Pyloric gland adenoma in the duodenal bulb: case report of a giant laterally spreading tumor treated with endoscopic submucosal dissection

A 55-year-old man underwent gastroduodenoscopy because of epigastric pain. Abdominal computed tomography revealed a uniformly enhanced mass (10 × 8 mm) within the duodenum, with no enlargement of lymph nodes ([Fig. 1]). A laterally spreading tumor, with a diameter of about 60 mm, was detected within the duodenal bulb ([Fig. 2]). The lesion involved the pylorus ring and had a positive lifting sign. Considering the difficulty of endoscopic resection of such a giant lesion in the duodenal bulb, surgical resection was proposed, but the patient refused. Therefore, a standard endoscopic submucosal dissection was performed ([Video 1]).

Fig. 1 Abdominal computed tomography revealed a uniformly enhanced mass within the duodenum, without enlargement of lymph nodes.

Fig. 2 A laterally spreading tumor in the duodenal bulb.

Video 1 Endoscopic submucosal dissection of a large laterally spreading pyloric gland adenoma in the duodenal bulb.

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Grossly, the resected tissue measured 60 × 50 mm ([Fig. 3]). The pathological examination revealed pyloric gland adenoma accompanied by some regional high grade intraepithelial neoplasia ([Fig. 4]). The lateral and vertical margins of the specimen were negative. No complications occurred during the procedure.

Fig. 3 The resected tumor was 60 × 50 mm in diameter.

Fig. 4 Histological examination revealed closely packed pyloric gland-type glands made up of cuboidal to columnar epithelial cells with pale to eosinophilic cytoplasm (hematoxylin and eosin, × 200).

A repeat gastroduodenoscopy about 1 year later showed no significant abnormalities in the duodenal bulb ([Fig. 5]).

Fig. 5 Repeat gastroduodenoscopy 1 year later showed no significant abnormalities in the duodenal bulb.

Pyloric gland adenoma (PGA) is a rare neoplasm, composed of tightly packed tubules (occasionally cystic dilation) with pyloric gland differentiation, which mainly occurs in the stomach [1]. Since the first description of PGA by Elster in 1976, few PGAs have been documented to originate from the duodenum [2] [3] and other extragastric sites; in addition, most reported PGAs have been < 25 mm [4]. Nowadays, PGA is a recognized precancerous disease, with a reported rate of association with adenocarcinoma ranging from 12 % to 47 % [5]. The risk of developing adenocarcinoma is associated with its size and the presence of high grade dysplasia [4]. Therefore, endoscopic removal of PGA is indicated. In our report, a rare giant duodenal PGA was described and successfully treated with endoscopic submucosal dissection.

Endoscopy_UCTN_Code_CCL_1AB_2AZ_3AB

^* These authors contributed equally to this work.

• References

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