pp. 791-796 ◽  
Author(s):  
Carole Rubino ◽  
Elisabeth Adjadj ◽  
Sylvie Guérin ◽  
Catherine Guibout ◽  
Akhtar Shamsaldin ◽  
...  
2021 ◽  
Vol 16 (1) ◽  
Author(s):  
Hongnan Zhen ◽  
Hui Guan ◽  
Jiabin Ma ◽  
Wenhui Wang ◽  
Shen Jing ◽  
...  

Abstract Background Neuroblastoma is a common extracranial malignant tumor in children. Its main treatment modality is a combination of chemotherapy, radiotherapy, and surgery. Given the advances in chemotherapy regimens and the widespread use of bone marrow transplantation over the decades, there has been improvement in treatment efficacy, which has led to prolonged patient survival. Accordingly, long-term complications have become a growing concern among physicians and patients. This study aimed to analyze the survival rate of patients with neuroblastoma and the risk factors for developing second malignant neoplasms (SMNs). Methods The SEER 18 Regs (1973–2015) and SEER 9 Regs (1973–2015) data of the surveillance, epidemiology, and end results (SEER) database of the US National Cancer Institute were adopted for survival and SMN analysis. Results The 5-, 10-, and 20-year overall survival rates of patients with neuroblastoma were 67%, 65%, and 62%, respectively. Among 38 patients with neuroblastoma who presented with SMNs, those with abdomen as the primary site accounted for the majority (63.2%), followed by those with thorax (26.3%) and other sites (10.5%). SMNs occurred more commonly in non-specific neuroblastoma (incidence: 0.87%) than ganglioneuroblastoma (incidence: 0.3%). Compared with the general population, the risk of SMN is significantly higher (SIR = 4.36). The risk of developing SMNs was significantly higher in the digestive system (SIR = 7.29), bones and joints (SIR = 12.91), urinary system (SIR = 23.48), brain and other nervous systems (SIR = 5.70), and endocrine system (SIR = 5.84). Multivariate analysis revealed that the year of diagnosis (OR = 2.138, 95% CI = 1.634–2.797, p < 0.001) was the only independent risk factor for developing SMNs. Conclusion This study identifies the risk factor for developing SMNs in patients with neuroblastoma, which could facilitate individualized screening for high-risk patients, to allow early diagnosis and treatment of SMNs.


Author(s):  
Tom Boterberg ◽  
Yen-Ch’ing Chang ◽  
Karin Dieckmann ◽  
Mark Gaze ◽  
Helen Woodman

Chapter 5 discusses care during and after radiotherapy for children and young people. During and immediately after treatment, children and young people receiving radiotherapy need monitoring for acute complications of treatment and may require supportive care. Following completion of treatment, a response assessment is needed, followed by ongoing surveillance for recurrence. If relapse occurs, consideration can be given to further treatment, which may be radical or palliative in intent. With the passing of time, the risks of relapse recede and monitoring for the late effects of treatment becomes more important. As the majority of patients will have some long-term sequelae, some of which can be ameliorated by timely intervention, patients should be followed in a multidisciplinary clinic. A detailed treatment summary will help predict the risk of complications and guide long-term follow-up. Patients, when they reach adult life, should be aware of possible problems, including fertility issues and second malignant neoplasms.


