Management of symptomatic tracheal pouches

International Journal of Pediatric Otorhinolaryngology (2007) 71, 527—531

www.elsevier.com/locate/ijporl

Management of symptomatic tracheal pouches§ Liane B. Johnson a,b, Robin T. Cotton a,b, Michael J. Rutter a,b,* a

Department of Otolaryngology/Head and Neck Surgery, University of Cincinnati College of Medicine, Cincinnati, OH, United States b Department of Paediatric Surgery, Division of Otolaryngology-Head and Neck Surgery, Dalhousie University, IWK Health Centre, 5850 University Avenue, P.O. Box 9700, Halifax, Nova Scotia, Canada B3K 6R8 Received 11 April 2005; received in revised form 27 October 2006; accepted 29 October 2006

KEYWORDS Tracheoesophageal fistula; Tracheal pouch; Tracheal diverticulum; Fulguration; Electrocoagulation; Diathermy

Summary Objective: Acquired tracheal pouches arise following tracheoesophageal fistula (TEF) repair, but are usually asymptomatic. Symptomatic tracheal pouches are rare, and the optimal management strategy debated. The evolution of our management to this challenging problem is presented. Design: A case series over a 5-year period. Setting: A tertiary care pediatric hospital. Patients: Children with a past history of TEF repair in whom severity of pouch-related respiratory symptomatology warranted surgical intervention. Main outcome measures: Symptomatology, bronchoscopic findings, number and type of surgical interventions required, complications, and outcomes. Results: Eleven patients required surgical intervention for a symptomatic tracheal pouch. Symptoms were due to stasis of secretions with associated pulmonary soiling in three children, severe tracheomalacia in six children, and respiratory obstruction related to the tip of a tracheotomy tube or endotracheal tube being displaced intermittently into the pouch in six children. Surgical management of the pouch included one fulguration with the CO2 laser, one with microlaryngeal instruments, Bugbee cautery in two, open resection in one, and transcervical division in another. The final five children had endoscopic pouch division with Clickline biopsy forceps. All children have had resolution of their pouch-related symptoms, although two remain tracheotomy-dependent. Conclusions: Symptomatic tracheal pouches are rare. Surgical intervention to divide the common party wall between the trachea and the pouch may alleviate associated

§

Presented at ABEA Meeting in Boca Raton, Florida, on May 12, 2002. * Corresponding author at: Division of Pediatric Otolaryngology, Cincinnati Children’s Hospital Medical Center, 3333 Burnet Avenue, Cincinnati, OH 45229-3039, United States. Tel.: +1 513 636 4356; fax: +1 513 636 8133. E-mail address: [email protected] (M.J. Rutter). 0165-5876/$ — see front matter # 2006 Elsevier Ireland Ltd. All rights reserved. doi:10.1016/j.ijporl.2006.10.024

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L.B. Johnson et al. respiratory symptomatology. The Clickline biopsy forceps is a safe, rapid, and effective method of dividing a tracheal pouch. # 2006 Elsevier Ireland Ltd. All rights reserved.

1. Introduction

2. Materials and methods

A tracheal diverticulum or pouch may be either congenital or acquired. Congenital diverticuli are rare narrow-mouthed small out-pouchings of tracheal mucosa. The origin is controversial and is thought to be either developmental or due to herniation of tracheal mucous membrane through an inherent muscular defect. These lesions may present in isolation or in association with other congenital anomalies [1,2]. Acquired lesions are less common. They are usually sequelae of tracheoesophageal fistula (TEF) repair, and the majority are shallow widemouthed pouches in the region of the carina. A subset of tracheal pouches may occur more proximally following the repair of a high TEF, and these are the pouches most likely to be both large and symptomatic. The clinical presentation comprises three categories: in one group of children pooling and stasis of secretions may occur within the pouch leading to chronic aspiration and recurrent pulmonary infections [2—4]. In a second group, the pouch is associated with severe tracheomalacia and airway obstruction. In the final group acute airway obstruction may occur if the tip of an endotracheal tube or tracheotomy tube is displaced into the pouch [5,6]. An individual child may have symptoms in more than one category, and placement of a long tracheotomy tube to bypass an area of severe tracheomalacia due to a tracheal pouch also risks displacement of the tip of the tube into the pouch, particularly during tracheotomy tube change. Tracheal pouch repair is reserved for symptomatic cases and is generally based on division of the shared diverticular and tracheal wall. Various techniques have been described [7], such as electrocautery, Nd:YAG laser, and surgical resection, which may be performed via transcervical, transthoracic, or endoscopic approaches [3]. The outcomes of reported cases vary from death due to fulminant bronchopneumonia and inability to ventilate [2], to successful decannulation following fulguration of the anterior diverticular wall. We present the largest series of acquired symptomatic tracheal pouches as a sequela to TEF repair and describe the evolution and outcome of our treatment approach.

