Cancer Research and Treatment

Original Articles

Pediatric cancer

Long-term Outcomes of Protocol-Based Treatment for Newly Diagnosed Medulloblastoma: Won Kee Ahn, Seung Min Hahn, Hong In Yoon, Jeongshim Lee, Eun Kyung Park, Kyu Won Shim, Dong Seok Kim, Chang-Ok Suh, Se Hoon Kim, Chuhl Joo Lyu, Jung Woo Han; Cancer Res Treat. 2024;56(2):652-664. Published online November 30, 2023; DOI: https://doi.org/10.4143/crt.2023.865

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Purpose
The Korean Society of Pediatric Neuro-Oncology (KSPNO) conducted treatment strategies for children with medulloblastoma (MB) by using alkylating agents for maintenance chemotherapy or tandem high-dose chemotherapy (HDC) with autologous stem cell rescue (ASCR) according to the risk stratification. The purpose of the study was to assess treatment outcomes and complications based on risk-adapted treatment and HDC.
Materials and Methods
Fifty-nine patients diagnosed with MB were enrolled in this study. Patients in the standard-risk (SR) group received radiotherapy (RT) after surgery and chemotherapy using the KSPNO M051 regimen. Patients in the high-risk (HR) group received two and four chemotherapy cycles according to the KSPNO S081 protocol before and after reduced RT for age following surgery and two cycles of tandem HDC with ASCR consolidation treatment.
Results
In the SR group, 24 patients showed 5-year event-free survival (EFS) and overall survival (OS) estimates of 86.7% (95% confidence interval [CI], 73.6 to 100) and 95.8% (95% CI, 88.2 to 100), respectively. In the HR group, more infectious complications and mortality occurred during the second HDC than during the first. In the HR group, the 5-year EFS and OS estimates were 65.5% (95% CI, 51.4 to 83.4) and 72.3% (95% CI, 58.4 to 89.6), respectively.
Conclusion
High intensity of alkylating agents for SR resulted in similar outcomes but with a high incidence of hematologic toxicity. Tandem HDC with ASCR for HR induced favorable EFS and OS estimates compared to those reported previously. However, infectious complications and treatment-related mortalities suggest that a reduced chemotherapy dose is necessary, especially for the second HDC.

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Ferroptosis Transcriptional Regulation and Prognostic Impact in Medulloblastoma Subtypes Revealed by RNA-Seq
Christophe Desterke, Yuanji Fu, Jenny Bonifacio-Mundaca, Claudia Monge, Pascal Pineau, Jorge Mata-Garrido, Raquel Francés
Antioxidants.2025; 14(1): 96. CrossRef

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Hematologic malignancy

Incidence and Features of Lymphoid Proliferation and Lymphomas after Solid Organ or Hematopoietic Stem Cell Transplantation in a National Database Cohort: Seung Min Hahn, Myeongjee Lee, JongHoon Hyun, Sungmin Lim, Ji-Man Kang, Jong Gyun Ahn, Dong Jin Joo, Inkyung Jung, Kyong Ihn; Cancer Res Treat. 2024;56(1):305-313. Published online July 18, 2023; DOI: https://doi.org/10.4143/crt.2023.647

Abstract PDF Supplementary Material PubReader ePub: Purpose
Post-transplantation lymphoproliferative disorders (PTLDs) after hematopoietic stem transplantation (HCT) or solid organ transplantation (SOT) result in poorer outcomes, including death. There are limited large cohort data on the incidence and natural course of PTLD in Asians.
Materials and Methods
We investigated PTLD using Korean national health insurance claims data of 47,518 patients who underwent HCT or SOT in 2008-2020. Patient demographics, time and type of PTLD diagnosis, type of PTLD treatment, and death data were collected. We used Fine and Gray subdistribution hazard models to calculate the cumulative incidence and risk factors for PTLD.
Results
During median follow-up of 5.32 years, PTLD occurred in 294 of 36,945 SOT patients (0.79%) and 235 of 10,573 HCT patients (2.22%). Cumulative incidence of PTLD were 0.49% at 1 year, 1.02% at 5 years, and 1.50% at 10 years post-transplantation. Age < 20 years (subdistribution hazard ratio [SHR] of 1.67 in age 10-19, SHR 1.51 in age 0-9), HCT (SHR 3.02), heart transplantation (SHR 2.27), and liver transplantation (SHR 1.47) were significant risk factors for PTLD. The presence of PTLD was associated with an increased risk of death (hazard ratio of 2.84). Overall, 5-year survival of PTLD patients was 68.9% (95% confidence interval, 64.9 to 73.2).
Conclusion
We observed a steady increase in PTLD over 10 years after HCT or SOT in this large cohort study. Pediatric age group, HCT, liver transplantation, and heart transplantation were suggested to be risk factors for PTLD, and PTLD was associated with a higher risk of death.

