Iatrogenic atlantoaxial rotatory subluxation after thyroidectomy in a pediatric patient:A case report

BACKGROUND Atlantoaxial rotatory subluxation(AARS)is an uncommon disease with a greater prevalence among children than adults,and it is mostly associated with trauma.Iatrogenic spinal injury accounts for a low percentage of injuries.However,in AARS,20%-40%of cases are associated with surgery,and 48%are caused by infection.Here,we describe our experience with a case of iatrogenic AARS after general anesthesia.CASE SUMMARY A 12-year-old girl presented with right-sided torticollis and cervical motion limit.The patient had undergone thyroidectomy 2 mo ago.Computed tomography revealed AARS with bilateral locked facets.Following the failure of repeated external reduction under general anesthesia,the patient underwent an open surgical reduction.The patient gained atlantoaxial alignment without any complications.Follow-up radiographs showed a normal appearance without instability.The cervical spine of children is more predisposed to injury due to anatomical and biomechanical differences.AARS secondary to infection and surgery is known as Grisel’s syndrome,which involves non-traumatic AARS.Several cases of AARS after surgery and other procedures with no evidence of inflammation have been reported.Our experience shows that surgery requiring hyperextension of the neck after general anesthesia should also be included as a risk factor.CONCLUSION Surgeons and anesthesiologists should be careful not to excessively extend the neck during pediatric surgery.Moreover,clinicians caring for pediatric patients with recent head and neck procedures must be aware of common AARS presentations.

Posterior pedicle screw fixation combined with local steroid injections for treating axial eosinophilic granulomas and atlantoaxial dislocation:A case report

BACKGROUND Eosinophilic granuloma(EG)is a proliferative condition that affects the cells of bone tissue.There are no specific clinical signs or imaging manifestations in the early stages of the disease,making it simple to overlook and misdiagnose.Because of the disease's rarity,there is presently no standardized treatment principle.There are few accounts of such occurrences affecting the axis among children.We discovered a case of a child whose EG resulted in atlantoaxial joint dislocation and destruction of the axial bone.CASE SUMMARY After having pharyngeal discomfort for more than six months without a clear explanation,a 6-year-old boy was brought to our hospital.Following a careful evaluation,the pathology indicated a strong likelihood of an axial EG.Ultimately,we decided to treat the boy with posterior pedicle screw fixation and local steroid injections.CONCLUSION EGs of the upper cervical spine are quite uncommon in children,and they are exceedingly easy to overlook or misdiagnose.Posterior pedicle screw fixation and local steroid injections are effective treatments for patients with axial EGs affecting the atlantoaxial junction.

Surgical treatment of atlantoaxial dysplasia and scoliosis in spondyloepiphyseal dysplasia congenita:A case report

BACKGROUND Spondyloepiphyseal dysplasia congenita(SEDC)is a rare autosomal dominant hereditary disease caused by COL2A1 mutations.SEDC primarily involves the skeletal system,with typical clinical manifestations,including short stature,hip dysplasia,and spinal deformity.Due to the low incidence of SEDC,there are only a few case reports regarding the surgical treatment of SEDC complicated with spinal deformities.CASE SUMMARY We report a case of a 16-year-old male patient with SEDC.He presented with typical short stature,atlantoaxial dysplasia,scoliosis,and hip dysplasia.Cervical magnetic resonance imaging showed spinal canal stenosis at the atlas level and cervical spinal cord compression with myelopathy.The scoliosis was a right thoracic curve with a Cobb angle of 65°.He underwent atlantoaxial reduction,decompression,and internal fixation from C1–C2 to relieve cervical myelopathy.Three months after cervical surgery,posterior correction surgery for scoliosis was performed from T3 to L4.Scoliosis was corrected from 66°to 8°and remained stable at 2-year follow-up.CONCLUSION This is the first case report of a patient with SEDC who successfully underwent surgery for atlantoaxial dysplasia and scoliosis.The study provides an important reference for the surgical treatment of SEDC complicated with spinal deformities.

