International Journal of Pediatric Otorhinolaryngology 78 (2014) 701–703
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Case Report
Postoperative recurrent tracheoesophageal fistula: An unusual complication of oxidized regenerated cellulose (Surgicel1) Zafer Dokumcu *, Kamer Polatdemir, Coskun Ozcan, Ata Erdener Department of Pediatric Surgery, Ege University Facuty of Medicine, 35100 Bornova, Izmir, Turkey
A R T I C L E I N F O
A B S T R A C T
Article history: Received 24 September 2013 Received in revised form 23 January 2014 Accepted 26 January 2014 Available online 6 February 2014
Oxidized regenerated cellulose (Surgicel1) is a commonly used material in Pediatric Surgery. We present a case of recurrent tracheoesophageal fistula (RTEF) repaired by Surgicel1. In this case, tracheoesophageal fistula (TEF) recurred due to migration of Surgicel into the tracheal and esophageal lumen. ß 2014 Elsevier Ireland Ltd. All rights reserved.
Keywords: Esophageal atresia Recurrent tracheoesophageal fistula Surgicel1
1. Introduction RTEF remains a severe and potentially dangerous complication of esophageal atresia (EA) repair. However, recurrence of the fistula may occur in about 5–10% of cases; and these can be difficult to repair [1]. Although there are number of reports of endoscopic obliteration of RTEF using diathermic obliteration, tissue adhesive or a combination of tissue adhesive and sclerosing agent [2–4], the rates of success of these techniques are limited [5]. Several attempts may be required for successful closure which can result in repeated and potentially unnecessary exposure to anesthetic risk. That is why, thoracotomy with fistula ligation and division is still the operation of choice. We present a case of EA with RTEF complicated by Surgicel1. 2. Case report A boy newborn was born after 40 weeks of gestation with a birth weight of 2500 g and brought to our neonatal unit for EA with distal TEF. No additional malformations were detected in postnatal examinations. He was operated by thoracotomy the next day and fistula ligation and primary anastomosis with circular myotomy were carried out under slight tension, interposing a pleural flap between trachea and esophagus. In the postoperative period, a transient anastomotic leak developed and healed spontaneously
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[email protected] (Z. Dokumcu). 0165-5876/$ – see front matter ß 2014 Elsevier Ireland Ltd. All rights reserved. http://dx.doi.org/10.1016/j.ijporl.2014.01.034
within a couple of days. Postoperative course was uneventful and he was discharged at 3rd postoperative week. He applied to our department 29 months after definitive operation suffering from recurrent pulmonary infections and coughing following oral feeding, particularly during the intake of fluids. RTEF was suspected and cineradiographic esophagography was carried out however fistula was not seen. Bronchoscopy and esophagoscopy were performed and RTEF was detected under direct vision. Diathermic obliteration and fibrin glue (Tisseel1, Baxter) was applied to the tract via a thin flexible catheter passed into the side port of the bronchoscope three times at intervals of four weeks, however all of them failed. Therefore, a second thoracotomy was carried out. Fistula which was located at the site of original TEF was divided, esophageal and tracheal defects were sutured and Surgicel1 along with surgical adhesive (BioGlue1, CryoLife) was applied on esophageal suture line to prevent leakage and recurrence. He was fed smoothly in postoperative period and discharged uneventfully. He was admitted to the hospital with same previous complaints at 1st month of the second thoracotomy. A thoracic computed tomography (CT) scan was obtained whereas RTEF was suspected (Fig. 1). Therefore bronchoscopy and esophagoscopy were performed. Surprisingly, endoscopic examination showed that Surgicel1 was not absorbed and it migrated into the tracheal and esophageal lumen, recanalizing the fistula tract (Fig. 2). Surgicel1 was not drawn to prevent enlargement of the fistula. Control endoscopy after three weeks revealed persistent TEF where Surgicel1 disappeared. The trachea was separated from esophagus sharply; tracheal and esophageal defects were sutured by a third thoracotomy. Any material was not interposed between trachea
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Fig. 1. CT scan images of N-type TEF (arrow) at tracheal (A) and esophageal (B) levels (CT, computerized tomography; TEF, tracheoesophageal fistula).
Fig. 2. (A and B) Bronchoscopic view of Surgicel1 (arrow) in TEF, (C) esophagoscopic view of Surgicel1 (arrow) at anastomosis level (TEF, tracheoesophageal fistula).
