Retro-odontoid pseudotumor: a potentially serious cervical spine incidental finding on cone beam computed tomography.

Retro-odontoid pseudotumor (ROP) is a benign proliferation of soft tissues posterior to the odontoid process. It can be associated with both inflammatory and noninflammatory arthritis. Altered biomechanics and atlantoaxial instability are the major contributing factors. There are no characteristic clinical findings. In the majority of cases, ROP is detected as an incidental finding in imaging. Early diagnosis of the lesion is important because of its proximity to the spinal canal and spinal cord. Clinical signs and symptoms range from pain to paralysis depending on the presence and level of spinal cord compression. In very rare cases, the lesion might cause sudden death of the patient. We report a case of ROP detected as an incidental finding in cone beam computed tomography of a patient examined for implant treatment planning.

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.

The Impact of C1 Anterior Arch Preservation on Spine Stability After Odontoidectomy: Systematic Review and Meta-Analysis.

BACKGROUND: Odontoidectomy for symptomatic irreducible ventral brainstem compression at the craniovertebral junction may result in spine instability requiring subsequent instrumentation. There is no consensus on the importance of C1 anterior arch preservation in prevention of iatrogenic instability. We conducted a systematic review of the impact of C1 anterior arch preservation on postodontoidectomy spine stability. METHODS: PubMed, Embase, Scopus, Web of Science, and Cochrane were searched following the PRISMA guidelines to include studies of patients undergoing odontoidectomy. Random-effect model meta-analyses were performed to compare spine stability between C1 anterior arch preservation versus removal and posttreatment outcomes between transoral approaches (TOAs) versus endoscopic endonasal approaches (EEAs). RESULTS: We included 27 studies comprising 462 patients. The most common lesions were basilar invagination (73.3%) and degenerative arthritis (12.6%). Symptoms included myelopathy (72%) and neck pain (43.9%). Odontoidectomy was performed through TOA (56.1%) and EEA corridors (34.4%). The C1 anterior arch was preserved in 16.7% of cases. Postodontoidectomy stabilization was performed in 83.3% patients. Median follow-up was 27 months (range, 0.1-145). Rates of spine instability were significantly lower (P = 0.004) when the C1 anterior arch was preserved. Postoperative clinical improvement and pooled complications were reported in 78.8% and 12.6% of patients, respectively, with no significant differences between TOA and EEA (P = 0.892; P = 0.346). Patients undergoing EEA had significantly higher rates of intraoperative cerebrospinal fluid leaks (P = 0.002). CONCLUSIONS: Odontoidectomy is safe and effective for treating craniovertebral junction lesions. Preservation of the C1 anterior arch seems to improve maintenance of spine stability. TOA and EEA show comparable outcomes and complication rates.

Operative Technique for Resection of a Ventral Transdural Retro-Odontoid Pannus: A 2-Dimensional Operative Video.

BACKGROUND: Retro-odontoid pseudotumors are rare inflammatory complications of atlantoaxial instability often associated with cervical degenerative disease and rheumatoid arthritis. While propagation of these lesions has been shown to cause spinal cord compression and cervical myelopathy, intradural extension has rarely been reported. METHODS: In this manuscript and 2-dimensional illustrative intraoperative video, we demonstrate cervical decompression, removal of the intradural component, and stabilization with C1-2 instrumentation using a posterior approach. A 71-year-old patient presented with progressive cervical myelopathy. Preoperative imaging demonstrated a large retro-odontoid pannus causing severe spinal cord compression and an associated contrast-enhancing intradural lesion, in the absence of obvious C1-2 instability or fractures on computed tomography scan. C1-2 posterior decompression and fusion were performed with maximally safe intradural pannus resection and ventral dural reconstruction. RESULTS: Postoperatively, the patient experienced significant improvement in myelopathic symptoms. Imaging demonstrated good spinal cord decompression with complete intradural pannus resection and debulking of the extradural component. CONCLUSIONS: Our outcome in this rare complication suggests a posterior approach may be effective in treating similar patients.

An Unusual Chordoma of the Odontoid Process: A Case Report and Literature Review.

The aim of this study is to present a rare case of chordoma in the odontoid process in which the tumor involved the odontoid process and compressed the spinal cord at the craniocervical junction. We report on the effectiveness and successful outcome of anterior microscopic tumor resection combined with posterior occipitocervical fixation and review the current standard treatment. A 39-year-old man presented with sudden dyspnea and quadriparesis caused by an unknown tumor compression at C2. Radiographic examination revealed a large destructive mass at C2 and heterogeneous enhancement. The patient received urgent surgical intervention by microscopic-assisted anterior tumor resection and posterior spinal fixation from the occiput to the C5 level. The pathohistologic reports for cytokeratins, epithelial membrane antigen, and S-100 protein were positive. The final diagnosis was chordoma of the odontoid process. At the 2-year follow-up, the patient's condition had improved, and a postoperative MRI showed no indication of tumor regrowth. Chordoma of the odontoid process or C2 body is very rare. The current standard management is wide tumor resection to prevent recurrence. The combined approach of anterior tumor resection with microscopic assistance and posterior stabilization of the occiput to C5 is the optimal treatment for this condition.