Application of transoral atlantoaxial reduction plate Ⅲ in treatment of complicated atlantoaxial dislocation

Objective To evaluate the biomechanical characteristies and the clinical advantage of transoral atlantoaxial reduction plate (TARP) Ⅲ. Methods Design of TARP-Ⅲ was based on TARP-Ⅱ. The screw hole in the axis was moved 1～2mm upwards

Atlantoaxial rotatory displacement in children

AIM To correlate the Pang and Lee class with the clinical course in a consecutive series of patients presenting with painful torticollis.METHODS Forty-seven dynamic rotational computed tomography(CT) scans in 35 patients were classified into one of the five types defined by Pang and Li, including types Ⅰ(atlantoaxial rotatopry fixation), Ⅱ("pathologic stickiness" without crossover of C1 on C2), Ⅲ("pathologic stickiness" with crossover of C1 on C2), Ⅳ(normal or muscular torticollis), and Ⅴ(diagnostic grey zone). The Pang and Li class was then compared with the radiologist's report, which was graded abnormal, diagnosis of rotatory subluxation or fixation, or non-diagnostic. Medical records were reviewed and the clinical course was compared among the five subtypes.RESULTS We reviewed 47 CT scans in 35 patients, and the majority were performed without sedation. The average age was 7.7 years(4-14 years old) and associated conditions included minor trauma(20%), surgical procedures around the head and neck(29%), and Grisels syndrome(20%). Twenty-six percent of our studies fell within the pathologic spectrum(5% type 1 or rotatory fixation, 21% types 2 and 3 or rotatory subluxation), while 45% were classified as muscular torticollis(45%) and 28% fell within the diagnostic grey zone. Seven radiologists interpreted these studies, and their interpretation was discordant in 45% of cases. Clinical resolution occurred in 27 of 29 cases for which follow-up was available. One of two patients with fixed rotatory subluxation required a C1-C2 arthrodesis.CONCLUSION The Pang and Li classification characterizes a spectrum of abnormalities in rotation to facilitate communication, although the indications for dynamic CT scan should be further defined.

Single door laminoplasty plus posterior fusion for posterior atlantoaxial dislocation with congenital malformation:A case report and review of literature

BACKGROUND Posterior atlantoaxial dislocation(PAD)is a rare type of upper cervical spine disease.We sought to describe a unreported case of old PAD with os odontoideum(OO)and atlas hypoplasia(AH)and our unique treatment approach consisting of C1 single door laminoplasty with C1-3 posterior fixation and fusion.CASE SUMMARY A 70-year-old male patient who suffered from progressive aggravating numbness and limb weakness for 4 years without trauma,was diagnosed with old PAD with OO and AH.The patient underwent closed reduction and C1 single door laminoplasty with C1-3 posterior fixation and fusion instead of C1 laminectomy with occipitocervical fusion.During the 3-year follow-up,he was able to walk by himself instead of using a wheelchair and with a±25°range of head rotation as well as a±10°range of flexion-extension.Three-year follow-up images showed satisfactory reduction and fusion.CONCLUSION C1 single door laminoplasty with cervical fusion in PAD combined with spinal cord compression could be a suitable and effective surgical option.Compared with laminectomy and occipitocervical fusion,it retains more cervical range of motion,has a smaller incision and provides an adequate bone grafting space for atlantoaxial fusion.

Surgical resection of intradural extramedullary tumors in the atlantoaxial spine via a posterior approach

BACKGROUND The anatomical features of the atlantoaxial spine increase the difficulty of complete and safe removal of atlantoaxial intradural extramedullary(IDEM)tumors.Studies concerning surgical interventions via a posterior approach are limited.AIM To investigate the safety and efficacy of atlantoaxial IDEM tumor resection using a one-stage posterior approach.METHODS We retrospectively analyzed clinical databases for one-stage atlantoaxial IDEM tumor resection via a posterior approach between January 2008 and January 2018.The analyzed data included tumor position,histopathological type,pre-and postoperative Japanese Orthopedic Association(JOA)scores and Nurick grades,postoperative complication and recurrence status.RESULTS A total of 13 patients who underwent C1-C2 Laminectomy and/or unilateral facetectomy via the posterior approach were enrolled in the study.In all cases reviewed,total tumor resection and concomitant C1-C2 fusion were achieved.The average follow-up was 35.3±6.9 mo(range,26-49 mo).A statistically significant difference was noted between the preoperative JOA score(11.2±1.1)and the score at the last final follow-up(15.6±1.0)(P<0.05).A statistically significant difference was noted between the preoperative Nurick grade(2.3±0.9)and that at the last follow-up(1.2±0.4)(P<0.05).However,no statistically significant difference was noted between the preoperative and last follow-up C1-2 Cobb angle and C2-7 Cobb angle(P>0.05).No mortalities,severe complications or tumor recurrence were observed during the follow-up period.CONCLUSION Total resection of atlantoaxial IDEM tumors is feasible and effective via a posterior approach.Surgical reconstruction should be considered to avoid iatrogenic kyphosis and improve spinal stability and overall clinical outcomes.

