Development of a Retro-Odontoid pseudotumor in the absence of atlantoaxial instability or rheumatoid arthritis Post-Laminoplasty: case report.

A retro-odontoid pseudotumor (ROP) is commonly associated with atlantoaxial instability (AAI) or rheumatoid arthritis (RA). However, we describe a patient with ROP in the absence of AAI or RA. An 81-year-old man who did not have a history of trauma to the head and neck admitted with neck pain, right upper extremity numbness, lower limb weakness, and walking disturbance. He had a history of C2 dome and C3-7 laminoplasty 10 years ago. Magnetic resonance imaging revealed a retro-odontoid mass with cervical cord compression. Dynamic radiography did not show signs of AAI. He underwent C1 laminectomy without fixation for the ROP. We speculated that the load on C1 and C2 increased because of the progression of kyphosis from C2 to C7 with increases in range of motion, which in turn caused change in the biomechanics of the cervical spine, leading to recurrent partial tear and degradation of the transverse ligament that induced formation of the ROP. Spinal surgeons should keep this complication in mind and inform patients about this potential postoperative complication.

Salvage Surgery for Symptomatic Recurrence of Retro-Odontoid Pseudotumor after a C1 Laminectomy.

We provide the first report of successful salvage surgery for a post-C1 laminectomy symptomatic recurrence of a retro-odontoid pseudotumor (ROP) that caused myelopathy. The 72-year-old Japanese woman presented with an ROP causing symptomatic cervical myelopathy. With ultrasonography support, we performed the enucleation of the ROP via a transdural approach and fusion surgery for the recurrence of the mass. At the final observation 2-year post-surgery, MRI demonstrated the mass's regression and spinal cord decompression, and the patient's symptoms had improved. Our strategy is an effective option for a symptomatic recurrence of ROP.

Imaging Characterization of Non-Rheumatoid Retro-Odontoid Pseudotumors: Comparison with Atlantoaxial Manifestation of Rheumatoid Arthritis.

Backgroundand Objectives: To date, imaging characterization of non-rheumatic retro-odontoid pseudotumors (NRROPs) has been lacking; therefore, NRROPs have been confused with atlantoaxial joint involvement of rheumatoid arthritis (RA). It is important to differentiate these two disease because the treatment strategies may differ. The purpose of this study is to characterize imaging findings of NRROPs and compare them with those of RA. Material and Methods: From January 2015 to December 2019, 27 patients (14 women and 13 men) with NRROPs and 19 patients (15 women and 4 men) with RA were enrolled in this study. We evaluated various imaging findings, including atlantoaxial instability (AAI), and measured the maximum diameter of preodontoid and retro-odontoid spaces with magnetic resonance imaging (MRI) and computed tomography (CT). Results: Statistical significance was considered for p < 0.05. AAI was detected in eight patients with NRROPs and in all patients with RA (p < 0.0001). Seventeen patients with NRROPs and six patients with RA showed spinal cord compression (p = 0.047). Compressive myelopathy was observed in 14 patients with NRROPs and in 4 patients with RA (p = 0.048). Subaxial degeneration was observed in 25 patients with NRROPs and in 9 patients with RA (p = 0.001). Moreover, C2-3 disc abnormalities were observed in 11 patients with NRROPs and in 2 patients with RA (p = 0.02). Axial and longitudinal diameter of retro-odontoid soft tissue and preodontoid and retro-odontoid spaces showed significant differences between NRROP and RA patients (p < 0.0001). Furthermore, CT AAI measurements were differed significantly between NRROP and RA patients (p < 0.05). Conclusions: NRROPs showed prominent retro-odontoid soft tissue thickening, causing compressive myelopathy and a high frequency of subaxial and C2-3 degeneration without AAI.

Dysphagia following C1 laminectomy and posterior atlantoaxial fixation for retro-odontoid pseudotumor: a case report.

