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Mitchel Seruya, Albert K. Oh, Michael J. Boyajian, Jeffrey C. Posnick and Robert F. Keating

Object

Patients with delayed presentation of isolated sagittal synostosis (ISS) pose unique surgical challenges. Intracranial hypertension can be missed in the absence of overt findings. Here, an algorithm is presented for the elucidation of intracranial hypertension and approaches to surgical treatment during calvarial reconstruction.

Methods

Patients with delayed presentation (age > 15 months) of ISS between 1997 and 2009 were identified. Symptoms, signs, and radiological evidence of intracranial hypertension were noted. Intraoperative management included calvarial reconstruction in conjunction with ventriculostomy for intracranial pressure (ICP) monitoring and CSF drainage in the setting of suspected intracranial hypertension.

Results

Seventeen patients underwent calvarial reconstruction for delayed presentation of ISS. The mean surgical age was 40.5 months (16.2–82.9 months), and the average follow-up was 34.2 months (0.6–92.2 months). Eleven patients with subtle findings of intracranial hypertension underwent ICP monitoring during calvarial reconstruction. The mean opening ICP was 23.5 cm H2O (16.5–29.5 cm H2O), and the mean closing ICP was 7.0 cm H2O (3.5–17.0 cm H2O). Nine (81.8%) of 11 monitored patients demonstrated intracranial hypertension (ICP ≥ 20 cm H2O); the other 2 had borderline increased ICP. Perioperative morbidity was 5.9%, with 1 patient medically treated for transient, new-onset intracranial hypertension. The postoperative Whitaker category was I in 94.1% of patients, II in 5.9%, and III/IV in 0%. There were no reoperations or deaths.

Conclusions

In this consecutive series, 11 of 17 patients with delayed presentation of ISS underwent ICP monitoring during calvarial reconstruction as a result of subtle clinical findings of intracranial hypertension. Nine (81.8%) of 11 monitored patients demonstrated intracranial hypertension. Calvarial reshaping along with ICP monitoring and CSF drainage facilitated reconstruction and resulted in good outcomes and the resolution of intracranial hypertension.

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Acalvaria

Case report

Ammar H. Hawasli, Thomas L. Beaumont, Timothy W. Vogel, Albert S. Woo and Jeffrey R. Leonard

Acalvaria is a rare congenital malformation characterized by an absence of skin and skull. The authors describe a newborn at an estimated 38 weeks gestational age who was delivered via cesarean section from a 32-year-old mother. Upon delivery, the child was noted to have a frontal encephalocele and an absence of calvaria including skull and skin overlying the brain. A thin membrane representing dura mater was overlying the cortical tissue. After multiple craniofacial operations, including repair of the encephalocele and application of cultured keratinocytes over the rostral defect, the patient demonstrated significant closure of the calvarial defect and was alive at an age of more than 17 months with near-average development.

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Kristen Radcliff, Christopher K. Kepler, Todd A. Rubin, Motasem Maaieh, Alan S. Hilibrand, James Harrop, Jeffrey A. Rihn, Todd J. Albert and Alexander R. Vaccaro

Object

The load-sharing score (LSS) of vertebral body comminution is predictive of results after short-segment posterior instrumentation of thoracolumbar burst fractures. Some authors have posited that an LSS > 6 is predictive of neurological injury, ligamentous injury, and the need for surgical intervention. However, the authors of the present study hypothesized that the LSS does not predict ligamentous or neurological injury.

Methods

The prospectively collected spinal cord injury database from a single institution was queried for thoracolumbar burst fractures. Study inclusion criteria were acute (< 24 hours) burst fractures between T-10 and L-2 with preoperative CT and MRI. Flexion-distraction injuries and pathological fractures were excluded. Four experienced spine surgeons determined the LSS and posterior ligamentous complex (PLC) integrity. Neurological status was assessed from a review of the medical records.

