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Management of severe cervical kyphosis in a patient with Larsen syndrome

Case report

Marci Madera, Alvin Crawford, and Francesco T. Mangano

✓ Larsen syndrome is a rare genetic disorder of the connective tissue that is characterized by multiple joint dislocations, distinctive deformities of the hands and feet, characteristic facial features, kyphoscoliosis, and segmentation anomalies of the vertebrae. Diverse treatment options, including conservative observation and surgical correction, have been reported for patients who present with cervical spine pathophysiology. Differences in surgical approaches, timing of the correction, and pre- or postoperative bracing have been reported. According to the authors, the present case is the first report of a pediatric patient with Larsen syndrome in whom an asymptomatic cervical instability was treated before neurological deterioration with synchronous anterior decompression and fixation, posterior fusion and fixation, and halo placement. A review of the literature on similar patients reveals the variety of practices associated with a diagnosis of Larsen syndrome.

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All-terrain vehicle–related injury

James T. Rutka

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Standardizing preoperative preparation to reduce surgical site infections among pediatric neurosurgical patients

Joshua K. Schaffzin, Katherine Simon, Beverly L. Connelly, and Francesco T. Mangano

OBJECTIVE

Surgical site infections (SSIs) are costly to patients and the health care system. Pediatric neurosurgery SSI risk factors are not well defined. Intraoperative protocols have reduced, but have not eliminated, SSIs. The effect of preoperative intervention is unknown. Using quality improvement methods, a preoperative SSI prevention protocol for pediatric neurosurgical patients was implemented to assess its effect on SSI rate.

METHODS

Patients who underwent a scheduled neurosurgical procedure between January 2014 and December 2015 were included. Published evidence and provider consensus were used to guide preoperative protocol development. The Model for Improvement was used to test interventions. Intraoperative and postoperative management was not standardized or modified systematically. Staff, family, and overall adherence was measured as all-or-nothing. In addition, SSI rates among eligible procedures were measured before and after protocol implementation.

RESULTS

Within 4 months, overall protocol adherence increased from 51.3% to a sustained 85.7%. SSI rates decreased from 2.9 per 100 procedures preintervention to 0.62 infections postintervention (p = 0.003). An approximate 79% reduction in SSI risk was identified (risk ratio 0.21, 95% CI 0.08–0.56; p = 0.001).

CONCLUSIONS

Clinical staff and families successfully collaborated on a standardized preoperative protocol for pediatric neurosurgical patients. Standardization of the preoperative phase of care alone reduced SSI rates. Attention to the preoperative in addition to the intraoperative and postoperative phases of care may lead to further reduction in SSI rates.

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Mesial temporal lobe morphology in intractable pediatric epilepsy: so-called hippocampal malrotation, associated findings, and relevance to presurgical assessment

James L. Leach, Reem Awwad, Hansel M. Greiner, Jennifer J. Vannest, Lili Miles, and Francesco T. Mangano

OBJECTIVE

Diagnostic criteria for hippocampal malrotation (HIMAL) on brain MRI typically include a rounded hippocampus, vertical collateral sulcus, and architectural blurring. Relationship to epileptogenesis remains speculative, and usefulness for surgical guidance is unknown. The study was performed to determine the prevalence of hippocampal rotational anomalies in a cohort of pediatric patients with intractable epilepsy undergoing evaluation for surgery and to determine the significance of this finding in the context of surgical planning.

METHODS

Forty-eight surgically treated children with intractable epilepsy were compared with matched healthy subjects; reviewers were blinded to surgical side. Each temporal lobe was evaluated for rounded hippocampus, blurring, vertical collateral sulcus, wide choroidal fissure, enlarged temporal horn, low fornix, hippocampal signal, and findings of hippocampal sclerosis. A mesial temporal lobe (MTL) score was calculated by summing the number of features, and the collateral sulcus angle (CSA) was measured in each temporal lobe. Surgical side, pathological diagnosis, and imaging findings elsewhere in the brain were tabulated. Presence of HIMAL, associated imaging features, and MTL score were compared between sides, between epilepsy and control groups, in relationship to side of surgery, and in relationship to postoperative outcome.

RESULTS

Only 3 epilepsy patients (6.2%) and no controls exhibited all 3 features of HIMAL (p = 0.12). Eight of 48 (16.7%) epilepsy versus 2 of 48 (4.6%) control subjects had both a rounded hippocampus and vertical collateral sulcus (suggesting HIMAL) (p = 0.045). In control and epilepsy subjects, most findings were more prevalent on the left, and the left CSA was more vertical (p < 0.0001). Epilepsy subjects had higher MTL scores (z = −2.95, p = 0.002) and more acute CSAs (p = 0.04) than controls. Only lateralizing raw MTL score had a significant association with surgical side (p = 0.03, OR 7.33); however, this was not significant when hippocampal sclerosis cases were excluded. HIMAL findings were more prevalent and MTL scores were higher in patients with resections involving the temporal lobes. On group analysis, HIMAL findings did not predict eventual surgical side and did not predict outcome, although the numbers are small. In 4 patients the abnormally rotated hippocampus was resected and showed hippocampal sclerosis and/or dysplastic changes on histopathology. All of these patients had a good outcome after surgery.