Author(s):  
Frank Speleman ◽  
Julie R. Park ◽  
Tara O. Henderson

Neuroblastoma, an embryonal tumor arising from neural crest–derived progenitor cells, is the most common solid tumor in childhood, with more than 700 cases diagnosed per year in the United States. In the past several decades, significant advances have been made in the treatment of neuroblastoma. Treatment advances reflect improved understanding of the biology of neuroblastoma. Although amplification of MYCN was discovered in the early 1980s, our understanding of neuroblastoma oncogenesis has advanced in the last decade as a result of high-throughput genomic analysis, exome and whole-genome sequencing, genome-wide association studies, and synthetic lethal drug screens. Our refined understanding of neuroblastoma biology and genetics is reflected in improved prognostic stratification and appropriate tailoring of therapy in recent clinical trials. Moreover, for high-risk neuroblastoma, a disease that was uniformly fatal 3 decades ago, recent clinical trials incorporating autologous hematopoietic transplant and immunotherapy utilizing anti-GD2 antibody plus cytokines have shown improved event-free and overall survival. These advances have resulted in a growing population of long-term survivors of neuroblastoma. Examination of the late effects and second malignant neoplasms (SMNs) in both older generations of survivors and more recently treated survivors will inform both design of future trials and surveillance guidelines for long-term follow-up. As a consequence of advances in understanding of the biology of neuroblastoma, successful clinical trials, and refined understanding of the late effects and SMNs of survivors, the promise of precision medicine is becoming a reality for patients with neuroblastoma.


1997 ◽  
Vol 89 (19) ◽  
pp. 1429-1439 ◽  
Author(s):  
L. B. Travis ◽  
R. E. Curtis ◽  
J. F. Fraumeni ◽  
J. D. Boice ◽  
H. Storm ◽  
...  

2004 ◽  
Vol 91 (11) ◽  
pp. 1905-1910 ◽  
Author(s):  
H C Jenkinson ◽  
M M Hawkins ◽  
C A Stiller ◽  
D L Winter ◽  
H B Marsden ◽  
...  

2006 ◽  
Vol 24 (18_suppl) ◽  
pp. 9007-9007
Author(s):  
R. Goldsby ◽  
C. Burke ◽  
R. Nagarajan ◽  
T. Zhou ◽  
Z. Chen ◽  
...  

9007 Background: The growing number of individuals surviving childhood cancer has increased the awareness and recognition of long-term sequelae. One of the most worrisome complications following cancer therapy is the development of second malignant neoplasms (SMN), in particular, late-occurring solid second malignancies related radiation therapy. Methods: We describe the incidence of solid organ SMN in survivors of pediatric malignant bone tumors (MBT) treated on legacy CCG/POG protocols from 1980 to 2005. This retrospective cohort study included 2,842 patients, 1,686 treated for osteosarcoma (OS) and 1,156 treated for Ewings Sarcoma (ES). The cohort included 56% males and 44% females, with a median age at primary diagnosis of 13 years. The median length of follow-up was 4.3 years (range: 0 to 20.9 years). Results: At the time of the analysis, 64% of patients in this study are alive. Seventeen patients with solid organ SMN were identified, and included three patients with breast cancer, three with malignant fibrous histiocytoma, two with osteosarcoma, and 9 patients with other solid organ malignancies. The standardized incidence ratio (SIR=observed/expected cases) was 2.9 (95% confidence interval [CI], 1.4–5.4) for patients treated for OS and 5.0 (95%CI 2.6–9.4) for patients treated for ES. The median time from diagnosis to develop solid organ SMN was 7 years (range: 1 to 13 years). The 10-year cumulative incidence of solid organ SMN for the entire cohort was 1% (95%CI 0.6–2%). In univariate analysis, treatment with etoposide, cyclophosphamide or radiotherapy were each associated with a higher than expected incidence of cancer with SIR of 4.8 (95% CI, 2.5–9.5), 5.8 (95% CI, 3.5–9.5) and 4.1 (95% CI, 2.4–7.1), respectively. Conclusions: Solid organ SMNs are rare after treatment for OS and ES, although higher in patients treated for ES. Recurrence remains the most significant problem for patients diagnosed with MBT and development of improved therapies with fewer long-term consequences remains paramount. However, solid organ cancers are likely to increase with longer follow-up. Therefore, surveillance should focus on monitoring for both recurrence of primary malignancies and development of SMN. No significant financial relationships to disclose.


