(Chest. 2006;130:906-909.)
© 2006
American College of Chest Physicians
First Use of Ventricular Septal Defect Occlusion Device for Endoscopic Closure of an Esophagorespiratory Fistula Using Bronchoscopy and Esophagoscopy*
Thomas Rabenstein, MD, PhD;
Christoph Boosfeld, PhD;
Rolf Henrich, MD and
Christian Ell, MD, PhD
* From the Department of Medicine II (Drs. Rabenstein, Henrich, and Ell), Dr. Horst-Schmidt Klinik Wiesbaden, Wiesbaden; and DRABO Medizintechnik–Amplatzer Support Europe (Dr. Boosfeld), Cologne, Germany.
Correspondence to: Thomas Rabenstein, MD, PhD, Klinik Innere Medizin II, Dr.-Horst-Schmidt-Klinik Wiesbaden, Akademisches Lehrkrankenhaus der Johannes-Gutenberg-Universität Mainz, Ludwig-Erhard-Str 100, 65199 Wiesbaden, Germany; e-mail: thomas.rabenstein{at}hsk-wiesbaden.de
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Abstract
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A 70-year-old woman presented with a persistent, nonmalignant esophagorespiratory fistula. Since other treatment options failed or were denied, an experimental nonsurgical therapy was performed. A self-expanding ventricular septal defect (VSD) occlusion device (Amplatzer; AGA Medical Corporation; Golden Valley, MN) was bronchoscopically introduced to close the fistula. The double umbrella-like occlusion device is made from nitinol mesh and closes luminal contact between the esophageal and bronchial walls, with its waist filling out the fistula itself. The geometry of the occluder system can in theory be designed according to individual purposes and needs. The performed treatment was safe and successful, and the patient remained asymptomatic for 1 year after the first presentation. The treatment of chronic nonmalignant esophagorespiratory fistulas can be difficult. The self-expanding VSD occluder system described in this case might be useful in patients who are not surgical candidates.
Key Words: Amplatzer • esophagorespiratory fistula • experimental treatment • interventional endoscopy • nonmalignant • nonsurgical treatment • therapeutic endoscopy • ventricular septal defect occluder
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Introduction
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The clinical picture of esophagorespiratory fistulas consists of chronic cough, bouts of cough when swallowing liquids, recurrent pulmonary infections, lung suppurations, and hemoptysis.123 In adults, communications between the esophagus and the tracheobronchial tree are mostly acquired.4 Most fistulas have a malignant origin. In these cases, implantation of covered stents either on the esophageal route or on the bronchial route is the treatment of choice because concomitant tumor stenosis prevents the migration of the self-expanding metal mesh prosthesis.56 Benign causes of such fistulas are iatrogenetic (ie, intubation, sclerotherapy, and surgery) and infections, inflammatory conditions, and corrosive ingestion. Developmental abnormalities are summarized as congenital fistulas; they are found three times more commonly to the right than to the left bronchial tree.78 A congenital fistula that remains asymptomatic until adulthood is rare.
The recommended therapy of esophagorespiratory fistulas is surgery that consists of the division of the fistula and the excision of any permanently damaged lung segments.478 Alternatively, a simple stapling has been advocated for elderly patients. Endoscopic obliteration has been recommended for patients who decline to undergo or are unfit for thoracotomy.9 We report on the first use of a ventricular septal defect (VSD) occlusion device (Amplatzer; AGA Medical Corporation; Golden Valley, MN) for combined bronchoscopic and esophagoscopic closure of a benign esophagorespiratory fistula.
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Case Report
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A 70-year-old woman presented with aggravated chronic cough and consecutive worsening of urinary incontinence. The cough was particularly tormenting at night; also, after drinking, the patient had bouts of cough, even though she had never had pneumonia or lung suppuration and did not show an increased body temperature. Laboratory findings including blood cell counts and C-reactive protein were completely normal. Functional pulmonary tests and blood gas analysis also showed no abnormalities. The clinical picture was attributed to an esophagorespiratory fistula of unknown origin, which had been diagnosed 20 years previously, after the accidental swallowing of a fish bone. Since the patient was only afflicted with tussive irritation, no treatment was performed.
Radiologic evaluation (CT and barium study) confirmed an esophagorespiratory fistula to the right pulmonary segment 6, but during routine esophagoscopy, the GI end of the fistula could not be found. Bronchoscopy showed retention of fluid in the distal pulmonary segments of the right side and a bronchial edema of segment 6. After esophageal application of methylene blue, the blue fluid was immediately visible in right segment 6, but the exact localization of the bronchial end of the fistula was not possible. Bronchial application of methylene blue into the right segment 6 finally allowed esophagoscopic identification of the GI end of the fistula and consecutive guide wire and catheter cannulation. After the fistula had been brushed with a cytology brush, 2 mL of fibrin glue were applied in order to induce inflammation-related closure. Five months after fibrin glue injection, the patient returned. Esophagoscopy and bronchoscopy showed an unchanged picture. The fistula was still open, and swallowed fluids flowed directly into the bronchial system (Fig 1
). The patient now agreed to an experimental treatment option by using the self-expanding VSD occluder (Fig 2
) for closing the symptomatic fistula under general anesthesia.
