Cancer Research and Treatment

Original Articles

Genitourinary cancer

Clear Cell Adenocarcinoma of Urethra: Clinical and Pathologic Implications and Characterization of Molecular Aberrations: Boram Song, Seok Hyun Lee, Jeong Hwan Park, Kyung Chul Moon; Cancer Res Treat. 2024;56(1):280-293. Published online September 11, 2023; DOI: https://doi.org/10.4143/crt.2023.577

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Purpose
This study aimed to evaluate the molecular features of clear cell adenocarcinoma (CCA) of the urinary tract and investigate its pathogenic pathways and possible actionable targets.
Materials and Methods
We retrospectively collected the data of patients with CCA between January 1999 and December 2016; the data were independently reviewed by two pathologists. We selected five cases of urinary CCA, based on the clinicopathological features. We analyzed these five cases by whole exome sequencing (WES) and subsequent bioinformatics analyses to determine the mutational spectrum and possible pathogenic pathways.
Results
All patients were female with a median age of 62 years. All tumors were located in the urethra and showed aggressive behavior with disease progression. WES revealed several genetic alterations, including driver gene mutations (AMER1, ARID1A, CHD4, KMT2D, KRAS, PBRM1, and PIK3R1) and mutations in other important genes with tumor-suppressive and oncogenic roles (CSMD3, KEAP1, SMARCA4, and CACNA1D). We suggest putative pathogenic pathways (chromatin remodeling pathway, mitogen-activated protein kinase signaling pathway, phosphoinositide 3-kinase/AKT/mammalian target of rapamycin pathway, and Wnt/β-catenin pathway) as candidates for targeted therapies.
Conclusion
Our findings shed light on the molecular background of this extremely rare tumor with poor prognosis and can help improve treatment options.

Citations

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Urethral clear cell adenocarcinoma in an adult female: A rare case report
Yacob Sheiferawe Seman, Michael Teklehaimanot Abera, Fadil Nuredin Abrar, Tesfaye Kebede Legesse, Mesfin Asefa Tola, Tsiyon Nigusie Alemu
Urology Case Reports.2025; 58: 102882. CrossRef
Two rare cases of primary clear cell adenocarcinoma of the urethra: clinical experience, case report and literature review
Bohao Jiang, Jiyuan Hu, Benqiao Wang, Xujia Liu, Ling Tong, Yitong Xu, Hao Zhang
Frontiers in Oncology.2025;[Epub] CrossRef
Association between CACNA1D polymorphisms and hypospadias in a southern Chinese population
Ye He, Binyao Li, Xinying Zhao, Lingling Pan, Yanqing Liu, Chaoting Lan, Fuming Deng, Wen Fu, Yan Zhang, Xiaoyu Zuo
Journal of Pediatric Urology.2024; 20(3): 438.e1. CrossRef
The L‐type calcium channel CaV1.3: A potential target for cancer therapy
Xuerun Liu, Boqiang Shen, Jingyi Zhou, Juan Hao, Jianliu Wang
Journal of Cellular and Molecular Medicine.2024;[Epub] CrossRef

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Sarcoma

Real-World Clinical Outcomes and Prognostic Factors for Patients with Advanced Angiosarcoma who Received Systemic Treatment: Changhee Park, Miso Kim, Yoonjin Kwak, Kyung Chul Moon, Se Hyun Kim, Bhumsuk Keam, Yu Jung Kim, Tae Min Kim, Dong-Wan Kim; Cancer Res Treat. 2021;53(4):1195-1203. Published online February 1, 2021; DOI: https://doi.org/10.4143/crt.2020.1337

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Purpose
Angiosarcoma is a highly aggressive mesenchymal tumor. Although systemic chemotherapy is often considered for the inoperable or metastatic angiosarcoma, the outcome of such treatment is unsatisfactory and poorly delineated.
Materials and Methods
We reviewed electronic medical records of 75 patients with angiosarcoma who were treated with systemic chemotherapy for inoperable or metastatic disease. Patients were classified as having liver involvement if they had either primary or metastatic hepatic lesions.
Results
Among the patients evaluated, 51 patients were male (68%) and 24 patients (32%) had primary cutaneous angiosarcoma. Liver involvement was present in 28 patients (37.3%). A total of 59 patients received first-line weekly paclitaxel (wPac) and showed an objective response rate (ORR) of 23.7% (n=14), a median progression free survival (mPFS) of 4.0 months (95% confidence interval [CI] 3.0–6.1), and a median overall survival (mOS) of 10.2 months (95% CI 7.0–14.6). Among patients without liver involvement, patients receiving wPac (n=35) had significantly prolonged mPFS (5.8 vs. 3.2 months, respectively, p=0.014) with a tendency for prolonged mOS (13.8 vs. 11.6 months, respectively, p=0.13) than those receiving other regimens (n=12). A total of 24 patients received second- or later-line pazopanib monotherapy and showed an ORR of 16.7% (n=4), a mPFS of 2.4 months (95% CI 1.8–4.3) and a mOS of 5.4 months (95% CI 3.5–NA).
Conclusion
Treatment with first-line wPac and later-line pazopanib seems to provide survival benefit, especially for patients with advanced angiosarcoma without liver involvement.

