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1/66. The Chiari II malformation: lesions discovered within the fourth ventricle.

    Structural lesions associated with the Chiari II malformation have been identified within the fourth ventricle in 8 patients. During the 42-month period encompassing the operations of the 7 patients treated surgically, only 9 other patients were explored without the discovery of some associated structural lesion. The patients ranged in age from 2 to 26 years. The following lesions were identified: glial or arachnoidal cysts (3 patients), glial or choroidal nodules (3 patients) and subependymoma (2 patients). These lesions were all situated in the roof of the fourth ventricle adjacent to or interspersed with the choroid plexus. Only the cystic lesions were identified by preoperative imaging. In only 1 case did the associated lesion, a 2-cm cyst, seem to contribute to the patient's clinical presentation. Structural lesions of the fourth ventricle associated with the Chiari II malformation are common in patients who are submitted to decompression. These lesions may be dysplasias of developmental origin, or they may be reactive lesions related to chronic compression and ischemia. They do not necessarily required biopsy or excision.
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2/66. Syringobulbia caused by delayed postoperative tethering of the cervical spinal cord - delayed complication of foramen magnum decompression for Chiari malformation.

    Postoperative tethering of the high cervical spinal cord is a rare cause of neurological deterioration after foramen magnum decompression (FMD) with duraplasty for Chiari type I malformation. A review of the literature revealed that only 5 cases have been reported. This entity is not widely known to occur as a complication of the common surgical procedure for Chiari type I malformation. A 17-year-old boy experienced rapidly progressive neurological deterioration over a 3-month period. FMD and duraplasty with lyophilized cadaver dura had been performed 8 years previously. Follow-up MR images showed that the cerebrospinal fluid (CSF) space dorsal to the cord was gradually disappearing and that syringobulbia had developed. Opening the dura mater of the posterior fossa revealed dense fibrous scarring, arachnoid thickening over the cervicomedullary area, and tethering the cord to the dura from the medulla to C2. The adhesions were dissected free, and the tethering was released. A syringosubarachnoid (SS) shunt was inserted and duraplasty was performed with an expended polytetrafluoroethylene sheet (Gore-Tex). Postoperative MR images demonstrated that the syringobulbia had completely collapsed and that a dorsal CSF space was present. Follow-up MR images provided significant information on the cervical spinal cord tethering after FMD with duraplasty for Chiari malformation. We encourage sharp surgical detethering and duraplasty with Gore-Tex to avoid retethering. Early recognition and treatment of this unusual but important complication are emphasized.
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3/66. Management of Chiari I malformations with holocord syringohydromyelia.

    The management of patients with Type I Chiari malformations (CM 1) with or without syringohydromyelia (SHM) has remained quite controversial, and many different surgical procedures have been advocated. Over the past several years, the authors have treated 7 children presenting with CM 1 and holocord syringohydromyelia with suboccipital decompression and duraplasty alone without intradural procedures. All children received MRI imaging at 2-4 months and 1 year postoperatively. On the early postoperative MRI examination, marked reduction in the syringohydromyelia was seen in 6 children, with minimal change in syrinx size in 1 child who was clinically improving after the operation. At 1 year, all children with early collapse remained collapsed, and the child with minimal early collapse demonstrated an approximately 50% reduction in syrinx size. Clinical follow-up (mean 30 months, range 21-50 months) showed good results in all patients: none of the children have required further neurosurgical intervention, and all have shown improvement in their preoperative function. One child with a 46 degrees scoliosis had a complete collapse of her SHM, but ultimately required spinal fusion. The presenting clinical findings, operative technique, MRI imaging and clinical outcomes will be discussed. The results from these 7 patients with CM 1 and holocord syringomyelia suggest that posterior fossa decompression alone (without intradural procedures) can provide excellent radiographic and clinical outcome.
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4/66. Respiratory failure caused by Chiari I malformation with associated syringomyelia.

    The authors report a case of a 38-year-old male with a Chiari I malformation and syringomyelia that presented with acute respiratory distress. Pulmonary function testing was consistent with respiratory muscle weakness. A suboccipital craniectomy and C1, 2, 3 laminectomies were performed on an emergent basis. Postoperatively the patient had significant improvement in his respiratory status associated with a reduction in the size of the syringomyelia on follow-up imaging.
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5/66. Presentation of a previously asymptomatic Chiari I malformation by a flexion injury to the neck.

