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Richard Leblanc

Wilder Penfield (1891–1976) is widely regarded as a dominant figure in 20th century neurobiology for his singular contributions to the structure-function relationship of the brain, his discovery of the language function of the supplementary motor area, the discovery (with Herbert Jasper and Brenda Milner) of the anatomy of recall, and his pioneering work in the surgical treatment of focal epilepsy. But another of his significant discoveries has escaped notice: the recognition that focal microgyria can generate epileptic seizures, and that these can be treated surgically. Penfield discussed the case of the patient through which this discovery was made during his Shattuck Lecture to the Massachusetts Medical Society in 1939. As Penfield gave only a fragmentary account of this case, the patient's chart was retrieved from the Montreal Neurological Institute archives, and his operative note and brain map, intraoperative photographs, and the histopathological and cytological examinations of the resected specimen were reviewed. Based on these primary sources, this paper provides a complete, detailed account of the first case in which microgyria was recognized as a cause of focal epilepsy, which was successfully treated surgically.

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Arnold H. Menezes, Scott C. Seaman, Matthew A. Howard III, Patrick W. Hitchon, and Elizabeth B. Takacs

OBJECTIVE

Tethered cord syndrome (TCS) has been well described in pediatric patients. Many recent reports of TCS in adult patients have grouped retethering patients with newly diagnosed ones without separately analyzing each entity and outcome. The authors reviewed their experience of newly diagnosed adult TCS patients to identify and explore TCS misdiagnosis, recognition, subtype pathology, and individual objective outcomes.

METHODS

This study included 24 adult patients (20 female and 4 male) who fit the criteria of being newly diagnosed and aged 20 years and older (age range 20–77 years). Preexisting dermal sinus was present in 6 patients, hypertrichosis in 5, skin tag/cleft/dimple and fatty subcutaneous masses in 5, scoliosis in 2, and neurological abnormalities in 4 patients. The pathology consisted of TCS with taut filum in 8 patients, conus lipoma with TCS in 7, diastematomyelia in 7, and cervical cord tethering in 2 patients. Of the 24 study patients, nondermatomal low-back or perineal pain occurred in 19 patients, bladder dysfunction in 21, and motor, sensory, and reflex abnormalities in 21 patients. Aggravating factors were repeated stretching, multiple pregnancies, heavy lifting, and repeated bending. Urological evaluation included bladder capacity, emptying, postvoid residuals, detrusor function, pelvic floor electromyography (EMG), bladder sensitivity, and sphincter EMG, which were repeated at 6 months and 1 year postoperatively. The follow-up was 1 to 30 years. Detailed postoperative neurological findings and separate patient outcome evaluations were recorded. Four of the 24 patients did not have an operation.

RESULTS

Resolution of pain occurred in 16 of the 19 patients reporting low-back or perineal pain. Motor and sensory complaints resolved in 17 of 20 patients. Regarding bladder dysfunction, in the 20 patients with available data, bladder function returned to normal in 12 patients, improved in 3 patients, and was unchanged in 5 patients. If the symptom duration was less than 6–8 months, there was recovery of all parameters of pain, bladder dysfunction, and neurological deficit, and recovery from hyperreflexia matched that from neurological deficit. Fifteen patients were employed preoperatively and returned to work, and an additional 3 others who were unable to work preoperatively were able to do so postoperatively.

CONCLUSIONS

Most adults with newly diagnosed TCS have unrecognized neurocutaneous abnormalities and neurological deficits. The triad of nondermatomal sacral or perineal pain, bladder dysfunction, and neurological deficit should not be confused with hip or degenerative lumbosacral disease. Addressing the primary pathology often leads to successful results.

