Endoscopic submucosal dissection in the management of bleeding duodenal tumors refractory to conventional therapy

Abstract

Background and Aims

Although isolated hamartomas are rare, they can cause bleeding that is refractory to medical treatment. Management strategies depend on factors such as tumor location, size, and bleeding intensity.

Methods

A 94-year-old woman experienced melena and a significant decrease in hemoglobin from 13 g/dL to 7 g/dL. An EGD revealed a large, bleeding pedunculated tumor in the duodenum. Previous interventions, including epinephrine injection and EMR, failed, necessitating multiple blood transfusions. After multidisciplinary consultation, endoscopic submucosal dissection was undertaken.

Results

En bloc resection of the duodenal lesion was performed successfully. The specimen was segmented and removed without causing any trauma to the duodenal wall.

Conclusions

Hamartomas in the upper GI tract, particularly in the stomach or duodenum, pose a greater bleeding risk. Endoscopic submucosal dissection effectively removes large duodenal tumors, especially when EMR is unfeasible. Segmenting the specimen during retrieval minimizes trauma to surrounding tissue, preserving duodenal integrity.

Background

GI hamartomas are benign tumors that can occur in both the upper and lower GI tract. Most hamartomas are linked to syndromes, such as Peutz-Jeghers syndrome, whereas isolated hamartomas are quite rare.¹ GI hamartomas may cause bleeding, which can be refractory to medical treatment.² The management of bleeding from hamartomas depends on various factors, including their location, size, and the rate of bleeding.

Case presentation

A 94-year-old woman presented with melena and a hemoglobin level that decreased to 7 g/dL, down from a baseline of 13 g/dL. On computed tomography scan, we noted an intramural gastric antral mass extending into the proximal duodenum. EGD revealed a large, bleeding pedunculated tumor in the duodenum. Attempts to manage the bleeding with epinephrine injection and hemostatic powder were unsuccessful. An EMR attempt also failed because the snare was unable to be placed around the lesion as the result of the tumor's size and location. The patient remained hospitalized because of recurrent bleeding, which necessitated multiple blood transfusions. After multidisciplinary discussions, the decision was made to proceed with endoscopic submucosal dissection (ESD) (Fig. 1, Video 1, available online at www.videogie.org).

Figure 1.

Large lesion with stigmata of recent bleeding in the duodenal bulb.

A submucosal injection was performed into the stalk of the lesion using 6% hydroxyethyl starch. A 1.5-mm needle-type ESD knife was used to initiate the mucosal incision (Fig. 2). The water-pressure method was used to facilitate entry into the submucosal layer. The water-pressure method gently exerts pressure on the mucosal flap, keeping it lifted, and maintaining a cushion. This provides a clear space for safely dissecting the submucosal layer.

Figure 2.

Mucosal incision of the lesion stalk.

A feeding artery was identified and cauterized with hemostatic forceps (Fig. 3). To prevent inadvertent injury to the duodenal wall behind the stalk, a hook-shaped ESD knife was used to continue the dissection (Fig. 4). The hook-knife prevented distal wall injury by lifting the tissue (away from the duodenal wall) before cutting. This offered better control of the depth of the cut while minimizing the risk of muscle injury. Traction techniques were applied to prevent tissue migration.

Figure 3.

Use of coagulation forceps for preemptive hemostasis.

Figure 4.

Use of a J-shaped knife to transect remainder of the stalk.

To facilitate retrieval, the specimen was segmented (Fig. 5). A pure cutting current (high cut, effect 4, 180 W) was used to transect the specimen. The specimen was then brought into the stomach and retrieved using a retrieval net. The mucosal defect was only visible under retroflexed view in the duodenal bulb (Fig. 6). Finally, the defect was closed using a hemostatic clip. On pathologic assessment, we noted glands with morphologic features somewhat distinct from adjacent normal Brunner’s glands favoring a hamartomaous lesion. No signs of dysplasia or deep invasion were found.

Figure 5.

Lesion segmentation to facilitate safe removal of the lesion.

Figure 6.

Mucosal defect after dissection of the duodenal lesion.

Conclusions

Hamartomas in the upper GI tract, particularly in the stomach or duodenum, are more likely to cause bleeding than those in the lower GI tract.² The overall risk of bleeding depends on factors such as the size, number, and location of the lesions. Large tumors in the duodenum can be removed using ESD, which offers an effective alternative when EMR is not feasible because of the tumor's size. In contrast, if the tumor is removed using an en bloc resection, it can be challenging to extract it without causing deep trauma to the surrounding tissue. By segmenting the specimen, the risk of deep trauma during retrieval is minimized, preserving the integrity of the duodenal wall.

Pantaleón Sánchez et al³ reported the successful treatment of a duodenal bulb hamartoma using ESD. Similarly, Abiko et al⁴ achieved success with ESD for a hamartoma located along the greater curvature of the stomach. In their approach, the authors used a clip with a line attachment before incision to stabilize the lesion. This area, characterized by a thicker gastric wall and greater accessibility, may facilitate a relatively straightforward dissection compared with the duodenum.

Both Pantaleón Sánchez et al and Abiko et al successfully performed hamartoma resections, achieving complete removal without any adverse events. Ohba et al⁵ introduced a modified ESD technique that uses a snare and grasping forceps, although specific details of this modification were not provided. Although the aforementioned cases illustrate successful ESD, it is crucial to recognize that these procedures are conducted by highly experienced endoscopists. Given the considerable risk of adverse events, particularly perforation, duodenal ESD should be attempted only by experts in the field.^6,7

In conclusion, ESD offers an effective therapeutic option for large pedunculated lesions, particularly when EMR is not feasible because of the tumor's size and location. Specimen segmentation should be considered to facilitate specimen retrieval and avoid deep trauma.

Patient Consent

The patient in this article has given written informed consent to publication of the case details.

Disclosures

C. C. Thompson has served as a consultant for Apollo Endosurgery, Boston Scientific, Endoquest Robotics, Enterasense, EnVision Endoscopy, Fujifilm, Fractyl, USGI Medical, Medtronic/Covidien, Olympus/Spiration, GI Dynamics, and Xenter; has served as an advisory board member for USGI Medical and Fractyl; has received research grant and support from USGI Medical, Apollo Endosurgery, Boston Scientific, Endoquest Robotics, ERBE, Fujifilm, Lumendi Olympus/Spiration, Aspire Bariatrics, and GI Dynamics; has served as a general partner for Blueframe Healthcare; has served as a founder of Enterasense, EnVision Endoscopy, and GI Windows; and holds stock and royalties for GI Windows. M. Ryou has served as a consultant for Olympus America, Fujifilm Medical Systems, Boston Scientific, ConMed, MicroTech, GI Windows Surgical, and EnteroSence. H. Aihara has served as a consultant for Olympus America, Fujifilm Medical Systems, Boston Scientific, ConMed, MicroTech, BioDevek, Johnson & Johnson MedTech, and Medtronic and Advisory Board Member for BioDevek and EndoQuest Robotics. All other authors disclosed no financial relationships.

Supplementary data

Video 1

Endoscopic submucosal dissection used in the management of bleeding duodenal hamartoma.