This series consists of a 5-year case series of all symptomatic patients diagnosed with a tracheal pouch at a tertiary care pediatric institution. Inclusion in this series required a diagnosis of a tracheal pouch on rigid airway endoscopy, with the severity of pouch-related respiratory symptomatology warranting surgical intervention. Gender, age, perinatal, and surgical histories were obtained. Patient histories, treatment approaches, and outcomes were evaluated. Treatment approaches were based on size and depth of the pouch, pre-existing tracheotomy, concomitant airway pathology, and surgeon preference. The use of microlaryngeal instruments, CO2 laser endoscopy, and two forms of cautery have all been tried successfully. Decannulation is the ultimate treatment outcome, while the primary treatment objective is the resolution of the pouch-related symptoms.

3. Results Eleven patients were identified, all of whom had had a single previous TEF repair. Gender distribution minimally favored females (6/11). Syndromic association was noted in five patients (VATER in four and partial trisomy 18 in one), including the only three premature infants in the series. No prenatal abnormalities were reported in any patient. Tracheal pouch diagnosis ranged from 1 month to 11 years following TEF repair. Presenting symptomatology included chronic cough (5/11), failure to extubate (6/11), severe tracheomalacia (5/11), recurrent pneumonia (1/11), and chronic aspiration (1/11). Four children presented with episodic lifethreatening respiratory obstruction associated with displacement of the tip of a tracheotomy tube or endotracheal tube into the tracheal pouch. Eight children were tracheotomy-dependent or intubated at the time of initial referral. The median age at diagnosis was 12.5 months while the average age was 38 months. Hospitalization following repair was generally under 48 h due to the presence of a tracheotomy. Persistence of acute airway obstruction required that one patient remain in hospital for up to 4 months and undergo three procedures. The number of procedural attempts to obliterate the tracheal pouches ranged from 1 to 3. Nine

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Table 1 Patient demographics, procedures, and outcomes Age/gender

Technique

# Attempts

Outcome

1 2 3 4

1 mo , 7 wk < 4 mo < 10 mo <

Clickline biopsy forceps Transthoracic a Clickline biopsy forceps Endoscopic division c

1 1 1 1

Decannulated Decannulated Tracheotomy b Decannulated

5

10 mo ,

Bugbee Clickline biopsy forceps

2 1

Tracheotomy, CPAP

6 7 8 9 10

15 mo , 30 mo < 6 yo , 9 yo , 11 yo ,

Bugbee Transcervical d Endoscopic division c Clickline biopsy forceps CO2 laser

2 1 1 1 1

Decannulated Decannulated Decannulated Decannulated Resolution of recurrent pneumonia e

a b c d e

Transthoracic resection at time of slide tracheoplasty. Tracheotomy-dependent due to severe tracheomalacia. Tracheoscopy with microlaryngeal scissors. Transcervical division at time of laryngotracheoplasty. Decannulated as a child, but developed recurrent pneumonia due to tracheal pouch.

patients had a single pouch repair, with all but one of this group successfully decannulated. The other tracheotomized patient required three procedures to fulgurate the pouch. Nine of 11 patients have been successfully decannulated while the remaining 2, despite the absence of a residual pouch, continue to be tracheotomy-dependent due to severe tracheobronchomalacia. The time to decannulation following division of the tracheal pouch ranged from 2 weeks to 7 months. All, but one of these patients, had associated airway anomalies including tracheomalacia (7/11), subglottic stenosis (4/11), bilateral true vocal cord paralysis (2/11), and complete tracheal rings (1/11). Overall follow-up for these patients varied depending on the associated underlying airway pathology as well as the availability of follow-up at their home hospital. All patients were referred from outside institutions, while seven of these were out-of-state patients. Overall follow-up from the time of pouch division ranged from 2 months, for the non-tracheotomized patient, to greater than 20 months. Table 1 summarizes the procedural outcomes including: one endoscopic CO2 laser, one transcervical division during laryngotracheoplasty, one transthoracic resection at the time of slide tracheoplasty, two tracheoscopies with microlaryngeal scissor division, two patients underwent Bugbee bovie [7] for a total of four procedures, and five endoscopic pouch division with the Clickline biopsy forceps (Karl Storz Endoscopy, Culver City, CA; Figs. 1 and 2).

