Multidisciplinary management of thoracic esophageal fistula secondary to traumatic upper thoracic fracture (T3–4) with associated discitis/osteomyelitis and spinal epidural abscess: illustrative case

Peter Schaible Midwestern University, Chicago College of Osteopathic Medicine, Chicago, Illinois;

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Paul Gordon Departments of Cardiovascular and Thoracic Surgery,

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Ramasamy Kalimuthu Plastic/Reconstructive Surgery,

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Ellen Omi Trauma Surgery, and

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Keith Schaible Neurological Surgery, Advocate Christ Medical Center, Oak Lawn, Illinois

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BACKGROUND

An esophageal fistula secondary to a traumatic upper thoracic (T3–4) fracture with resultant thoracic discitis/osteomyelitis and an epidural abscess with neurological compromise is a rare clinical entity. Early diagnosis is critical for an optimal clinical outcome avoiding grave and progressive spinal dissemination with structural instability and neurological deterioration.

OBSERVATIONS

The following case, not clearly described previously in the literature, highlights the clinical course and multidisciplinary approach to management including a single-stage posterior cervicothoracic (C3–T6) decompression with vertebral reconstruction with an expandable interbody cage (T2–4) and posterior cervicothoracic fusion and instrumentation (C3–T6), followed by direct esophageal fistula closure with AlloDerm and a vascularized latissimus dorsi muscle flap.

LESSONS

Early diagnosis and the potential treatment of a posttraumatic esophageal fistula requires a multidisciplinary approach.

ABBREVIATIONS

CT = computed tomography; CVA = cerebrovascular accident; DVT = deep vein thrombosis; MRI = magnetic resonance imaging; SEA = spinal epidural abscess; VAS = visual analog scale; VO = vertebral osteomyelitis

BACKGROUND

An esophageal fistula secondary to a traumatic upper thoracic (T3–4) fracture with resultant thoracic discitis/osteomyelitis and an epidural abscess with neurological compromise is a rare clinical entity. Early diagnosis is critical for an optimal clinical outcome avoiding grave and progressive spinal dissemination with structural instability and neurological deterioration.

OBSERVATIONS

The following case, not clearly described previously in the literature, highlights the clinical course and multidisciplinary approach to management including a single-stage posterior cervicothoracic (C3–T6) decompression with vertebral reconstruction with an expandable interbody cage (T2–4) and posterior cervicothoracic fusion and instrumentation (C3–T6), followed by direct esophageal fistula closure with AlloDerm and a vascularized latissimus dorsi muscle flap.

LESSONS

Early diagnosis and the potential treatment of a posttraumatic esophageal fistula requires a multidisciplinary approach.

ABBREVIATIONS

CT = computed tomography; CVA = cerebrovascular accident; DVT = deep vein thrombosis; MRI = magnetic resonance imaging; SEA = spinal epidural abscess; VAS = visual analog scale; VO = vertebral osteomyelitis

An esophageal fistula is a transmural disruption of the esophagus that subsequently leads to the leakage of intraluminal contents into the mediastinum1 and surrounding tissue. This can cause a local infectious/inflammatory response and septic complications. Additional sequelae include progressive spinal dissemination and potential neurological compromise. Esophageal perforations are associated with serious complication and mortality rates ranging from 10% to 25%, when therapy is initiated within 24 hours of perforation, but can rise to 40% to 60%, when the treatment is delayed beyond 48 hours.2 The diagnosis of an esophageal fistula is often difficult to make, because clinical findings can be subtle and mimic other disorders.3

Esophageal fistulas related to spinal fracture are uncommon and generally not on the radar of most treating physicians, so special awareness is paramount. The proximity of the esophagus to the spinal column between C5 and T4 results in its vulnerability to spinal fracture, particularly hyperextension-type injuries, and possible esophageal disruptions and fistula formations.4

The treatment of an esophageal fistula can involve conservative management with enteral diversion or direct closure of the defect with direct repair and or in combination with vascularized tissue.5–7

An esophageal fistula complication has been documented in the cervical spine, with incidence rates ranging from 0% to 3.4%,8,9 although this complication in the high thoracic spine has not been as well documented. Inoue et al.4 documented a summary of high thoracic spine cases with spinal trauma, which included esophageal perforation (Supplemental Table 1). Six of the seven cases had been rapidly diagnosed and conservatively treated and had a good prognosis. The one operative case had no complications. Only one case reported a death, which was due to septic complications.

