Case Report
Copyright ©The Author(s) 2020. Published by Baishideng Publishing Group Inc. All rights reserved.
World J Gastroenterol. May 7, 2020; 26(17): 2111-2118
Published online May 7, 2020. doi: 10.3748/wjg.v26.i17.2111
Multiple carcinosarcomas of the esophagus with adeno-carcinomatous components: A case report
Hiroshi Okamoto, Hiroshi Kikuchi, Hiroshi Naganuma, Takashi Kamei
Hiroshi Okamoto, Takashi Kamei, Department of Gastroenterological Surgery, Graduate School of Medicine, Tohoku University, Sendai 980-8574, Japan
Hiroshi Kikuchi, Department of Surgery, South Miyagi Medical Center, Miyagi 989-1253, Japan
Hiroshi Naganuma, Department of Pathology, Sendai City Hospital, Sendai 982-8502, Japan
Author contributions: Okamoto H is the main author of this article; Kikuchi H was the patient’s surgeon and prepared the manuscript; Naganuma H performed the pathological examination; Kamei T reviewed the manuscript; all authors have read and approved the final manuscript.
Informed consent statement: The patient provided informed written consent for all aspects of care described in this manuscript.
Conflict-of-interest statement: The authors have no conflicts to declare.
CARE Checklist (2016) statement: The authors have read the CARE Checklist (2016), and the manuscript was prepared and revised according to the CARE Checklist (2016).
Open-Access: This article is an open-access article that was selected by an in-house editor and fully peer-reviewed by external reviewers. It is distributed in accordance with the Creative Commons Attribution NonCommercial (CC BY-NC 4.0) license, which permits others to distribute, remix, adapt, build upon this work non-commercially, and license their derivative works on different terms, provided the original work is properly cited and the use is non-commercial. See: http://creativecommons.org/licenses/by-nc/4.0/
Corresponding author: Hiroshi Okamoto, MD, PhD, Assistant Professor, Doctor, Surgeon, Department of Gastroenterological Surgery, Graduate School of Medicine, Tohoku University, 1-1, Seiryo-machi, Aoba-ku, Sendai 980-8574, Japan. hi-ok-0531@med.tohoku.ac.jp
Received: February 29, 2020
Peer-review started: February 29, 2020
First decision: April 2, 2020
Revised: April 8, 2020
Accepted: April 28, 2020
Article in press: April 28, 2020
Published online: May 7, 2020
Processing time: 67 Days and 23.1 Hours
Abstract
BACKGROUND

Carcinosarcoma (spindle cell carcinoma) of the esophagus is an extremely rare event; the etiology and origins of this neoplasm have not yet been determined. Epithelial-mesenchymal transition (EMT) has been associated with invasion and metastasis, and may be related to the generation of a stem cell population within this tumor.

CASE SUMMARY

We present the case of a 61-year-old male with nausea and fever. Upper gastrointestinal endoscopy revealed the presence of type 1 and 0-IIc lesions located 35 cm from the incisors toward the esophago-gastric junction. Thoracoscopic esophagectomy was performed. Macroscopic analysis revealed three polypoid lesions in the abdominal esophagus that accompanied the main lesion in the lower thoracic esophagus and 0-IIc lesions that spread continuously with them. Histologically, the lesions included proliferating spindle cells. Adeno-carcinomatous components were detected in a section near the foot, and squamous cell carcinoma was identified in the mucosa at the base of the tumor. The patient was diagnosed with multiple carcinosarcomas, staged at pT1b (SM3), pN1 (#110, #7), cM0, Stage II (sarcomatous metastasis to the lymph nodes). Spindle cells did not express E-cadherin but were positive for EMT markers, including zinc finger E-box-binding homeobox 1, TWIST, and snail family transcriptional repressor 2. The patient has experienced no recurrence at 5 years and 2 mo after surgery.

CONCLUSION

This report suggests that multiple sarcomatous tumors may be generated from primary squamous cell carcinoma via mechanisms related to EMT.

Keywords: Esophagus; Multiple carcinosarcomas; Multiple spindle cell carcinomas; Epithelial-mesenchymal transition; Epithelial-mesenchymal transition markers; Case report

Core tip: Epithelial-mesenchymal transition (EMT) has been associated with cancer invasion and metastasis as epithelial cells acquire the ability to migrate and invade; these observations have been related to the generation of a stem cell population within the neoplasm. We report here a resected case of multiple carcinosarcomas with adeno-carcinomatous components that may have developed secondary to EMT. Detailed elucidation of the relationship between the pathogenesis of carcinosarcoma and EMT are among the future challenges in this field.