Results

Forty-four patients were included in the study. There were 4 patients for whom all observers assigned an LSS > 6, recommending operative treatment. Eleven patients had LSSs ≤ 6 across all observers, suggesting that nonoperative treatment would be appropriate. There was moderate interobserver agreement (0.43) for the overall LSS and fair agreement (0.24) for an LSS > 6. Correlations between the LSS and the PLC score averaged 0.18 across all observers (range −0.02 to 0.34, p value range 0.02–0.89). Correlations between the LSS and the American Spinal Injury Association motor score averaged −0.12 across all observers (range −0.25 to −0.03, p value range 0.1–0.87). Correlations describing the relationship between an LSS > 6 and the treating physician's decision to operate averaged 0.17 across all observers (range 0.11–0.24, p value range 0.12–0.47).

Conclusions

The LSS does not uniformly correlate with the PLC injury, neurological status, or empirical clinical decision making. The LSSs of only one observer correlated significantly with PLC injury. There were no significant correlations between the LSS as determined by any observer and neurological status or clinical decision making.

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D. Greg Anderson, Worawat Limthongkul, Amirali Sayadipour, Christopher K. Kepler, James S. Harrop, Mitchell Maltenfort, Alexander R. Vaccaro, Alan Hilibrand, Jeffrey A. Rihn and Todd J. Albert

Object

Lumbar degenerative spondylolisthesis (LDS) is common and has generally been characterized as a homogeneous disease entity in the literature and in clinical practice. Because disease variability has not been carefully characterized, stratification of treatment recommendations based on scientific evidence is currently lacking. In this study, the authors analyzed radiographic parameters of patients with LDS at the L4–5 level to better characterize this entity.

Methods

Demographic data were collected from 304 patients (200 women and 104 men) with LDS at the L4–5 level. Plain radiographs including anteroposterior, lateral, and flexion-extension lateral radiographs were analyzed for disc height, segmental angulation, segmental translation, and osteophyte formation. Correlations were sought between the variables of age, sex, disc height, segmental angulation, segmental translation, and osteophyte formation.

Results

The mean patient age was 63.8 years (range 40–86 years). The mean mid-disc height was 7 mm (range 0–14 mm) on the neutral lateral view. The mean angulation between the superior endplate of L-5 and the inferior endplate of L-4 was 6° of lordosis (range 13° of kyphosis to 23° lordosis) on the neutral lateral view. The mean angular change between flexion and extension lateral radiographs was 5° (range 0°–17°). The mean translation on the neutral lateral view was 6 mm (range 0–15 mm). The mean change in translational between flexion and extension was 2 mm (range 0–11 mm). Twenty patients (7%) exhibited spondylolisthesis only on the flexion view. A significant positive correlation was found between the change in angulation and the change in translation on flexion and extension views (ρ = 0.18, p = 0.001). No significant correlation was found between anterior osteophyte size and mobility with flexion-extension radiographs.

Conclusions

The wide range in all radiographic parameters for LDS confirms the heterogeneous nature of this condition and suggests that a grading system to subclassify LDS may be clinically useful. On flexion and extension radiographs, increased translational motion correlated with increased angular motion. Anterior osteophyte size was not found to be predictive of segmental stability. This data set should prove beneficial to those seeking to subcategorize LDS in the future.

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Robert J. Maciunas, Brian N. Maddux, David E. Riley, Christina M. Whitney, Mike R. Schoenberg, Paula J. Ogrocki, Jeffrey M. Albert and Deborah J. Gould

Object

The severity of Tourette syndrome (TS) typically peaks just before adolescence and diminishes afterward. In some patients, however, TS progresses into adulthood, and proves to be medically refractory. The authors conducted a prospective double-blind crossover trial of bilateral thalamic deep brain stimulation (DBS) in five adults with TS.

Methods

Bilateral thalamic electrodes were implanted. An independent programmer established optimal stimulator settings in a single session. Subjective and objective results were assessed in a double-blind randomized manner for 4 weeks, with each week spent in one of four states of unilateral or bilateral stimulation. Results were similarly assessed 3 months after unblinded bilateral stimulator activation while repeated open programming sessions were permitted.

Results

In the randomized phase of the trial, a statistically significant (p < 0.03, Friedman exact test) reduction in the modified Rush Video-Based Rating Scale score (primary outcome measure) was identified in the bilateral on state. Improvement was noted in motor and sonic tic counts as well as on the Yale Global Tic Severity Scale and TS Symptom List scores (secondary outcome measures). Benefit was persistent after 3 months of open stimulator programming. Quality of life indices were also improved. Three of five patients had marked improvement according to all primary and secondary outcome measures.