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Peter G. Passias, Haddy Alas, Sara Naessig, Han Jo Kim, Renaud Lafage, Christopher Ames, Eric Klineberg, Katherine Pierce, Waleed Ahmad, Douglas Burton, Bassel Diebo, Shay Bess, D. Kojo Hamilton, Munish Gupta, Paul Park, Breton Line, Christopher I. Shaffrey, Justin S. Smith, Frank Schwab, Virginie Lafage, and the International Spine Study Group

OBJECTIVE

The goal of this study was to assess the conversion rate from baseline cervical alignment to postoperative cervical deformity (CD) and the corresponding proximal junctional kyphosis (PJK) rate in patients undergoing thoracolumbar adult spinal deformity (ASD) surgery.

METHODS

The operative records of patients with ASD with complete radiographic data beginning at baseline up to 3 years were included. Patients with no baseline CD were postoperatively stratified by Ames CD criteria (T1 slope–cervical lordosis mismatch [TS-CL] > 20°, cervical sagittal vertical axis [cSVA] > 40 mm), where CD was defined as fulfilling one or more of the Ames criteria. Severe CD was defined as TS-CL > 30° or cSVA > 60 mm. Follow-up intervals were established after ASD surgery, with 6 weeks postoperatively defined as early; 6 weeks–1 year as intermediate; 1–2 years as late; and 2–3 years as long-term. Descriptive analyses and McNemar tests identified the CD conversion rate, PJK rate (< −10° change in uppermost instrumented vertebra and the superior endplate of the vertebra 2 levels superior to the uppermost instrumented vertebra), and specific alignment parameters that converted.

RESULTS

Two hundred sixty-six patients who underwent ASD surgery (mean age 59.7 years, 77.4% female) met the inclusion criteria; 103 of these converted postoperatively, and the remaining 163 did not meet conversion criteria. Thirty-eight patients converted to CD early, 26 converted at the intermediate time point, 29 converted late, and 10 converted in the long-term. At conversion, the early group had the highest mean TS-CL at 25.4° ± 8.5° and the highest mean cSVA at 33.6 mm—both higher than any other conversion group. The long-term group had the highest mean C2–7 angle at 19.7° and the highest rate of PJK compared to other groups (p = 0.180). The early group had the highest rate of conversion to severe CD, with 9 of 38 patients having severe TS-CL and only 1 patient per group converting to severe cSVA. Seven patients progressed from having only malaligned TS-CL at baseline (with normal cSVA) to CD with both malaligned TS-CL and cSVA by 6 weeks. Conversely, only 2 patients progressed from malaligned cSVA to both malaligned cSVA and TS-CL. By 1 year, the former number increased from 7 to 26 patients, and the latter increased from 2 to 20 patients. The revision rate was highest in the intermediate group at 48.0%, versus the early group at 19.2%, late group at 27.3%, and long-term group at 20% (p = 0.128). A higher pelvic incidence–lumbar lordosis mismatch, lower thoracic kyphosis, and a higher thoracic kyphosis apex immediately postoperatively significantly predicted earlier rather than later conversion (all p < 0.05). Baseline lumbar lordosis, pelvic tilt, and sacral slope were not significant predictors.

CONCLUSIONS

Patients with ASD with normative cervical alignment who converted to CD after thoracolumbar surgery had varying radiographic findings based on timing of conversion. Although the highest number of patients converted within 6 weeks postoperatively, patients who converted in the late or long-term follow-up intervals had higher rates of concurrent PJK and greater radiographic progression.

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Takeshi Imai, Daisuke Sakai, Jordy Schol, Toshihiro Nagai, Akihiko Hiyama, Hiroyuki Katoh, Masato Sato, and Masahiko Watanabe

BACKGROUND

Rubinstein-Taybi syndrome (RTS) is a rare disorder with a range of congenital anomalies. Although 40% to 60% of patients with RTS have scoliotic deformities, few reports discuss the outcomes of correctional surgery and postoperative care. To raise awareness of the clinical features of RTS and surgical considerations, the authors report on the surgical treatment of a pediatric patient with RTS accompanied by scoliosis.

