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Manipadam JM, Bains SPS, Mahesh S, Emmanuel AM, Ramesh H. Thoracoscopic Esophagectomy for a Huge Leiomyosarcoma. Surg J (N Y) 2019; 5:e163-e169. [PMID: 31650035 PMCID: PMC6805235 DOI: 10.1055/s-0039-1696729] [Citation(s) in RCA: 1] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Subscribe] [Scholar Register] [Received: 03/09/2019] [Accepted: 07/17/2019] [Indexed: 12/25/2022] Open
Abstract
Esophageal leiomyosarcoma is the commonest of all esophageal sarcomas but yet has a very low incidence. These tumors have been resected by the open approach so far. We describe the steps and challenges involved in the thoracoscopic excision of a huge leiomyosarcoma of the esophagus.
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Affiliation(s)
- John Mathew Manipadam
- Department of GI Surgery and Liver Transplantation, VPS Lakeshore Hospital and Research Centre, Kochi, Kerala, India
| | - Satinder Pal Singh Bains
- Department of GI Surgery and Liver Transplantation, VPS Lakeshore Hospital and Research Centre, Kochi, Kerala, India
| | - S Mahesh
- Department of GI Surgery and Liver Transplantation, VPS Lakeshore Hospital and Research Centre, Kochi, Kerala, India
| | - Ami Maria Emmanuel
- Department of Pathology, VPS Lakeshore Hospital and Research Centre, Kochi, Kerala, India
| | - H Ramesh
- Department of GI Surgery and Liver Transplantation, VPS Lakeshore Hospital and Research Centre, Kochi, Kerala, India
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2
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Ebi M, Sakamoto K, Inoue S, Ozeki T, Kimura M, Kondo R, Sugiyama T, Yamamoto K, Adachi K, Yoshimine T, Yamaguchi Y, Tamura Y, Izawa S, Hijikata Y, Funaki Y, Ogasawara N, Sasaki M, Kasugai K. Esophageal Leiomyosarcoma Diagnosed by Endoscopic Ultrasound-guided Fine-needle Aspiration Biopsy and Cured with Surgical Resection. Intern Med 2019; 58:2479-2483. [PMID: 31118374 PMCID: PMC6761329 DOI: 10.2169/internalmedicine.2219-18] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Figures] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Indexed: 12/12/2022] Open
Abstract
Esophageal leiomyosarcomas are rare. We herein present the case of an 82-year-old patient who underwent upper gastrointestinal endoscopy, which revealed a submucosal tumor of 30 mm in diameter that was in contact with the esophagus. Endoscopic ultrasound-guided fine needle aspiration biopsy was performed and the histopathological findings indicated esophageal leiomyosarcoma. Surgical resection was performed. On histopathological examination, the tumor was found to consist of spindle cells with deep chromatin nuclei. The tumor was finally diagnosed as esophageal leiomyosarcoma. We were able to diagnose early-stage esophageal leiomyosarcoma using endoscopic ultrasound-guided fine-needle aspiration biopsy (EUS-FNA). EUS-FNA is mostly recommended as a diagnostic tool for esophageal submucosal tumors.