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Pediatric cancer

Epidemiologic and Clinical Outcomes of Pediatric Renal Tumors in Korea: A Retrospective Analysis of The Korean Pediatric Hematology and Oncology Group (KPHOG) Data: Kyung-Nam Koh, Jung Woo Han, Hyoung Soo Choi, Hyoung Jin Kang, Ji Won Lee, Keon Hee Yoo, Ki Woong Sung, Hong Hoe Koo, Kyung Taek Hong, Jung Yoon Choi, Sung Han Kang, Hyery Kim, Ho Joon Im, Seung Min Hahn, Chuhl Joo Lyu, Hee-Jo Baek, Hoon Kook, Kyung Mi Park, Eu Jeen Yang, Young Tak Lim, Seongkoo Kim, Jae Wook Lee, Nack-Gyun Chung, Bin Cho, Meerim Park, Hyeon Jin Park, Byung-Kiu Park, Jun Ah Lee, Jun Eun Park, Soon Ki Kim, Ji Yoon Kim, Hyo Sun Kim, Youngeun Ma, Kyung Duk Park, Sang Kyu Park, Eun Sil Park, Ye Jee Shim, Eun Sun Yoo, Kyung Ha Ryu, Jae Won Yoo, Yeon Jung Lim, Hoi Soo Yoon, Mee Jeong Lee, Jae Min Lee, In-Sang Jeon, Hye Lim Jung, Hee Won Chueh, Seunghyun Won, the Korean Pediatric Hematology and Oncology Group (KPHOG); Cancer Res Treat. 2023;55(1):279-290. Published online August 11, 2022; DOI: https://doi.org/10.4143/crt.2022.073

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Purpose
Renal tumors account for approximately 7% of all childhood cancers. These include Wilms tumor (WT), clear cell sarcoma of the kidney (CCSK), malignant rhabdoid tumor of the kidney (MRTK), renal cell carcinoma (RCC), congenital mesoblastic nephroma (CMN) and other rare tumors. We investigated the epidemiology of pediatric renal tumors in Korea.
Materials and Methods
From January 2001 to December 2015, data of pediatric patients (0–18 years) newly-diagnosed with renal tumors at 26 hospitals were retrospectively analyzed.
Results
Among 439 patients (male, 240), the most common tumor was WT (n=342, 77.9%), followed by RCC (n=36, 8.2%), CCSK (n=24, 5.5%), MRTK (n=16, 3.6%), CMN (n=12, 2.7%), and others (n=9, 2.1%). Median age at diagnosis was 27.1 months (range 0-225.5) and median follow-up duration was 88.5 months (range 0-211.6). Overall, 32 patients died, of whom 17, 11, 1, and 3 died of relapse, progressive disease, second malignant neoplasm, and treatment-related mortality. Five-year overall survival and event free survival were 97.2% and 84.8% in WT, 90.6% and 82.1% in RCC, 81.1% and 63.6% in CCSK, 60.3% and 56.2% in MRTK, and 100% and 91.7% in CMN, respectively (p < 0.001).
Conclusion
The pediatric renal tumor types in Korea are similar to those previously reported in other countries. WT accounted for a large proportion and survival was excellent. Non-Wilms renal tumors included a variety of tumors and showed inferior outcome, especially MRTK. Further efforts are necessary to optimize the treatment and analyze the genetic characteristics of pediatric renal tumors in Korea.

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Congenital Mesoblastic Nephroma Mimic Wilms Tumor on 18F-FDG PET/CT and PET/MR
Wenzhu Hu, Chunxia Qin, Fuqiang Shao, Mengting Li, Xiaoli Lan
Clinical Nuclear Medicine.2024; 49(4): 353. CrossRef
Progress towards Therapies for Solid Renal Tumors in Children
洁林
Advances in Clinical Medicine.2024; 14(06): 245. CrossRef

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The Efficacy of Alternate Systemic Intravenous Chemotherapy and Intra-arterial Chemotherapy Approach for Eye Globe Salvage in Retinoblastoma: Jung Woo Han, Christopher Seungkyu Lee, Seung Min Hahn, Won Kee Ahn, Hyo Sun Kim, Hyeseon Yun, Sung Chul Lee, Byung Moon Kim, Dong Joon Kim, Chuhl Joo Lyu; Cancer Res Treat. 2023;55(1):270-278. Published online May 24, 2022; DOI: https://doi.org/10.4143/crt.2021.1537