OBSERVATIONS

A 14-year-old girl with RTS presented with low back pain associated with progressive scoliosis. Because of jaw hypoplasia, videolaryngoscopy-mediated intubation was chosen. A single-stage T4–L3 posterior corrective fusion with instrumentation was successfully performed. Physical and imaging findings were analyzed up to 2 years after correction. The main thoracic Cobb angle was corrected from 73° to 12° and maintained for 2 years after surgery. The patient’s low back pain resolved.

LESSONS

Careful consideration of RTS-associated complications and preoperative planning, including the use of videolaryngoscopy-mediated intubation, anesthesia selection, and postoperative care, proved crucial. Scoliosis may appear in many variations in rare diseases such as RTS. Publication of case reports such as this one is needed to provide detailed information about strategies and considerations for correcting scoliotic deformities in patients with RTS.

Open access

Nathaniel R. Ellens and Howard J. Silberstein

BACKGROUND

Coronavirus disease 2019 (COVID-19) is known to cause more severe symptoms in the adult population, but pediatric patients may experience severe neurological symptoms, including encephalopathy, seizures, and meningeal signs. COVID-19 has also been implicated in both ischemic and hemorrhagic cerebrovascular events. This virus inhibits angiotensin-converting enzyme 2, decreasing angiotensin (1–7), decreasing vagal tone, disrupting blood pressure autoregulation, and contributing to a systemic vascular inflammatory response, all of which may further increase the risk of intracranial hemorrhage. However, there has only been one reported case of intracranial hemorrhage developing in a pediatric patient with COVID-19.

OBSERVATIONS

The authors discuss the first case of a pediatric patient with COVID-19 presenting with intracranial hemorrhage. This patient presented with lethargy and a bulging fontanelle and was found to have extensive intracranial hemorrhage with hydrocephalus. Laboratory tests were consistent with hyponatremia and vitamin K deficiency. Despite emergency ventriculostomy placement, the patient died of his disease.

LESSONS

This case demonstrates an association between COVID-19 and intracranial hemorrhage, and the authors have described several different mechanisms by which the virus may potentiate this process. This role of COVID-19 may be particularly important in patients who are already at a higher risk of intracranial hemorrhage, such as those with vitamin K deficiency.

Open access

Ketevan Mikeladze, Anton Konovalov, Andrey Bykanov, Evgeniy Vinogradov, and Sergey Yakovlev

BACKGROUND

The authors report on four clinical cases with intraarterial verapamil administration to resolve vasospasm in patients who underwent surgery for intracranial tumors. Iatrogenic subarachnoid hemorrhage after tumor resection and subsequent vasospasm (an increase in the systolic linear velocity of blood flow through the M1 segment of the middle cerebral artery of more than 250 cm/sec; Lindegaard index: 4.1) were observed in four patients during the early postoperative period after the removal of intracerebral tumors. Each vasospasm case was confirmed by angiography data, was clinically significant, and manifested as the development of a neurological deficit.

OBSERVATIONS

Resolution of vasospasm with the intraarterial administration of verapamil was achieved in all four cases as confirmed by angiographic data in all four cases and complete regression of neurological symptoms in two cases. In all four presented cases, vasospasm was resolved; unfortunately, the resolution did not always lead to significant clinical improvement. However, lethal outcomes were avoided in two cases, and almost full recoveries were achieved in the other two.

LESSONS

The authors believe that the removal of intracranial tumors can cause expected and potential complications, such as cerebral vasospasm, which must be diagnosed and treated in a timely manner.

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Cory McFall, Alexandra D. Beier, Kelsey Hayward, Emily C. Alberto, Randall S. Burd, Bethany J. Farr, David P. Mooney, Kristin Gee, Jeffrey S. Upperman, Mauricio A. Escobar Jr., Nicole G. Coufal, Helen A. Harvey, and Gerald Gollin

OBJECTIVE

The authors sought to evaluate the contemporary management of pediatric open skull fractures and assess the impact of variations in antibiotic and operative management on the incidence of infectious complications.

