Objective To elucidate the clinical outcomes of craniocervical realignment surgery in patients with craniovertebral junction (CVJ) kyphosis accompanied by negative sagittal imbalance, and to identify radiological predictors associated with favorable outcomes.
Methods A retrospective analysis was performed on 28 patients who underwent craniocervical realignment between 2014 and 2022 for CVJ kyphosis with accompanying negative sagittal imbalance. Clinical outcomes were evaluated using the Neck Disability Index (NDI), visual analogue scale for neck pain, and the Japanese Orthopaedic Association (JOA) score. Radiographic parameters included the C0–2 angle and the C2–7 sagittal vertical axis (SVA). Favorable outcomes were defined as an improvement of more than 20 points in the NDI and a JOA recovery rate exceeding 50%. Multiple linear regression and receiver operating characteristic (ROC) curve analyses were conducted to identify independent predictors and to determine optimal threshold values.
Results Significant improvements in both clinical outcomes and radiographic alignment were observed in association with craniocervical realignment surgery. Patients who achieved favorable outcomes exhibited greater postoperative changes in the C0–2 angle and the C2–7 SVA. Multivariate analysis identified changesm in the C0–2 angle (p=0.019) and C2–7 SVA (p=0.010) as independent predictors of NDI improvement, while age (p=0.033) and C2–7 SVA change (p=0.037) were independently associated with the JOA recovery rate. ROC curve analysis determined optimal cutoff values of ≥10.65° for C0–2 angle change and ≥19.2 mm for C2–7 SVA change, with corresponding area under the curve values of 0.872 and 0.802, respectively.
Conclusion Craniocervical realignment appears to be a viable surgical option for patients with CVJ kyphosis and negative sagittal imbalance. Postoperative changes in C0–2 angle and C2–7 SVA were found to be associated with favorable clinical and functional outcomes, suggesting their potential role as prognostic factors.
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Objective Lesions of the ventral craniovertebral junction are difficult to access owing to their deep location and proximity to critical neurovascular and pharyngeal structures. In this study, we aimed to describe the surgical technique and clinical outcomes of the endoscopic endonasal transnasopharyngeal approach for ventral craniovertebral junction lesions and highlight key considerations regarding approach selection, airway management, and occipitocervical stabilization.
Methods We retrospectively reviewed 7 patients who underwent the endoscopic endonasal transnasopharyngeal approach for ventral craniovertebral junction lesions. The analysis included preoperative planning for surgical access, intraoperative technique, postoperative management, airway and nutritional strategies, and the need for occipitocervical fixation. One representative case is presented to illustrate key technical steps.
Results Of the 7 patients, 6 had neoplastic lesions and 1 had basilar invagination. Despite a relatively large mean lesion size of 39.4 mm, subtotal or greater resection was achieved in 5 of the 6 tumor cases. Occipitocervical fixation was performed in 2 cases. Two patients underwent prophylactic tracheostomy because of anticipated airway compromise. Of the 5 orally intubated cases, 3 were extubated immediately and 2 by postoperative day 2. Oral feeding resumed by day 10 in 6 cases. No postoperative infections or cerebrospinal fluid leakage occurred. One patient experienced transient velopharyngeal insufficiency, which resolved spontaneously.
Conclusion The endoscopic endonasal transnasopharyngeal approach is a safe and effective option for ventral craniovertebral junction lesions when appropriately selected. Careful preoperative evaluation and individualized management of airway and spinal stability are essential for favorable outcomes.
