Cardiovasc Intervent Radiol DOI 10.1007/s00270-015-1147-7

CASE REPORT

Closure of Nonmalignant Tracheoesophageal Fistula Using an Atrial Septal Defect Occluder: Case Report and Review of the Literature Ping Jiang1 • Ji Liu2 • Dong Yu3 • Bing Jie3 • Sen Jiang3

Received: 15 April 2015 / Accepted: 20 May 2015  Springer Science+Business Media New York and the Cardiovascular and Interventional Radiological Society of Europe (CIRSE) 2015

Abstract Tracheoesophageal fistula (TEF) is a lifethreatening condition for which there are several management techniques. We present a case of nonmalignant TEF closure using an atrial septal defect (ASD) occluder. A 53-year-old man with a severe TEF was admitted to our hospital for TEF caused by stenting of an esophagogastric anastomotic stricture. He was successfully treated with closure of the TEF using an endotracheal ASD occluder. Three hundred and eighteen days after placement of the occluder, he suddenly developed a severe cough after dilatation of the esophagogastric anastomosis and spontaneously coughed out the occluder. The fistula was repaired and complete closure that was confirmed on esophagography. He had no recurrence of fistula during the follow-up period of 13 months.

& Sen Jiang [email protected] Ping Jiang [email protected] Ji Liu [email protected] Dong Yu [email protected] Bing Jie [email protected] 1

Department of Emergency, Shanghai Pulmonary Hospital, Tongji University School of Medicine, Shanghai, China

2

Department of Anesthesiology, Shanghai Pulmonary Hospital, Tongji University School of Medicine, Shanghai, China

3

Department of Radiology, Shanghai Pulmonary Hospital, Tongji University School of Medicine, 507 Zhengmin Road, Shanghai 200433, China

Keywords Tracheoesophageal fistula  Atrial septal defect occluder  Radiology intervention  Nonsurgical treatment Abbreviations ASD Atrial septal defect TEF Tracheoesophageal fistula

Introduction Most tracheoesophageal fistulas (TEFs) are malignant and often result from tumor growth or recurrence in esophageal and lung carcinomas [1, 2]. Nonmalignant TEF is rare, and causes can include congenital dysplasia, complication of surgery or sent placement, trauma, infection, and long-term intubation [3–10]. Surgery is usually required to treat nonmalignant TEF [3, 4]. We present a case of refractory TEF treated with nonsurgical invasive endotracheal closure using an atrial septal defect (ASD) occluder. A brief review of the literature is also presented.

Case Report A 53-year-old man was admitted to our hospital in December 2013 for treatment of refractory TEF. Before this admission, he had been diagnosed and treated at a local hospital. He had a history of moderately differentiated squamous cell carcinoma in the mid esophagus and had undergone radical esophagectomy (left transthoracic esophagectomy and esophagogastrostomy above the aortic arch) in February 2008. After esophagectomy, he

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P. Jiang et al.: Closure of Nonmalignant Tracheoesophageal Fistula Using an Atrial Septal…

developed a benign stricture at the anastomotic site and underwent endoscopic dilatation about once a month for the next 5 years. In October 2013, the patient elected to undergo stenting with an 18 9 60 mm covered selfexpanding nitinol stent (Micro-Tech Co. Ltd., Nanjing, China) because he could no longer tolerate repeated dilatations. Eight days after stent placement, he presented with coughing associated with eating and drinking. A TEF at the upper part of stent was found on esophagoscopy, and the stent was then removed. The patient was treated with placement of a covered tracheal stent and injection of fibrin glue, but failed to achieve closure of the fistula. The patient was then transferred to our hospital for further care. Upon arrival to our emergency department, the patient was severely malnourished and short of breath. Auscultation of the lungs revealed diffuse moist rales bilaterally. His body mass index was 14.4 kg/m2, and his serum albumin concentration was 22 g/L. Arterial blood gas analysis revealed a PO2 of 56 mmHg. Enhanced CT scan results displayed a TEF 6.5 mm in greatest dimension 2 cm above the anastomosis, evidence of esophagectomy, and severe pulmonary infection in both lungs (Fig. 1A, C). The patient was offered surgical or nonsurgical closure using an ASD occluder and opted for the minimally invasive closure. The procedure was performed in the catheterization laboratory. The occluder system (CeraTM ASD occluder; LifeTech Scientific; Shenzhen, China) consists of a selfexpandable, double-umbrella device covered with polyester