METHODS

The records of children who presented from 2009 to 2017 to 6 pediatric trauma centers with an open calvarial skull fracture were reviewed. Data collected included mechanism and anatomical site of injury; presence and depth of fracture depression; antibiotic choice, route, and duration; operative management; and infectious complications.

RESULTS

Of the fractures among the 138 patients included in the study, 48.6% were frontal and 80.4% were depressed; 58.7% of patients underwent fragment elevation. The average duration of intravenous antibiotics was 4.6 (range 0–21) days. Only 53 patients (38.4%) received a single intravenous antibiotic for fewer than 4 days. and 56 (40.6%) received oral antibiotics for an average of 7.3 (range 1–20) days. Wounds were managed exclusively in the emergency department in 28.3% of patients. Two children had infectious complications, including a late-presenting hardware infection and a superficial wound infection. There were no cases of meningitis or intracranial abscess. Neither antibiotic spectrum or duration nor bedside irrigation was associated with the development of infection.

CONCLUSIONS

The incidence of infectious complications in this population of children with open skull fractures was low and was not associated with the antibiotic strategy or site of wound care. Most minimally contaminated open skull fractures are probably best managed with a short duration of a single antibiotic, and emergency department closure is appropriate unless there is significant contamination or fragment elevation is necessary.

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Corey T. Walker, Jakub Godzik, Santiago Angel, Juan Pedro Giraldo, Jay D. Turner, and Juan S. Uribe

OBJECTIVE

Coronal malalignment (CM) in adult spinal deformity is associated with poor outcomes and remains underappreciated in the literature. Recent attempts at classifying CM indicate that some coronal shifts may be more difficult to treat than others. To date, outcomes for circumferential minimally invasive surgery (cMIS) of the spine in the context of these new CM classifications are unreported.

METHODS

A retrospective evaluation of patients with degenerative scoliosis (Cobb angle > 20°) consecutively treated with cMIS at a single institution was performed. Preoperative and 1-year postoperative standing radiographs were used to make the comparisons. Clinical outcome measures were compared. Patients were subgrouped according to the preoperative distance between their C7 plumb line and central sacral vertical line (C7-CSVL) as either coronally aligned (type A, C7-CSVL < 3 cm); shifted ≥ 3 cm toward the concavity (type B); or shifted ≥ 3 cm toward the convexity (type C) of the main lumbar curve.

RESULTS

Forty-two patients were included (mean age 67.7 years). Twenty-six patients (62%) were classified as type A, 5 patients (12%) as type B, and 11 patients (26%) as type C. An average of 4.9 segments were treated. No type A patients developed postoperative CM. All type B patients had CM correction. Six of the 11 type C patients had CM after surgery. Overall, there was an improvement in the C7-CSVL (from 2.4 to 1.8 cm, p = 0.04). Among subgroups, only type B patients improved (from 4.5 to 0.8 cm, p = 0.002); no difference was seen for type A patients (from 1.2 to 1.4 cm, p = 0.32) or type C patients (from 4.3 to 3.1 cm, p = 0.11). Comparing type C patients with postoperative CM versus those without postoperative CM, patients with CM had worse visual analog scale back scores at 1 year (5 vs 1, p = 0.01). Moreover, they had higher postoperative L4 tilt angles (11° vs 5°, p = 0.01), indicating inadequate correction of the lumbosacral fractional curve.

CONCLUSIONS

cMIS improved coronal alignment, curve magnitudes, and clinical outcomes among patients with degenerative scoliosis. It did not result in CM in type A patients and was successful at improving the C7-CSVL in type B patients. Type C patients remain the most difficult to treat coronally, with worse visual analog scale back pain scores in those with postoperative CM. Regional coronal restoration of the lumbosacral fracture curve should be the focus of correction in cMIS for these patients.