CONCLUSIONS

While increased in prevalence in children with intractable epilepsy, imaging findings of HIMAL did not have preoperative lateralizing utility in this group. Findings of HIMAL (including round hippocampus, architectural blurring, and vertical collateral sulcus) did not predict outcome after surgery, although the small number of patients with these findings limits evaluation. In the small number of patients in which the malrotated hippocampus was removed, outcome was good. Further research is needed to continue to define this association in children with intractable epilepsy, focusing on a temporal lobe cohort.

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Pediatric neurosurgical injuries associated with all-terrain vehicle accidents: a 10-year experience at St. Louis Children’s Hospital

Francesco T. Mangano, Jose A. Menendez, Matthew D. Smyth, Jeffrey R. Leonard, Prithvi Narayan, and Tae Sung Park

Object

All-terrain vehicles (ATVs) have been characterized as inherently unstable and are associated with significant pediatric injuries in the US. The authors performed a study to analyze data obtained in pediatric patients who had sustained neurological injuries in ATV-related accidents, identify potential risk factors, and propose preventive measures. The study is based on a 10-year experience at the St. Louis Children’s Hospital.

Methods

The authors retrospectively analyzed data obtained in all patients admitted to the St. Louis Children’s Hospital between 1993 and 2003, limiting their focus to pediatric cases involving ATV-related accidents. A total of 185 patients were admitted with these criteria. Sixty-two patients (33.5%) suffered neurological injuries; there were 42 male and 20 female patients whose age ranged from 2 to 17 years. The most common injuries included skull fracture (37 cases) and closed head injury (30 cases). There were 39 cases of intracranial hemorrhage and 11 of spinal fracture. A total of 15 types of neurosurgical procedure were performed: six craniotomies for hematoma drainage, five craniotomies for elevation of depressed fractures, two procedures to allow placement of an intracranial pressure monitor, one to allow placement of an external ventricular drain, and one to allow the insertion of a ventriculoperitoneal shunt. Two patients had sustained spinal cord injury, and three procedures were performed for spinal decompression or stabilization. The duration of hospital stay ranged from 1 to 143 days (mean 6.6 days). Fifty-seven patients (30.8%) were eventually discharged from the hospital, three (1.6%) were transferred to another hospital, two (1.1%) died, and 123 (66.4%) required in-patient rehabilitation.

Conclusions

Children suffered significant injuries due to ATV accidents. In passengers there was a statistically significant increased risk of neurological injury. The relative risk of neurological injury in patients not wearing helmets was higher than that in those who wore helmets, but the difference did not reach statistical significance. Further efforts must be made to improve the proper operation and safety of ATVs, both through the education of parents and children and through the creation of legislation requiring stricter laws concerning ATV use.

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Frameless Pinless Stereotaxy

Francesco T. Mangano and Matthew D. Smyth

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Early programmable valve malfunctions in pediatric hydrocephalus

Francesco T. Mangano, Jose A. Menendez, Tracy Habrock, Prithvi Narayan, Jeffrey R. Leonard, Tae Sung Park, and Matthew D. Smyth

Object

The use of adjustable differential pressure valves has been recommended to improve ventriculoperitoneal (VP) shunt performance in selected patients; however, published data are scarce regarding their clinical reliability. Recently, the identification of a number of malfunctioning programmable valves during shunt revision surgery in children prompted a retrospective review of valve performance in this patient cohort.

Methods

The authors performed a retrospective chart analysis of 100 patients with programmable valve shunts and 89 patients with nonprogrammable valve shunts implanted at the St. Louis Children's Hospital between April 2002 and June 2004. They noted the cause of hydrocephalus, the type of shunt malfunction, and cerebrospinal fluid (CSF) protein levels. Regular clinical follow up ranged from 1 to 26 months, with a mean follow-up time of 9.75 months for patients with programmable valves and 10.4 months for patients with nonprogrammable valves.

Patient ages ranged from 2 weeks to 18 years. One hundred patients had 117 programmable valves implanted, and 35 of these patients (35%) underwent shunt revision because of malfunction. The programmable valve itself malfunctioned in nine patients who had undergone shunt revision (11.1%/year of follow up). The nonprogrammable valve group had no valve malfunctions. The overall VP shunt revision rate in the nonprogrammable valve group was 20.2%. No significant differences were identified when CSF protein levels and specific malfunction types were compared within the programmable valve and nonprogrammable valve groups.

Conclusions

In this study the authors demonstrated an annualized intrinsic programmable valve malfunction rate of 11.1%, whereas during the same period no intrinsic valve malfunctions were noted with nonprogrammable valve systems for similar causes of hydrocephalus. The CSF protein levels did not correlate with observed valve malfunction rates. Further evaluation in a prospective, randomized fashion will elucidate specific indications for programmable valve systems and better determine the reliability of these valves in the pediatric population.