the TEF tract, preserving a proportion of the tracheal aspect of the fistula tract. Transthoracic exposure is excellent when the TEF is distal, thus it is more easily divided close to the tracheal lumen, with a resultant shallow pouch, and less likely to produce post-operative symptoms. Additionally a distal tracheal TEF is less likely to have

4. Discussion Lequien et al. [4] propose that acquired tracheal pouches originate following an unequal division of

Fig. 1

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L.B. Johnson et al.

Fig. 2

associated symptoms secondary to tracheomalacia or risk displacement of an endotracheal tube or tracheotomy tube into the pouch. Meanwhile a proximal TEF is more difficult to tackle surgically as it is poorly exposed through a traditional transthoracic approach, and the tracheal aspect of the TEF is difficult to access. Therefore ligation of a proximal TEF is more likely to be accomplished closer to the esophageal aspect of the TEF, leaving a larger deeper tracheal pouch. This, in turn, is more likely to be symptomatic as tracheomalacia is more pronounced in the tracheal segment corresponding to the tracheal pouch. The severity of the tracheomalacia may require intubation or tracheotomy for airway stabilization, either of which risks displacement of the tip of the ventilating tube into the pouch with a resultant abrupt inability to ventilate. All the techniques described herein have been successful. Procedure selection has evolved based on our past experience, the patient’s associated airway pathology and the efficacy of the technique to yield the most optimal outcome. Drawbacks of the CO2 laser are based on the expense of the equipment, the specialized staff training, and safety measures. Set-up time and overall costs are greater than the other techniques despite a similar surgical outcome, and thus with advances in endoscopic instrumentation the CO2 fell out of favor. Endoscopic division with microlaryngeal scissors is fast and effective, but mild bleeding can hinder proper visualization of the pouch intraoperatively, therefore increasing the inherent risk of the procedure and contributing to the risk of pouch recurrence. This leads to a trial with the Bugbee cautery which enhanced overall visualization by providing hemostasis during pouch division. Bugbee cauterization must be performed cautiously as the tissue coagulum penetrates more deeply than what is immediately apparent intraoperatively. Due to the risk of overly aggressive cauterization with the Bugbee bovie, in our experience, multiple procedures are required to achieve complete fulguration of the pouch. Clickline biopsy forceps is a true-cut endoscopic instrument designed for pediatric laparoscopic procedures,

which uses diathermy for instant coagulation at the time of pouch division. It is the most optimal of the techniques used at our institution to date. It minimizes bleeding, and is a rapid and effective procedure that is comparable in safety and speed to microlaryngeal scissors, while visualization is enhanced due to the associated cauterization. Technically the Clickline biopsy forceps is placed endoscopically through the larynx, while being guided by a ventilating tracheoscope placed through the tracheostoma by an assistant. The endoscopic image provided by the tracheoscope is displayed and the surgeon maneuvers the Clickline biopsy forceps into the pouch, with the common party wall of the pouch lying between the jaws of the forceps. With the diathermy set on a coagulation setting of 30 W, the biopsy forceps are closed resecting a segment of the common party wall, then opened, advanced down the pouch, and this continues till the apex of the pouch is reached. If the pouch is very wide, this technique may be used on the lateral aspects of the pouch bilaterally, allowing a central wedge of the pouch to be resected. The tracheotomy tube is then replaced with the tip of the tube bypassing the apex of the pouch. A minimum of 48-h post-operative observation is recommended to ensure that no residual symptomatology is present and that the post-surgical changes have not altered the tracheal dynamics.

5. Conclusion This series of symptomatic tracheal pouches reveals that patients may develop respiratory symptomatology as late as 11 years following distal TEF repair. Symptomatic tracheal pouches tend to present with respiratory symptoms associated with retained secretions in older children, or with acute obstructive symptoms associated with displacement of a tracheotomy tube or endotracheal tube into a proximal pouch. Associated tracheomalacia is commonly present. An evolution of the surgical management of symptomatic tracheal pouches has shown that an endoscopic approach with Clickline biopsy forceps is fast, safe and effective, and yields optimal results with, to date, no recurrence of symptoms.

Disclaimer None of the authors have derived financial gain, nor do they have a financial interest in the products mentioned in this text.

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