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Howard L. Weiner, P. David Adelson, Douglas L. Brockmeyer, Cormac O. Maher, Nalin Gupta, Matthew D. Smyth, Andrew Jea, Jeffrey P. Blount, Jay Riva-Cambrin, Sandi K. Lam, Edward S. Ahn, Gregory W. Albert and Jeffrey R. Leonard

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Deepak Khatri, Jaskaran Singh Gosal, Kuntal Kanti Das, Kamlesh Bhaisora and Arun Kumar Srivastava

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Shawyon Baygani, Kristin Zieles and Andrew Jea

OBJECTIVE

The purpose of this study is to determine if the preoperative Pediatric Quality of Life Inventory (PedsQL) score is predictive of short- and intermediate-term PedsQL outcomes following Chiari decompression surgery. The utility of preoperative patient-reported outcomes (PROs) in predicting pain, opioid consumption, and long-term PROs has been demonstrated in adult spine surgery. To the best of the authors’ knowledge, however, there is currently no widely accepted tool to predict short-, intermediate-, or long-term outcomes after pediatric Chiari decompression surgery.

METHODS

A prospectively maintained database was retrospectively reviewed. Patients who had undergone first-time decompression for symptomatic Chiari malformation were identified and grouped according to their preoperative PedsQL scores: mild disability (score 80–100), moderate disability (score 60–79), and severe disability (score < 60). PedsQL scores at the 6-week, 3-month, and/or 6-month follow-ups were collected. Preoperative PedsQL subgroups were tested for an association with demographic and perioperative characteristics using one-way ANOVA or chi-square analysis. Preoperative PedsQL subgroups were tested for an association with improvements in short- and intermediate-term PedsQL scores using one-way ANOVA and a paired Wilcoxon signed-rank test controlling for statistically different demographic characteristics when appropriate.

RESULTS

A total of 87 patients were included in this analysis. According to their preoperative PedsQL scores, 28% of patients had mild disability, 40% had moderate disability, and 32% had severe disability. There was a significant difference in the prevalence of comorbidities (p = 0.009) and the presenting symptoms of headaches (p = 0.032) and myelopathy (p = 0.047) among the subgroups; however, in terms of other demographic or operative factors, there was no significant difference. Patients with greater preoperative disability demonstrated statistically significantly lower PedsQL scores at all postoperative time points, except in terms of the parent-reported PedsQL at 6 months after surgery (p = 0.195). Patients with severe disability demonstrated statistically significantly greater improvements (compared to preoperative scores) in PedsQL scores at all time points after surgery, except in terms of the 6-week and 6-month PROs and the 6-month parent-reported outcomes (p = 0.068, 0.483, and 0.076, respectively).

CONCLUSIONS

Patients with severe disability, as assessed by the PedsQL, had lower absolute PedsQL scores at all time points after surgery but greater improvement in short- and intermediate-term PROs. The authors conclude that the PedsQL is an efficient and accurate tool that can quickly assess patient disability in the preoperative period and predict both short- and intermediate-term surgical outcomes.

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Jacob Archer, Meena Thatikunta and Andrew Jea

The transoral transpharyngeal approach is the standard approach to resect the odontoid process and decompress the cervicomedullary spinal cord. There are some significant risks associated with this approach, however, including infection, CSF leak, prolonged intubation or tracheostomy, need for nasogastric tube feeding, extended hospitalization, and possible effects of phonation. Other ventral approaches, such as transmandibular and circumglossal, endoscopic transcervical, and endoscopic transnasal, are also viable alternatives but are technically challenging or may still traverse the nasopharyngeal cavity. Far-lateral and posterior extradural approaches to the craniocervical junction require extensive soft-tissue dissection. Recently, a posterior transdural approach was used to resect retro-odontoid cysts in 3 adult patients. The authors present the case of a 12-year-old girl with Down syndrome and significant spinal cord compression due to basilar invagination and a retro-flexed odontoid process. A posterior transdural odontoidectomy prior to occiptocervical fusion was performed. At 12 months after surgery, the authors report satisfactory clinical and radiographic outcomes with this approach.

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Jonathan E. Martin, Brandon G. Rocque, Andrew Jea, Richard C. E. Anderson, Joshua Pahys and Douglas Brockmeyer

OBJECTIVE

Hypermobility of the craniocervical junction (CCJ) in patients with Down syndrome (DS) is common. Whereas atlantoaxial (C1–2) hypermobility is well characterized, occipitoatlantal (Oc–C1) laxity is recognized but poorly defined. A clear understanding of the risks associated with DS-related hypermobility is lacking. Research efforts to address the topic of axial cervical spine instability in the patient with DS require a reliable and reproducible means of assessing CCJ mobility. The authors conducted a pilot study comparing two methods of quantifying motion of the CCJ on dynamic (flexion/extension) plain radiographs: the delta–condyle-axial interval (ΔCAI) and the delta–basion-axial interval (ΔBAI) methods.

