Spine Surgery and Related Research
The Japanese Society for Spine Surgery and Related Research
image
Transoral Mandibular Tongue-Splitting Approach in Upper Cervical Epidural Abscess: A Case Report and Review of the Literature
Volume: 4 , Issue: 2
Doi: 10.22603/ssrr.2019-0090

Table of Contents

Highlights

Notes

Abstract

Introduction The transoral mandibular tongue-splitting approach is typically performed for the treatment of upper cervical tumor and instability but has not been performed for the treatment of upper cervical epidural abscess (UCEA). We report the first case of UCEA successfully treated with a transoral mandibular tongue-splitting approach. Technical Note A 62-year-old man who had medical histories of tracheotomy with intubation and dermatopathy due to radiation therapy for the treatment of nasopharyngeal carcinoma presented with neck pain and limb weakness. The imaging examination revealed bone erosion of C2-C4 vertebrae and abscess at the level of C2-C4, supporting a diagnosis of UCEA. The transcervical approach could not be used for treatment; therefore, the transoral mandibular tongue-splitting approach was used successfully to perform decompression, debridement, and iliac bone grafting. Subsequently, we reviewed the literature pertaining to the use of the transoral mandibular tongue-splitting approach. The approach can be invasive and cause some complications. However, no fatal complications have been reported, and all patients demonstrated a favorable neurological outcome with reduced neurological deficits. Conclusions This case and subsequent literature review suggest that the transoral mandibular tongue-splitting approach may be effective for the improvement of neurological outcomes without fatal complications in patients with UCEA. There may be an increasing number of patients with UCEA requiring the transoral mandibular tongue-splitting approach due to the increasing prevalence of immunocompromized status and the aging population.

Keywords
Kobayashi, Morimoto, Maeda, Toda, Hirata, Yoshihara, and Mawatari: Transoral Mandibular Tongue-Splitting Approach in Upper Cervical Epidural Abscess: A Case Report and Review of the Literature

Introduction

The transoral mandibular tongue-splitting approach has been performed mainly for the treatment of upper cervical tumor1-6). However, the approach is technically demanding and invasive with many possible complications1-6). Thus, the indication may be limited to patients requiring surgical treatment with a wide operative field and anterior stabilization. An upper cervical epidural abscess (UCEA) can cause fatal respiratory failure and/or tetraplegia, and emergent surgical intervention is required in patients with progressive limb paralysis7). Although the transoral approach is frequently used for the treatment of UCEA8-17), it may be inappropriate if a wide operative field and anterior stabilization are required18-27). For such patients, the transoral mandibular tongue-splitting approach may be useful, but it has not been used for the treatment of UCEA due to its invasiveness and possible complications18-27). Here, we describe a patient with UCEA who was successfully treated via transoral mandibular tongue-splitting approach.

Technical Note

A 62-year-old man presented with neck pain and limb weakness. He had medical histories of tracheotomy with intubation and dermatopathy due to radiation therapy for the treatment of nasopharyngeal carcinoma (Fig. 1). Laboratory tests revealed an elevated inflammatory response, and computed tomography (CT) of the cervical spine demonstrated bone erosion in C2-C4 vertebrae (Fig. 2A). Subsequent T2-weighted magnetic resonance imaging revealed a high signal intensity lesion (Fig. 2B), indicating an abscess in C2-C4. Bacterial cultures were negative. The patient was diagnosed with UCEA, and intravenous antibiotics were administered (tazobactam/piperacillin). However, the patient exhibited deterioration of limb weakness, and repeat CT revealed progression of the erosion in C2-C4 vertebrae, as well as UCEA-related spinal cord compression. To prevent progressive neurological deficits due to the abscess, the patient underwent decompression, debridement, and iliac bone grafting by the transoral mandibular tongue-splitting approach.

