Cervical squamous cell carcinoma coexisting with ectopic gastric mucosa treated by endoscopic submucosal dissection: A case report.

A 69-year-old Japanese man underwent an endoscopic submucosal dissection (ESD) for early gastric cancer 2 years prior to admission at Fukuchiyama City Hospital. A follow-up esophagogastroduodenoscopy (EGD) revealed a butterfly-shaped brownish area in the cervical esophagus. A successful endoscopic submucosal dissection of the esophageal lesion was performed for total biopsy. Histopathological analysis of the resected specimen revealed moderately differentiated squamous cell carcinoma of the esophagus, cervical esophagus tumor type 0-IIb, a tumor diameter of 14x8 mm, T1b pathological findings (invading into submucosa), infiltrative growth α, lymphatic invasion (-), venous invasion (-), a horizontal tumor free margin and a vertical tumor free margin. The results demonstrated that the squamous cell carcinoma had invaded into the submucosal layer under the ectopic gastric mucosa. After endoscopic treatment, chemoradiation therapy was administered to the patient. Copyright: © Tsuji et al.

Introduction

Esophageal cancer is the sixth common cause of cancer associated deaths worldwide (1). It is often difficult to notice esophageal cancer using conventional esophagogastroduodenoscopy (EGD) with white-light imaging (WLI). Many clinical studies have reported on the diagnostic performance by image-enhanced endoscopy with narrow-band imaging (NBI) (2,3). Moreover, endoscopic surveillance of Barrett's esophagus has become a foundation of the management of esophageal adenocarcinoma, and this trend has accelerated with recent developments in advanced endoscopic imaging and treatment technologies (4).

Endoscopic submucosal dissection is a lower invasive therapy for superficial esophageal cancers. However, esophageal treatment requires more skill than gastric resection because of the technical difficulties related with the narrow location and thin wall in esophagus usually. It is more difficult to distinguish between ectopic gastric mucosa and early cancer, it rarely reported that the case for patient to undergo magnifying endoscopy with NBI and endoscopic submucosal dissection (ESD) for an esophageal adenocarcinoma in the upper thoracic esophagus (5). Therefore, we have reported a first case of squamous cell carcinoma coexisting with ectopic gastric mucosa treated by endoscopic submucosal dissection. I confirmed that the patient provided informed consent for the use of this data with a consent form.

Case report

A 69-year-old Japanese man underwent an ESD for early gastric cancer 2 years prior to his admission. A follow-up EGD with NBI system showed a butterfly-shaped brownish area (Fig. 1) in the cervical esophagus. The patient's history included hypertension and chronic kidney disease, and his family history included a brother with gastric and colonic cancer. The patient had smoked for 20 years beginning when he was 20 years old.

Figure 1

Esophagogastroduodenoscopy with a narrow-band imaging system revealed a 15 mm butterfly-shaped brownish area at the first endoscopic examination.

NBI magnifying endoscopy, revealed that the patient's butterfly-shaped brownish area had type B2 vessels (per the Japanese esophageal Society expansion endoscopic classification (6) in 2/3 of its area (Fig. 2A and B). In addition, 1/3 of this area showed a gastric mucosal pattern (Fig. 2C).

Figure 2

Endoscopic images of esophageal carcinoma. (A) Narrow-band imaging revealed an irregular intra-papillary capillary loop in esophageal mucosa and an irregular microsurface pattern and microvascular pattern in gastric mucosa. (B) Magnifying endoscopy revealed type B2 vessels in 2/3 of this lesion. (C) One-third of this area exhibited a gastric mucosal pattern. (D) Endoscopic image of the unstained lesion after spraying with Lugol's solution.

The endoscopic image of the same lesion after spraying with Lugol's solution showed that the lesion remained unstained (Fig. 2D). A squamous cell carcinoma was diagnosed based on the biopsy from the lesion, which revealed type B1-2 vessels.

Because of a localization in cervical esophagus, the surgical operation method will be Pharyngolaryngoesophagectomy. Considering that endoscopic treatment will be more beneficial for patient, we performed an endoscopic submucosal dissection (ESD) of the esophageal lesion for the purpose of doing a total biopsy. We completed that endoscopic surgery as planned (Fig. 3A-C). The histopathological analysis of the resected specimen revealed squamous cell carcinoma (moderately differentiated) of the esophagus, Ce, 0- IIb, 14x8 mm, pT1b(SM1), INFα, ly0(D2-40), v0(EVG), pHM0, pVM0 (Fig. 4A-D). The squamous cell carcinoma invaded into the submucosal layer under the ectopic gastric mucosa.

Figure 3

Macroscopic findings. (A) Macroscopic appearance of the resected specimen exhibiting the esophageal cancer as a red lesion. (B) Squamous cell carcinoma unstained in the specimen after spraying with Lugol's solution. (C) The red line demonstrates squamous cell carcinoma of the mucosal layer. The blue line marks the squamous cell carcinoma of the submucosal layer. The green line indicates the ectopic gastric mucosa.

Figure 4

Histological examination of the endoscopically resected specimen. (A) Microscopy revealed that the esophageal lesion was located in the submucosa layer (H&E staining; magnification, x20). (B) High-magnification image demonstrating tumor invasion into the ubmucosal layer under the ectopic gastric mucosa (H&E staining; magnification, x100). (C) Esophageal carcinoma invaded deeply into the submucosal layer (H&E staining; magnification, x20). (D) H&E staining revealed massive infiltration of esophagus cancer cells (magnification, x100). H&E, hematoxylin and eosin.

Discussion

We performed an endoscopic submucosal dissection of the cervical esophageal lesion for a total biopsy as planned. The reason why I determined that ESD would be more benefical is a location of esophageal cancer. Because of a localization of cervical esophagus, the operation will be pharyngolaryngoesophagectomy. Needless to say, blood cancer biomarker of squamous cell cancer was normal. Metastatic lesion was not detected in all body CT scan.

The reported frequency of ectopic gastric mucosa of the upper esophagus is 11% (7). Approximately 30 case reports of adenocarcinoma occurring from ectopic gastric mucosa of the esophagus have been reported since 1950(8), but our search of the literature revealed no reported case of squamous cell carcinoma neighboring and invading ectopic gastric mucosa. As another rare case, there are a report to undergo ESD for esophageal adenocarcinoma with enteroblastic differentiation arising from ectopic gastric mucosa in the esophagus (9). In our case, the squamous cell carcinoma invaded into the submucosal layer under the ectopic gastric mucosa. From now, it is necessary for us to consider how to be invaded into submucosal layer pathologically. We have thus apparently reported the first case of a patient with squamous cell carcinoma coexisting with ectopic gastric mucosa treated by endoscopic submucosal dissection.