Purpose: Although dysphagia is known potential complication of cervical spine surgery, it rarely occurs after a posterior approach. We describe an unusual case of a retro-odontoid pseudotumor that suffered dysphagia following a C1 laminectomy and posterior atlantoaxial fixation.Materials and methods: A 79-year-old man presented with progressive tetraparesis and bladder and bowel dysfunction due to severe compression to cervical cord at C1 from a retro-odontoid pseudotumor. After C1 laminectomy and atlantoaxial fixation, the symptoms improved, but dysphagia and aspiration developed, associated with pharyngeal and esophageal stases on videofluoroscopy.Results and conclusions: Possible explanations for postoperative dysphagia include limitation of cervical spine motion, and cervical cord reperfusion injury in addition to the baseline anterior osteophyte and aging. This is the first case of dysphagia developing after laminectomy and posterior atlantoaxial fixation not involving the occipital bone.

Thinking beyond pannus: a review of retro-odontoid pseudotumor due to rheumatoid and non-rheumatoid etiologies.

Retro-odontoid pseudotumor, or mass-like retro-odontoid soft tissue thickening, is an uncommon but important imaging finding that may be associated with rheumatoid arthritis, crystal deposition diseases, as well as non-inflammatory conditions such as cervical degenerative changes and mechanical alterations. Retro-odontoid pseudotumor is commonly associated with atlantoaxial microinstability or subluxation. MRI and CT have an important role in the detection and diagnosis of retro-odontoid pseudotumor. However, due to a wide range of imaging characteristics and ambiguous etiology, it is a frequently misunderstood entity. The purpose of this article is to review relevant anatomy of the craniocervical junction; describe various imaging appearances, pathophysiology and histology in both rheumatoid and non-rheumatoid etiologies; and discuss differential diagnosis of retro-odontoid pseudotumor in order to help guide clinical management.

Post-operative regression of retro-odontoid pseudotumors treated with and without fusion.

PURPOSE: Retro-odontoid pseudotumor is common in elderly people and is a cause of cervical myelopathy. The goal of the study was to investigate surgical procedures, outcomes, and post-operative spontaneous regression of posterior cervical retro-odontoid pseudotumors. METHODS: The subjects were 29 patients who underwent surgery for myelopathy due to a retro-odontoid pseudotumor around the craniocervical region at 9 facilities and were followed-up for an average of 54 months (range 12-96 months). Data were collected in a multicenter review of a retrospective database. Comparisons were performed between cases treated with and without fusion. RESULTS: The JOA recovery rate at final follow-up did not differ significantly between the fusion (n = 17, including all 15 patients with atlantoaxial subluxation) and non-fusion (n = 12) groups. However, pseudotumor regression was significantly more frequent in the fusion group (100% vs. 42%, p < 0.01). In all patients, regression cases had significantly higher rates of contrast enhancement of the pseudotumor on pre-operative T1 gadolinium-enhanced MRI (68% vs. 14%, p = 0.013) and of JOA recovery (50% vs. 30%, p < 0.01). CONCLUSIONS: Regression of pseudotumor occurred in all cases treated with fusion surgery. There was a significant difference in pseudotumor regression with or without fusion, and regression was significantly related to gadolinium enhancement on MRI. Therefore, it is preferable to use fusion surgery for a retro-odontoid pseudotumor that shows contrast enhancement, even if there is no apparent instability pre-operatively. These slides can be retrieved under Electronic Supplementary Material.

[Retro-odontoid pseudotumor in diffuse idiopathic skeletal hyperostosis]. / Pseudotumor retroodontoideo en un paciente con hiperostosis esquelética idiopática difusa.

Retro-odontoid pseudotumors are lesions caused by inflammatory granulation or reactive soft tissue hypertrophy from chronic atlantoaxial subluxation. However, one-third of the cases reported in the medical literature did not show atlantoaxial instability clearly. The authors present the case of a 76-year-old man previously diagnosed with diffuse idiopathic skeletal hyperostosis who presented with severe progressive myelopathy. A magnetic resonance imaging of his cervical spine revealed a retro-odontoid predural mass, which caused a severe compression of the cervical spinal cord. The patient underwent a posterior laminectomy of the atlas and an occipitocervical fusion. After surgery, the pseudotumor was considerably smaller and the neurological symptoms improved.