Due to the unfortunate caudocranial orientation of the fistula, a so-called guidewire loop had to be performed. A guidewire (Terumo 0.035; Terumo Corporation; Tokyo, Japan; 150 cm) was introduced into the esophagus and then extracted via the fistula and the bronchial pathway using a biopsy forceps. Subsequently, the wire was exchanged for a 260-cm Amplatzer super-stiff guidewire. The course of this wire was now from stomach through the esophagus, then the fistula and trachea, and finally out of the patient through a side port of the anesthesia mask. Over this wire, we introduced a regular Amplatzer delivery sheath (8F/60 cm/45° tip) into the esophagus and advanced a 6-mm Amplatzer muscular VSD occluder (model 9-VSD-MUSC-006) under fluoroscopic and gastroscopic (visual) control. The distal umbrella of the occluder pushed in the esophagus. Under tension of the whole assembly, the proximal umbrella unfolded in the bronchus of segment 6, while the waist filled the fistula itself (Fig 3, 4
). Procedure time was 65 min, with radiation time of 10 min. A single fever peak of 38.9°C developed 2 h after the procedure, which was treated with IV applied imipenem for 3 days. No further side effects or complications were detected. On day 1 after the procedure, endoscopic evaluation showed correct position of occluder and closure of the fistula. The patient could thus be discharged without any medical complaints.
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Figure 3.. Top, A: the VSD occluder through the bronchial route; the proximal position is controlled endoscopically by esophagoscopy. Bottom, B: the VSD occluder during release; the esophageal end is completely expanded.
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Ten weeks after placement of the occluder, the patient returned for a routine control visit. She reported a symptom-free period of 3 weeks. In week 4 after insertion of the occluder, the chronic cough had recurred; in week 9, halitosis, expectorations, and minor hemoptysis developed. Clinically, she had no fever, and C-reactive protein as well as leukocytes and hemoglobin levels were normal. Chest radiography showed that the occluder was slightly dislocated toward the bronchial system, and the esophageal end was only incompletely expanded. CT ruled out pulmonary abscess and infiltration. During esophagoscopy, the esophageal end of the fistula was clearly visible; fluoroscopically, the occluder appeared retracted into the mediastinum and was incompletely expanded. However, esophageally applied fluid did not reach the bronchial system. Bronchoscopy revealed increased mucosal vulnerability in segment 6 to be the origin of hemoptysis, but the bronchial end of the occluder could not be reached for extraction. The patient recovered under antibiotic therapy (piperacillin and combactam, three 4.5-g doses per day IV for 3 days and then mofloxacin, 400 mg po qd for 10 days) and could be discharged 5 days after hospitalization.
Six months after the procedure, another follow-up examination was performed. The patient felt good, and she had neither fever nor cough or hemoptysis. Bronchoscopically, the occluder could not be reached because of the peripheral position; however, there were no signs of mucosal injury or hemorrhage. Esophagoscopy displayed a small fistula opening, but fluoroscopically the occluder was now completely expanded closely below the esophageal wall. Contrast studies proved complete closure of the fistula by the implanted VSD occluder (Fig 5
). One year after closure of the fistula, the patient reported one further period of expectorations and minor hemoptysis, which were treated successfully with oral antibiotics for 8 days (mefloxacin, 400 mg/d). The initial chronic cough and the aspirations at night did not recur. Endoscopic and radiologic evaluation showed no changes: the fistula was closed by the completely expanded occluder, although the esophageal umbrella was lying in the mediastinum below the esophageal wall.
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Discussion
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This is the first report on the flexible endoscopic closure of a chronic, nonmalignant esophagorespiratory fistula by the use of a self-expanding VSD occluder system.10 The reported fistula was probably acquired because a congenital fistula that remains asymptomatic until adulthood is rare. Increasing problems induced by chronic fluid aspiration developed after a long symptom-free period. Fortunately, no major infectious complications had occurred, but a therapeutic intervention to close the chronic fistula and thus avoid future complications seemed necessary. Esophagoscopic intervention by fibrin glue injection failed to close the fistula, and the patient did not agree with the surgical treatment.
Finally an experimental method using the VSD occluder system was offered to close the fistula, and the patient agreed. The double umbrella-like occlusion device is made from nitinol mesh; it closes luminal contact between the esophageal and bronchial walls, with its waist filling out the fistula itself. The geometry of the occluder system can in theory be designed according to individual purposes and needs, but for this first experimental use we chose a standardized method.11 The prerequisites for placement of the system could be achieved by advanced endoscopic techniques for localization and guidewire cannulation of the fistula. Finally, it was only possible to introduce the system via the bronchial route, since an almost 180° angle between the fistula and esophagus thwarted a transesophageal approach. Correct positioning was achieved under fluoroscopic and gastroscopic guidance. The VSD occluder is able to be repositioned and retrieved,11 and we were prepared to extract and replace it with another occluder if the waist would not match with the length of the fistula. Fortunately, this was not necessary, as the fistula was closed completely after spontaneous retraction of the occluder through the esophageal wall and expansion in this position.
Compared to vascular applications in which the VSD occluder is completely covered by an endothelial layer after a very short time, this cannot be expected in GI or bronchial use. But as long as the discs of the device are positioned closely to the organ walls and do not induce luminal obstruction, no long-term problems may be expected in this respect. Additionally, the device can be extracted if any significant problems occur. Another significant difference compared to vascular use is the possibility of combined radiologic and direct visible control during placement and release during endoscopic procedures. Thus, the use of VSD or atrial septal defect occluder systems is also attractive for endoscopic treatment of other acquired fistulas. For example, we used an atrial septal defect occluder for successful closure of a biliodigestive fistula between the bulbus duodeni and common bile duct, which had induced ascending food impaction, cholangitis, and bile sludge formation (sump syndrome).
The treatment of chronic nonmalignant esophagorespiratory fistulas can be difficult. The self-expanding VSD occluder system described in this case might be useful in patients who are not surgical candidates.
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Acknowledgements
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We thank Sabine Nunius for translation of this article.
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Footnotes
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Abbreviation: VSD = ventricular septal defect
The authors have reported to the ACCP that no significant conflicts of interest exist with any companies/organizations whose products or services may be discussed in this article.
Received for publication October 3, 2005.
Accepted for publication December 19, 2005.
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References
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Abstract
Introduction
