Retrograde device-assisted lumen-apposing metal stent insertion for candy cane syndrome

Abstract

Background and Aims

Candy cane syndrome is a rare adverse event of gastric bypass or gastrectomy, where a blind jejunal pouch fills with food, causing dilation and compression of the efferent limb, leading to obstructive symptoms like vomiting and regurgitation. Surgical resection is curative but technically challenging, and endoscopic treatment using lumen-apposing metal stent (LAMS) insertion has been attempted.

Methods

This case describes a retrograde LAMS insertion in a patient with previous total gastrectomy who presented a decade later with dysphagia and food regurgitation. Imaging revealed an enlarged blind jejunal pouch and migration of the esophagojejunostomy anastomosis above the diaphragm and into the thoracic cavity. A tandem endoscopic approach with an ultraslim gastroscope and an echoendoscope was used to place a LAMS from the efferent limb into the blind pouch. The gastroscope is used to assist with instillation of saline and endoscopic visualization of the LAMS in the blind pouch to ensure safe deployment. A retrograde approach from the efferent limb to the blind pouch allows a larger and more stable target for puncture, and a better LAMS axis for effective diversion of food.

Results

He gained 9 kg over 4 months after the procedure and was able to tolerate a normal diet. The LAMS was planned for removal after 10 to 12 months to allow longer indwelling time and greater rate of patency.

Conclusions

Endoscopic management with EUS-guided LAMS insertion is a promising alternative to surgical resection for candy cane syndrome.

Introduction

Candy cane syndrome (CCS) is a type of blind pouch syndrome that may develop after gastric bypass surgery or gastrectomy, often presenting with upper GI symptoms such as nausea, food regurgitation, postprandial vomiting, and abdominal pain.¹ In CCS, the blind jejunal pouch preferentially fills with ingested food and progressively dilates, compressing the efferent jejunal limb. This in turn exacerbates preferential filling and dilatation of the blind pouch, and adverse events such as pouch rupture have been described.² Surgical resection of the dilated blind pouch is curative but may be technically challenging. Endoscopic treatment using antegrade insertion of a lumen-apposing metal stent (LAMS) has been reported in 2 patients, redirecting flow of food from the blind pouch to the efferent limb.^3,4 To our knowledge, we report the first case of a retrograde approach for LAMS insertion for CCS, assisted by a tandem ultraslim gastroscope.

Case report

A 64 year-old man underwent subtotal colectomy and prophylactic total gastrectomy with esophagojejunostomy (EJ) for transverse colon cancer and SMAD4 gene mutation. He then underwent low anterior resection and subsequent proctectomy with ileal pouch anal anastomosis for disease recurrence. He presented 10 years after his initial surgery with progressive solid and liquid dysphagia, as well as regurgitation of ingested food 15 minutes after meals. Upper GI endoscopy showed a moderate amount of food residue at the distal esophagus proximal to the intact and normal-looking EJ anastomosis. The blind jejunal pouch was dilated, and an esophageal diverticulum was seen just proximal to it. The efferent jejunal limb was normal. Cross-sectional imaging showed migration of the EJ anastomosis above the diaphragm into the thoracic cavity (Fig. 1). There was no recurrent locoregional disease or distal metastases. Surgical management was deemed technically challenging because of hostile abdomen from previous abdominal surgeries and possible thoracotomy because of the migration of the EJ anastomosis, and EUS-guided LAMS insertion was proposed as an alternative (Video 1, available online at www.videogie.org).

Figure 1.

Coronal view of a computed tomography scan of the chest showing the esophagojejunostomy anastomosis above the diaphragm (arrow), the dilated blind pouch (asterisk), and the efferent limb with a nasojejunal tube in situ (arrowhead).

Initial endoscopy was performed with an ultraslim gastroscope (GIF-XP180N; Olympus, Tokyo, Japan), which was used to visualize the blind jejunal pouch and the efferent limb. The gastroscope was positioned in the blind pouch, while a linear echoendoscope (GF-UCT180; Olympus) was traversed alongside the gastroscope and into the efferent limb. Fluoroscopic confirmation of their respective positions was performed with intraluminal contrast (Fig. 2A). The gastroscope was used to fill the blind pouch with normal saline up to 27 × 39 cm. Under EUS and fluoroscopic guidance, a 20- × 10-mm LAMS (Hot Axios; Boston Scientific, Marlborough, Mass, USA) was then inserted upward from the efferent limb into the blind pouch with a single puncture using an electrocautery-enhanced delivery system (Figs 2B and C, 3) under a pure cutting current (effect 5, 100 W; Erbe Elektromedizin GmbH, Tübingen, Germany). The gastroscope in the blind pouch provided additional endoscopic confirmation of the LAMS positioning during deployment of the distal flange (Fig. 4). There was good flow of intraluminal contrast seen from the blind pouch toward the efferent limb (Fig. 5)

Figure 2.

Fluoroscopic images of LAMS deployment. A, Gastroscope position was confirmed with intraluminal contrast instilled into the blind pouch. The echoendoscope was alongside a nasojejunal tube in the efferent limb. B, A single puncture was created using an electrocautery-enhanced delivery system from the efferent limb into the blind pouch. C, Deployment of LAMS. LAMS, Lumen-apposing metal stent.

Figure 3.

Endosonographic images of LAMS deployment. A, After saline filling of the blind pouch up to 27 × 39 mm. B, Deployment of LAMS distal flange. LAMS, Lumen-apposing metal stent.

Figure 4.

Endoscopic images of LAMS deployment. A, LAMS distal flange in the blind pouch. B, After deployment of the LAMS, efferent limb (arrow), dilated blind pouch (arrowhead), and esophageal diverticulum (asterisk) were seen. LAMS, Lumen-apposing metal stent.

Figure 5.

Fluoroscopic image after LAMS deployment. The LAMS axis allowed effective downward flow of contrast from the blind pouch to the efferent limb. LAMS, Lumen-apposing metal stent.

The procedure was uncomplicated, and the patient was discharged feeling well after 3 days. On review 1 week after discharge, he was asymptomatic and had gained 3 kg, and an abdominal radiograph showed that the LAMS was stable in position. He gained more weight, weighing from 38 kg to 47 kg (9 kg) over the 4 months since the procedure, and was able to tolerate a normal diet. The LAMS was planned for removal after 10 to 12 months, to allow longer indwelling time and greater rate of patency.

Conclusions

Endoscopic management with EUS-guided LAMS insertion is a promising alternative to surgical resection for CCS. A retrograde approach from the efferent limb to the blind pouch allows a larger and more stable target for puncture and facilitates a better LAMS axis for effective diversion of food. An ultraslim gastroscope is used to assist with instillation of saline and endoscopic visualization of the LAMS in the blind pouch to ensure safe deployment. There are limitations, including the need for a second endoscopy tower and a slightly reduced working space.

Patient Consent

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

Disclosures

C.J.L. Khor has served as a consultant for Boston Scientific, Erbe, and Fujifilm. D.M.Y. Tan has served as a consultant for Boston Scientific, Pentax Medical, and Olympus. All other authors disclosed no financial relationships.

Supplementary data

Video 1

EUS-guided LAMS insertion for candy cane syndrome.