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

Abstract PDF Supplementary Material PubReader ePub: 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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Tandem High-Dose Chemotherapy Increases the Risk of Secondary Malignant Neoplasm in Pediatric Solid Tumors: Hana Lim, Minji Im, Eun Seop Seo, Hee Won Cho, Hee Young Ju, Keon Hee Yoo, Sung Yoon Cho, Jong-Won Kim, Do Hoon Lim, Ki Woong Sung, Ji Won Lee; Cancer Res Treat. 2024;56(2):642-651. Published online November 24, 2023; DOI: https://doi.org/10.4143/crt.2023.999

Abstract PDF Supplementary Material PubReader ePub

Purpose
This study aimed to investigate the incidence and risk factors for secondary malignant neoplasms (SMN) in pediatric solid tumors, focusing on the effects of tandem high-dose chemotherapy (HDCT).
Materials and Methods
Patients (aged < 19 years) diagnosed with or treated for pediatric solid tumors between 1994 and 2014 were retrospectively analyzed. The cumulative incidence of SMN was estimated using competing risk methods by considering death as a competing risk.
Results
A total of 1,435 patients (413 with brain tumors and 1,022 with extracranial solid tumors) were enrolled. Seventy-one patients developed 74 SMNs, with a 10-year and 20-year cumulative incidence of 2.680±0.002% and 10.193±0.024%, respectively. The types of SMN included carcinoma in 28 (37.8%), sarcoma in 24 (32.4%), and hematologic malignancy in 15 (20.3%) cases. Osteosarcoma and thyroid carcinoma were the most frequently diagnosed tumors. Multivariate analysis showed that radiotherapy (RT) > 2, 340 cGy, and tandem HDCT were significant risk factors for SMN development. The SMN types varied according to the primary tumor type; carcinoma was the most frequent SMN in brain tumors and neuroblastoma, whereas hematologic malignancy and sarcomas developed more frequently in patients with sarcoma and retinoblastoma, respectively.
Conclusion
The cumulative incidence of SMN in pediatric patients with solid tumors was considerably high, especially in patients who underwent tandem HDCT or in those who received RT > 2,340 cGy. Therefore, the treatment intensity should be optimized based on individual risk assessment and the long-term follow-up of pediatric cancer survivors.

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Rising Prevalence of Low-Frequency PPM1D Gene Mutations after Second HDCT in Multiple Myeloma
Katja Seipel, Nuria Z. Veglio, Henning Nilius, Barbara Jeker, Ulrike Bacher, Thomas Pabst
Current Issues in Molecular Biology.2024; 46(8): 8197. 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.

Citations

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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
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
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
Therapeutic Targeting of EZH2 and BET BRD4 in Pediatric Rhabdoid Tumors
Yukitomo Ishi, Yongzhan Zhang, Ali Zhang, Takahiro Sasaki, Andrea Piunti, Amreena Suri, Jun Watanabe, Kouki Abe, Xingyao He, Hiroaki Katagi, Pankaj Bhalla, Manabu Natsumeda, Lihua Zou, Ali Shilatifard, Rintaro Hashizume
Molecular Cancer Therapeutics.2022; 21(5): 715. CrossRef
Molecular targeted therapies for pediatric atypical teratoid/rhabdoid tumors
Chang Zhang, Hao Li
Pediatric Investigation.2022; 6(2): 111. CrossRef
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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Case Report

Mixed Testicular Germ Cell Tumor Presenting as Metastatic Pure Choriocarcinoma Involving Multiple Lung Metastases That Was Effectively Treated with High-dose Chemotherapy: Sang-Cheol Lee, Kyoung Ha Kim, Sung Han Kim, Nam Su Lee, Hee Sook Park, Jong-Ho Won; Cancer Res Treat. 2009;41(4):229-232. Published online December 31, 2009; DOI: https://doi.org/10.4143/crt.2009.41.4.229

Abstract PDF PubReader ePub

Choriocarcinoma in the testis is very rare, and it represents less than 1% (0.3%) of all the testicular germ cell tumors. It is a particularly aggressive variant of non-seminoma tumor, which is characterized by a high serum β-HCG level and multiple lung metastases. The optimal management for this disease remains undefined. We report here on a case of choriocarcinoma with multiple lung metastases, and the patient has achieved continuous remission for 2 years after combination chemotherapy of BEP (bleomycin, etoposide and cisplatin) and sequential high-dose chemotherapy with autologous peripheral stem cell rescue.

