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Huang Y, Wang Y, Kong B, Zheng F, Ding Y, Hayford B, Guo J, Qin R. Pulmonary artery intimal sarcoma mimicking pulmonary thromboembolism: A case report and literature review. Oncol Lett 2025; 29:169. [PMID: 39963321 PMCID: PMC11831714 DOI: 10.3892/ol.2025.14915] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 09/15/2024] [Accepted: 01/09/2025] [Indexed: 02/20/2025] Open
Abstract
Pulmonary artery intimal sarcoma (PAIS) is a rare and highly aggressive form of hemangiosarcoma. The non-specific clinical manifestations, signs and routine imaging findings of this sarcoma often mimic those of pulmonary thromboembolism (PTE), resulting in frequent misdiagnosis as PTE prior to pathological confirmation in most patients. This delay in achieving an accurate diagnosis poses challenges for promptly initiating optimal treatment and further contributes to the unfavorable prognosis. The current study presents the case of a 68-year-old female who presented with acute chest tightness and dyspnea. Transthoracic echocardiography revealed the presence of atypical echogenic structures within the primary pulmonary artery, while computed tomography pulmonary angiography showed marked filling defects in the main trunk and branches of the pulmonary artery. The patient was initially misdiagnosed with PTE but did not respond well to anticoagulant therapy. Subsequent surgical resection confirmed the diagnosis of PAIS through pathological examination. Despite postoperative treatment with molecular-targeted antitumor drugs, the patient experienced tumor recurrence and intrapulmonary metastasis, ultimately succumbing to disease progression. This exceptional case is being presented to enhance the clinical understanding of PAIS, to encourage further extensive research for reliable diagnostic approaches and to provide further data that will help form efficacious therapeutic strategies to ameliorate the unfavorable prognosis of affected patients.
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Affiliation(s)
- Yan Huang
- Department of Cardiology, Jiangsu University Affiliated People's Hospital, Zhenjiang Clinical Medical College of Nanjing Medical University, Zhenjiang, Jiangsu 212002, P.R. China
| | - Yiming Wang
- Department of Cardiology, Jiangsu University Affiliated People's Hospital, Zhenjiang Clinical Medical College of Nanjing Medical University, Zhenjiang, Jiangsu 212002, P.R. China
| | - Biko Kong
- Department of Cardiology, Jiangsu University Affiliated People's Hospital, Zhenjiang Clinical Medical College of Nanjing Medical University, Zhenjiang, Jiangsu 212002, P.R. China
| | - Feng Zheng
- Department of Ultrasound, Jiangsu University Affiliated People's Hospital, Zhenjiang, Jiangsu 212002, P.R. China
| | - Yi Ding
- Department of Medical Imaging, Jiangsu University Affiliated People's Hospital, Zhenjiang, Jiangsu 212002, P.R. China
| | - Boamah Hayford
- Research Center of Fluid Machinery Engineering and Technology, Jiangsu University, Zhenjiang, Jiangsu 212013, P.R. China
| | - Junfang Guo
- Department of Cardiology, Jiangsu University Affiliated People's Hospital, Zhenjiang Clinical Medical College of Nanjing Medical University, Zhenjiang, Jiangsu 212002, P.R. China
| | - Rong Qin
- Department of Medical Oncology, Jiangsu University Affiliated People's Hospital, Zhenjiang, Jiangsu 212002, P.R. China
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Kashizaki F, Chen H, Tsuchiya N, Matsumoto S, Osada R, Miyasaka A, Kaneko M, Kikuchi A, Kojima Y, Yumoto K, Hashiyama N, Osawa H, Koizumi H, Mo M, Takahashi K, Kaneko T. Clinical Features of Primary Pulmonary Artery Sarcoma: A Systematic Review and Pooled Analysis. Arch Bronconeumol 2024:S0300-2896(24)00484-8. [PMID: 39827013 DOI: 10.1016/j.arbres.2024.12.012] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Received: 07/22/2024] [Revised: 12/07/2024] [Accepted: 12/13/2024] [Indexed: 01/22/2025]
Abstract