Post-tracheotomy, post-intubation, and post-dermatopathy due to radiation therapy for the treatment of nasopharyngeal carcinoma.
Figure 1.
Post-tracheotomy, post-intubation, and post-dermatopathy due to radiation therapy for the treatment of nasopharyngeal carcinoma.
Sagittal plane of CT scan before treatment (A). Sagittal T2-weighted MRI before treatment (B). These images show bone erosion of C2-C3 vertebrae (A) and cervical epidural abscess at the C2-C3 levels (B).
Figure 2.
Sagittal plane of CT scan before treatment (A). Sagittal T2-weighted MRI before treatment (B). These images show bone erosion of C2-C3 vertebrae (A) and cervical epidural abscess at the C2-C3 levels (B).

A Nelaton tube was inserted into the nasal cavity and advanced to the oral cavity to elevate the soft palate. Transoral mandibular tongue splitting (Fig. 3) was achieved by an otolaryngologist and an oral surgeon. After debridement of the UCEA and infected granulation tissue, C2 vertebrae were excavated. Iliac bone grafting was performed to stabilize the excavated C2 vertebrae. Soft tissues and tongue were sutured, followed by mandibular fixation using a titanium miniplate. A halo-vest was placed to prevent postoperative dislocation of the bone graft.

Transoral mandibular tongue-splitting approach. Median mandibular splitting, median longitudinal incision of the tongue, partial resection of eroded lesion of C2-C3 vertebrae, debridement of the abscess, suturing tongue, and titanium plate fixation of the mandible were performed.
Figure 3.
Transoral mandibular tongue-splitting approach. Median mandibular splitting, median longitudinal incision of the tongue, partial resection of eroded lesion of C2-C3 vertebrae, debridement of the abscess, suturing tongue, and titanium plate fixation of the mandible were performed.

Operative culture revealed Pseudomonas aeruginosa , and high-dose intravenous empirical antibiotic therapy was continued. One week after the first operation (Fig. 4A), the patient underwent posterior occipitocervical arthrodesis (Oc-C4) with instrumentation (Synthes GmbH., Eimattstrasse, Oberdorf, Switzerland) (Fig. 4B).

Postoperative images. (A) Sagittal plane of CT scan. (B) Lateral radiograph.
Figure 4.
Postoperative images. (A) Sagittal plane of CT scan. (B) Lateral radiograph.

His symptoms, such as severe cervical pain and neurological deficit, rapidly improved following surgery. Perioperative incidental dural tear was detected, which may have been influenced by radiation therapy28). There were some minor complications, such as limited jaw mobility and superficial mucosal infections. However, meningitis, malocclusion, and a reduction in the occlusal force were not noted. Intravenous antibiotics were switched to oral administration of ciprofloxacin for a total of 6 months, with normalization of the inflammatory response and imaging confirmation of abscess disappearance. Although a bridging callus was not noted due to radiation therapy, the dislocation of the bone graft and implant was not detected at 2 months after surgery. Despite this progress, the patient died of progressive nasopharyngeal carcinoma 6 months postoperatively.

Discussion

The transoral mandibular tongue-splitting approach is used mainly for tumor resection and stabilization of the upper cervical spine1-6). Despite its wide operative field, it is technically demanding and invasive with many complications1-6).

We reviewed the literature pertaining to the transoral mandibular tongue-splitting approach (Table 1). In all reported cases, preoperative tracheotomy was performed to prevent respiratory failure1-6). Cervical instability was treated with single-stage or two-stage posterior instrumentation1-6). Considering the blood loss and surgical time, this approach could be invasive. However, the neurological outcome was favorable in all patients1-6). Four of 19 patients died of progressive cancer at >4 months postoperatively; all patients without a history of cancer achieved full recovery.

