Journal articles on the topic 'Bone cancers in children and adolescents'
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1
Badar, Farhana, and Shahid Mahmood. "Epidemiology of cancers in Lahore, Pakistan, among children, adolescents and adults, 2010–2012: a cross-sectional study part 2." BMJ Open 7, no. 12 (December 2017): e016559. http://dx.doi.org/10.1136/bmjopen-2017-016559.
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ObjectivesTo estimate the cancer incidence by age group for the Lahore district population within the Punjab Cancer Registry (PCR), Pakistan. The average annual population of Lahore was 9.8 million in 2010–2012. This is a sequel to a study published earlier.DesignA cross-sectional study.SettingThe registry has 19 centres in Lahore reporting their data to the coordinating office located within the Shaukat Khanum Memorial Cancer Hospital and Research Center (SKMCH&RC), Lahore, Pakistan.ParticipantsData existing in the PCR database, based on a confirmed diagnosis of cancer from 1 January 2010 to 31 December 2012, among the Lahore residents, were reviewed.Outcome measuresCancer counts and the age-standardised incidence rates (ASIR) per 100 000 population were computed by gender, cancer site/type and age group (0–14, 15–19 and ≥20 years).ResultsBetween 2010 and 2012, of the 15 840 new cancers diagnosed, 57% were in females. The ASIRs in age groups 0–14, 15–19 and ≥20 years, among females, were: 6.1, 8.4 and 170.7, respectively, and among males, 9.3, 12.2 and 104.5, respectively. The common diagnoses in children, adolescents and adults were: (1) among females: leukaemia: 2.2; bone tumour: 1.4 and breast cancer: 79.2, respectively, and (2) among males: leukaemia: 3.6; bone tumour: 2.4 and prostate cancer: 10.7, respectively.ConclusionsThe ASIR was higher in adult women than in men, but it was lower in girls and young women than their corresponding male counterparts. Leukaemia was the most common diagnosis in children and bone tumour in adolescents, regardless of gender. Among women, breast cancer and, in men, prostate cancer, were the leading cancer types, in adults. These estimates could be used for the expansion of health coverage in the region including setting-up low cost, diagnostic tests for early detection of cancers.
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Martinez-Rodriguez, Cesar, and Ma del Rocio Banos-Lara. "HMPV in Immunocompromised Patients: Frequency and Severity in Pediatric Oncology Patients." Pathogens 9, no. 1 (January 10, 2020): 51. http://dx.doi.org/10.3390/pathogens9010051.
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Cancer is the first cause of death by disease in childhood globally. The most frequent types of cancers in children and adolescents are leukemias, followed by brain and central nervous system tumors and lymphomas. The recovery rate of cancer in children is around 80% in developed countries and up to 30% in developing countries. Some of the main causes of complications in children and adolescents with cancer are respiratory viral infections, mainly in bone marrow-transplanted patients. Respiratory viruses have been detected in the bronchoalveolar lavage or nasal wash specimens from cancer patients with or without respiratory illness symptoms. Human metapneumovirus (HMPV) is within the ten most common viruses that are encountered in samples from pediatric patients with underlying oncology conditions. In most of cases, HMPV is found as the only viral agent, but co-infection with other viruses or with bacterial agents has also been reported. The discrepancies between the most prevalent viral agents may be due to the different populations studied or the range of viral agents tested. Some of the cases of infection with HMPV in cancer patients have been fatal, especially in those who have received a hematopoietic stem cell transplant. This review seeks to show a general view of the participation of HMPV in respiratory illness as a complication of cancer in childhood and adolescence.
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Małek, Anna, Olga Wysokińska, Klara Iwaniszyn-Zapołoch, Karolina Zadrożna, Piotr Sidorczuk, Mateusz Fabiś, Bartłomiej Wójcik, Justyna Żyga, and Katarzyna Klajda. "Diagnosis of malignant bone tumors in children." Journal of Education, Health and Sport 12, no. 11 (November 8, 2022): 334–40. http://dx.doi.org/10.12775/jehs.2022.12.11.044.
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Bone cancers occur mainly in adolescents and young adults, and benign lesions are more frequent than malignant ones. The most common malignant tumors include osteosarcoma, Ewing's sarcoma and chondrosarcoma. The course of the disease may not be obvious and symptoms may be ambiguous, so it is important to catch them early and take appropriate action. Laboratory tests such as CRP, ESR, LDH, ALP may prove useful in the diagnosis of bone neoplasms in children. All children with suspected malignant bone cancer should undergo an x-ray examination. MRI, CT scans are performed to expand the diagnosis and determine the extent of the disease. The basis of diagnosis is histopathological examination. The result of this study is very important in the selection of therapeutic procedures, it influences decisions regarding the scope and degree of radicality of the operation. It is crucial that the entire diagnostic process is carried out in highly specialized centers that have extensive experience in the diagnosis and treatment of malignant bone tumors in children. It is most beneficial to conduct diagnosis and treatment at the same center, and preferably by the same team of specialists. This team should include doctors from orthopedics, radiology, oncology, radiation therapy and pathomorphology. Conducting diagnosis and treatment in highly specialized centers has a positive effect on the prognosis and quality of life of patients. The purpose of this article is to highlight the early and uncharacteristic symptoms of bone cancer in children and to present the complexity of the diagnostic process, as well as the importance of carrying it out in highly specialized centers with extensive experience in treating pediatric bone malignancies.
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Close, Allison, Brittani Seynnaeve, Kimberly Miller, and Louis Rapkin. "Sex-specific mortality trends in adolescents and young adults with cancer from 2007 to 2016." Journal of Clinical Oncology 37, no. 15_suppl (May 20, 2019): 1570. http://dx.doi.org/10.1200/jco.2019.37.15_suppl.1570.
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1570 Background: Adolescents and young adults (AYA) aged 15-39 years make up approximately 70,000 new oncology cases in the USA. Historically, mortality from cancer has smaller incremental improvements in AYA patients when compared to children and older adults, and not much is known about current sex-specific trends. We assessed overall and sex specific AYA mortality for the last 10 years (2007-2016). Methods: Trends in age-adjusted mortality rates per 100,000 (1972-2016) were obtained from the CDC’s National Center for Health Statistics (NCHS). Average annual percent changes (AAPCs) in relative survival were analyzed using NCI’s JPSurv webtool and mortality AAPCs were quantified using Joinpoint regression analysis. Results: Overall declines in mortality are similar in AYA men and women from 1972-2016, with 54% and 51% decline, respectively. In the most recent 10 years of data (2007 to 2016), combined sex AYA mortality AAPC’s declined by about 0.8% per year, slightly slower than declines in children <15 years (1.3% per year) and adults ages >40 years (1.5% per year). Among AYA males there have been 10 year AAPC mortality declines in leukemia (-1.8%), Hodgkin lymphoma (HL) (-5.1%), Non-Hodgkin lymphoma (NHL) (-4.1%) and melanoma of the skin (-3.4%). For AYA females, 10 year AAPC mortality declines occurred in leukemia (-1.9%), ovarian (-1.5%), HL (-10%), NHL (-4.9%) and melanoma (-2.8%). These declines have been offset by stable or increasing mortality rates for several common AYA cancers, including colorectal cancer (CRC) (1.1%) and bone and joint cancer (0.6%) in AYA males. AYA females have experienced mortality increases for CRC (0.6%), bone and joint cancer (0.5%) and uterine corpus cancer (2.8%). Conclusions: In general, mortality rates for both AYA men and women have declined over the past 10 years due to decreased mortality in hematologic malignancies and melanoma. Despite overall improvement, tumor categories in both AYA males and female such as CRC, bone and joint cancer, and uterine corpus cancers show increasing mortality. These diseases require specific investigations by both pediatric and adult researchers.
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Badar, Farhana, and Shahid Mahmood. "Cancer in Lahore, Pakistan, 2010–2019: an incidence study." BMJ Open 11, no. 8 (August 2021): e047049. http://dx.doi.org/10.1136/bmjopen-2020-047049.
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ObjectivesTo study the cancer incidence rates over 10 years (2010–2019), in Lahore, Pakistan.DesignAn incidence study.SettingThe population-based Punjab Cancer Registry was established in 2005 in Lahore, which is the provincial metropolis of the province of Punjab (five rivers), and is located in the northeast region of Pakistan. The coordinating office of the Registry is located within Shaukat Khanum Memorial Cancer Hospital and Research Center. Both the active and passive forms of data collection are used.ParticipantsResidents of the district of Lahore diagnosed with cancer. The average annual population of Lahore was estimated at 11.1 million.Outcome measuresCancer counts and incidence rates per 100 000 population, by age-group, sex and cancer site/type, over 10 years.ResultsIn Lahore, from 2010 to 2019, 58 394 incident cases were reported, with the majority seen in females (57.1%). Adults accounted for 92.2%, adolescents 2.2% and children 5.6% of the total cases. Per 100 000 population, the age-standardised incidence rate was 103.4 for females and 65.6 for males. Among females, the highest incidence rates were recorded for breast cancer (76.7) in adults, bone tumour (1.2) in adolescents and lymphoid leukaemia (1.6) in children, and among males, prostate cancer (10.7) in adults, bone tumour (2.2) in young adults and lymphoid leukaemia (2.4) in children. The age-specific incidence rates peaked in the 60–70 year group, reaching a high of 420 per 100 000 in women and 330 per 1 00 000 men.ConclusionsIn Lahore, the incidence rates for cancers of the breast, prostate, lymphoid leukaemia and bone were among the highest documented. More cases were recorded in females than in males. The results reported could be used as a reference point for assessing the effectiveness of future interventions.