Circumferential Pannus at C1-C2 Causing Spinal Cord Compression.

Periodontoid pannus formation is a pathologic condition caused by a multitude of different etiologies, however, it is most commonly due to rheumatoid arthritis. In these cases, the pannus is typically located in the retro-odontoid space ventral to the spinal cord, leading to progressive neural compression. We describe in this report, a patient who presented with progressive high cervical myelopathy, who on imaging revealed both a retro-odontoid pannus and a posterior C1-C2 mass causing severe circumferential compression of the spinal cord. The patient was successfully treated with a C1-C2 laminectomy and occipitocervical fusion. Periodontoid pannus is a common entity; however, the presence of a C1-C2 posterior pannus is a unique finding. To our knowledge, circumferential pannus at C1-C2 causing neural compression is a clinical entity that has not been previously reported.

The Impact of Cervical Degeneration and Sagittal Balance on Retro-Odontoid Soft-Tissue Thickness.

OBJECTIVE: To investigate whether retro-odontoid soft-tissue thickness (ROSTT) is associated with cervical degeneration, cervical spine mobility, and sagittal balance of cervical spine. METHODS: The data of 151 patients who presented at our hospital with cervical spondylosis were reviewed. The ROSTT was measured using T1-weighted sagittal cervical magnetic resonance imaging findings. The assessment of the degree of cervical intervertebral disc degeneration (IVDD) was conducted using sagittal T2-weighted imaging. The T1 slope (T1S), C0-C2 angle, C1-C2 angle, C2-C7 angle, C1-C7 sagittal vertical axis and C2-C7 sagittal vertical axis were measured. The range of motion was assessed by measuring the flexion-extension radiographs. According to the ROSTT, those measuring less than 3 mm were classified as normal group and those measuring larger than 3 mm were classified as thickened group. RESULTS: The thickened group had larger cervical IVDD grade, age, C2-C7 angle, and T1S compared to the normal group (all P < 0.05). Additionally, the C0-C2 angle was significantly smaller in the thickened group than in the normal group (P < 0.05). ROSTT showed a negative correlation with C0-C2 angle (r = -0.181, P < 0.05), but positive correlations with both C2-C7 angle (r = 0.255, P < 0.05) and T1S (r = 0.240, P < 0.05). Furthermore, ROSTT was positively correlated with age (r = 0277, P < 0.05) and cervical IVDD grade (Spearman, r = 0.299, P < 0.05). CONCLUSIONS: Cervical sagittal balance and cervical degeneration have a significant impact on ROSTT. Patients with a higher T1S and severe cervical degeneration are more likely to result in greater ROSTT.

Cervical Myelopathy Caused by Non-Rheumatic Retro-Odontoid Pseudotumor: An Investigation of Underlying Mechanisms and Optimal Surgical Strategy.

STUDY DESIGN: Retrospective case-control study. OBJECTIVE: This study aimed to identify the underlying pathologies of non-rheumatic retro-odontoid pseudotumors (NRPs), which would help establish an appropriate surgical strategy for myelopathy caused by NRP. METHODS: We identified 35 patients with myelopathy caused by NRP who underwent surgery between 2006 and 2017. An age- and sex-matched control group of 70 subjects was selected from patients with degenerative cervical myelopathy. Radiographic risk factors for NRP were compared between cases and controls. We also assessed surgical outcomes following occipital-cervical (O-C) fusion, atlantoaxial (C1-2) fusion, or C1 laminectomy. RESULTS: Patients with NRP had significantly lower C1 sagittal inner diameter, C2-7 range of motion (ROM), C2-7 Cobb angle, and C7 tilt, as well as significantly higher C1-2 ROM, atlantodental interval (ADI), and C1-2 to O-C7 ROM ratio. Multivariate regression analysis revealed that ADI, C2-7 ROM, and C7 tilt were independent risk factors for NRP. Neurological recovery and pseudotumor size reduction were comparable among surgical procedures, whereas post-operative cervical spine function was significantly lower in the O-C fusion group than in the other groups. CONCLUSION: Non-rheumatic retro-odontoid pseudotumor was associated with an increase in ADI, suggesting that spinal arthrodesis surgery is a reasonable strategy for NRP. C1-2 fusion is preferable over O-C fusion because of the high prevalence of ankylosis in the subaxial cervical spine. Given that 29% of patients with NRP have C1 hypoplasia, such cases can be treated by posterior decompression alone. Our study highlights the need to select appropriate surgical procedures based on the underlying pathology in each case.