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Purpose
The advances in the treatment of retinoblastoma have enabled salvaging the globe in advanced stages with intra-arterial chemotherapy (IAC). We developed a strategy of alternate application of systemic intravenous chemotherapy (IVC) and IAC (referred to as alternate systemic IVC and IAC; ASIAC) to reduce central nervous metastases during IAC and examined its efficacy and safety in eye globe salvage in this study.
Materials and Methods
Between January 2010 and February 2021, 43 eyes of 40 patients received ASIAC treatment for retinoblastoma at the Yonsei Cancer Center, Yonsei University Health System. Their medical records were reviewed retrospectively to evaluate the eye salvage rate (ESR), defined from diagnosis to enucleation. High-risk retinoblastoma was defined as group D or E by the International Classification of Retinoblastoma.
Results
The study enrolled 38 and five cases of high-risk and low-risk retinoblastoma, respectively. In total, 178 IAC and 410 IVC courses were administered, with a median of 4 (interquartile range [IQR], 3.0 to 5.0) IAC and 9 (IQR, 6.0 to 11) IVC courses per eye, respectively. The 5-year ESR was 60.4%±8.7% for the whole cohort, 100% for low-risk retinoblastoma, and 53.6%±9.8% for high-risk retinoblastoma. Among those diagnosed since 2015, the 5-year ESR for high-risk retinoblastoma was 63.5%±14.0%. Fifteen eyes underwent enucleation; no viable tumor was found in three enucleated eyes. There were no deaths in this cohort.
Conclusion
Primary IAC-IVC (i.e., ASIAC) for patients with retinoblastoma was tolerable and effective in salvaging the eye and maintaining survival.

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Selective ophthalmic arterial injection using a balloon catheter for retinoblastoma: a seven-year clinical evaluation
Sota Oguro, Yi Ning Chen, Takashi Yamane, Makoto Mohri, Shigenobu Suzuki
Japanese Journal of Ophthalmology.2024; 68(4): 346. CrossRef
Hematological Second Primary Malignancy in Pediatric Retinoblastoma: A Case Report and Systematic Review
Seung Hyun Park, Hyun Young Park, Heejin Kim, Jung Woo Han, Jin Sook Yoon
Ophthalmic Plastic & Reconstructive Surgery.2024; 40(5): 487. CrossRef

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Children’s Hepatic Tumors International Collaboration-Hepatoblastoma Stratification (CHIC-HS) System for Pediatric Patients with Hepatoblastoma: A Retrospective, Hospital-Based Cohort Study in South Korea: Pyeong Hwa Kim, Hyun Joo Shin, Hee Mang Yoon, Young Hun Choi, Jung-Man Namgoong, Dae Yeon Kim, Kyung-Nam Koh, Mi-Jung Lee, Haesung Yoon, Chuhl Joo Lyu, Jung Woo Han, Seung Min Hahn, Young Ah Cho; Cancer Res Treat. 2022;54(1):253-258. Published online March 24, 2021; DOI: https://doi.org/10.4143/crt.2021.265

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Purpose
In 2017, the Children’s Hepatic Tumors International Collaboration-Hepatoblastoma Stratification (CHIC-HS) system was introduced. We aimed to evaluate the accuracy of CHIC-HS System for the prediction of event-free survival (EFS) in Korean pediatric patients with hepatoblastoma.
Materials and Methods
This two-center retrospective study included consecutive Korean pediatric patients with histopathologically confirmed hepatoblastoma from March 1988 through September 2019. We compared EFS among four risk groups according to the CHIC-HS system. Discriminatory ability of CHIC-HS system was also evaluated using optimism-corrected C-statistics. Factors associated with EFS were explored using multivariable Cox regression analysis.
Results
We included 129 patients (mean age, 2.6±3.3 years; female:male, 63:66). The 5-year EFS rates in the very low, low, intermediate, and high-risk groups, according to the CHIC-HS system were 90.0%, 82.8%, 73.5%, and 51.3%, respectively. The CHIC-HS system aligned significantly well with EFS outcomes (p=0.004). The optimism-corrected C index of CHIC-HS was 0.644 (95% confidence interval [CI], 0.561 to 0.727). Age ≥ 8 (vs. age ≤ 2; hazard ratio [HR], 2.781; 95% CI, 1.187 to 6.512; p=0.018), PRE-Treatment EXTent of tumor (PRETEXT) stage IV (vs. PRETEXT I or II; HR, 2.774; 95% CI, 1.228 to 5.974; p=0.009), and presence of metastasis (HR, 2.886; 95% CI, 1.457 to 5.719; p=0.002), which are incorporated as the first three nodes in the CHIC-HS system, were independently associated with EFS.
Conclusion
The CHIC-HS system aligned significantly well with EFS outcomes in Korean pediatric patients with hepatoblastoma. Age group, PRETEXT stage, and presence of metastasis were independently associated with EFS.

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The importance of age as a prognostic predictor of childhood hepatoblastoma: an analysis of single-center childhood hepatoblastoma in China
Zhen Li, Hong Jiang, Yuan-Qi Wang, Wen-Qing Wang, Li-Bin Huang, Jun-Cheng Liu, Hong-Man Xue
BMC Pediatrics.2025;[Epub] CrossRef
Elevated serum uric acid is associated with the risk of advanced staging and vascular involvement in patients with hepatoblastoma: a 14-year retrospective study
Yunlan Zhou, Jinning Li, Yanhui Ma, Mengjie Tang, Xiaojun Yuan, Lisong Shen
Frontiers in Oncology.2023;[Epub] CrossRef

