Second malignant neoplasms in children: an update from the Late Effects Study Group.

1985 ◽  
Vol 3 (4) ◽  
pp. 532-538 ◽  
Author(s):  
A T Meadows ◽  
E Baum ◽  
F Fossati-Bellani ◽  
D Green ◽  
R D Jenkin ◽  
...  
Keyword(s):  
Risk Factors ◽  
Late Effects ◽  
Wilms Tumor ◽  
Soft Tissue Sarcomas ◽  
Malignant Neoplasms ◽  
Study Group ◽  
Second Malignant Neoplasms ◽  
Bone Sarcomas ◽  
Radiation Genetic ◽  
Survivors Of Childhood Cancer

This paper presents an update from the Late Effects Study Group on 292 cases of second malignant neoplasms (SMN) occurring in individuals who were diagnosed with their first neoplasm in childhood. Data are presented regarding the types of first and second neoplasm, the therapy administered, and the predisposing factors. Of the 292 cases (308 SMN), the most common primary was retinoblastoma followed by Hodgkin's disease, soft-tissue sarcomas, and Wilms' tumor. This is not similar to the relative frequency of these cancers in children but rather reflects specific risk factors. Bone sarcomas were the most common SMN among the 208 SMN developing in previously irradiated sites while acute leukemia was the most common SMN unassociated with radiation. Known predisposing conditions to cancer were present in 73 cases; retinoblastoma was the most common of these, followed by neurofibromatosis. There were ten patients with three and three patients with four malignant neoplasms. In 14 patients, the cause of SMN was not suggested by known risk factors as these patients had negative family histories and received no radiation or chemotherapy. We note, therefore, that although most cases of SMN in survivors of childhood cancer can be attributed to radiation, genetic disease, chemotherapy, or combinations of these, unrecognized predisposition or chance may also play a role.

scholarly journals Early and Late Mortality After Diagnosis of Wilms Tumor

2009 ◽  
Vol 27 (8) ◽  
pp. 1304-1309 ◽  
Author(s):  
Cecilia A. Cotton ◽  
Susan Peterson ◽  
Patricia A. Norkool ◽  
Janice Takashima ◽  
Yevgeny Grigoriev ◽  
...  
Keyword(s):  
Late Effects ◽  
Cox Regression ◽  
Wilms Tumor ◽  
Malignant Neoplasms ◽  
Calendar Period ◽  
Original Diagnosis ◽  
Diagnosis Rate ◽  
Second Malignant Neoplasms ◽  
Causes Of Mortality ◽  
Standardized Mortality Ratios

Purpose To assess rates and causes of mortality in patients with Wilms tumor (WT). Methods Through 2002, 6,185 patients enrolled onto the National Wilms Tumor Study between 1969 and 1995 were actively observed. Deaths were classified on the basis of medical records as the result of original disease, late effects (including second malignant neoplasms [SMNs], cardiac causes, pulmonary disease, and renal failure), or other causes. Standardized mortality ratios (SMRs) and Cox regression were used to assess the effects of sex, age, and calendar period of diagnosis on mortality. Results Within 5 years of WT diagnosis, 819 deaths occurred, and 159 deaths occurred among 4,972 known 5-year survivors. The SMR was 24.3 (95% CI, 22.6 to 26.0) for the first 5 years, was 12.6 (95% CI, 10.0 to 15.7) for the next 5 years, and remained greater than 3.0 thereafter. For deaths in the first 5 years, the mortality risk decreased by 5-year calendar period of diagnosis (rate ratio [RR] = 0.78 per period). No such trend occurred for later deaths. Among 5-year survivors, 62 deaths were attributed to late effects of treatment or disease, including 27 to SMNs. A trend of decreased risk with calendar period of diagnosis was observed for late-effects mortality (RR = 0.86; 95% CI, 0.67 to 1.10) and for SMN mortality (RR = 0.82; 95% CI, 0.55 to 1.21). Conclusion Although the survival outlook for WT patients has improved greatly over time, survivors remain at elevated risk for death many years after their original diagnosis.

scholarly journals Outcome and Late Complications of Hepatoblastomas Treated Using the Japanese Study Group for Pediatric Liver Tumor 2 Protocol

2020 ◽  
Vol 38 (22) ◽  
pp. 2488-2498 ◽  
Author(s):  
Eiso Hiyama ◽  
Tomoro Hishiki ◽  
Kenichiro Watanabe ◽  
Kohmei Ida ◽  
Yuka Ueda ◽  
...  
Keyword(s):  
Late Effects ◽  
Low Dose ◽  
Liver Tumors ◽  
Survival Rates ◽  
Late Complications ◽  
Malignant Neoplasms ◽  
Study Group ◽  
Hematopoietic Stem ◽  
Japanese Study ◽  
Pediatric Liver

