Endoscopic submucosal dissection of squamous cell carcinoma accompanied by adenoid cystic carcinoma of the esophagus

Adenoid cystic carcinoma of the esophagus is a rare tumor and difficult to detect early [1] [2]. Esophageal squamous cell carcinoma (ESCC) has a characteristic magnifying endoscopic appearance [3]. However, adenoid cystic carcinoma of the esophagus can have atypical endoscopic features.

An 84-year-old man underwent esophagogastroduodenoscopy (EGD) owing to retrosternal discomfort. EGD showed a flat (0-IIb) and slightly reddish lesion of 15 × 20 mm in the middle esophagus ([Fig. 1 a]). The lesion appeared as a brownish area on narrow-band imaging (NBI) endoscopy ([Fig. 1 b, c]). Endoscopic ultrasound (EUS) revealed the lesion primarily involved the mucosal layer of the esophagus with a hypoechoic area ([Fig. 1 d]). NBI magnification revealed that the intrapapillary capillary loop pattern appeared to be type B1 ([Fig. 2], [Video 1]) based on the magnifying endoscopic classification of the Japan Esophageal Society. But near the anal area of this 2 × 2-mm lesion, the loop pattern appeared irregular and of the fine reticular (R) type ([Fig. 2 b]) (red arrow). The endoscopic diagnosis was ESCC and the depth was mainly T1a-EP or T1a-LPM. Biopsy pathology suggested a high grade intraepithelial neoplasia (HGIN).

Fig. 1 Preoperative endoscopy.

Fig. 2 Narrow-band imaging with magnification.

Video 1 Endoscopic features and endoscopic submucosal dissection of squamous cell carcinoma accompanied by adenoid cystic carcinoma of the esophagus.

This patient was eligible for endoscopic therapy. Therefore, an en bloc resection was performed by endoscopic submucosal dissection (ESD) ([Fig. 3 a–f]). From the second to eighth tissue strips, hematoxylin and eosin (H&E) stain showed HGIN with focally invasive SCC in the lamina propria ([Fig. 3 g]) (blue circle). In the sixth strip, there are epithelioid cells arranged in a cribriform, tubular and solid architecture, which have no relation to the surface squamous epithelium and are restricted to the lamina propria ([Fig. 4 a–c]). Immunohistochemical analysis showed that these abnormally arranged epithelioid cells were positive for P40, SOX-10 and CD117, which was diagnosed as adenoid cystic carcinoma ([Fig. 4 d, e, f]). The pathological diagnosis was: (1) ESCC,0-IIb, pT1a(LPM), ly(-), v(-), HM0, VM0, pR0, 14 × 16 mm (in 27 × 35 mm); (2) EACC,0-IIb, pT1a(MM), ly(-), v(-), HM0, VM0, pR0, 2 × 2 mm (in 27 × 35 mm) ([Fig. 5]). Endoscopic control at 6 months showed the presence of a regular scar with no signs of residual disease or recurrence. Adenoid cystic carcinoma of the esophagus lacked a typical magnifying endoscopic appearance. R-type vessels can be atypical magnifying endoscopic features that help us to detect lesions early [4] [5].

Fig. 3 Intraoperative endoscopy, postoperative specimen and hematoxylin and eosin (H&E) stain.

Fig. 4 H&E stain and immunostaining of the 6th tissue strips in resected specimen.

Fig. 5 The specimen (Lugol stain) showing invasion depth of cancer on serial section (color code).

Endoscopy_UCTN_Code_TTT_1AO_2AG

Competing interests

The authors declare that they have no conflict of interest.

• References

• 1 Morisaki Y, Yoshizumi Y, Hiroyasu S. et al. Adenoid cystic carcinoma of the esophagus: report of a case and review of the Japanese literature. Surg Today 1996; 26: 1006-1009
• 2 Gregg JB, Stamler FW. Unusual neoplasms of the esophagus: review of literature and report of a case. AMA Arch Otolaryngol 1954; 59: 159-169
• 3 Kumagai Y, Inoue H, Nagai K. et al. Magnifying endoscopy, stereoscopic microscopy, and the microvascular architecture of superficial esophageal carcinoma. Endoscopy 2002; 34: 369-375
• 4 Yoshikawa K, Kinoshita A, Hirose Y. et al. Endoscopic submucosal dissection in a patient with esophageal adenoid cystic carcinoma. World J Gastroenterol 2017; 23: 8097-8103
• 5 Miwako A, Mika M, Takako Y. et al. Classification of the Japan Esophageal Society for magnifying endoscopy and the depth of tumor invasion-type r vessels and histopathological features. Stomach and Intestine 2014; 49: 213-221

Publication History

Article published online:
22 September 2022