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.

Retro-Odontoid Pseudotumor Without Radiographic Instability with Congenital C1 Assimilation and C2-C3 Fusion: A Case Report.

CASE: The authors present a case of retro-odontoid pseudotumor (ROP) with congenital C1 assimilation and C2-C3 block vertebra without radiological instability who presented with cervical myelopathy with spastic quadriparesis. The patient was managed with occipitocervical fusion and C1 laminectomy. She had rapid neurological recovery in 3 months postoperatively and at 2 years had complete resolution of the retro-odontoid mass. CONCLUSION: C1 assimilation without apparent radiographic instability as a cause of ROP is underappreciated. This case report and review of literature highlight that C1 assimilation and C2-C3 fusion can lead to ROP even in the absence of apparent radiographic instability with posterior atlantoaxial fusion alone providing good results.

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.

Experience in surgical treatment of type â ¡ odontoid fractures: A report of two cases and review of the literature.

Two cases of type Ⅱ odontoid fractures were reported to share our experience in surgery treatment of such cases. A 33-year-old woman with comminuted type Ⅱ odontoid fracture and a 42-year-old man with fracture end hardened type Ⅱ odontoid fracture received surgical treatment in our hospital. Though imaging examination suggested that these two patients were suitable for anterior screw fixation, we encountered difficulties during the operation. The two patients eventually underwent posterior C1-C2 fusion surgery and recovered well. According to the experience of these two cases, we found that the fracture line angle and the degree of comminution are two important factors affecting surgical decision-making. Although anterior screw fixation is the ideal choice for type Ⅱ odontoid fractures with anterior superior to posterior inferior fracture line, it may not be the best choice for comminuted or fracture end hardened type Ⅱ odontoid fractures.

Dorsal Atlantoaxial Ligament Hypertrophy as a Cause for Clinical Signs in Dogs with Dens Abnormalities.

The objective of this study is to suggest clinical and subclinical atlantoaxial (AA) instability as a cause for dorsal AA ligament hypertrophy responsible for clinical signs in dogs with dens abnormalities. Clinical information from five dogs with malformed dens and dorsal spinal cord compression at the AA junction was collected. All dogs had neck pain, associated with tetraparesis in three cases. Radiological examination revealed hypoplastic dens in two dogs and a defect in its ossification in the other three. Stress views were able to demonstrate obvious AA instability only in two cases, but it was suspected in the other owing to response to surgical fixation of the joint and the presence of a dorsal compressive band, which was considered an enlarged dorsal AA ligament. Surgical and histopathological examination of compressive tissue confirmed hypertrophy of the ligament. Long-term prognosis in the four operated cases, either by dorsal decompression and ventral fixation or by ventral fixation alone, was excellent. A malformed dens can cause subclinical instability, unnoted in dynamic studies. As instability may lead to hypertrophy of joint ligaments, soft tissue changes (specifically dorsal AA ligament hypertrophy) points out this instability and the need for joint fixation if surgical management is required.

Morphometric Analysis of the Lateral Atlantoaxial Joints in Patients with an Old Type II Odontoid Fracture and Atlantoaxial Dislocation: A Study Based on CT Analysis.

MINI: The morphological features of the lateral atlantoaxial joints (LAJs) in patients with old type II odontoid fractures and atlantoaxial dislocation have not been fully analyzed. Our study found the changes in morphological features of the LAJs in some patients, and revealed the causes and consequences of the changes in morphological features of the LAJs.

Retrospective cohort study. We aimed to study and interpret the morphological features of the lateral atlantoaxial joints (LAJs) in patients with old type II odontoid fractures and atlantoaxial dislocation (AAD). The causes and consequences of the morphological changes of the LAJs in patients with old type II odontoid fractures and AAD have not been analyzed before. A retrospective study was performed on patients with old type II odontoid fracture and AAD. These patients were placed in group A (unstable or reducible dislocation) or group B (irreducible or bony dislocation) depending on the type of dislocation they had. We documented the morphological changes and measured the inclination angle of the articular surface of the LAJs in computerized tomographic images and compared the results collected for the two groups. The association between the history of injury and the inclination angle of the articular surface of the LAJs was assessed using the Pearson correlation coefficient. A total of 29 patients were enrolled (16 patients in group A, and 13 patients in group B). The inclination angle in group A was significantly greater than that in group B ( P  < 0.001). Patients in group B were more likely to have fish-lip-like changes, dome-like changes, or collapse of the LAJs. Age, history, atlantodental interval, and Japanese Orthopedic Association score were also significant differences between the two groups ( P  < 0.05). There was a negative correlation between the history of injury and the inclination angle of the articular surfaces of the LAJs (left: r = −0.726, P  < 0.001; right: r = −0.795, P  < 0.001). The morphological changes of the LAJs could progressively become more pronounced during the evolution of the disease. Level of Evidence: 4.

