Cancer Research and Treatment

Original Articles

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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Clinical Characteristics and Treatment Outcomes of Childhood Acute Promyelocytic Leukemia in Korea: A Nationwide Multicenter Retrospective Study by Korean Pediatric Oncology Study Group: Kyung Mi Park, Keon Hee Yoo, Seong Koo Kim, Jae Wook Lee, Nack-Gyun Chung, Hee Young Ju, Hong Hoe Koo, Chuhl Joo Lyu, Seung Min Han, Jung Woo Han, Jung Yoon Choi, Kyung Taek Hong, Hyoung Jin Kang, Hee Young Shin, Ho Joon Im, Kyung-Nam Koh, Hyery Kim, Hoon Kook, Hee Jo Baek, Bo Ram Kim, Eu Jeen Yang, Jae Young Lim, Eun Sil Park, Eun Jin Choi, Sang Kyu Park, Jae Min Lee, Ye Jee Shim, Ji Yoon Kim, Ji Kyoung Park, Seom Gim Kong, Young Bae Choi, Bin Cho, Young Tak Lim; Cancer Res Treat. 2022;54(1):269-276. Published online April 20, 2021; DOI: https://doi.org/10.4143/crt.2021.313

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Purpose
Acute promyelocytic leukemia (APL) is a rare disease in children and there are some different characteristics between children and adult. We aimed to evaluate incidence, clinical characteristics and treatment outcomes of pediatric APL in Korea.
Materials and Methods
Seventy-nine pediatric APL patients diagnosed from January 2009 to December 2016 in 16 tertiary medical centers in Korea were reviewed retrospectively.
Results
Of 801 acute myeloid leukemia children, 79 (9.9%) were diagnosed with APL. The median age at diagnosis was 10.6 years (range, 1.3 to 18.0). Male and female ratio was 1:0.93. Thirty patients (38.0%) had white blood cell (WBC) count greater than 10×10⁹/L at diagnosis. All patients received induction therapy consisting of all-trans retinoic acid and chemotherapy. Five patients (6.6%) died during induction chemotherapy and 66 patients (86.8%) achieved complete remission (CR) after induction chemotherapy. The causes of death were three intracranial hemorrhage, one cerebral infarction, and one sepsis. Five patients (7.1%) suffered a relapse during or after maintenance chemotherapy. The estimated 4-year event-free survival and overall survival (OS) rates were 82.1%±4.4%, 89.7%±5.1%, respectively. The 4-year OS was significantly higher in patients with initial WBC < 10×10⁹/L than in those with initial WBC ≥ 10×10⁹/L (p=0.020).
Conclusion
This study showed that the CR rates and survival outcomes in Korean pediatric APL patients were relatively good. The initial WBC count was the most important prognostic factor and most causes of death were related to serious bleeding in the early stage of treatment.

Citations

Citations to this article as recorded by

Management of Acute Promyelocytic Leukemia at Extremes of Age
Sabine Kayser, Shannon E. Conneely
Cancers.2023; 15(14): 3637. CrossRef
Current Challenges of Asian National Children's Cancer Study Groups on Behalf of Asian Pediatric Hematology and Oncology Group
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
Childhood acute promyelocytic leukemia in a pediatric cancer referral center in Baghdad, Iraq. Improved results with ATRA extended consolidation
Anna Maria Testi, Mazin Faisal Al-Jadiry, Hasanein Habeeb Ghali, Samaher Abdulrazzaq Fadhil, Amir Fadhil Al-Darraji, Raghad Majid Al-Saeed, Ahmed Hatem Sabhan, Safaa A. Faraj Al-Badri, Wisan Majeed Abed, Najiha Ahmed Ameen, Ruaa Zaki Al-Tameemi, Arabiya I
Leukemia & Lymphoma.2022; 63(12): 2940. CrossRef
Death due to unsuspected acute myeloid leukaemia: an unusual forensic diagnosis
Lila Krebs-Drouot, Georgia Karpathiou, Virginie Scolan, Carolyne Bidat-Callet, Baptiste Boyer, Michel Péoc’h
Forensic Science, Medicine and Pathology.2022; 19(1): 60. CrossRef
Successful Treatment of Isolated Central Nervous System Relapse with Intrathecal Chemotherapy in an Adolescent with Acute Promyelocytic Leukemia
Haerim Song, Eun Sang Yi
Clinical Pediatric Hematology-Oncology.2022; 29(2): 70. CrossRef