The atlantoaxial (C1–2) junction is among the most technically demanding regions for cervical spine surgery owing to its complex osseoligamentous anatomy and proximity to critical neurovascular structures. Numerous posterior fixation constructs have been developed to optimize biomechanical rigidity and promote arthrodesis. Since Gallie’s introduction of posterior wiring with autologous bone grafts in 1939, evolving techniques have focused on enhancing fusion rates while minimizing risk to adjacent structures. This paper outlines the historical evolution of C1–2 posterior instrumentation, current fixation strategies, bone fusion techniques, and reduction methods. A systematic literature search identified 61 relevant studies on C1–2 fusion. Additional references were manually reviewed to provide a comprehensive context. Of these, 41 studies were narratively summarized to outline the historical and conceptual evolution of C1–2 fusion techniques, while the remaining 20 post-2000 studies on contemporary surgical modifications were systematically reviewed and tabulated for technical details and clinical outcomes. C1–2 fusion techniques have evolved significantly over time. Early methods primarily involved posterior wiring with autologous bone grafts, but later transitioned to rigid segmental fixation using pedicle screw constructs, resulting in improved fusion rates and clinical outcomes. Interarticular fusion, when concurrently performed, enhances the biological fusion environment, contributing to favorable clinical results. C1 lateral mass, posterior arch, pedicle screws and C2 pedicle, lamina screws give us much stronger stability and higher fusion rates. Interarticular fusion using local bone also gives us technical easiness guaranteeing high fusion rate overcoming inconvenience of wiring and iliac bone harvest. Interarticular height reduction and interarticular fusion should be discriminated.
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Objective To explore a surgical technique for completing ventral bone decompression and C1–2 plate-screw fixation in the craniocervical junction (CVJ) through nasal approach by stage I at the imaging and physical anatomy levels, and to evaluate its feasibility.
Methods Radiographic parameters of 80 patients with basilar invagination (BI) and 56 with normal CVJ anatomy were retrospectively analyzed. Three-dimensional (3D) reconstructions were performed in 31 patients with BI. Key anatomical landmarks, screw entry points, and fixation trajectories were evaluated. Customized plate-screw constructs were designed. Finally, surgical feasibility was tested on a 3D-printed anatomical model and a cadaveric.
Results In 80 BI patients, the average distances between 4 screw insertion points were 16.04 mm, 21.10 mm, 6.83 mm, and 7.10 mm. C2 lateral mass oblique lengths were 16.81 mm (right) and 17.12 mm (left); C1 lengths were 18.71 mm (right) and 19.07 mm (left), with significant differences between C1 and C2 (p<0.001). A 28.5×14.1-mm titanium plate with 16 mm screws was successfully implanted via the nasal route in the polyether ether ketone 3D-printed BI model and the cadaveric. Radiology indicated that the screws were all in the lateral mass and the plates fit tightly.
Conclusion In BI, transnasal odontoidectomy and plate-screw fixation of C1–2 are feasible theoretically. This may enable a new alternative approach for nasal minimally invasive decompression and immobilization, following the completion of biomechanics and clinical trials.
Objective To investigate the developmental defects caused by knockdown of best1 gene in zebrafish as a model for a subtype of craniovertebral junction (CVJ) malformation.
Methods Two antisense morpholinos (MOs) were designed targeting zebrafish best1 to block translation (ATG-MO) or to disrupt splicing (I3E4-MO). MOs were microinjected into fertilized one-cell embryos. Efficacy of splicing MO was confirmed by reverse transcription-polymerase chain reaction. Phenotypes were analyzed and quantified by microscopy at multiple developmental stages. Neuronal outgrowth was assessed in transgenic zebrafish expressing green fluorescent protein in neurons. Skeletal ossification was visualized by Calcein staining.
Results Knockdown of best1 resulted in zebrafish embryos with shorter body length, curved axis, low survival rate, microcephaly, reduced eye size, smaller head and brain, impaired neuronal outgrowth, and reduced ossification of craniofacial and vertebral bone.
Conclusion Best1 gene plays critical roles in ophthalmologic, neurological and skeletal development in zebrafish. A patient with a premature stop codon in BEST1 gene exhibited similar phenotypes, implying a subtype of CVJ malformation.
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Objective To identify potential risk factors for cerebrospinal fluid (CSF) leakage after craniovertebral junction (CVJ) anomaly surgery and to provide a reference for clinical practice.
Methods Sixty-six patients who underwent elective CVJ anomaly surgery during a 6-year period (April 2013 to September 2019) were retrospectively included. Research data were collected from the patients’ medical records and imaging systems. Patients were divided into CSF leak and no CSF leak groups. Univariate tests were performed to identify potential risk factors. For statistically significant variables in the univariate tests, a logistic regression test was used to identify independent risk factors for CSF leakage.