OBJECTIVES Primary pulmonary artery sarcoma (PPAS) is a rare disease with unclear clinical manifestations. Advances in imaging devices have improved diagnostic capabilities, potentially affecting clinical characteristics and overall survival (OS); however, details remain unclear. This study conducted a pooled analysis of case reports and series to analyse the clinical characteristics and OS of PPAS in the era of advanced medical devices. METHODS Data were sourced from PubMed and CINAHL, focusing on studies published between 1 January 2014 and 31 December 2023. The study included patients diagnosed with PPAS, with extracted data covering demographics, diagnosis, treatments, and survival. RESULTS Overall, 643 patients were included (mean age: 52.6±13.1 years; 50.4% were female). Initially, 70.6% were diagnosed with pulmonary thromboembolism (PTE), and 15.4% were suspected of having PPAS. Among these, 93.9% and 55.2% showed suggestive findings on computed tomography (CT)-integrated positron emission tomography with 2-deoxy-2-18F-fluoro-d-glucose (18F-FDG PET/CT) and CT, respectively, with 98.2% confirmed before death. The right main pulmonary artery was the most affected site on CT (72.3%). Surgery and chemotherapy were performed in 81.4% and 66.4% of patients, respectively. The median OS was 31 months, with surgery extending OS across all stages and chemotherapy benefiting stages III-IV. Longer OS was achieved in patients who underwent complete surgical resection. CONCLUSIONS 18F-FDG PET/CT and multi-detector-row CT can differentiate PTE from PPAS. These medical devices may contribute to improved OS.
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Affiliation(s)
- Fumihiro Kashizaki
- Department of Respiratory Medicine, Yokohama Minami Kyosai Hospital, Yokohama, Japan.
| | - Hao Chen
- Department of Respiratory Medicine, Yokohama Minami Kyosai Hospital, Yokohama, Japan
| | - Nanami Tsuchiya
- Department of Respiratory Medicine, Yokohama Minami Kyosai Hospital, Yokohama, Japan
| | - Sachiko Matsumoto
- Department of Respiratory Medicine, Yokohama Minami Kyosai Hospital, Yokohama, Japan
| | - Reeko Osada
- Department of Respiratory Medicine, Yokohama Minami Kyosai Hospital, Yokohama, Japan
| | - Atsushi Miyasaka
- Department of Respiratory Medicine, Yokohama Minami Kyosai Hospital, Yokohama, Japan
| | - Mai Kaneko
- Department of Respiratory Medicine, Yokohama Minami Kyosai Hospital, Yokohama, Japan
| | - Akitomo Kikuchi
- Department of General Thoracic Surgery, Yokohama Minami Kyosai Hospital, Yokohama, Japan
| | - Yui Kojima
- Department of Pathology, Yokohama Minami Kyosai Hospital, Yokohama, Japan
| | - Kentaro Yumoto
- Department of Respiratory Medicine, Yokohama Minami Kyosai Hospital, Yokohama, Japan
| | - Naoki Hashiyama
- Department of Cardiovascular Surgery, Yokohama Minami Kyosai Hospital, Yokohama, Japan
| | - Hiroyuki Osawa
- Department of General Thoracic Surgery, Yokohama Minami Kyosai Hospital, Yokohama, Japan
| | - Harumi Koizumi
- Department of Respiratory Medicine, Yokohama Minami Kyosai Hospital, Yokohama, Japan
| | - Makoto Mo
- Department of Cardiovascular Surgery, Yokohama Minami Kyosai Hospital, Yokohama, Japan
| | - Kenichi Takahashi
- Department of Respiratory Medicine, Yokohama Minami Kyosai Hospital, Yokohama, Japan
| | - Takeshi Kaneko
- Department of Respiratory Medicine, Yokohama City University Hospital, Yokohama, Japan
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Zhang DY, Su L, Wang YW. Malignant solitary fibrous tumor in the central nervous system treated with surgery, radiotherapy and anlotinib: A case report. World J Clin Cases 2022; 10:631-642. [PMID: 35097089 PMCID: PMC8771389 DOI: 10.12998/wjcc.v10.i2.631] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 05/03/2021] [Revised: 11/01/2021] [Accepted: 12/08/2021] [Indexed: 02/06/2023] Open
Abstract
BACKGROUND Solitary fibrous tumor (SFT) of the central nervous system is rare. It is predominantly benign and rarely malignant. There is no established standardized treatment regimen for malignant intracranial SFTs.