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Clinical Outcome of Relapsed or Refractory Burkitt Lymphoma and Mature B-Cell Lymphoblastic Leukemia in Children and Adolescents: Hyery Kim, Eun Sil Park, Soo Hyun Lee, Hong Hoe Koo, Hyo Sun Kim, Chuhl Joo Lyu, So Eun Jun, Young Tak Lim, Hee Jo Baek, Hoon Kook, Ji Won Lee, Hyoung Jin Kang, Kyung Duk Park, Hee Young Shin, Hyeo Seop Ahn; Cancer Res Treat. 2014;46(4):358-365. Published online July 21, 2014; DOI: https://doi.org/10.4143/crt.2013.047

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Purpose
Despite the rapid improvement in survival rate from Burkitt lymphoma and mature B-cell lymphoblastic leukemia (B-ALL) in children, a small subset of patients do not respond to first-line chemotherapy or experience relapse (RL). Herein, we report the clinical characteristics and outcomes of these patients.
Materials and Methods
RL or refractory Burkitt lymphoma and mature B-ALL in 125 patients diagnosed from 1990 to 2009 were retrospectively analyzed.
Results
Nineteen patients experienced RL or progressive disease (PD). Among them, 12 patients had PD or RL less than six months after initial treatment and seven had late RL. Seven patients achieved complete response (CR), 11 had PD, and one had no more therapy. Six patients who achieved CR survived without evidence of disease and four of them underwent high-dose chemotherapy (HDC) followed by stem cell transplantation (SCT). However, 11 patients who failed to obtain CR eventually died of their disease. Five-year overall survival (OS) was 31.6±10.7%. OS of patients with late RL was superior to that of patients with early RL (57.1±18.7%, vs. 16.7±10.8%, p=0.014). Achievement of CR after reinduction had significant OS (p < 0.001). OS for patients who were transplanted was superior (p < 0.01). In multivariate analysis, achievement of CR after reinduction chemotherapy showed an association with improved OS (p=0.05).
Conclusion
Late RL and chemotherapy-sensitive patients have the chance to achieve continuous CR using HDC/SCT, whereas patients who are refractory to retrieval therapy have poor prognosis. Therefore, novel salvage strategy is required for improvement of survival for this small set of patients.