Results The overall prevalence of CSF leakage was 13.64%. Univariate tests showed that a basion-dental interval (BDI) > 10 mm and occipitalized atlas had significant intergroup differences (p < 0.05). Multivariate analysis indicated that a BDI > 10 mm was an independent risk factor for CSF leakage, and patients with CVJ anomalies with a BDI > 10 mm were more likely to have postoperative CSF leaks (odds ratio, 14.67; 95% confidence interval, 1.48–30.88; p = 0.004).
Conclusion It is necessary to maintain vigilance during CVJ anomaly surgery in patients with a preoperative BDI > 10 mm to avoid postoperative CSF leaks.
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Objective Diseases of the craniovertebral junction (CVJ) are commonly associated with deformity, malalignment, and subsequent myelopathy. The misaligned CVJ might cause compression of neuronal tissues and subsequently clinical symptoms. The triangular area (TA), measured by magnetic resonance imaging/images (MRI/s), is a novel measurement for quantification of the severity of compression to the brain stem. This study aimed to assess the normal and pathological values of TA by a comparison of patients with CVJ disease to age- and sex-matched controls. Moreover, postoperative TAs were correlated with outcomes.
Methods Consecutive patients who underwent surgery for CVJ disease were included for comparison to an age- and sex-matched cohort of normal CVJ persons as controls. The demographics, perioperative information, and pre- and postoperative 2-year cervical MRIs were collected for analysis. Cervical TAs were measured and compared.
Results A total of 201 patients, all of whom had pre- or postoperative MRI, were analyzed. The TA of the CVJ deformity group was larger than the healthy control group (1.62 ± 0.57 cm2 vs. 1.01 ± 0.18 cm2, p < 0.001). Moreover, patients who had combined anterior odontoidectomy and posterior laminectomy with fixation had the greatest reduction in the TA (1.18 ± 0.58 cm2).
Conclusion In CVJ deformity, the measurement of the cervical TA could indicate the severity of brain stem compression. After surgery, the TA had a varying degree of improvement, which could represent the efficacy of surgery.
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Objective To summarize the vertebral artery (VA) pattern of 96 “sandwich” atlantoaxial dislocation (AAD) patients and to describe the strategies of reducing the injury of VA during surgery.
Methods From 2009 to 2020, we retrospectively reviewed the 3-dimensional computed tomography angiography data of 96 AAD patients combined with atlas occipitalization and C2–3 fusion, which were diagnosed as “sandwich” AAD and 96 patients as control group patients who were without atlas occipitalization, C2–3 fusion and any other cervical bone deformity at our institution. The variations of each side of VA were described in 3 different parts (C0–1, C1–2, and C2–3) according to the characteristics of the 3-part pathological structures in “sandwich” subgroup.
Results One hundred ninety-two sides of VAs in every group of patients were analyzed and every VA was described separately at 3 different level regions. There were different variations in these 3 different regions: 4 variations in the upper fusion region, 5 variations in the sandwiched region, and 6 variations in the lower fusion region in sandwich AAD patients. And the rate of VA deformity in sandwich AAD patients was much higher and more types of VA variations existed.
Conclusion In “sandwich” AAD patients, deformities of vertebral arteries in craniovertebral junction are more common, and the same VA may have deformities at different levels that severely affect surgical procedures. Therefore, preoperative imaging examination of VA for “sandwich” AAD patients is vital of guiding surgeons to avoid injury of VA during surgery.
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Objective The conventional criteria for defining the basilar invagination (BI) focus on the relationship of odontoid tip to basion and opisthion, landmarks that are intrinsically variable especially in presence of occipitalised atlas. A universal single reference line is proposed that helps in unequivocally establishing the diagnosis of BI, may be relevant in establishing both Goel types A and B BI, as well as in differentiating a ‘very high’ from ‘regular’ BI.
