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 Table of Contents  
CASE REPORT
Year : 2022  |  Volume : 3  |  Issue : 1  |  Page : 64-66

Tracheoesophageal fistula masquerading as dysphagia and aspiration in traumatic tetraplegia


1 Department of Physical Medicine and Rehabilitation, AIIMS, Rishikesh, Uttarakhand, India
2 Department of Orthopedics, AIIMS, Rishikesh, Uttarakhand, India

Date of Submission27-Jun-2020
Date of Decision30-Jun-2020
Date of Acceptance18-Nov-2020
Date of Web Publication04-Jun-2021

Correspondence Address:
Dr. Raj Kumar Yadav
T-47, Shivalik Nagar, BHEL Ranipur, Haridwar - 249 403, Uttarakhand
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/JME.JME_101_20

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How to cite this article:
Neyaz O, Yadav RK, Sharma R, Kandwal P. Tracheoesophageal fistula masquerading as dysphagia and aspiration in traumatic tetraplegia. J Med Evid 2022;3:64-6

How to cite this URL:
Neyaz O, Yadav RK, Sharma R, Kandwal P. Tracheoesophageal fistula masquerading as dysphagia and aspiration in traumatic tetraplegia. J Med Evid [serial online] 2022 [cited 2022 Jul 5];3:64-6. Available from: http://www.journaljme.org/text.asp?2022/3/1/64/344304




  Introduction Top


Dysphagia after acute traumatic cervical spinal cord injury (CSCI) has been noted in up to 41% of cases.[1],[2] As this dysphagia is mostly due to soft tissue swelling resulting from trauma or anterior spinal surgery, it usually subsides in 2 weeks post-injury.[3] The causes of prolonged dysphagia after anterior cervical surgery are difficult to define. Usual causes are protrusion of implants or graft, developing of infections or haematoma, injuries to the pharyngeal plexus, vagus nerve, glossopharyngeal nerve or hypoglossal nerve.[4] Tracheoesophageal fistula (TEF) as a cause of dysphagia and aspiration in traumatic tetraplegia has not been reported commonly in the literature. Here, the authors describe a rare case of TEF as a cause of prolong dysphagia and aspiration in a traumatic CSCI.


  Case Report Top


A 26-year-old unmarried male presented as traumatic tetraplegia with neurogenic bladder and bowel secondary to C5 and C6 subluxation due to fall from height in June of 2019. His findings on clinical examination were as follows: motor level: right - C5, left - C4; sensory level: right - C4, left - C4; neurological level of injury - C4; American Spinal Injury Association Impairment Scale-A. He underwent anterior cervical discectomy of C5-C6 and fusion with left iliac crest tricortical bone graft [Figure 1]. On the second post-operative day, he developed a tense swelling over the surgical site along with surgical emphysema. On ultrasound, an ill-defined collection with internal echoes, deep to the left sternocleidomastoid muscle extending into tracheoesophageal groove, was seen. Thus, an oesophageal injury was suspected. Neck exploration was done, and an oesophageal rent was found which was repaired using sternocleidomastoid muscle flap. In view of inadequate cough reflex, a tracheostomy was planned. As the oesophageal rent was well above the carina and it was adequately repaired, an elective tracheostomy was performed under direct vision as per the standard surgical procedure and it was uneventful. The tracheostomy tube used was single lumen Portex cuffed of size 7.5. Cuff pressure was monitored with a cuff manometer and it was kept at 20 cm of water. Enteral feeding was started with 16 French soft rubber nasogastric tube (NGT). Surgical emphysema subsided gradually. A Gastrografin swallow study done 3 weeks post-operatively showed no leak [Figure 2].
Figure 1: Anterior cervical discectomy C5-C6 and fusion with left iliac crest tricortical bone graft

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Figure 2: No leak in Gastrograffin swallow study

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An oral feeding trial was initiated, during which the patient's oxygen saturation dropped to 33%. On auscultation, breath sounds were reduced with the wheezing present over the left lower part of the chest. Hence, the trial had to be aborted. The patient underwent exercises and training for improving respiratory muscle strength, cough reflex and secretion management. Meanwhile, some repeat oral feeding trials in the presence of NGT were done, but they also failed.

After a month, there was an adequate improvement in cough reflex and respiratory muscle strength. NGT was removed, as it may itself cause dysphagia.[5] An oral feeding trial was given, but the patient developed desaturation (SpO2 58%), tachycardia (140/min) and hypotension (90/60 mmHg). Tracheal suctioning was done, high-flow oxygen at the rate of 10 L/min started and 16 French soft rubber NGT was re-inserted under all aseptic precaution. After this, his vitals improved within 5 min.

Multiple failed oral feeding trials, with or without NGT, even after improvement in cough reflex and respiratory muscle strength, made suspicion of some rare cause for this. An upper gastrointestinal endoscopy was done to look for it, which revealed a fistulous communication between oesophagus and trachea of size 10 mm × 8 mm, at 1 cm below the upper oesophageal sphincter. Along with it, NGT-induced mucosal injury and Grade C reflux esophagitis were also seen [Figure 3]. Thus, a diagnosis of TEF was made. As the patient was a poor candidate for surgery, a decision to manage the TEF conservatively was taken. Percutaneous endoscopic gastrostomy was performed for long-term feeding. Proton-pump inhibitors were given for reflux esophagitis. A focused rehabilitation protocol was continued for improving the respiratory condition of the patient and building the nutritional status.
Figure 3: Upper gastrointestinal endoscopy picture