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Brion, Régis, Laura Regnier, Mathilde Mullard, Jérome Amiaud, Françoise Rédini, and Franck Verrecchia. "LIM Kinases in Osteosarcoma Development." Cells 10, no. 12 (December 15, 2021): 3542. http://dx.doi.org/10.3390/cells10123542.
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Tumorigenesis is a long-term and multistage process that often leads to the formation of metastases. During this pathological course, two major events appear to be crucial: primary tumour growth and metastatic expansion. In this context, despite research and clinical advances during the past decades, bone cancers remain a leading cause of death worldwide among paediatric cancer patients. Osteosarcomas are the most common malignant bone tumours in children and adolescents. Notwithstanding advances in therapeutic treatments, many patients succumb to these diseases. In particular, less than 30% of patients who demonstrate metastases at diagnosis or are poor responders to chemotherapy survive 5 years after initial diagnosis. LIM kinases (LIMKs), comprising LIMK1 and LIMK2, are common downstream effectors of several signalization pathways, and function as a signalling node that controls cytoskeleton dynamics through the phosphorylation of the cofilin family proteins. In recent decades, several reports have indicated that the functions of LIMKs are mainly implicated in the regulation of actin microfilament and the control of microtubule dynamics. Previous studies have thus identified LIMKs as cancer-promoting regulators in multiple organ cancers, such as breast cancer or prostate cancer. This review updates the current understanding of LIMK involvement in osteosarcoma progression.
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Jin, Hye Young, and Jun Ah Lee. "Low bone mineral density in children and adolescents with cancer." Annals of Pediatric Endocrinology & Metabolism 25, no. 3 (September 30, 2020): 137–44. http://dx.doi.org/10.6065/apem.2040060.030.
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Taskinen, Mervi, Ulla M. Saarinen-Pihkala, Liisa Hovi, Kim Vettenranta, and Outi Mäkitie. "Bone health in children and adolescents after allogeneic stem cell transplantation." Cancer 110, no. 2 (2007): 442–51. http://dx.doi.org/10.1002/cncr.22796.
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Mostoufi-Moab, Sogol, and Leanne M. Ward. "Skeletal Morbidity in Children and Adolescents during and following Cancer Therapy." Hormone Research in Paediatrics 91, no. 2 (November 27, 2018): 137–51. http://dx.doi.org/10.1159/000494809.
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Skeletal abnormalities are common in children and adolescents diagnosed and treated for a malignancy. The spectrum ranges from mild pain to debilitating osteonecrosis and fractures. In this review, we summarize the impact of cancer therapy on the developing skeleton, provide an update on therapeutic strategies for prevention and treatment, and discuss the most recent advances in musculoskeletal research. Early recognition of skeletal abnormalities and strategies to optimize bone health are essential to prevent long-term skeletal sequelae and diminished quality of life in childhood cancer survivors.
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Metayer, Catherine, Charles F. Lynch, E. Aileen Clarke, Bengt Glimelius, Hans Storm, Eero Pukkala, Timo Joensuu, et al. "Second Cancers Among Long-Term Survivors of Hodgkin’s Disease Diagnosed in Childhood and Adolescence." Journal of Clinical Oncology 18, no. 12 (June 12, 2000): 2435–43. http://dx.doi.org/10.1200/jco.2000.18.12.2435.
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PURPOSE: To quantify the risk of second cancers among long-term survivors of Hodgkin’s disease (HD) diagnosed before 21 years of age and to explore sex-, age-, and site-related differences. PATIENTS AND METHODS: We analyzed data from 5,925 pediatric HD patients, including 2,646 10-year and 755 20-year survivors, who were reported to 16 population-based cancer registries in North America and Europe between 1935 and 1994. RESULTS: A total of 157 solid tumors (observed/expected ratio [O/E] = 7.0; 95% confidence interval [CI], 5.9 to 8.2.) and 26 acute leukemias (O/E = 27.4; 95% CI, 17.9 to 40.2) were reported. Risk of solid tumors remained significantly increased among 20-year survivors (O/E = 6.6, observed [O] = 40, cumulative risk = 6.5%) and persisted for 25 years (O/E = 4.6, O = 15, cumulative risk = 11.7%). Temporal trends for cancers of thyroid, female breast, bone/connective tissue, stomach, and esophagus were consistent with the late effects of radiotherapy. Greater than 50-fold increased risks were observed for tumors of the thyroid and respiratory tract (one lung and one pleura) among children treated before age 10. At older ages (10 to 16 years), the largest number of second cancers occurred in the digestive tract (O/E = 19.3) and breast (O/E = 22.9). Risk of solid tumors increased with decreasing age at HD on a relative but not absolute scale. CONCLUSION: Children and adolescents treated for HD experience significantly increased risks of second cancers at various sites for 2 to 3 decades. Although our results reflect the late effects of past therapeutic modalities, they underscore the importance of lifelong follow-up of pediatric HD patients given early, more aggressive treatments.
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Sharma, Krishna Sagar, Sabita Panthi, Kishor Pradhananga, and Bhaktaman Shrestha. "Institutional Audit of Pediatric and Adolescent Malignancy in Nepal." Nepalese Journal of Cancer 4, no. 1 (October 7, 2020): 78–83. http://dx.doi.org/10.3126/njc.v4i1.31847.
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Background: There have been no records of the incidence of pediatric malignancy recorded in Nepal until recently. The aim of this study is to analyze the cases of pediatric malignancy reported in 2006 in order to find out the relative frequency and geographical distribution of childhood malignancy throughout Nepal.
Methods: All the data for 2006 were collected from the Hospital-based Cancer Registry of Nepal (B P Koirala Memorial Cancer Hospital Registry Programme). All the cases included in the report were analyzed for geographical distribution, age, sex and relative frequency of the various types of childhood malignancies. The cancers were all classified according to the International Classification of Childhood Cancer (ICCC) and separated into 12 major groupings.
Result: A total 343 children and adolescents from seven tertiary care hospitals located in the western, middle and eastern regions of Nepal were registered in the Hospital-based Cancer Registry Program of Nepal. A majority of the patients were from the eastern and mid-eastern regions. The others were from the western, far-western and mid-western regions. Very few of them were from the far-western and mid-western regions. The males had a higher reported rate of malignancies than the females, 60% vs. 40%. The adolescent population (13-19 years old) had 43% of the malignancies: a significant number. Leukemia (33%), lymphoma (18%) and bone tumors (13%) were the first, second and third most common cancers among the 12 groups. The number of reported cases has increased each year from 2003 to 2006.
Discussion: The Hospital-based Cancer Registry was started in 2003. At that time not much attention was given during the collection of data to making note of the different variables. The incidence of pediatric malignancies has not been known till now. This study shows that the relative frequencies of pediatric malignancies and leukemiaare the same as in western countries. Knowledge of the national incidence is necessary in order to make proper policies for the treatment of children with cancer and for research in the field of pediatric oncology. Brain tumors are the second most common cancer in developed countries but in our study it is the fifth most common malignancy: 20% Vs 5%. We have a higher percentage of bone tumors 10% vs. 5% unlike other developed countries where population based registry data are available.
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Malkin, David, Kim E. Nichols, Kristin Zelley, and Joshua D. Schiffman. "Predisposition to Pediatric and Hematologic Cancers: A Moving Target." American Society of Clinical Oncology Educational Book, no. 34 (May 2014): e44-e55. http://dx.doi.org/10.14694/edbook_am.2014.34.e44.
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Our understanding of hereditary cancer syndromes in children, adolescents, and young adults continues to grow. In addition, we now recognize the wide variation in tumor spectrum found within each specific cancer predisposition syndrome including the risk for hematologic malignancies. An increased understanding of the genetic mutations, biologic consequences, tumor risk, and clinical management of these syndromes will improve patient outcome. In this article, we illustrate the diversity of molecular mechanisms by which these disorders develop in both children and adults with a focus on Li-Fraumeni syndrome, hereditary paraganglioma syndrome, DICER1 syndrome, and multiple endocrine neoplasia syndrome. This is followed by a detailed discussion of adult-onset tumors that can occur in the pediatric population including basal cell carcinoma, colorectal cancer, medullary thyroid cancer, and adrenal cortical carcinoma, and the underlying hereditary cancer syndromes that these tumors could indicate. Finally, the topic of leukemia predisposition syndromes is explored with a specific focus on the different categories of syndromes associated with leukemia risk (genetic instability/DNA repair syndromes, cell cycle/differentiation, bone marrow failure syndromes, telomere maintenance, immunodeficiency syndromes, and transcription factors/pure familial leukemia syndromes). Throughout this article, special attention is made to clinical recognition of these syndromes, genetic testing, and management with early tumor surveillance and screening.
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Gordon, Bruce G., Dennis D. Weisenburger, Warren G. Sanger, James O. Armitage, and Peter F. Coccia. "Peripheral T-Cell Lymphoma in Children and Adolescents: Role of Bone Marrow Transplantation." Leukemia & Lymphoma 14, no. 1-2 (January 1994): 1–10. http://dx.doi.org/10.3109/10428199409049645.
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Lones, Mark A., Sherrie L. Perkins, Richard Sposto, Nicole Tedeschi, Marshall E. Kadin, Carl R. Kjeldsberg, John F. Wilson, David L. Zwick, and Mitchell S. Cairo. "Non-Hodgkin’s Lymphoma Arising in Bone in Children and Adolescents Is Associated With an Excellent Outcome: A Children’s Cancer Group Report." Journal of Clinical Oncology 20, no. 9 (May 1, 2002): 2293–301. http://dx.doi.org/10.1200/jco.2002.06.017.