Methods Study design – case-control study. In 268 patients (group I with BI [n = 89] including Goel type A BI [n = 66], Goel type B BI [n = 23], and group II controls [n = 179]), the perpendicular distance between odontoid tip and line subtended between posterior tip of hard palate-internal occipital protuberance (P-IOP line) was measured. Logistic regression analysis determined factors influencing the proposed parameter (p < 0.05).
Results In patients with a ‘very high’ BI (n = 5), the odontoid tip intersected/or was above the P-IOP line. In patients with a ‘regular’ BI (n = 84), the odontoid tip was 6.56 ± 3.9mm below the P-IOP line; while in controls, this distance was 12.53 ± 4.28 mm (p < 0.01). In Goel type A BI, the distance was 7.01 ± 3.78 mm and in type B BI, it was 5.07 ± 4.19 mm (p = 0.004). Receiver-operating characteristic curve analysis identified 9.0 mm (8.92–9.15 mm) as the cut-point for diagnosing BI using the odontoid tip-P-IOP line distance as reference.
Conclusion The odontoid tip either intersecting the P-IOP line (very high BI) or being < 9 mm below the P-IOP line (Goel types A and B BI) is recommended as highly applicable criteria to establish the diagnosis of BI. This parameter may be useful in establishing the diagnosis in all varieties of BI.
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Objective Developmental bony craniovertebral junction (CVJ) anomalies seem to have a genetic basis and also abnormal joint morphology causing atlantoaxial dislocation (AAD) and basilar invagination (BI).
Methods DNA extracted polymerase chain reaction single-stranded conformation polymorphism (SSCP) performed for mutation screening of FBN1 gene (n = 50 cases+ 50 age/sex-matched normal; total: 100). Samples with a deviated pattern of bands in SSCP were sequenced to detect the type of variation. Computed tomography (CT) scans of 100 patients (15–45 years old) compared with an equal number of age/sex-matched controls (21.9 ± 8.2 years). Joint parameters studied: sagittal joint inclination (SI), craniocervical tilt (CCT), coronal joint inclination (CI).
Results Thirty-nine samples (78%) showed sequence variants. Exon 25, 26, 27, and 28 showed variable patterns of DNA bands in SSCP, which on sequencing gives various types of DNA sequence variations in intronic region of the FBN1 gene in 14%, 14%, 6%, and 44% respectively. CT radiology:SI and CCT correlated with both BI and AAD (p < 0.01). The mean SI value in controls: 83.35° ± 8.65°, and in patients with BI and AAD:129° ± 24.05°. Mean CCT in controls: 60.2° ± 9.2°, and in patients with BI and AAD: 86.0° ± 18.1°. Mean CI in controls:110.3° ± 4.23°, and in cases: 125.15° ± 16.4°.
Conclusion The study showed mutations in FBN1 gene (reported in Marfan syndrome). There is also an alteration of joint morphology, correlating with AAD and BI severity. Hence, we propose a double-hit hypothesis: the presence of weak ligaments (due to FB1 gene alterations) and abnormal joint morphology may contribute to AAD and BI.
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Craniovertebral junction (CVJ) trauma is a challenging clinical condition. Being a highly mobile functional unit at the junction of the skull and the vertebral column, traumatic events in this area may produce devastating neurological complications and death. Additionally, many of the CVJ traumatic injuries can be left undiagnosed or even raise difficult treatment dilemmas. We present a literature review in the format of recommendations on the diagnosis and management of different scenarios for upper cervical trauma and produce recommendations, which can be applicable to various areas of the globe.
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Objective Joint manipulation for craniovertebral junction instability is often hindered by the C2-ganglion (C2G). Our study aims to compare the surgical outcome among patients with or without C2G preservation and discuss the technical nuances.
Methods We did a prospective, bicentric study and included all the operated patients with craniovertebral junction anomaly. The outcome was assessed by the Pain Numeric Rating Scale, Patient Satisfactions Score, and Stony Brook Scar Evaluation Scale. The fusion was assessed using Lenke fusion grade.