Table 1.
Characteristics of Reported Cases of Transoral Mandibular Tongue-Splitting Approach.
ReferenceYearAge (years)/ SexPrimary diseaseLevelTracheotomySurgical procedureBlood loss (mL)Surgical time (h)Complication (n)
Vishteh et al1199911/MOccipitocervical instabilityOc-C2DoneSingle-stage procedure: transoral bilateral sagittal split mandibular osteotomy combined with soft palate-splitting approach (unspecified)Not availableNot availableLimited jaw mobility (4)
Superficial mucosal infection (2)
Macroglossia (1)
Micrognathia (1)
Retrognathia (1)
20/FKlippel-Feil anomalyOc-C2DoneNot availableNot available
49/FRheumatoid arthritis/basilar invaginationOc-C2DoneNot availableNot available
68/FRheumatoid arthritis/basilar invaginationOc-C2DoneNot availableNot available
Hiromasa et al2201223/FUpper cervical tumorC2-C3DoneTwo-stage procedure: first stage, posterior debridement and fusion; second stage, transoral mandibular tongue-splitting extirpation (unspecified)Not availableNot availableNone
65/MUpper cervical tumorC1-C2DoneTwo-stage procedure: first stage, posterior debridement and fusion; second stage, transoral mandibular soft palate-splitting extirpation (unspecified)Not availableNot available
66/FUpper cervical tumorC2-C3DoneTwo-stage procedure: first stage, posterior debridement and fusion; second stage, transoral mandibular tongue-splitting extirpation (unspecified)Not availableNot available
Ortega-Porcayo et al3201443/MUpper cervical tumorC2-C4DoneTwo-stage procedure: first stage, posterior bilateral laminectomies and facetectomies of C2-C4 and instrumentation (Occiput-C5-C6-C7); second stage, transoral mandibular tongue-splitting approach with anterior screw-plate fixation (C2-C4)21009None
23/FUpper cervical tumorC2-C3DoneTwo-stage procedure: first stage, posterior bilateral laminectomies and facetectomies of C2-C4 and instrumentation (C1-C3-C4); second stage, transoral mandibular tongue-splitting approach using anterior titanium cage with bone matrix fixation (C1-C4)9005
Logroscino et al4200459/MUpper cervical tumorC2DoneTwo-stage procedure: first stage, transoral mandibular tongue-splitting approach with anterior screw-plate fixation (C2); second stage, posterior instrumentation (Occiput-C2-C3-C4)Not availableNot availableNone
63/FUpper cervical tumorC2DoneNot availableNot available
Stulík et al5200727/MUpper cervical tumorC2DoneTwo-stage procedure: first stage, posterior bilateral laminectomies and facetectomies of C2 and instrumentation (C1-C3-C4); second stage, transoral mandibular tongue-splitting approach using anterior cage with iliac bone graft fixation (C1-C3)3008Superficial mucosal infection (1)
Liquorrhea (1)
Menon et al6201935/MOccipitocervical instabilityC2-C3DoneSingle-stage procedure: transoral mandibular tongue-splitting approach with anterior screw and/or plate fixation (C2), with/without posterior instrumentation (unspecified)Not availableNot availableSuperficial mucosal infection (1)
26/MOccipitocervical instabilityC2-C3DoneNot availableNot available
46/FUpper cervical tumorC2DoneNot availableNot available
67/MUpper cervical tumorC2DoneNot availableNot available
38/MUpper cervical tumorC2DoneNot availableNot available
51/MUpper cervical tumorC2DoneNot availableNot available
Current case62/MUpper cervical epidural abscessC2-C4DoneTwo-stage procedure: first stage, transoral mandibular tongue-splitting approach with iliac bone graft (C3); second stage, posterior instrumentation (Occiput-C2-C3-C4-C5)2847.5Limited jaw mobility (1)
Superficial mucosal infection (1)

Some authors have reported that UCEA was successfully treated by posterior stabilization and decompression, which provides a wide operative field and is a common approach for orthopedic surgeon13,29). However, we selected transoral mandibular tongue-splitting approach for five reasons in this case: first, anterior debridement and stabilization were needed, considering the abscess location and C2-C3 vertebral erosion. Second, posterior stabilization and decompression risked promoting implant infection. Third, the transcervical approach was contraindicated due to neck dermatopathy after radiation therapy. Fourth, postoperative management was relatively simple in terms of tracheotomy and intubation, which was placed for the treatment of nasopharyngeal carcinoma. Fifth, cooperation between otolaryngologists and oral surgeons is necessary in our hospital to successfully perform the technique.

