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Safety of neuroangiography and embolization in children: complication analysis of 697 consecutive procedures in 394 patients

Ning Lin, Edward R. Smith, R. Michael Scott, and Darren B. Orbach

OBJECT

The safe treatment of children using catheter-based angiography and embolization poses unique challenges because of the technical factors regarding the size and fragility of access and target vessels, as well as unique pediatric cerebrovascular pathologies. The complication rates for neurointerventional procedures in children have not been established.

METHODS

The records of a consecutive cohort of pediatric patients who underwent neuroangiography and/or embolization between 2007 and 2013 were reviewed retrospectively to identify both intraprocedural and postprocedural complications. Demographic and clinical risk factors were analyzed with a multivariate logistic regression model.

RESULTS

The 697 consecutive procedures consisted of 429 diagnostic angiograms and 268 embolizations (mean age of patients 11.1 years; range 4 days to 18 years; 217 females). There were 130 intracranial, 122 extracranial, and 16 spinal embolizations. Pathologies included 28 intracranial arteriovenous malformations (AVMs), 12 spinal AVMs, 19 aneurysms, 29 vein of Galen malformations, 29 dural arteriovenous fistulas, 96 extracranial AVMs, 39 tumors, 3 strokes, and 13 others. Overall, 2 intraprocedural and 1 postprocedural complication (0.7%) occurred in the diagnostic group, all of which were nonneurological events. In the embolization group, 7 intraprocedural and 11 postprocedural complications (6.7%) were observed. Of these complications, 15 were nonneurological events (5.6%), 1 was a short-term neurological event (0.4%), and 2 were long-term neurological events (0.7%).

CONCLUSIONS

Neither the technical challenges posed by children’s access and target vessels nor the unique neuro-vascular pathologies seen in children need result in an elevated morbidity rate related to neuroangiography and embolization. At a dedicated high-volume center, the complication rates may be lower than those for comparable procedures performed in adults.

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Foramen magnum stenosis from overgrowth of the opisthion in a child with achondroplasia

Case report

Ruchira M. Jha, Paul Klimo Jr., and Edward R. Smith

Achondroplasia has a known association with foramen magnum stenosis that can result in cervicomedullary compression, which is most often due to a hypertrophied posterior occipital rim and an undersized transverse diameter. The authors present a unique case of a child with achondroplasia with symptomatic craniocervical compression from marked overgrowth of his opisthion anterior to the posterior arch of the atlas. This 22-month-old child with achondroplasia presented with severe respiratory and motor disabilities, including progressive quadriparesis and apneic episodes requiring continuous positive airway pressure. Magnetic resonance imaging and CT scans revealed marked foramen magnum stenosis from overgrowth of the opisthion, a hypoplastic C-1 ring, and spinal cord edema at the cervicomedullary junction. Foramen magnum decompression and a C-1 laminectomy were performed. Postoperatively, steady motor improvement has been observed and the patient no longer requires ventilatory support. To the authors' knowledge, this is the first report of this unusual anatomical entity.

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Preoperative transdural collateral vessels in moyamoya as radiographic biomarkers of disease

Armide Storey, R. Michael Scott, Richard Robertson, and Edward Smith

OBJECTIVE

The prevalence of angiographically evident preoperative transdural collateral vessels in moyamoya is not well documented. The authors hypothesized that transdural collaterals could be used as radiographic biomarkers of disease, and that their presence is associated with more advanced moyamoya arteriopathy at diagnosis, which is a harbinger of more frequent operative complications and a predictor of better long-term angiographic results following surgery.

METHODS

The study consists of a single-institution case series of patients with moyamoya who underwent pial synangiosis between 2005 and 2013.

RESULTS

Moyamoya was diagnosed in a total of 204 patients (n = 121 [59%] female, 83 [41%] male); the average age at surgery was 9.5 years (range 0.4–35 years). Radiographically, 154 (75%) had bilateral disease for a total of 308 affected hemispheres; 152 (75%) had radiographic stroke; and 190 (93%) had “ivy sign” on FLAIR MRI, indicating slow flow. Of the 358 hemispheres, 324 were treated operatively. On preoperative angiography, 107 patients (52%) had transdural collaterals in 176 affected hemispheres (49%). The Suzuki stage was higher in patients with collaterals (3.4 vs 3.0, p = 0.002). Of 324 treated hemispheres, 84 (26%) had collaterals within the surgical field. Complications included 12 strokes (3.7% stroke rate/hemisphere), with 5 (42%) directly attributable to interruption of transdural collaterals. On 1-year postoperative arteriograms available in 222 hemispheres, Matsushima grades trended better in patients with preoperative collaterals (1.5 vs 1.8 [A = 1, B = 2, C = 3]; p < 0.003).

CONCLUSIONS

Transdural collaterals are present in nearly half of all preoperative arteriograms in patients with moyamoya. These collaterals are more common in advanced disease, are associated with stroke as a perioperative complication, and may suggest increased capacity to produce surgical collaterals postoperatively. These data support the utility of preoperative arteriography.

