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Erik C. Brown, Kunal Gupta and Christina Sayama

Lateral meningocele syndrome (LMS) is a rare genetic connective tissue disorder. It is associated with morphological changes similar to those of other connective tissue disorders, with the unique distinction of multiple, often bilateral and large, lateral meningoceles herniating through the spinal foramina. In some cases, these lateral meningoceles can cause pain and discomfort due to their presence within retroperitoneal tissues or cause direct compression of the spinal nerve root exiting the foramen; in some cases compression may also involve motor weakness. The presence of lateral meningoceles imposes unique challenges related to CSF flow dynamics, especially with concurrent Chiari malformation, which also occurs with increased frequency in individuals with LMS.

The authors present the case of a 6-month-old female with LMS with multiple lateral meningoceles throughout the thoracic and lumbar spine. The infant experienced a focal neurological abnormality due to enlargement of her lateral meningoceles following decompression of a symptomatic Chiari malformation and endoscopic third ventriculostomy. The finding was reversed through implantation of a ventriculoperitoneal shunt, which reduced the burden of CSF upon the lateral meningoceles. Such a case compels consideration that CSF flow dynamics in addition to altered connective tissue play a role in the presence of lateral meningoceles in patients within this and similar patient populations.

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Christina M. Sayama, James K. Liu and William T. Couldwell

✓Cerebral vasospasm remains a major source of morbidity and death in patients with aneurysmal subarachnoid hemorrhage (SAH). When vasospasm becomes refractory to maximal medical management consisting of induced hypertension and hypervolemia and administration of calcium channel antagonists, endovascular therapies should be considered. The primary goal of endovascular treatment is to increase cerebral blood flow to prevent cerebral infarction. Two of the more frequently studied endovascular treatments are transluminal balloon angioplasty and intraarterial papaverine infusion. These two have been used either alone or in combination for the treatment of vasospasm. Other pharmacological vasodilating agents currently being investigated are intraarterial nimodipine, nicardipine, verapamil, and milrinone. Newer intraarterial agents, such as fasudil and colforsin daropate, have also been investigated. In this article the authors review the current options in terms of endovascular therapies for treatment of cerebral vasospasm. The mechanism of action, technique of administration, clinical effect and outcomes, and complications of each modality are discussed.

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Christina Sayama, Tsulee Chen, Gregory Trost and Andrew Jea

Pediatric spine fractures constitute 1%–3% of all pediatric fractures. Anywhere from 20% to 60% of these fractures occur in the thoracic or lumbar spine, with the lumbar region being more affected in older children. Younger children tend to have a higher proportion of cervical injuries. The pediatric spine differs in many ways from the adult spine, which can lead to increased ligamentous injuries without bone fractures. The authors discuss and review pediatric lumbar trauma, specifically focusing on epidemiology, radiographic findings, types and mechanisms of lumbar spine injury, treatment, and outcomes.

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Carolyn S. Quinsey, Katie Krause, Lissa C. Baird, Christina M. Sayama and Nathan R. Selden

OBJECTIVE

The relationship between a tethered cord (TC) and neurofibromatosis type 1 (NF1) and NF2 is not known. The purpose of this study was to define the incidence of TC in pediatric neurosurgical patients who present with NF.

METHODS

The authors performed a single-institution (tertiary care pediatric hospital) 10-year retrospective analysis of patients who were diagnosed with or who underwent surgery for a TC and/or NF. Clinical and radiological characteristics were analyzed, as was histopathology.

RESULTS

A total of 424 patients underwent surgery for a TC during the study period, and 67 patients with NF were seen in the pediatric neurosurgery clinic. Of these 67 patients, 9 (13%) were diagnosed with a TC, and filum lysis surgery was recommended. Among the 9 patients with NF recommended for TC-release surgery, 4 (44%) were female, the mean age was 8 years (range 4–14 years), the conus position ranged from L1–2 to L-3, and 3 (33%) had a filum lipoma, defined as high signal intensity on T1-weighted MR images. All 9 of these patients presented with neuromotor, skeletal, voiding, and/or pain-related symptoms. Histopathological examination consistently revealed dense fibroconnective tissue and blood vessels.

CONCLUSIONS

Despite the lack of any known pathophysiological relationship between NF and TC, the incidence of a symptomatic TC in patients with NF1 and NF2 who presented for any reason to this tertiary care pediatric neurosurgery clinic was 13%. Counseling patients and families regarding TC symptomatology might be indicated in this patient population.