Atlantoaxial Intradural Synovial Cyst Mimicking an Extradural Lesion Adjacent to a Retro-odontoid Pseudotumor: A Case Report.

Atlantoaxial synovial cysts can very rarely penetrate the dura mater into the thecal sac and cause direct neural compression. Several case reports have been available on "intradural synovial cysts" (IDSCs). In this study, we report on a case with an atlantoaxial IDSC mimicking an extradural lesion. A 90-year-old man was diagnosed with a cystic lesion located laterally to the atlantoaxial joint adjacent to the retro-odontoid pseudotumor (ROP) causing cervical spinal cord compression. Thus, surgical removal was planned. On preoperative examination, the cyst, which had a two-layer structure showing a T2-isointense small mass inside a T2-hyperintense lesion, was thought to be located in the extradural region. However, operative findings showed that the cyst was located inside the dura mater. Histopathological examination suggested a synovial cyst. No recurrence of the cyst was observed until the latest follow-up after 3 years, and the ROP decreased in size. Almost all IDSCs reported previously were observed in the medial site of the atlantoaxial joint. In our case, however, the cyst was observed adjacent to the posteromedial site of the right atlantoaxial joint and the ROP, mimicking an extradural lesion. We had no knowledge regarding the IDSC before the surgery and assumed an extradural lesion. Albeit rare, the existence of such a condition should be considered.

Cervical Myelopathy Due to Idiopathic Retro-odontoid Pseudotumor.

OBJECTIVE: A retro-odontoid pseudotumor (ROP) is commonly associated with atlantoaxial instability or rheumatoid arthritis. However, ROP in the absence of atlantoaxial instability or rheumatoid arthritis, which is termed idiopathic ROP (IROP), is a rare condition. The pathomechanisms and optimal treatment strategies for IROP remain controversial. The aim of the present study was to evaluate the radiographic and clinical characteristics of IROP patients and to assess the efficiency of atlantoaxial/occipitocervical fusion on IROP regression. METHODS: Data from 5 patients diagnosed with IROP were retrospectively reviewed. Posterior atlantoaxial or occipitocervical fixation and fusion were performed in 4 patients and C1 posterior arch resection alone in 1 patient. The patients' features, surgical procedures, and complications were recorded. The retro-odontoid soft tissue thickness was measured on preoperative and postoperative magnetic resonance imaging to evaluate IROP regression. RESULTS: The mean follow-up time was 37 months. ROP regression was achieved in patients who received atlantoaxial/occipitocervical fusion, but not for the patient with C1 posterior resection alone. There were no observed neurovascular complications associated with surgery. CONCLUSIONS: IROP was related to a restricted range of motion of the subaxial spine. Upper cervical fixation is an optional treatment that produces IROP regression over time. By contrast, direct removal of the IROP is unnecessary.

Regression of retro-odontoid pseudotumor following botulinum injection into cervical muscles.