Results One hundred seventy-one patients (88 in group A and 83 in group B) were included. The most common symptom was spastic quadriparesis (n = 165, 96.5%) with median Nurick grade 3.3. Thirteen patients had suboccipital numbness and 12 patients had paraesthesia. Mean blood loss in group A was 490 ± 96.2 mL and group B was 525 ± 45.7 mL; median operative time was 217.9 and 162.2 minutes in the groups A and B, respectively (p < 0.05). At the follow-up (median, 46.8 months), Lenke fusion grade A was achieved in 92.4% and grade B in 7.6%. A trend suggesting better functional outcomes (numbness, parestheisa, scar outcome, and postoperative ulcer formation) in group A was seen with all 6 patients, who underwent O-C2 fixation, developed pressure sore.
Conclusion Our results support ganglion preservation, especially in the subset of patients where occipital plating is required. Although the study fails to show any statistical significance, we suggest that one should always start with an ‘intent’ of preservation as the functional outcome is better.
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Craniovertebral junction (CVJ) deformity is a challenging pathology that can result in progressive deformity, myelopathy, severe neck pain, and functional disability, such as difficulty swallowing. Surgical management of CVJ deformity is complex for anatomical reasons; given the discreet relationships involved in the surrounding neurovascular structures and intricate biochemical issues, access to this region is relatively difficult. Evaluation of the reducibility, CVJ alignment, and direction of the mechanical compression may determine surgical strategy. If CVJ deformity is reducible, posterior in situ fixation may be a viable solution. If the deformity is rigid and the C1–2 facet is fixed, osteotomy may be necessary to make the C1–2 facet joint reducible. C1–2 facet release with vertical reduction technique could be useful, especially when the C1–2 facet joint is the primary pathology of CVJ kyphotic deformity or basilar invagination. The indications for transoral surgery are becoming as narrow as a treatment for CVJ deformity. In this article, we will discuss CVJ alignment and various strategies for the management of CVJ deformity and possible ways to prevent complications and improve surgical outcomes.
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Chiari I malformation is characterized by the downward displacement of cerebellar tonsils through the foramen magnum. While discussing the treatment options for Chiari I malformation, the points of focus include: (1) Has the well-established procedure of posterior fossa decompression become outdated and has been replaced by posterior C1–2 stabilization in every case? (2) In case posterior stabilization is required, should a C1–2 stabilization, rather than an occipitocervical fusion, be the only procedure recommended? The review of literature revealed that when there is bony instability like atlantoaxial dislocation (AAD), occipito-atlanto-axial facet joint asymmetry or basilar invagination (BI) associated with Chiari I malformation, one should address the anterior bony compression as well as perform stabilization. This takes care of the compromised canal at the foramen magnum and re-establishes the cerebrospinal fluid flow along the craniospinal axis; and also provides treatment for CVJ instability. In the cases with a pure Chiari I malformation without AAD or BI and with completely symmetrical C1–2 joints, however, posterior fossa decompression with or without duroplasty is sufficient to bring about neurological improvement. The latter subset of cases with pure Chiari I malformation have, thus, shown significant (>70%) rates of neurological improvement with posterior fossa decompression alone. A C1–2 posterior stabilization is a more stable construct due to the strong bony purchase provided by the C1–2 lateral masses and the short lever arm of the construct. However, in the cases with significant bleeding from paravertebral venous plexus; a very high BI, condylar hypoplasia and occipitalized atlas; gross C1–2 rotation or vertical C1–2 joints with unilateral C1 or C2 facet hypoplasia, as well as the presence of subaxial scoliosis; maldevelopment of the lateral masses and facet joints (as in very young patients); or, the artery lying just posterior to the C1–2 facet joint capsule (being endangered by the C1–2 stabilization procedure), it may be safer to perform an occipitocervical rather than a C1–2 fusion.