CASE SUMMARY We present a rare case of SFT in a 9-year-old girl with a space-occupying effect in the frontal-parietal lobes. She underwent craniotomy, and the mass was resected. Immunohistochemistry examination of the specimen showed that Ki-67 proliferation index staining was highly positive in 80% of tumor cells. Whole exome sequencing of the surgical tissue showed 38 somatic gene mutations and 1 gene amplification such as fibroblast growth factor receptor 4 or TP53. At 1.5 mo after surgery, head magnetic resonance imaging revealed that the tumor had recurred. The patient received 60 Gy and 30 fractions of intensity modulated radiotherapy. The patient then received anlotinib 8 mg po qd for 1-14 d of a 21 d cycle. Following this regimen, the patient achieved stable disease for > 17 mo. Magnetic resonance imaging at 1.5 year after surgery showed that the tumor had not progressed.
CONCLUSION This is the first reported case of SFT of the central nervous system treated with surgery, radiotherapy and anlotinib. This regimen may be an effective treatment option for malignant intracranial SFT patients.
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Affiliation(s)
- Dong-Yong Zhang
- Department of Neurosurgery, the First Affiliated Hospital of China Medical University, Shenyang 110001, Liaoning Province, China
| | - Lan Su
- Department of Genetron Health, Genetron Health, Beijing 110024, China
| | - Yi-Wei Wang
- Department of Anatomy, Shenyang Medical College, Shenyang 110034, Liaoning Province, China
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Wu Y, Zhang K, Guan J, Wu W, Zhang J, Chen H. Treatment with Anlotinib After Chemotherapy and EGFR-TKI Resistance in Lung Adenosquamous Carcinoma with Concurrent EGFR and PIK3CA Mutations: A Case Report and Literature Review. Cancer Manag Res 2021; 13:7047-7053. [PMID: 34526820 PMCID: PMC8437381 DOI: 10.2147/cmar.s326094] [Citation(s) in RCA: 6] [Impact Index Per Article: 1.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 06/24/2021] [Accepted: 08/30/2021] [Indexed: 11/23/2022] Open
Abstract
Concurrent mutations of epidermal growth factor receptor (EGFR) and phosphatidylinositol-4,5-bisphosphate 3-kinase catalytic subunit alpha (PIK3CA) in non-small cell lung cancer (NSCLC) are rare, and the presence of concurrent mutations may complicate treatment. Herein, we report a case of primary lung adenosquamous carcinoma with concurrent EGFR 21 (L858R) and PIK3CA (H1047R/E545K) mutations, and the results of a literature review to help management and treatment. A 49-year-old female was admitted our department for coughing and excessive sputum production for more than 1 month. Computed tomography (CT) of the chest identified a lesion, and a CT-guided needle biopsy was performed. Pathological examination and immunohistochemistry (IHC) staining confirmed a diagnosis of primary lung adenosquamous carcinoma. Amplification refractory mutation system-polymerase chain reaction (ARMS-PCR) gene sequencing demonstrated mutations in both EGFR 21 (L858R) and PIK3CA (H1047R/E545K) mutations in adenocarcinoma (AC) component. She was treated with pemetrexed plus platinum-based chemotherapy and an EGFR-tyrosine kinase inhibitor (TKI). Disease progression occurred with gefitinib or osimertinib as maintenance therapy. A repeat CT-guided needle biopsy was performed, and generation sequencing (NGS) revealed EGFR 21 (L858R) and PIK3CA (H1047R/E545K) mutations. Anlotinib monotherapy was then administered as the third-line treatment, and there was a PR. The patient is currently still receiving treatment and follow-up. To our knowledge, there is little evidence that anlotinib is beneficial when there are concurrent EGFR and PIK3CA mutations. PIK3CA mutations are associated with poor therapeutic effects and short survival time. Concurrent EGFR and PIK3CA mutations do not respond to EGFR-TKI treatment. Chemotherapy should be given in combination with a TKI and can prolong the progression-free survival (PFS) and overall survival (OS) of patients with lung cancer.