PURPOSE We report here the outcomes and late effects of the Japanese Study Group for Pediatric Liver Tumors (JPLT)-2 protocol, on the basis of cisplatin-tetrahydropyranyl-adriamycin (CITA) with risk stratification according to the pretreatment extent of disease (PRETEXT) classification for hepatoblastoma (HB). PATIENTS AND METHODS From 1999 to 2012, 361 patients with untreated HB were enrolled. PRETEXT I/II patients were treated with up-front resection, followed by low-dose CITA (stratum 1) or received low-dose CITA, followed by surgery and postoperative chemotherapy (stratum 2). In the remaining patients, after 2 cycles of CITA, responders received the CITA regimen before resection (stratum 3), and nonresponders were switched to ifosfamide, pirarubicin, etoposide, and carboplatin (ITEC; stratum 4). Intensified chemotherapeutic regimens with autologous hematopoietic stem-cell transplantation (SCT) after resection were an optional treatment for patients with refractory/metastatic disease. RESULTS The 5-year event-free and overall survival rates of HB patients were 74.2% and 89.9%, respectively, for stratum 1, 84.8% and 90.8%%, respectively, for stratum 2, 71.6% and 85.9%%, respectively, for stratum 3, and 59.1% and 67.3%%, respectively, for stratum 4. The outcomes for CITA responders were significantly better than those for nonresponders, whose outcomes remained poor despite salvage therapy with a second-line ITEC regimen or SCT. The late effects, ototoxicity, cardiotoxicity, and delayed growth, occurred in 61, 18, and 47 patients, respectively. Thirteen secondary malignant neoplasms (SMNs), including 10 leukemia, occurred, correlating with higher exposure to pirarubicin and younger age at diagnosis. CONCLUSION The JPLT-2 protocol achieved up-front resectability in PRETEXT I/II patients with no annotation factors, and satisfactory survival in patients who were CITA responders in the remaining patients. However, outcomes for CITA nonresponders were unsatisfactory, despite therapy intensification with ITEC regimens and SCT. JPLT-2 had a relatively low incidence of cardiotoxicity but high rates of SMNs.

Neuroblastoma: A Tough Nut to Crack

2016 ◽  
pp. e548-e557 ◽  
Author(s):  
Frank Speleman ◽  
Julie R. Park ◽  
Tara O. Henderson
Keyword(s):  
Clinical Trials ◽  
Late Effects ◽  
Association Studies ◽  
Genomic Analysis ◽  
The United States ◽  
Malignant Neoplasms ◽  
Genome Wide Association Studies ◽  
Second Malignant Neoplasms ◽  
Tumor In Childhood

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.

Synchronous Wilms Tumor and Fibrolamellar Hepatocellular Carcinoma: Report of a Case

2000 ◽  
Vol 3 (5) ◽  
pp. 492-496 ◽  
Author(s):  
Anirban Maitra ◽  
Dharamdas M. Ramnani ◽  
Linda R. Margraf ◽  
Adi F. Gazdar
Keyword(s):  
Hepatocellular Carcinoma ◽  
Solid Tumors ◽  
Molecular Analysis ◽  
Wilms Tumor ◽  
P53 Mutation ◽  
Malignant Neoplasms ◽  
Fibrolamellar Hepatocellular Carcinoma ◽  
First Report ◽  
Second Malignant Neoplasms

Fibrolamellar hepatocellular carcinoma (FHCC) is a unique histologic variant of HCC that occurs in a younger subset of patients than classical HCC, and is associated with a better prognosis. Wilms tumor (WT) is a malignant embryonal neoplasm of the kidney and is one of the most common solid tumors of childhood, occurring at an estimated frequency of 1 in 8000 to 10,000 births. Although second malignant neoplasms (SMNs) following therapy for WTs have been reported in the liver, the coexistence of HCC and WT is extremely rare. We present the first report of a synchronous ana-plastic WT and FHCC in a previously healthy 4-year-old girl. Despite the presence of focal immunohistochemical positivity for p53 in the WT, molecular analysis failed to reveal a germline or somatic p53 mutation, and was inconclusive in establishing a clonal relation between the two tumors.