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PURPOSE: Non-Hodgkin’s lymphoma (NHL) arising in bone is a heterogeneous histologic type of NHL that includes large-cell lymphoma, lymphoblastic lymphoma, and small noncleaved-cell lymphoma. NHL arising in bone is well recognized in adults but is less well characterized and infrequent in children and adolescents. PATIENTS AND METHODS: We performed a retrospective review of Children’s Cancer Group (CCG) studies treating children and adolescents with NHL over a 20-year period (CCG-551, CCG-501, CCG-502, CCG-503, CCG-552, CCG-5911, and CCG-5941) and determined the response and event-free survival (EFS) rates in 31 patients with NHL arising in bone. RESULTS: The patients ranged in age from 3 to 17 years (median, 11 years; mean, 11 years), and 64.5% were male. All 31 (100%) patients achieved complete response. For 31 patients with NHL arising in bone, the product-limit estimated 5-year EFS was 83.8% ± 6.7%. EFS in 17 patients with localized disease (Murphy stages I and II) was 94.1% ± 5.7%, and EFS in 14 patients with disseminated disease (Murphy stage III) was 70.7% ± 12.4% (log-rank P = .10). EFS in 17 patients treated with chemotherapy and radiation was 70.1% ± 11.2%, and EFS in 14 patients treated with chemotherapy without radiation was 100% (P = .03). EFS in 26 patients with histology-directed treatment (LSA2-L2 or ADCOMP for lymphoblastic, other therapy for nonlymphoblastic) was 92.2% ± 5.3%, and in five patients with nonhistology-directed treatment it was 40.0% ± 21.9% (P < .001). CONCLUSION: NHL arising in bone is a heterogeneous type of NHL that makes up approximately 2.0% of NHL in children and adolescents on CCG studies. Response and survival in this young age group seem superb, with histology-directed treatment protocols without radiation in both localized and disseminated disease.
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Escobedo-Monge, Marlene Fabiola, Enrique Barrado, Joaquín Parodi-Román, María Antonieta Escobedo-Monge, María Carmen Torres-Hinojal, and José Manuel Marugán-Miguelsanz. "Magnesium Status and Ca/Mg Ratios in a Series of Children and Adolescents with Chronic Diseases." Nutrients 14, no. 14 (July 18, 2022): 2941. http://dx.doi.org/10.3390/nu14142941.
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Magnesium (Mg) is an essential divalent cation involved in various enzymatic reactions that regulate vital biological functions. The main goal was to evaluate Mg status and its association with nutritional indicators in 78 children and adolescents with chronic diseases. We assessed anthropometric, biochemical, diet, body composition, and bone densitometry valuations. Serum Mg and Ca levels were determined using the standardized method and diet calcium (Ca) and Mg consumption by a prospective 72 h diet survey. Mean serum Ca (9.9 mg/dL), Mg (2.08 mg/dL) dietary Ca (102% DRI: Dietary Reference Intake), and Mg intake (105% DRI) were normal. A total of 45% had hypomagnesemia, 12% had hypermagnesemia, and 26% and 24% had inadequate and high Mg intake, respectively. Only 6% of patients had poor Mg intake and hypomagnesemia, and 54% and 90% of our series had an elevated serum Ca/Mg ratio > 4.70 (mean 4.79) and a low Ca/Mg intake ratio < 1.70 (mean 1.06), respectively. Both Ca/Mg ratios were linked with the risk of developing other chronic conditions such as cardiovascular disease, type 2 diabetes, syndrome metabolic, and even several cancers. Therefore, 79% of children and adolescents with chronic diseases were at elevated risk of having abnormal Mg status and developing other chronic illnesses.
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Shafi, Ahmed Mohamed Abdel, Sarah Fendius, and Wael Ibrahim Awad. "Ewing sarcoma of the rib presenting with hemothorax." Asian Cardiovascular and Thoracic Annals 27, no. 9 (June 13, 2019): 779–81. http://dx.doi.org/10.1177/0218492319858560.
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Ewing sarcoma is a malignant primitive neuroectodermal tumor, primarily affecting the long bones in young children and adolescents. It is a rare disease but is the most common malignancy of the chest wall, accounting for 6%–16% of chest wall cancers. We report a case of Ewing sarcoma of the rib presenting with massive hemothorax in a teenage boy, with rapid disease progression.
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Fox, E., B. C. Widemann, P. O. Whitcomb, A. Aikin, E. Dombi, M. Lodish, C. A. Stratakis, S. Steinberg, S. A. Wells, and F. M. Balis. "Phase I/II trial of vandetanib in children and adolescents with hereditary medullary thyroid carcinoma." Journal of Clinical Oncology 27, no. 15_suppl (May 20, 2009): 10014. http://dx.doi.org/10.1200/jco.2009.27.15_suppl.10014.
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10014 Background: Mutations in the RET protooncogenecause hereditary medullary thyroid carcinoma (MTC) including Multiple Endocrine Neoplasia (MEN) Type 2A, Type 2B and familial MTC. Vandetanib inhibits VEGFR, EGFR, and RET tyrosine kinases and is active in adults with hereditary MTC. Methods: We are conducting a trial of vandetanib in children and adolescents with RET mutations and MTC. Safety is evaluated at each dose level in adolescents (13–18 years) prior to enrolling children (5–12 years). In the absence of dose limiting toxicity (DLT), intrapatient dose escalation is permitted after cycle 2. To assess bone toxicity, growth plate volume is measured using MRI. Response is monitored using tumor measurements (RECIST), serum biomarkers, calcitonin (CTN), and carcinoembryonic antigen (CEA). Results: Five adolescents and 2 children were enrolled at the 100 mg/m2 dose level, 3 adolescents were dose escalated to 150 mg/m2. Six have MEN2B (M918T RET mutation). Median (range) baseline CTN was 12,200 pg/mL (2,300–67,000) and CEA was 104 mcg/L (5–325). Dose limiting diarrhea was observed in 1/5 adolescents at the 100 mg/m2 and 1/3 adolescents escalated to 150 mg/m2. No DLTs were observed in children receiving 100 mg/m2. Non-DLT included elevated TSH (n = 6), rash (n = 5), anorexia (n = 3), diarrhea (n = 2), hypertension (n = 1), and fatigue (n = 1). Median (range) percent change in growth plate volume during therapy was -18% (-44% to + 50%). All patients had linear growth during therapy. Serum CTN and CEA decreased by ≥ 50% in 6/7 and 2/7 patients, respectively. Tumor size decreased in 6/6 patients with M918T RET mutations; 2 achieved RECIST partial response after 6 and 12 cycles. Conclusions: Preliminary results suggest that vandetanib has activity in children and adolescents with MEN2B associated MTC. Vandetanib 100 mg/m2 was well tolerated. Linear growth was not impaired. [Table: see text]
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Calvo, Felipe A., David Ortiz de Urbina, Luis Sierrasesúmaga, Oscar Abuchaibe, Ignacio Azinovic, Federico Antillon, Manuel Santos, and José Canadell. "Intraoperative radiotherapy in the multidisciplinary treatment of bone sarcomas in children and adolescents." Medical and Pediatric Oncology 19, no. 6 (1991): 478–85. http://dx.doi.org/10.1002/mpo.2950190606.
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Bordbar, Mohammadreza, Ali Sarfaraz, Sezaneh Haghpanah, Omidreza Zekavat, Soheila Zareifar, and Tahereh Zarei. "The Outcome of Children With Malignant Bone Tumors: A Single-Center Experience." Global Pediatric Health 8 (January 2021): 2333794X2110422. http://dx.doi.org/10.1177/2333794x211042238.
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Malignant bone tumors (MBT) account for 3% to 5% of cancers in children younger than 15 years. We aimed to report the outcome of children with MBT in 10 years in Southern Iran. During the study period, 100 patients (57 Osteosarcoma, 43 Ewing sarcoma) with an M/F ratio of 1.56 and a median age of 13.8 years (3.8-17.9) were diagnosed. Metastasis occurred in 27% of patients, mostly in the first 3 months of diagnosis. The mean survival time of MBT altogether was 94.1 months (95% CI: 86.5-101.7). The 5-year overall survival and event-free survivals were 85.2% (95% CI: 74%-91.8%) and 69.2% (95% CI: 56%-79%), respectively. Metastasis was the only independent risk factor of death in our study cohort (Hazard ratio 36.7, 95% CI: 4.8-282.6, P = .001) MBT in children mostly occur in adolescent boys. About one-third of them become metastatic, which is significantly associated with poor outcomes.
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Kim, Eun Ho, Mi-Sook Kim, Akihisa Takahashi, Masao Suzuki, Guillaume Vares, Akiko Uzawa, Akira Fujimori, Tatsuya Ohno, and Sei Sai. "Carbon-Ion Beam Irradiation Alone or in Combination with Zoledronic acid Effectively Kills Osteosarcoma Cells." Cancers 12, no. 3 (March 16, 2020): 698. http://dx.doi.org/10.3390/cancers12030698.
