A, Computed tomographic image of the cervical spine, sagittal view, demonstrates spondylodiscitis with bony destruction. B, A T1-weighted postcontrast magnetic resonance image shows enhanced epidural abscess.
Computed tomographic image of the cervical spine, sagittal view, shows appropriate bony fusion following iliac crest bone graft reconstruction.
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D’Souza JN, Luginbuhl AJ, Goldman RA, Heller JE, Curry JM, Cognetti DM. Cervical Spine Spondylodiscitis After Esophageal Dilation in Patients With a History of Laryngectomy or Pharyngectomy and Pharyngeal Irradiation. JAMA Otolaryngol Head Neck Surg. 2016;142(5):467–471. doi:https://doi.org/10.1001/jamaoto.2015.3038
Dysphagia is a frequently reported sequela of treatment for head and neck cancer and is often managed with esophageal dilation in patients with dysphagia secondary to hypopharyngeal or esophageal stenosis. Reported complications of esophagoscopy with dilation include bleeding, esophageal perforation, and mediastinitis. These, though rare, can lead to substantial morbidity or mortality. In patients who have undergone irradiation, tissue fibrosis and devascularization may contribute to a higher incidence of these complications.
To describe the occurrence of cervical spine spondylodiscitis (CSS) following esophageal dilation in patients with a history of laryngectomy or pharyngectomy and irradiation with or without chemotherapy.
Design, Setting, and Participants
Medical records from a 5-year period (January 1, 2009, through December 31, 2014) in an academic tertiary care center were searched for patients with a history of laryngopharyngeal irradiation and a diagnosis of CSS following esophageal dilation. Four eligible patients were identified.
Main Outcomes and Measures
Recognition and treatment of CSS in the study population.
A total of 1221 patients underwent esophageal dilation for any reason. Of these, 247 patients carried a diagnosis of head and neck cancer at the following sites: piriform sinus, larynx, hypopharynx, epiglottis, oropharynx, base of the tongue, and tonsil. Of these, 4 patients with a diagnosis of CSS following esophageal dilation were included in this assessment. Prompt diagnosis and multidisciplinary management of CSS with intravenous antibiotics as well as spinal surgical debridement and stabilization led to recovery of full ability to take food by mouth in 3 of the 4 included patients. One patient remained dependent on the feeding tube.
Conclusion and Relevance
In patients with a history of laryngopharyngeal irradiation and esophageal dilation, complaints of neck pain or upper extremity weakness should trigger immediate evaluation for CSS; if present, prompt therapy is essential for prevention of substantial morbidity and mortality.
Head and neck cancer therapy has traditionally involved multimodality treatment including surgical resection, irradiation, and chemotherapy. The rise in use of chemoradiation as the primary treatment is owing to a preference for organ-sparing therapy.1 Primary surgical treatment with adjuvant therapy for advanced-stage head and neck cancer and salvage laryngectomy in patients who develop recurrence after prior chemoradiation or radiation alone are mainstays of management. Despite the refined delivery of radiation, critical structures necessary for normal deglutition are affected during primary or adjuvant treatment resulting in soft-tissue fibrosis, edema, and ultimately dysphagia.2,3 Patient factors known to increase the risk of permanent dysphagia after irradiation include older patient age, primary site in the larynx or hypopharynx, larger tumor size, larger radiation portal size, and higher radiation dose to the pharyngeal constrictors.4
The severity of dysphagia after head and neck cancer treatment is increasingly becoming a measure of functional outcome.5 It is estimated that up to 59% of patients undergoing irradiation for head and neck carcinoma have some degree of oropharyngeal dysfunction and aspiration.6 Pharyngeal or esophageal stenosis is seen in 15% to 30% of patients who undergo primary or salvage laryngectomy.7,8 The management of dysphagia includes modified swallow techniques, neuromuscular electrical stimulation, diet modifications, and surgical procedures. Esophagoscopy with bougie dilation is a frequently used for postradiation dysphagia and carries an increased risk of esophageal injury ranging from esophageal microtrauma to full-thickness perforation.9 Compromised vascularity in irradiated tissues may allow persistent infection to develop at these vulnerable sites of microtrauma. Cervical spine spondylodiscitis (CSS) following hypopharyngeal dilation is rare and has not been well described in the literature.9
In this case series, we describe 4 patients with history of laryngopharyngeal irradiation for head and neck cancer and development of CSS within weeks to months of flexible esophagoscopy with bougie dilation for dysphagia.
