CASE REPORT
An Autopsy Case of Iatrogenic Tracheoesophageal Fistula Secondary to Tracheostomy Ritesh G. Menezes, MD, DNB,* Sadip Pant, MD,Þ Sampath Chandra Prasad, MS, DNB,þ Jagadish Rao Padubidri, MD, DNB,§ Prashanth Prabhu, MS,|| Francis N.P. Monteiro, MD, DNB,¶ Tanuj Kanchan, MD,§ Raghavendra Babu Yallapur Prahalad, MD,§ Prashantha Bhagavath, MD, DNB,¶ Manjunath Sathyanarayan Achar, MD,¶ and Savita Lasrado, MS# Abstract: Tracheoesophageal fistula (TEF) is an uncommon but lifethreatening sequel of prolonged use of a cuffed tracheostomy tube. We describe a patient who developed a TEF after a tracheostomy done for management of head injury due to a road traffic mishap. The patient subsequently died of sepsis after bilateral bronchopneumonia. To the best of our knowledge, this is the first autopsy case of iatrogenic TEF after tracheal intubation reported in forensic literature. Key Words: autopsy, trauma, airway, airway management, trachea, tracheostomy, tracheostomy tube, tracheal intubation, tracheoesophageal fistula (Am J Forensic Med Pathol 2014;35: 77Y79)
T
racheoesophageal fistula (TEF) is a life-threatening complication caused by a communication between the esophagus and the tracheobronchial tree within the neck or the thorax. It is an uncommon and a difficult problem to manage. Tracheoesophageal fistulas can be classified as congenital or acquired. Acquired TEFs can be further classified as malignant or nonmalignant. The most common etiology of nonmalignant acquired TEF is tracheal injury related to a cuffed tracheostomy tube in an unconscious patient. Placement of tracheostomy tubes results in trauma to the mucosa because of increased pressure on the tracheal wall secondary to cuff inflation. This results in ischemic damage to the tracheal mucosa, causing necrosis at the site. Patients with TEF are at an increased risk for aspiration pneumonia from gastric contents that reflux through the TEF into the tracheobronchial tree. We describe a patient who developed a TEF after a tracheostomy done for management of head injury due to a road traffic mishap. The patient subsequently died of sepsis after bilateral bronchopneumonia.
Manuscript received May 20, 2012; accepted July 18, 2013. From the *Forensic Medicine Division, Department of Pathology, College of Medicine, King Fahd Hospital of the University, University of Dammam, Dammam, Saudi Arabia; †Department of Internal Medicine, University of Arkansas for Medical Sciences, Little Rock, AR; ‡Department of Otorhinolaryngology Y Head & Neck Surgery, and §Department of Forensic Medicine, Kasturba Medical College, Mangalore, India; ||Department of Otorhinolaryngology Y Head & Neck Surgery, and ¶Department of Forensic Medicine, Kasturba Medical College, Manipal, India; and #Department of Otorhinolaryngology Y Head & Neck Surgery, Father Muller Medical College, Mangalore, India. Kasturba Medical College, Mangalore and Kasturba Medical College, Manipal are affiliated with Manipal University, India. The authors report no conflict of interest. Reprints: Sadip Pant, MD, Department of Internal Medicine, University of Arkansas for Medical Sciences, 4301 W Markham, Little Rock, AR 72205. E-mail: [email protected]; [email protected]. Copyright * 2014 by Lippincott Williams & Wilkins ISSN: 0195-7910/14/3502Y0077 DOI: 10.1097/PAF.0000000000000042
Am J Forensic Med Pathol
&
Volume 35, Number 2, June 2014
CASE REPORT A 33-year-old man presented to a tertiary care referral hospital with cough and heavy expectoration after tracheostomy for management of head injury due to a road traffic mishap 1 month back. The patient had been diagnosed to have a right frontoparietal subdural hematoma after the mishap for which he underwent a burr-hole surgery and evacuation of the hematoma. A cuffed tracheostomy tube was inserted along with a nasogastric tube postoperatively. In the immediate postoperative period, the patient improved considerably. However, in the third postoperative week, the patient developed cough with copious expectoration. At that point, he was referred. After a chest radiograph, a diagnosis of bilateral bronchopneumonia was