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Effective Treatment of Low-Grade Myofibroblastic Sarcoma with Apatinib: A Case Report and Literature Review

Low-grade myofibroblastic sarcoma (LGMS) is a rare, poorly differentiated, malignant tumor. The disease mainly occurs in the head and neck and rarely metastasizes at any age. Currently, there is no consensus regarding the treatment of metastatic LGMS. Here, we report the case of a 45-year-old man wh...

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Autores principales: Lin, Yan, Gao, Xing, Liu, Ziyu, Liu, Zhihui, Li, Yongqiang, Liang, Rong, Liao, Zhiling, Ye, Jiazhou
Formato: Online Artículo Texto
Lenguaje:English
Publicado: Dove 2022
Materias:
Acceso en línea:https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9188403/
https://www.ncbi.nlm.nih.gov/pubmed/35698620
http://dx.doi.org/10.2147/PGPM.S359492
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author Lin, Yan
Gao, Xing
Liu, Ziyu
Liu, Zhihui
Li, Yongqiang
Liang, Rong
Liao, Zhiling
Ye, Jiazhou
author_facet Lin, Yan
Gao, Xing
Liu, Ziyu
Liu, Zhihui
Li, Yongqiang
Liang, Rong
Liao, Zhiling
Ye, Jiazhou
author_sort Lin, Yan
collection PubMed
description Low-grade myofibroblastic sarcoma (LGMS) is a rare, poorly differentiated, malignant tumor. The disease mainly occurs in the head and neck and rarely metastasizes at any age. Currently, there is no consensus regarding the treatment of metastatic LGMS. Here, we report the case of a 45-year-old man who was admitted to our hospital with cough for two weeks and abdominal pain for one week. Preoperative computed tomography revealed a large mass (116×35 mm) in both the lungs and jejunal mass (maximum diameter, 32 mm). The tumor was excised, and immunohistochemical staining was positive for SMA and CD117 and negative for desmin and CD34, indicating a case of LGMS. The patient was effectively treated with apatinib (250 mg/day) after failure of imatinib, liposomal doxorubicin, and ifosfamide. The progression-free survival time was 8.5 months, and the overall survival time was 17 months after treatment with apatinib. No grade 3 or 4 side effects were observed, except hand-foot syndrome. Whole-exome sequencing (WES) and RNA sequencing (RNA-seq) were performed on the patient’s jejunal tumor and para-cancerous tissue samples, and bioinformatics analysis was performed on the results. WES identified five mutations in MKI67, OR2J2, EPPK1, FCGBP, and OR10G4. RNA-seq revealed that 1422 genes were upregulated and 1890 genes were downregulated. The differentially expressed genes were mainly enriched in the phosphatidylinositol 3-kinase (PI3K) signaling pathway, neuroactive ligand-receptor interaction signaling pathway, and cAMP signaling pathway. Our study indicated that apatinib may be a potential novel and effective treatment for LGMS.
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spelling pubmed-91884032022-06-12 Effective Treatment of Low-Grade Myofibroblastic Sarcoma with Apatinib: A Case Report and Literature Review Lin, Yan Gao, Xing Liu, Ziyu Liu, Zhihui Li, Yongqiang Liang, Rong Liao, Zhiling Ye, Jiazhou Pharmgenomics Pers Med Case Report Low-grade myofibroblastic sarcoma (LGMS) is a rare, poorly differentiated, malignant tumor. The disease mainly occurs in the head and neck and rarely metastasizes at any age. Currently, there is no consensus regarding the treatment of metastatic LGMS. Here, we report the case of a 45-year-old man who was admitted to our hospital with cough for two weeks and abdominal pain for one week. Preoperative computed tomography revealed a large mass (116×35 mm) in both the lungs and jejunal mass (maximum diameter, 32 mm). The tumor was excised, and immunohistochemical staining was positive for SMA and CD117 and negative for desmin and CD34, indicating a case of LGMS. The patient was effectively treated with apatinib (250 mg/day) after failure of imatinib, liposomal doxorubicin, and ifosfamide. The progression-free survival time was 8.5 months, and the overall survival time was 17 months after treatment with apatinib. No grade 3 or 4 side effects were observed, except hand-foot syndrome. Whole-exome sequencing (WES) and RNA sequencing (RNA-seq) were performed on the patient’s jejunal tumor and para-cancerous tissue samples, and bioinformatics analysis was performed on the results. WES identified five mutations in MKI67, OR2J2, EPPK1, FCGBP, and OR10G4. RNA-seq revealed that 1422 genes were upregulated and 1890 genes were downregulated. The differentially expressed genes were mainly enriched in the phosphatidylinositol 3-kinase (PI3K) signaling pathway, neuroactive ligand-receptor interaction signaling pathway, and cAMP signaling pathway. Our study indicated that apatinib may be a potential novel and effective treatment for LGMS. Dove 2022-06-07 /pmc/articles/PMC9188403/ /pubmed/35698620 http://dx.doi.org/10.2147/PGPM.S359492 Text en © 2022 Lin et al. https://creativecommons.org/licenses/by-nc/3.0/This work is published and licensed by Dove Medical Press Limited. The full terms of this license are available at https://www.dovepress.com/terms.php and incorporate the Creative Commons Attribution – Non Commercial (unported, v3.0) License (http://creativecommons.org/licenses/by-nc/3.0/ (https://creativecommons.org/licenses/by-nc/3.0/) ). By accessing the work you hereby accept the Terms. Non-commercial uses of the work are permitted without any further permission from Dove Medical Press Limited, provided the work is properly attributed. For permission for commercial use of this work, please see paragraphs 4.2 and 5 of our Terms (https://www.dovepress.com/terms.php).
spellingShingle Case Report
Lin, Yan
Gao, Xing
Liu, Ziyu
Liu, Zhihui
Li, Yongqiang
Liang, Rong
Liao, Zhiling
Ye, Jiazhou
Effective Treatment of Low-Grade Myofibroblastic Sarcoma with Apatinib: A Case Report and Literature Review
title Effective Treatment of Low-Grade Myofibroblastic Sarcoma with Apatinib: A Case Report and Literature Review
title_full Effective Treatment of Low-Grade Myofibroblastic Sarcoma with Apatinib: A Case Report and Literature Review
title_fullStr Effective Treatment of Low-Grade Myofibroblastic Sarcoma with Apatinib: A Case Report and Literature Review
title_full_unstemmed Effective Treatment of Low-Grade Myofibroblastic Sarcoma with Apatinib: A Case Report and Literature Review
title_short Effective Treatment of Low-Grade Myofibroblastic Sarcoma with Apatinib: A Case Report and Literature Review
title_sort effective treatment of low-grade myofibroblastic sarcoma with apatinib: a case report and literature review
topic Case Report
url https://www.ncbi.nlm.nih.gov/pmc/articles/PMC9188403/
https://www.ncbi.nlm.nih.gov/pubmed/35698620
http://dx.doi.org/10.2147/PGPM.S359492
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