Conclusions

Bilateral thalamic DBS appears to reduce tic frequency and severity in some patients with TS who have exhausted other available means of treatment.

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Jennifer M. Strahle, Rukayat Taiwo, Christine Averill, James Torner, Chevis N. Shannon, Christopher M. Bonfield, Gerald F. Tuite, Tammy Bethel-Anderson, Jerrel Rutlin, Douglas L. Brockmeyer, John C. Wellons III, Jeffrey R. Leonard, Francesco T. Mangano, James M. Johnston, Manish N. Shah, Bermans J. Iskandar, Elizabeth C. Tyler-Kabara, David J. Daniels, Eric M. Jackson, Gerald A. Grant, Daniel E. Couture, P. David Adelson, Tord D. Alden, Philipp R. Aldana, Richard C. E. Anderson, Nathan R. Selden, Lissa C. Baird, Karin Bierbrauer, Joshua J. Chern, William E. Whitehead, Richard G. Ellenbogen, Herbert E. Fuchs, Daniel J. Guillaume, Todd C. Hankinson, Mark R. Iantosca, W. Jerry Oakes, Robert F. Keating, Nickalus R. Khan, Michael S. Muhlbauer, J. Gordon McComb, Arnold H. Menezes, John Ragheb, Jodi L. Smith, Cormac O. Maher, Stephanie Greene, Michael Kelly, Brent R. O’Neill, Mark D. Krieger, Mandeep Tamber, Susan R. Durham, Greg Olavarria, Scellig S. D. Stone, Bruce A. Kaufman, Gregory G. Heuer, David F. Bauer, Gregory Albert, Jeffrey P. Greenfield, Scott D. Wait, Mark D. Van Poppel, Ramin Eskandari, Timothy Mapstone, Joshua S. Shimony, Ralph G. Dacey Jr., Matthew D. Smyth, Tae Sung Park and David D. Limbrick Jr.

OBJECTIVE

Scoliosis is frequently a presenting sign of Chiari malformation type I (CM-I) with syrinx. The authors’ goal was to define scoliosis in this population and describe how radiological characteristics of CM-I and syrinx relate to the presence and severity of scoliosis.

METHODS

A large multicenter retrospective and prospective registry of pediatric patients with CM-I (tonsils ≥ 5 mm below the foramen magnum) and syrinx (≥ 3 mm in axial width) was reviewed for clinical and radiological characteristics of CM-I, syrinx, and scoliosis (coronal curve ≥ 10°).

RESULTS

Based on available imaging of patients with CM-I and syrinx, 260 of 825 patients (31%) had a clear diagnosis of scoliosis based on radiographs or coronal MRI. Forty-nine patients (5.9%) did not have scoliosis, and in 516 (63%) patients, a clear determination of the presence or absence of scoliosis could not be made. Comparison of patients with and those without a definite scoliosis diagnosis indicated that scoliosis was associated with wider syrinxes (8.7 vs 6.3 mm, OR 1.25, p < 0.001), longer syrinxes (10.3 vs 6.2 levels, OR 1.18, p < 0.001), syrinxes with their rostral extent located in the cervical spine (94% vs 80%, OR 3.91, p = 0.001), and holocord syrinxes (50% vs 16%, OR 5.61, p < 0.001). Multivariable regression analysis revealed syrinx length and the presence of holocord syrinx to be independent predictors of scoliosis in this patient cohort. Scoliosis was not associated with sex, age at CM-I diagnosis, tonsil position, pB–C2 distance (measured perpendicular distance from the ventral dura to a line drawn from the basion to the posterior-inferior aspect of C2), clivoaxial angle, or frontal-occipital horn ratio. Average curve magnitude was 29.9°, and 37.7% of patients had a left thoracic curve. Older age at CM-I or syrinx diagnosis (p < 0.0001) was associated with greater curve magnitude whereas there was no association between syrinx dimensions and curve magnitude.

CONCLUSIONS

Syrinx characteristics, but not tonsil position, were related to the presence of scoliosis in patients with CM-I, and there was an independent association of syrinx length and holocord syrinx with scoliosis. Further study is needed to evaluate the nature of the relationship between syrinx and scoliosis in patients with CM-I.