made. There was frank regurgitation of feeds through the tracheostomy site. An endoscopy was done through the tracheostoma. An obliquely oriented 10.5-cm fistula was seen in the posterior wall of the trachea at the level of the fifth and sixth tracheal rings. The posterior wall of the trachea communicated with the esophagus, forming a complete TEF. A decision was taken to perform a feeding jejunostomy. The cuff of the portex tube was kept distal to the fistula with minimal pressure. Good bronchial suctioning and chest physiotherapy were given to bring out the secretions from the lungs. Despite this, the patient’s condition deteriorated, and he developed generalized septicemia. The patient eventually died of the condition in the days to follow. A medicolegal postmortem examination was performed on the deceased patient in Manipal, South India. The deceased patient was a poorly nourished and moderately built man. A tracheostomy wound was present on the front of the neck in the midline (Fig. 1), with an evidence of a jejunostomy wound on the left side of the front of the abdominal wall. Reflection of the scalp revealed evidence of a burr hole on the right half of the frontal bone with connective tissue in situ. The undersurface of the dura mater showed reddish brown discoloration in the right frontoparietal region. The brain was edematous and congested. A TEF (Fig. 2) was identified 4 cm below the tracheostomal opening on the anterior tracheal wall. The posterior wall of the trachea communicated with the esophageal lumen through the anterior esophageal wall (Fig. 3). Adhesions were present between the posterior tracheal wall and the anterior esophageal wall. Cut surface of both lungs showed purulent material. Estimated one hundred milliliters of straw-colored peritoneal fluid with peritoneal adhesions were also present. The cause of death was opined as sepsis after pulmonary complications secondary to TEF that resulted from management of head injury sustained in a road traffic mishap.
DISCUSSION There has been a paradigm shift in the etiology of acquired TEFs during the last few decades. Although infection used to be the predominant etiology of acquired TEFs, malignancy, iatrogenesis, and trauma constitute the major burden of the disease www.amjforensicmedicine.com
Copyright © 2014 Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
77
Am J Forensic Med Pathol
Menezes et al
&
Volume 35, Number 2, June 2014
FIGURE 3. Esophageal opening of the TEF. FIGURE 1. Tracheostomy wound present on the front of the neck in the midline.
at present.1 Malignancy remains the most common cause of acquired TEF by far.2 Nonmalignant causes include posttraumatic, as in foreign body ingestion and blunt or penetrating trauma, prolonged endotracheal intubation, or prolonged insertion of a cuffed tracheostomy tube; postinflammatory, secondary to esophagitis, caustic injury, and mediastinitis; and postoperative, after surgery of the esophagus or the trachea.2 In patients with human immunodeficiency virus infection, the commonly present esophagitis due to candida or mycobacteria may result in TEF formation.3 The incidence of TEF is 0.5% in patients who undergo tracheostomy with positive pressure ventilation.4 High cuff pressures, high airway pressures, excessive motion of the tracheal tube, prolonged duration of intubation, respiratory infections, esophageal infections, concomitant nasogastric tube insertion, and advanced age are factors favoring formation of a TEF.4 Intracuff pressure greater than 22 mm Hg causes decreased capillary perfusion of the tracheal mucosa, and pressure greater than 36.66 mm Hg leads to total ischemia of the tracheal epithelium.5 The friction caused by the movement of the cuff against the tracheal wall causes abrasion of the tracheal mucosa, which is compounded by the presence of a nasogastric tube.3 This results in ulcerative tracheitis, which, if infected, leads to further
