Early oesophageal carcinoma with a defect of the oesophageal muscularis propria: a rare case report

Abstract

We herein report a rare case of early oesophageal cancer combined with an oesophageal muscularis propria defect discovered under endoscopic submucosal dissection. The surgeon did not damage the muscularis propria during endoscopic resection; however, we observed the mediastinal cavity as well as the later occurrence of subcutaneous emphysema. Consequently, the patient was considered to have a muscularis propria defect. This phenomenon has not been reported in the literature to date.

Introduction

The latest definition of early oesophageal cancer is the presence of cancer cells confined to the oesophageal mucosal or submucosal layer without lymph node metastasis. ¹ Endoscopic resection is an approach used to remove such cancerous lesions. The goal of endoscopic submucosal dissection (ESD) is to preserve the integrity of the muscularis propria, enabling resection of early gastrointestinal tumours with minimal changes to the gastrointestinal anatomy. Oesophageal diverticula can be easily detected by endoscopy in patients with muscularis propria defects; however, if these defects do not result in diverticulum formation, then they are difficult to diagnose by endoscopy or other imaging methods. This is the first report of successful removal of early oesophageal cancer in a patient with an oesophageal muscularis propria defect. The patient’s defect was blocked using an oesophageal metal-covered stent.

Case report

A man in his early 70 s presented to the Department of Gastroenterology of Shenzhen Second People’s Hospital to undergo ESD for treatment of high-grade oesophageal intraepithelial neoplasia. Preoperative enhanced computed tomography of the chest indicated no abnormalities in the oesophageal wall and no surrounding lymphatic enlargement. Intraoperative oesophagogastroduodenoscopy (Olympus, Tokyo, Japan) showed a type 0 to IIa mucosal lesion in the oesophagus (approximately 29–38 cm from the incisors). The widest part of the lesion was half of the peripheral diameter of the oesophagus, and the entire lesion was white with a red marginal area (Figure 1(a)). Chromoendoscopy with 1% Lugol’s iodine solution demonstrated a well-demarcated unstained area (Figure 1(b)). A mixture of normal saline, indigo carmine, and sodium hyaluronate was injected into the submucosa. A circumferential incision was made outside the markers with an incision depth to the submucosa (Figure 1(c)). Dissection was performed in the submucosa to avoid damage to the muscularis propria (Figure 1(d)). On the anal side of the lesion, a defect of the muscularis propria was found. The edge of the propria around the defect was neat and smooth, with no cutting damage seen; it was only covered by a white outer membrane (Figure 2(a)). The patient immediately developed subcutaneous cervical emphysema. To avoid adverse events associated with an excessive operation time, such as mediastinal infection, aggravated subcutaneous emphysema, and serious pneumothorax, endoscopic piecemeal mucosal resection was performed. A 12-cm-long oesophageal metal-covered stent was placed to seal the defect (Figure 2(b)). Postoperative pathologic examination suggested oesophageal intramucosal carcinoma, and no muscularis propria component was observed in any of the resected specimens (Figure 2(c)). The oesophageal metal-covered stent was still well fixed after 3 months. Oesophageal mucosal hyperplasia on the oral side of the stent was observed (Figure 3(a)). The metal-covered stent was removed, and the defect in the original muscularis propria healed well (Figure 3(b)).

Figure 1.

Endoscopic presentation of the lesion and the resection procedure. (a) A type 0 to IIa mucosal lesion was present in the oesophagus (approximately 29–38 cm from the incisors). The widest part of the lesion was half of the peripheral diameter of the oesophagus, and the entire lesion was white with a red marginal area. (b) Chromoendoscopy with 1% Lugol’s iodine solution demonstrated a well-demarcated unstained area. (c) A circumferential incision was performed outside the markers with the incision depth to the submucosa and (d) Dissection was performed in the submucosa to avoid damage to the muscularis propria.

Figure 2.

Endoscopic treatment for defect of the muscularis propria and postoperative pathology. (a) On the anal side of the lesion, a defect of the muscularis propria was found. The edge of the propria around the defect was neat and smooth, with no cutting damage seen; only a white outer membrane covered the defect. (b) A 12-cm-long oesophageal metal-covered stent was placed to seal the defect and (c) No muscularis propria component was observed in any of the resected specimens.

Figure 3.

Postoperative status. (a) The oesophageal metal-covered stent was still well fixed after 3 months. Oesophageal mucosal hyperplasia was observed on the oral side of the stent and (b) The metal-covered stent was removed, and the defect in the original muscularis propria healed well (green arrow).

Publication of this report was approved by the Clinical Research Ethics Committee of Shenzhen Second People’s Hospital (2023-204-01PJ). The reporting of this study conforms to the CARE guidelines (for case reports). ²

Discussion

Muscularis propria defects in the digestive tract are clinically rare and may be caused by both congenital and acquired factors. Congenital factors include developmental disorders, whereas acquired defects may be caused by local selective ischaemia.^3–5 Many reports have described muscularis propria defects in the stomach and intestines, with a high incidence among infants and young children. ⁶ However, reports of oesophageal muscularis propria defects are quite rare.

A digestive tract diverticulum is a common manifestation of the absence of the muscularis propria. Absence of the muscularis propria increases the pressure in the gastrointestinal tract, causing the mucosal layer to be squeezed outside the lumen and resulting in diverticular-like depressions after long-term extrusion. Reports of oesophageal diverticula in patients with oesophageal cancer are not uncommon. Fu et al. ⁷ reported a case of superficial oesophageal cancer in an oesophageal diverticulum. The oesophageal cancer was removed by ESD without intraoperative or postoperative perforation, and there was no recurrence or metastasis of the oesophageal cancer after 42 months of follow-up. ⁷ The success of the surgery in such cases is due to the preoperative design. With an understanding of local anatomical changes, the design of the operation is more appropriate, and the operation is conducted with greater caution. By contrast, in the present case, we did not know that a defect was present in the muscularis propria below the lesion. Thus, a preoperative operational design could not be applied. Sudden intraoperative defects can also introduce unexpected challenges for the operator or even make it difficult to accurately determine if the sudden change in the patient’s condition is due to the defect or an operation-induced perforation. Therefore, when faced with this problem in the present case, we took intraoperative emergency measures; that is, we asked two other senior chief physicians to examine the defect together and then determined the diagnosis and treatment plan according to the specific situation. For doctors with limited experience or hospitals with limited medical resources, this problem could have disastrous consequences.

Oesophageal metal-covered stent placement has been shown to be an effective method for closure of perforations associated with oesophageal cancer. ⁸ This is the first report to describe successful application of the oesophageal metal-covered stent technique for the occlusion of an oesophageal muscularis propria defect. The defect was successfully closed after removal of the stent, suggesting that an oesophageal metal-covered stent is an effective therapy for oesophageal muscularis propria defects.

In conclusion, we have presented a case of early oesophageal cancer with a defect of the oesophageal muscularis propria. The cancer was successfully treated by performing ESD combined with endoscopic piecemeal mucosal resection, and we blocked the muscularis propria defect with an oesophageal metal-covered stent. Serious complications were thus prevented, and a good long-term prognosis was achieved.