Early reoccurrence of traumatic posterior atlantoaxial dislocation without fracture:A case report

BACKGROUND In general,atlantoaxial dislocation is rare due to the stability of the C1-C2 complex.Traumatic atlantoaxial dislocations are usually anterior and accompanied by odontoid fractures.Posterior atlantoaxial dislocations are rare,and complete posterior dislocation without associated fracture is even more rare.A case of early recurrence of posterior atlantoaxial dislocation without fracture being in therapy of first closed reduction and then open reduction has not been previously reported.CASE SUMMARY A 45-year-old female presented with traumatic posterior atlantoaxial dislocation(TPAD)of C1-C2 without associated fractures,and Frankel Grade B spinal cord function.She was successfully managed by immediate closed reduction under skull traction.Unexpectedly,17 d later,re-dislocation was discovered.On day 28,closed reduction was performed as before but failed.Then,open reduction and posterior internal fixation with autologous iliac bone grafts was performed.By 6 mo after surgery,atlantoaxial joint fusion was achieved,and neurological function had recovered to Frankel Grade E.At 12 mo follow-up,she had lost only 15°of cervical rotation,and atlantoaxial complex instability in joint flexing and extending were no longer observed under fluoroscopy.CONCLUSION Early assessment of transverse ligament is critical for TPAD without fracture avoiding re-dislocation after closed reduction.

Posterior Atlantoaxial Fusion Using C1 Lateral Mass and C2 Pedicle by Polyaxial Screw in Atlantoaxial Instability

Objective: The aim of this study is to evaluate clinical and radiological outcomes in subjects of atlantoaxial instability which were operated using C1 lateral mass and C2 pedicle screw (Harmes technique). Materials and Methods: Twenty patients with atlantoaxial instability were scheduled at our clinic for atlantoaxial fusion using polyaxial C1 lateral mass and C2 pedicle screw between January 2008 and March 2014. Results: Trauma was the most common cause of atlantoaxial instability, seen in 18 (90%) patients. Modes of trauma were road traffic accident (75%) and falling (15%). Osteoarthritis was observed in 10% of cases. Patients were followed up with radiographs and clinical examinations. Satisfactory screw placement and reduction were achieved in all patients with the average union time of 3.5 months. There were no implant failures, nonunions, vertebral artery injuries or C2 nerve root injuries. Only, surgical site infections occur in 20% of cases that are improved with medical treatment. Conclusions: C1 lateral mass and C2 pedicle polyaxial screw fixation is an effective technique for the fusion of the atlantoaxial complex. It provides the highest fusion rates without any risk of vertebral injury.

Atlantoaxial joint fusion using anterior transarticular screw fixation and bone grafting for atlantoaxial joint instability

Objective To evaluate the clinical application of atlantoaxial joint fusion using anterior transarticular screw fixation and bone grafting for atlantoaxial joint instability. Methods Twenty-three cases of atlantoaxial joint instability were

Atlantoaxial pedicle screw fixation for old odontoid fracture combined with atlantoaxial instability

Objective To explore the clinical effect of the trans-atlantoaxial pedicle screw-rod internal fixation and fusion in treatment of old odontoid fracture combined with atlantoaxial instability. Methods The study involved 48 patients with

Atlantoaxial pedicle screw system for treatment of unstable atlantoaxial dislocation post traction

Objective To explore the technique and clinical outcome of the atlantoaxial pedicle screw system in the treatment of the unstable altantoaxial dislocation post traction.