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Full‐endoscopic technique for posterior fossa decompression in Chiari malformation type I: An anatomical feasibility study in human cadavers I. Dolas, A. G. Yorukoglu, A. Sencer, Tugrul Cem Unal, C. I. Gulsever, A. Aydoseli, Y. Aras, P. A. Sabanci, S. Ruetten Clinical Anatomy.2023; 36(4): 660. CrossRef
Manual Therapy and Exercise for the Management of Chronic Neck Pain With Multiple Neurovascular Comorbidities: A Case Report Zach Mayo, Christopher Keating Cureus.2023;[Epub] CrossRef
C1-2 Fusion in Atlantoaxial Dislocation and Basilar Invagination with and without Chiari Malformation: Clinical/Radiological and Craniometric Results Haitao Zhu, Rui Xu, Shengxi Wang, Haijian Xia, Yi Yan, Yongzhi Xia World Neurosurgery.2023; 175: e1017. CrossRef
A rare clinical case of syringomyelia progression in the presence of Chiari I malformation following the surgery E. E. Rostorguev, N. S. Kuznetsova, A. A. Maslov, V. E. Hatyushin, B. V. Matevosyan, G. A. Reznik, O. V. Pandova, E. V. Shalashnaya South Russian Journal of Cancer.2023; 4(3): 44. CrossRef
Mobility Assessment Using Multi-Positional MRI in Children with Cranio-Vertebral Junction Anomalies Flavie Grenier-Chartrand, Maxime Taverne, Syril James, Lelio Guida, Giovanna Paternoster, Klervie Loiselet, Kevin Beccaria, Volodia Dangouloff-Ros, Raphaël Levy, Timothée de Saint Denis, Thomas Blauwblomme, Roman Hossein Khonsari, Nathalie Boddaert, Sandr Journal of Clinical Medicine.2023; 12(21): 6714. CrossRef
Research process, recap, and prediction of Chiari malformation based on bicentennial history of nomenclature and terms misuse Yunsen He, Mengjun Zhang, Xiaohong Qin, Caiquan Huang, Ping Liu, Ye Tao, Yishuang Wang, Lili Guo, Mingbin Bao, Hongliang Li, Zhenzhen Mao, Nanxiang Li, Zongze He, Bo Wu Neurosurgical Review.2023;[Epub] CrossRef
Relevance of C1/2 Facet Configurations and Clivus-Canal-Angles for Adult Patients with Chiari I Malformation with and without Basilar Invagination Jörg Klekamp World Neurosurgery.2022; 162: e156. CrossRef
Development and Validation of a Multivariable Model to Predict Surgical Outcome in Patients with Chiari malformations Shishir Kumar, Komal P. Chandrachari, Kiran Mariswamappa, Shibu V. Pillai, Vishwas H. Vijayendra, Sathyanarayana Lale, Thimappa Hegde Neurology India.2022; 70(3): 905. CrossRef
The management of Chiari malformation type 1 and syringomyelia in children: a review of the literature Veronica Saletti, Mariangela Farinotti, Paola Peretta, Luca Massimi, Palma Ciaramitaro, Saba Motta, Alessandra Solari, Laura Grazia Valentini Neurological Sciences.2021; 42(12): 4965. CrossRef
Prognostic significance of C1–C2 facet malalignment after surgical decompression in adult Chiari malformation type I: a pilot study based on the Chicago Chiari Outcome Scale Michael Lumintang Loe, Tito Vivas-Buitrago, Ricardo A. Domingo, Johan Heemskerk, Shashwat Tripathi, Bernard R. Bendok, Mohamad Bydon, Alfredo Quinones-Hinojosa, Kingsley Abode-Iyamah Journal of Neurosurgery: Spine.2021; 34(2): 171. CrossRef
Chiari malformation type I and basilar invagination originating from atlantoaxial instability: a literature review and critical analysis Arthur Wagner, Lukas Grassner, Nikolaus Kögl, Sebastian Hartmann, Claudius Thomé, Maria Wostrack, Bernhard Meyer Acta Neurochirurgica.2020; 162(7): 1553. CrossRef
The battle against an invisible enemy Manjul Tripathi, Sandeep Mohindra Acta Neurochirurgica.2020; 162(11): 2923. CrossRef
Central Atlantoaxial Instability: A New Clinical Entity? Justin S. Smith Neurospine.2019; 16(2): 212. CrossRef
The Role of Atlantoaxial Instability on Chiari, Ossification of the Posterior Longitudinal Ligament, Spondylosis and Stenosis K. Daniel Riew Neurospine.2019; 16(2): 214. CrossRef
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