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Affiliation(s)
- Masahide Ebi
- Department of Gastroenterology, Aichi Medical University School of Medicine, Japan
| | - Kazumasa Sakamoto
- Department of Gastroenterology, Aichi Medical University School of Medicine, Japan
| | - Satoshi Inoue
- Department of Gastroenterology, Aichi Medical University School of Medicine, Japan
| | - Tomonori Ozeki
- Department of Gastroenterology, Aichi Medical University School of Medicine, Japan
| | - Mikitoshi Kimura
- Department of Gastroenterology, Aichi Medical University School of Medicine, Japan
| | - Riki Kondo
- Department of Gastroenterology, Aichi Medical University School of Medicine, Japan
| | - Tomoya Sugiyama
- Department of Gastroenterology, Aichi Medical University School of Medicine, Japan
| | - Kazuhiro Yamamoto
- Department of Gastroenterology, Aichi Medical University School of Medicine, Japan
| | - Kazunori Adachi
- Department of Gastroenterology, Aichi Medical University School of Medicine, Japan
| | - Takashi Yoshimine
- Department of Gastroenterology, Aichi Medical University School of Medicine, Japan
| | - Yoshiharu Yamaguchi
- Department of Gastroenterology, Aichi Medical University School of Medicine, Japan
| | - Yasuhiro Tamura
- Department of Gastroenterology, Aichi Medical University School of Medicine, Japan
| | - Shinya Izawa
- Department of Gastroenterology, Aichi Medical University School of Medicine, Japan
| | - Yasutaka Hijikata
- Department of Gastroenterology, Aichi Medical University School of Medicine, Japan
| | - Yasushi Funaki
- Department of Gastroenterology, Aichi Medical University School of Medicine, Japan
| | - Naotaka Ogasawara
- Department of Gastroenterology, Aichi Medical University School of Medicine, Japan
| | - Makoto Sasaki
- Department of Gastroenterology, Aichi Medical University School of Medicine, Japan
| | - Kunio Kasugai
- Department of Gastroenterology, Aichi Medical University School of Medicine, Japan
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Yamamoto Y, Nishisaki H, Koma YI, Sawai H, Sakai A, Mimura T, Kushida S, Tsumura H, Sakamoto T, Tobimatsu K, Miki I, Sakuma T, Tsuda M, Mano M, Hirose T, Inokuchi H. Polypoid leiomyosarcoma of the esophagus treated by endoscopic submucosal dissection. Dig Endosc 2015; 27:700-3. [PMID: 25597630 DOI: 10.1111/den.12437] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.3] [Reference Citation Analysis] [Abstract] [Key Words] [Track Full Text] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 11/26/2014] [Accepted: 01/14/2015] [Indexed: 01/04/2023]
Abstract
We report a rare case of polypoid leiomyosarcoma of the esophagus that was treated by endoscopic submucosal dissection (ESD). A 63-year-old man with complaints of progressive dysphagia was referred to Hyogo Cancer Center for treatment of esophageal tumor. Esophagoscopy revealed a polypoid tumor 25 mm in diameter on the left side of the upper esophagus. Despite several biopsy specimens, the diagnosis could not be confirmed. Computed tomography showed a protruded, homogeneously enhancing mass in the upper esophagus, but no lymph node enlargement or metastasis. After 1.5 months, the esophagogram showed a filling defect 47 mm in diameter in the upper esophagus. Given this rapid tumor growth, en bloc resection was done by ESD for therapeutic diagnosis. After this treatment, the tumor seemed to grow larger, showing a short stalk and occupying the esophageal lumen. Histopathologically, the tumor comprised pleomorphic spindle cells with mitosis. Tumor invasion involved the lumina propria mucosae and contact with the muscularis mucosae, but not involving the submucosa. Immunohistochemical examination showed positive staining for smooth muscle actin and HHF35, but negative for desmin, caldesmon, CD34, c-kit, DOG1, ALK, S-100 protein and cytokeratin. These histopathological findings were compatible with a diagnosis of esophageal leiomyosarcoma derived from the muscularis mucosae.