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Arthur Hosmann, Carmen Angelmayr, Andreas Hopf, Steffen Rauscher, Jonas Brugger, Lavinia Ritscher, Isabelle Bohl, Philipp Schnackenburg, Adrian Engel, Walter Plöchl, Markus Zeitlinger, Andrea Reinprecht, Karl Rössler, and Andreas Gruber

OBJECTIVE

Intrahospital transport for CT scans is routinely performed for neurosurgical patients. Particularly in the sedated and mechanically ventilated patient, intracranial hypertension and blood pressure fluctuations that might impair cerebral perfusion are frequently observed during these interventions. This study quantifies the impact of intrahospital patient transport on multimodality monitoring measurements, with a particular focus on cerebral metabolism.

METHODS

Forty intrahospital transports in 20 consecutive patients suffering severe aneurysmal subarachnoid hemorrhage (SAH) under continuous intracranial pressure (ICP), brain tissue oxygen tension (pbtO2), and cerebral microdialysis monitoring were prospectively included. Changes in multimodality neuromonitoring data during intrahospital transport to the CT scanner and the subsequent 10 hours were evaluated using linear mixed models. Furthermore, the impact of risk factors at transportation, such as cerebral vasospasm, cerebral hypoxia (pbtO2 < 15 mm Hg), metabolic crisis (lactate-pyruvate ratio [LPR] > 40), and transport duration on cerebral metabolism, was analyzed.

RESULTS

During the transport, the mean ICP significantly increased from 7.1 ± 3.9 mm Hg to 13.5 ± 6.0 mm Hg (p < 0.001). The ICP exceeded 20 mm Hg in 92.5% of patients; pbtO2 showed a parallel rise from 23.1 ± 13.3 mm Hg to 28.5 ± 23.6 mm Hg (p = 0.02) due to an increase in the fraction of inspired oxygen during the transport. Both ICP and pbtO2 returned to baseline values thereafter. Cerebral glycerol significantly increased from 71.0 ± 54.9 µmol/L to 75.3 ± 56.0 µmol/L during the transport (p = 0.01) and remained elevated for the following 9 hours. In contrast, cerebral pyruvate and lactate levels were stable during the transport but showed a significant secondary increase 1–8 hours and 2–9 hours, respectively, thereafter (p < 0.05). However, the LPR remained stable over the entire observation period. Patients with extended transport duration (more than 25 minutes) were found to have significantly higher levels of cerebral pyruvate and lactate as well as lower glutamate concentrations in the posttransport period.

CONCLUSIONS

Intrahospital transport and horizontal positioning during CT scans induce immediate intracranial hypertension and an increase in cerebral glycerol, suggesting neuronal injury. Afterward, sustained impairment of neuronal metabolism for several hours could be observed, which might increase the risk of secondary ischemic events. Therefore, intrahospital transport for neuroradiological imaging should be strongly reconsidered and only indicated if the expected benefit of imaging results outweighs the risks of transportation.

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Takaya Yasuda, Yoshitaka Kurosaki, Ryota Ishibashi, Kensuke Takada, and Masaki Chin

Trigeminal meningocele is a rare disease that results in rhinorrhea. Treatments with endoscopic approaches and open craniotomies have high recurrence rates, and controversy regarding the most effective surgical strategy for trigeminal meningocele is ongoing.

The authors report a case of a 13-year-old female patient with a diagnosis of trigeminal meningocele determined after she presented with a history of intermittent headaches, suspected rhinorrhea, and recurrent meningitis. In addition to the conventional method of covering the efflux point of CSF and filling the inside of the meningocele with fascial tissues, the authors selectively closed the influx point of CSF from the prepontine cistern to the meningocele using an anterior transpetrosal approach. On the basis of the preoperative images, the authors hypothesized that the influx point of CSF could not be observed under the microscopic direct view and instead used a flexible endoscope. A check valve–like structure with one-way communication of CSF from the prepontine cistern into the cystic cavity was identified and was closed.

At the time of this report, 36 months postoperatively, the patient had no indications of recurrence. Although cases of trigeminal meningoceles are infrequently encountered and require a tailored approach, the results in this case thus far indicate that the use of an endoscope and open craniotomy is an effective strategy for surgical treatment.