The incidence of spinal epidural abscesses has increased due to the increasing prevalence of immunocompromized status and the aging population30,31). Moreover, the main source of UCEA is contiguous spread, such as that in the case of otorhinolaryngologic disease and tooth extraction8-27). Thus, there may be an increasing number of patients with UCEA requiring anterior debridement and stabilization.

This is the first report of the successful treatment of UCEA with transoral mandibular tongue-splitting approach, which may prevent neurological deficits without fatal complications in patients with UCEA.

Conflicts of Interest: The authors declare that there are no relevant conflicts of interest.

Author Contributions: Takaomi Kobayashi, Tadatsugu Morimoto, and Kazumasa Maeda wrote and prepared the manuscript, and all of the authors participated in the study design. All authors have read, reviewed, and approved the article.

Informed Consent: The patient and his family provided consent for submission of the case for publication.

References

1. 

Vishteh AG, Beals SP, Joganic EF, et al. Bilateral sagittal split mandibular osteotomies as an adjunct to the transoral approach to the anterior craniovertebral junction. Technical note. J Neurosurg. 1999;90(2):267-70.

2. 

Hiromasa K, Shin K, Seiji A, et al. Transoral anterior approach using median mandibular splitting in upper spinal tumor extirpation. Oral Surg Oral Med Oral Pathol Oral Radiol. 2012;114(5):e12-6.

3. 

Ortega-Porcayo LA, Cabrera-Aldana EE, Arriada-Mendicoa N, et al. Operative technique for en bloc resection of upper cervical chordomas: Extended transoral transmandibular approach and multilevel reconstruction. Asian Spine J. 2014;8(6):820-6.

4. 

Logroscino CA, Casula S, Rigante M, et al. Transmandible approach for the treatment of upper cervical spine metastatic tumors. Orthopedics. 2004;27(10):1100-3.

5. 

Stulík J, Kozák J, Sebesta P, et al. Total spondylectomy of C2: a new surgical technique. Acta Chir Orthop Traumatol Cech. 2007;74(2):79-90.

6. 

Menon KV, Al Saqri H, Kumar R, et al. The median labio-mandibulo-glossotomy approach to the upper cervical spine: A personal series and tips and pearls. Indian Spine J. 2019;2(1):92-8.

7. 

Kobayashi T, Ureshino H, Hotta K, et al. Timing of surgical interventions for upper cervical epidural abscess: a case report and review of the literature. Eur J Orthop Surg Traumatol. 2019;29(6):1365-6.

8. 

Keogh S, Crockard A. Staphylococcal infection of the odontoid peg. Postgrad Med J. 1992;68(795):51-4.

9. 

Suchomel P, Buchvald P, Barsa P, et al. Pyogenic osteomyelitis of the odontoid process: single stage decompression and fusion. Spine (Phila Pa 1976). 2003;28(12):E239-44.

10. 

Reid PJ, Holman PJ. Iatrogenic pyogenic osteomyelitis of C1 and C2 treated with transoral decompression and delayed occipitocervical arthrodesis. Case report. J Neurosurg Spine. 2007;7(6):664-8.

11. 

Ruskin J, Shapiro S, McCombs M, et al. Odontoid osteomyelitis. An unusual presentation of an uncommon disease. West J Med. 1992;156(3):306-8.

12. 

Wiedau-Pazos M, Curio G, Grüsser C. Epidural abscess of the cervical spine with osteomyelitis of the odontoid process. Spine (Phila Pa 1976). 1999;24(2):133-6.

13. 

Zigler JE, Bohlman HH, Robinson RA, et al. Pyogenic osteomyelitis of the occiput, the atlas, and the axis. A report of five cases. J Bone Joint Surg Am. 1987;69(7):1069-73.

14. 

Young WF, Weaver M. Isolated pyogenic osteomyelitis of the odontoid process. Scand J Infect Dis. 1999;31(5):512-5.

15. 