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Effectiveness and Safety of Clofarabine Monotherapy or Combination Treatment in Relapsed/Refractory Childhood Acute Lymphoblastic Leukemia: A Pragmatic, Non-interventional Study in Korea: Jung Yoon Choi, Che Ry Hong, Kyung Taek Hong, Hyoung Jin Kang, Seongkoo Kim, Jae Wook Lee, Pil Sang Jang, Nack-Gyun Chung, Bin Cho, Hyery Kim, Kyung-Nam Koh, Ho Joon Im, Jong Jin Seo, Seung Min Hahn, Jung Woo Han, Chuhl Joo Lyu, Eu Jeen Yang, Young Tak Lim, Keon Hee Yoo, Hong Hoe Koo, Hoon Kook, In Sang Jeon, Hana Cho, Hee Young Shin; Cancer Res Treat. 2021;53(4):1184-1194. Published online January 4, 2021; DOI: https://doi.org/10.4143/crt.2020.289

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Purpose
Effectiveness and safety of clofarabine (one of the treatment mainstays in pediatric patients with relapsed/refractory acute lymphoblastic leukemia [ALL]) was assessed in Korean pediatric patients with ALL to facilitate conditional coverage with evidence development.
Materials and Methods
In this multicenter, prospective, observational study, patients receiving clofarabine as mono/combination therapy were followed up every 4-6 weeks for 6 months or until hematopoietic stem cell transplantation (HSCT). Response rates, survival outcomes, and adverse events were assessed.
Results
Sixty patients (2-26 years old; 65% B-cell ALL, received prior ≥ 2 regimen, 68.3% refractory to previous regimen) were enrolled and treated with at least one dose of clofarabine; of whom 26 (43.3%) completed 6 months of follow-up after the last dose of clofarabine. Fifty-eight patients (96.7%) received clofarabine combination therapy. Overall remission rate (complete remission [CR] or CR without platelet recovery [CRp]) was 45.0% (27/60; 95% confidence interval [CI], 32.4 to 57.6) and the overall response rate (CR, CRp, or partial remission [PR]) was 46.7% (28/60; 95% CI, 34.0 to 59.3), with 11 (18.3%), 16 (26.7%), and one (1.7%) patients achieving CR, CRp, and PR, respectively. The median time to remission was 5.1 weeks (95% CI, 4.7 to 6.1). Median duration of remission was 16.6 weeks (range, 2.0 to 167.6 weeks). Sixteen patients (26.7%) proceeded to HSCT. There were 24 deaths; 14 due to treatment-emergent adverse events.
Conclusion
Remission with clofarabine was observed in approximately half of the study patients who had overall expected safety profile; however, there was no favorable long-term survival outcome in this study.

Citations

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Revolutionizing cancer treatment: ROS-induced apoptosis via nanoformulated alkaloids
Swathi Putta, Santhosh Kumar Chinnaiyan, Ramadevi Korni, Venkata Radha Gadela
Journal of Drug Delivery Science and Technology.2025; 104: 106556. CrossRef
Clofarabine

Reactions Weekly.2023; 1958(1): 155. CrossRef
Patient-Level Meta-analysis of Clofarabine in Acute Lymphoblastic Leukemia
Sima Jeha, Hiroaki Goto, André Baruchel, Emmanuelle Boëlle-Le Corfec, Christine Geffriaud-Ricouard, Rob Pieters, Hee Young Shin
Advances in Therapy.2023; 40(12): 5447. CrossRef
Novel Treatments for Pediatric Relapsed or Refractory Acute B-Cell Lineage Lymphoblastic Leukemia: Precision Medicine Era
Shang Mengxuan, Zhou Fen, Jin Runming
Frontiers in Pediatrics.2022;[Epub] CrossRef

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Central nervous system

Atypical Teratoid/Rhabdoid Tumor of the Central Nervous System in Children under the Age of 3 Years: Meerim Park, Jung Woo Han, Seung Min Hahn, Jun Ah Lee, Joo-Young Kim, Sang Hoon Shin, Dong-Seok Kim, Hong In Yoon, Kyung Taek Hong, Jung Yoon Choi, Hyoung Jin Kang, Hee Young Shin, Ji Hoon Phi, Seung-Ki Kim, Ji Won Lee, Keon Hee Yoo, Ki Woong Sung, Hong Hoe Koo, Do Hoon Lim, Hyung Jin Shin, Hyery Kim, Kyung-Nam Koh, Ho Joon Im, Seung Do Ahn, Young-Shin Ra, Hee-Jo Baek, Hoon Kook, Tae-Young Jung, Hyoung Soo Choi, Chae-Yong Kim, Hyeon Jin Park, Chuhl Joo Lyu; Cancer Res Treat. 2021;53(2):378-388. Published online October 28, 2020; DOI: https://doi.org/10.4143/crt.2020.756