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James K. Liu, Christina M. Sayama, Clough Shelton and Joel D. MacDonald

✓Some evidence in the literature supports the topical application of papaverine to the cochlear nerve to prevent internal auditory artery vasospasm and cochlear ischemia as a method of enhancing the ability to preserve hearing during acoustic neuroma surgery. The authors report a case of transient facial nerve palsy that occurred after papaverine was topically applied during a hearing preservation acoustic neuroma removal. A 58-year-old woman presented with tinnitus and serviceable sensorineural hearing loss in her right ear (speech reception threshold 15 dB, speech discrimination score 100%). Magnetic resonance imaging demonstrated a 1.5-cm acoustic neuroma in the right cerebellopontine angle (CPA). A retrosigmoid approach was performed to achieve gross-total resection of the tumor. During tumor removal, a solution of 3% papaverine soaked in a Gelfoam pledget was placed over the cochlear nerve. Shortly thereafter, the quality of the facial nerve stimulation deteriorated markedly. Electrical stimulation of the facial nerve did not elicit a response at the level of the brainstem but was observed to elicit a robust response more peripherally. There were no changes in auditory brainstem responses. Immediately after surgery, the patient had a House–Brackmann Grade V facial palsy on the right side. After several hours, this improved to a Grade I. At the 1-month follow-up examination, the patient exhibited normal facial nerve function and stable hearing.

Intracisternal papaverine may cause a transient facial nerve palsy by producing a temporary conduction block of the facial nerve. This adverse effect should be recognized when topical papaverine is used during CPA surgery.

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Ben A. Strickland, Christina Sayama, Valentina Briceño, Sandi K. Lam, Thomas G. Luerssen and Andrew Jea

OBJECT

In a previous study, the authors reported on their experience with the use of sublaminar polyester bands as part of segmental spinal constructs. However, the risk of neurological complications with sublaminar passage of instrumentation, such as spinal cord injury, limits the use of this technique. The present study reports the novel use of subtransverse process polyester bands in posterior instrumented spinal fusions of the thoracic and lumbar spines and sacrum or ilium in 4 patients.

METHODS

The authors retrospectively reviewed the demographic and procedural data of patients who had undergone posterior instrumented fusion using subtransverse process polyester bands.

RESULTS

Four patients, ranging in age from 11 to 22 years, underwent posterior instrumented fusion for neuromuscular scoliosis (3 patients) and thoracic hyperkyphosis (1 patient). There were 3 instances of transverse process fracture, with application and tensioning of the polyester band in 1 patient. Importantly, there was no instance of spinal cord injury with subtransverse process passage of the polyester band. The lessons learned from this technique are discussed.

CONCLUSIONS

This study has shown the “Eleghia” technique of passing subtransverse process bands to be a technically straightforward and neurologically safe method of spinal fixation. Pedicle screws, laminar/pedicle/transverse process hooks, and sublaminar metal wires/bands have been incorporated into posterior spinal constructs; they have been widely reported and used in the thoracic and lumbar spines and sacrum or ilium with varying success. This report demonstrates the promising results of hybrid posterior spinal constructs that include the Eleghia technique of passing subtransverse process polyester bands. This technique incorporates technical ease with minimal risk of neurological injury and biomechanical stability.

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Sohum K. Desai, Christina Sayama, Daniel Vener, Alison Brayton, Valentina Briceño, Thomas G. Luerssen and Andrew Jea

OBJECT

The authors have previously reported on their early experience with sublaminar polyester bands in spine surgery. In this paper, the authors describe the use of sublaminar polyester bands in long-segment posterior instrumented spinal fusions from the upper thoracic spine to the ilium in 21 children and transitional adults with progressive neuromuscular scoliosis. Transitional adults were patients older than 18 years of age with a spinal disorder of pediatric onset, such as spina bifida. This dedicated study represents the first reported use of polyester bands in spine surgery for neuromuscular scoliosis in this patient population in the US.

METHODS

The authors retrospectively reviewed the demographics and procedural data of patients who underwent posterior instrumented fusion using sublaminar polyester bands for neuromuscular scoliosis.

RESULTS

Twenty-one pediatric and adult transitional patients, ranging in age from 10 to 20 years (mean 14 years), underwent posterior instrumented fusion for progressive neuromuscular scoliosis. The average coronal Cobb angle measured 66° before surgery (range 37°–125°). Immediately after surgery, the mean coronal Cobb angle was 40° (range 13°–85°). At last follow-up, the average coronal Cobb angle was maintained at 42° (range 5°–112°). Regarding sagittal parameters, thoracic kyphosis was restored by 8%, and lumbar lordosis improved by 20% after surgery. Mean follow-up duration was 17 months (range 2–54 months). One patient with an aborted procedure due to loss of intraoperative evoked potentials was excluded from the analysis of radiographic outcomes. Mean surgical time was 7 hours 43 minutes (range 3 hours 59 minutes to 10 hours 23 minutes). All patients received either a 12- or 24-mg dose of recombinant human bone morphogenetic protein–2. Average estimated blood loss was 976 ml (range 300–2700 ml). Complications directly related to the use of sublaminar instrumentation included transient proprioceptive deficit (1 patient) and prolonged paraparesis (1 patient). Other complications noted in this series included disengagement of the rod from an iliac screw (1 patient), proximal junctional kyphosis (1 patient), noninfected wound drainage (2 patients), and perioperative death (1 patient). The lessons learned from these complications are discussed.