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Affiliation(s)
| | | | - Yu-ichiro Koma
- Department of Pathology, Kobe University Graduate School of Medicine, Hyogo, Japan
| | - Hiroaki Sawai
- Department of Gastroenterology, Hyogo Cancer Center, Hyogo, Japan
| | - Aya Sakai
- Department of Gastroenterology, Hyogo Cancer Center, Hyogo, Japan
| | - Takuya Mimura
- Department of Gastroenterology, Hyogo Cancer Center, Hyogo, Japan
| | - Saeko Kushida
- Department of Gastroenterology, Hyogo Cancer Center, Hyogo, Japan
| | - Hidetaka Tsumura
- Department of Gastroenterology, Hyogo Cancer Center, Hyogo, Japan
| | - Takeshi Sakamoto
- Department of Gastroenterology, Hyogo Cancer Center, Hyogo, Japan
| | | | - Ikuya Miki
- Department of Gastroenterology, Hyogo Cancer Center, Hyogo, Japan
| | - Toshiko Sakuma
- Department of Diagnostic Pathology, Hyogo Cancer Center, Hyogo, Japan
| | - Masahiro Tsuda
- Department of Gastroenterology, Hyogo Cancer Center, Hyogo, Japan
| | - Masayuki Mano
- Department of Clinical Laboratory, Osaka Medical Center, Osaka, Japan
| | - Takanori Hirose
- Department of Diagnostic Pathology, Hyogo Cancer Center, Hyogo, Japan
| | - Hideto Inokuchi
- Department of Gastroenterology, Hyogo Cancer Center, Hyogo, Japan
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Abstract
Leiomyosarcoma of the esophagus is a rare malignant tumor with slow growth and late metastasis. The aim of this study was to reassess the clinical characteristics and treatment modality in one of the largest series of esophageal leiomyosarcomas from a single institution. From February 1973 to December 2011, 12 cases of esophageal leiomyosarcoma were identified. The principal symptoms included progressive dysphagia in 11 cases (91.7%), retrosternal/back pain in four (33.3%), weight loss in four (33.3%), upper gastrointestinal hemorrhage in two (16.7%), and emesis in two (16.7%). The average duration of symptoms was 10.6 months. The location of the primary tumor was in the middle thoracic esophagus in five cases, and lower thoracic esophagus in seven cases. Six cases were classified as the polypoid type, five cases as the infiltrative type, and only one case as the intramural type. All 12 of the patients underwent esophagectomies, and radical resections were achieved in these patients. Based on the Kaplan-Meier Method, the 3-, 5-, and 10-year survival rates were 80.0%, 58.3%, and 31.1%, respectively, with a median survival of 63 months. Five-year survival rates for patients with polypoid or intramural tumors (n = 7) was 83.3%, and for patients with infiltrative tumor (n = 5) it was 25.0%. One of the patients had tumor resected four times and survived for 161 months. In conclusion, patients presenting with esophageal leiomyosarcomas have an excellent prognosis, and radical resection may achieve acceptable results.
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Affiliation(s)
- B H Zhang
- Thoracic Surgery, Cancer Hospital and Institute, Chinese Academy of Medical Sciences and Peking Union Medical College, Beijing, China
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Jang SS, Kim WT, Ko BS, Kim EH, Kim JO, Park K, Lee SW. A case of rapidly progressing leiomyosarcoma combined with squamous cell carcinoma in the esophagus. World J Gastroenterol 2013; 19:5385-5388. [PMID: 23983445 PMCID: PMC3752576 DOI: 10.3748/wjg.v19.i32.5385] [Citation(s) in RCA: 2] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [Key Words] [MESH Headings] [Track Full Text] [Download PDF] [Journal Information] [Submit a Manuscript] [Subscribe] [Scholar Register] [Received: 03/12/2013] [Revised: 06/15/2013] [Accepted: 07/18/2013] [Indexed: 02/06/2023] Open
Abstract
Esophageal leiomyosarcoma is a rare tumor that accounts for less than 1% of all malignant esophageal tumors. Esophageal leiomyosarcoma combined with squamous cell carcinoma is even rarer than solitary leiomyosarcoma. We experienced a case of leiomyosarcoma combined with squamous cell carcinoma that progressed very rapidly.
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Abstract
Leiomyosarcomas of the esophagus are rare, malignant, smooth-muscle tumors. The presenting symptoms are indistinguishable from other esophageal neoplasms, though the history may be longer due to the slow growth of these tumors. Barium studies may show large intramural masses with ulceration or tracking, expansile intraluminal masses or areas of luminal narrowing. Endoscopic biopsies may give a high false negative rate especially in cases where the mucosa is intact. The treatment of choice is surgical excision. Synchronous and metachronous metastases do not preclude surgery, provided the metastases are also resectable. Prognosis is better than in patients with squamous esophageal cancer. The role of adjuvant radiotherapy and chemotherapy is controversial. We report a 40-year-old man who presented to us with dysphagia and was found to have a leiomyosarcoma of the esophagus. He was treated successfully with esophagectomy and is disease-free after 7 years. We review the literature on esophageal leiomyosarcomas and their management.