Abstract PDF PubReader ePub

Purpose
Atypical teratoid/rhabdoid tumor (ATRT) is a highly aggressive malignancy with peak incidence in children aged less than 3 years. Standard treatment for central nervous system ATRT in children under the age of 3 years have not been established yet. The objective of this study was to analyze characteristics and clinical outcomes of ATRT in children aged less than 3 years.
Materials and Methods
A search of medical records from seven centers was performed between January 2005 and December 2016.
Results
Forty-three patients were enrolled. With a median follow-up of 90 months, 27 patients (64.3%) showed at least one episode of disease progression (PD). The first date of PD was at 160 days after diagnosis. The 1- and 3-year progression-free survivals (PFS) were 51.2% and 28.5%, respectively. The 1- and 3-year overall survivals were 61.9% and 38.1%, respectively. The 3-year PFS was improved from 0% in pre-2011 to 47.4% in post-2011. Excluding one patient who did not receive any further therapy after surgery, 27 patients died due to PD (n=21), treatment-related toxicity (n=5), or unknown cause (n=1). In univariate analysis, factors associated with higher 3-year PFS were no metastases, diagnosis after 2011, early adjuvant radiotherapy, and high-dose chemotherapy (HDCT). In multivariate analysis, the use of HDCT and adjuvant radiotherapy remained significant prognostic factors for PFS (both p < 0.01).
Conclusion
Aggressive therapy including early adjuvant radiotherapy and HDCT could be considered to improve outcomes of ATRT in children under the age of 3 years.

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Metabolic profiling of patient-derived organoids reveals nucleotide synthesis as a metabolic vulnerability in malignant rhabdoid tumors
Marjolein M.G. Kes, Francisco Morales-Rodriguez, Esther A. Zaal, Terezinha de Souza, Natalie Proost, Marieke van de Ven, Marry M. van den Heuvel-Eibrink, Jeroen W.A. Jansen, Celia R. Berkers, Jarno Drost
Cell Reports Medicine.2025; 6(1): 101878. CrossRef
Supratentorial ATRT in a young Infant: Expanding the diagnostic spectrum beyond medulloblastoma
Ali Msheik, Mohamad Aoun, Youssef Fares
Interdisciplinary Neurosurgery.2024; 35: 101857. CrossRef
Radiation Therapy Plays an Important Role in the Treatment of Atypical Teratoid/Rhabdoid Tumors: Analysis of the EU-RHAB Cohorts and Their Precursors
Sabine Frisch, Hanna Libuschewski, Sarah Peters, Joachim Gerß, Katja von Hoff, Rolf-Dieter Kortmann, Karolina Nemes, Stefan Rutkowski, Martin Hasselblatt, Torsten Pietsch, Michael C. Frühwald, Beate Timmermann
International Journal of Radiation Oncology*Biology*Physics.2024; 119(4): 1147. CrossRef
An adult with recurrent atypical teratoid rhabdoid tumor of the spine
Antoinette J Charles, Vanessa L Smith, C Rory Goodwin, Margaret O Johnson
CNS Oncology.2024;[Epub] CrossRef
Dynamic Survival Risk Prognostic Model and Genomic Landscape for Atypical Teratoid/Rhabdoid Tumors: A Population-Based, Real-World Study
Sihao Chen, Yi He, Jiao Liu, Ruixin Wu, Menglei Wang, Aishun Jin
Cancers.2024; 16(5): 1059. CrossRef
ESTRO-SIOPE guideline: Clinical management of radiotherapy in atypical teratoid/rhabdoid tumors (AT/RTs)
Beate Timmermann, Claire Alapetite, Karin Dieckmann, Rolf-Dieter Kortmann, Yasmin Lassen-Ramshad, John H. Maduro, Monica Ramos Albiac, Umberto Ricardi, Damien C. Weber
Radiotherapy and Oncology.2024; 196: 110227. CrossRef
Development and epigenetic regulation of Atypical teratoid/rhabdoid tumors in the context of cell-of-origin and halted cell differentiation
Laura Huhtala, Goktug Karabiyik, Kirsi J Rautajoki
Neuro-Oncology Advances.2024;[Epub] CrossRef
Comparative treatment results of children with atypical teratoid/rhabdoid tumor of the central nervous system in the younger age group
L. V. Olkhova, O. G. Zheludkova, L. S. Zubarovskaya, A. S. Levashov, A. Yu. Smirnova, Yu. V. Dinikina, Yu. V. Kushel, A. G. Melikyan, S. K. Gorelyshev, M. V. Ryzhova, Yu. Yu. Trunin, A. G. Gevorgyan, O. B. Polushkina, V. E. Popov, L. P. Privalova, N. B. Y
Russian Journal of Pediatric Hematology and Oncology.2023; 10(1): 11. CrossRef
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Chi-kong Li, Purna Kurkure, Ramandeep Singh Arora, Bow Wen Chen, Kirill Kirgizov, Yasuhiro Okamoto, Panya Seksarn, Yongmin Tang, Keon Hee Yoo, Bharat Agarwal, Godfrey C.F. Chan, Rashmi Dalvi, Hiroki Hori, Muhammad Saghir Khan, Alice Yu, Akira Nakagawara
JCO Global Oncology.2023;[Epub] CrossRef
Survival and Malignant Transformation of Pineal Parenchymal Tumors: A 30-Year Retrospective Analysis in a Single-Institution
Tae-Hwan Park, Seung-Ki Kim, Ji Hoon Phi, Chul-Kee Park, Yong Hwy Kim, Sun Ha Paek, Chang-Hyun Lee, Sung-Hye Park, Eun Jung Koh
Brain Tumor Research and Treatment.2023; 11(4): 254. CrossRef
Atypical Teratoid/Rhabdoid Tumor in Taiwan: A Nationwide, Population-Based Study
Yen-Lin Liu, Min-Lan Tsai, Chang-I Chen, Noi Yar, Ching-Wen Tsai, Hsin-Lun Lee, Chia-Chun Kuo, Wan-Ling Ho, Kevin Li-Chun Hsieh, Sung-Hui Tseng, James S. Miser, Chia-Yau Chang, Hsi Chang, Wen-Chang Huang, Tai-Tong Wong, Alexander T. H. Wu, Yu-Chun Yen
Cancers.2022; 14(3): 668. CrossRef
Atypical Teratoid Rhabdoid Tumor: A Possible Oriented Female Pathology?
Cinzia Baiano, Rosa Della Monica, Raduan Ahmed Franca, Maria Laura Del Basso De Caro, Luigi Maria Cavallo, Lorenzo Chiariotti, Tamara Ius, Emmanuel Jouanneau, Teresa Somma
Frontiers in Oncology.2022;[Epub] CrossRef
Clinical predictors of survival for patients with atypical teratoid/rhabdoid tumors
Vismaya S. Bachu, Pavan Shah, Adrian E. Jimenez, Adham M. Khalafallah, Jignesh Tailor, Debraj Mukherjee, Alan R. Cohen
Child's Nervous System.2022; 38(7): 1297. CrossRef
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Molecular Cancer Therapeutics.2022; 21(5): 715. CrossRef
Molecular targeted therapies for pediatric atypical teratoid/rhabdoid tumors
Chang Zhang, Hao Li
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The results of multicenter treatment of atypical teratoid/rhabdoid tumors of the central nervous system in children under 3 years
L. V. Olkhova, O. G. Zheludkova, L. S. Zubarovskaya, A. Yu. Smirnova, Yu. V. Dinikina, Yu. V. Kushel, A. G. Melikyan, S. K. Gorelyshev, M. V. Ryzhova, Yu. Yu. Trunin, E. I. Shults, A. G. Gevorgyan, S. V. Gorbatykh, A. N. Kislyakov, V. E. Popov, L. P. Priv
Pediatric Hematology/Oncology and Immunopathology.2021; 20(2): 121. CrossRef

