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| CASE REPORT |
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| Year : 2023 | Volume
: 14
| Issue : 1 | Page : 93-96 |
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Osteoradionecrosis of the occipitocervical junction: A rare case of C1 anterior arch disruption
Masaki Sakamoto1, Takayoshi Shimizu2, Atsushi Suehiro3, Shuichi Matsuda2
1 Department of Orthopaedic Surgery, Kyoto University, Graduate School of Medicine, Kyoto, Japan
2 Department of Orthopedic Surgery, Kyoto University Graduate School of Medicine, Kyoto, Japan
3 Department of Otolaryngology-Head and Neck Surgery, Kyoto University Graduate School of Medicine, Kyoto, Japan
| Date of Submission | 21-Nov-2022 |
| Date of Acceptance | 04-Dec-2022 |
| Date of Web Publication | 13-Mar-2023 |
Correspondence Address:
Takayoshi Shimizu
Department of Orthopedic Surgery, Kyoto University Graduate School of Medicine, 54 Kawahara-Cho, Shogoin, Sakyo, Kyoto 606-8507
Japan
 Source of Support: None, Conflict of Interest: None  | Check |
DOI: 10.4103/jcvjs.jcvjs_145_22
 Abstract | | |
We report the case of a 57-year-old man who developed osteoradionecrosis (ORN) at the occipitocervical (OC) junction after radiation therapy for nasopharyngeal carcinoma. During soft-tissue debridement using a nasopharyngeal endoscope, the anterior arch of the atlas (AAA) was spontaneously disrupted, which was later spat out. Radiographic examination revealed complete disruption of the AAA that caused OC instability. We performed posterior OC fixation. The patient experienced successful postoperative pain relief. AAA disruption secondary to ORN at OC junction can cause severe instability. Posterior OC fixation alone may be an effective procedure if the necrotic pharyngeal region is mild and endoscopically controllable.
Keywords: Anterior arch, occipitocervical, osteoradionecrosis, posterior fixation
How to cite this article:
Sakamoto M, Shimizu T, Suehiro A, Matsuda S. Osteoradionecrosis of the occipitocervical junction: A rare case of C1 anterior arch disruption. J Craniovert Jun Spine 2023;14:93-6 |
How to cite this URL:
Sakamoto M, Shimizu T, Suehiro A, Matsuda S. Osteoradionecrosis of the occipitocervical junction: A rare case of C1 anterior arch disruption. J Craniovert Jun Spine [serial online] 2023 [cited 2023 Mar 25];14:93-6. Available from: https://www.jcvjs.com/text.asp?2023/14/1/93/371564 |
| Introduction | |  |
Osteoradionecrosis (ORN) commonly occurs in the mandible as a complication of chemoradiotherapy for head-and-neck cancers. The etiology is ischemic necrosis and fibrosis of irradiated soft tissue and the subsequent necrosis of the adjacent bone nourished by it.[1] Only 0.7%–1% of patients with nasopharyngeal carcinoma develop ORN in the cervical spine, most of which are asymptomatic.[2],[3] ORN in the occipitocervical (OC) junction is rare, but anatomical destruction in this area can cause devastating OC instability and be life-threatening. While there have been case reports describing ORN in the OC junction (clivus, C1, or C2), the optimal surgical strategy remains unclear. We report a case in which the patient developed spontaneous disruption of the anterior arch of the atlas (AAA) secondary to ORN and presented with a unique clinical episode successfully treated with posterior OC fixation alone.
| Case Report | | |
A 57-year-old man was diagnosed with T3N1M0 poorly differentiated squamous cell carcinoma of the posterior nasopharyngeal wall and received 70 Gy of intensity-modulated radiation therapy combined with thrice-weekly cisplatin. A tracheotomy was performed because the soft tissue of the posterior pharyngeal wall was repeatedly infected, and the abscess flowed into the trachea, leading to aspiration pneumonitis. Although the patient experienced no recurrence later, he required periodic debridement through a nasopharyngeal endoscope for a few months. Five years later, when endoscopic debridement was performed, the otolaryngologist realized that a bone fragment had dropped from the posterior pharyngeal wall into the piriform sinus. At night, after he went home, he spat out the fragment. Subsequently, he gradually complained of neck pain which became intractable even with opioid use. Plain radiography and computed tomography (CT) revealed complete disruption of the anterior AAA [Figure 1]a, [Figure 1]b, [Figure 1]c. The ipsilateral C0-C1 facet spontaneously fused, likely due to osteoarthrosis after facet involvement in ORN [Figure 1]d. These findings were not observed in the images taken 5 years preoperatively [Figure 1]e and [Figure 1]f. The posterior AAA was simultaneously fractured [Figure 1]g. Magnetic resonance imaging (MRI) showed relatively low intensity in the C2 dens and clivus, indicating possible osteonecrosis, while the posterior structures were intact [Figure 1]h. [Figure 2]a and [Figure 2]b show the nasal endoscopic findings before the episode. The necrotic anterior arch was exposed from the posterior pharyngeal wall and was extremely unstable. We performed a micro-CT scan of the fragment that the patient brought to us and confirmed that it was the anterior AAA [Figure 2]c and [Figure 2]d. The patient was neurologically intact; however, his neck pain was severe. | Figure 1: (a) Preoperative lateral radiograph demonstrating a defect of the anterior arch of the atlas, (b and c) CT images showing anterior arch disruption, (d) the right C0-C1 facet is auto-fused (white arrow), (e and f) images taken 5 years preoperatively (intact anterior arch and C0-C1 facet), (g) fractured posterior arch of the atlas (white arrow), and (h) sagittal T1-weighted MR image showing low intensity in the clivus and C2 body, indicating osteonecrosis. CT: Computed tomography, MR: Magnetic resonance