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Moyamoya disease with mesial temporal sclerosis

Case report

Subash Lohani, Joseph R. Madsen, Ann M. Bergin, and Edward R. Smith

The combination of moyamoya syndrome and symptomatic mesial temporal sclerosis (MTS) has not previously been reported. The authors present the case of a 5-year-old boy with symptomatic MTS who developed progressive moyamoya syndrome. This combination of progressive moyamoya and a structural seizure focus presented a unique clinical problem, with the natural history of MTS predicting a high likelihood of needing resection in the future, which could be challenging following any type of moyamoya-related revascularization surgery. In anticipation of this problem, the patient underwent resection of the right inferior and mesial temporal lobe followed by right pial synangiosis as a 1-day combined operation. Postoperatively he recovered well without any neurological deficits and had an uneventful hospital stay. This case of moyamoya is unique in its association with MTS, and for the simultaneous operations for pial synangiosis and temporal lobectomy, highlighting the importance of surgical planning in patients with dual pathological processes.

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Cavernous malformations of the basal ganglia in children

Clinical article

Bradley A. Gross, Edward R. Smith, and R. Michael Scott

Object

Cavernous malformations (CMs) of the basal ganglia are relatively rare lesions that can lead to considerable neurological impairment because of their eloquent location. The authors reviewed the clinical course and surgical outcome of a series of children with basal ganglia CMs.

Methods

The authors retrospectively reviewed the operative experience of the senior author (R.M.S.) and the 1997–2011 database of Boston Children's Hospital for children with CM of the basal ganglia (which includes CM of the caudate and/or lentiform nucleus and excludes CM of the thalamus). They evaluated baseline demographics, presenting signs, operative outcomes, and condition at long-term follow-up visits and compared these characteristics among patients who underwent surgery and those who were observed.

Results

Of 180 children with a diagnosis of CM, 11 (6%) had CM of the basal ganglia. The mean age at diagnosis was 9.3 years, and the male/female ratio was 1.8:1. Presenting signs were as follows: hemorrhage (8 children), incidental lesions (2), and seizures (1); 2 children had choreiform movement disorders. Treatment was observation or surgery. Observation was chosen for 5 children either because the lesions were asymptomatic (2 children) or because the risk for neurological dysfunction after attempted excision was believed to be high (3 children). These 5 children were observed over a combined total of 30.4 patient-years; none experienced neurological deterioration or symptomatic hemorrhage from their lesions. The other 6 children underwent microsurgical resection of the lesion because they were symptomatic from hemorrhage or increasing mass effect. All 6 of these children had hemorrhagic lesions, of which the smallest dimension was at least 1.5 cm. Of these 6 lesions, 5 were excised completely, and over a combined total of 46 patient-years of follow-up, no rebleeding or late neurological deterioration after surgery was reported.

Conclusions

In this patient population, the natural history of small and asymptomatic CMs of the basal ganglia was benign. The children with large (> 1.5 cm) symptomatic lesions underwent excision; neurological impairment was apparently minimal, and no hemorrhage or neurological deterioration occurred later.

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In-hospital mortality rates after ventriculoperitoneal shunt procedures in the United States, 1998 to 2000: relation to hospital and surgeon volume of care

Edward R. Smith, William E. Butler, and Fred G. Barker II

Object. Death after ventriculoperitoneal (VP) shunt surgery is uncommon, and therefore it has been difficult to study. The authors used a population-based national hospital discharge database to examine the relationship between annual hospital and surgeon volume of VP shunt surgery in pediatric patients and in-hospital mortality rates.

Methods. All children in the Nationwide Inpatient Sample (1998–2000, age 90 days—18 years) who underwent VP shunt placement or shunt revision as the principal procedure were included. Main outcome measures were in-hospital mortality rates, length of stay (LOS), and total hospital charges.

Overall, 5955 admissions were analyzed (253 hospitals, 411 surgeons). Mortality rates were lower at high-volume centers and for high-volume surgeons. In terms of hospital volume, the mortality rate was 0.8% at lowest-quartile-volume centers (< 28 admissions/year) and 0.3% at highest-quartile-volume centers (> 121 admissions/year). In terms of surgeon volume, the mortality rate was 0.8% for lowest-quartile-volume providers (< nine admissions/year) and 0.1% for highest-quartile-volume providers (> 65 admissions/year). After multivariate adjustment for demographic variables, emergency admission and presence of infection, hospital volume of care remained a significant predictor of death (odds ratio [OR] for a 10-fold increase in caseload 0.38; 95% confidence interval [CI] 0.18–0.81). Surgeon volume of care was statistically significant in a similar multivariate model (OR for a 10-fold increase in caseload 0.3; 95% CI 0.13–0.69). Length of stay was slightly shorter and total hospital charges were slightly higher at higher-volume centers, but the differences were not statistically significant.

Conclusions. Pediatric shunt procedures performed at high-volume hospitals or by high-volume surgeons were associated with lower in-hospital mortality rates, with no significant difference in LOS or hospital charges.