Spinal epidural abscess (SEA) comprises 0.2 to 2 cases per 10,000 hospital admissions.10 Symptoms associated with SEA are local pain, low-grade fever, and progressive neurological deficit.10 In a meta-analysis with more than 1,000 cases of SEA, Arko et al.11 noted the most common infections are caused by Staphylococcus aureus (63.6%) and Streptococcus species (6.8%). When the SEA is located in the anterior epidural space, it is frequently associated with vertebral osteomyelitis.12 Patients can be successfully treated conservatively by using antibiotics where neurological deficits are absent.10

Vertebral osteomyelitis (VO) is another uncommon disease with an incidence rate from 1:100,000 to 1:250,000.13 VO may develop as hematogenous spread, postsurgery, or due to trauma to the spine,14 and if conservative treatment fails, surgery may be advised.15 The indications for surgery can include epidural abscess formation, progressive neurological deficit, severe destruction of endplates with mechanical instability or segmental kyphosis, septic pseudarthrosis, severe pain, and failure of conservative treatment.13

Illustrative Case

A 74-year-old male with a history of high blood pressure, coronary artery disease, diabetes, peripheral arterial disease, deep vein thrombosis (DVT), and a previous cerebrovascular accident (CVA) with some right-sided residual weakness presented to an outside emergency room following a fall. He reported missing a step, falling backward on a set of concrete stairs, and hitting his back and head consistent with a hyperextension type injury. No loss of consciousness was reported. The patient complained of upper thoracic pain (visual analog scale [VAS] score 7/10), which was sharp and aggravated with movement. The patient had full strength 5/5 in all four extremities but had chronic paresthesia, likely from diabetes. All imaging studies were negative except for thoracic computed tomography (CT; Fig. 1A and B), which suggested a T3 vertebral fracture extending through the vertebral body and a paraspinal hematoma. There was a T4 pars fracture, but alignment was maintained and there was no canal encroachment. Magnetic resonance imaging (MRI) confirmed no canal involvement (Fig. 1C). On admission, the patient noted difficulty swallowing. He had a history of a previous CVA, and with this concern in mind, the patient underwent brain MRI and magnetic resonance angiography, which ruled out acute stroke. The patient was treated conservatively with a thoracic-lumbar-sacral orthosis brace and was discharged with a follow-up in 4 weeks.

FIG. 1
FIG. 1

Initial CT scans (A and B) and sagittal T2-weighted MRI scan (C) displaying “beak-like” fracture. Sagittal T1-weighted MRI scan (D) prior to laminectomy. Sagittal T1-weighted MRI scans (E and F) postlaminectomy with progressive spinal dissemination.

At the 4-week neurosurgery follow-up visit, the patient complained of pain (VAS score 4/10), and the thoracic radiograph showed no signs of progressive subluxation or vertebral collapse. At 6 weeks, as an outpatient via his primary care physician, the patient received a course of antibiotics for aspiration pneumonia. Subsequently, 7 weeks from his initial injury, the patient returned to the emergency department with an inability to ambulate for the previous 4 days with a complaint of “sweating at night” with low-grade fevers. His lower extremity was assessed at 0 to 1/5 proximal and 2/5 distal strength with decreased reflexes and decreased light touch sensation. Thoracic MRI without contrast (Fig. 1D) displayed significant inflammatory changes, enhancement consistent with epidural abscess, associated spinal cord compression, as well as progressive vertebral changes. The patient was taken to the operating room for a posterior T2–4 laminectomy. At the time of the operation, cultures were taken and found to be negative. Postoperatively, the patient initially seemed to have preserved movement in his legs. Two days postoperatively, the patient was noted to have neurological deterioration with no movement in his legs and with new-onset upper-extremity weakness.

The patient underwent further imaging. MRI (Fig. 1E and F) suggested extensive cervical inflammatory involvement with extension of the thoracic epidural abscess into the cervical spine region and a concern for an esophageal fistula. The patient underwent a single-stage posterior cervicothoracic decompressive laminectomy with posterolateral vertebral thoracic corpectomy (T3) with vertebral reconstruction with an expandable interbody cage (T2–4) and posterior cervicothoracic lateral fusion and instrumentation (C3–T6). Intraoperative cultures grew Lactobacillus. Eight days later, the combined services of plastic and thoracic surgery performed a bronchoscopy, esophagoscopy (Supplemental Fig. 1), and right-sided thoracotomy with direct closure of the esophageal fistula with AlloDerm and myocutaneous vascularized latissimus dorsi flap. The myocutaneous flap was raised and inserted around the esophagus. Initially postoperatively, the patient improved with upper extremity movement; however, no lower extremity improvement was noted. Later the patient had a tracheostomy. The postoperative course was complicated by pulmonary embolus, DVT, multiorgan failure, and continued respiratory insufficiency, and the patient died 47 days later. Postoperative radiography (Fig. 2) demonstrated good implant position, and subsequent radiographs approximately 1 month later continued to demonstrate no evidence of implant failure with maintained implant positioning.