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  Discussion Top


Acquired tracheoesophageal fistulae (ATEF) are mostly malignant and rarely benign.[6] Benign ATEF are usually caused by blunt or penetrating trauma, granulomatous infections and iatrogenic or chemical injuries. In nonventilated patients, the usual presentation is uncontrolled coughing when swallowing, dysphagia, breathlessness, chest pain, voice hoarseness, haemoptysis and repeated pneumonia. In ventilated patients, repeated respiratory tract infections, inability to wean from the ventilator and unexplained weight loss may lead to suspicion of TEF.[7]

In the present case, after anterior cervical discectomy, an oesophageal rent had been found post-operatively. According to a systematic review by Halani et al., oesophageal injuries are rare but well-known complications of anterior cervical spine surgeries.[8] Hardware failure, intraoperative injury by operative tools, graft penetration and erosion by hardware are the reasons for early oesophageal injuries in these surgeries. Subsequently, the patient developed TEF, despite good oesophageal repair and keeping the cuff pressure low at 20 cm H2O. A similar case has been reported by Hameed et al., where ATEF was developed but due to high cuff pressure of 40 cm H2O.[9] Ischaemic damage to the trachea depends on the balance between mucosal perfusion pressure and the pressure exerted by the cuff. In patients with long-term use of a cuffed tracheal tube, even low cuff pressure can cause mucosal damage. Concurrent use of NGT, frequent chest infection, poor nutritional status and excessive motion of tracheostomy tube are additional contributing factors.[9] All these factors were present in the described case.

The diagnosis of TEF is usually based on a combination of clinical, radiographic and endoscopic findings. Contrast-enhanced oesophagography has been used traditionally. In mechanically ventilated patients, computed tomography (CT) is the alternative. Endoscopy and/or bronchoscopy are procedures of choice for confirming the imaging findings and localising the fistula.[10] Benign ATEF are mostly treated surgically wherever possible. Surgical options include oesophageal diversion, resection and anastomosis, direct closure and repair, collar oesophagostomy with gastrostomy or jejunostomy.[11] Single stenting with oesophageal or airway stent is recommended for benign ATEF >5 mm in patients who are not fit for surgery or needing time for definitive surgery. Smaller fistulas are treated with local endoscopic therapies (e.g. clipping, fibrin glue).[12]

In the present case, diagnosis was made using endoscopy and non-surgical management was done, as he was tetraplegic with poor recovery, had bad nutritional status and insufficient respiratory reserve. Conservative therapeutic measures for ATEF include eliminating oral intake and optimising nutrition through a gastrostomy or jejunostomy tubes, using proton-pump inhibitors, keeping the head of the bed elevated, providing adequate oxygenation, good suctioning and managing pulmonary infections promptly to prevent dreaded complications.[12]

Acknowledgement

We acknowledge (1) Gupta Suvrat, DNB (PMR), Senior Resident, Department of Physical Medicine and Rehabilitation, AIIMS Rishikesh, and (2) Patra Binayak, Department of Physical Medicine and Rehabilitation, AIIMS Rishikesh.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
  References Top

1.
Shem K, Castillo K, Wong SL, Chang J, Kolakowsky-Hayner S. Dysphagia and respiratory care in individuals with tetraplegia: Incidence, associated factors, and preventable complications. Top Spinal Cord Inj Rehabil 2012;18:15-22.  Back to cited text no. 1
    
2.
Shem K, Castillo K, Wong S, Chang J. Dysphagia in individuals with tetraplegia: Incidence and risk factors. J Spinal Cord Med 2011;34:85-92.  Back to cited text no. 2
    
3.
Hayashi T, Fujiwara Y, Sakai H, Kubota K, Kawano O, Mori E, et al. The time course of dysphagia following traumatic cervical spinal cord injury: A prospective cohort study. Spinal Cord 2019;28:1-5.  Back to cited text no. 3
    
4.
Yue WM, Brodner W, Highland TR. Persistent swallowing and voice problems after anterior cervical discectomy and fusion with allograft and plating: A 5- to 11-year follow-up study. Eur Spine J 2005;14:677-82.  Back to cited text no. 4
    
5.
Hayashi T, Fujiwara Y, Sakai H, Maeda T, Ueta T, Shiba K. Risk factors for severe dysphagia in acute cervical spinal cord injury. Spinal Cord 2017;55:940-3.  Back to cited text no. 5
    
6.
Bouayyad S, Beena M, Nigam A. A rare case of acquired benign tracheoesophageal fistula. J Surg Case Rep 2020;2:1-3.  Back to cited text no. 6
    
7.
Diddee R, Shaw IH. Acquired tracheo-oesophageal fistula in adults. Contin Educ Anaesth Crit Care Pain 2006;6:105-8.  Back to cited text no. 7
    
8.
Halani SH, Baum GR, Riley JP, Pradilla G, Refai D, Rodts GE Jr., et al. Esophageal perforation after anterior cervical spine surgery: A systematic review of the literature. J Neurosurg Spine 2016;25:285-91.  Back to cited text no. 8
    
9.
Hameed AA, Mohamed H, Al-Mansoori M. Acquired tracheoesophageal fistula due to high intracuff pressure. Ann Thorac Med 2008;3:23-5.  Back to cited text no. 9
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10.
Bixby BA, Maddock SD, Reddy CB, Iravani A, Ansari SA. Acquired esophago respiratory fistulae in adults. Shanghai Chest 2020;4:4.  Back to cited text no. 10
    
11.
Marulli G, Mammana M, Natale G, Rea F. Surgical treatment of acquired benign tracheoesophageal fistulas. J Vis Surg 2018;4:123.  Back to cited text no. 11
    
12.
Majid A, Kheir F. Tracheo and broncho-esophageal fistulas in adults. In: Colt HG, editor. UpToDate. Waltham, Mass: UpToDate; 2020. Available from: https://www.uptodate.com. [Last accessed on 2020 Apr 28].  Back to cited text no. 12
    


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