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Behavioral Disturbance Causing Sudden Respiratory Distress in a 3-Year-Old After Tracheocutaneous Fistula Closure: A Case Report

Ekeoduru, Rhashedah A. MD; Aijazi, Hassan M. MD; Graham-Carlson, Amy D. MD

doi: 10.1213/XAA.0000000000000512
Case Reports: Case Report
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A 3-year-old boy underwent tracheostomy at age 5 months for respiratory failure. The tracheostomy tube was removed a year later but a tracheocutaneous fistula developed requiring fistulectomy and primary skin closure. After an initial uneventful course in the postanesthesia care unit, the patient became agitated, began to scream, and suddenly developed rapidly progressing subcutaneous emphysema over his chest, face, and abdomen. Orotracheal intubation was emergently performed and chest radiograph revealed pneumothorax and pneumomediastinum. The child was taken to the operating room for placement of a tracheostomy tube.

From the Department of Pediatric Anesthesiology, University of Texas Health Science Center at Houston, Children’s Memorial Hermann Hospital, McGovern Medical School, Houston, Texas.

Accepted for publication January 6, 2017.

Funding: None.

The authors declare no conflicts of interest.

Address correspondence to Rhashedah Ekeoduru, MD, University of Texas Health Science Center at Houston, Children’s Memorial Hermann Hospital, 6431 Fannin St, Ste.5.020, Houston, TX 77030. Address e-mail to rhashedah.a.ekeoduru@uth.tmc.edu.

Advances in the care of critically ill neonates have led to an increase in the number of children requiring prolonged mechanical ventilation and tracheostomy. Tracheocutaneous fistulas (TCFs) develop in 6.2% to 37.1% of children after tracheal decannulation, and can cause chronic cough, skin irritation, recurrent infections, and phonation difficulty.1 It is now recommended to close persistent, symptomatic TCFs. Rare, but previously described complications of TCF closure are postoperative subcutaneous emphysema, pneumothorax, and pneumomediastinum leading to respiratory distress. This report discusses a case in which these complications likely occurred secondary to a behavioral disturbance (ie, temper tantrum), an association which has not been previously described.

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Consent

The patient’s family gave written permission for publication.

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CASE DESCRIPTION

A 3-year-old 10.8 kg, former 34-week premature infant developed respiratory failure because of severe respiratory syncytial viral infection at 2 weeks of age requiring extracorporeal membrane oxygenation and tracheostomy. The tracheostomy tube was removed at age 2, but decannulation was complicated by a TCF. He presented for surgical repair of the fistula.

General anesthesia was induced using inhaled sevoflurane, followed by propofol infusion to facilitate airway examination while maintaining spontaneous ventilation. Direct laryngoscopy and bronchoscopy revealed grade 1 subglottic stenosis (20%) and a large 8-mm TCF. A 4.0-mm internal diameter (ID) microcuffed endotracheal tube was placed. The fistula tract was excised and the skin closed in layers over a drain to facilitate egress of air. Before emergence from anesthesia, 0.5 μg/kg dexmedetomidine was administered to supplement earlier administration of 2 μg/kg fentanyl. The patient was extubated awake and calm with spontaneous respiration of 26 breaths per minute.

During the first 30 minutes in the postanesthesia care unit, the patient remained calm, breathing at a rate between 20 and 28 breaths per minute, with peripheral oxygen saturation of 98% to 100% on blow-by oxygen. Shortly thereafter the child became agitated and began to cry. This was attributed to pain based on a FLACC (face, legs, activity, cry, consolability) score of 7. Morphine 0.05 mg/kg was administered and the patient calmed. Slight bulging was noted at the TCF closure site. Both the otolaryngology and anesthesiology teams promptly assessed the patient. The child was not in distress and had minimal surgical site swelling, thus close monitoring was continued in the postanesthesia care unit.

One hour later the child became upset about wanting to go home and began vigorously screaming. His tantrum continued despite attempts to console him by the care team and his mother. He acutely developed facial and neck swelling that spread to his chest (Figure 1). Oxygen saturation transiently decreased to a nadir of 74% but improved to 95% with supplemental oxygen via facemask. Propofol 3 mg/kg was administered followed by emergent tracheal intubation with a 3.5-mm ID microcuffed endotracheal tube using a C-MAC videolaryngoscope (Karl Storz, Tuttlingen, Germany). The sutures were reopened at bedside to facilitate air egress. Chest radiograph revealed extensive subcutaneous air overlying the face, neck, and chest. Pneumomediastinum and right-sided pneumothorax were also visualized (Figure 2). The patient was taken to the operating room for placement of a 3.5-mm (ID) tracheostomy tube.

Figure 1.

Figure 1.

Figure 2.

Figure 2.

The child was sedated for 24 hours postoperatively with 0.2 μg/kg/h dexmedetomidine. He was ventilated using volume-controlled ventilation with a tidal volume of 76 mL, respiratory rate of 22 breaths per minute, and fraction of inspired oxygen (Fio2) of 0.40. Serial chest radiographs showed a nonexpanding pneumothorax, thus a thoracostomy tube was not placed. Craniofacial emphysema gradually resolved, and the tracheostomy tube was removed on the fourth postoperative day while maintaining dexmedetomidine sedation. The patient was monitored in the intensive care unit overnight then transferred to the ward. He was discharged on the sixth postoperative day without further complication.