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Affiliation(s)
- Yonghui Wu
- Department of Cardiothoracic Surgery, The Third Affiliated Hospital of Sun Yat-Sen University, Guangzhou, Guangdong, 510630, People's Republic of China
| | - Kai Zhang
- Department of Cardiothoracic Surgery, The Third Affiliated Hospital of Sun Yat-Sen University, Guangzhou, Guangdong, 510630, People's Republic of China
| | - Jiexia Guan
- Department of Pathology, The Third Affiliated Hospital of Sun Yat-Sen University, Guangzhou, Guangdong, 510630, People's Republic of China
| | - Weibin Wu
- Department of Cardiothoracic Surgery, The Third Affiliated Hospital of Sun Yat-Sen University, Guangzhou, Guangdong, 510630, People's Republic of China
| | - Jian Zhang
- Department of Cardiothoracic Surgery, The Third Affiliated Hospital of Sun Yat-Sen University, Guangzhou, Guangdong, 510630, People's Republic of China
| | - Huiguo Chen
- Department of Cardiothoracic Surgery, The Third Affiliated Hospital of Sun Yat-Sen University, Guangzhou, Guangdong, 510630, People's Republic of China
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Zou J, Yang X, Duan J, Wang J, Yang Z, Luo D, Liu L, Chen J, Nie J. A Case Report of Targeted Therapy with Anlotinib in a Patient with Advanced Breast Metaplastic Carcinoma. Onco Targets Ther 2021; 14:4599-4607. [PMID: 34475766 PMCID: PMC8408045 DOI: 10.2147/ott.s318645] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.5] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 05/11/2021] [Accepted: 08/11/2021] [Indexed: 12/13/2022] Open
Abstract
Metaplastic breast carcinoma (MBC) is a rare malignant breast tumor, and no effective chemotherapy unique to metaplastic carcinoma exists. As MBC is typically “triple negative”, endocrine therapy and molecular therapy targeted to Her2 might not be favorable, resulting in a poor prognosis. Anlotinib is currently being tested in patients with breast or cancer. Here, we report a successful case in which anlotinib was used to treat MBC. A 54-year-old female patient visited the hospital after the discovery of a left breast tumor 10 months prior, and tumor redness and swelling had lasted for more than one month. After admission, relevant examinations were performed. After left breast tumor puncture revealed left emulsified biological cancer, the tumor significantly increased in size, and bleeding was obvious after 2 cycles of the “EC” chemotherapy regimen. The curative effect was evaluated as progressive disease (PD). After two cycles of chemotherapy with the “PCb” regimen, the efficacy was still PD. The Karnofsky performance status (KPS) score of the patient after 4 cycles of chemotherapy was 60 points, with severe anemia, and she could not tolerate chemotherapy. The patient was given radiotherapy to stop bleeding, and the tumor further increased in size during radiotherapy. The curative effect was evaluated as PD. After a multidisciplinary consultation in our hospital, we initiated oral anlotinib (12 mg; 2 weeks on, 1 week off). The tumor significantly decreased in size after taking anlotinib, and the efficacy was evaluated as PR. Adverse reactions during treatment were controlled, and progression-free survival (PFS) reached up to 25+ months. The follow-up is ongoing. The patient has provided written informed consent for the case details and images to be published, and at the same time institutional approval was required to publish the case details, we report this case.