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Osteosarcoma (OSA) is the most common malignant bone tumor in children and adolescents. The overall five-year survival rate for all bone cancers is below 70%; however, when the cancer has spread beyond the bone, it is about 15–30%. Herein, we evaluated the effects of carbon-ion beam irradiation alone or in combination with zoledronic acid (ZOL) on OSA cells. Carbon-ion beam irradiation in combination with ZOL significantly inhibited OSA cell proliferation by arresting cell cycle progression and initiating KHOS and U2OS cell apoptosis, compared to treatments with carbon-ion beam irradiation, X-ray irradiation, and ZOL alone. Moreover, we observed that this combination greatly inhibited OSA cell motility and invasion, accompanied by the suppression of the Pi3K/Akt and MAPK signaling pathways, which are related to cell proliferation and survival, compared to individual treatments with carbon-ion beam or X-ray irradiation, or ZOL. Furthermore, ZOL treatment upregulated microRNA (miR)-29b expression; the combination with a miR-29b mimic further decreased OSA cell viability via activation of the caspase 3 pathway. Thus, ZOL-mediated enhancement of carbon-ion beam radiosensitivity may occur via miR-29b upregulation; co-treatment with the miR-29b mimic further decreased OSA cell survival. These findings suggest that the carbon-ion beam irradiation in combination with ZOL has high potential to increase OSA cell death.
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Lu, Ko-Hsiu, Renn-Chia Lin, Jia-Sin Yang, Wei-En Yang, Russel J. Reiter, and Shun-Fa Yang. "Molecular and Cellular Mechanisms of Melatonin in Osteosarcoma." Cells 8, no. 12 (December 12, 2019): 1618. http://dx.doi.org/10.3390/cells8121618.
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Osteosarcoma, the most common primary bone malignancy, occurs most frequently in adolescents with a peak of incidence at 11–15 years. Melatonin, an indole amine hormone, shows a wide range of anticancer activities. The decrease in melatonin levels simultaneously concurs with the increase in bone growth and the peak age distribution of osteosarcoma during puberty, so melatonin has been utilized as an adjunct to chemotherapy to improve the quality of life and clinical outcomes. While a large amount of research has been conducted to understand the complex pleiotropic functions and the molecular and cellular actions elicited by melatonin in various types of cancers, a few review reports have focused on osteosarcoma. Herein, we summarized the anti-osteosarcoma effects of melatonin and its underlying molecular mechanisms to illustrate the known significance of melatonin in osteosarcoma and to address cellular signaling pathways of melatonin in vitro and in animal models. Even in the same kind of osteosarcoma, melatonin has been sparingly investigated to counteract tumor growth, apoptosis, and metastasis through different mechanisms, depending on different cell lines. We highlighted the underlying mechanism of anti-osteosarcoma properties evoked by melatonin, including antioxidant activity, anti-proliferation, induction of apoptosis, and the inhibition of invasion and metastasis. Moreover, we discussed the drug synergy effects of the role of melatonin involved and the method to fortify the anti-cancer effects on osteosarcoma. As a potential therapeutic agent, melatonin is safe for children and adolescents and is a promising candidate for an adjuvant by reinforcing the therapeutic effects and abolishing the unwanted consequences of chemotherapies.
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Kube, Stefanie J., Claudia Blattmann, Stefan S. Bielack, Leo Kager, Peter Kaatsch, Thomas Kühne, Benjamin Sorg, et al. "Secondary malignant neoplasms after bone and soft tissue sarcomas in children, adolescents, and young adults." Cancer 128, no. 9 (February 23, 2022): 1787–800. http://dx.doi.org/10.1002/cncr.34110.
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Kafa, Satria Kharimul Qolbi. "The Influence of Visual, Auditorial and Kinesthetic Learning Styles on the Learning Achievement of Cancer Fighting Children." Pedagogia : Jurnal Pendidikan 11, no. 2 (August 23, 2022): 89–98. http://dx.doi.org/10.21070/pedagogia.v11i2.1198.
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Cancer is a dangerous disease with a high mortality rate. From ancient times until now, cancer has various types of cancer of the blood, eye, bone and others. Every human being can get the disease regardless of age, starting from children to adults. Children up to adolescents are school children, so when a child is diagnosed with cancer, it will affect his education. Therefore, this study discusses the effect of visual, auditory, and kinesthetic learning styles on the learning achievement of those diagnosed with cancer as children cancer fighters. This research uses experimental methods through 3 types of styles with data sources of interviews, observation and documentation in the form of reports on teaching and learning activities. The data is processed by Anova Variant Analysis. From the results of these calculations, there are effects of visual, auditory, and kinesthetic learning styles on the learning achievement of children who fight cancer.
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Suryanarayan, Kaveri, Jonathan J. Shuster, Sarah S. Donaldson, Robert E. Hutchison, Sharon B. Murphy, and Michael P. Link. "Treatment of Localized Primary Non-Hodgkin's Lymphoma of Bone in Children: A Pediatric Oncology Group Study." Journal of Clinical Oncology 17, no. 2 (February 1999): 456. http://dx.doi.org/10.1200/jco.1999.17.2.456.
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PURPOSE: The treatment of primary lymphoma of bone (PLB) in children has traditionally included radiotherapy to the primary site; more recently, it has included systemic chemotherapy. Because of concern about the untoward effects of treatment in a disease that is curable, we attempted to determine whether radiotherapy can be safely excluded from treatment. PATIENTS AND METHODS: The results of three consecutive Pediatric Oncology Group (POG) studies were examined to determine the impact on outcome of radiotherapy as adjunctive treatment in children and adolescents receiving chemotherapy for early-stage primary lymphoma of bone. RESULTS: From 1983 to 1997, 31 patients with localized PLB were entered onto POG studies of early-stage non-Hodgkin's lymphoma (NHL). Between 1983 and 1986, seven patients were treated with 8 months of chemotherapy with irradiation (XRT) of the primary site. After 1986, patients were treated without XRT; four received 8 months of chemotherapy, and 20 received 9 weeks of chemotherapy. Primary sites were the femur (nine), tibia (eight), mandible (five), mastoid (one), maxilla (one), zygomatic arch (one), rib (one), clavicle (one), scapula (one), ulna (one), talus (one), and calcaneous (one). Histologic classification revealed 21 cases of large-cell lymphoma, five cases of lymphoblastic lymphoma, two cases of small, noncleaved-cell lymphoma, and three cases of NHL that could not be classified further. One patient relapsed at a distant site 22 months after completion of therapy. There have been no deaths. CONCLUSION: Localized PLB is curable in most children and adolescents with a 9-week chemotherapy regimen of modest intensity, and radiotherapy is an unnecessary adjunct.
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Lipton, Jeffrey M., Christine L. S. Molmenti, Pooja Desai, Alexander Lipton, Steven R. Ellis, and Adrianna Vlachos. "Early Onset Colorectal Cancer: An Emerging Cancer Risk in Patients with Diamond Blackfan Anemia." Genes 13, no. 1 (December 26, 2021): 56. http://dx.doi.org/10.3390/genes13010056.
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Diamond Blackfan anemia (DBA) is a rare inherited bone marrow failure syndrome, the founding member of a class of disorders known as ribosomopathies. Most cases result from loss of function mutations or deletions in 1 of 23 genes encoding either a small or large subunit-associated ribosomal protein (RP), resulting in RP haploinsufficiency. DBA is characterized by red cell hypoplasia or aplasia, poor linear growth and congenital anomalies. Small case series and case reports demonstrate DBA to be a cancer predisposition syndrome. Recent analyses from the Diamond Blackfan Anemia Registry of North America (DBAR) have quantified the cancer risk in DBA. These studies reveal the most prevalent solid tumor, presenting in young adults and in children and adolescents, to be colorectal cancer (CRC) and osteogenic sarcoma, respectively. Of concern is that these cancers are typically detected at an advanced stage in patients who, because of their constitutional bone marrow failure, may not tolerate full-dose chemotherapy. Thus, the inability to provide optimal therapy contributes to poor outcomes. CRC screening in individuals over the age of 50 years, and now 45 years, has led to early detection and significant improvements in outcomes for non-DBA patients with CRC. These screening and surveillance strategies have been adapted to detect familial early onset CRC. With the recognition of DBA as a moderately penetrant cancer risk syndrome a rational screening and surveillance strategy will be implemented. The downstream molecular events, resulting from RP haploinsufficiency and leading to cancer, are the subject of significant scientific inquiry.
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Savitskaya, Yulia, Genaro Rico, Luis Linares, Ernesto Delgado, Elisa Martinez, Rene Tellez, Erendira Estrada, Norma Marin, and Clemente Ibarra. "Circulating biomarker-IgM complexes in the serum of pediatric patients with osteosarcoma and Ewing tumor (127.9)." Journal of Immunology 188, no. 1_Supplement (May 1, 2012): 127.9. http://dx.doi.org/10.4049/jimmunol.188.supp.127.9.
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Abstract Osteosarcoma (OS) and Ewing tumor (ET) together account for the majority of musculoskeletal sarcomas diagnosed in patients of all ages and nearly 90% of those occurring in children and adolescents. Natural IgM antibodies to tumor antigen have been reported in patients with early-stage cancer, and a panel of serum antibodies can detect cancer many years prior to imaging detection. Circulating immune complexes formed by tumor antigens and immunoglobulin M (IgM) represent a novel class of biomarkers with diagnostic value for early cancer detection. In the present study, we investigated the presence of ANG-IgM, VEGF-IgM, bFGF-IgM, PDGF-IgM in the sera of children with bone tumors. The study included 174 pediatric patients with newly diagnosed bone tumors. Express ELISA techniques have been developed to quantify immune complexes in INR. Serum samples from all patients were analyzed for the presence of biomarker-IgM immune complexes. We compared the serum level of angiogenic factors and immune complexes in children with different types of bone tumors. The highest concentration of VEGF-IgM was detected in ET patients and ANG-IgM in OS patients. The present study demonstrated increased ANG-IgM expression in the sera OS patients and a positive correlation between its expression levels and tumor vascularity evaluated by CTA. Our results have potential applications as biomarkers for controlling unwanted angiogenesis, early diagnosis and response to therapy in children with bone tumors.
