Complete Atlantooccipital Assimilation with Basilar Invagination and Atlantoaxial Subluxation Treated Non-Surgically: A Case Report.
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SUBMITTER: Electricwala AJ
PROVIDER: S-EPMC5501708 | biostudies-other | 2017 Jun
REPOSITORIES: biostudies-other
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Project description:Introduction:Atlantoaxial rotatory subluxation (AARS) is not uncommon in paediatric emergencies, however, the complications might be fatal. Long onset before presentation is correlated with higher recurrence and persistent deformity. There is no consensus on the treatment of AARS yet. Selected patients may benefit from conservative approaches; however, retention might be difficult, and subluxation may recur. Presentation of case:A 6-year-old boy was admitted to our institution with AARS for three months before admission. Typical Cock-Robin position was observed. Computed tomography (CT) indicated AARS Fielding and Hawkins grade III. We treated the case conservatively by closed reduction and cervical traction using Gardner-Wells tongs. However, poor compliance resulted recurrence of subluxation, so we decided to fuse the atlantoaxial joint using transarticular screws, posterior wiring, and autologous bone grafting. Posterior fusion resulted in a satisfactory outcome, in which the wound healed accordingly. Six months of follow up examination revealed normal motoric and sensory function. The patient was able to perform daily activities with no significant issues. Discussion:Patients with fixed deformity of more than three weeks have a higher rate for recurrence or persistent deformity, as reduction is harder and difficult to maintain. The use of posterior wiring alone is limited in maintaining reduction, while using transarticular screws alone is considered better in maintaining reduction; however, not providing it. Conclusion:The use of posterior cervical fusion using C-wire, transarticular screws, and autologous bone grafting may be applied in recurrent case of AARS to ensure adequate reduction and fixation of the atlantoaxial joint.
| S-EPMC7198983 | biostudies-literature
Project description:ObjectiveThis study evaluated the feasibility and efficacy of quantitative reduction and fixation to treat basilar invagination (BI) with atlantoaxial dislocation (AAD).MethodsPosterior occipitocervical angle (POCA), occiput-C2 angle (Oc-C2A), clivusaxial angle (CAA), and C2-7 angle (C2-7A) were considered for quantitative reduction. Twelve patients with BI complicated with AAD received posterior interarticular release and individualized cage implantation to restore vertical dislocation. The POCA was adjusted using cantilever technology to further reduce the horizontal dislocation and adjust lower cervical vertebral angle. All patients received a radiological follow-up for ≥12 months. Improvements in spinal cord function were evaluated using Japanese Orthopedic Association (JOA) score.ResultsAll the patients received successful quantitative reduction for BI-AAD, and bony fusion was achieved without spinal cord injury after surgery for 12 months. The JOA score was improved significantly to 15.2 ± 0.9 twelve months after surgery (p < 0.01). Radiological follow-up revealed that individualized cage and POCA play vital roles in quantitative correction: (1) distance of the dens above McRae's line and atlantodens interval were restored to normal level, respectively; (2) changes in Oc-C2 angle (ΔOc-C2A), C2-7 angle (ΔC2-7A), clivus-axial angle (ΔCAA), and POCA (ΔPOCA) were all caused by changes in axis tilt. Based on the changes of radiological parameter we deduced the formula for quantitative reduction by linear regression analysis: -ΔPOCA = ΔOc-C2A = -ΔC2-7A = ΔCAA.ConclusionQuantitative posterior reduction by individualized cage and adjusting ΔPOCA is feasible for treating BI with AAD.
| S-EPMC7538363 | biostudies-literature
Project description:Study Design Case report and literature review. Objective To describe a case of nontraumatic atlantoaxial rotatory subluxation (Grisel syndrome) and to review clinical and radiologic aspects, physiopathology, and treatment of this lesion. There is no well-established protocol in the management of patients without spontaneous reduction. The authors discuss the available strategies to achieve reduction and when to operate on these patients. Methods Case presentation of a 7-year-old patient who presented with torticollis ∼1 week after the onset of an upper airway infection. There was no history of head or neck trauma. Computed tomography demonstrated atlantoaxial rotatory subluxation and a normal atlantodental interval. Results The patient was treated with nonsteroidal anti-inflammatory drugs and antibiotics and by progressively increasing the soft cervical collar height. Clinical reduction of the subluxation occurred after 48 hours. He wore the rigid collar for 6 weeks. At that moment, the patient was completely asymptomatic and follow-up cervical spine radiograph demonstrated an anatomical C1-C2 relation. The patient was instructed to return to daily life activities in a gradual manner. Conclusions Grisel syndrome should be considered in the differential diagnosis of torticollis, especially in children. The management can be planned according to the classification of Fielding and Hawkins. The initial treatment involves medicines, injury reduction, and cervical spine immobilization. Surgical treatment is indicated only in cases of failure of conservative treatment, recurrences of subluxation, and irreducible subluxations.
| S-EPMC4111947 | biostudies-other
Project description:INTRODUCTION:Recently, a novel hypothesis has been proposed concerning the origin of craniovertebral junction (CVJ) abnormalities. Commonly found in patients with these entities, atlantoaxial instability has been suspected to cause both Chiari malformation type I and basilar invagination, which renders the tried and tested surgical decompression strategy ineffective. In turn, C1-2 fusion is proposed as a single solution for all CVJ abnormalities, and a revised definition of atlantoaxial instability sees patients both with and without radiographic evidence of instability undergo fusion, instead relying on the intraoperative assessment of the atlantoaxial joints to confirm instability. METHODS:The authors conducted a comprehensive narrative review of literature and evidence covering this recently emerged hypothesis. The proposed pathomechanisms are discussed and contextualized with published literature. CONCLUSION:The existing evidence is evaluated for supporting or opposing sole posterior C1-2 fusion in patients with CVJ abnormalities and compared with reported outcomes for conventional surgical strategies such as posterior fossa decompression, occipitocervical fusion, and anterior decompression. At present, there is insufficient evidence supporting the hypothesis of atlantoaxial instability being the common progenitor for CVJ abnormalities. Abolishing tried and tested surgical procedures in favor of a single universal approach would thus be unwarranted.