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Affiliation(s)
- C S Pramesh
- Division of Thoracic Surgery, Department of Surgical Oncology, Tata Memorial Hospital, Mumbai, India.
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Visioli A, Daniel FJ. Leiomyosarcoma of the oesophagus: a case report and literature review of leiomyosarcoma. AUSTRALASIAN RADIOLOGY 1997; 41:160-5. [PMID: 9153814 DOI: 10.1111/j.1440-1673.1997.tb00704.x] [Citation(s) in RCA: 3] [Impact Index Per Article: 0.1] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 02/04/2023]
Abstract
Leiomyosarcomas are uncommon tumours. Oesophageal leiomyosarcomas are even rarer. A case is presented of this rare tumour which on review of the literature seems to be the first patient surviving 22 years from the original treatment. We include a literature review of the clinical behaviour and management of leiomyosarcoma.
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Affiliation(s)
- A Visioli
- Peter MacCallum Cancer Institute, Melbourne, Australia
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Koga H, Iida M, Suekane H, Aoyagi K, Yao T, Kimura Y, Masuda N, Fujishima M. Rapidly growing esophageal leiomyosarcoma: case report and review of the literature. ABDOMINAL IMAGING 1995; 20:15-9. [PMID: 7894289 DOI: 10.1007/bf00199635] [Citation(s) in RCA: 20] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 01/27/2023]
Abstract
We evaluated a 72-year-old woman who was experiencing dysphagia. Esophageal leiomyosarcoma was diagnosed by barium meal study, upper gastrointestinal endoscopy, endoscopic ultrasonography (EUS), by computed tomography (CT). A barium meal study and esophagoscopy performed 3 months before the diagnosis of esophageal leiomyosarcoma showed no abnormalities. Therefore, the tumor appeared to have grown rapidly during the 3-month period.
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Affiliation(s)
- H Koga
- Second Department of Internal Medicine, Faculty of Medicine, Kyushu University, Fukuoka, Japan
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Rowley H, Viani L, Leen E, Broe P, Keane T. Leiomyosarcoma of the upper oesophagus--a difficult pre-operative diagnosis. Ir J Med Sci 1994; 163:501-2. [PMID: 7806442 DOI: 10.1007/bf02967094] [Citation(s) in RCA: 0] [Impact Index Per Article: 0] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/27/2023]
Abstract
The case of a 62 year old female with a leiomyosarcoma of the upper oesophagus is described. A pharyngolaryngo-oesophagectomy was performed for adequate excision of the tumour. Included is a discussion on the management of leiomyosarcoma of the upper aerodigestive tract.
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Affiliation(s)
- H Rowley
- Department of Otolaryngology, Beaumont Hospital, Dublin
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Moran CA, Suster S, Perino G, Kaneko M, Koss MN. Malignant smooth muscle tumors presenting as mediastinal soft tissue masses. A clinicopathologic study of 10 cases. Cancer 1994; 74:2251-60. [PMID: 7922976 DOI: 10.1002/1097-0142(19941015)74:8<2251::aid-cncr2820740808>3.0.co;2-4] [Citation(s) in RCA: 71] [Impact Index Per Article: 2.3] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/27/2023]
Abstract
BACKGROUND Smooth muscle tumors presenting as mediastinal soft tissue masses are extremely rare and often are mistaken for other neoplastic conditions. METHODS Ten cases of patients with malignant smooth muscle tumors presenting as mediastinal soft tissue masses were studied and correlated with their clinical behavior. Tissues were examined histologically and with immunohistochemical stains in all cases, and by electron microscopy in two cases. RESULTS The patients' ages ranged from 26 to 71 years (mean, 56 years); three were women, and seven were men. Three cases were located in the anterior mediastinum and seven in the posterior mediastinum. The patients with anterosuperior mediastinal tumors all presented with signs and symptoms referable to their lesions; the patients with posterior mediastinal masses (with the exception of one) were all asymptomatic. Grossly, the lesions were well circumscribed and unencapsulated, ranging from 6 to 18 cm in greatest dimension and showed a homogeneous, rubbery cut surface with prominent cystic and myxoid areas. The tumors in all patients appeared to arise from the soft tissues within the mediastinum and were unrelated to adjacent structures. In three patients, the tumors compressed and displaced the