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Avnet, Sofia, Luigi Falzetti, Alberto Bazzocchi, Chiara Gasperini, Fulvia Taddei, Enrico Schileo, and Nicola Baldini. "Individual Trajectories of Bone Mineral Density Reveal Persistent Bone Loss in Bone Sarcoma Patients: A Retrospective Study." Journal of Clinical Medicine 11, no. 18 (September 15, 2022): 5412. http://dx.doi.org/10.3390/jcm11185412.
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Multiagent chemotherapy offers an undoubted therapeutic benefit to cancer patients, but is also associated with chronic complications in survivors. Osteoporosis affects the quality of life of oncologic patients, especially at the paediatric age. However, very few studies have described the extent of loss of bone mineral density (BMD) in bone sarcoma patients. We analysed a retrospective series of children and adolescents with primary malignant bone tumours (52 osteosarcoma and 31 Ewing sarcoma) and retrieved their BMD at diagnosis and follow-up as Hounsfield units (HU). We studied their individual BMD trajectories before and after chemotherapy up to 5 years, using routine chest CT scan and attenuation thresholds on T12 vertebrae ROI. At one year, bone sarcoma patients showed significant bone loss compared to diagnosis: 17.6% and 17.1% less for OS and EW, respectively. Furthermore, a bone loss of more than 49.2 HU at one-year follow-up was predictive of the persistence of a reduced bone mass over the following 4 years, especially in patients with EW. At 4 years, only 26% and 12.5% of OS and EW, respectively, had recovered or improved their BMD with respect to the onset, suggesting a risk of developing morbidities related to a low BMD in those subjects.
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Hejazi, Sasan, Mehran Noroozi, Mohammad Amin Kuhestani, Farid Ghazizadeh, Mohammad Karmiyar, Ebrahim Sadeghi, Ahad Ghazavi, and Hamidreza Houshmand. "Effective factors on clinical outcomes of children with osteosarcoma and Ewing bone tumors." Immunopathologia Persa 6, no. 2 (May 23, 2020): e26-e26. http://dx.doi.org/10.34172/ipp.2020.26.
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Introduction: Malignant bone tumors account for 6% of childhood malignancies. The peak incidence rate for bone cancer is in adolescents. Geographic and race differences in incidence rate of bone tumors are important for correct diagnosis and treatment. Objectives: This study aimed to investigate the demographic features of patients with malignant bone tumors and prognostic factors. Patients and Methods: In a retrospective study, records of all patients diagnosed and treated for malignant bone tumor from March 1996 to February 2014 were investigated. Data were collected using checklists and analyzed using SPSS software. Results: The most common type of bone tumor was Ewing sarcoma (73.7%) in patients who were over 10 years old (87.5%). Primary site of disease in most cases of osteosarcoma and Ewing sarcoma was distal femur (50% and 27.3%, respectively). Most common clinical manifestation was local pain and mass (88.5%). Regarding metastasis, 72.7% of osteosarcoma patients and 32.4% of Ewing sarcoma patients had metastasis. There was not statistically relationship between survival rate and gender, age group and stage of disease at diagnosis time. One, three and five-year survival rate was lower than the previous studies. Conclusion: The incidence of Ewing sarcoma is more then osteosarcoma in West Azerbaijan. In our study there were no statistically important differences in survival rates of patients with metastasis at diagnosis time and those without metastasis.
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Galila, Mokhtar M., Ebeid FS Fatma, Ishak AI Sherif, Ragab A. Iman, and Yousef AS Khaled. "Functional And Survival Outcome Of Egyptian Children And Adolescents With Malignant Bone Tumors: An Experience In A Setting Of Limited Health Resource." Forum of Clinical Oncology 9, no. 1 (July 12, 2019): 3–9. http://dx.doi.org/10.2478/fco-2018-0001.
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Abstract Objective Evaluate outcome of paediatric malignant bone tumours at Ain Shams University, Egypt, from January 2003 to July 2016. Methods Retrospective data analysis regarding clinico-epidemiological aspects, treatment outcomes, survival analysis and musculoskeletal tumour society score (MSTS score). Results The study included 37 patients; 22 had Ewing sarcoma (ES) and 15 had osteosarcoma, male: female ratio 0.85:1, median ages of 11. The overall frequency was 2.3% among all cancers. There is wide range of time lag until diagnosis. Patients with ES were significantly younger than those with osteosarcoma were. Swelling was the most common presenting symptom and femur was the most common affected site. Fifteen patients fulfilled MSTS criteria; most of them had excellent MSTS score, which significantly affected by type of surgery. ES patients were treated with POG#9354/CCG#7942 protocols and osteosarcoma with CCG#7921 protocol. Limb salvage was the most common type for surgical local control. Most common cause of death was relapse, whereas infection was the most common complication of treatment. 1-year, 2-year, 3-year overall-survival of osteosarcoma were 93.3%, 40%, and 13.3% respectively and 77.3%, 40.9%, and 18.2% respectively for ES. 1-year, 2-year, 3-year event-free-survival were 80%, 40%, and 13.3% respectively and 72.7%, 22.7%, and 18.2% respectively for ES patients. Conclusion Although survival rates for malignant bone tumours are still unsatisfactory, the functional outcome of extremity tumours after limb salvage procedures is promising
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Chen, Zhuo-Yuan, Huiqin Yang, Jie Bu, Qiong Chen, Zhen Yang, and Hui Li. "Prognosis Implication of a Novel Metabolism-Related Gene Signature in Ewing Sarcoma." Journal of Oncology 2021 (December 10, 2021): 1–14. http://dx.doi.org/10.1155/2021/3578949.
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Ewing sarcoma (ES) is one of the most common bone cancers in adolescents and children. Growing evidence supports the view that metabolism pathways play critical roles in numerous cancers (He et al. (2020)). However, the correlation between metabolism-associated genes (MTGs) and Ewing sarcoma has not been investigated systematically. Here, based on the univariate Cox regression analysis, we get survival genes from differentially expressed genes (DEGs) from Gene Expression Omnibus (GEO) cohort. Multivariate Cox regression analysis and least absolute shrinkage and selection operator (LASSO) regression analysis were employed to establish the MTG signature. Comprehensive survival analyses including receiver operating characteristic (ROC) curves and Kaplan–Meier analysis were applied to estimate the independent prognostic value of the signature. The ICGC cohort served as the validation cohort. A nomogram was constructed based on the risk score of the MTG signature and other independent clinical variables. The CIBERSORT algorithm was applied to estimate immune infiltration. In addition, we explored the correlation between MTG signature and immune checkpoints. Collectively, this work presents a novel MTG signature for prognostic prediction of Ewing sarcoma. It also suggests six genes that are potential prognostic indicators and therapeutic targets for ES.
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Wells, R. J., W. G. Woods, J. D. Buckley, L. F. Odom, D. Benjamin, I. Bernstein, D. Betcher, S. Feig, T. Kim, and F. Ruymann. "Treatment of newly diagnosed children and adolescents with acute myeloid leukemia: a Childrens Cancer Group study." Journal of Clinical Oncology 12, no. 11 (November 1994): 2367–77. http://dx.doi.org/10.1200/jco.1994.12.11.2367.
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PURPOSE The objectives of this study were to determine if the addition of etoposide, thioguanine, and dexamethasone to daunorubicin and cytarabine (five-drug regimen) during induction would improve remission induction rates and survival of children with acute myeloid leukemia (AML) when compared with the standard regimen of cytarabine and daunorubicin (7 + 3) and whether allogeneic bone marrow transplantation (BMT) or intensive chemotherapy consolidation with or without maintenance would give a superior outcome. PATIENTS AND METHODS A total of 591 assessable children with AML entered Childrens Cancer Group (CCG) trial 213 between January 1986 and February 1989. The status of patients as of September 1, 1992 forms the basis of this report. The results were compared with previous AML studies. RESULTS The projected survival rate of all patients at 5 years is 39% (event-free survival [EFS] rate, 31%), which is superior to that of the prior CCG study (P = .01). The induction rate was 79% for 7 + 3 and 76% for the five-drug regimen (not significant). Comparisons of BMT to chemotherapy favored BMT, but these differences do not always reach statistical significance (eg, 5-year disease-free survival [DFS] rate, 46% v 38% [P = .06] with donor available and 54% v 37% [P = .002] if treated according to protocol intent). No benefit for maintenance therapy was found and, in some comparisons, it was inferior to discontinuation of therapy (5-year survival rate, 46% v 68%, P < .01). CONCLUSION The 5-year EFS rate of patients with AML is 31% and has improved. The five-drug induction regimen is no better than standard induction, BMT appears superior to chemotherapy, and maintenance therapy was not beneficial.
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Goldsby, Robert, Cynthia Burke, Rajaram Nagarajan, Tianni Zhou, Zhengjia Chen, Neyssa Marina, Debra Friedman, Joseph Neglia, Paul Chuba, and Smita Bhatia. "Second solid malignancies among children, adolescents, and young adults diagnosed with malignant bone tumors after 1976." Cancer 113, no. 9 (November 1, 2008): 2597–604. http://dx.doi.org/10.1002/cncr.23860.
