Esophageal stenosis is a common complication of laryngeal cancer treatments,1 commonly requiring repeated bougie dilations. Esophageal perforations are rare (0.04−0.40%).2 Subsequent spondylodiscitis and epidural abscess are even rarer, exceptionally reported, with only few cases in the literature to our knowledge. The treatment of this condition, in this selected group of patients, where it is usual to have anterior neck soft tissue problems due to previous surgical approaches and radiation therapy, can become a challenge for the surgeon, limiting the usual management of other causes of spondylodiscitis where an anterior approach debridement and fixation would surely be the first option. We present the first case published, to the best of our knowledge, of successful treatment by posterior fixation and specific intravenous antibiotic therapy.

A 75-year-old male with a history of larynx carcinoma treated with laryngectomy and radiotherapy 30 years earlier, with the consequent deterioration of soft tissues of the anterior neck and progressive esophagus stricture secondary to the treatment, leading to multiple esophageal dilations. Four weeks after an apparently uneventful dilation procedure, the patient consulted at the Emergency Department for neck stiffness, pain and fever. At admission in the infectious disease department, he presented weakness to left shoulder abduction (M:3/5). Leukocytosis (18,690/mL) and elevated C-reactive-protein (25.55 mg/dl, normal <1 mg/dl) were remarkable. Lumbar puncture showed hypoglycorrhachia (3 mg/dl) and an elevated protein count (883 mg/dl). Blood cultures were positive for Peptostreptococcus. Bacterial meningitis was the initial diagnosis in the medical department and was treated with antibiotics according to sensitivity without improvement.

Further image studies were performed. Cervical X-ray showed hypodense areas of C5/C6 vertebral bodies and prevertebral calcification compatible with an abscess (Fig. 1A). Computed tomography (CT) demonstrated a prevertebral collection and a radiolucency of anterior cortical margin of C5-C6 (Fig. 1B). Magnetic resonance imaging (MRI) reported a prevertebral collection with sinus tract from the esophagus, C5/C6 osteomyelitis and an epidural abscess with subarachnoid space occupancy and associated increased uptake of cervicodorsal perimedular ependyma (Fig. 1C). The patient developed a spondylodiscitis and epidural abscess after a previous, four weeks before, neglected esophageal perforation.

Fig. 1.

A. Cervical lateral radiograph: hypodense lesions of C5/C6 vertebral bodies with slight listhesis from C4 to C5. Prevertebral calcification is compatible with abscess formation and increased anterior soft tissue volume (arrows); B. Cervical lateral computed tomography shows erosion and radiolucency of the anterior aspect of C5/C6 vertebral bodies (arrow); C and D. Cervical sagittal T2 and STIR weighted contrast magnetic resonance showing prevertebral collection, increased prevertebral space (double arrow) associated with osteomyelitic signs in the C5/C6 vertebrae, sinus tract from the esophagus and epidural abscess with mild obliteration signs of the spinal cord (arrow).

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Patient received treatment with antibiotics and a Philadelphia collar after his admission. Ten days later, without clinical improvement of shoulder abduction we consider the need for a surgical debridement. Surgical treatment for spondylodiscitis is usually performed through an anterior approach that involves debridement, vertebral body curettage or resection and anterior fixation and fusion. However, in the reported patient, after careful evaluation of the serious soft tissue-damaged state of the anterior neck, the option of doing an anterior approach was discarded. The option of a conservative management with a cervical collar was discarded due to the risk of progressive neurological deterioration. The patient was treated with a posterior approach and fixation by bilateral facet joints C4-C6 and pedicle C7 screws without debridement, associated with 8 weeks of antibiotics. A nasogastric tube was placed in the esophagus for parenteral nutrition during this period although the dilaceration caused by the dilation was probably closed spontaneously. The patient showed progressive improvement, regaining strength in the upper limb (M: 4/5). A 6-month control MRI showed resolution of C5/C6 osteomyelitis and no evidence of abscesses or sinus tracts, although it showed intramedullary expansive high signal (C4-T11), anterior medullary synechiae, and persistent dural and arachnoid enhancement throughout the cord (Fig. 2). After 6 months, total strength was regained without signs of infectious processes after an 8-year follow-up.

Fig. 2.

A. Lateral cervical radiograph at six months post-operation: posterior fixation with bilateral facet screws at C4-C5-C6 and pedicle screws at C7 six months after surgery; B. MRI at 6 moths post- operation showed resolution of C5/C6 osteomyelitis and no evidence of abscesses or sinus tracts, although it showed intramedullary expansive high signal (C4-T11), anterior medullary synechiae, and persistent dural and arachnoid enhancement throughout the cord.

