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Topic Editor Jerry W. Oakes

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The Chiari I malformation

JNSPG 75th Anniversary Invited Review Article

Samuel G. McClugage III and W. Jerry Oakes

As with many pathologies, the course of our understanding of the Chiari I malformation (CIM) has developed extensively over time. The early descriptions of the Chiari malformations by Hans Chiari in 1891 opened the door for future classification and research on this topic. However, even over a long timeframe, our understanding of the pathophysiology and, more importantly, treatment, remained in its infancy. As recently as the 1970s, CIM was not discussed in popular neurology textbooks. Syringomyelia is listed as a degenerative disorder with no satisfactory treatment. Radiation therapy was considered an option in treatment, and surgery was thought to play no role. During the last 40 years, equivalent to the duration of a neurosurgical career, our understanding of the pathophysiology and natural history of CIM, coupled with modern MRI, has improved the treatment paradigm for this patient population. More importantly, it has given us evidence confirming that CIM is a disorder responsive to surgical intervention, giving patients once thought to be destined for lifelong disability a comparatively normal life after treatment. The purpose of this article is to offer a review of CIM and its important associated entities. The authors will discuss the evolution in understanding of the Chiari malformation and, importantly, distinguish between symptomatic CIM and asymptomatic tonsillar ectopia, based on imaging and presenting symptomatology. They will discuss techniques for surgical intervention, expected outcomes, and complications after surgery. Proper patient selection for surgery based on appropriate symptomatology is tantamount to achieving good surgical outcomes in this population, separating those who can be helped by surgery from those who are unlikely to improve. While our knowledge of the Chiari malformations continues to improve through the efforts of clinical and basic science researchers, surgeons, and patients, our current understanding of these entities represents a monumental improvement in patient care over a relatively short time period.

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Scott Elton and W. Jerry Oakes

Dermal sinus tracts in the spine range from asymptomatic pits to tracts with significant disease. These tracts may be associated with lesions that tether the spinal cord and can either become infected or produce neurological deficits. Over time the treatment of these lesions has varied little: complete resection and intradural exploration are the standard surgical interventions. The authors review their experience with 23 dermal sinus tracts treated in the last 19 years by the senior author. The clinical findings, radiographic appearance, treatment, and pathological findings of these lesions will be discussed. The authors will also provide a summary of the literature covering these lesions.

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W. Jerry Oakes

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R. Shane Tubbs and W. Jerry Oakes

✓Self-mutilation after deafferentation injuries has rarely been reported in humans. The authors report on a 16-year-old girl who was born with a myelomeningocele. In adolescence it was noted that concurrent with her spinal cord becoming symptomatically tethered she began to self-mutilate her digits. A rare manifestation of the tethered spinal cord may be dysesthesias that led to self-mutilation.

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R. Shane Tubbs and W. Jerry Oakes

✓The authors present the case of a child diagnosed as having Beckwith—Wiedemann syndrome and Chiari I malformation. Hemihypertrophy is associated with Beckwith—Wiedemann syndrome and has been described in conjunction with Chiari I malformation. The authors hypothesize that the hemihypertrophy that may involve the skull base and Chiari I malformation found in their patient are not spurious findings but are pathologically related, perhaps by slight dysmorphologies of the posterior cranial fossa.

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R. Shane Tubbs and W. Jerry Oakes

✓The authors present a case of an adolescent with achondroplasia and symptoms of neurogenic claudication who, interestingly, also presented with intermittent priapism. Both the neurogenic claudication and priapism were relieved following a thoracolumbar laminectomy. To our knowledge, this is the first report of a dwarf with thoracolumbar spinal canal stenosis presenting with priapism.

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R. Shane Tubbs and W. Jerry Oakes

Object. The cisternal portion of the trochlear nerve (fourth cranial nerve) can easily be injured during intracranial surgical operations. To help minimize the chance of such injury by promoting a thorough understanding of the anatomy of this nerve and its relationships to surrounding structures, the authors present this anatomical study.

Methods. In this study, in which 12 cadaveric heads (24 sides) were used, the authors describe exact distances between the trochlear nerve and various surrounding structures. Also described are relatively safe areas in which to manipulate or enter the tentorium, and these are referenced to external landmarks.

Conclusions. This information will prove useful in planning and executing surgical procedures in and around the free edge of the tentorium cerebelli.