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Mullard, Mathilde, Marie Cadé, Sarah Morice, Maryne Dupuy, Geoffroy Danieau, Jérome Amiaud, Sarah Renault, et al. "Sonic Hedgehog Signature in Pediatric Primary Bone Tumors: Effects of the GLI Antagonist GANT61 on Ewing’s Sarcoma Tumor Growth." Cancers 12, no. 11 (November 19, 2020): 3438. http://dx.doi.org/10.3390/cancers12113438.
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Osteosarcoma (OS) and Ewing’s sarcoma (ES) are the most common malignant bone tumors in children and adolescents. In many cases, the prognosis remains very poor. The Sonic hedgehog (SHH) signaling pathway, strongly involved in the development of many cancers, regulate transcription via the transcriptional factors Gli1-3. In this context, RNAseq analysis of OS and ES cell lines reveals an increase of some major compounds of the SHH signaling cascade in ES cells, such as the transcriptional factor Gli1. This increase leads to an augmentation of the transcriptional response of Gli1 in ES cell lines, demonstrating a dysregulation of Gli1 signaling in ES cells and thus the rationale for targeting Gli1 in ES. The use of a preclinical model of ES demonstrates that GANT61, an inhibitor of the transcriptional factor Gli1, reduces ES primary tumor growth. In vitro experiments show that GANT61 decreases the viability of ES cell, mainly through its ability to induce caspase-3/7-dependent cell apoptosis. Taken together, these results demonstrates that GANT61 may be a promising therapeutic strategy for inhibiting the progression of primary ES tumors.
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Leonard, Mary B. "Assessment of bone health in children and adolescents with cancer: Promises and pitfalls of current techniques." Medical and Pediatric Oncology 41, no. 3 (July 11, 2003): 198–207. http://dx.doi.org/10.1002/mpo.10337.
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Weiss, Aaron R., Elizabeth R. Lyden, James R. Anderson, Douglas S. Hawkins, Sheri L. Spunt, David O. Walterhouse, Suzanne L. Wolden, et al. "Histologic and Clinical Characteristics Can Guide Staging Evaluations for Children and Adolescents With Rhabdomyosarcoma: A Report From the Children's Oncology Group Soft Tissue Sarcoma Committee." Journal of Clinical Oncology 31, no. 26 (September 10, 2013): 3226–32. http://dx.doi.org/10.1200/jco.2012.44.6476.
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Purpose To simplify the recommended staging evaluation by correlating tumor and clinical features with patterns of distant metastasis in newly diagnosed patients with embryonal rhabdomyosarcoma (ERMS) or alveolar rhabdomyosarcoma (ARMS). Patients and Methods Patient data from the Intergroup Rhabdomyosarcoma Study Group and the Children's Oncology Group over two periods were analyzed: 1991 to 1997 and 1999 to 2004. We used recursive partitioning analyses to identify factors (including histology, age, regional nodal and distant metastatic status, tumor size, local invasiveness, and primary site) that divided patients into subsets with the most different rates of metastatic disease. Results Of the 1,687 patients analyzed, 5.7% had lung metastases, 4.8% had bone involvement, and 6% had bone marrow (BM) involvement. Rhabdomyosarcoma (RMS) without local invasion (T1) had a low rate of metastasis for all distant sites, especially ERMS (0% bone, 0% BM). ARMS with local invasion (T2) had a higher rate of metastasis for all distant sites (13% lung, 18% bone, 23% BM). ERMS, T2 also had a higher rate of metastatic lung involvement (9%). The likelihood of bone or BM involvement increased in the presence of lung metastases (41% with, 6% without). Regional nodal metastases (N1) predicted a high rate of metastasis in all distant sites (14% lung, 14% bone, 18% BM). A staging algorithm was developed. Conclusion Staging studies in childhood RMS can be tailored to patients' presenting characteristics. Bone marrow aspirate and biopsy and bone scan are unnecessary in at least one third of patients with RMS.
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Kutluk, M. Tezer, and Akif Yeşilipek. "Pediatric Cancer Registry in Turkey 2009-2021 (TPOG & TPHD)." Journal of Clinical Oncology 40, no. 16_suppl (June 1, 2022): e22020-e22020. http://dx.doi.org/10.1200/jco.2022.40.16_suppl.e22020.
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e22020 Background: The pediatric cancers is in the global agenda to improve the survival rates which is still low in LMICs although it is more than 80% in HICs. More than 300.000 pediatric cancer cases annually are expected in children and adolescents aged 0-14 globally. Registry is the first step of an efficient cancer control. Here, we present the most updated results of the pediatric cancer registry fromTurkey. Methods: Turkish Pediatric Oncology Group and Turkish Pediatric Hematology Association has established the pediatric cancer registry in 2002. The childhood cancer cases registered between 2009-2021 was included in this analysis. International Childhood Cancer Classification System was used for the classification. Essential demographic findings, ICD-O-3 morphology and topography codes were recorded for each case. Results: During the 13 years from 2009 to 2021, 24080 cases were registered. For all cases, median age was 6.7 year (0-19; M/F 13461/10609, 4 hermaphrodite, 6 unknown). Age distribution was 0-4 yrs, 40.8%; 5-9 yrs, 23.8%; 10-14 yrs, 23.3%; 15-19 yrs, 12.1%) The distribution of the tumor types were [number of cases, percentage of total, median age yrs, M/F]: Leukemia (5819, 24.2%, 5.5, 3366/2453); Lymphoma & other RES tumors (4446, 18.5%, 9.8, 2956/1487, 1 hermaphrodite & 2 unknown); CNS [brain & spinal] (3730, 15.5%, 6.8, 2061/1668, 1 unkown); Symphatetic system (1965, 8.2%, 2.3, 1010/955); Retinoblastoma (675, 2.8%, 1.4, 375/300); Renal (1160, 4.8%, 3.1, 557/601, 1 hermaphrodite & 1 unknown); Liver (409, 1.7%, 2.2, 234/175); Malignant bone (1584, 6.6%, 12.6, 864/720); Soft tissue sarcomas (1726, 7.2%, 7.7, 983/743); Germ cell (1593, 6.6%, 9.6, 588/1001, 2 hermaphrodite, 2 unknown); Carcinoma & other malignant epithelial (804, 3.3%, 13.5, 381/423); Other/non-specific malignant (169, 0.7%, 7.9, 86/83). Five year survival rate was found as 72.3%. Conclusions: The registry shows that the survival rates for children and adolescents have been improved to 72% which reflects the status of the pediatric cancer care in Turkey. The data from this work became a valuable source for all stakeholders in national and international level working on improvement the pediatric cancer control.
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Purz, Sandra, Christine Mauz-Körholz, Dieter Körholz, Dirk Hasenclever, Antje Krausse, Ina Sorge, Kathrin Ruschke, et al. "[18F]Fluorodeoxyglucose Positron Emission Tomography for Detection of Bone Marrow Involvement in Children and Adolescents With Hodgkin's Lymphoma." Journal of Clinical Oncology 29, no. 26 (September 10, 2011): 3523–28. http://dx.doi.org/10.1200/jco.2010.32.4996.
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Purpose Currently, a routine bone marrow biopsy (BMB) is performed to detect bone marrow (BM) involvement in pediatric Hodgkin's lymphoma (HL) stage greater than IIA. [18F]fluorodeoxyglucose positron emission tomography (FDG-PET) is increasingly used for the initial staging of HL. The value of using FDG-PET to detect BM involvement has not been sufficiently defined. We compared the results of BMBs and FDG-PET for the diagnosis of BM involvement in a large pediatric group with HL. Patients and Methods The initial staging of 175 pediatric patients with newly diagnosed classical HL stage greater than IIA was determined by using BMB, FDG-PET, chest computed tomography (CT), and magnetic resonance imaging (MRI) or CT of the neck, abdomen, and pelvis. Staging images were prospectively evaluated by a central review board. Skeletal regions that were suggestive of BM involvement by either method were re-evaluated by using different imaging modalities. In suspicious cases, bone scintigraphy was performed. If follow-up FDG-PET scans were available, the remission of skeletal lesions during treatment was evaluated. Results BMB results were positive in seven of 175 patients and were identified by FDG-PET. FDG-PET scans showed BM involvement in 45 patients. In addition, the lesions of 32 of these 45 patients had a typical multifocal pattern. In 38 of 39 follow-up positron emission tomography scans, most of the skeletal lesions disappeared after chemotherapy. There was no patient with skeletal findings suggestive of BM involvement by MRI or CT with a negative FDG-PET. Conclusion FDG-PET is a sensitive and specific method for the detection of BM involvement in pediatric HL. The sensitivity of a BMB appears compromised by the focal pattern of BM involvement. Thus, FDG-PET may safely be substituted for a BMB in routine staging procedures.
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Eapen, Mary, Mary M. Horowitz, John P. Klein, Richard E. Champlin, Fausto R. Loberiza, Olle Ringdén, and John E. Wagner. "Higher Mortality After Allogeneic Peripheral-Blood Transplantation Compared With Bone Marrow in Children and Adolescents: The Histocompatibility and Alternate Stem Cell Source Working Committee of the International Bone Marrow Transplant Registry." Journal of Clinical Oncology 22, no. 24 (December 15, 2004): 4872–80. http://dx.doi.org/10.1200/jco.2004.02.189.