Background: Posterior fixation of C1/2 has become more commonly performed to treat retro-odontoid pseudotumor (ROP). Here, we report a 60-year-old female with cervical dystonia (CD), whose ROP regressed and whose quadriparesis improved after a series of cervical intramuscular botulinum injections. Case Description: A 60-year-old female with 30 years of CD newly presented with a progressive quadriparesis. When the MRI showed ROP compression the cervical spinal cord, she refused surgery and underwent multiple cervical muscular botulinum injections over the next 2 years. Following these injections, the patient's quadriparesis improved as the ROP regressed on subsequent MR studies. Conclusion: Over a 2-year period, multiple cervical botulinum injections caused regression of a retro-odontoid cervical pseudotumor improvement in the patient's quadriparesis.

Factors Associated with Retro-Odontoid Pseudotumor in Long-Term Hemodialysis Patients.

OBJECTIVE: Hemodialysis has been reported to be associated with retro-odontoid pseudotumor (ROP), but its clinical characteristics have not been well described. The purpose of the present study was to investigate the factors associated with ROP in hemodialysis patients. METHODS: A retrospective clinical study of hemodialysis patients was conducted with the evaluation of computed tomography and magnetic resonance imaging of cervical spinal lesions at a single institution from 2012 to 2020. The patients' characteristics and radiographic findings were assessed. A case-control analysis was performed between patients with ROP (ROP group) and patients without ROP (control group). RESULTS: We analyzed 46 patients. The mean duration of hemodialysis (± standard deviation) was 21.5 ± 11.8 years. The mean retro-odontoid soft tissue thickness was 4.3 ± 0.3 mm and was correlated with the duration of hemodialysis (r = 0.46, P < 0.01). Thirty patients (65.2%) were included in the ROP group. The ROP group showed a significantly longer duration of hemodialysis (24.9 ± 11.2 years vs. 15.2 ± 10.3 years, P < 0.01) and a higher incidence of osteolytic lesions in the atlantoaxial joint compared with the control group (60.0% vs. 18.8%, P < 0.01). Logistic regression analysis revealed the atlantoaxial osteolytic lesions are associated with retro-odontoid pseudotumor in hemodialysis patients (odds ratio, 5.1; 95% confidence interval, 1.1-24.2; P = 0.04). CONCLUSIONS: The existence of ROP in hemodialysis patients was associated with osteolytic lesions in the atlantoaxial joint. The finding of atlantoaxial erosive lesions in long-term hemodialysis patients requires spine surgeons to carefully evaluate the presence of ROP.

Combined C1-2 transarticular screw fixation and C1 laminoplasty with interlaminar bone transplant for retro-odontoid pseudotumor: illustrative case.

BACKGROUND: The surgical treatment for retro-odontoid pseudotumor (ROP) remains controversial. Posterior C1 laminectomy without fixation occasionally leads to postoperative enlargement of ROP. Because its pathogenesis is associated with atlantoaxial instability, atlantoaxial fixation with C1 laminectomy is commonly performed. Atlantoaxial fixation usually includes transarticular fixation and screw-rod fixation. However, these methods have some issues. That is, in transarticular fixation with C1 laminectomy, the bone grafting area is challenging to obtain. In screw-rod fixation with C1 laminectomy, extensive bleeding occurs at times if the lateral atlantoaxial joint is exposed to the transplant bone grafts. OBSERVATIONS: Herein, the authors report a case of ROP treated with combined C1-2 transarticular screw fixation and C1 laminoplasty with interlaminar bone transplant, which can help achieve adequate spinal cord decompression and can simultaneously secure the bone grafting area between the laminae. In addition, the volume of intraoperative blood loss decreased. Postoperative magnetic resonance imaging showed sufficient decompression of the spinal cord at the C1 level, and computed tomography performed after 6 months revealed bone union between the C1 and C2 laminae. LESSONS: Combined C1-2 transarticular screw fixation and C1 laminoplasty with interlaminar bone transplant is a useful method for ROP.

A case of nonrheumatoid retro-odontoid pseudotumor in Klippel-Feil syndrome with C1 occipitalization.