Hematology ◽  
2014 ◽  
Vol 2014 (1) ◽  
pp. 190-197 ◽  
Author(s):  
Lewis B. Silverman

Abstract Cure rates for children and adolescents with acute lymphoblastic leukemia (ALL) have improved dramatically over the last few decades. With this success has come increasing recognition of the adverse late effects of treatment. The significant long-term sequelae in the earliest cohort of long-term survivors treated in the 1970s and 1980s are well documented. To reduce the incidence of these late effects, the majority of pediatric patients treated on more contemporary regimens receive less intensive treatment than did those treated 30-40 years ago. However, current therapies are not risk free; children treated with contemporary regimens remain at risk for developing long-term toxicities, including cardiac dysfunction, osteonecrosis, neurocognitive impairment, and second malignant neoplasms. One of the great challenges facing clinical investigators today is to identify interventions that will reduce the frequency and severity of long-term toxicities without adversely affecting cure rates. The use of dexrazoxane as a cardioprotectant (to prevent anthracycline-associated cardiotoxicity) and alternate-week dosing of dexamethasone (to reduce the risk of osteonecrosis) are examples of 2 such successful strategies. This article provides an overview of the long-term toxicities associated with current therapies and reviews results of clinical trials designed to minimize the burden of cure in long-term survivors.


2020 ◽  
Vol 7 (2) ◽  
pp. 115-119
Author(s):  
Yu. K. Toshina ◽  
Yu. V. Dinikina ◽  
A. S. Egorov ◽  
A. Yu. Smirnova ◽  
M. B. Belogurova

Neuroblastoma (NB) is the most common extra-cranial solid tumor in infants with the most heterogeneous clinical course to compare with other malignant diseases. Due to intensive multimodal anticancer treatment there are an increased number of survivors and issues related to long-term effects are becoming increasingly important. One of them is the risk of secondary malignant neoplasms. This article represents a clinical case of T-cell lymphoblastic leukemia in a child aged 2 years and 5 months who received combined antitumor therapy for NB with an intermediate risk group under the age of one year. We observed literature data to investigate the incidence of second malignant neoplasms in patients with NB for the period from 1948 to 2018 and analyzed risk factors.


2021 ◽  
Author(s):  
Hongnan Zhen ◽  
Hui Guan ◽  
Jiabin Ma ◽  
Wenhui Wang ◽  
Shen Jing ◽  
...  

Abstract Background: Neuroblastoma is a common extracranial malignant tumor in children. Its main treatment modality is a combination of chemotherapy, radiotherapy, and surgery. Given the advances in chemotherapy regimens and the widespread use of bone marrow transplantation over the decades, there has been improvement in treatment efficacy, which has led to prolonged patient survival. Accordingly, long-term complications have become a growing concern among physicians and patients. This study aimed to analyze the survival rate of patients with neuroblastoma and the risk factors for developing second malignant neoplasms (SMNs).Methods: The SEER 18 Regs (1973-2015) and SEER 9 Regs (1973-2015) data of the Surveillance, Epidemiology, and End Results (SEER) database of the US National Cancer Institute were adopted for survival and SMN analysis.Results: The 5-, 10-, and 20-year overall survival rates of patients with neuroblastoma were 67%, 65%, and 62%, respectively. Among 38 patients with neuroblastoma who presented with SMNs, those with abdomen as the primary site accounted for the majority (63.2%), followed by those with thorax (26.3%) and other sites (10.5%). SMNs occurred more commonly in non-specific neuroblastoma (incidence: 0.87%) than ganglioneuroblastoma (incidence: 0.3%). Compared with the general population, the risk of SMN is significantly higher (SIR=4.36). The risk of developing SMNs was significantly higher in the digestive system (SIR =7.29), bones and joints (SIR = 12.91), urinary system (SIR = 23.48), brain and other nervous systems (SIR = 5.70), and endocrine system (SIR = 5.84). Multivariate analysis revealed that the year of diagnosis (OR=2.138, 95%CI=1.634-2.797, p<0.001) was the only independent risk factor for developing SMNs.Conclusion: This study identifies the risk factor for developing SMNs in patients with neuroblastoma, which could facilitate individualized screening for high-risk patients, to allow early diagnosis and treatment of SMNs.


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