This study was approved by the Thomas Jefferson University Hospital (Jefferson Hospital) institutional review board, which waived written informed consent.
A retrospective review of the Jefferson Hospital electronic medical records for adults between January 1, 2009, and December 31, 2014, revealed that 1221 patients underwent esophageal dilation for any reason. Of these, 247 patients carried a diagnosis of head and neck cancer at the following sites: piriform sinus, larynx, hypopharynx, epiglottis, oropharynx, base of the tongue, and tonsil. Medical records were reviewed for history of laryngopharyngeal irradiation, diagnosis of CSS, timing of CSS presentation from dilation, cervical neck level of CSS, clinical symptoms, surgical management of the spine, surgical management of the pharynx, microbial cultures, and clinical outcome. Of the 247 patients with head and neck cancer and esophageal dilation performed at Jefferson Hospital, 3 were subsequently diagnosed with CSS. In addition, 1 patient with a history of laryngopharyngeal irradiation who underwent esophageal dilation at another institution and was referred to Jefferson Hospital for management of subsequent CSS was included.
A man in his 70s with a history of laryngeal cancer had been treated with a 66-Gy dose of radiation to the larynx 20 years prior to presentation. Ten years after that, he experienced a second occurrence of laryngeal cancer, which was treated with total laryngectomy and adjuvant chemoradiotherapy (radiation dose unavailable). Approximately 1 year prior to presentation, he began to experience progressive dysphagia, and he was treated with flexible esophagoscopy with serial bougie dilations every 3 to 5 weeks. Several months into this course of treatment, he began to experience neck pain and fevers; imaging revealed a retropharyngeal collection, raising concern for CSS. He was considered a high operative risk and was managed medically with antibiotics.
When he presented to our institution for a second opinion, imaging revealed progressive destruction in the cervical vertebral bodies and epidural abscess. After multidisciplinary consultation, the neurosurgery and head and neck services collaborated to perform surgical debridement and reconstruction procedures. The details of operative management and outcome are listed in the Table. An infectious disease consultation was obtained, and a polymicrobial infection was diagnosed; he began a 6-week intravenous regimen of daptomycin, metronidazole, and ciprofloxacin. Subsequently, he was switched to an oral ciprofloxacin and sulfamethoxazole-trimethoprim regimen, and a plan was developed for lifelong antibiotic suppression therapy under the guidance of treating infectious disease team. He remained dependent on a percutaneous endoscopic gastrostomy (PEG) tube for nutrition but eventually was able to take a limited diet by mouth. He died of unrelated causes 1 year later.
A man in his 70s with a history of laryngeal cancer treated with total laryngectomy and conventional radiation therapy (dose unavailable) developed progressive dysphagia 24 years after treatment. He underwent flexible esophagoscopy with bougie dilation of a hypopharyngeal stricture without perioperative antibiotics to a maximum dilation of 48 Fr (16 mm).
Ten days after this dilation surgery, he presented to the emergency department with fever (40°C) and complained of increasing neck pain not relieved by narcotic pain medication. He was found to be hypotensive and bacteremic. Computed tomographic (CT) and magnetic resonance imaging (MRI) scans of the spine revealed retropharyngeal air, C6-C7 CSS, and a communicating epidural abscess. Two prominent osteophytes were implicated in this perforation.
His medical and surgical management is detailed in the Table. Immediately thereafter, and following spine cultures positive for polymicrobial bacteria plus Candida albicans, he began a 6-week regimen of intravenous ertapenem and fluconazole. He recovered well from surgery and returned to an oral diet.
A man in his 60s with a history of T2N2bM0 squamous cell carcinoma of the pyriform sinus was treated with transoral robotic resection, neck dissection, and adjuvant chemoradiotherapy. He received 60 Gy of radiation to the primary site, and each side of his neck was treated with 56 to 60 Gy following surgical resection. He developed a hypopharyngeal stricture and dysphagia 3 months after treatment completion for which he underwent flexible esophagoscopy with bougie dilation with perioperative antibiotics. Postdilation esophagoscopy confirmed no evidence of perforation or mucosal injury. He was noted on biopsy to have a persistent hypopharyngeal radiation ulcer without evidence of cancer recurrence.
