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We report on a 57-year-old woman with frequent and powerful retrocollis. The involuntary movements combined with rheumatoid arthritis and osteoporosis led to a fracture of the axial odontoid process. Neurologists with a focus on movement disorders should screen for diseases putting the patient at risk for spinal lesions and consider early treatment.
Rheumatoid arthritis is a chronic, multisystem disease that causes significant morbidity and early mortality. A common source of morbidity in severe rheumatoid arthritis is involvement of the cervical spine. Sudden death due to subluxation of the odontoid process into the medulla oblongata is a dramatic but rare complication. We report the case of a patient who died suddenly. Computerized tomography scan, autopsy, and clinical findings were correlated. Studies of cervical involvement in rheumatoid arthritis were analyzed for cases of vertical subluxation. The case reports of sudden deaths and patients treated for vertical subluxation were reviewed; they revealed a trend toward surgery in patients with neurological abnormalities. However, a comparison of clinical outcome does not demonstrate improved survival with surgical intervention. Randomized trials comparing surgical with nonsurgical therapy are needed.
Os odontoideum (OO) and absentia odontoidei (AO) are rare conditions, which mainly are of interest because they may cause atlanto-axial instability. Traditionally they are considered to be congenital lesions. However, during the latest decades a number of acquired cases have been reported. Presently it is generally believed that the two conditions may exist in both a congenital and an acquired version. A review of the literature about OO fails to disclose any solid evidence in support of the classical theory of congenitality. It is concluded that OO and AO are diseases of childhood and adolescence. They are probably always acquired, and result from avascular necrosis of the odontoid process during the growth process.
The authors have reviewed 94 cases of fractures of the odontoid process, 21 of the with neurological signs. The conclusions were as follows: 1) Conservative treatment in 26 cases led to 9 non-unions; 2) 27 recent fractures were operated on (arthrodesis) with 24 fusions; 3) 54 cases were treated more than 30 days after the accident: 15 ot them demonstrated neurological involvement, 34 of them were operated on; 4) The surgical technique is fully described, usually the operation aimed at fusing C1 to C2; 5) One patient died, none was aggravated, all were somewhat improved on the neurological standpoint; 6) On a functional standpoint neck mobility was less decreased after conservative treatment than after cervical fusion.
The authors propose a theory concerning the pathogeny of the mobile odontoid. They believe that this malformation could result from abnormal shortness of the transverse ligament of the atlas.
Treatment results in 104 patients with odontoid fractures were reviewed. There were 2 type I, 62 type II, 32 type III fractures and eight epiphysiolyses in children <7 years old. Thirty-seven patients were managed nonoperatively using plaster casts, cervical braces, or halo devices. Sixty-seven patients were treated surgically including anterior screw fixation (ASF), posterior fusion (PF), and transoral anterior fusion (TAF). Plaster casts and cervical braces were effective for type I fractures and epiphysiolyses only. Halo devices provided successful results in stable type III fractures. ASF is the treatment of choice for most type II and unstable III fractures including some old cases. PF also provided successful union, although impaired cervical motion remained. It should be reserved for irreducible fractures, established nonunions, and as a salvage procedure. TAF should be limited to exceptional cases requiring anterior spinal cord decompression.
Eleven patients with documented Morquio's syndrome who had dysplasia of the odontoid process and resulting atlanto-axial instability were reviewed. They were found to be at risk for acute traumatic quadriparesis, chronic myelopathy of a variable and often rapid rate of progression, and sudden death by respiratory arrest. The evidence suggests that early prophylactic posterior cervical fusion is indicated for patients with this malformation and that once quadriparesis is established, recovery of function is limited.
The authors describe a new technique for treatment fractures of the dens. An anterior approach (technique of Boehler) is used. It provides for one-stage fusion and no additional disturbance of C1-C2 rotation. Under AP and lateral guidance the body of C2 is drilled medially, and so are the site of fracture. Then comes the gradual impaction of a conical implant of biocompatible resorbable implant [*], tip cephalad. The biopolymer is used for grafting purposes in all type of fractures of the odontoid process, which permits the latter to be fused without using screws or any other metal implant.
Marked evanescent swelling of the prevertebral soft tissues has been found in 2 patients with undisplaced fractures of the odontoid process. In one patient the significance of the swelling was appreciated only in retrospect and after shift had occurred at the fracture site. The experience gained from this patient alerted the radiologist and the clinician so that any further damage could be averted in the second patient. All patients with head injuries require radiography of the cervical spine. Soft-tissue swelling anterior to the atlas should be viewed in a serious light.