inflammation and necrosis, causing destruction of the trachea and erosion through the esophagus.6 Although it may not be present in all cases, the presence of persistent choking or coughing while swallowing (Ono sign)3 should make one suspicious. Air leak around the cuff and gastric distension may occur. Auscultation over the abdomen may reveal air movement synchronous with respiration. There may be evidence of mediastinal and cervical emphysema or even pneumothorax.7 Contrast radiograph of the esophagus demonstrates the fistula in almost all cases. Esophagoscopy and bronchoscopy will directly visualize the opening of the fistula.3 Appropriate antibiotics to control infection, chest physiotherapy to improve the pulmonary function, and improvement of the nutritional status form the mainstay of initial management of the patient.3 Conservative approach is advised in patients on mechanical ventilation until the patient is weaned off. Patients with several comorbid conditions and requiring prolonged mechanical ventilation are usually not good candidates for surgery with high mortality rates.8 However, direct surgical repair of the fistula is the treatment of choice whenever patient condition is fit for general anesthesia.3 Various approaches are described depending on the site of the fistula. For example, cervical approach is preferred for fistulae above the clavicle, and right posterolateral thoracotomy is preferred for fistulae of the thoracic region.3 A three-layer closure of the fistula supports healing and prevents recurrent fistula formation. Transposition of the transverse colon is done in cases of large fistulae.3 Failure of the closure may occur if the patient requires persistent prolonged ventilator support in the postoperative period. If so, the cuff should be placed distal to the suture line and the patient must not be fed through the nasogastric tube, and instead, a feeding jejunostomy is done.7 Other complications after TEF repair include tracheal stenosis (2.5%-22%), pneumonia, and esophageal anastomosis leakage9 (10%). The overall complication rate is 25% to 50%, and surgical mortality9 is 10% to 18.9%. Autopsy cases of iatrogenic tracheal laceration and perforation after endotracheal intubation are reported in the literature.10,11 However, to the best of our knowledge, this is the first autopsy case of iatrogenic TEF after tracheal intubation reported in the literature. REFERENCES
FIGURE 2. Tracheoesophageal fistula.
78
www.amjforensicmedicine.com
1. Grillo HC. Acquired tracheoesophageal fistula and bronchoesophageal fistula. In: Grillo HC, ed. Surgery of the Trachea and Bronchi. New York, NY: BC Dekker Inc; 2003:341Y356.
* 2014 Lippincott Williams & Wilkins
Copyright © 2014 Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
Am J Forensic Med Pathol
&
Volume 35, Number 2, June 2014
Iatrogenic TEF Secondary to Tracheostomy
2. Diddee R, Shaw IH. Acquired tracheo-oesophageal fistula in adults. Contin Educ Anaesth Crit Care Pain. 2006;6:105Y108.
7. Schmitz GL. Acquired tracheoesophageal fistula. Otolaryngol Clin North Am. 1979;12:823Y827.
3. Darbari A. Non malignant tracheo-esophageal fistula: our experience. Indian J Thorac Cardiovasc Surg. 2005;21:272Y276.
8. Mathisen DJ, Grillo HC, Wain JC, et al. Management of acquired nonmalignant tracheoesophageal fistula. Ann Thorac Surg. 1991;52:759Y765.
4. Payne DK, Anderson WM, Romero MD, et al. Tracheoesophageal fistula formation in intubated patients: risk factors and treatment with high-frequency jet ventilation. Chest. 1990;98;161Y164. 5. Seegobin RD, van Hasselt GL. Endotracheal cuff pressure and tracheal mucosal blood flow: endoscopic study of effects of four large volume cuffs. Br Med J (Clin Res Ed). 1984;288:965Y968. 6. Abbey NC, Green DE, Cicale MJ. Massive tracheal necrosis complicating endotracheal intubation. Chest. 1989;95:459Y460.
* 2014 Lippincott Williams & Wilkins
9. Marzelle J, Dartevelle P, Khalife J, et al. Surgical management of acquired post-intubation trachea-oesophageal fistulas: 27 patients. Eur J Cardiothorac Surg. 1989;3:499Y502. 10. Pazanin L, Misak VB, Goreta N, et al. Iatrogenic tracheal laceration causing asphyxia. J Forensic Sci. 2008;53:1185Y1187. 11. Sato H, Tanaka T, Kasai K, et al. Perforation of the trachea by an endotracheal tube: an autopsy case. Int J Legal Med. 2009;123:513Y516.