METHODS

Dynamic radiographs from a cohort of 10 patients with DS were evaluated according to prescribed standards. Independent movement of Oc–C1, C1–2, and Oc–C2 was calculated. Interrater and intrarater reliability for CCJ mobility was then calculated for both techniques.

RESULTS

Measurement using the ΔCAI technique had excellent fidelity with intraclass correlation coefficients (ICCs) of 0.77, 0.71, and 0.80 for Oc–C1, C1–2, and Oc–C2, respectively. The ΔBAI technique had lower fidelity, yielding respective ICCs of 0.61, 0.65, and 0.50.

CONCLUSIONS

This pilot study suggests that ΔCAI is a superior measurement technique compared to ΔBAI and may provide reliable assessment of the mobility of the CCJ on dynamic radiographs in the pediatric patient with DS. The use of reliable and reproducible measurement techniques strengthens the validity of research derived from pooled database efforts.

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Lourdes C. Eco, Alison Brayton, William E. Whitehead and Andrew Jea

Chordomas are histologically benign tumors with local aggressive behavior. They arise from embryological remnants of the notochord at the clivus, mobile spine, and sacrum. Chordomas are rare tumors in the pediatric age group. Their surgical management is difficult, given their propensity for inaccessible anatomical regions, and proximity to critical neurovascular structures. While en bloc resection with surgical margins has been advocated as the preferred approach for chordomas, tumor characteristics and violation of adjacent anatomical boundaries may not allow for safe en bloc resection of the tumor. Here, the authors present the case of a C1 chordoma in a 5-year-old boy with epidural and prevertebral extension. The patient’s treatment consisted of a far-lateral approach for resection of the tumor and C1 arch, followed by circumferential reconstruction of the craniocervical junction with an expandable cage spanning the skull base to C2, and posterior occipitocervical spinal instrumentation. At 42 months after surgery, the patient remains neurologically intact with stable oncological status, and no evidence of craniocervical junction instrumentation failure.

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Arvind C. Mohan, Howard L. Weiner, Carrie A. Mohila, Adekunle Adesina, Murali Chintagumpala, Daniel Curry, Andrew Jea, Jonathan J. Lee, Sandi K. Lam, William E. Whitehead, Robert Dauser, Daniel Yoshor and Guillermo Aldave

OBJECTIVE

The indication for and timing of surgery for epilepsy associated with low-grade mixed neuronal-glial tumors may be controversial. The purpose of this study was to evaluate the effect of resection and associated variables on epilepsy and on progression-free survival (PFS).

METHODS

A retrospective chart review of patients treated between 1992 and 2016 was conducted to identify individuals with epilepsy and low-grade gliomas or neuronal-glial tumors who underwent resective surgery. Data analyzed included age at epilepsy onset, age at surgery, extent of resection, use of electrocorticography, the number of antiepileptic drugs (AEDs) before and after surgery, the presence of dysplasia, Engel class, histological findings, and PFS. The institutional review board protocol was specifically approved to conduct this study.

RESULTS

A total of 107 patients were identified. The median follow-up was 4.9 years. The most common pathology was dysembryoplastic neuroepithelial tumor (36.4%), followed by ganglioglioma (31.8%). Eighty-four percent of patients had Engel class I outcomes following surgery. Gross-total resection was associated with a higher likelihood of an Engel class I outcome (90%) as compared to subtotal resection (58%) (p = 0.0005). Surgery reduced the AED burden, with 40% of patients requiring no AEDs after surgery (p < 0.0001). Children with neurodevelopmental comorbidities (n = 5) uniformly did not experience seizure improvement following resection (0% vs 83% overall; p < 0.0001). Electrocorticography was used in 33% of cases and did not significantly increase class I outcomes. PFS was 90% at 5 years. Eleven percent of tumors recurred, with subtotal resection more likely to result in recurrence (hazard ratio 5.3, p = 0.02). Histological subtype showed no significant impact on recurrence.

CONCLUSIONS

Gross-total resection was strongly associated with Engel class I outcome and longer PFS. Further studies are needed to elucidate the suitable time for surgery and to identify factors associated with oncological transformation.