Subluxación atlanto-axoidea secundaria a absceso retrofaríngeo en el adulto (síndrome de Grisel). Revisión de la literatura a propósito de un caso clínico / Atlantoaxial subluxation secondary to retropharyngeal abcess in adults (Grisel's syndrome). Review of literature about a clinical case

El síndrome de Grisel (SG) se trata de una subluxación atlanto-axoidea no traumática producida luego de un proceso inflamatorio, infeccioso o posquirúrgico de la región superior de cuello. Es una complicación característica, aunque actualmente infrecuente, de la faringitis bacteriana aguda en la edad pediátrica. La presentación en adultos es aún más infrecuente y requiere un manejo específico. Presentamos el caso de una paciente de 68 años con antecedentes de leucemia mieloide crónica, que se presentó con poliartritis infecciosa, cervicalgia, tortícolis e impotencia cervical. La radiografía, la TC y la RM cervical demostraron una subluxación C1-C2 adyacente a un absceso faríngeo, diagnosticándose un SG. Al finalizar el tratamiento antibioticoterápico, la paciente debió ser intervenida quirúrgicamente debido al fracaso de la reducción cerrada. Debido a su incidencia tan baja, no existen guías clínicas para el manejo de estos pacientes; la colaboración interdisciplinaria es fundamental para establecer el diagnóstico oportuno que permita instaurar el tratamiento conservador y prevenir tanto la cirugía como las complicaciones y las secuelas de la fijación de la articulación atlanto-axoidea

Grisel syndrome (GS) is a non-traumatic atlanto-axial subluxation produced after an inflammatory, infectious or postsurgical process of the upper neck region. It is a characteristic, although currently uncommon complication of acute bacterial pharyngitis in the pediatric age. Presentation in adults is even more infrequent and requires specific management. We present the case of a 68-year-old patient with a history of chronic myeloid leukemia, who presented with infectious polyarthritis, cervicalgia, torticollis and cervical impotence. Cervical radiography, CT, and MRI showed a C1-C2 subluxation adjacent to a pharyngeal abscess, so GS was diagnosed. After antibiotic therapy, the patient had to undergo surgery due to the failure of the closed reduction. Due to its low incidence, there are no clinical guidelines for the management of these patients; The interdisciplinary collaboration is fundamental to establish the opportune diagnosis that allows to establish the conservative treatment and prevent the surgery, as well as the complications and sequelae of the fixation of the atlanto-axial joint

Arnold-Chiari malformation type I and the posterior dislocation of the odontoid process aggravate prolonged weaning in a patient with severe viral pneumonia: a case report.

BACKGROUND: Prolonged and difficult weaning is associated with higher rates of complications and mortality. Therefore, it is important to identify the associated factors. CASE PRESENTATION: We describe our experience with a 37-year-old man diagnosed with severe viral pneumonia (influenza A). He presented with acute respiratory failure type I on admission. During intubation, his blood pressure and heart rate decreased, and epinephrine and norepinephrine were administered. Although his clinical condition improved 8 days after intensive care unit (ICU) admission, he experienced difficulty weaning. He remained conscious but had a poor spontaneous cough with sputum production and weak limb muscle strength. His cough reflex was absent during bronchoscopic sputum suction, and he used abdominal breathing during the T-tube test. Magnetic resonance imaging revealed an Arnold-Chiari malformation type I, posterior dislocation of the odontoid process, and syringomyelia, with compression and deformation of the medulla and high cervical cord. The patient was successfully weaned from the ventilator at 20 days after ICU admission. CONCLUSIONS: Arnold-Chiari malformation type I and posterior dislocation of the odontoid process, which aggravate medullary compression and increase the risk of cervical nerve injury, might be a rare factor affecting prolonged weaning in critical illness.

Cervical CT-Dependent Diagnosis of Crowned Dens Syndrome in Calcium Pyrophosphate Dihydrate Crystal Deposition Disease.