Seven weeks later he presented to another facility with upper extremity weakness, and imaging revealed an epidural abscess, bone destruction, discitis, and retropharyngeal air. He underwent urgent drainage along with decompression and an anterior cervical fusion. Postoperatively, he developed a pharyngocutaneous fistula through his neck wound and was transferred to our facility for further management.
Examination and imaging revealed a pharyngeal defect and retropharyngeal collection, C3-C4 CSS, and enlargement of the epidural abscess. Again, after multidisciplinary consultation, the patient was managed medically and surgically as summarized in the Table. Immediately thereafter, he began a 6-week regimen of intravenous ceftriaxone and metronidazole, after which he began a long-term regimen of oral moxifloxacin. He experienced a full neurologic recovery and was able to resume an oral diet.
A man in his 70s with a history of tongue base cancer treated with radiation therapy (doses at the primary site to 66 Gy and neck to 60 Gy) subsequently developed laryngeal cancer, which was treated with laser excision. He experienced recurrence of the laryngeal cancer 5 years later and underwent total laryngectomy and reirradiation to 60 Gy. He developed significant dysphagia 5 months after treatment completion and underwent hypopharyngeal dilation by flexible esophagoscopy with bougienage uneventfully, with improvement in swallowing function. He did receive preoperative antibiotics, and no laceration or perforation was seen on postdilation esophagoscopy.
Approximately 10 weeks following dilation, a surveillance positron emission tomographic–CT scan revealed intense activity in the lower cervical spine. At that time, the patient was experiencing neck pain but had no neurological deficits. A follow-up MRI of the spine revealed C5-C6 CSS (Figure 1), and he was medically and surgically managed similarly to cases 1 through 3, as summarized in the Table. The pharynx remained intact, and no soft-tissue reconstruction was required. Cervical spine stabilization was carried out with iliac crest bone graft (Figure 2). Postoperatively, and after speciation of spine cultures, he began a 6-week regimen of intravenous ciprofloxacin. He recovered well and returned to an oral diet.
Dysphagia is a well-known complication of head and neck cancer treatment. It affects patient nutrition, healing, and quality of life. Dysphagia in posttreatment head and neck cancer is multifactorial. Critical structures such as the tongue, larynx, and pharyngeal musculature receive high doses of radiation during the course of treatment resulting in mucositis, denervation atrophy, and fibrosis. Use of concurrent chemotherapy is also associated with higher rates of long-term dysphagia.3 Studies of have found that lower radiation doses are associated with lower rates of dysphagia, and doses over 60 Gy are significantly associated with an increased risk of stricture, PEG tube dependence, and aspiration.10,11 Strictures of the hypopharynx and upper esophagus are mainly due to fibrosis caused by progressive obliterative endarteritis leading to esophageal wall ischemia, and over time, the damaged tissue is replaced by dense collagen scar.12,13 On esophagoscopy, the presentation of these conditions can range from membranous rings to total obliteration of esophageal lumen.14
Esophageal stricture after irradiation has been considered a low-frequency complication, generally estimated at less than 3% of all patients treated for head and neck cancer. A 2008 retrospective study by Vu et al15 postulated a much higher rate of upper esophageal stricture and stenosis following total laryngectomy and irradiation: 44% of their patients complained of posttreatment dysphagia, and on barium esophagogram, 33% of the entire study population had an esophageal stricture.
Operative examination with flexible or rigid esophagoscopy is the method of choice to fully evaluate persistent dysphagia. Reported complications of esophageal dilation include bleeding, esophageal perforation, and mediastinitis. In esophagoscopy with dilation, microtrauma caused by dilators may cause bacterial translocation and seeding of adjacent tissues. Transient bacteremia associated with esophagoscopy with dilation has been estimated to occur in 45% to 100% of procedures.9
Esophageal perforation in the cervical spine most commonly takes place at the Killian triangle, an area of natural weakness between the inferior pharyngeal constrictor and the cricopharyngeus muscle. The Killian triangle is found in close proximity to C4-C6 anterior vertebral bodies, placing them at risk when infectious organisms are introduced through microtrauma or frank perforation.16 In this region, the esophagus directly overlies the cervical vertebrae, which are covered only by a thin layer of prevertebral fascia.17 In laryngopharyngectomy patients, the Killian triangle no longer exists, and the anatomical area of weakness is located at the neopharyngeal anastomosis site. Here again, the neopharynx-esophagus junction directly overlies the cervical vertebrae, with thin prevertebral fascia alone as a barrier to bacterial spread.