FIG. 2
FIG. 2

Radiological imaging performed after a single-stage posterior cervicothoracic (C3–T6) decompression with vertebral reconstruction with an expandable interbody cage (T2–4) and posterior cervicothoracic fusion and instrumentation (C3–T6), displaying good screw and cage placement.

Patient Informed Consent

The necessary patient informed consent was obtained in this study.

Discussion

Observations

An upper thoracic fracture with its proximity to the esophagus makes that organ vulnerable to injury and subsequent disseminated infectious sequalae. Early diagnosis and treatment of esophageal fistula formation, which is generally not on the radar of most treating clinicians, can significantly impact a patient’s clinical course and outcome.

A traumatic esophageal fistula associated with a ventral lower cervical and an upper thoracic fracture are uncommon and generally not on the radar of most care providers. The proximity of the esophagus to the lower cervical and upper thoracic spine allows for a possible site of perforation and subsequent luminal disruption. Early features of an esophageal fistula can be associated with symptoms of esophageal dysfunction such as difficulty swallowing, cough, aspiration, and aspiration pneumonia as in our patient. Pain with swallowing and reflux/gastroesophageal reflux disease symptoms can also occur. Imaging studies including spinal CT and spinal MRI should be scrutinized for esophageal dilation, compression, and distortion, as well as paraoesophageal collections and inflammatory changes involving both the esophagus and surrounding structures. Our patient’s presenting CT and MRI studies (Fig. 3) indicated concerning features of proximal esophageal dilatation and esophageal compression from a vertebral osteophyte/fracture. Our patient’s subsequent MRI with contrast (Fig. 4 and 5) further demonstrated esophageal fistula formation with ventral paraspinal abscess.

FIG. 3
FIG. 3

Initial thoracic CT scan demonstrating anatomical proximity of the esophagus (star) with the vertebral fracture. The arrow indicates a prominent ventral osteophyte vertebral fracture, or “beaked” fracture. The circle indicates the trachea.

FIG. 4
FIG. 4

Thoracic MRI scans with proximal esophageal dilatation and esophageal distortion with compression from a beaked vertebral osteophyte/fracture (arrows). Stars indicate the esophagus, and circles indicate the trachea.

FIG. 5
FIG. 5

Sagittal (A and B) and axial (C and D) thoracic T1-weighted MRI scans with contrast displaying a periesophageal fistula/abscess (red arrows). Stars indicate the esophagus, and circles indicate the trachea.

MRI examination can be hampered by a narrow field of view, imaging artifacts associated with pulsatile flow with enlarged vessels, and hollow airway structures. Swallowing studies can be informative and diagnostic, but in our patient, who did undergo a video swallowing examination, this examination only included oropharyngeal and neck views; thus, swallowing studies need to evaluate the entire esophagus down to the gastroesophageal junction. Endoscopic evaluation with esophagoscopy can provide confirmation of intraluminal disruption or defect. Thoracic examination and open thoracotomy allow for direct esophageal inspection and subsequent repair of the fistula. Early diagnosis of an esophageal fistula can limit the progressive disseminated infectious and inflammatory changes. Such changes can result in progressive spinal involvement with discitis, osteomyelitis, and associated destructive spinal changes and neurological compromise with paraplegia and quadriplegia representing catastrophic neurological sequalae.

The management of esophageal fistulas can involve a conservative approach with antibiotics and enteral diversion. A failure to respond to conversative management, extensive esophageal defects/perforation, or an extensive disseminated process may require surgical intervention, and such requires a multidisciplinary approach. Incorporating the traditional posterior spinal approach with the lateral extracavitary approach allows for a generous exposure to address both dorsal and ventral pathologies including, as in our case, infectious and structural but also neoplastic conditions. Using this approach as described for upper thoracic (T3–4) conditions offers a significant advantage over a posterior approach alone, an anterior approach alone, or a combination of both anterior and posterior approaches. Urgent neurosurgical intervention may be needed to limit neurological progression. Esophageal fistula closure requires a multidisciplinary approach involving thoracic surgery and plastic surgery or ear, nose, and throat surgery.