One stage anterior release and posterior fusion for the treatment of irreducible atlantoaxial dislocation secondary to os odontoideum

Objective To evaluate clinical effect of the ventral release through high anterior cervical retropharyngeal approach and one stage posterior fusion for the treatment ofirreducible atlantoaxial dislocation (IAAD) secondary

Three-dimensional CT angiography study on the relations between the vertebral artery and atlantoaxial joint

Background The vertebral artery （VA） and atlantoaxial joint （AAJ）, with complicated structures, are located in the depths of the head-neck boundary area, the regional anatomy of which cannot be shown globally and directly. This study aims to evaluate three-dimensional CT angiography （3DCTA） in displaying the AAJ, atlantoaxial segment of the vertebral artery （ASVA） and the identification of their interrelations. Methods Sixty-eight subjects without pathology of the ASVA and AAJ were selected from head-neck CTA examination. All the 3D images were formed with volume rendering （VR） together with techniques of separating, fusing, opacifying and false-coloring （SFOF）. On the 3D images, the ASVA and AAJ were observed, and their interrelations were measured. Results All the 3DCTA images were of high quality and up to our requirements. They could clearly and directly show the ASVA, ascending along the AAJ. There were 5 curves in the course of the ASVA, of which 2 curves were away from the atlantoaxial joint, one in the 2rid curve of 0.0 mm-5.4 mm, the other in the 4th of 2.6 mm-9.2 mm. There was no significant difference in the measurements between left and right （P 〉0.05）. The curved parts of the ASVA slightly expanded, with the biggest diameter of 5.6 mm in the 4th curve. Statistical comparison shows that the left ASVA is larger than the right （P 〈0.05）. Variations of the ASVA were found in 8 cases and of the AAJ in 12. Conclusions 3DCTA can globally and directly demonstrate the structures of the AAJ, ASVA and their interrelations. The 3D imaging data make up and enrich the research contents of regional anatomy and lay the foundation for related study and applications.

C1-C2 pedicle screw fixation for adolescent with os odontoideum associated atlantoaxial dislocation and a compound reduction technique for irreducible atlantoaxial dislocation

To the Editor: Os odontoideum (OO) is a rare congenital anomaly of the second cervical vertebra that is defined as occurring when an independent ossicle is separated from the base of the odontoid process?⑴ In general, it requires surgeries for the patients who suffer from OO-associated atlantoaxial dislocation (AAD), especially for young patients due to their immature bones and the unavoidable tumbles and physical impacts in their daily lives.

Advances in the treatment of cervical rheumatoid: Less surgery and less morbidity

Rheumatoid arthritis is a chronic systemic inflammatory disease that often affects the cervical spine.While it was initially thought that cervical involvement was innocuous,natural history studies have substantiated the progressive nature of untreated disease.Over the past 50 years,there has been further elucidation in the pathophysiology of the disease,as well as significant advancements in medical and surgical therapy.The introduction of disease modifying drugs and biologic agents has reduced the amount of patients with advanced stages of the disease needing surgery.Advancement in instrumentation techniques has improved patient outcomes and fusion rates.The introduction of endoscopic approaches for ventral decompression may further lower surgical morbidity.In this review,we give a brief overview of the pertinent positives of the disease.A discussion of historical techniques and the evolution of surgical therapy into the modern era is provided.With improved medical therapies and lessinvasive approaches,we will likely continue to see less advanced cases of disease and less surgical morbidity.Nonetheless,a thorough understanding of the disease is crucial,as its systemic involvement and need for continued medical therapy have tremendous impact on overall complications and outcomes even in patients being seen for standard degenerative disease with comorbid rheumatoid.

Conservative Treatment in Grisel’s Syndrome Following Tonsillectomy

Grisel’s syndrome is a rare complication that is characterized by non-traumatic atlantoaxial subluxation. It is most commonly seen in children after an upper respiratory tract infection or neck surgery, such as an adenoidectomy, tonsillectomy and mastoidectomy. Grisel’s Syndrome is characterized by torticollis and a limited range of motion of the neck with pain. Here the authors report a case of a Malian 5-year-old girl with Grisel’s syndrome following tonsillectomy. The patient’s complaints were fully resolved after 2 weeks of conservative treatment.