fabric (Fig. 2). Because endoesophageal placement of the ASD occluder for closure of TEF has been reported to result in its migration into the airway [9], we chose an endotracheal approach, leaving the larger, distal umbrella in the esophagus, to prevent device migration. The patient received intravenous anesthesia, endotracheal intubation, and ventilator assistance. Via the endotracheal tube, a 4-French C2 catheter (Cordis Corp; Miami Lakes, FL, USA) with a 0.035 inch, 150-cm guidewire (Terumo Corp; Tokyo, Japan) was inserted through the fistula into the thoracic stomach under direct visualization using a pediatric bronchoscope. From the catheter, a 260-cm Amplatzer guidewire (Cordis Corp) was exchanged, and a 45-cm, 7-French renal artery sheath (Flexor Check-Flo introducer sheath; Cook Medical; Bloomington, IN, USA) was inserted into the esophageal side (Fig. 3A). From the sheath, an ASD occluder with a 10 mm diameter waist, a 22 mm diameter distal umbrella, and an 18 mm diameter proximal umbrella was then introduced and released from the esophageal to the tracheal-side under endoscopic and fluoroscopic control (Fig. 3B–D). Three hours after the procedure, the patient underwent esophagography, which showed complete closure of the fistula (Fig. 4A). The patient resumed an oral diet after the procedure and was discharged 3 days postoperatively. Before discharge, we instructed the patient to have the device removed when the re-epithelialized mucosa was seen to cover the esophageal-side umbrella during endoscopic dilatation therapy at his local hospital. After discharge from our hospital, he

Fig. 1 A, C Pre-closure CT scan showing a large tracheoesophageal fistula (TEF) and severe pulmonary infection in both lungs. B, D CT scan 3 months post closure showing good position of the atrial septal defect (ASD) occluder and significant resolution of pneumonia

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P. Jiang et al.: Closure of Nonmalignant Tracheoesophageal Fistula Using an Atrial Septal…

Fig. 2 The ASD occluder device used for closure the TEF

continued to undergo endoscopic dilatation about once a month. Three months after the procedure, his first followup examination was performed. He felt good, and had neither cough nor fever. His body mass index increased to 19.2 kg/m2. Esophagography and CT scan were performed and showed closure of the fistula, good position of occluder, and significant resolution of pneumonia (Fig. 1B, D). Five months after the procedure, the patient informed us that endoscopy had revealed complete coverage of the esophageal-side umbrella with re-epithelialized mucosa and granulation tissue. However, he had decided not to have the occluder removed. Three hundred eighteen days after the procedure, he had presented with sudden severe cough after dilatation of the esophagogastric anastomosis and spontaneously coughed out the occluder. The patient was quickly brought to our hospital, where esophagography and CT scan showed closure of the fistula (Fig. 4B). The shapes of the removed occluder umbrellas were normal. By the end of the 13-month follow-up period, the

Fig. 3 A A 7-French sheath is inserted into the esophageal side through the tracheal intubation. B, C The occluder is introduced and released through the sheath. D Good shape and positioning of the occluder are seen 10 min after procedure

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P. Jiang et al.: Closure of Nonmalignant Tracheoesophageal Fistula Using an Atrial Septal…

Fig. 4 A Esophagography performed 3 h after the procedure, showing complete closure of the fistula. B Esophagography showing complete closure of the fistula after the occluder was coughed out by the patient

Table 1 Summary of previous articles Reference/ year

Patient no.