Citations

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Testicular Mixed Germ Cell Tumor Presenting with Upper Gastrointestinal Bleeding: A Case Report
Emilija Nikolovska Trpchevska, Beti Todorovska, Magdalena Bogdanovska Todorovska, Meri Trajkovska, Dafina Nikolova, Darko Dzambaz, Gjorgji Deriban, Fana Licoska-Josifovikj
PRILOZI.2023; 44(2): 47. CrossRef
Gastrointestinal Bleeding Secondary to Metastatic Duodenal Choriocarcinoma in a Patient with Concomitant Peptic Ulcer Disease
Ahmed Elfiky, Asmaa Mokhtar, Mira Alsheikh, Hassan Almoussawi, Stephen Mulrooney, Haruhiko Sugimura
Case Reports in Gastrointestinal Medicine.2021; 2021: 1. CrossRef
Gastrointestinal Bleeding with Hemodynamic Repercussion due to a Gastric Metastatic Lesion of a Testicular Choriocarcinoma in a Previously Asymptomatic Young Adult
Omar Al Salman, Fahad Malik, Shashank Trivedi, Marwah Alchalabi, Shobhana Chaudhari
GE - Portuguese Journal of Gastroenterology.2020; 27(3): 212. CrossRef
Choriocarcinomas May Be Presented with Abnormal Manifestations
Alpaslan Tanoglu, Tolga Duzenli
International Journal of Cancer Management.2017;[Epub] CrossRef
Metastatic Testicular Choriocarcinoma: A Rare Cause of Upper GI Bleeding
Kirsty Lowe, Jacqueline Paterson, Sharon Armstrong, Shaun Walsh, Max Groome, Craig Mowat
ACG Case Reports Journal.2016; 3(1): 36. CrossRef
Metastatic choriocarcinoma induced separate simultaneous intracerebral haemorrhages: a very rare occurrence and its novel association with Klinefelter syndrome
Maximilian Olavi Joret, Robert M Starke, John Scotter, Peter Heppner
BMJ Case Reports.2015; : bcr2015212777. CrossRef
Contrast-Enhanced Ultrasonography of the Testes
Ounali S. Jaffer, Paul S. Sidhu
Ultrasound Clinics.2013; 8(4): 509. CrossRef
Multiparametric ultrasonography of the testicles
Tobias De Zordo, Daniel Stronegger, Leo Pallwein-Prettner, Chris J. Harvey, Germar Pinggera, Werner Jaschke, Friedrich Aigner, Ferdinand Frauscher
Nature Reviews Urology.2013; 10(3): 135. CrossRef
A Case of Gastrointestinal Bleeding due to Duodenal Metastasis from a Testicular Choriocarcinoma
Magdalene Vardaros, Miral Subhani, Kaleem Rizvon, Vladimir Gotlieb, Paul Mustacchia, Lester Freedman, Vikas Garg, Jaspreet Singh, Ghulam Siddiqui
Journal of Gastrointestinal Cancer.2013; 44(2): 234. CrossRef
Unravelling mechanisms of cisplatin sensitivity and resistance in testicular cancer
R. Koster, M.A.T.M. van Vugt, H. Timmer-Bosscha, J.A. Gietema, S. de Jong
Expert Reviews in Molecular Medicine.2013;[Epub] CrossRef

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Original Articles

High-Dose Chemotherapy of Cyclophosphamide, Thiotepa, and Carboplatin (CTCb) Followed by Autologous Stem-Cell Transplantation for Metastatic Breast Cancer Patients: A 6-Year Follow-Up Result: Hee-Jung Sohn, Sang-Hee Kim, Gyeong-Won Lee, Shin Kim, Hye Jin Kang, Jin-Hee Ahn, Sung-Bae Kim, Sang-We Kim, Woo Kun Kim, Cheolwon Suh; Cancer Res Treat. 2005;37(1):24-30. Published online February 28, 2005; DOI: https://doi.org/10.4143/crt.2005.37.1.24

Abstract PDF PubReader ePub

Purpose

The benefit of high-dose chemotherapy (HDC) for metastatic breast cancer (MBC) is controversial. We evaluated the efficacy and safety of HDC with cyclophosphamide, thiotepa, and carboplatin (CTCb) followed by autologous stem-cell transplantation (ASCT) for MBC patients.

Materials and Methods

From September 1994 to December 1999, 23 MBC patients were enrolled. All the patients received 2 to 10 cycles of induction chemotherapy. Before transplantation, 12 patients were in complete response (CR), nine were in partial response (PR), and two had progressive disease (PD). The HDC regimen consisted of cyclophosphamide 1,500 mg/m²/day, thiotepa 125 mg/m²/day and carboplatin 200 mg/m²/day intravenously for 4 consecutive days.