    Flexion injury and/or whiplash injury to the neck in car accidents are usually trivial injuries with no serious neurological deficits. Our intention was to point out the importance to proceed with diagnostic procedures if neurological deficits do occur in order to reveal the true cause of the deficit. The paper presents the case of a 35-year-old woman who sustained a flexion injury to the neck. A relatively trivial injury to the neck promoted a progressive neurological deterioration. The standard diagnostic procedures (x-ray, computed tomography scan) were normal. Further diagnostics with magnetic resonance imaging was required to reveal an underlying Chiari I malformation. Finally, the operative decompression of the craniocervical junction was performed. Following the surgical treatment, the patient's clinical symptoms regressed. One year after her discharge, she remains in good physical condition. To our knowledge, this case is the first report of the manifestation of Chiari I malformation in the adult as a result of a flexion or whiplash injury of the neck. This unusual case suggests that in a trivial flexion injury to the neck sustained in a car accident, which presents with serious neurological dysfunction, and where the standard diagnostic procedures are normal, the possibility of underlying congenital abnormality, such as Chiari I malformation should be considered.
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6/66. Attenuation of somatosensory evoked potentials during positioning in a patient undergoing suboccipital craniectomy for Chiari I malformation with syringomyelia.

    Intraoperative electrophysiologic monitoring can diminish the risk of neurologic injury by enabling the detection of injury at a time when it can be reversed or minimized. This report describes a 14-year-old girl with a Chiari's malformation type I and syringomyelia who underwent a suboccipital decompression and dural patch grafting with concurrent somatosensory evoked potentials. When the patient was turned into the prone position and the neck was flexed, the left-sided somatosensory evoked potential deteriorated. After the patient's neck was repositioned, the left median nerve potential improved but did not return to baseline. Postoperatively, the patient had decreased proprioception of her left arm, which completely resolved at 2-week follow-up. This single case report does not establish the need for routine somatosensory evoked potential monitoring. Nevertheless, deterioration of the potential in this case led directly to a change in the surgical positioning, which may have significantly reduced the chances of a permanent neurologic injury.
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keywords = operative
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7/66. Chiari type I presenting as left glossopharyngeal neuralgia with cardiac syncope.

    Glossopharyngeal neuralgia is an uncommon craniofacial pain syndrome that is occasionally associated with cardiac syncope. However, we relate Chiari I syndrome as a cause of this clinical picture for the first time in the literature. The authors analyze the relevant literature and discuss the pathogenesis and treatment of associated syndromes. We describe the case of a 45-year-old female patient who presented with a 3-year history of left glossopharyngeal neuralgia with occasional dysphagia and episodes of syncope when eating or swallowing. The pain was not disseminated to the right side and was fairly well controlled by carbamazepine. The syncopal attacks had a duration of about 10 sec. Neurological examination elicited a faint dysphonia associated with paradoxical dysphagia. The cranial magnetic resonance imaging scan showed a bilateral herniation of the cerebellar tonsils through the foramen magnum, more evidently on the left side. The patient received a suboccipital craniectomy and resection of the posterior arch of C1. The dura mater was opened, and we found both tonsils displaced into the foramen magnum extending caudally toward the C1 level. Both tonsils were compressing the brainstem and especially the low cranial nerves bilaterally. The lower cranial nerves were compressed between the posterior inferior cerebellar artery (pica) and the herniated cerebellar tonsil on both sides. Both PICAs were dissected by microsurgical technique and all the arachnoid adherences were cut. The left tonsil was resected by means of ultrasonic aspirator. Duraplasty was performed with the occipital pericranium. The paroxysmal pain attacks and the syncopal picture disappeared immediately after the operation. The patient was discharged on the 7th postoperative day. One year later, she was free of symptoms. This case provides clinical evidence of close connections between the glossopharyngeal and vagus nerves, which control visceral sensation; and compression of the IX and X nerves between the herniated tonsil and pica or vertebral artery may cause an irritative sensory phenomenon, which is the origin of the algic sensation and the cardiac syncope by means of cross talk between the fibers of the same nerve.
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8/66. Endoscopic third ventriculostomy for treatment of noncommunicating syringomyelia associated with a Chiari I malformation and hydrocephalus: case report and pathophysiological considerations.