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Affiliation(s)
- Jieya Zou
- The Third Department of Breast Surgery, The Third Affiliated Hospital of Kunming Medical University, Yunnan Cancer Hospital, Kunming, 650118, People's Republic of China
| | - Xiaojuan Yang
- The Third Department of Breast Surgery, The Third Affiliated Hospital of Kunming Medical University, Yunnan Cancer Hospital, Kunming, 650118, People's Republic of China
| | - Jiajun Duan
- The Third Department of Breast Surgery, The Third Affiliated Hospital of Kunming Medical University, Yunnan Cancer Hospital, Kunming, 650118, People's Republic of China
| | - Ji Wang
- The Third Department of Breast Surgery, The Third Affiliated Hospital of Kunming Medical University, Yunnan Cancer Hospital, Kunming, 650118, People's Republic of China
| | - Zhuangqing Yang
- The Third Department of Breast Surgery, The Third Affiliated Hospital of Kunming Medical University, Yunnan Cancer Hospital, Kunming, 650118, People's Republic of China
| | - Dan Luo
- Department of Pathology, The Third Affiliated Hospital of Kunming Medical University, Yunnan Cancer Hospital, Kunming, 650118, People's Republic of China
| | - Lihua Liu
- The Third Department of Breast Surgery, The Third Affiliated Hospital of Kunming Medical University, Yunnan Cancer Hospital, Kunming, 650118, People's Republic of China
| | - Junyao Chen
- The Third Department of Breast Surgery, The Third Affiliated Hospital of Kunming Medical University, Yunnan Cancer Hospital, Kunming, 650118, People's Republic of China
| | - Jianyun Nie
- The Third Department of Breast Surgery, The Third Affiliated Hospital of Kunming Medical University, Yunnan Cancer Hospital, Kunming, 650118, People's Republic of China
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Fang S, Cheng W, Zhang M, Yang R. Association of TP53 Mutations with Response to Anlotinib Treatment in Advanced Non-Small Cell Lung Cancer. Onco Targets Ther 2020; 13:6645-6650. [PMID: 32753898 PMCID: PMC7354100 DOI: 10.2147/ott.s257052] [Citation(s) in RCA: 6] [Impact Index Per Article: 1.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 04/09/2020] [Accepted: 06/15/2020] [Indexed: 12/24/2022] Open
Abstract
Multitargeted antiangiogenic drugs have demonstrated significant antitumor activity against a variety of solid tumors. Anlotinib, a novel oral multitargeted antiangiogenic tyrosine kinase inhibitor, was approved as a third-line treatment for advanced NSCLC in China. However, predictive biomarkers are currently insufficient and are urgently required. Herein, we report three pre-treated cases of advanced NSCLC with TP53 mutations, wherein these patients showed partial response to anlotinib. Moreover, the three patients have achieved a progression-free survival of 8, 6.5, and 5 months, respectively. The main toxicities were hypertension, hand-foot syndrome and fatigue. In conclusion, TP53 mutations may represent a biomarker for predicting salutary effects of anlotinib.
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Affiliation(s)
- Shencun Fang
- Department of Respiratory Medicine, The Affiliated Brain Hospital of Nanjing Medical University, Nanjing, Jiangsu, People's Republic of China
| | - Wanwan Cheng
- Department of Respiratory Medicine, The Affiliated Brain Hospital of Nanjing Medical University, Nanjing, Jiangsu, People's Republic of China
| | - Meiling Zhang
- Department of Oncology, The First Affiliated Hospital of Nanjing Medical University, Nanjing, Jiangsu, People's Republic of China
| | - Rusong Yang
- Department of Thoracic Surgery, The Affiliated Brain Hospital of Nanjing Medical University, Nanjing, Jiangsu, People's Republic of China
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