Citations

Citations to this article as recorded by

The experience of relapsed Burkitt lymphoma treatment with targeted drugs and autologous/allogeneic stem cell transplantation
T. T. Valiev, A. A. Khachatryan, S. V. Goryacheva, N. A. Batmanova, K. I. Kirgizov, S. R. Varfolomeeva
Oncohematology.2024; 19(1): 40. CrossRef
Refractory Burkitt Lymphoma: Diagnosis and Interventional Strategies
Francesco Malfona, Anna Maria Testi, Sabina Chiaretti, Maria Luisa Moleti
Blood and Lymphatic Cancer: Targets and Therapy.2024; Volume 14: 1. CrossRef
MYC upstream region orchestrates resistance to PI3K inhibitors in cancer cells through FOXO3a-mediated autophagic adaptation
Rosa Bordone, Devon Michael Ivy, Rodrigo D’Amico, Martina Barba, Miriam Gaggianesi, Fiorella Di Pastena, Bianca Cesaro, Francesca Bufalieri, Alessio Balzerano, Enrico De Smaele, Giuseppe Giannini, Lucia Di Marcotullio, Alessandro Fatica, Giorgio Stassi, L
Oncogene.2024; 43(46): 3349. CrossRef
Prognostic Factors and Treatment Outcome of Relapsing and Refractory Pediatric Mature B-cell Non-Hodgkin Lymphoma, Children’s Cancer Hospital Egypt Experience
Samah F. Semary, Hany Abdel Rahman, Naglaa Elkinaai, Iman Zaky, Nouran Nagy, Sherine Salem, Asmaa Hamoda
Journal of Pediatric Hematology/Oncology.2023; 45(6): e757. CrossRef
Chronic Recurrent Multifocal Osteomyelitis Associated With Inflammatory Bowel Disease Successfully Treated With Infliximab
Shinhyeung Kwak, Dongsub Kim, Joon-sik Choi, Yoonsun Yoon, Eun Sil Kim, Mi Jin Kim, So-Young Yoo, Jong Sup Shim, Yon Ho Choe, Yae-Jean Kim
Pediatric Infection & Vaccine.2022; 29(2): 96. CrossRef
Burkitt lymphoma
Cristina López, Birgit Burkhardt, John K. C. Chan, Lorenzo Leoncini, Sam M. Mbulaiteye, Martin D. Ogwang, Jackson Orem, Rosemary Rochford, Mark Roschewski, Reiner Siebert
Nature Reviews Disease Primers.2022;[Epub] CrossRef
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Xueqian Li, Meng Zhou, Jiaqian Qi, Yue Han
Clinical Lymphoma Myeloma and Leukemia.2021; 21(3): e227. CrossRef
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Cancers.2021; 13(9): 2075. CrossRef
Long non-coding RNAs as the critical regulators of doxorubicin resistance in tumor cells
Ghazaleh Khalili-Tanha, Meysam Moghbeli
Cellular & Molecular Biology Letters.2021;[Epub] CrossRef
EBV BCL-2 homologue BHRF1 drives chemoresistance and lymphomagenesis by inhibiting multiple cellular pro-apoptotic proteins
Leah Fitzsimmons, Rachel Cartlidge, Catherine Chang, Nenad Sejic, Laura C. A. Galbraith, Chathura D. Suraweera, Deborah Croom-Carter, Grant Dewson, Rosemary J. Tierney, Andrew I. Bell, Clare Shannon-Lowe, Marco J. Herold, Alan B. Rickinson, Peter M. Colma
Cell Death & Differentiation.2020; 27(5): 1554. CrossRef
Treatment of relapsed/refractory paediatric aggressive B‐cell non‐Hodgkin lymphoma
Maria L. Moleti, Anna M. Testi, Robin Foà
British Journal of Haematology.2020; 189(5): 826. CrossRef
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Jin Kyung Suh, Yi-Jin Gao, Jing-Yan Tang, Shiann-Tarng Jou, Dong-Tsamn Lin, Yoshiyuki Takahashi, Seiji Kojima, Ling Jin, Yonghong Zhang, Jong Jin Seo
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Rituximab is highly effective in children and adolescents with Burkitt lymphoma in Risk Groups R2 to R4
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Knockdown of lncRNA MCM3AP-AS1 Attenuates Chemoresistance of Burkitt Lymphoma to Doxorubicin Treatment via Targeting the miR-15a/EIF4E Axis

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Outcomes of adults with relapsed or refractory Burkitt and high‐grade B‐cell leukemia/lymphoma
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Case Reports

Staphylococcal Endocarditis Presenting with a Renal Infarct in a Patient with Acute Lymphoblastic Leukemia: Meong Hi Son, Eun Sil Park, Ji-Hyun Seo, Jae-Young Lim, Chan-Hoo Park, Hyang-Ok Woo, Hee-Shang Youn; Cancer Res Treat. 2008;40(3):151-154. Published online September 30, 2008; DOI: https://doi.org/10.4143/crt.2008.40.3.151

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We present here a patient with acute lymphoblastic leukemia (ALL) and who developed infective endocarditis during induction chemotherapy with prednisolone, L-asparaginase (Leunase®), vincristine and adriamycin. The patient did not have a history of a central venous catheter. Sharp flank pain and fever occurred on the 25^th day of induction chemotherapy. In addition, a renal infarct and movable vegetations on the mitral valve were detected on the abdominal computed tomography (CT) and echocardiography. S. aureus was identified in the cultured blood. While the patient achieved remission, follow-up echocardiography revealed the vegetation had increased in size and an abscess pocket had developed despite the antibiotics and heparin therapy. Consequently, ten days after the diagnosis of infective endocarditis, a successful mitral valvuloplasty was performed without complications. The patient is currently on maintenance chemotherapy while in remission.