Results

After ASCT, 13 patients (56%) had a CR, five (22%) had a PR, three (13%) had no change, while two (9%) showed a PD. Seventeen patients relapsed or progressed during the median follow-up of 78 months. The median progression-free survival (PFS) time was 11 months and the median overall survival (OS) time was 23 months. The 5-year PFS and OS rates were 22% and 25%, respectively. On the multivariate analyses, less than 4 involved lymph nodes was predictive of a better PFS and OS.

Conclusion

HDC with CTCb for MBC has acceptable toxicity; however, this treatment does not show a survival benefit.

Citations

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Real-world Experience of Improvement in the Survival of Lymphoma and Myeloma Patients with Autologous Stem Cell Transplantation over a 25-year Period
Hyungwoo Cho, Shin Kim, Kyoungmin Lee, Jung Sun Park, Cheolwon Suh
The Korean Journal of Medicine.2021; 96(6): 501. CrossRef

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Conventional Treatments in Patients with Hodgkin's Disease: Jong Beom Park, Chul Won Seo, Sang Hee Kim, Kyung No Lee, Hun Ho Song, Soon Seo Park, Hyo Jung Kim, Yung Joo Min, Jin Hee Park, Sung Joon Choe, Jung Koon Kim, Tae Won Kim, Dae Yung Jang, Je Hwan Lee, Sung Bae Kim, Sang Wee Kim, Koo Hyung Lee, Jung Sin Lee, Woo Keon Kim; J Korean Cancer Assoc. 1999;31(4):821-829.

Abstract PDF: PURPOSE
We conducted this study to determine the efficacy of conventional treatments for patients with Hodgkin's disease and identify the patients who have poor prognosis and need high-dose chemotherapy and autologous stem cell transplantation.
MATERIALS AND METHODS
Between Jun. 1989 and Dec. 1997, 50 patients were enrolled and 39 patients were evaluable. Patients were treated with radiotherapy (5 patients) or combination chemotherapy (21 patients) or combined chemotherapy and radiotherapy (13 patients) according to their disease stage. Chemotherapy regimens were C-MOPP (cyclo- phosphamide, vincristine, procarbazine, and prednisone), MOPP (mechlorethamine, vin- cristine, procarbazine, and prednisone), ABVD (doxorubicin, bleomycin, vinblastine, and dacarbazine), alternating C-MOPP/ABVD, and MOPP/ABV hybrid. Radiation therapy was performed when there was residual tumor after chemotherapy or bulky disease. The response to treatments was analyzed by clinical stage I-II and stage III-IV patients group, respectively.
RESULTS
The complete response rate was 76.9% for total patients, 83.3% for stage I-II patients, and 71.4% for stage III-IV patients. Of the 30 patients achieving complete response, four (13.3%) relapsed at 6, 12, 22, and 28 months after complete response, respectively. The median follow-up duration was 24 months. Nine patients died. Four patients died of Hodgkins disease. Three-year overall survival rate was 72.9% for total patients, 72.5% for stage I-II patients, and 70% for stage III-IV patients. Two-year disease- free survival rate was 77.6% for total patients, 79% for stage I-II stage patients, and 73.9% for stage III-IV patients. The prognostic factor analysis showed that performance status affected the disease-free survival rate.
CONCLUSION
Conventional treatments in patients with Hodgkins disease showed results comparable to previous studies. But we were unable to identify the patients, who need high-dose chemotherapy and autologous stem cell transplantation, because of small number of study patients and short follow up duration.

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High-Dose Chemotherapy with Vandervilt Regimen and CSF Support for High-Risk Aggressive Non-Hodgkin's Lymphoma: Bong Seog Kim, Jeong Hoon Yang, Kyung Tae Kim, Baek Yeol Ryoo, Tae You Kim, Young Hyuck Im, Jhin Oh Lee, Tae Woong Kang, Yoon Koo Kang; J Korean Cancer Assoc. 1998;30(1):137-149.

Abstract PDF: PURPOSE
To detennine the therapeutic effect and toxicities of high-dose chemotherapy with Vanderbilt regimen and colany-stimulating factors(CSF) support for high-risk aggressive non-Hodgkin's lymphoma(NHL).
MATERIALS AND METHODS
Between Aug. 1995 and Mar. 1997, 40 patients with high-risk aggressive NHLs were treated with high-dose chemotherapy with Vandebilt regimen and CSF support. If the complete response(CR) was induced, four cycles of CHOP were administered for the maintenance of response. In cases of lymphoblastic lymphomas, CNS prophyiaxis with cranial irradiation and intrathecal methotrexate was done after CR.
RESULTS
CR was achieved after Vanderbilt regimen in 62.5%(25/40) of the total patients. CR rste in refractory group(12.5%: 1/8) was significantly lower than in other groups (75%: 24/32)(p=0.001). With a median follow-up of 14 months, the failure free survival (FFS) was 0~18+ months(median 6.1 months). The overall FFS rate at one year was 31.7%. The 1-year FFS rate in refractory group(0%) was significantly lower than in other patients groups(41%)(p=0.001). The range of survival time was 0.5~18+ months, and median survival time was 6.2 months. Grade 4 leukopenia was observed in 100% of chemotherapy cycles and its median duration was 7 days. However, only one patient died due to treatment-relate sepsis. Non-hematological toxicities were tolerable and all reversible.
CONCLUSION
High-dose chemotherapy with Vanderbilt regimen was effcctive for induction of CR in high-risk aggressive NHL patients and safe with the CSF support. However, poor CR rate in reftactory group and poor FFS in other groups indicate that a new, more intensive approach is needed for the induction of CR in refractory group and for the maintenance of CR in other high-risk patient groups.

