Successful treatment of advanced pancreatic leiomyosarcoma treated with gemcitabine plus nab -paclitaxel: a case report
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CASE REPORT
Successful treatment of advanced pancreatic leiomyosarcoma treated with gemcitabine plus nab‑paclitaxel: a case report and literature review Yoshinori Kikuchi1 · Yusuke Nishikawa1 · Makoto Amanuma1 · Yui Kishimoto1 · Kensuke Takuma1 · Megumi Wakayama2 · Kazutoshi Shibuya2 · Naoki Okano1 · Hideaki Shimada3,4 · Yoshinori Igarashi1 Received: 21 July 2020 / Accepted: 23 September 2020 © The Japan Society of Clinical Oncology 2020
Abstract Pancreatic leiomyosarcoma (PLMS) is an extremely rare tumor that accounts for 0.1% of pancreatic malignancies, and its chemotherapy has yet to be established. Generally, soft-tissue sarcoma chemotherapy is standard treatment with doxorubicin (DXR) alone. However, the effectiveness of gemcitabine (GEM) plus docetaxel (DOC) has been shown in uterine leiomyoma. In contrast, the GEM plus nab-paclitaxel (PTX) regimen has been established as first-line chemotherapy for unresectable pancreatic cancer. For this study, we selected the GEM plus nab-PTX regimen for patients with PLMS, achieving success in approximately 10 months. From a search on PubMed, we found only 12 cases of PLMS (including this case) that underwent chemotherapy. Our case is the first reported patient to have survived more than 2 years with chemotherapy alone. In a nude mouse model, the GEM plus DOC regimen was shown to significantly decrease tumor size when compared with DXR in leiomyosarcoma, and the GEM plus nab-PTX regimen was reported to significantly reduce necrosis when compared with DXR alone, GEM alone, DOC alone, nab-PTX alone and GEM plus DOC in soft-tissue sarcoma. GEM plus nab-PTX therapy might therefore be the first choice for soft-tissue sarcoma and leiomyosarcoma. This is the first reported case of PLMS treated with GEM plus nab-PTX. Keywords Pancreatic leiomyosarcoma · Chemotherapy · Gemcitabine · Nab-paclitaxel
Introduction Primary pancreatic leiomyosarcoma (PLMS), first reported in 1951 by Ross CF [1], is an extremely rare mesenchymal pancreatic tumor that accounts for 0.1% of pancreatic malignancies [2]. PLMS has a dismal prognosis; Xu et al. reported * Yoshinori Kikuchi y‑[email protected]‑u.ac.jp 1
Division of Gastroenterology and Hepatology, Department of Internal Medicine, Faculty of Medicine, Toho University, 6‑11‑1 Omori‑nishi, Ota‑ku, Tokyo 143‑8541, Japan
2
Department of Surgical Pathology (Omori), Toho University, Tokyo, Japan
3
Division of General and Gastroenterological Surgery, Department of Surgery (Omori), Toho University, Tokyo, Japan
4
Department of Gastroenterological Surgery and Clinical Oncology, Toho University Graduate School of Medicine, Tokyo, Japan
that the median survival time and 3-year survival rate were 12 ± 6.3 months and 0%, respectively [3]. Almost all cases of PLMS are from diagnostic and surgical reports, with very few reports on its chemotherapy, of which there were only three cases, including our case, on effective chemotherapy. Our case is the first patient reported to survive more than 2 years with chemotherapy alone. Doxorubicin (
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