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Is there a “July phenomenon” in pediatric neurosurgery at teaching hospitals?

Edward R. Smith, William E. Butler, and Fred G. Barker II

Object

Concern for patient safety, among other reasons, recently prompted sweeping changes in resident work policies in the US. Some have speculated that the arrival of new interns and residents at teaching hospitals each July might cause an annual transient increase in poor patient outcomes and inefficient care.

Methods

Data were analyzed for 4323 craniotomies for tumor resection and 22,072 shunt operations performed in pediatric patients between 1988 and 2000 in US nonfederal hospitals (Nationwide Inpatient Sample, Healthcare Cost and Utilization Project, Agency for Healthcare Research and Quality, Rockville, MD). In-hospital mortality rates, discharge outcome, complications, and efficiency measures (length of stay [LOS] and hospital charges) for patients treated in July and August were compared with similar data for patients in other months.

There were no significant increases in any adverse end point for either tumor or shunt operations in July and August. Odds ratios (95% confidence interval [CI]) for outcome of tumor craniotomies performed in July and August compared with outcome for tumor craniotomies performed in other months were as follows: for mortality rate, 0.43 (0.14–1.32); for adverse discharge disposition, 1.03 (0.71–1.51); for neurological complications, 1.00 (0.63–1.59); for transfusion, 0.70 (0.41–1.19). Hospital charges were 0.5% lower (range −6 to 5%) in July and August, and LOS was 3% shorter (range −8 to 3%). Odds ratios (95% CI) for July or August shunt surgery compared with shunt surgery performed in other months were as follows: for mortality rate, 0.96 (0.58–1.60); for adverse discharge disposition, 0.85 (0.66–1.11); for neurological complications, 1.27 (0.75–2.16); for transfusion, 0.81 (0.48–1.37). Hospital charges were 0.2% higher in July and August (range −3 to 3%), and LOS was 3% shorter (range −5 to 0.5%).

Conclusions

Although moderate increases in some adverse end points could not be excluded, there was no evidence that brain tumor or shunt surgery performed in pediatric patients at US teaching hospitals during July and August is associated with more frequent adverse patient outcome or inefficient care than similar surgery performed during other months.

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Introduction. Translational research advances in the evaluation and management of moyamoya disease

Edward R. Smith, Giuseppe Lanzino, Gary K. Steinberg, and Bin Xu

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Introduction

Incidentally discovered lesions

Cormac O. Maher, Paul Klimo Jr., and Edward R. Smith

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Protective effects of tirilazad mesylate and metabolite U-89678 against blood-brain barrier damage after subarachnoid hemorrhage and lipid peroxidative neuronal injury

Sarah L. Smith, Heidi M. Scherch, and Edward D. Hall

✓ The 21-aminosteroid lipid-peroxidation inhibitor, tirilazad mesylate (U-74006F), recently was shown in a large multinational Phase III clinical trial to decrease mortality and improve neurological recovery in patients 3 months after onset of aneurysmal subarachnoid hemorrhage (SAH). A major tirilazad metabolite in animals and man, U-89678 is formed when the 4–5 double bond in the A-ring is reduced and has been postulated to contribute significantly to tirilazad's neuroprotective effects. In the first experiment of the present study, the authors compared the effects of tirilazad and U-89678 on acute blood-brain barrier (BBB) damage in rats subjected to SAH via injection of 300 µl of autologous nonheparinized blood under the dura of the left cortex. The rats were treated by intravenous administration of either 0.3 or 1.0 mg/kg of tirilazad or U-89678 10 minutes before and 2 hours after SAH, and BBB damage was quantified according to the extravasation of the protein-bound Evans' blue dye into the injured cortex 3 hours post-SAH. The results revealed that 0.3 and 1.0 mg/kg tirilazad significantly reduced SAH-induced BBB damage 35.2% (p < 0.05) and 60.6% (p < 0.0001), respectively, in comparison to treatment with vehicle. The 0.3- and 1.0-mg/kg doses of U-89678 also decreased injury by 39.1% (p < 0.05) and 21.3% (not significant), respectively. In the second experiment, the investigators assessed the relative abilities of tirilazad and U-89678 to protect cultured neurons from iron-induced lipid peroxidative injury. Fetal mouse spinal cord cells were pretreated with 3, 10, or 30 µM tirilazad or U-89678 for 1 hour and then exposed to 200 µM ferrous ammonium sulfate (FAS) for 40 minutes. Cell viability was measured in terms of the uptake of [3H]α-(methyl)-aminoisobutyric acid 45 minutes after the FAS treatment. Both compounds enhanced neuronal survival in a concentration-dependent fashion. Although the two were equally efficacious, U-89678 was slightly more potent than its parent. On the basis of these findings, the authors conclude that the tirilazad metabolite, U-89678, possesses vaso- and neuroprotective properties that are essentially equivalent to the parent 21-aminosteroid. Hence, U-89678 probably contributes to the protective effects of tirilazad in SAH and other insults to the central nervous system.