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DISCUSSION

This case highlights a potentially life-threatening complication following closure of a TCF. Approximately, 2500 to 4500 tracheotomies are performed on children in the United States each year for upper airway obstruction, respiratory failure, and/or prolonged mechanical ventilation.1 After decannulation, the tracheostomy site usually closes spontaneously. Failure of tract closure and persistent TCF occurs in 6.2% to 37.1% of children because of epithelialization and cicatricial scarring.1 Factors contributing to TCF include age of the child when tracheostomy is performed, technique used, and duration of tracheostomy. Studies indicate that TCF usually does not develop when cannulation lasts <16 weeks, but can increase to 50% when a child is cannulated for >1 year.2

Complications that arise from a TCF and warrant closure include skin irritation, difficulty phonating, weak cough, aspiration, and recurrent respiratory infections. Despite the presence of these symptoms, closure is contraindicated in patients who are ventilator dependent, have chronic respiratory infections, moderate to severe tracheomalacia, or history of difficult intubation.

TCF may be surgically closed via primary closure in layers or by secondary intention. TCF is most commonly treated with a primary closure because of improved cosmetic outcome. The fistula tract is resected, the tracheal defect is sutured, and the overlying tissue is closed in layers. Complications can include subcutaneous emphysema, pneumothorax, pneumomediastinum, respiratory distress, and hypoxia.

During secondary closure, a fistulectomy is performed followed by recannulation with a smaller tracheostomy tube.3 The tracheostomy is decannulated within a few days, and the wound is allowed to gradually close. Compared with primary closure, this option takes longer to heal and is more likely to cause scarring, although there is no difference in complication rates between the 2 techniques.1 In higher-risk patients, like the one presented here, who had a large tracheal defect, secondary closure may be preferable.4

Symptomatic emphysema is a feared complication of TCF closure and is caused by the escape of air from the trachea through the suture lines into subcutaneous tissue, the mediastinum, and pleural spaces. This can lead to life-threatening respiratory compromise. Air leak can occur following both primary and secondary TCF closure and is commonly caused by increased tracheal pressure during coughing.1 Severe complications can also occur in appropriate surgical candidates. Lee et al5 describe a case in which a 3-year-old without respiratory infection, pulmonary pathology, or cough and a tracheal defect of < 5 mm developed pneumomediastinum, pneumothorax, and respiratory distress after primary closure of a TCF.

There are techniques that may reduce complications. One technique is to place a drain to allow air to escape during coughing.6 In addition, a valsalva maneuver should be performed before emergence to check for a tracheal leak. The anesthesiologist can spray the vocal cords with lidocaine to reduce coughing during emergence. It may be advisable to extubate these patients fully awake to avoid upper airway obstruction. Treatment of obstruction with positive pressure bag mask ventilation can worsen an air leak.

Although coughing is the most common cause of raised intratracheal pressure causing air egress, our patient had a postoperative tantrum, which caused a significant air leak. The child emerged from anesthesia smoothly and calmly with no signs of air leak or respiratory distress before the outburst. On further investigation, the child had a history of behavioral difficulty exemplified by crying spells and self-inflicted violence. Several bruises were noted on the forehead preoperatively. A history of behavioral disturbance has not been previously described as a predictor of adverse postoperative outcome following TCF closure. We posit that behavioral pathology should confer a higher risk, leading to careful determination of surgical closure technique. If the surgeon prefers primary closure, a drain should be left in place and intubation equipment should be at the bedside. Symptomatic air leaks following TCF closure can occur rapidly. It is recommended that children be monitored for a minimum of 24 hours postoperatively, with some proposing to keep children undergoing primary closure intubated overnight. However, one retrospective review did not demonstrate significant correlation between the duration of intubation postoperatively and adverse outcomes.2

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CONCLUSIONS

Repair of TCF is an increasingly common surgery because of improved neonatal survival rates and increased incidence of tracheostomy. Postoperative course can be complicated by significant air leaks leading to life-threatening subcutaneous emphysema, pneumomediastinum, and pneumothorax. It is important to note risk factors that increase this complication, such as chronic cough, recent respiratory tract infection, and a tracheal defect larger than 5 mm. We believe that pediatric patients with a history of behavioral disturbances (including, but not limited to, age-related tantrums) should also be considered high-risk patients. High-risk patients should preferentially undergo secondary closure. If primary closure is warranted, then we believe this high-risk group would benefit from close postoperative monitoring in an intensive care setting with strong consideration for 24- to 48-hour sedation.

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DISCLOSURES

Name: Rhashedah A. Ekeoduru, MD.

Contribution: This author helped review the literature, care for the patient, and write and edit the manuscript.

Name: Amy D. Graham-Carlson, MD.

Contribution: This author helped review the literature, care for the patient, and write and edit the manuscript.

Name: Hassan M. Aijazi, MD.

Contribution: This author helped review the literature, care for the patient, and write and edit the manuscript.

This manuscript was handled by: Hans-Joachim Priebe, MD, FRCA, FCAI.

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REFERENCES

1. Osborn AJ, de Alarcon A, Hart CK, Cotton RT, Rutter MJ. Tracheocutaneous fistula closure in the pediatric population: should secondary closure be the standard of care. Otolaryngol Head Neck Surg. 2013;149:766771.
2. Gallagher TQ, Hartnick CJ. Tracheocutaneous fistula closure. Adv Otorhinolaryngol. 2012;73:7679.
3. Geyer M, Kubba H, Hartley B. Experiences of tracheocutaneous fistula closure in children: how we do it. Clin Otolaryngol. 2008;33:359369.
4. Stern Y, Cosenza M, Walner DL, Cotton RT. Management of persistent tracheocutaneous fistula in the pediatric age group. Ann Otol Rhinol Laryngol. 1999;108:880883.
5. Lee BH, Sarah GE, Rosbe KW, Alemi S. Pneumothorax after tracheostomy closure with successful nonsurgical management. J Clin Anesth. 2016;31:115118.
6. Wiel E, Fayoux P, Vilette B. Complications of surgical closure of trachea-cutaneous fistula in pediatric patients-two case reports. Int J Pediatr Otorhinolaryngol. 2000;52:9799.
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