    OBJECTIVE AND IMPORTANCE: A Chiari I malformation associated with syringomyelia and hydrocephalus is a rare condition. We report the successful use of endoscopic third ventriculostomy for the treatment of this pathological entity. The successful use of this technique in such a case has not been previously described, and the results allow us to speculate on the pathophysiological mechanism involved. CLINICAL PRESENTATION: A 34-year-old woman presented with headaches, a motor deficit of the right upper limb, and gait dyspraxia. Magnetic resonance imaging scans demonstrated dilation of all ventricles, compression of the retrocerebellar cerebrospinal fluid space, downward displacement of the tonsils, and syringomyelia. syringomyelia involved the cervicodorsal cord below C3, with a syrinx-free segment between C1 and C3 and no enlargement of the rostral part of the central canal. INTERVENTION: Endoscopic third ventriculostomy resulted in prompt improvement of the clinical symptoms. Postoperative magnetic resonance imaging scans demonstrated shrinkage of the syrinx and return of the cerebellar tonsils to their physiological positions. CONCLUSION: This experience demonstrates that endoscopic third ventriculostomy, which is a simple, safe technique, may be the treatment of choice for associated Chiari I malformations, hydrocephalus, and syringomyelia (even the noncommunicating type).
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9/66. Resolution of Chiari malformation after repair of a congenital thoracic meningocele: case report and literature review.

    OBJECTIVE AND IMPORTANCE: Many theories have been proposed regarding potential causative factors for Chiari malformations. An unusual case is described in which regression of a congenital Chiari malformation was observed after repair of a thoracic meningocele without direct surgical intervention to decompress the craniocervical junction. This supports the importance of an in utero craniospinal pressure gradient as a potential cause for congenital, but reversible, cerebellar herniation. CLINICAL PRESENTATION: A newborn baby was observed to have a thoracic meningocele. magnetic resonance imaging scan revealed a concomitant Chiari malformation. No neurological deficits were present at initial examination. INTERVENTION: The patient underwent surgical closure of the thoracic meningocele and untethering of the spinal cord at the site of the dural defect. A postoperative magnetic resonance imaging scan obtained 3 months after the operation revealed complete resolution of the cerebellar herniation. CONCLUSION: The resolution of the Chiari malformation in this child may have resulted from restoration of normal cerebrospinal fluid flow and elimination of the meningocele-related cerebrospinal fluid pressure gradient between the intracranial and intraspinal compartments.
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10/66. Maternal Arnold-Chiari type I malformation and syringomyelia: a labor management dilemma.

    Arnold-Chiari type I malformations consists of elongation of the cerebellar tonsils with their displacement below the foramen magnum. syringomyelia is an associated cyst that accumulates cerebrospinal fluid in the cord that can impinge on local nerve fibers. pregnant women with either of these disorders are of special concern due to the potential risk of brain stem herniation and or spinal column compression from physiological changes that occur during labor. We present two cases. The first case is a patient with syringomyelia who was admitted in labor with worsening peripheral neurological symptoms. Epidural anesthesia was placed and she underwent an uncomplicated cesarean delivery with resolution of her symptoms postpartum. The second case is a patient with an Arnold-Chiari type I malformation and syringomyelia who presented in labor. The patient had an epidural placed and was allowed to progress to complete dilation and effacement at 2 station. She underwent a successful operative vaginal delivery without voluntary maternal expulsive efforts. Both patients had uncomplicated postpartum courses. Although these are rare disorders with significant potential morbidity, labor can be managed by either mode of delivery with careful patient selection. We caution that this review has insufficient numbers of patients to address the safety and efficacy of either delivery mode but rather focuses on alternatives for delivery. This report is the first to document a case of a patient with an arnold-chiari malformation and syringomyelia successfully managed in labor with a vaginal delivery.
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