www.amjforensicmedicine.com
Copyright © 2014 Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
79
An Autopsy Case of Iatrogenic Tracheoesophageal Fistula Secondary to Tracheostomy Ritesh G. Menezes, MD, DNB,* Sadip Pant, MD,Þ Sampath Chandra Prasad, MS, DNB,þ Jagadish Rao Padubidri, MD, DNB,§ Prashanth Prabhu, MS,|| Francis N.P. Monteiro, MD, DNB,¶ Tanuj Kanchan, MD,§ Raghavendra Babu Yallapur Prahalad, MD,§ Prashantha Bhagavath, MD, DNB,¶ Manjunath Sathyanarayan Achar, MD,¶ and Savita Lasrado, MS# Abstract: Tracheoesophageal fistula (TEF) is an uncommon but lifethreatening sequel of prolonged use of a cuffed tracheostomy tube. We describe a patient who developed a TEF after a tracheostomy done for management of head injury due to a road traffic mishap. The patient subsequently died of sepsis after bilateral bronchopneumonia. To the best of our knowledge, this is the first autopsy case of iatrogenic TEF after tracheal intubation reported in forensic literature. Key Words: autopsy, trauma, airway, airway management, trachea, tracheostomy, tracheostomy tube, tracheal intubation, tracheoesophageal fistula (Am J Forensic Med Pathol 2014;35: 77Y79)
T
racheoesophageal fistula (TEF) is a life-threatening complication caused by a communication between the esophagus and the tracheobronchial tree within the neck or the thorax. It is an uncommon and a difficult problem to manage. Tracheoesophageal fistulas can be classified as congenital or acquired. Acquired TEFs can be further classified as malignant or nonmalignant. The most common etiology of nonmalignant acquired TEF is tracheal injury related to a cuffed tracheostomy tube in an unconscious patient. Placement of tracheostomy tubes results in trauma to the mucosa because of increased pressure on the tracheal wall secondary to cuff inflation. This results in ischemic damage to the tracheal mucosa, causing necrosis at the site. Patients with TEF are at an increased risk for aspiration pneumonia from gastric contents that reflux through the TEF into the tracheobronchial tree. We describe a patient who developed a TEF after a tracheostomy done for management of head injury due to a road traffic mishap. The patient subsequently died of sepsis after bilateral bronchopneumonia.
Manuscript received May 20, 2012; accepted July 18, 2013. From the *Forensic Medicine Division, Department of Pathology, College of Medicine, King Fahd Hospital of the University, University of Dammam, Dammam, Saudi Arabia; †Department of Internal Medicine, University of Arkansas for Medical Sciences, Little Rock, AR; ‡Department of Otorhinolaryngology Y Head & Neck Surgery, and §Department of Forensic Medicine, Kasturba Medical College, Mangalore, India; ||Department of Otorhinolaryngology Y Head & Neck Surgery, and ¶Department of Forensic Medicine, Kasturba Medical College, Manipal, India; and #Department of Otorhinolaryngology Y Head & Neck Surgery, Father Muller Medical College, Mangalore, India. Kasturba Medical College, Mangalore and Kasturba Medical College, Manipal are affiliated with Manipal University, India. The authors report no conflict of interest. Reprints: Sadip Pant, MD, Department of Internal Medicine, University of Arkansas for Medical Sciences, 4301 W Markham, Little Rock, AR 72205. E-mail: [email protected]; [email protected]. Copyright * 2014 by Lippincott Williams & Wilkins ISSN: 0195-7910/14/3502Y0077 DOI: 10.1097/PAF.0000000000000042
Am J Forensic Med Pathol
&
Volume 35, Number 2, June 2014
CASE REPORT A 33-year-old man presented to a tertiary care referral hospital with cough and heavy expectoration after tracheostomy for management of head injury due to a road traffic mishap 1 month back. The patient had been diagnosed to have a right frontoparietal subdural hematoma after the mishap for which he underwent a burr-hole surgery and evacuation of the hematoma. A cuffed tracheostomy tube was inserted along with a nasogastric tube postoperatively. In the immediate postoperative period, the patient improved considerably. However, in the third postoperative week, the patient developed cough with copious expectoration. At that point, he was referred. After a chest radiograph, a diagnosis of bilateral bronchopneumonia