CONCLUSIONS

Pedicle screws, laminar/pedicle/transverse process hooks, and sublaminar metal wires have been incorporated into posterior spinal constructs and widely reported and used in the thoracic and lumbar spines and sacrum with varying success. This report demonstrates the satisfactory radiological outcomes of hybrid posterior spinal constructs in pediatric and adult neuromuscular scoliosis that include sublaminar polyester bands that promise the technical ease of passing sublaminar instrumentation with the immediate biomechanical rigidity of pedicle screws and hooks. However, the high neurological complication rate associated with this technique (2/21, or 10%) tempers the acceptable radiographic outcomes.

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Christina M. Sayama, Anne G. Osborn, Steven S. Chin and William T. Couldwell

Object

Brain capillary telangiectasias (BCTs) are small, clinically benign, angiographically occult lesions that are usually incidental findings. Large capillary telangiectasias have not been reported previously as most BCTs are very small. Symptomatic BCTs are also rare, with few reports in the literature. The authors review the clinical manifestations, imaging, and histopathological characteristics of BCTs to further elucidate the diagnostic and clinical features of these vascular malformations.

Methods

The authors completed a retrospective radiological review of all cases of BCTs in the neuroradiology database at the University of Utah involving patients treated between January 1993 and December 2007. The MR imaging scans were reviewed, and the BCT was measured in 2 dimensions. They arbitrarily chose > 1 cm to define a large BCT as a majority of these lesions were smaller than that. The medical chart and the electronic database were used to gather each patient's clinical information.

Results

One hundred thirty patients were identified in the archived neuroradiology database of capillary telangiectasias. Cases involving 105 patients with definite capillary telangiectasias were reviewed, and from these, 7 patients were identified to have a large capillary telangiectasia measuring > 1 cm. Upon further review, 2 of these patients were identified as having symptoms likely related to their capillary telangiectasia. These 2 cases are reported in the article. No patients with smaller BCTs were found to have symptoms related to their lesion.

Conclusions

Brain capillary telangiectasias are small vascular malformations that rarely cause symptoms. They are often overlooked on imaging because of their clinically benign nature; however, they have been misdiagnosed as glial tumors in the past. Specific MR imaging sequences (T1-weighted postcontrast and gradient refocused echo) are valuable in aiding diagnosis, as histopathological diagnosis is often not possible. These cases highlight that BCTs can cause symptoms, a finding that may actually be related to the size of the lesion (28.6% of large BCTs in this series were symptomatic, whereas none of the small ones were).

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Extranodal NK/T-cell lymphoma presenting as a pituitary mass

Case report and review of the literature

James K. Liu, Christina Sayama, Steven S. Chin and William T. Couldwell

✓ Primary pituitary lymphomas (PPLs) are rare tumors of the central nervous system, and most are of B-cell origin. Extranodal NK/T-cell lymphomas are uncommon neoplasms that are highly aggressive and show a strong association with Epstein–Barr virus. They most commonly affect the nasal cavity and paranasal sinuses; manifestation as a primary pituitary tumor has never been described. The authors report a case of NK/T-cell lymphoma of the pituitary gland and review 17 cases of PPL from the literature.

All patients had been evaluated at presentation for clinical, neuroimaging, and histopathological findings. Patients who had systemic lymphoma with secondary involvement of the pituitary gland were excluded. The mean patient age was 55.5 years (range 26–86 years); the male/female ratio was 13:5. The most common presentation was pituitary insufficiency (72%), followed by headache (56%), diplopia (39%), visual loss (28%), and fever (22%). Thirteen patients (72%) exhibited anterior hypopituitarism and seven (39%) had diabetes insipidus at presentation. Magnetic resonance imaging demonstrated enhancing parasellar masses with diffuse enlargement of the pituitary gland (94%), suprasellar extension (44%), cavernous sinus extension (39%), and stalk thickening (22%). Thirteen patients (72%) had B-cell lymphoma, four (22%) had T-cell lymphoma, and one (6%) had NK/T-cell lymphoma.

Primary pituitary lymphomas are rare entities with a range of clinical presentations and neuroimaging findings that are unique from those of patients who present with pituitary adenomas. The pathological entity of NK/T-cell lymphoma is distinct, and its course is very aggressive with a poor prognosis.