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Loss of Heterozygosity at Chromosome 16q Is a Negative Prognostic Factor in Korean Pediatric Patients with Favorable Histology Wilms Tumor: A Report of the Korean Pediatric Hematology Oncology Group (K-PHOG): Jun Eun Park, O Kyu Noh, Yonghee Lee, Hyoung Soo Choi, Jung Woo Han, Seung Min Hahn, Chuhl Joo Lyu, Ji Won Lee, Keon Hee Yoo, Hong Hoe Koo, Seon-Yong Jeong, Ki Woong Sung; Cancer Res Treat. 2020;52(2):438-445. Published online September 10, 2019; DOI: https://doi.org/10.4143/crt.2019.313

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Purpose
Loss of heterozygosity (LOH) at chromosomes 1p and 16q is a poor prognostic factor in favorable histology Wilms tumor (FHWT). This study investigated the prevalence of LOH at 1p and 16q and evaluated its prognostic value in Korean children with FHWT. Materials and Methods We analyzed 101 FHWT patients who were diagnosed between 1996 and 2016 in Korean Society of Pediatric Hematology Oncology Group hospitals. Using paraffin-embedded kidney tissue samples sent from each center, we reviewed LOH at 1p and 16q in each patient and assessed the prognostic value of LOH status for clinical parameters affecting event-free survival (EFS).
Results
Of the 101 patients, 12 (11.9%) experienced recurrence; the 3-year EFS was 87.6%. LOH at 1p or 16q was detected in 19 patients (18.8%), with five having LOH at both 1q and 16q. The frequency of LOH at 1p was higher among younger patients (p=0.049), but there was no difference in LOH prevalence according to tumor stage. In the multivariate analysis, LOH at 16q was a significant negative prognostic factor affecting EFS (3-year EFS, 73.7% vs. 91.1%; hazard ratio, 3.95; p=0.037), whereas LOH at 1p was not (p=0.786). Conclusion LOH at 16q was a significant negative prognostic factor affecting outcome in Korean pediatric FHWT patients. Due to the small sample size of this study, large-scale multicenter trials are warranted to investigate the prognostic value of LOH at 1p and 16q in Korean children with FHWT.