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A Case of Primary Splenic Angiosarcoma Associated with Kasabach-Merritt Syndrome: Jin Seok Jeon, Gyu Taek Lee, Ki Ju Han, Jae Ho Byun, Seung Kyu Park, Jong Ho Won, Seung Ho Baick, Dae Sik Hong, Hee Sook Park, Hae Kyung Lee, So Young Jin; J Korean Cancer Assoc. 1997;29(2):352-357.

Abstract PDF: PURPOSE
Primary malignant vascular neoplasms of the spleen are rare. It has been known that the prognosis was very poor and the splenectomy before rupture could increase survival. No effective chemotherapeutic protocol for angiosarcomas has yet to be established but patients with or without metastatic disease may be treated by chemotherapy. MATERIAL AND METHODS: We experienced a case of primary splenic angiosarcoma in a 42-year-old woman with multiple purpuric skin rashes associated with consumptive coagulopathy:the Kasabach-Merritt syndrome. The CT showed spleen is diffusely enlarged and inhomogenously enhanced with multiple metastasis in the liver. The splenectomy was done and angiosarcoma was diagnosed. We treated her with conventional combination chemotherapy and obtained partial response. For additional response, high-dose chemotherapy and stem cell rescue with autologous peripheral blood stem cell transplantation was done.
RESULT
Afer splenectomy, platelet count return to normal. The follow up abdominal CT scan after treatment showed complete disappeared multiple metastatic lesions in the both lobe of liver and the patient has continued to do well four months following discharge.
CONCLUSION
We herein report our experience of a splenic angiosarcoma whose multiple hepatic metastases responded well to the high-dose chemotherapy.

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High - dose Chemotherapy with Hematopoietic Stem Cell Support for Malignant Lymphoma: Young Suk Park, Jung Ae Lee, Woo Sung Min, Seonyang Park, Jing Shin Lee, Hugh Chul Kim, Hoon Kook, Jong Ho Won, Dae Sik Hong, Hee Sook Park; J Korean Cancer Assoc. 1996;28(2):316-326.

Abstract PDF: Patients with intermediate or high-grade non-Hodgkin's lymphoma(NHL) have been reported a 40% to 70% cure rate when treated with conventional chemotherapy or radiotherapy. However, most of the patients who do not attain complete remission(CR) or who relapae after chemotherapy are incurable using conventional salvage therapies and these individuals have potential for cure with high-dose therapy with reinfusion of stem cells derived from bone marrow or peripheral blood. Between February 1993 and September 1995, 26 patients with aggressive, relapsed and/or refractory malignant lymphoma were treated with high-dose chemotherapy with either autologous peripheral blood stem cell(25 patients) or bone marrow(l patient) support in 7 university hospitals in Korea. The median age was 39 years(range, l7 to 69) and male to female ratio was 4.2: 1. The common histologic types were diffuse large cell(42%), immunoblastic type(15%) of non-Hodgkin's lymphoma. The rate of complete remission was 61%(14/23) and overall remission rate was 78%(18/ 23) for the patients with measurable disease. Three patients were treated as the consolidation therapy in the status of complete remission. The median duration of remission was not reached right now. The median survival time was 7.8 months for all patients(the median follow up time; 9 months). The median time to recovery to a neutrophil count more than 0.5 x 10(9)/L was 13 days(range, 8 to 43), and to 1 x 10(9)/L was 22 days(range, 8 to 53). The median time to recovery of platelet count more than 50 x l0(9)/L and 100 x 10(9)/L were 20 days(range, 8 to 45) and 21 days(ranae, ll to 60). Non-hematologic side effects were fever, nausea and vomiting, and liver toxicity. Two toxic deaths occurred due to cardiovascular disease, mainly congestive heart failure. Based on high complete remission rate and tolerable toxicity, high-dose chemotherapy with hematopietic stem cell support appears to be a promising treatment modality for the patients with aggressive, relapsed and/or refractory malignant lymphoma.

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