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Characterization of abnormal diffusion properties of supratentorial brain tumors: a preliminary diffusion tensor imaging study

Weihong Yuan, Scott K. Holland, Blaise V. Jones, Kerry Crone, and Francesco T. Mangano

Object

Diffusion tensor (DT) imaging was used in children with supratentorial tumors to evaluate the anisotropic diffusion properties between different tumor grades and between tumors and adjacent and contralateral white matter.

Methods

In this retrospective review, the authors review the cases of 16 children (age range 1–18 years) who presented to their institution with supratentorial tumors and were treated between 2004 and 2007. Eleven patients had low-grade and 5 had high-grade tumors. Fractional anisotropy (FA), mean diffusivity, and axial (λ) and radial (λ) eigenvalues within selected regions were studied. Mitotic index, necrosis, and vascularity of the tumors were compared with DT imaging parameters.

Results

The mean diffusivity was significantly higher in low-grade than in high-grade tumors (p = 0.04); the 2 tumor grades also significantly differed for both λ (p < 0.05) and λ (p < 0.05). Mean diffusivity values in low-grade tumors were significantly higher than in adjacent normal-appearing white matter (NAWM; p = 0.0004) and contralateral NAWM (p = 0.0001). In both low- and high-grade tumors, the FA was significantly lower than in NAWM (p < 0.0001 and p < 0.03, respectively) and contralateral NAWM (p < 0.0001 and p < 0.003, respectively). Tumor cellularity highly correlated with mean diffusivity and λand λ.

Conclusions

Diffusion tensor imaging is a useful tool in the evaluation of supratentorial tumors in children. The mean diffusivity appears to be a significant marker in differentiating tumors grades. Findings related to λ and λ within tumor groups and between tumors and NAWM may be an indirect manifestation of the combined effects of axonal injury, demyelination, and tumor mass within the cranial compartment.

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Utility of the material community deprivation index as a metric to identify at-risk children for severe traumatic brain injury

Amber L. Gaulden, Stephen Trinidad, Suzanne Moody, Meera Kotagal, Francesco T. Mangano, and Smruti K. Patel

OBJECTIVE

Pediatric traumatic brain injury (TBI) is a significant cause of morbidity and mortality with lasting effects including neurological deficits and psychological comorbidities. Recent studies have shown that social determinants of health are key factors that impact clinical outcomes in other pediatric traumatic injuries, suggesting that these health disparities may have a significant impact on patients sustaining TBI as well. The purpose of this study was to retrospectively review a cohort of pediatric patients diagnosed with TBI and elucidate the relationships among socioeconomic deprivation, patient-specific demographics, and morbidity and mortality.

METHODS

The authors conducted a retrospective cross-sectional analysis of pediatric patients (≤ 18 years of age) treated for TBI at a level I pediatric trauma center between 2016 and 2020. Patients with concussion-related injuries without intracranial findings and those with nonaccidental trauma were excluded from the study. In addition to evaluating basic patient demographics, the authors geocoded patient addresses to allow identification of the patient’s home census tract using the material community deprivation index (MCDI). The MCDI is a unique composite index score created by the combination of six census variables and ranges from 0 to 1 in severity.

RESULTS

Of the 513 patients included in this study, 71 (13.8%) were diagnosed with severe TBI, 28 (5.5%) with moderate TBI, and 414 (80.7%) with mild TBI. Patients in quartile 4 (MCDI ≥ 0.45) were at a significantly higher risk of having a severe TBI than patients in quartile 1 (OR 2.29, 95% CI 1.1–4.71; p = 0.02). Black patients were more likely to have a firearm-related TBI (OR 3.74, 95% CI 2.01–8.7; p = 0.018) than non-Black patients. Patients who lived in a neighborhood with a lower MCDI were significantly more likely to be discharged home than those who lived in an area with a higher MCDI (OR 2.78, 95% CI 7.90–32.93; p < 0.001).

CONCLUSIONS

This study demonstrated that inequities continue to exist within the pediatric TBI population and that the MCDI is a valuable tool to identify at-risk subpopulations. More specifically, patients who lived in a neighborhood with a higher MCDI were at higher risk of sustaining a severe TBI. By partnering with communities, families, and policymakers, healthcare providers could serve as advocates for these patients and work to minimize the social disparities that continue to exist.

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Tethered cord syndrome: a review of the literature from embryology to adult presentation

Dean A. Hertzler II, John J. DePowell, Charles B. Stevenson, and Francesco T. Mangano

Tethered cord syndrome (TCS) is a clinical condition of various origins that arises from tension on the spinal cord. Radiographic findings may include the conus medullaris in a lower than normal position, fatty infiltration of the filum terminale, lipomyelomeningocele, myelomeningocele, myelocystocele, meningocele, split cord malformations, dermal sinus, anorectal malformations, and intraspinal tumors. The clinical constellation of signs and symptoms associated with TCS may include dermatologic, urological, gastrointestinal, neurological, and orthopedic findings. The current review focuses on TCS by age group of the more common causes of the condition, including myelomeningocele, lipomyelomeningocele, as well as the adult presentation of occult TCS. Pertinent review of the neuroembryology and normal anatomical position of the conus medullaris is included.