Endoscopic obliteration of recurrent tracheoesophageal fistula

Digestive Diseases and Sciences, Vol. 38, No. 2 (February 1993), pp. 374-377

CASE REPORT

Endoscopic Obliteration of Recurrent Tracheoesophageal Fistula Y. V A N D E N P L A S , MD, PhD, R. H E L V E N , RN, H. DEROP, RN, A. M A L F R O O T , MD, PhD, T. De B A C K E R , MD, T. B E Y E N S , MD, A. V A N D E V E L D E , MD, B. D E S P R E C H I N S , MD, W. L A U R E Y S , MD, G. DEVIS, MD, Phi), and P. D E C O N I N C K , MD, PhD KEY WORDS: (tracheoesophageal) fistula; endoscopy; obliteration; Histoacryl; Cyanoacrylate.

Recurrent tracheoesophageal fistula (TEF) is not an u n c o m m o n complication following surgical repair of esophageal atresia and T E F , with an incidence b e t w e e n 5 and 15% (1). H o w e v e r , the diagnosis is often overlooked. W h e n e v e r a child presents with a persistent cough, with h e a v y coughing immediately after swallowing some liquid, with chronic and recurrent p u l m o n a r y infection, and with failure to thrive, one should keep in mind the possible occurr e n c e of refistulization. A careful investigation needs to be carried out, including chest x-rays, b r o n c h o s c o p y , and e s o p h a g o s c o p y . The surgical treatment of recurrent T E F with division and closure through a t h o r a c o t o m y can cause serious difficulties because of dense adhesions (2) and m a y be unsuccessful because of p o o r blood supply to b o t h ends of the divided and sutureligated fistula. Therefore, a successful endoscopic obliteration technique opens interesting perspectives of less aggressive treatment. CASE REPORT

A 12-year-old girl was referred to our unit for endoscopy of the upper gastrointestinal tract because of clinical recurrence of a TEF. She was born prematurely (birth weight 2.050 kg) with an esophageal atresia with a wide gap, and a TEF. She had a gastrostomy, ligation of the TEF, and cervical esophagostomy shortly after birth. At 7 months, the TEF recurred and was divided again, as an end-to-end anastomosis of both ends of the esophagus Manuscript received December 30, 1991; revised manuscript received June 12, 1992; accepted June 18, 1992. From the Units of Pediatric Gastroenterology, Adult Gastroenterology, Pediatric Pneumology, Pediatric Surgery, Pediatric Anesthesia, Pediatric Radiology, Academisch Ziekenhuis Kinderen, Vrije Universiteit Brussel, Brussels, Belgium. Address for reprint requests: Dr. Yvan Vandenplas, Academic Children's Hospital, Free University of Brussels, Laarbeeklaan 101, 1090 Brussels, Belgium. 374

was carried out in one layer with nonabsorbable sutures. Postoperatively a stenosis of the anastomosis occurred, necessitating dilatations. Esophagoscopy at 3 years showed no stenosis anymore, but a short, blind fistula tract at the site of the anastomosis. At age 7, an important gastroesophageal reflux was documented, causing pneumopathy. Treatment with cisapride, antibiotics, and respiratory physical therapy was successful, but no definitive cure was obtained. This patient has associated progressing scoliosis and thoracic malformations for which she has been wearing a Milwaukee brace since the age of 11. More than 1 year ago, the patient presented with symptoms suggestive of recurrent TEF (nocturnal coughing, ingestion-related coughing, halitosis, and chest pain, which was attributed to the corset). She had to sleep in an upright position, propped up with pillows. There was a chronic severe pulmonary infection, making surgery possibly life-threatening. Endoscopy with an Olympus GIF XP20 confirmed a large fistula at the anastomosis, situated at 23 cm from her teeth. The fistula was about 0.6 cm diameter, about 2 cm long, and in direct connection to the fight middle bronchus, as visualized during fluoroscopy after the injection of contrast medium (Figures 1 and 2). A first attempt to close the fistula was made by injecting 4 ml Tissucol (fibrinogen and thrombin, mostly used to stop diffuse bleeding) through a small polyethylene catheter. The polyethylene catheter (and the very careful manipulation of the latter) is of the greatest importance to prevent adherence to the instrument and occlusion of the channels and suction valves of the endoscope. All therapeutic endoscopies were performed under general anesthesia. However, 24 hr later the product had disappeared as shown with a control fluoroscopy. Four milliliters of Histoacryl (n-butyl-z-cyanoacrylate) was injected at a second obliteration attempt, and in combination with submucosal injection of 2 ml NaC1 30% at a third attempt (3). (Cyanoacrylate is not available in the United States.) Both were successful only very temporarily: after 2 and 10 days, respectively, symptoms reoccurred, and refistulization was demonstrated by fluoroscopy and esophagoscopy under sedation. During a fourth session, 8 ml Histoacryl was injected directly into the fistula followed by 4 Digestive Diseases and Sciences, Vol. 38, No. 2 (February 1993)

0163-2116/93/02004)374507.00/0 9 1993 Plenum Publishing Corporation

TRACHEOESOPHAGEAL FISTULA OBLITERATION

Fig 1. Endoscopic view of the fistula.