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Management of laryngeal tumors is often complicated by esophageal stricture,3 particularly if radiation therapy has been associated, requiring dilations and causing microtrauma that could induce direct inoculation of intestinal bacteria into adjacent tissues.1 An even more threatening complication is a perforation. The Killian triangle is an anatomically weak area (between the inferior pharyngeal constrictor and the cricopharyngeous muscle) more susceptible to perforations. Its proximity to C4–C6 vertebrae places them at high risk of infection by direct inoculation of bacteria through esophageal perforation.1 Several causes of direct inoculation of bacteria into adjacent tissues of the cervical spine with subsequent infection secondary to esophageal injuries have been reported, such as esophageal diverticulitis, spontaneous rupture,4 complications of stent placements,5 gastrointestinal endoscopies, anterior cervical fusions6 traumatic esophageal injuries7–10 and or surgical procedures as a pharyngolaryngectomy.11

Spondylodiscitis after perforations as a consequence of esophageal dilations is exceptionally described in the literature. Ekbom first reported a case of cervical spondylodiscitis and epidural abscess after a dilation with paresis of an upper extremity, managed with posterior drainage of the abscess that later recurred. Needing further treatment (anterior approach, corpectomy and fusion) nevertheless, cerebrospinal leakage and a persistent fistula developed (Table 1).2

Later, Radulovic reported another case of perforation, causing a rapidly progressive infection, with neurological involvement that underwent drainage of the abscess, with a good outcome after 6 weeks of antibiotics. At two-year follow-up, spontaneous fusion of intervertebral bodies (C2-C4) and paresis of the deltoid muscle were present.12 D’Souza reported 4 cases of cervical spondylodiscitis after reviewing 1221 patients with esophageal dilations; of these, only one had neurological involvement. All were surgically managed with debridement and stabilization.1

Neglecting epidural abscesses related to spondylodiscitis is likely considering its uncommon occurrence and initial subtle symptoms, presenting the classic triad (localized pain, neurologic deficit and fever) only in 37% of the cases.11–14 After onset of neurological symptoms, a rapid progressive compression of neurovascular structures can be expected without rapid intervention.13,14 Although in some series there was no neurological impairment, the incidence of neurological deficit ranges from 40 to 55 % of cervical spondylodiscitis cases reported in the literature.11

Management of spondylodiscitis depends on potential instability, the presence of neurological and progressive symptoms,11 and the overall state of the patient. Conservative management includes intravenous antibiotics based on isolated bacteria, analgesics and cervical bracing.14 Most authors recommend radical debridement, stable reconstruction using anterior only surgery, when needed, and antibiotic therapy. Management of cervical epidural abscesses remains controversial. Most authors recommend immediate decompression and drainage due to the potential risk of expansion, secondary neurological compression and vascular thrombosis. The prognosis of epidural abscesses is inversely proportionate to the duration and severity of symptoms. However, medical management is still a viable option when the infectious germ has been identified, if strict vigilance is ensured.11,15

In the reported case, the usual management would have been to perform an anterior debridement and instrumentation. However, considering the status of the prevertebral cervical skin and soft tissues with fibrosis and adherence related to the history of radiotherapy and surgery, the presence of a traqueostomy, and having identified the infecting bacteria by blood cultures, we decided to perform a posterior cervical instrumentation to stabilize and fix the affected spinal segment and the use of intravenous antibiotic therapy for four weeks that was followed by another 4 weeks of oral antibiotics, obtaining a complete resolution of the infection. An anterior approach, regardless of its difficulty in the case presented, could have been the source of multiple further complications, including greater esophageal perforation.

In cases of spondylodiscitis and epidural abscess, usual management would be anterior debridement and instrumentation. However, after considering the status of prevertebral cervical tissues with fibrosis, adherences related to the history of radiotherapy and surgery, the presence of traqueostomy, and identified infecting bacteria, a posterior fixation without anterior debridement and intravenous antibiotic specific therapy can could be a successful treatment.

This study was performed in line with the principles of the Declaration of Helsinki.

According to Institutional Review Board, the informed consent is not required.

Authors consent to publish this report in NEUROCIRUGIA (ASTUR).

The authors have no relevant financial or non-financial interests to disclose.

This research did not receive specific support from public sector agencies, the commercial sector, or non-profit organizations.

Not applicable. All data are in the text

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All authors contributed equally to this paper.