A 69-year-old-female with a history of rheumatoid arthritis since 1975 had suffered from dysesthesia of extremities since October 1989. Radiating pain and weakness occurred when she tried to stand up on Dec. 25 in 1989. She was admitted to our hospital in October 1990. Physical examination showed emaciation, hypesthesia of extremities, hypesthesia over the right chest and back, impaired vibration and position sense, and hyperreflexia. Laboratory findings revealed that the erythrocyte sedimentation rate was elevated to 46mm/hr, rheumatoid factor (RF) to 83.1IU/ml and CRP to 3.7mg/dl. Her blood sugar was high and she was diagnosed as having diabetes mellitus. Cervical X ray film showed atlanto-axial subluxation. A pseudotumor around the odontoid process bulging into the spinal canal and compression of the upper cervical cord was observed by MRI. In spite of administration of bucillamine (100mg/day), the size of pseudotumor did not change. Methotrexate (MTX) at a dose of 5mg/week was started in February 1991 and the pseudotumor decreased in size with a concurrent reduction of ESR, RF and CRP. However, the high intensity lesion by T2 weighed image did not change and dysesthesia persisted. The pseudotumor was thought to be due to pannus and it was revealed that MTX was effective for reduction. The persistent dysesthesia was probably due to the degeneration of the upper cervical cord, although diabetic neuropathy may also have played a role.
Clinical and X-ray examination of 140 persons demonstrated a wide range of developmental disorders of the odontoid process, from neglible deviations from the average normal structure to total absence of the process. From the pathogenetic standpoints, some of the variants may be characterized as possessing normal structural features, others are developmental anomalies and defects which call for suboccipital decompression, laminectomy for freeing the compressed bulbarspinal parts of the brain or stabilization of the disturbed craniovertebral relationship.
A patient with platybasia of the skull and temporomandibular joint ankylosis is presented. Relief of brain stem compression by the impingement of the odontoid through a soft, hypoplastic clivus was done by first doing a mandibular condylectomy to open the mouth. A transoral-transpharyngeal route was then employed to excise the odontoid process.
Primary screw fixation of a Type II odontoid fracture or non-union is an attractive alternative to posterior atlanto-axial arthrodesis in that normal cervical motion can be maintained. Eight cervical cadaver spines, ranging in age from 17-90 years, were used for study. Type II fractures of the dens were created using an osteotome. Simulated fractures were fixed using one or two 3.5-mm bone screws. After testing each screw fixation technique, the screws were removed and a posterior C1-C2 brooks sublaminar wiring was performed using four 18-gauge wires with wooden blocks to simulate bone graft. No significant differences were found between bending and torsional stiffnesses for the one-screw and two-screw specimens. No significant differences were found between one- and two-screw fixation when compared with primary C1-C2 wiring in torsion. One- or two-screw fixation was as stiff as primary C1-C2 wiring in bending. One or two screws offers similar stability for fixation for a dens fracture. One- and two-screw fixation is at least as stiff as primary C1-C2 wiring in torsion and one- or two-screw fixation is stiffer than primary C1-C2 wiring in bending.
One case of fracture of the atlas associated with rupture of the transverse ligament and three cases of atlantal fracture with associated fractures of the odontoid process are reported. In such cases, halo immobilization is recommended until the atlantal fracture has healed, followed by atlanto-axial fusion for instability of the cervical spine.
Anomalous development of the odontoid is uncommon, and its clinical significance lies in its potential for producing serious neurologic sequelae due to atlantoaxial instability. Although there are several recognized variations (aplasia, hypoplasia, and os odontoideum), clinically they share the same signs and symptoms, and the treatment is identical. Symptoms are usually due to instability of the atlantoaxial joint, with compression of the spinal cord and anteriorly against the axis or posteriorly from the ring of the atlas. Patients may present with no symptoms, with persistent neck complaints, with transient or permanent neurologic deficits, or with sudden death. Symptoms from cranial nerve irritation seldom occur, but occasionally symptoms of cerebral and brain stem ischemia are noted as a result of compression of the vertebral arteries in the area of the atlas. If the condition is suspected, the diagnosis usually can be confirmed on lateral flexion-extension roentgenograms. Special techniques are often required, particularly lateral laminagrams, and flexion-extension stress roentgenograms are necessary to determine the presence and degree of atlantoaxial instability. The role of prophylactic surgical stabilization is not yet established. If instability greater than 5 mm. is demonstrated or the patient has clinical findings of neurologic compromise, surgical fusion should be performed.
The presence of primary position downbeat nystagmus indicates disease at the cervicomedullary junction where craniovertebral anomalies are commonly encountered. This paper reports a patient whose only symptom was disabling oscillopsia and downbeat nystagmus secondary to basilar impression and Arnold-Chiari malformation. He experienced total resolution of his symptoms following transoral removal of the odontoid process. In the past it was felt that ocular signs and symptoms did not improve with surgery for craniovertebral anomalies. In light of this report, the ocular symptoms of craniovertebral anomalies may be considered indications in themselves for surgical intervention.
Scoliosis surgery presents the anesthetist with specific clinical challenges. Since scoliosis is the most common problem for which patients of congenitally short stature present to the operating room, the preoperative evaluation of dwarfs is discussed here in the context of a patient with spondyloepiphyseal dysplasia congenita. In the case described, many of the deformities associated with dwarfism were present. The significance of these to the preoperative, intraoperative, and postoperative care is discussed. Consideration is given to cervical spine abnormalities, congenital absence of the odontoid process, pulmonary function abnormalities, and mucopolysaccharidosis (a syndrome which may compromise airway management). The intraoperative monitoring of somatosensory evoked potentials and their significance are also discussed.
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