Circumferential endoscopic submucosal dissection of a 14-cm long-segment Barrett’s esophagus with multifocal adenocarcinoma: a case report

Abstract

Background and Aims

Barrett's esophagus (BE) is a recognized precursor to esophageal adenocarcinoma (EAC), with an annual progression risk of up to 7% in cases involving high-grade dysplasia (HGD). Endoscopic therapy is the standard treatment for dysplastic BE and early-stage EAC, typically involving ablation techniques, such as radiofrequency ablation and cryotherapy, for flat BE and endoscopic resection methods, including EMR and more recently endoscopic submucosal dissection (ESD), for nodular lesions.

Methods

This article describes the case of a patient with an ultralong segment of BE (14 cm) and multifocal EAC who was successfully treated with circumferential ESD.

Results

A complete en bloc resection of a 16-cm segment was achieved. Histopathologic analysis confirmed the presence of BE with HGD and multifocal EAC (T1b). Although the patient developed an esophageal stricture postprocedure, it was effectively managed endoscopically.

Conclusions

The patient is currently doing well and remains under routine endoscopic and radiographic surveillance. Circumferential ESD represents a safe and effective approach for the endoscopic resection of extensive BE and early EAC and should be considered a viable alternative to surgical intervention.

Background

Barrett’s esophagus (BE) is the only known precursor to esophageal adenocarcinoma (EAC), with annual progression rates of 0.33% to 7%, depending on dysplasia grade.^1,2 Long-segment BE (LSBE) (≥3 cm) carries a higher risk than short-segment BE (<3 cm).^3,4 Dysplastic BE and early EAC are often treatable endoscopically. To our knowledge, this article presents the longest-documented BE segment removed via endoscopic submucosal dissection (ESD), underscoring its clinical importance.

Case

A 72-year-old man presented with nonsteroidal anti-inflammatory drug–induced peptic ulcer disease that was managed with proton pump inhibitor. EGD revealed LSBE. Biopsies (4 quadrants every 2 cm) confirmed high-grade dysplasia (HGD) at 25 and 27 cm and “at least” intramucosal EAC at 29, 31, 33, and 35 cm. A CT/positron emission tomography only showed increased metabolic activity in the distal esophagus.

At our institution, EGD confirmed LSBE (C14M14) with a 1-mm nodule at 25 cm (Fig. 1). Although not a reliable tool to distinguish T1a from T1b, EUS (Fig. 1) detected a T1a disease and identified a small (6 mm) benign-appearing paraesophageal isoechoic lymph node (at 27 cm, positron emission tomography nonavid) that was not biopsied because of the risk of seeding. After multidisciplinary discussion in the tumor board, ESD was recommended over esophagectomy or EMR and radiofrequency ablation. The potential intraprocedure and postprocedure adverse events (eg, long stricture) were discussed with the patient, who elected to proceed with ESD.

Figure 1.

A, Long-segment Barrett’s esophagus with small nodule (red arrow). B, Sonographic evaluation of esophageal adenocarcinoma showing no definite mass or concerning lymph nodes.

Procedure

The patient was supine, with the endoscopist positioned at the head. BE was marked proximally and distally using a hybrid knife. Following submucosal injection of a methylene blue–saline solution, circumferential incision was performed distally (1 cm distal to BE). Three straight submucosal tunnels (at 2, 10, and 6 o'clock) were created proximally until the gastric lumen was observed distally. The first tunnel was performed at 6 o'clock, as it is the most difficult tunnel because of pooling of fluids. The tunnels were connected, fully detaching the distal specimen from the esophageal wall. Intact mucosa was left in between the proximal incisions to maintain traction (Fig. 2, Video 1, available online at www.videogie.org). Those areas were incised at the end of resection before the specimen was retrieved using a rat-tooth forceps (Fig. 2). Few superficial muscle injuries were observed (Sydney classification type III) without perforation. The final length of the specimen was 16 cm (incision was 1 cm proximal and distal to BE). An intermittent pneumatic compression device was used to reduce deep vein thrombosis risk.

Figure 2.

A, Three incisions and tunnels were performed proximally. B, Submucosal dissection and connecting submucosal tunnels. C, Circumferential defect after endoscopic submucosal dissection. D, Esophageal circumferential specimen after completing the procedure (measuring approximately 16 cm).

To minimize stricture risk, triamcinolone was injected into the cautery ulcer base. Two fully covered stents (19 × 100 mm and 23 × 150 mm) were placed with and secured using 3 MANTIS clips (Boston Scientific, Marlborough, Mass, USA). The procedure, completed in 10 hours and 33 minutes, was technically successful. Pathology confirmed multifocal well- to moderately differentiated adenocarcinoma (pT1b, SM1) in a background of HGD, with no lymphovascular invasion (Fig. 3). All lateral and deep margins were negative. Figure 4 is showing the important events during this patient's care (Fig. 4).

Figure 3.

A, High-grade dysplasia (HGD) at the proximal end of the specimen (H&E, orig. mag. × 10). B, HGD at the distal end of the specimen (H&E, orig. mag. × 10). C, Adenocarcinoma with invasion into the submucosa (H&E, orig. mag. × 20).

Figure 4.

A timeline figure showing the important events during this patient care and time lapses between those events.

Outcome

Next day, the patient was discharged on levofloxacin and proton pump inhibitor. A week later, he was readmitted with chest pain; imaging revealed stents in place, and he was treated symptomatically. One month later, he returned with stent intolerance. Difficult extraction caused trauma and a small esophageal leak, necessitating placement of a new stent, treatment with piperacillin-tazobactam (Zosyn), fluconazole, and total parenteral nutrition. Managed conservatively, he was discharged 8 days later with amoxicillin–clavulanic acid (Augmentin) and fluconazole. Four weeks later, EGD was performed to retrieve the stent, and an esophagram showed no residual leak. A long esophageal stricture developed and was successfully treated with 5 biweekly dilations, triamcinolone injections (1 mL/4 quadrants every 1 cm), and a 4-week course of prednisolone (30 mg). Despite the resolution of the stricture, the patient had poor oral intake and lost 13 kg. Mild depression was diagnosed, and mirtazapine (15 mg daily) was initiated to address both poor appetite and depression.

The patient is currently doing well, without dysphagia and a 14-kg weight gain, returning to baseline. He undergoes endoscopic surveillance with biopsies (4 quadrants every 1 cm) every 3 months, with no signs of recurrence. Six-month EUS showed no lymphadenopathy and a stable paraesophageal lymph node. At 1-year surveillance, CT was negative for recurrence. Over 18 months, he has had 11 EGDs in total.

Conclusion

LSBE with early EAC can be effectively managed with circumferential ESD after multidisciplinary approach. Although circumferential ESD has been previously reported, this case represents the longest resection in humans documented in the literature, to our knowledge.^5,6

Prophylactic stent placement may reduce, but not eliminate, stricture risk. Based on observation, smaller stents may promote healing, whereas larger ones can delay healing because of localized ischemia. Oral steroids can help reduce stricture formation. Proper patient selection and post-ESD management are crucial for optimal outcomes.

Patient Consent

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

Disclosure

All authors disclosed no financial relationships.

Supplementary data

Video 1

A video showing the procedure and postoperative course in details.