esophagus without infiltrating its wall, and in one patient, the tumor was found in close proximity, although unattached, to a large vessel. Histologically, the lesions exhibited a spectrum of morphologic appearances that ranged from low grade leiomyosarcoma with mild-to-moderate nuclear atypia and low mitotic activity (< 3/10 high power fields [HPFs]), to high grade tumors with marked nuclear pleomorphism, extensive areas of necrosis, and high mitotic activity (> 10 mitoses/10 HPFs). One case was characterized by a striking epithelioid morphology with large, round cells arranged in small clusters; another was associated with an incidental microscopic focus of thymic seminoma in the adjacent thymus. Immunohistochemical stains in all cases showed positive labeling of the tumor cells with smooth muscle actin, desmin, and vimentin antibodies. Electron microscopy in two cases showed features of smooth muscle differentiation, i.e., spindle cells surrounded by basal lamina material, immature cell junctions, and abundant intracytoplasmic filaments with focal condensations. All patients were treated with surgical excision. On follow-up, three patients with Stage IIIb and IVa tumors died 2-7 years after surgery, and two patients with Stage Ib and IIb were alive and well 4 and 6 years after surgery, respectively. CONCLUSION Leiomyosarcomas may arise as primary tumors originating from mediastinal soft tissues in both anterior and posterior compartments. Because of their large size and frequent areas of cystic and myxoid degeneration, they may be confused histologically with neural or other neoplasms. As with their counterparts in other soft tissue locations, histologic grade and clinical stage are the most useful parameters for assessing prognosis.
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Affiliation(s)
- C A Moran
- Department of Pulmonary and Mediastinal Pathology, Armed Forces Institute of Pathology, Washington, D.C. 20306-6000
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Aimoto T, Sasajima K, Kyono S, Uchida E, Watanabe M, Yamashita K, Takubo K, Onda M. Leiomyosarcoma of the esophagus: report of a case and preoperative evaluation by CT scan, endoscopic ultrasonography and angiography. GASTROENTEROLOGIA JAPONICA 1992; 27:773-9. [PMID: 1468608 DOI: 10.1007/bf02806531] [Citation(s) in RCA: 8] [Impact Index Per Article: 0.2] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Subscribe] [Scholar Register] [Indexed: 12/27/2022]
Abstract
A case of esophageal leiomyosarcoma, for which CT scan, endoscopic ultrasonography (EUS) and angiography were employed for preoperative diagnosis is reported. CT scan identified an exophytic mass which had no rim-enhancement in the lower end of the esophagus. EUS revealed a homogeneous mass originating from the muscularis propria. Angiography showed a mildly hypervascular tumor. Histologically, the resected specimen was a leiomyosarcoma. These results suggest that the evaluation by EUS combined with CT scan and angiography is useful in differentiating smooth muscle tumors from other submucosal tumors.
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Affiliation(s)
- T Aimoto
- First Department of Surgery, Nippon Medical School, Tokyo, Japan
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Choh JH, Khazei AH, Ihm HJ. Leiomyosarcoma of the esophagus: report of a case and review of the literature. J Surg Oncol 1986; 32:223-6. [PMID: 3736064 DOI: 10.1002/jso.2930320409] [Citation(s) in RCA: 29] [Impact Index Per Article: 0.7] [Reference Citation Analysis] [Abstract] [MESH Headings] [Track Full Text] [Journal Information] [Subscribe] [Scholar Register] [Indexed: 01/07/2023]
Abstract
Leiomyosarcoma of the esophagus is a very rare neoplasm, the total number of reported cases in the English literature being 44 patients. We describe another case of the esophageal leiomyosarcoma to add to the recorded experience of this rare tumor. The therapeutic and prognostic implications of the tumor are discussed, and the related literature is reviewed. In spite of the progress in the thoracic surgical care of patients and the progress in the field of oncology, overall prognosis of patients with leiomyosarcoma of the esophagus remains unchanged. Patients with polypoid lesions and well-differentiated histologic features appear to show better chance of survival. Simple excision of the lesion can be as effective as radical excision, especially in well-localized lesions.
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