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PurposePeripheral-blood stem cells (PBSC) may be used as an alternative to bone marrow (BM) for allogeneic transplantation. Despite lack of data on PBSC transplantation in children, there has been a change in clinical practice, with increasing numbers of children receiving PBSC allografts.Patients and MethodsWe compared the results of 143 PBSC and 630 BM transplants from human leukocyte antigen–identical sibling donors in children aged 8 to 20 years with acute leukemia. PBSC transplant recipients were older, and were more likely to have advanced leukemia, receive growth factors post-transplantation, and have undergone transplantation more recently. Risks of acute and chronic graft-versus-host disease (GVHD), treatment-related mortality, relapse, treatment failure (relapse or death), and overall mortality were compared using Cox proportional hazards regression to adjust for potentially confounding factors.ResultsHematopoietic recovery was faster after PBSC transplantation. Risks of grade 2 to 4 acute GVHD were similar, but chronic GVHD risk was higher after PBSC transplantation (relative risk [RR], 1.85; 95% CI, 1.28 to 2.66; P = .001). In contrast to reports in adults, treatment-related mortality (RR, 1.89; 95% CI, 1.28 to 2.80; P = .001), treatment failure (RR, 1.31; 95% CI, 1.03 to 1.68; P = .03), and mortality (RR, 1.38; 95% CI, 1.07 to 1.79; P = .01) were higher after PBSC transplantation. Risks of relapse were similar.ConclusionThese data suggest poorer outcomes after PBSC compared with BM transplantation in children after adjusting for relevant risk factors. Given the trend toward increased use of PBSC allografts in children, prospective clinical trials are required to determine their appropriate role in this group of patients.
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Lézot, Frédéric, Isabelle Corre, Sarah Morice, Françoise Rédini, and Franck Verrecchia. "SHH Signaling Pathway Drives Pediatric Bone Sarcoma Progression." Cells 9, no. 3 (February 26, 2020): 536. http://dx.doi.org/10.3390/cells9030536.
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Primary bone tumors can be divided into two classes, benign and malignant. Among the latter group, osteosarcoma and Ewing sarcoma are the most prevalent malignant primary bone tumors in children and adolescents. Despite intensive efforts to improve treatments, almost 40% of patients succumb to the disease. Specifically, the clinical outcome for metastatic osteosarcoma or Ewing sarcoma remains poor; less than 30% of patients who present metastases will survive 5 years after initial diagnosis. One common and specific point of these bone tumors is their ability to deregulate bone homeostasis and remodeling and divert them to their benefit. Over the past years, considerable interest in the Sonic Hedgehog (SHH) pathway has taken place within the cancer research community. The activation of this SHH cascade can be done through different ways and, schematically, two pathways can be described, the canonical and the non-canonical. This review discusses the current knowledge about the involvement of the SHH signaling pathway in skeletal development, pediatric bone sarcoma progression and the related therapeutic options that may be possible for these tumors.
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Rosolen, Angelo, Sherrie L. Perkins, C. Ross Pinkerton, R. Paul Guillerman, John T. Sandlund, Catherine Patte, Alfred Reiter, and Mitchell S. Cairo. "Revised International Pediatric Non-Hodgkin Lymphoma Staging System." Journal of Clinical Oncology 33, no. 18 (June 20, 2015): 2112–18. http://dx.doi.org/10.1200/jco.2014.59.7203.
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Purpose Treatment and prognosis of pediatric non-Hodgkin lymphoma (NHL) have improved dramatically in the last 30 years. However, the St Jude NHL staging classification for pediatric NHL was developed more than 35 years ago. The most recent Lugano lymphoma staging classification focused on adult lymphoma. Furthermore, major limitations of the current pediatric NHL staging classification include lack of consideration of new distinct pediatric NHL histologic entities; absence of recognition of frequent skin, bone, kidney, ovarian, and other organ involvement; and lack of newer precise methods to detect bone marrow and CNS involvement, minimal disease quantification, and highly sensitive imaging technologies. Methods An international multidisciplinary expert panel convened in Frankfurt, Germany, in 2009 at the Third International Childhood, Adolescent and Young Adult NHL Symposium to develop a revised international pediatric NHL staging system (IPNHLSS), addressing limitations of the current pediatric NHL staging system and creating a revised classification. Evidence-based disease distribution and behavior were reviewed from multiple pediatric cooperative group NHL studies. Results A revised IPNHLSS was developed incorporating new histologic entities, extranodal dissemination, improved diagnostic methods, and advanced imaging technology. Conclusion This revised IPNHLSS will facilitate more precise staging for children and adolescents with NHL and facilitate comparisons of efficacy across different treatment strategies, various institutions, multicenter trials, and cooperative groups by allowing for reproducible pediatric-based staging at diagnosis and relapse.
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Müller, Daniel A., and Unai Silvan. "On the biomechanical properties of osteosarcoma cells and their environment." International Journal of Developmental Biology 63, no. 1-2 (2019): 1–8. http://dx.doi.org/10.1387/ijdb.190019us.
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Although rare among the general population, bone malignancies have a high rate of incidence among children and adolescents and are associated with high mortality rates. Osteosarcoma (also known as osteogenic sarcoma) is the most frequent primary cancer of bone and shows a high tendency to metastasize to the lung. Despite the frequent use of osteosarcoma-derived cell lines in basic biomechanical research and for the evaluation of cell responses to new biomaterials, the mechanical phenotype and the differences between osteosarcoma cells and related cell types, such as mesenchymal cells, osteoblasts and osteocytes, remain largely unknown. In the present review we summarize current knowledge of the biophysical and mechanical properties of the niche of primary osteosarcomas and of the malignant cells, and discuss the impact of these features on the progression of malignancy.
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Das, Aparna, Farnaz Nobi, Gobinda Banik, and Md Azizul Kahhar. "Vertebral Compression Fractures as a Presenting Feature of AcuteLymphoblastic Leukemia." Journal of Medicine 17, no. 2 (October 23, 2016): 120–21. http://dx.doi.org/10.3329/jom.v17i2.30078.
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Acute lymphoblastic leukemia is one of the most common malignancies in children and adolescent accounting for 30% of all the cancers. We report an interesting case of Acute lymphoblastic leukaemia(ALL) with an unusual presentation.This 15year- old girl came with a progressive low back pain for three months following a history of fall for 5 month ago with intact consciousness. The physical examination showed gibbus over lumbar spine with tenderness. Images showed generalized osteopenia with multiple vertebral body collapse.Complete blood count revealed pancytopenia with raised ESRwithout any blast cell. Bone marrow aspirate was suggestive of ALL.Young ALL patients usually present with symptoms due to cytopenias, fever and bone pains. Although asymptomatic skeletal involvement may be present in patients with ALL, rarely patients present with pathological fractures. Therefore a high index of suspicion is needed to diagnose such case.J MEDICINE July 2016; 17 (2) : 120-121
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Gillespie, Anne Ingalls, and Madalynn Neu. "Youth and Pet Survivors: Exploring the Experiences of Pediatric Oncology and Bone Marrow Transplant Patients in a Virtual Animal-Assisted Therapy Pen Pal Program." Journal of Pediatric Oncology Nursing 37, no. 6 (August 4, 2020): 368–76. http://dx.doi.org/10.1177/1043454220944122.
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Introduction: YAPS™ (Youth and Pet Survivors™) is a form of virtual animal-assisted therapy (AAT), a pen pal program designed for children and adolescents with cancer and/or having a bone marrow transplant (BMT) to engage in virtual visits with a dog or a cat (who has also been treated for cancer or serious medical illness) through letter writing and pictures. The purpose of this qualitative descriptive study was to explore the experiences of YAPS participants over time and to explore how virtual AAT may be an additional or alternative intervention to the traditional form of AAT, which involves live visits with animals, primarily dogs. Method: Open-ended, face-to-face interviews were conducted throughout the participants’ involvement with their animal pen pal. Interviews were digitally recorded. Data were analyzed using a content analysis method. Results: Fifteen children and adolescents, aged 7 to 16 years, participated. Three main themes and five subthemes were found, including connection, shared experience, and friendship. Themes suggested that a virtual AAT letter writing program can provide a source of fun and a way to process the cancer experience with a dog or cat pen pal who has also faced cancer or serious medical treatment. Discussion: Interventions that promote well-being for pediatric oncology and BMT patients are needed, and virtual AAT seems to be one such intervention suited for those who have an affinity for animals and enjoy letter writing. The findings of this study also presented an exciting and intriguing gap for further research in virtual AAT.
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Liao, Nannan, Till Koehne, Jan Tuckermann, Ioanna Triviai, Michael Amling, Jean-Pierre David, Thorsten Schinke, and Julia Luther. "Osteoblast-specific inactivation of p53 results in locally increased bone formation." PLOS ONE 16, no. 11 (November 18, 2021): e0249894. http://dx.doi.org/10.1371/journal.pone.0249894.