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Acute Lymphoblastic Leukemia with Infective Endocarditis Presented with Unusual Intracardiac Mass
Ali Sadeghpour Tabaei, Leili Koochakzadeh, Mohammadrafie Khorgami, Sepehr Sadeghpour Tabaei
Case Reports in Cardiology.2017; 2017: 1. CrossRef

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Carboxypeptidase-G2 Rescue in a Patient with High Dose Methotrexate-induced Nephrotoxicity: Eun Sil Park, Kyung Hee Han, Hyoung Soo Choi, Hee Young Shin, Hyo Seop Ahn; Cancer Res Treat. 2005;37(2):133-135. Published online April 30, 2005; DOI: https://doi.org/10.4143/crt.2005.37.2.133

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A 13 year-old girl with osteosarcoma and pulmonary tumor recurrence developed acute renal failure following high dose methotrexate (12 g/m²) therapy, she had previously tolerated high dose methotrexate and her renal and hepatic functions were normal. Briefly, 48 hours after beginning methotrexate infusion her methotrexate concentration and creatinine level were 1338.8 µM/L and 5.8 mg/dl, respectively. Grade IV oral mucositis and neutropenia with fever developed at 144 hours after MTX infusion. Hydration and alkalinization were continued and leucovorin rescue was intensified based on the plasma MTX concentrations. Plasma exchange was performed twice and hemodialysis 3 times without problems, but methotraxate and creatinine levels remained high, 91.9 µM/L, and 2.5 mg/dl, respectively. After 3 courses of hemodialysis carboxypeptidase-G2 (CPDG2) was administered at 50 U/kg, intravenously over 5 minutes. After 15 minutes of CPDG2 (Voraxaze™) infusion, her plasma MTX concentration was 0.91 µM/L and no rebound elevation or side effects developed. Thirteen days post-MTX infusion her renal function had normalized. We report here our experience of a dramatic methotrexate level reduction caused by CPDG2 administration.

Citations

Citations to this article as recorded by

Extracorporeal Treatment for Methotrexate Poisoning
Marc Ghannoum, Darren M. Roberts, David S. Goldfarb, Jesper Heldrup, Kurt Anseeuw, Tais F. Galvao, Thomas D. Nolin, Robert S. Hoffman, Valery Lavergne, Paul Meyers, Sophie Gosselin, Tudor Botnaru, Karine Mardini, David M. Wood
Clinical Journal of the American Society of Nephrology.2022; 17(4): 602. CrossRef
Comparable efficacy with varying dosages of glucarpidase in pediatric oncology patients
Jeffrey R. Scott, Yinmei Zhou, Cheng Cheng, Deborah A. Ward, Hope D. Swanson, Alejandro R. Molinelli, Clinton F. Stewart, Fariba Navid, Sima Jeha, Mary V. Relling, Kristine R. Crews
Pediatric Blood & Cancer.2015; 62(9): 1518. CrossRef
Resumption of high‐dose methotrexate after acute kidney injury and glucarpidase use in pediatric oncology patients
Anthony M. Christensen, Jennifer L. Pauley, Alejandro R. Molinelli, John C. Panetta, Deborah A. Ward, Clinton F. Stewart, James M. Hoffman, Scott C. Howard, Ching‐Hon Pui, Alberto S. Pappo, Mary V. Relling, Kristine R. Crews
Cancer.2012; 118(17): 4321. CrossRef

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