and esophagus. Postoperative course was uneventful and he has been going well at postoperative one year. Timeline of patient’s management is depicted in Table 1. 3. Discussion The most serious problem following primary repair of EA with TEF is RTEF which occurs up to 10% of all large series. It is believed that many recurrent fistulas are caused by anastomotic leakage that results in infection in the area of the repair, particularly when the site of tracheal closure is very close to the anastomosis [6]. Probably, RTEF was also related to the anastomotic leak in our case. Although, RTEF typically occurs in the early postoperative period, it may not be detected for months to years. The diagnosis of RTEF is confirmed on bronchoscopy or cineradiographic esophagography but cineradiography may fail to reveal the fistula [6,7]. That is why; combined bronchoscopy and esophagoscopy can assist in diagnosing these patients. We could also detect RTEF on endoscopic examination in our case. Table 1 Timeline of patient’s management. Age
Diagnosis – intervention
Newborn
EA + TEF Thoracotomy, fistula ligation + primary anastomosis (Pleural flap) RTEF Bronchoscopy diathermy + fibrin glue RTEF Bronchoscopy diathermy + fibrin glue RTEF Bronchoscopy diathermy + fibrin glue RTEF Thoracotomy, fistula ligation + Surgicel1 – BioGlue1 RTEF? Bronchoscopy–esophagoscopy (Surgicel1 migrating into tracheal and esophageal lu¨men) RTEF? Bronchoscopy (Surgicel1 dissappeared recanalizing TEF) RTEF Thoracotomy and fistula ligation
29 months 30 months 31 months 33 months 34 months
35 months 42 months
RTEFs remain a therapeutic challenge because therapeutic approaches have been associated with substantial rates of morbidity, mortality, and re-recurrences [6]. There have been a number of reports describing endoscopic techniques such as electrocautery followed by fibrin glue and other tissue adhesives, and laser in the repair of the recurrent fistula tract [5,8]. However the success rates of endoscopic interventions are questionable. It has been proposed that endoscopic approach may only works temporarily and it may permit to delay the definitive surgical closure saving time for infant growth and clinical conditions improvement [9]. Therefore, the golden standard care for these cases is still operative closure. We tried three applications of endoscopic obliteration by diathermy and fibrin glue for the repair of RTEF in our case but all of them failed and we had to perform thoracotomy and fistula ligation, applying fibrin glue and oxidized cellulose on esophageal suture line. Reinforcement of the suture line with membranes of absorbable materials was suggested for minimizing the risk of leakage by providing strength to the cut tissue [10]. Finley performed fibrin glue-oxidized cellulose sandwich for laparoscopic wedge resection of small renal lesions [11]. Surgicel1 is a bio-absorbable material and can be left in the surgical bed. The exact mechanism of absorption is not well understood, but macrophage processing is presumed to play a role [12,13]. Once saturated with blood, it swells into a gelatinous mass which aids in the formation of a clot. Absorption begins as early as 18th hour after placement of the material into the surgical bed [13]. However, Surgicel1 was not absorbed at 1st postoperative month and caused recanalization of TEF, moving into the tracheal and esophageal lumen in our case. We thought that Surgicel1 penetrated the tracheal and esophageal lumens postoperatively either by expansion or migration. It has been reported that its tendency to swell once placed increases the risk of its use in closed or bone walled spaces [14]. There have been several cases of post-thoracotomy paraplegia owing to the compressive effects of Surgicel1 in adults and children undergoing thoracotomy for pulmonary, esophageal pathologies and resection of mediastinal neuroblastoma [14–16]. They speculated that pressure differences on closure of the thoracic cavity
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caused Surgicel1 to migrate into the spinal canal through the foramen [16–18]. Closure of the thoracotomy with rib approximation could produce a compressive force on the Surgicel1 forcing it into the tracheal and esophageal lumen in our case, too. To the best of our knowledge, this is the first reported case of RTEF via this mechanism after repair of TEF by Surgicel1 and the first reported case in a child. In conclusion, Surgicel1 can complicate the surgical area especially for cases undergoing thoracotomy by migrating through the suture lines into the organs with lumen or foramen. Therefore, it should be used judiciously and, the over-liberal use of Surgicel1 should be avoided for these cases. References [1] S.W. Bruch, R.B. Hirschl, A.G. Coran, The diagnosis and management of recurrent tracheoesophageal fistulas, J. Pediatr. Surg. 45 (2010) 337–340. [2] L. Rangecroft, G.h. Bush, C. Lister, I.M. Irving, Endoscopic diathermy obliteration of recurrent tracheoesophageal fistulae, J. Pediatr. Surg. 19 (1984) 41–43. [3] H.J. Pampino, Endoscopic closure of tracheo-oesophageal fistula, Z. Kinderchir. 27 (1979) 90–93. [4] A.Y. Al-Samarrai, K. Jessen, K. Haque, Endoscopic obliteration of a recurrent tracheoesophageal fistula, J. Pediatr. Surg. 22 (1987) 993. [5] K.T. Tzifa, E.L. Maxwell, P. Chait, A.L. James, V. Forte, S.H. Ein, et al., Endoscopic treatment of congenital H-Type and recurrent tracheoesophageal fistula with electrocautery and histoacryl glue, Int. J. Pediatr. Otorhinolaryngol. 70 (2006) 925–930. [6] A.G. Coran, Redo esophageal surgery: the diagnosis and management of recurrent tracheoesophageal fistula, Pediatr. Surg. Int. 10 (2013) 995–999.
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