| S-EPMC7295832 | biostudies-literature
Project description:ObjectiveDevelopmental bony craniovertebral junction (CVJ) anomalies seem to have a genetic basis and also abnormal joint morphology causing atlantoaxial dislocation (AAD) and basilar invagination (BI).MethodsDNA 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).ResultsThirty-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°.ConclusionThe 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.
| S-EPMC7788422 | biostudies-literature
Project description:The aim was to report a rare case of isolated traumatic atlantoaxial rotatory subluxation without ligamentous injury. Management consisted of analgesia, sedation, and application of a halo skull traction device. After removing halo skull traction, full reduction and recovery were achieved without instability.
| S-EPMC4826932 | biostudies-literature
Project description:BackgroundBecause of the severity and fatal outcome of traumatic vertical atlantoaxial dislocation (AAD), most patients may die in the early post-traumatic period. The post-injury management of patients with vertical AAD has been rarely reported. Improper treatment may lead to disastrous outcome and further aggravate the neurologic symptoms.Case presentationThis report describes the perioperative management and outcome of a rare improperly treated patient with traumatic vertical AAD. The severe pulmonary infection of this patient prevented further surgery for vertical AAD. After placement of a halo vest, combined with effective antibiotic drug treatment, the patient's pulmonary infection was brought under control. The patient underwent atlantoaxial fusion using C1 lateral mass screws and C2 pedicle screws with the assistance of the halo vest. A computed tomography scan at 1 year follow-up indicated that the bone graft was fused and the patient was able to walk independently.ConclusionSkull traction is contraindicated in patients with traumatic vertical AAD. Application of a halo vest can be used for temporary fixation of the cervical spine and atlantoaxial fixation should be performed to maintain the stability of atlantoaxial articulation.
| S-EPMC9891968 | biostudies-literature
Project description:Introduction: Snapping biceps femoris tendon over the fibular head is an uncommon cause of snapping knee. We report a rare case of knee pain secondary to subluxation of the long head of the biceps femoris over the fibular head in an athlete with no history of trauma. This case was treated surgically by a modification of Kennedy procedure. Case Report: A 21-year-old Lebanese athletic male presented for a nauseating feeling of something snapping over his lateral knee since the age of 12 when performing self-defense sports with no history of trauma. Snapping begins when the patient flexes above 80° during squatting, cycling, gym exercises, and climbing stairs. Conclusion: A symptomatic snapping biceps femoris tendon is a rare phenomenon with limited reports in the literature. When conservative treatment fails, surgery may be the only successful treatment. A modification of the Kennedy procedure was used. Rerouting of the subluxating superior aspect of the tendon without further dissection and release appears to be an effective treatment that can result in symptom resolution as long as no anatomic variants are present.
| S-EPMC9499056 | biostudies-literature
Project description:OBJECTIVE:The technique of distraction, compression, extension, and reduction (DCER) is effective to reduce, realign, and relieve cranio-spinal compression through posterior only approach. METHODS:Study included all patients with atlantoaxial dislocation and basilar invagination (BI) with occipitalized C1 arch. Study techniques included Nurick grading, computed tomography scan to study atlanto-dental interval, BI, hyper-lordosis, and neck tilt. Sagittal inclination (SI), coronal inclination (CI), cranio-cervical tilt, presence of pseudo-joints, and anomalous vertebral artery were also noted. Patients underwent DCER with/without joint remodeling or extra-articular distraction (EAD) based on the SI being <100°, 100°-160°, or >160° respectively. In cases with pseudo-joints, joint remodeling was performed in type I and EAD in type II. Customized 'bullet shaped' PSC spacers (n=124) and prototype of the universal craniovertebral junction reducer (UCVJR, n=36) were useful. RESULTS:A total of 148 patients with average age 27.25±17.43 years, ranging from 3 to 71 years (87 males) were operated. Nurick's grading improved from 3.14±1.872 to 1.22±1.17 (p<0.0001). Fifty-two percent of total joints (n=154/296 joints) were either type I (19%)/type II (33%) pseudo-j oints. All traditional indices such as Chamberlein line, McRae line, atlanto-dental interval, and Ranawat line improved (p<at least 0.001). BI, SI, and CI values correlated with type of pseudo-joints (p<0.0001). Side of neck tilt correlated with the type of pseudo-joint (p<0.0001). Cervical hyperlordosis improved significantly (p<0.0001). CONCLUSION:Occipito-C2 pseudo-joints are important in determining the severity of BI. Asymmetrical pseudo-joint causes coronal/neck tilt. Type of pseudo-joint can strategize by DCER. Customized instruments and implants make technique safe, effective and easier.
| S-EPMC6603831 | biostudies-literature
Project description:Brachytelephalangic chondrodysplasia punctata (CDPX1) is characterized by brachytelephalangy and nasomaxillary hypoplasia, in addition to stippled epiphyses. Some reports have described infants with CDPX1 who exhibited cervical spinal stenosis. However, the natural course of cervical spinal lesions in this condition has not been elucidated. Here, we report a very rare adolescent case of CDPX1, which demonstrated progressive myelopathy caused by atlantoaxial subluxation and a subsequent retroodontoid pseudotumor, successfully treated with surgery. Our case highlights a new clinically important fact that upper cervical spinal lesions in CDPX1 can deteriorate even after childhood and thus need close monitoring.
| S-EPMC6369497 | biostudies-literature