was made. There was frank regurgitation of feeds through the tracheostomy site. An endoscopy was done through the tracheostoma. An obliquely oriented 10.5-cm fistula was seen in the posterior wall of the trachea at the level of the fifth and sixth tracheal rings. The posterior wall of the trachea communicated with the esophagus, forming a complete TEF. A decision was taken to perform a feeding jejunostomy. The cuff of the portex tube was kept distal to the fistula with minimal pressure. Good bronchial suctioning and chest physiotherapy were given to bring out the secretions from the lungs. Despite this, the patient’s condition deteriorated, and he developed generalized septicemia. The patient eventually died of the condition in the days to follow. A medicolegal postmortem examination was performed on the deceased patient in Manipal, South India. The deceased patient was a poorly nourished and moderately built man. A tracheostomy wound was present on the front of the neck in the midline (Fig. 1), with an evidence of a jejunostomy wound on the left side of the front of the abdominal wall. Reflection of the scalp revealed evidence of a burr hole on the right half of the frontal bone with connective tissue in situ. The undersurface of the dura mater showed reddish brown discoloration in the right frontoparietal region. The brain was edematous and congested. A TEF (Fig. 2) was identified 4 cm below the tracheostomal opening on the anterior tracheal wall. The posterior wall of the trachea communicated with the esophageal lumen through the anterior esophageal wall (Fig. 3). Adhesions were present between the posterior tracheal wall and the anterior esophageal wall. Cut surface of both lungs showed purulent material. Estimated one hundred milliliters of straw-colored peritoneal fluid with peritoneal adhesions were also present. The cause of death was opined as sepsis after pulmonary complications secondary to TEF that resulted from management of head injury sustained in a road traffic mishap.
DISCUSSION There has been a paradigm shift in the etiology of acquired TEFs during the last few decades. Although infection used to be the predominant etiology of acquired TEFs, malignancy, iatrogenesis, and trauma constitute the major burden of the disease www.amjforensicmedicine.com
Copyright © 2014 Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
77
Am J Forensic Med Pathol
Menezes et al
&
Volume 35, Number 2, June 2014
FIGURE 3. Esophageal opening of the TEF. FIGURE 1. Tracheostomy wound present on the front of the neck in the midline.
at present.1 Malignancy remains the most common cause of acquired TEF by far.2 Nonmalignant causes include posttraumatic, as in foreign body ingestion and blunt or penetrating trauma, prolonged endotracheal intubation, or prolonged insertion of a cuffed tracheostomy tube; postinflammatory, secondary to esophagitis, caustic injury, and mediastinitis; and postoperative, after surgery of the esophagus or the trachea.2 In patients with human immunodeficiency virus infection, the commonly present esophagitis due to candida or mycobacteria may result in TEF formation.3 The incidence of TEF is 0.5% in patients who undergo tracheostomy with positive pressure ventilation.4 High cuff pressures, high airway pressures, excessive motion of the tracheal tube, prolonged duration of intubation, respiratory infections, esophageal infections, concomitant nasogastric tube insertion, and advanced age are factors favoring formation of a TEF.4 Intracuff pressure greater than 22 mm Hg causes decreased capillary perfusion of the tracheal mucosa, and pressure greater than 36.66 mm Hg leads to total ischemia of the tracheal epithelium.5 The friction caused by the movement of the cuff against the tracheal wall causes abrasion of the tracheal mucosa, which is compounded by the presence of a nasogastric tube.3 This results in ulcerative tracheitis, which, if infected, leads to further