Burns TC, Mindea SA, Pendharkar AV, et al. Endoscopic transnasal approach for urgent decompression of the craniocervical junction in acute skull base osteomyelitis. J Neurol Surg Rep. 2015;76(1):e37-42.

16. 

Kubo S, Takimoto H, Hosoi K, et al. Osteomyelitis of the odontoid process associated with meningitis and retropharyngeal abscess-case report. Neurol Med Chir (Tokyo). 2002;42(10):447-51.

17. 

Kurimoto M, Endo S, Ohi M, et al. Pyogenic osteomyelitis of an invaginated odontoid process with rapid deterioration of high cervical myelopathy: A case report. Acta Neurochir (Wien). 1998;140(10):1093-4.

18. 

Curry JM, Cognetti DM, Harrop J, et al. Cervical discitis and epidural abscess after tonsillectomy. Laryngoscope. 2007;117(12):2093-6.

19. 

Deshmukh VR. Midline trough corpectomies for the evacuation of an extensive ventral cervical and upper thoracic spinal epidural abscess. J Neurosurg Spine. 2010;13(2):229-33.

20. 

Bartels JW, Brammer RE. Cervical osteomyelitis with prevertebral abscess formation. Otolaryngol Head Neck Surg. 1990;102(2):180-2.

21. 

Mirouse G, Journe A, Casabianca L, et al. Bartonella henselae osteoarthritis of the upper cervical spine in a 14-year-old boy. Orthop Traumatol Surg Res. 2015;101(4):519-22.

22. 

Anton K, Christoph R, Cornelius FM. Osteomyelitis and pathological fracture of the axis. Case illustration. J Neurosurg. 1999;90(1):162.

23. 

Al-Hourani K, Frost C, Mesfin A. Upper cervical epidural abscess in a patient with Parkinson disease: a case report and review. Geriatr Orthop Surg Rehabil. 2015;6(4):328-33.

24. 

Haridas A, Walsh DC, Mowle DH. Polymicrobial osteomyelitis of the odontoid process with epidural abscess: case report and review of literature. Skull Base. 2003;13(2):107-11.

25. 

Paul CA, Kumar A, Raut VV, et al. Pseudomonas cervical osteomyelitis with retropharyngeal abscess: an unusual complication of otitis media. J Laryngol Otol. 2005;119(10):816-8.

26. 

Papp Z, Czigléczki G, Banczerowski P. Multiple abscesses with osteomyelitis and destruction of both the atlas and the axis in a 4-week-old infant. Spine (Phila Pa 1976). 2003;38(19):E1228-30.

27. 

Aranibar RJ, Del Monaco DC, Gonzales P. Anterior microscopic transtubular (MITR) surgical approach for cervical pyogenic C1-2 abscess: A case report. Int J Spine Surg. 2015;9:56.

28. 

Yokogawa N, Murakami H, Demura S, et al. Postoperative cerebrospinal fluid leakage associated with total en bloc spondylectomy. Orthopedics. 2015;38(7):e561-6.

29. 

Fukutake T, Kitazaki H, Hattori T. Odontoid osteomyelitis complicating pneumococcal pneumonia. Eur Neurol. 1998;39(2):126-7.

30. 

Baker AS, Ojemann RG, Swartz MN, et al. Spinal epidural abscess. N Engl J Med. 1975;293(10):463-8.

31. 

Vakili M, Crum-Cianflone NF. Spinal epidural abscess: a series of 101 cases. Am J Med. 2017;130(12):1458-63.

https://www.researchpad.co/tools/openurl?pubtype=article&doi=10.22603/ssrr.2019-0090&title=Transoral Mandibular Tongue-Splitting Approach in Upper Cervical Epidural Abscess: A Case Report and Review of the Literature&author=Takaomi Kobayashi,Tadatsugu Morimoto,Kazumasa Maeda,Yu Toda,Hirohito Hirata,Tomohito Yoshihara,Masaaki Mawatari,&keyword=upper cervical epidural abscess (UCEA),transoral approach,mandibular tongue-splitting,transcervical approach,neurological outcome,complication,&subject=Technical Note,