Citations

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Loss of heterozygosity for chromosomes 16q in Wilms tumors predicts outcomes: A meta-analysis
Yuan-Hua Song, Wen-Ling Li, Zhen Yang, Yan Gao, Zhi-Ping Feng
World Journal of Gastrointestinal Oncology.2024; 16(5): 2159. CrossRef
Inter-Ethnic Variations in the Clinical, Pathological, and Molecular Characteristics of Wilms Tumor
Kia Teng Lim, Amos H. P. Loh
Cancers.2024; 16(17): 3051. CrossRef
Characterization of malignant kidney tumors in childhood by 18F-FDG PET/CT
Jinyu Gou, Xueli Ji, Shuqi Wu, Hui Wang, Suyun Chen
World Journal of Urology.2024;[Epub] CrossRef
Epidemiologic and Clinical Outcomes of Pediatric Renal Tumors in Korea: A Retrospective Analysis of The Korean Pediatric Hematology and Oncology Group (KPHOG) Data
Kyung-Nam Koh, Jung Woo Han, Hyoung Soo Choi, Hyoung Jin Kang, Ji Won Lee, Keon Hee Yoo, Ki Woong Sung, Hong Hoe Koo, Kyung Taek Hong, Jung Yoon Choi, Sung Han Kang, Hyery Kim, Ho Joon Im, Seung Min Hahn, Chuhl Joo Lyu, Hee-Jo Baek, Hoon Kook, Kyung Mi Pa
Cancer Research and Treatment.2023; 55(1): 279. CrossRef
Research Advances of Prognostic Risk Factors of Wilms Tumor
建宇汪
World Journal of Cancer Research.2023; 13(02): 51. CrossRef
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Clinical Risk Factors Influencing Dental Developmental Disturbances in Childhood Cancer Survivors: Chung-Min Kang, Seung Min Hahn, Hyo Sun Kim, Chuhl Joo Lyu, Jae-Ho Lee, Jinae Lee, Jung Woo Han; Cancer Res Treat. 2018;50(3):926-935. Published online October 10, 2017; DOI: https://doi.org/10.4143/crt.2017.296

Abstract PDF Supplementary Material PubReader ePub

Purpose
Although studies regarding dental developmental disturbances after childhood cancer treatment have increased, they have many limitations. Studies analyzing the significance of independent clinical risk factors with regard to the dental health status are also rare. We aimed to investigate the risk factors for dental developmental disturbances, particularly severe disturbances, in childhood cancer survivors (CCS).
Materials and Methods
Oral examinations and retrospective reviews of medical and panoramic radiographs were performed for 196 CCS (mean age, 15.6 years). Cancer type, age at diagnosis, treatment modality, type and accumulated dose of administered drugs, and dose and site of radiation were recorded. Dental developmental disturbances were diagnosed using panoramic radiographs and graded for severity according to the Modified Dental Defect Index (MDDI). Descriptive statistics and multivariate analyseswere performed to determine the association between dental abnormalities and clinical factors.
Results
In total, 109 CCS (55.6%) exhibited at least one dental anomaly, and the median value of MDDI was 2.5. Microdontia (30.6%) was the most prevalent anomaly, followed by tooth agenesis (20.4%), V-shaped roots (14.8%), and taurodontism (10.2%). Multivariate analysis revealed that a young age at diagnosis (≤ 3 years), a history of hematopoietic stem cell transplantation, the use of multiple classes of chemotherapeutic agents (≥ 4 classes), and the use of heavy metal agents were significant risk factors for severe dental disturbances.
Conclusion
CCS with any of the above risk factors for severe developmental disturbances should be comprehensively followed up to minimize adverse consequences to their dental development and preserve their future dental health.