Lessons

Thoracic esophageal fistula secondary to upper thoracic spine fracture is uncommon and not easily recognized. A multidisciplinary management approach and early diagnosis and treatment of this complex diagnosis can limit the potential devastating spinal and neurological sequalae.

Author Contributions

Conception and design: P Schaible, Gordon, K Schaible. Acquisition of data: P Schaible, K Schaible. Analysis and interpretation of data: P Schaible, K Schaible. Drafting the article: P Schaible, Gordon, K Schaible. Critically revising the article: P Schaible, Kalimuthu, Omi, K Schaible. Reviewed submitted version of manuscript: P Schaible, Kalimuthu, Omi, K Schaible. Approved the final version of the manuscript on behalf of all authors: P Schaible. Administrative/technical/material support: P Schaible. Study supervision: P Schaible, Kalimuthu. Patient care: Gordon.

Supplemental Information

Online-Only Content

Supplemental material is available with the online version of the article.

Supplemental Table 1 and Fig. 1. https://thejns.org/doi/suppl/10.3171/CASE23344.

References

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    Abila AW, Nditika ME, Kipkemoi RD, Ondigo S, Khwa-Otsyula BO. Primary repair of esophageal perforation: case report. Int J Surg Case Rep. 2020;71:159162.

  • 2

    Kaman L, Iqbal J, Kundil B, Kochhar R. Management of esophageal perforation in adults. Gastroenterol Res. 2010;3(6):235244.

  • 3

    Jones WG 2nd, Ginsberg RJ. Esophageal perforation: a continuing challenge. Ann Thorac Surg. 1992;53(3):534543.

  • 4

    Inoue T, Abe M. Esophageal perforation associated with fracture of the upper thoracic spine from blunt trauma: a case report [published correction appears in Spinal Cord Ser Cases. 2016 Jul 21; 2:16019]. Spinal Cord Ser Cases. 2016;2:15034.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 5

    Whyte RI, Iannettoni MD, Orringer MB. Intrathoracic esophageal perforation. The merit of primary repair. J Thorac Cardiovasc Surg. 1995;109(1):140146.

  • 6

    Wright CD, Mathisen DJ, Wain JC, Moncure AC, Hilgenberg AD, Grillo HC. Reinforced primary repair of thoracic esophageal perforation. Ann Thorac Surg. 1995;60(2):245249.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 7

    Jougon J, Mc Bride T, Delcambre F, Minniti A, Velly JF. Primary esophageal repair for Boerhaave’s syndrome whatever the free interval between perforation and treatment. Eur J Cardiothorac Surg. 2004;25(4):475479.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 8

    Patel NP, Wolcott WP, Johnson JP, et al. Esophageal injury associated with anterior cervical spine surgery. Surg Neurol. 2008;69(1):2024, 24.

  • 9

    Tasiou A, Giannis T, Brotis AG, et al. Anterior cervical spine surgery-associated complications in a retrospective case-control study. J Spine Surg. 2017;3(3):444459.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 10

    Reihsaus E, Waldbaur H, Seeling W. Spinal epidural abscess: a meta-analysis of 915 patients. Neurosurg Rev. 2000;23(4):175204, discussion 205.

  • 11

    Arko L 4th, Quach E, Nguyen V, Chang D, Sukul V, Kim BS. Medical and surgical management of spinal epidural abscess: a systematic review. Neurosurg Focus. 2014;37(2):E4.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 12

    Sendi P, Bregenzer T, Zimmerli W. Spinal epidural abscess in clinical practice. QJM. 2008;101(1):112.

  • 13

    Gorensek M, Kosak R, Travnik L, Vengust R. Posterior instrumentation, anterior column reconstruction with single posterior approach for treatment of pyogenic osteomyelitis of thoracic and lumbar spine. Eur Spine J. 2013;22(3):633641.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 14

    Graeber A, Cecava ND. Vertebral Osteomyelitis. In: StatPearls. [Internet]. StatPearls Publishing; 2023.

  • 15

    Dai LY, Chen WH, Jiang LS. Anterior instrumentation for the treatment of pyogenic vertebral osteomyelitis of thoracic and lumbar spine. Eur Spine J. 2008;17(8):10271034.

    • PubMed
    • Search Google Scholar
    • Export Citation

Supplementary Materials

  • Collapse
  • Expand
  • FIG. 1

    Initial CT scans (A and B) and sagittal T2-weighted MRI scan (C) displaying “beak-like” fracture. Sagittal T1-weighted MRI scan (D) prior to laminectomy. Sagittal T1-weighted MRI scans (E and F) postlaminectomy with progressive spinal dissemination.