Os Odontoideum: Report of Eight Cases and Review of the Literature

Background: Os odontoideum is a separate odontoid process from the body of the axis. It is a rare anomaly of the craniovertebral junction with evidence of both congenital and acquired causes but the aetiology still debated in the literature. It can lead to instability of the atlantoaxial joint and places the spinal cord at significant risk. Methods: Eight cases of os odontoideum were symptomatic at time of presentation, submitted for general and neurological examination, and investigated with different radiological modalities and managed with surgical fixation and stabilization. These patients followed for one year postoperatively. Results: The study includes six male patients and two female patients with median age 18.7 years. All patients presented with myelopathy and showed clinical evidence of atlantoaxial instability. They submitted for complete radiological investigations (plain x ray, CT scan and MRI) craniocervical junction. They underwent cervical surgical stabilization, two cases by atlantoaxial arthrodesis with wiring, two atlantoaxial fixation by lateral mass screw one of them with transoral odontoiectomy, two atlantoaxial fixations by a Rosen’s Hooks, and two cases craniocervical fixation by Tie frame. Death postoperatively recorded in two patients. Follow-up one year revealed resolution of symptoms in two patients and four patients remained stationary but without deterioration. Conclusion: Os odontoideum is a rare but sometimes lethal abnormality that requires radiological and clinical judgement to ensure the best choice of management and optimum patient’s care.

Chordomas of the upper cervical spine： clinical characteristics and surgical management of a series of 21 patients

Background Chordomas of the upper cervical spine are rare and present unique surgical challenge.This study aimed to describe the clinical characteristics and surgical management of patients with chordomas of the upper cervical spine.Methods Twenty-one patients with chordomas of the upper cervical spine who were treated in Peking University Third Hospital from January 1999 to October 2012 were retrospectively analyzed.Survival was calculated by the Kaplan-Meier method and was compared between groups using the log-rank test.Results The postoperative diagnosis was classical chordoma in 20 cases and chondroid chordoma in one case.The mean operative time was 9.5 hours （range 6-17 hours）,and the mean blood loss was 2 812 ml （range 700-4 800 ml）.There were two postoperative deaths.Unilateral vertebral artery ligation was performed in six patients,cervical nerve roots were cut in six patients,and the external branch of the superior laryngeal nerve was repaired after being cut in one case.Two patients developed postoperative velopharyngeal incompetence,and loosening of the occipitocervical screws was observed in one patient.The recurrence rate was 66.7% （10/15） after a mean follow-up period of 46.8 months （range 14-150 months）.The 5-and 10-year overall survival rates were （39.8±13.1）% and （31.9±12.7）%,respectively.There was a significant difference in survival rate between patients who underwent surgery and those who did not.Conclusion In spite of the high rates of recurrence and complications after surgical treatment of chordomas of the upper cervical spine,intralesional resection combined with adjuvant radiotherapy remains the optimal treatment to prolong survival.

Surgical treatment of a complex craniocervical malformation combined with posterior cranial fossa teratoma:a case report and literature review

Background:Basilar invagination(BI)with atlantoaxial dislocation(AAD)is not uncommon in patients with scoliosis,Klippel-Feil syndrome(KFS),and other bone deformities.Cases with combinations of the abovementioned dislocations and deformities with posterior cranial fossa teratoma are rare in the clinic and difficult to handle.Case presentation:This case presents a 34-year-old woman diagnosed with atlantoaxial dislocation and posterior cranial fossa mass.After two surgeries,the posterior cranial teratoma was completely removed with satisfactory atlantoaxial reduction.The postoperative 1-year follow-up examination showed that the bone graft fusion was successful,without remaining significant dysfunction.Conclusions:The surgical risk of irreducible atlantoaxial dislocation combined with posterior cranial fossa tumor is huge.Thus,it needs to be fully preoperatively evaluated and managed carefully in accordance with sound surgical principles.