ml Aethoxysclerol in the submucosa surrounding the fistula. At the present time, the girl has been asymptomatic for more than 12 months and is doing well. Her pulmonary function has cleared, and weight gain has been significant (from 28 to 36 kg). The patient eats and drinks normally, without any deglutition difficulties. A control esophageal barium study did not reveal a fistula; at endoscopy some Histoacryl was still visible. Since the obliteration, she sleeps in a normal horizontal position. DISCUSSION We could find only one case report on the associated use of Histoacryl and Aethoxysclerol (4), in a premature infant with a very small fistula, needing injection of 0.5 ml Histoacryl and 0.6 ml Aethoxysclerol only. A few earlier reports in the Swiss and German literature (published more than 10 years ago) on the use of tissue adhesives or diathermia for closure of recurrent TEF should be mentioned (5, 6). Digestive Diseases and Sciences, Vol. 38, No. 2 (February 1993)

The association of both products is, in our opinion, essential indeed for successful obliteration. Histoacryl is a well-known powerful tissue adhesive. It has been used alone successfully to close a small bronchopleural fistula (7). Aethoxysclerol is a alcohol-based substance used for endoscopic obliteration of esophageal varices and for hemostasis in bleeding gastrointestinal ulcers. The substance causes an inflammatory process in the injected submucosa, leading to local edema and fibrotic changes. This mechanism is probably of great importance for closure of a large fistula. In our patient, NaCI 30%, a less irritating product, was first injected for the same purpose, but without success. [Submucosal NaC1 30% injection is advised for the treatment of persistent rectal prolapse (3).] To our knowledge, no studies have been published on the pulmonary toxicity of cyanoacrylate. However, the clinical situation of our patient allowed us to inject the product in the fistula since it 375

VANDENPLAS ET AL

Fig 2. Endoscopic close-up of the fistula, prior to obliteration.

was known that in case of complications or failure of the obliteration, a surgical procedure (middle lobectomy) would have been necessary, and this procedure was the only alternative to obliteration. The question arises whether endoscopic obliteration of a recurrent TEF is preferable to surgical repair, since the latter has a high morbidity and mortality rate. In one series, the mortality is reported to be as high as 50% (8). We think that endoscopic obliteration can be considered in each case of recurrent TEF as an alternative approach to high-risk surgical interventions. Relapse of the fistula might occur with both procedures. Even if surgery would appear to be necessary, the endoscopic obliteration of the fistula prior to surgery could be considered, in order to improve the pulmonary infection that is inevitable in patients with a recurrent TEF. The injection of quite a large volume of the tissue adhesive in the fistula (probably and inevitably also some in the bronchus) may be necessary. It was

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clinically very well tolerated in our patient. The asymptomatic follow-up period of 12 months indicates the long-term safety of the procedure. In conclusion, endoscopic obliteration might offer a safe and elegant alternative to major high-risk surgery for the treatment of recurrent TEF in patients in whom surgery would be life-threatening. SUMMARY

We report a successful endoscopic obliteration of a large recurrent tracheoesophageal fistula (diameter 0.6 cm, length 2.0 cm) in a 12-year-old girl, using a combination of Histoacryl (n-butyl-z-cyanoacrylate) and Aethoxysclerol injected through a polyethylene catheter. The severe pulmonary infection, which rendered surgery potentially life threatening, disappeared after the endoscopic closure. Since the obliteration, now over 12 months ago, the girl is asymptomatic. Endoscopic obliteration is a worthwhile technique and should be considered as an Digestive Diseases and Sciences, Vol. 38, No. 2 (February 1993)

T R A C H E O E S O P H A G E A L F I S T U L A OBLITERATION a l t e r n a t i v e to s u r g e r y in p a t i e n t s p r e s e n t i n g with a c o m p l i c a t e d r e c u r r e n t t r a c h e o e s o p h a g e a l fistula. 5.

REFERENCES 1. Cudmore RE: Oesophageal atresia and tracheo-esophageal fistula. InNeonatal Surgery. J Lister, IM Irving (eds). London, Butterworths, 1990, pp 246-247 2. Slim MS, Tabry IF: Left extrapleural approach for the repair of recurrent tracheoesophageal fistula. J Thorac Cardiovasc Surg 68:654-675, 1974 3. Kay NRM, Zachary RB: The treatment of rectal prolapse in children with injections of 30 percent saline solution. J Pediatr Surg 5:334, 1970 4. Izzidien A1-Samarrai AY, Jessen K, Haque K: Endoscopic

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obliteration of a recurrent tracheoesophageal fistula. J Pediatr Surg 22:993, 1987 Rangecroft L, Bush GH, Lister J, Irving IM: Endoscopic diathermy obliteration of recurrent tracheoesophageal fistulae. J Pediatr Surg 19:41-43, 1984 Gdanietz K, Krause I: Plastic adhesives for closing esophagotraheal fistulae in children. Z Kinderchir 17(suppl):137-138, 1975 Malfroot A, Vantussenbroeck F, VanNootn C, Dab I: Endoscopic diagnosis and closure of a bronchopleural fistula. Pediatr Plumonol 11:280-282, 1991 Wayson EE, Garnjobst W, Chandler JJ: Esophageal atresia with tracheo-oesophageal fistula. Am J Surg 110:162-167, 1965

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