Age (year)/sex

Cause of fistula

Procedural access

Outcome

Result of occluder

Follow-up

Scordamaglio [7]/2009

1

73/M

Long-term intubation

Trachea

Successful closure

Remove at 4 months after procedure

4 month and 1 week

Repici [8]/ 2010

2

58/M

Complication of esophagectomy

Esophagus

Placement

8 month

Coppola [9]/ 2010

3

83/M

Ingestion of dental amalgam

Esophagus

Successful closure before migration

Migration into airway at 2 months after procedure

2 month

Miller [10]/ 2014

4

72/F

Complication of stenting

Trachea

Successful closure before migration

Migration into airway at 3 months after procedure

3 month

Present case

5

53/M

Complication of stenting

Trachea

Successful closure

Migration into airway at 318 days after procedure

13 month

Successful closure

patient had experienced no further symptoms of TEF; however, at that time he committed suicide because of personal problems.

Discussion TEF is a life-threatening condition that should be treated immediately after diagnosis. Interventional and/or endoscopic placement of covered esophageal and/or airway

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stents is the preferred management for malignant TEF [1, 2]. Various surgical techniques can be used to treat nonmalignant TEF [3]. Chaturvedi et al. [4] reported a surgical method that employed an obturator device to treat nonmalignant TEF. Several nonsurgical invasive techniques to treat nonmalignant TEF, such as temporary stent placement, fibrin glue injection, and closure using an Amplatzer device, have been described [5–10]. These nonsurgical invasive techniques can benefit patients who decline, or are not candidates for, surgery.

P. Jiang et al.: Closure of Nonmalignant Tracheoesophageal Fistula Using an Atrial Septal…

It has been reported that an atrial or ventricular septal occluder, which was developed to treat cardiovascular disease, can be used in the treatment of bronchopleural, bronchoesophageal, and TEF [7–12]. The ASD occluder system consists of a self-expandable, double-umbrellas device covered with polyester fabric. The CeraTM ASD occluder (LifeTech Scientific), has double umbrellas with a 4-mm connecting waist with a self-centering mechanism. Closure of nonmalignant TEF using an ASD occluder has been reported in only four cases prior to the present case (Table 1) [7–10], all of which had successful closure the fistula after the procedure. Scordamaglio et al. [7] first reported endotracheal ASD occluder closure of benign TEF and removed the device 4 months postoperatively, when epithelialization over the tracheal-side umbrella had occurred, but local repair and complete closure of fistula had occurred by postoperative day 7. In three of the other four cases (Table 1), the delayed complication of device migration into the airway was seen at long-term follow-up. There are several possible explanations for device migration: (1) esophageal peristalsis, (2) extrusion by food and/or another external force, (3) enlargement of a fragile fistula, and (4) an inappropriately sized occluder that is too small for the fistula. Theoretically, an endotracheal approach can prevent migration of an ASD occluder by placing the large, distal umbrella in the esophagus. In our case, the esophageal umbrella was completely covered by re-epithelialized mucosa and granulation tissue 5 months after procedure, but the patient chose not to remove the device. Migration occurred after dilatation of the esophagogastric anastomosis, but the fistula was completely closed. The migration might have resulted from extrusion because of dilatation therapy. However, the complication of migration due to enlargement of the fistula has been reported for both endoesophageal and endotracheal approaches [9, 10]. For closure of an ASD, the diameter of the occluder is usually 2–8 mm greater than the diameter of the defect. We used an ASD occluder with a 10 mm diameter waist to close a 6.5 mm TEF, and Scordamaglio et al. [7] used a 20 mm occluder to close a 5 mm TEF. Unfortunately, the sizes of the fistulas and ASD occluders were not described in the other cases [8–10]. In conclusion, closure of nonmalignant TEF using an ASD occluder may be useful in patients presenting with refractory TEF who are not surgical candidates. However, the delayed complication of device migration into the

airway should be seriously considered. The device may be able to be removed when re-epithelialized mucosa completely covers the umbrella on the esophageal side. Conflict of interest All authors declare that they have no conflict of interest. Statement of Human and Animal Rights formal consent is not required.

For this type of study

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Closure of Nonmalignant Tracheoesophageal Fistula Using an Atrial Septal Defect Occluder: Case Report and Review of the Literature.

Tracheoesophageal fistula (TEF) is a life-threatening condition for which there are several management techniques. We present a case of nonmalignant T...
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