Vertebral spondylodiscitis or osteomyelitis is a relatively uncommon condition in general, and only 6.5% of all cases are identified within the cervical spine.18 Mullen et al16 describe 7 cases of CSS directly linked to esophageal perforation following dilation or stenting. The CSS mortality rate remains high owing to its frequently insidious presentation leading to a delay in diagnosis. Patients with CSS are at risk for extension of the infection into an epidural abscess with resultant paralysis requiring immediate attention.
Management of CSS is dependent on patient factors such as neurologic deficits, spinal instability, and overall clinical status. Medical management options include appropriate intravenous antibiotics, analgesia, and immobilization of the cervical spine using a collar or a halo vest. Choice of antibiotics is typically dependent on organisms isolated from cultures; in empirical treatment, antibiotics used typically target Staphylococcus aureus, the most frequent isolate in pyogenic vertebral spondylodiscitis.17 In the postdilation patient population, additional organisms isolated are typically polymicrobial, including Enterococcus, Proteus, and Fusobacterium species.16 Surgical management is considered after the failure of antibiotic therapy or in cases of progressive neurologic symptoms, spinal instability, bony destruction, or identification of an epidural or paravertebral abscess on imaging.8,13,14 Surgical management should combine repair of pharyngeal defect, typically with the use of a muscle flap, with simultaneous debridement and stabilization of cervical spine.
The 4 patients in this series presented with upper extremity and/or neck complaints, fever, and a recent history of esophageal dilation. In all cases, imaging showed evidence of CSS. All patients required operative stabilization by a spine team. In 3 of the 4 cases, a pharyngeal defect was identified, requiring a muscular flap repair.
Once operative repair with both spine stabilization and pharyngeal coverage was performed, all patients were managed with bacterial culture–appropriate, long-term intravenous antibiotics under the guidance of a treating infectious disease team, enteral access via PEG tube or nasogastric tube, and nothing by mouth status. Before resumption of oral intake, swallow studies were performed to confirm lack of residual leak. Of the 4 patients described, 3 survived with no residual dysphagia, and 1 died of unrelated causes 1 year after spine stabilization.
Dysphagia is a common complaint following laryngectomy and irradiation for head and neck cancer. Patients with a history of laryngopharyngeal irradiation and esophageal dilation should be considered at high risk for complications, including CSS. Clinical complaints and symptoms, even in a delayed setting, warrant a high index of suspicion for serious infectious complications and require prompt evaluation and management to avoid serious, even life-threatening sequelae.
Accepted for Publication: October 12, 2015.
Corresponding Author: Jill N. D’Souza, MD, Department of Otolaryngology–Head and Neck Surgery, Thomas Jefferson University Hospital, 925 Chestnut St, Sixth Floor, Philadelphia, PA 19147 (email@example.com).
Published Online: March 24, 2016. doi:10.1001/jamaoto.2015.3038.
Author Contributions: Dr D’Souza had full access to all of the data in the study and takes responsibility for the integrity of the data and the accuracy of the data analysis.
Study concept and design: D’Souza, Luginbuhl, Goldman, Curry, Cognetti.
Acquisition, analysis, or interpretation of data: D’Souza, Goldman, Heller, Curry, Cognetti.
Drafting of the manuscript: D’Souza, Luginbuhl, Goldman, Curry, Cognetti.
Critical revision of the manuscript for important intellectual content: Luginbuhl, Goldman, Heller, Curry, Cognetti.
Statistical analysis: Luginbuhl, Cognetti.
Administrative, technical, or material support: D’Souza, Curry, Cognetti.
Study supervision: Luginbuhl, Goldman, Heller, Cognetti.
Conflict of Interest Disclosures: None reported.
Previous Presentation: This research was presented as a poster at the 2014 American Head and Neck Society Meeting; July 28-30, 2014; New York, New York.
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