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 | Figure 2: (a) Bone fragment in the epipharynx observed through nasal endoscope, (b) the fragment that was disrupted from the posterior pharyngeal wall (white arrow), (c) the bone fragment that the patient kept after they spat it out, and (d) a micro-CT image of the fragment. CT: Computed tomography
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Posterior OC fixation (C0-C2) was performed using an occipital plate and C2 pedicle screws. Because the C0-C1 facet was auto-fused, we preferred OC fixation rather than C1-C2 fixation. Supplemental C1-C2 posterior wiring with an iliac crest autograft was added. The patient achieved successful pain relief immediately postoperatively [Figure 3]a. One-year postoperative nasal endoscopy demonstrated the resolution of the soft-tissue defect in the posterior pharyngeal wall [Figure 3]b. One-year postoperative CT and magnetic resonance (MR) images showed no progression of the necrotic area in the clivus and C2 body but rather a slight improvement in T1-low intensity, which may indicate spontaneous bone healing [Figure 3]c and [Figure 3]d. The patient was completely free of pain medication use. | Figure 3: One-year postoperative images. (a) Lateral radiograph, (b) endoscopic image of the posterior pharyngeal wall showing resolution of soft-tissue defect at the area of disruption of the anterior arch of the atlas, (d) sagittal CT, and (d) MR images showing no progression of the necrotic area in the clivus and C2 body but rather a slight improvement of T1-low intensity. CT: Computed tomography, MR: Magnetic resonance
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| Discussion | | |
Chemoradiation therapy is one of the primary treatments for head-and-neck cancer.[4] However, irradiated patients may suffer from complications including skin necrosis, oral mucositis, and ORN (a postradiation complication first reported by Regaud in 1922).[5] The incidence of ORN caused by radiation therapy has been reported to be approximately 3%.[6] ORN commonly develops 2–4 years after the completion of radiation treatments. Previous studies have documented that over 60 Gy of radiation is a risk factor for ORN.[1],[7] Meanwhile, most patients with primary oral and oropharyngeal cancer undergo 60–70 Gy of radiation therapy; therefore, they are at high risk of developing ORN. ORN in the OC junction has rarely been reported. Most reported cases presented with neck pain, likely due to OC instability.[8] When a patient with a history of radiotherapy after head-and-neck cancer complains of neck pain, physicians should pay careful attention to ORN at the OC junction.
MRI is useful for diagnosis. Patients with ORN tend to show more contiguous involvement of the atlantoaxial or atlanto-occipital bones with intervening joint changes, reactive vertebral marrow edema, and reactive paravertebral muscle edema, while patients with metastasis tend to show cervical lymphadenopathy. MRI is also helpful in tracking postoperative spontaneous bone healing. Our case showed chronological improvement in MR intensity inside the affected C2 vertebra.
Treatment strategies should be carefully selected depending on the situation. Surgical intervention is selected for patients with OC (or cervical) instability, myelopathy, or severe radiculopathy. Conservative treatment, including antibiotic therapy, can be indicated in those without the abovementioned clinical signs and those who cannot tolerate surgery. The surgical methods differ on a case-by-case basis. Some reports used a free flap with vascularized fibula to reconstruct the posterior pharynx.[9] Although this may be necessary in cases of massive soft-tissue defects, spontaneous resolution of the soft tissue could be expected after OC stabilization with minimal endoscopic debridement, as observed in our case. Broad-spectrum antibiotics are often required depending on the presence of soft-tissue infection and osteomyelitis. Some authors used hyperbaric oxygen therapy (HBO). Donovan et al. reported the favorable effect of HBO in early-stage ORN.[10] However, comparable data with control cases in cervical ORN have not yet been reported. Further studies with more evidence are needed to determine the effect of HBO.
In conclusion, we report a rare case of ORN at the OC junction that presented with an extremely unique clinical episode. Anterior arch disruption secondary to ORN of the atlas can cause severe OC instability, requiring surgical intervention. Posterior OC fixation alone may be an effective procedure, whereas the necrotic pharyngeal region is mild and endoscopically controllable.
Declaration of patient consent
The authors certify that they have obtained an appropriate patient consent form. In the form, the patient has given his consent for his images and other clinical information to be reported in the journal. The patient understands that his name and initials will not be published and due efforts will be made to conceal his identity.
Financial support and sponsorship
Nil.
Conflicts of interest
There are no conflicts of interest.
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