  • FIG. 2

    Radiological imaging performed after a single-stage posterior cervicothoracic (C3–T6) decompression with vertebral reconstruction with an expandable interbody cage (T2–4) and posterior cervicothoracic fusion and instrumentation (C3–T6), displaying good screw and cage placement.

  • FIG. 3

    Initial thoracic CT scan demonstrating anatomical proximity of the esophagus (star) with the vertebral fracture. The arrow indicates a prominent ventral osteophyte vertebral fracture, or “beaked” fracture. The circle indicates the trachea.

  • FIG. 4

    Thoracic MRI scans with proximal esophageal dilatation and esophageal distortion with compression from a beaked vertebral osteophyte/fracture (arrows). Stars indicate the esophagus, and circles indicate the trachea.

  • FIG. 5

    Sagittal (A and B) and axial (C and D) thoracic T1-weighted MRI scans with contrast displaying a periesophageal fistula/abscess (red arrows). Stars indicate the esophagus, and circles indicate the trachea.

  • 1

    Abila AW, Nditika ME, Kipkemoi RD, Ondigo S, Khwa-Otsyula BO. Primary repair of esophageal perforation: case report. Int J Surg Case Rep. 2020;71:159162.

  • 2

    Kaman L, Iqbal J, Kundil B, Kochhar R. Management of esophageal perforation in adults. Gastroenterol Res. 2010;3(6):235244.

  • 3

    Jones WG 2nd, Ginsberg RJ. Esophageal perforation: a continuing challenge. Ann Thorac Surg. 1992;53(3):534543.

  • 4

    Inoue T, Abe M. Esophageal perforation associated with fracture of the upper thoracic spine from blunt trauma: a case report [published correction appears in Spinal Cord Ser Cases. 2016 Jul 21; 2:16019]. Spinal Cord Ser Cases. 2016;2:15034.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 5

    Whyte RI, Iannettoni MD, Orringer MB. Intrathoracic esophageal perforation. The merit of primary repair. J Thorac Cardiovasc Surg. 1995;109(1):140146.

  • 6

    Wright CD, Mathisen DJ, Wain JC, Moncure AC, Hilgenberg AD, Grillo HC. Reinforced primary repair of thoracic esophageal perforation. Ann Thorac Surg. 1995;60(2):245249.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 7

    Jougon J, Mc Bride T, Delcambre F, Minniti A, Velly JF. Primary esophageal repair for Boerhaave’s syndrome whatever the free interval between perforation and treatment. Eur J Cardiothorac Surg. 2004;25(4):475479.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 8

    Patel NP, Wolcott WP, Johnson JP, et al. Esophageal injury associated with anterior cervical spine surgery. Surg Neurol. 2008;69(1):2024, 24.

  • 9

    Tasiou A, Giannis T, Brotis AG, et al. Anterior cervical spine surgery-associated complications in a retrospective case-control study. J Spine Surg. 2017;3(3):444459.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 10

    Reihsaus E, Waldbaur H, Seeling W. Spinal epidural abscess: a meta-analysis of 915 patients. Neurosurg Rev. 2000;23(4):175204, discussion 205.

  • 11

    Arko L 4th, Quach E, Nguyen V, Chang D, Sukul V, Kim BS. Medical and surgical management of spinal epidural abscess: a systematic review. Neurosurg Focus. 2014;37(2):E4.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 12

    Sendi P, Bregenzer T, Zimmerli W. Spinal epidural abscess in clinical practice. QJM. 2008;101(1):112.

  • 13

    Gorensek M, Kosak R, Travnik L, Vengust R. Posterior instrumentation, anterior column reconstruction with single posterior approach for treatment of pyogenic osteomyelitis of thoracic and lumbar spine. Eur Spine J. 2013;22(3):633641.

    • PubMed
    • Search Google Scholar
    • Export Citation
  • 14

    Graeber A, Cecava ND. Vertebral Osteomyelitis. In: StatPearls. [Internet]. StatPearls Publishing; 2023.

  • 15

    Dai LY, Chen WH, Jiang LS. Anterior instrumentation for the treatment of pyogenic vertebral osteomyelitis of thoracic and lumbar spine. Eur Spine J. 2008;17(8):10271034.

    • PubMed
    • Search Google Scholar
    • Export Citation

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