Citations

Citations to this article as recorded by

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Híttalo Carlos Rodrigues De Almeida, Cleomar Donizeth Rodrigues, Luiz Pedro Mendes De Azevedo, Aronita Rosenblatt, Márcia Maria Fonseca Da Silveira, Ana Paula Veras Sobral
International Journal of Paediatric Dentistry.2025; 35(1): 45. CrossRef
A Case of Severe Early Childhood Caries Occurring in a Childhood Cancer Patient
Tatsuya Akitomo, Noriko Niizato, Shunya Ikeda, Yuya Ito, Eimi Tabata, Chieko Mitsuhata, Ryota Nomura
Children.2025; 12(3): 261. CrossRef
Dental Health of Childhood Cancer Survivors—A Report From the Swiss Childhood Cancer Survivor Study (SCCSS)
Carina Nigg, Corinne Matti, Philippa Jörger, Andre O. von Bueren, Cornelia Filippi, Tamara Diesch‐Furlanetto, Zuzana Tomášiková, Claudia E. Kuehni, Grit Sommer
Pediatric Blood & Cancer.2025;[Epub] CrossRef
Prevalence of, and risk factors for, dental sequelae in adolescents who underwent cancer therapy during childhood
Judit Rabassa‐Blanco, Lluís Brunet‐Llobet, Paula Marcote‐Sinclair, Sol Balsells‐Mejía, María Genoveva Correa‐Llano, Jaume Miranda‐Rius
Oral Diseases.2024; 30(2): 604. CrossRef
A Narrative Review of the Association between Dental Abnormalities and Chemotherapy
Tatsuya Akitomo, Yasuko Tsuge, Chieko Mitsuhata, Ryota Nomura
Journal of Clinical Medicine.2024; 13(16): 4942. CrossRef
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Elinor Halperson, Hanan Badarneh, Ella Zion, Helly Kruchenezki, Gal Goldstein, Sagui Gavri, David Zangen, Avia Fux-Noy
Frontiers in Oral Health.2024;[Epub] CrossRef
Dental dysplasia in childhood cancer survivors: a case series of permanent tooth abnormalities
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K. Seremidi, K. Kavvadia, A. Kattamis, A. Polychronopoulou
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Anna Jodłowska, Lidia Postek-Stefańska
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Assessment of Risk Factors for Dental Developmental Disorders in Pediatric Cancer Survivors
Jihyun Lee, Hyung-Jun Choi, Jaeho Lee, Je Seon Song, Chung-Min Kang
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Elinor Halperson, Vered Matalon, Gal Goldstein, Shirly Saieg Spilberg, Karin Herzog, Avia Fux-Noy, Aviv Shmueli, Diana Ram, Moti Moskovitz
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Anna Jodłowska, Lidia Postek-Stefańska
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Ana Zulijani, Martina Žigante, Luka Morelato, Berislav Perić, Ana Milardović
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Kristine Eidal Tanem, Einar Stensvold, Petter Wilberg, Anne B. Skaare, Petter Brandal, Bente Brokstad Herlofson
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Jaehyun Kim, Hee Jin Lim, Ja Hyeong Ku, Yoon-Ah Kook, Nack-Gyun Chung, Yoonji Kim, Nazmul Haque
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Patrycja Proc, Joanna Szczepańska, Małgorzata Zubowska, Beata Zalewska-Szewczyk, Wojciech Młynarski
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Dental late effects of antineoplastic treatment on childhood cancer survivors: Radiographic findings
Kyriaki Seremidi, Katerina Kavvadia, Antonis Kattamis, Argyro Polychronopoulou
International Journal of Paediatric Dentistry.2021; 31(6): 742. CrossRef
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Egle Immonen, Atte Nikkilä, Timo Peltomäki, Liisa Aine, Olli Lohi
Pediatric Blood & Cancer.2021;[Epub] CrossRef
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Juliette Stolze, Kim C. E. Vlaanderen, Frederique C. E. D. Holtbach, Jop C. Teepen, Leontien C. M. Kremer, Jacqueline J. Loonen, Eline van Dulmen-den Broeder, Marry M. van den Heuvel-Eibrink, Helena J. H. van der Pal, Birgitta Versluys, Margriet van der H
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Ryo Kameoka, Tomomi Kawakami, Miho Maeda, Tsukasa Hori, Ayako Yanagisawa, Toshiomi Shirase
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Priyanshi Ritwik, Tammuella E. Chrisentery-Singleton
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Kyriaki Seremidi, Dimitrios Kloukos, Argy Polychronopoulou, Antonis Kattamis, Katerina Kavvadia
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Outcomes of Treatment for Malignant Peripheral Nerve Sheath Tumors: Different Clinical Features Associated with Neurofibromatosis Type 1: In Kyung Hwang, Seung Min Hahn, Hyo Sun Kim, Sang Kyum Kim, Hyo Song Kim, Kyoo‑Ho Shin, Chang Ok Suh, Chuhl Joo Lyu, Jung Woo Han; Cancer Res Treat. 2017;49(3):717-726. Published online December 1, 2016; DOI: https://doi.org/10.4143/crt.2016.271

Abstract PDF Supplementary Material PubReader ePub

Purpose
Malignant peripheral nerve sheath tumors (MPNSTs) are a rare subtype of sarcoma that occur spontaneously or in association with neurofibromatosis type 1 (NF-1). This study aimed to clinically differentiate these types of MPNSTs.
Materials and Methods
The study reviewed 95 patients diagnosed with and treated for MPNST at Yonsei University Health System, Seoul, Korea over a 27-year period. The clinical characteristics, prognostic factors, and treatment outcomes of sporadic MPNST (sMPNST) and NF-1 associated MPNST (NF-MPNST) cases were compared.
Results
Patients with NF-MPNST had a significantly lower median age (32 years vs. 45 years for sMPNST, p=0.012), significantly larger median tumor size (8.2 cm vs. 5.0 cm for sMPNST, p < 0.001), and significantly larger numbers of imaging studies and surgeries (p=0.004 and p < 0.001, respectively). The 10-year overall survival (OS) rate of the patients with MPNST was 52±6%. Among the patients with localized MPNST, patients with NF-MPNST had a significantly lower 10-year OS rate (45±11% vs. 60±8% for sMPNST, p=0.046). Univariate analysis revealed the resection margin, pathology grade, and metastasis to be significant factors affecting the OS (p=0.001, p=0.020, and p < 0.001, respectively). Multivariate analysis of the patients with localized MPNST identified R2 resection and G1 as significant prognostic factors for OS.
Conclusion
NF-MPNST has different clinical features from sMPNST and requires more careful management. Further study will be needed to develop specific management plans for NF-MPNST.

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