PURPOSE: The purpose of this study is to assess the presence of crowned dens syndrome in patients with calcium pyrophosphate disease. We report 34 patients with crowned dens syndrome in one of the largest series from a single tertiary medical center in North America. METHODS: A retrospective chart review was conducted at the University of Kansas Medical Center from November 1, 2005-November 1, 2017. A total of 191 patients with calcium pyrophosphate disease were identified. The available cervical computed tomography scans were analyzed by a musculoskeletal radiologist for the presence of periodontoid calcifications and erosions. RESULTS: Of the 191 patients with calcium pyrophosphate disease, 57 had cervical computed tomography scans; 34 of them (34/57, 59.64%) had periodontoid calcifications. Only 12/34 patients were formally evaluated and diagnosed by rheumatologists with crowned dens syndrome. Twenty-two of 34 were either not seen by a rheumatologist or were not diagnosed with crowned dens syndrome. The median age was 78.5 years, with 73.52% over 70 years old; 24/34 (70.58%) were female; 17/34 patients (50%) were symptomatic; 28/34 (82.35%) had additional sites of chondrocalcinosis on available radiographs; 8 (28.57%) had 3 or more sites of chondrocalcinosis in typical calcium pyrophosphate disease locations. Six patients did not have any radiographs. CONCLUSION: Crowned dens syndrome is an under-recognized entity that should be considered in elderly patients with neck pain in the setting of calcium pyrophosphate disease. Our data demonstrated a high percentage (about 60%) of patients with calcium pyrophosphate disease who had cervical computed tomography findings consistent with crowned dens syndrome. This underscores the importance of performing cervical computed tomography when evaluating patients with neck pain and calcium pyrophosphate disease.

Posterior Screw-Rod Fixation and Selective Axial Loosening for the Treatment of Atlantoaxial Instability or Dislocation Caused by Os Odontoideum: A Case Series for a Single Posterior Approach.

OBJECTIVE: To evaluate the effect of screw-rod fixation and selective axial loosening in the treatment of atlantoaxial instability or dislocation (including reducible and irreducible) caused by os odontoideum (OO) via a single posterior approach. METHODS: A consecutive series of patients with OO surgically treated in our hospital were retrospectively analyzed. For atlantoaxial instability and reducible atlantoaxial dislocation, C1-C2 screw-rod fixation and fusion were performed. OO combined with irreducible atlantoaxial dislocation was reduced after posterior axial loosening, followed by screw-rod fixation and fusion. The general information, clinical data, and radiographic data were compared between the 2 different procedures. RESULTS: There were 41 patients with an average age of 40.6 ± 21.7 years. All the patients underwent posterior reduction and C1-2 screw rod fixation, 6 with axial loosening and 35 without axial loosening. The clinical manifestations and radiographic data significantly improved after the operation with a low rate of complications. Except for clivus-canal angle and visual analogue score of cervical pain, there were no differences in clinical and radiographic data between the 2 procedures. CONCLUSIONS: Posterior screws-rod fixation and selective axial loosening is appropriate for treating OO complicated with atlantoaxial instability or dislocation (including reducible and irreducible) without the need for anterior decompression.

Regression of Retro-odontoid Pseudotumor Using External Orthosis without Atlantoaxial Fusion: A Case Report.

CASE: We treated an 87-year-old woman with noninflammatory retro-odontoid pseudotumor induced by atlantoaxial instability (AAI) using only a Philadelphia collar. Because of perioperative risk factors, fusion surgery was canceled; nevertheless, the neurological symptoms improved gradually. After 11 months of nonoperative treatment, the follow-up magnetic resonance imaging demonstrated that the pseudotumor's size was obviously diminished. Orthosis was applied for 2 years, and after its removal, the pseudotumor's size remained the same at the final 7-year follow-up. CONCLUSIONS: External orthosis is one treatment option for pseudotumor with AAI, especially in patients with significant comorbidities for whom surgical procedures are relatively contraindicated.

Familial Os Odontoideum: Proatlas Segmentation Abnormality.

BACKGROUND: Os odontoideum is a smooth, independent ossicle separated from a hypoplastic odontoid process located cranially in the expected position of the odontoid tip. The pathogenesis of os odontoideum remains controversial, and several etiological theories have been suggested for congenital, developmental, and traumatic origins. CASE DESCRIPTION: We have reported symptomatic familial cases of dystopic os odontoideum in 2 elderly sisters who denied any traumatic history of the head and neck. Both patients were treated surgically, with successful outcomes achieved. CONCLUSIONS: When examining the etiology of os odontoideum, checking for the existence of a traumatic history is important. However, a more important matter is to consider morphological segmentation abnormalities of the proatlas. Segmentation abnormalities of the proatlas could be closely connected to the development of familial os odontoideum.