Background: Patients with both nonrheumatoid retro-odontoid pseudotumors (ROPTs) and congenital craniocervical junction (CCJ) abnormalities are rare. Here, a 73-year-old female presented with neck pain and myelopathy due to MR-documented ROPT with intramedullary hyperintensity at the CCJ warranting an occipital-cervical fusion. Case Description: A 73-year-old female originally developed occipitalgia and became quadriparetic within the subsequent 7 months. The cervical MR showed a ROPT with intramedullary hyperintensity at the CCJ. Further, the CT demonstrated C1 occipitalization and a congenital C2-3 fusion without radiological instability. After she underwent an occipito-C2 fusion, her symptoms improved. Conclusion: For patients with C1 occipitalization and a Klippel-Feil syndrome, ROPT may occur due to loading of C1-2 complex. These patients typically favorably respond to occipito-C2 fusion.

Retro-Odontoid and Retro-C2 Body Pseudotumor, Pannus, and/or Cyst. A Study Based on Analysis of 63 Cases.

OBJECTIVE: The potential significance of relationship of atlantoaxial instability with retro-odontoid pseudotumor, pannus, and/or cyst (RPC) is analyzed. METHODS: We searched the database of patients with craniovertebral junction-related instability treated by atlantoaxial fixation from January 2000 to March 2020. We identified 63 patients in whom there was an RPC in the region posterior to the odontoid process or posterior to the posterior aspect of the C2 body. RESULTS: The RPC was solid in 10 patients, predominantly cystic in 39 patients, and had both solid and cystic components in 14 patients. The vertical dimension of the RPC varied from 4.5 to 10.5 mm (average, 7.9 mm) and the transverse dimension ranged from 2 to 5.2 mm (average, 3.4 mm). In 51 patients, the RPC was in the midline and in 9 patients, it was eccentric in location. In 38 patients, there was erosion of the C2 body adjoining the RPC. Analysis of investigations showed that in 100% of patients, the dome of the RPC was in line with the most susceptible point of potential bone compression of neural structures. The RPC was eccentric in location in 9 patients presenting with torticollis. RPC indicated the presence of atlantoaxial instability even when there were no other positive and validated radiologic indicators. After atlantoaxial stabilization, the RPC spontaneously regressed or disappeared. Direct resection of the RPC was neither performed nor was necessary in any case. CONCLUSIONS: RPCs are secondary consequences of atlantoaxial instability and need not be primarily addressed by surgical resection. Their location suggests that they might have a neural protective function.

Preoperative Cyst Formation as a Predictive Feature of Spontaneous Regression of Retro-Odontoid Pseudotumor After Posterior Fusion.

OBJECTIVE: Although the spontaneous regression of pseudotumors after posterior fusion has been reported, the predictive factors remain unclear. We examined the radiological features that predict for the regression of retro-odontoid pseudotumors after posterior fusion, with a specific focus on cyst formation. METHODS: We included 28 patients with a diagnosis of retro-odontoid pseudotumor using preoperative magnetic resonance imaging. The radiographic parameters and pseudotumor thickness were measured pre- and postoperatively. The regression rate for each pseudotumor was calculated. The presence of a cyst around the retro-odontoid pseudotumor was investigated. If present, the cyst thickness was measured. To elucidate the predictors for the postoperative regression of pseudotumors, the patients were divided into 2 cohorts: the regression group with a regression rate >40% and the no-regression group with a regression rate of <40%. Multivariate logistic regression analysis, including the demographic data and preoperative radiographic parameters as independent variables, was performed. RESULTS: The mean pseudotumor size had decreased significantly from 8.8 ± 3.6 mm preoperatively to 5.3 ± 2.0 mm postoperatively (P < 0.0001). The mean regression rate was 35.9% during a magnetic resonance imaging follow-up period of 8.6 months (range, 6-12 months). Cystic lesions were noted in 10 patients (35.7%) preoperatively. The mean cyst size was 4.7 ± 1.9 mm. All cysts were located dorsal to the pseudotumors and were involved at the maximum spinal compression levels. Nevertheless, all the cysts had disappeared postoperatively. Multivariate logistic regression analysis revealed that the pseudotumor regression group had had a significantly greater proportion of cysts (57.1% vs. 14.3%; odds ratio, 11.7; P = 0.013). CONCLUSIONS: The presence of cystic lesions protruding from retro-odontoid pseudotumors might serve as a predictive factor for the spontaneous regression of pseudotumors after posterior fusion.