Citations

Citations to this article as recorded by

Punch Biopsy as a Diagnostic Keystone for Metastatic Cardiac Angiosarcoma Treated With Anthracycline-Based Chemotherapy: A Case Report
Aonghus Joyce, Gráinne Murphy, Cynthia C Heffron, David Aherne, Richard M Bambury
Cureus.2025;[Epub] CrossRef
Clinicopathological features, treatment outcomes, and prognostic factors of angiosarcoma: a 21-year experience at one center
Chenyan Fang, Xiaoting Zeng, Qing Ji, Tao Zhu, Meiyu Fang, Jun Cao
Orphanet Journal of Rare Diseases.2025;[Epub] CrossRef
Chest Wall Angiosarcoma with an Initial Presentation of Multiple Lung Metastases: a Case Report
Hitoshi Suzuki, Mari Shinoda, Daisuke Ito, Shin Shomura, Kentaro Inoue, Kazuo Fukutome, Akira Shimamoto, Hisamichi Yuda
Haigan.2024; 64(7): 917. CrossRef
Hepatic Angiosarcoma with Peliosis Hepatis
Kensuke Kitsugi, Kazuhito Kawata, Moe Matsumoto, Masahiro Umemura, Tomohiko Hanaoka, Maho Yamashita, Shingo Takatori, Jun Ito, Kazuyoshi Ohta, Takeshi Chida, Hidenao Noritake, Takafumi Suda
Internal Medicine.2023; 62(8): 1157. CrossRef
Conversion surgery for recurrent hepatic angiosarcoma after systemic chemotherapy with paclitaxel
Yuta Ushida, Takafumi Sato, Tomotaka Kato, Yasuyuki Shigematsu, Hiromichi Ito, Takeshi Suzuki, Yosuke Inoue, Yoshihiro Ono, Atsushi Oba, Yu Takahashi
Clinical Journal of Gastroenterology.2022; 15(2): 427. CrossRef
Management of Cutaneous Angiosarcoma: an Update Review
Siwei Bi, Ai Zhong, Xiya Yin, Jingyi Li, Ying Cen, Junjie Chen
Current Treatment Options in Oncology.2022; 23(2): 137. CrossRef
Results of NC-6300 (Nanoparticle Epirubicin) in an Expansion Cohort of Patients with Angiosarcoma
Richard F Riedel, Victoria Chua, Ania Moradkhani, Natalie Krkyan, Amir Ahari, Atsushi Osada, Sant P Chawla
The Oncologist.2022; 27(10): 809. CrossRef

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Radiation-Induced Sarcoma: A 15-Year Experience in a Single Large Tertiary Referral Center: Kyung Su Kim, Ji Hyun Chang, Noorie Choi, Han-Soo Kim, Ilkyu Han, Kyung Chul Moon, Il Han Kim, Hak Jae Kim; Cancer Res Treat. 2016;48(2):650-657. Published online September 9, 2015; DOI: https://doi.org/10.4143/crt.2015.171

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Purpose
The purpose of this study is to report on the incidence and the experience in management of radiation-induced sarcoma (RIS) at a large single center in Korea for 15 years.
Materials and Methods
We retrospectively reviewed the sarcoma registry of a large institution from January 2000 to April 2014.
Results
Out of the 3,674 patients listed in the registry, 33 patients (0.9%) diagnosed with RIS were identified. The median latency of RIS was 12.1 years. The number of cases of RIS increased from four cases in the years 2000-2003 to 14 cases in the years 2012-2014. The most common histology was osteosarcoma (36.4%). The median follow-up period was 23.1 months, the median overall survival (OS) of all patients was 2.9 years, and their 5-year survival rate was 44.7%. Univariate and multivariate analyses showed association of the age at diagnosis (p=0.01) and the treatment aim (p=0.001) with the OS. The median OS and the 5-year survival rate of patients treated with curative surgery (n=19) were 9.6 years and 65%, respectively, and of the conservatively treated patients, 0.7 years and 0% (n=14). Re-irradiation was delivered to nine patients, and radiation toxicity was observed in five patients.
Conclusion
In this study, RIS accounted for 0.9% of the cases of sarcoma, with increasing incidence. Despite the association of curative resection with increased survival, it could be applied to only 58% of the patients. Considering the limited treatment options for RIS, conduct of a genetic study to identify the underlying mechanism of RIS is needed.

Citations

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Case Report

A Case of Erdheim-Chester Disease with Asymptomatic Renal Involvement: Hyun Jung Lee, Kyoung Yul Lee, Dong-Yeop Shin, Yun Gyoo Lee, Se Youn Choi, Kyung Chul Moon, Il-Kyu Han, Tae Min Kim; Cancer Res Treat. 2012;44(2):146-150. Published online June 30, 2012; DOI: https://doi.org/10.4143/crt.2012.44.2.146

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Erdheim-Chester disease is a rare non-Langerhans-cell histiocytosis involving bones and multiple organs. Its clinical course can vary, from an asymptomatic state to a fatal disease, with renal involvement being a common cause of death. A 41-year-old man presented with a 10-month history of bilateral lower limb pain. Left perirenal soft-tissue infiltration had been found incidentally two years earlier. No progression of the lesion or deterioration of renal function was observed for a period of two years. At admission, plain radiography and magnetic resonance imaging of the patient's lower limbs showed patchy osteosclerosis. Biopsy of the tibia revealed histiocytic infiltration, which was found to be positive for CD68 and negative for CD1a. This report describes an unusual case of Erdheim-Chester disease involving a stationary course of disease with no specific treatment for a long period of time.

Citations

Citations to this article as recorded by

Radiological assessment of extremity bone involvement in Erdheim-Chester disease: a systematic review of case reports
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Reply to Commentary on "A Case of Erdheim-Chester Disease with Asymptomatic Renal Involvement"
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