inflammation and necrosis, causing destruction of the trachea and erosion through the esophagus.6 Although it may not be present in all cases, the presence of persistent choking or coughing while swallowing (Ono sign)3 should make one suspicious. Air leak around the cuff and gastric distension may occur. Auscultation over the abdomen may reveal air movement synchronous with respiration. There may be evidence of mediastinal and cervical emphysema or even pneumothorax.7 Contrast radiograph of the esophagus demonstrates the fistula in almost all cases. Esophagoscopy and bronchoscopy will directly visualize the opening of the fistula.3 Appropriate antibiotics to control infection, chest physiotherapy to improve the pulmonary function, and improvement of the nutritional status form the mainstay of initial management of the patient.3 Conservative approach is advised in patients on mechanical ventilation until the patient is weaned off. Patients with several comorbid conditions and requiring prolonged mechanical ventilation are usually not good candidates for surgery with high mortality rates.8 However, direct surgical repair of the fistula is the treatment of choice whenever patient condition is fit for general anesthesia.3 Various approaches are described depending on the site of the fistula. For example, cervical approach is preferred for fistulae above the clavicle, and right posterolateral thoracotomy is preferred for fistulae of the thoracic region.3 A three-layer closure of the fistula supports healing and prevents recurrent fistula formation. Transposition of the transverse colon is done in cases of large fistulae.3 Failure of the closure may occur if the patient requires persistent prolonged ventilator support in the postoperative period. If so, the cuff should be placed distal to the suture line and the patient must not be fed through the nasogastric tube, and instead, a feeding jejunostomy is done.7 Other complications after TEF repair include tracheal stenosis (2.5%-22%), pneumonia, and esophageal anastomosis leakage9 (10%). The overall complication rate is 25% to 50%, and surgical mortality9 is 10% to 18.9%. Autopsy cases of iatrogenic tracheal laceration and perforation after endotracheal intubation are reported in the literature.10,11 However, to the best of our knowledge, this is the first autopsy case of iatrogenic TEF after tracheal intubation reported in the literature. REFERENCES
FIGURE 2. Tracheoesophageal fistula.
78
www.amjforensicmedicine.com
1. Grillo HC. Acquired tracheoesophageal fistula and bronchoesophageal fistula. In: Grillo HC, ed. Surgery of the Trachea and Bronchi. New York, NY: BC Dekker Inc; 2003:341Y356.
* 2014 Lippincott Williams & Wilkins
Copyright © 2014 Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
Am J Forensic Med Pathol
&
Volume 35, Number 2, June 2014
Iatrogenic TEF Secondary to Tracheostomy
2. Diddee R, Shaw IH. Acquired tracheo-oesophageal fistula in adults. Contin Educ Anaesth Crit Care Pain. 2006;6:105Y108.
7. Schmitz GL. Acquired tracheoesophageal fistula. Otolaryngol Clin North Am. 1979;12:823Y827.
3. Darbari A. Non malignant tracheo-esophageal fistula: our experience. Indian J Thorac Cardiovasc Surg. 2005;21:272Y276.
8. Mathisen DJ, Grillo HC, Wain JC, et al. Management of acquired nonmalignant tracheoesophageal fistula. Ann Thorac Surg. 1991;52:759Y765.
4. Payne DK, Anderson WM, Romero MD, et al. Tracheoesophageal fistula formation in intubated patients: risk factors and treatment with high-frequency jet ventilation. Chest. 1990;98;161Y164. 5. Seegobin RD, van Hasselt GL. Endotracheal cuff pressure and tracheal mucosal blood flow: endoscopic study of effects of four large volume cuffs. Br Med J (Clin Res Ed). 1984;288:965Y968. 6. Abbey NC, Green DE, Cicale MJ. Massive tracheal necrosis complicating endotracheal intubation. Chest. 1989;95:459Y460.
* 2014 Lippincott Williams & Wilkins
9. Marzelle J, Dartevelle P, Khalife J, et al. Surgical management of acquired post-intubation trachea-oesophageal fistulas: 27 patients. Eur J Cardiothorac Surg. 1989;3:499Y502. 10. Pazanin L, Misak VB, Goreta N, et al. Iatrogenic tracheal laceration causing asphyxia. J Forensic Sci. 2008;53:1185Y1187. 11. Sato H, Tanaka T, Kasai K, et al. Perforation of the trachea by an endotracheal tube: an autopsy case. Int J Legal Med. 2009;123:513Y516.
www.amjforensicmedicine.com
Copyright © 2014 Lippincott Williams & Wilkins. Unauthorized reproduction of this article is prohibited.
79