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Spinal instrumentation in infants, children, and adolescents: a review

JNSPG 75th Anniversary Invited Review Article

Stephen Mendenhall, Dillon Mobasser, Katherine Relyea and Andrew Jea

OBJECTIVE

The evolution of pediatric spinal instrumentation has progressed in the last 70 years since the popularization of the Harrington rod showing the feasibility of placing spinal instrumentation into the pediatric spine. Although lacking in pediatric-specific spinal instrumentation, when possible, adult instrumentation techniques and tools have been adapted for the pediatric spine. A new generation of pediatric neurosurgeons with interest in complex spine disorder has pushed the field forward, while keeping the special nuances of the growing immature spine in mind. The authors sought to review their own experience with various types of spinal instrumentation in the pediatric spine and document the state of the art for pediatric spine surgery.

METHODS

The authors retrospectively reviewed patients in their practice who underwent complex spine surgery. Patient demographics, operative data, and perioperative complications were recorded. At the same time, the authors surveyed the literature for spinal instrumentation techniques that have been utilized in the pediatric spine. The authors chronicle the past and present of pediatric spinal instrumentation, and speculate about its future.

RESULTS

The medical records of the first 361 patients who underwent 384 procedures involving spinal instrumentation from July 1, 2007, to May 31, 2018, were analyzed. The mean age at surgery was 12 years and 6 months (range 3 months to 21 years and 4 months). The types of spinal instrumentation utilized included occipital screws (94 cases); C1 lateral mass screws (115 cases); C2 pars/translaminar screws (143 cases); subaxial cervical lateral mass screws (95 cases); thoracic and lumbar spine traditional-trajectory and cortical-trajectory pedicle screws (234 cases); thoracic and lumbar sublaminar, subtransverse, and subcostal polyester bands (65 cases); S1 pedicle screws (103 cases); and S2 alar-iliac/iliac screws (56 cases). Complications related to spinal instrumentation included hardware-related skin breakdown (1.8%), infection (1.8%), proximal junctional kyphosis (1.0%), pseudarthroses (1.0%), screw malpositioning (0.5%), CSF leak (0.5%), hardware failure (0.5%), graft migration (0.3%), nerve root injury (0.3%), and vertebral artery injury (0.3%).

CONCLUSIONS

Pediatric neurosurgeons with an interest in complex spine disorders in children should develop a comprehensive armamentarium of safe techniques for placing rigid and nonrigid spinal instrumentation even in the smallest of children, with low complication rates. The authors’ review provides some benchmarks and outcomes for comparison, and furnishes a historical perspective of the past and future of pediatric spine surgery.

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Kyung Shin Kang, Jeff Lastfogel, Laurie L. Ackerman, Andrew Jea, Alexander G. Robling and Sunil S. Tholpady

OBJECTIVE

Cranial defects can result from trauma, infection, congenital malformations, and iatrogenic causes and represent a surgical challenge. The current standard of care is cranioplasty, with either autologous or allogeneic material. In either case, the intrinsic vascularity of the surrounding tissues allows for bone healing. The objective of this study was to determine if mechanotransductive gene manipulation would yield non–weight-bearing bone regeneration in a critical size calvarial defect in mice.

METHODS

A mouse model of Sost deletion in Sost knockout (KO) mice was created in which the osteocytes do not express sclerostin. A critical size calvarial defect (4 mm in diameter) was surgically created in the parietal bone in 8-week-old wild-type (n = 8) and Sost KO (n = 8) male mice. The defects were left undisturbed (no implant or scaffold) to simulate a traumatic calvariectomy model. Eight weeks later, the animals were examined at necropsy by planimetry, histological analysis of new bone growth, and micro-CT scanning of bone thickness.

RESULTS

Defects created in wild-type mice did not fill with bone over the study period of 2 months. Genetic downregulation of sclerostin yielded animals that were able to regenerate 40% of the initial critical size defect area 8 weeks after surgery. A thin layer of bone covered a significant portion of the original defect in all Sost KO animals. A statistically significant increase in bone volume (p < 0.05) was measured in Sost KO mice using radiodensitometric analysis. Immunohistochemical analysis also confirmed that this bone regeneration occurred through the Wnt pathway and originated from the edge of the defect; BMP signaling did not appear to be affected by sclerostin.

CONCLUSIONS

Mechanical loading is an important mechanism of bone formation in the cranial skeleton and is poorly understood. This is partially due to the fact that it is difficult to load bone in the craniomaxillofacial skeleton. This study suggests that modulation of the Wnt pathway, as is able to be done with monoclonal antibodies, is a potentially efficacious method for bone regeneration that requires further study.