Second Malignant Neoplasms Following Multiple Myeloma: A Cohort Study On 744 Patients Treated 1997-2011

Blood ◽  
2013 ◽  
Vol 122 (21) ◽  
pp. 3100-3100 ◽  
Author(s):  
Martina Kleber ◽  
Kerstin Höck ◽  
Gabriele Ihorst ◽  
Bernd Koch ◽  
Ralph Waesch ◽  
...  
Keyword(s):  
Risk Factors ◽  
Multiple Myeloma ◽  
Alkylating Agents ◽  
Malignant Neoplasm ◽  
Individual Risk ◽  
Malignant Neoplasms ◽  
Seer Database ◽  
Educational Grant ◽  
Second Malignant Neoplasms

Abstract Introduction Second malignant neoplasms after first-line therapy and maintenance-therapy have been reported in clinical studies; however, no risk factors have been established. Moreover, second malignant neoplasms is gaining increasingly more attention as multiple myeloma patients live longer and data from randomized studies suggest there are associations between certain newer drugs and the risk of developing second malignant neoplasms. Clinical trials are not statically powered to define risk factors for second malignant neoplasms. Methods We have conducted a large study designed to define the rate of second malignant neoplasms in a well-characterized clinical multiple myeloma cohort. We identified 744 consecutive patients treated at our institution between 1997-2011 and analyzed 1. onset of second malignant neoplasms, 2. patient characteristics (age, gender, familiar risks, smoking status, immune status), 3. frequency of different neoplasms, 4. potential multiple myeloma specific risk factors and 5. possible treatment-related risk factors (novel agents, autologous stem cell transplantation [single vs. tandem (t)-ASCT], allogeneic (allo-) SCT, frequency of alkylating agents, cycles/lines of therapy and ionizing radiation). Results 118 (16%) multiple myeloma patients developed second malignant neoplasms. Prior or synchronous malignant neoplasms were observed in 83 patients (63%) and second malignant neoplasms developed subsequent to multiple myeloma in 49 (37%) patients. Overall, most (77%) of these neoplasms were solid tumors; whereas hematological neoplasms with 23% were prominently observed subsequent to multiple myeloma. Furthermore, multiple myeloma who developed second malignant neoplasms (versus those who did not) were older, predominantly male, had IgG-MM and more CTx-cycles, use of steroids, alkylators, lenalidomide/thalidomide and radiotherapy, but lacked laboratory abnormalities nor did they have more ASCTs or bortezomib therapy. The risk of dying of multiple myeloma due to disease progression remained substantially higher than that of developing a second malignant neoplasm (cumulative incidence at 20 years: 78% vs. 11%, respectively). However, since multiple myeloma prognosis increases and death at 20 years of follow-up decreases, the development of second malignant neoplasms remains highly relevant (Figure 1; comparison of the SEER database with our UKF data). Conclusions Matching the SEER database with our data (Figure 1) confirms the rate of second malignant neoplasms at 20 years of follow-up at around 10%, whereas death from other causes (multiple myeloma) seems to substantially decrease. Further comparison is currently under way and will expand our knowledge on the development of second malignant neoplasms. Our prior (Hasskarl J.,..Engelhardt M. 2011) and previous analyses suggest that physicians need to discuss individual risk-benefit ratios with patients and stay updated as more knowledge becomes available on this topic. It is likely that second malignant neoplasms in multiple myeloma patients remain important given that the prognosis in multiple myeloma has substantially increased and patients live significantly longer. Disclosures: Kleber: Celgene: Educational grant Other. Engelhardt:Celgene: Educational grant Other.

scholarly journals Risk of solid subsequent malignant neoplasms after childhood Hodgkin lymphoma—Identification of high‐risk populations to guide surveillance: A report from the Late Effects Study Group

Cancer ◽  
2018 ◽  
Vol 125 (8) ◽  
pp. 1373-1383 ◽  
Author(s):  
Anna S. Holmqvist ◽  
Yanjun Chen ◽  
Jennifer Berano Teh ◽  
Canlan Sun ◽  
Jillian M. Birch ◽  
...  
Keyword(s):  
High Risk ◽  
Hodgkin Lymphoma ◽  
Late Effects ◽  
Malignant Neoplasms ◽  
Study Group ◽  
High Risk Populations

scholarly journals Late effects of treatment for wilms' tumor. A report from the national wilms' tumor study group

Cancer ◽  
1991 ◽  
Vol 67 (2) ◽  
pp. 331-336 ◽  
Author(s):  
Audrey E. Evans ◽  
Patricia Norkool ◽  
Ilonka Evans ◽  
Norman Breslow ◽  
Giulio J. D'Angio
Keyword(s):  
Late Effects ◽  
Wilms Tumor ◽  
Study Group ◽  
Tumor Study ◽  
Tumor Study Group

scholarly journals Balancing cure and long-term risks in acute lymphoblastic leukemia

Hematology ◽  
2014 ◽  
Vol 2014 (1) ◽  
pp. 190-197 ◽  
Author(s):  
Lewis B. Silverman
Keyword(s):  
Acute Lymphoblastic Leukemia ◽  
Late Effects ◽  
Pediatric Patients ◽  
Lymphoblastic Leukemia ◽  
Malignant Neoplasms ◽  
Second Malignant Neoplasms ◽  
Current Therapies ◽  
Cure Rates ◽  
Long Term Survivors