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Inactivation of the tumor suppressor p53 (encoded by the Trp53 gene) is relevant for development and growth of different cancers, including osteosarcoma, a primary bone tumor mostly affecting children and young adolescents. We have previously shown that deficiency of the ribosomal S6 kinase 2 (Rsk2) limits osteosarcoma growth in a transgenic mouse model overexpressing the proto-oncogene c-Fos. Our initial aim for the present study was to address the question, if Rsk2 deficiency would also influence osteosarcoma growth in another mouse model. For that purpose, we took advantage of Trp53fl/fl mice, which were crossed with Runx2Cre transgenic mice in order to inactivate p53 specifically in osteoblast lineage cells. However, since we unexpectedly identified Runx2Cre-mediated recombination also in the thymus, the majority of 6-month-old Trp53fl/fl;Runx2-Cre (thereafter termed Trp53Cre) animals displayed thymic lymphomas, similar to what has been described for Trp53-deficient mice. Since we did not detect osteosarcoma formation at that age, we could not follow our initial aim, but we studied the skeletal phenotype of Trp53Cre mice, with or without additional Rsk2 deficiency. Here we unexpectedly observed that Trp53Cre mice display a unique accumulation of trabecular bone in the midshaft region of the femur and the humerus, consistent with its previously established role as a negative regulator of osteoblastogenesis. Since this local bone mass increase in Trp53Cre mice was significantly reduced by Rsk2 deficiency, we isolated bone marrow cells from the different groups of mice and analyzed their behavior ex vivo. Here we observed a remarkable increase of colony formation, osteogenic differentiation and proliferation in Trp53Cre cultures, which was unaffected by Rsk2 deficiency. Our data thereby confirm a critical and tumorigenesis-independent function of p53 as a key regulator of mesenchymal cell differentiation.
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Stiller, C. A., S. S. Bielack, G. Jundt, and E. Steliarova-Foucher. "Bone tumours in European children and adolescents, 1978–1997. Report from the Automated Childhood Cancer Information System project." European Journal of Cancer 42, no. 13 (September 2006): 2124–35. http://dx.doi.org/10.1016/j.ejca.2006.05.015.
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Bryant, Michele L., Mary A. Worthington, and Kerry Parsons. "Treatment of Osteoporosis/Osteopenia in Pediatric Leukemia and Lymphoma." Annals of Pharmacotherapy 43, no. 4 (March 31, 2009): 714–20. http://dx.doi.org/10.1345/aph.1l567.
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Objective: To evaluate the efficacy and safety of various treatment options for osteopenia and osteoporosis secondary to cancer treatment in pediatric patients undergoing cancer therapy. Data Sources: A systematic search of PubMed (1949–November 2008) and international Pharmaceutical Abstracts (to November 2008) was conducted using the following search terms: osteoporosis, osteopenia, pediatrics, cancer, neoplasms, chemotherapy, bisphosphonates, calcium, vitamin D, calcitonin, and physical therapy. Study Selection and Data Extraction: All prospective studies that evaluated various osteoporosis treatment options in pediatric patients undergoing chemotherapy were included. Results from studies evaluating bisphosphonates and other treatments in children with osteoporosis due to other causes were also included if important safety and efficacy data were provided. Most commonly reported primary efficacy endpoints included comparisons of bone density parameters measured before and after treatment. Data Synthesis: Four clinical studies and 2 case reports describing treatment with bisphosphonates, specifically alendronate and Pamidronate, for osteoporosis or osteopenia in pediatric cancer patients were identified. Results from the trials showed that these medications were efficacious in reducing bone mineral density loss during cancer therapy and were well tolerated in this special population. Primary efficacy endpoints included improvements in Z-scores measured by dual-energy X-ray absorptiometry scans. The most commonly reported adverse effects included hypocalcemia, mild stomach upset, and infusion-related hyperpyrexia. Four additional clinical trials involving the treatment of osteoporosis or osteopenia in children and adolescents who developed bone degeneration after chronic steroid therapy are also included. In these trials, treatment options such as calcitonin, and calcium and vitamin D supplementation were also shown to be beneficial. Conclusions: The clinical trials published to date are limited to only a few conducted in small populations of patients diagnosed with lymphoblastic leukemia or non–Hodgkin's lymphoma. However, alendronate and Pamidronate both appeared to be effective options in improving bone mineral density scores with minimal adverse effects.
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Zhao, Qian, Jianyong Yan, Wen Li, Ye Yang, Lu You, and Chenguang Qin. "Molecular Mechanism of Gas Anesthetics on the Invasion, Metastasis, and Chemosensitivity of Osteosarcoma Cells." Computational and Mathematical Methods in Medicine 2021 (November 3, 2021): 1–6. http://dx.doi.org/10.1155/2021/6000385.
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Background. Osteosarcoma is one of the most prominent bone cancers which has a predominant occurrence in children and adolescents. This study is focused on determining the effects of treatment of gas anesthetics on invasion, metastasis, and chemosensitivity in the progression of osteosarcoma cells. Material and Methods. The biological effects of the common gas anesthetics—desflurane, isoflurane, and sevoflurane—on osteosarcoma cells were studied and compared. The biological assays were performed for analysis of cell migration and proliferation. Results. Isoflurane and sevoflurane have shown significant inhibition in the osteosarcoma cells at clinically relevant concentrations. Desflurane has shown less potent action on cell migration and inhibition. All three gas anesthetics have shown inhibition in cell proliferation. The effective antiproliferative action was at a clinically significant dose. At low millimolar concentrations, cell apoptosis was moderately affected. Drug combination analysis with chemotherapeutic drugs showed relevant inhibition in cell migration. All three agents showed significant augmentation of chemotherapeutic drugs in suppression and inhibition of inducing apoptosis. The antimigration action is likely to affect the PI3K/AKT pathway and IGF-1. Conclusion. The study demonstrates the proposed mechanisms of gas anesthetics and their differential effects on osteosarcoma cells and their survival, migration, growth, and chemosensitivity.
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Dong, Zihe, Zhipeng Liao, Yonglin He, Chengye Wu, Zixiang Meng, Baolong Qin, Ge Xu, et al. "Advances in the Biological Functions and Mechanisms of miRNAs in the Development of Osteosarcoma." Technology in Cancer Research & Treatment 21 (January 2022): 153303382211173. http://dx.doi.org/10.1177/15330338221117386.
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Osteosarcoma is one of the most common primary malignant bone tumors, mainly occurring in children and adolescents, and is characterized by high morbidity and poor prognosis. MicroRNAs, a class of noncoding RNAs consisting of 19 to 25 nucleotides, are involved in cell proliferation, invasion, metastasis, and apoptosis to regulate the development and progression of osteosarcoma. Studies have found that microRNAs are closely related to the diagnosis, treatment, and prognosis of osteosarcoma patients and have an important role in improving drug resistance in osteosarcoma. This paper reviews the role of microRNAs in the pathogenesis of osteosarcoma and their clinical value, aiming to provide a new research direction for diagnosing and treating osteosarcoma and achieving a better prognosis.
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Lassmann, Michael, Christoph Reiners, and Markus Luster. "Dosimetry and thyroid cancer: the individual dosage of radioiodine." Endocrine-Related Cancer 17, no. 3 (September 2010): R161—R172. http://dx.doi.org/10.1677/erc-10-0071.
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Adjuvant therapy of differentiated thyroid cancer with radioactive iodine (131I) is a standard procedure for the ablation of remnant thyroid tissue following surgery and for the treatment of iodine-avid metastases. Presently, there are two dosimetric concepts for the treatment of thyroid cancer using radioiodine: a) the bone marrow dose limited approach and b) lesion-based dosimetry. Both concepts and their clinical applications are described. In addition, the use of 124I as a diagnostic and dosimetric agent is discussed. Treatment of children and adolescents with radioiodine requires special precautions; individualized approaches in this setting are reviewed. The limitations of treatments aiming at high absorbed doses are addressed as well as the doses to normal organs. Finally, new concepts for further elaborating the potential of thyroid cancer treatment using 131I are introduced.
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He, Tao, Xiaohong Lin, Chaohua Yang, Zhiyu Chen, Linbang Wang, Qiaochu Li, Jingjin Ma, et al. "Theaflavin-3,3 ′ -Digallate Plays a ROS-Mediated Dual Role in Ferroptosis and Apoptosis via the MAPK Pathway in Human Osteosarcoma Cell Lines and Xenografts." Oxidative Medicine and Cellular Longevity 2022 (October 25, 2022): 1–20. http://dx.doi.org/10.1155/2022/8966368.
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Globally, osteosarcoma (OS) is the most prevalent form of primary bone cancer in children and adolescents. Traditional neoadjuvant chemotherapy regimens have reached a bottleneck; thus, OS survivors have unsatisfactory outcomes. Theaflavin-3,3 ′ -digallate (TF3) exhibits potent anticancer properties against many human cancers. Nevertheless, the biological effects and the underlying molecular mechanism of TF3 in human OS remain unclear. The objective of this study was to investigate the effects of TF3 on human OS cell lines and mouse xenograft models. The results showed that TF3 reduced cell viability, suppressed cell proliferation, and caused G0/G1 cell cycle arrest in both MG63 and HOS cell lines in a concentration-dependent manner. TF3 also altered the homeostatic mechanisms for iron storage in the examined cell lines, resulting in an excess of labile iron. Unsurprisingly, TF3 caused oxidative stress through reduced glutathione (GSH) exhaustion, reactive oxygen species (ROS) accumulation, and the Fenton reaction, which triggered ferroptosis and apoptosis in the cells. TF3 also induced MAPK signalling pathways, including the ERK, JNK, and p38 MAPK pathways. Furthermore, oxidative stress was shown to be the primary reason for TF3-induced proliferation inhibition, programmed cell death, and MAPK pathway activation in vitro. Moreover, TF3 exhibited markedly strong antitumour efficacy in vivo in mouse models. In summary, this study demonstrates that TF3 concomitantly plays dual roles in apoptotic and ferroptotic cell death by triggering the ROS and MAPK signalling pathways in both in vitro and in vivo models.