Retro-odontoid pseudotumor concomitant with proximal adjacent cervical spondylotic myelopathy after multilevel anterior cervical discectomy and fusion.

INTRODUCTION AND IMPORTANCE: Adjacent cervical spondylotic myelopathy (CSM) following anterior cervical discectomy and fusion (ACDF) presenting as a retro-odontoid pseudotumor (ROP) is uncommon. This consequence adversely affects hand function, causes gait imbalance and results in other disabilities for the patient. This report describes the successful surgical treatment of a patient with ROP associated with adjacent CSM following multilevel ACDF of the subaxial cervical vertebrae by performing posterior cervical decompression and fusion. CASE PRESENTATION: A 60-year-old-male presented with progressive, disabling cervical myelopathy. He had undergone ACDF C3-C7 for treatment of CSM 16 years ago and his symptoms had fully resolved. Magnetic resonance imaging (MRI) revealed severe cervical spinal cord compression caused by a retro-odontoid mass at the C1-C2 level with upper adjacent segment disease (ASD) of C1-C3. The patient received C1-C3 posterior cervical spinal fusion by C1 lateral mass C2 and C3 pedicle screw fixation and C1-C3 laminectomy. After the surgery, he was able to ambulate independently and the myelopathic symptoms were significantly improved at the 6 months follow-up. CLINICAL DISCUSSION: Retro-odontoid pseudotumor concomitant with proximal ASD following ACDF is a rare occurrence. Both diagnosis and surgical management are challenging. CONCLUSIONS: Posterior cervical decompression and fusion of C1-C3 is an effective option for treatment of severe cervical spinal cord compression by a retro-odontoid mass at the C1-C2 level combined with ASD after ACDF.

Symptomatic retro-odontoid pseudotumor causing calcium pyrophosphate dihydrate deposition combined with multilevel cervical spondylotic myelopathy.

INTRODUCTION: Symptomatic retro-odontoid pseudotumor (ROP) caused by cervical compression and myelopathy is rare. Pathological diagnosis is recommended for differential diagnosis including the following: inflammatory disease, primary bone tumor, metastatic disease and calcium pyrophosphate dihydrate deposition (CPPD) also known as "crowned dens syndrome". The authors report a rare case of ROP caused by CPPD deposition combined with multilevel cervical spondylotic myelopathy (MCSM) which was treated by tumor resectioning using a transoral approach combined with posterior decompression and fusion. CASE PRESENTATION: A 66-year-old male presented with progressive neck pain and spastic gait with no history of trauma. Radiographic imaging revealed degenerative change involving the atlanto-axial and atlanto-occipital joints with calcified enhancing soft tissue around the odontoid process causing cord compression and cervical instability at the C1-C2 level combined with MCSM and spinal cord compression at C3 to C7. Microscopic assisted transoral tumor resection combined with posterior decompression and fusion was performed at the occiput to T2. The pathology report describes a rhomboid-shaped crystal caused by calcium pyrophosphate dihydrate deposition (CPPD) disease. At the 6-month follow-up following the operation, the patient's neck pain and spastic gait were improved compared to the preoperative examination. DISCUSSION: Cervical compression and myelopathy from ROP causing CPPD combined with MCSM is rare. Pathology diagnosis and surgical management are highly recommended. CONCLUSION: In this case, a combined surgical approach: tumor resection using a transoral approach and a posterior approach for decompression and fusion at occiput to T2 was an effective option for this condition.