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.

scholarly journals Second Malignant Neoplasms in Patients With Rhabdomyosarcoma

2021 ◽  
Vol 11 ◽  
Author(s):  
Hongnan Zhen ◽  
Zhikai Liu ◽  
Hui Guan ◽  
Jiabin Ma ◽  
Wenhui Wang ◽  
...  
Keyword(s):  
Risk Factors ◽  
Soft Tissues ◽  
Survival Rates ◽  
Malignant Neoplasm ◽  
The United States ◽  
Malignant Neoplasms ◽  
Second Malignant Neoplasm ◽  
Second Malignant Neoplasms ◽  
Risk Patients ◽  
Independent Risk Factors

ObjectiveRhabdomyosarcoma (RMS) is a rare malignant tumor. The main treatment modality is comprehensive with chemotherapy, radiotherapy, and surgery. With the advancement in recent decades, patient survival has been prolonged, and long-term complications are attracting increasing attention among both physicians and patients. This study aimed to present the survival of patients with RMS and analyze the risk factors for the development of a second malignant neoplasm (SMN).MethodsThe Surveillance, Epidemiology, and End Results (SEER) Program 18 registry database from 1973 to 2015 of the National Cancer Institute of the United States was used for the survival analyses, and the SEER 9 for the SMN analysis.ResultsThe 5-, 10-, and 20-year overall survival rates of the patients with RMS were 45%, 43%, and 33%, respectively. The risk of SMN was significantly higher in patients with RMS compared to the general population (SIR=1.95, 95% CI: 1.44 – 2.57, p < 0.001). The risk of developing SMN was increased in multiple locations, including the bones and joints (SIR = 35.25) soft tissues including the heart (SIR = 22.5), breasts (SIR = 2.10), male genital organs (SIR = 118.14), urinary system (SIR = 2.36), brain (SIR = 9.21), and all nervous system organs (SIR = 8.59). The multivariate analysis indicated that RMS in the limbs and earlier diagnosis time were independent risk factors for the development of SMN. Patients with head and neck (OR = 0.546, 95% CI: 0.313 – 0.952, p = 0.033) and trunk RMS (OR = 0.322, 95% CI: 0.184 – 0.564. p < 0.001) and a later diagnosis time were less likely to develop SMN (OR = 0.496, 95% CI: 0.421 – 0.585, p < 0.001).ConclusionThis study describes the risk factors associated with the development of SMN in patients with RMS, which is helpful for the personalized screening of high-risk patients with RMS.

Second Malignant Neoplasms: Assessment and Strategies for Risk Reduction

2012 ◽  
Vol 30 (30) ◽  
pp. 3734-3745 ◽  
Author(s):  
Marie E. Wood ◽  
Victor Vogel ◽  
Andrea Ng ◽  
Lewis Foxhall ◽  
Pamela Goodwin ◽  
...  
Keyword(s):  
Health Care Providers ◽  
Late Effects ◽  
Relative Survival ◽  
Risk Groups ◽  
Etiologic Factor ◽  
Malignant Neoplasms ◽  
Care Providers ◽  
Number Of Patients ◽  
Second Malignant Neoplasms ◽  
Screening And Prevention

Improvements in early detection, supportive care, and treatment have resulted in an increasing number of cancer survivors, with a current 5-year relative survival rate for all cancers combined of approximately 66.1%. For some patients, these survival advances have been offset by the long-term late effects of cancer and its treatment, with second malignant neoplasms (SMNs) comprising one of the most potentially life-threatening sequelae. The number of patients with SMNs is growing, with new SMNs now representing about one in six of all cancers reported to the National Cancer Institute's Surveillance, Epidemiology, and End Results (SEER) Program. SMNs reflect not only the late effects of therapy but also the influence of shared etiologic factors (in particular, tobacco and excessive alcohol intake), genetic susceptibility, environmental exposures, host effects, and combinations of factors, including gene-environment interactions. For selected SMNs, risk is also modified by age at exposure and attained age. SMNs can be categorized into three major groups according to the predominant etiologic factor(s): (1) treatment-related, (2) syndromic, and (3) those due to shared etiologic exposures, although the nonexclusivity of these groups should be underscored. Here we provide an overview of SMNs in survivors of adult-onset cancer, summarizing the current, albeit limited, clinical evidence with regard to screening and prevention, with a focus on the provision of guidance for health care providers. The growing number of patients with second (and higher-order) cancers mandates that we also further probe etiologic influences and genetic variants that heighten risk, and that we better define high-risk groups for targeted preventive and interventional clinical strategies.

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