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Julius Griauzde, Joseph J. Gemmete, Aditya S. Pandey and Neeraj Chaudhary

OBJECT

A CSF leak can be difficult to locate in patients who present with spontaneous intracranial hypotension (SIH). The purpose of this case series was to describe the authors’ experience with intrathecal preservative-free normal saline challenge coupled with contrast-enhanced MR myelography (CEMRM), which was used to provoke and detect a CSF leakage site in patients with SIH.

METHODS

The authors performed a retrospective review of the records of patients who underwent preservative-free normal saline challenge followed by intrathecal gadolinium (Gd) contrast infusion and MR myelography from 2010 to 2012.

RESULTS

The records survey identified 5 patients who underwent 6 procedures. Intrathecal preservative-free normal saline challenge followed by CEMRM identified a CSF leak during 5 of the 6 procedures. Previous CT myelograms were available from 4 patients, which did not reveal a leakage site. A CT myelogram of 1 patient showed a single leak, but the authors’ saline challenge-CEMRM technique identified multiple additional leakage sites. Three patients exhibited transient postprocedural symptoms related to the saline infusion, but no long-term or permanent adverse effects related to the procedure were observed.

CONCLUSIONS

Instillation of preservative-free normal saline into the thecal sac followed by intrathecal Gd infusion is a safe technique that may increase the detection of a CSF leak on MR myelography images in patients with SIH.

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Sravanthi Koduri, D. Andrew Wilkinson, Julius M. Griauzde, Joseph J. Gemmete and Cormac O. Maher

Moyamoya syndrome predisposes patients to ischemic or hemorrhagic stroke due to progressive narrowing of intracranial vessels with subsequent small-vessel collateralization. Dural arteriovenous fistulae (DAVFs) are most commonly noted after venous sinus or cortical vein thrombosis and are believed to be primarily due to venous hypertension and elevated sinus pressures, although there is no known association with moyamoya syndrome, or with surgical treatment for moyamoya disease (MMD). The authors present the case of a 14-year-old girl with Down syndrome treated using pial synangiosis for MMD who subsequently was noted to have bilateral DAVFs. This case provides a new perspective on the origins and underlying pathophysiology of both moyamoya syndrome and DAVFs, and also serves to highlight the importance of monitoring the moyamoya population closely for de novo cerebrovascular changes after revascularization procedures.

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Vijay M. Ravindra, Stephen F. Kralik, Julius Griauzde, Nisha Gadgil, Melissa A. LoPresti and Sandi Lam

OBJECTIVE

Moyamoya disease is a progressive occlusive arteriopathy for which surgical revascularization is indicated. In this retrospective study, the authors investigated the use of preoperative CT perfusion with the aim of establishing pathological data references.

METHODS

The authors reviewed the medical records of children with moyamoya disease treated surgically at one institution between 2016 and 2019. Preoperative CT perfusion studies were used to quantify mean transit time (MTT), cerebral blood volume (CBV), cerebral blood flow (CBF), and time to peak (TTP) for the anterior, middle, and posterior cerebral artery vascular territories for each patient. CT perfusion parameter ratios (diseased/healthy hemispheres) and absolute differences were compared between diseased and normal vascular territories (defined by catheter angiography studies). Sensitivity, specificity, and positive (PPV) and negative (NPV) predictive values for CT perfusion parameters for severe angiographic moyamoya were calculated.

RESULTS

Nine children (89% female) had preoperative CT perfusion data; 5 of them had evidence of unilateral hemispheric disease and 4 had bilateral disease. The mean age at revascularization was 77 months (range 40–144 months). The etiology of disease was neurofibromatosis type 1 (3 patients), Down syndrome (2), primary moyamoya disease (2), cerebral proliferative angiopathy (1), and sickle cell disease (1). Five patients had undergone unilateral revascularization. Among these patients, pathological vascular territories demonstrated increased MTT in 66% of samples, increased TTP in 66%, decreased CBF in 47%, and increased CBV in 87%. Severe moyamoya (Suzuki stage ≥ 4) had diseased/healthy ratios ≥ 1 for MTT in 78% of cases, for TTP in 89%, for CBF in 67%, and for CBV in 89%. The MTT and TTP region of interest ratio ≥ 1 demonstrated 89% sensitivity, 67% specificity, 80% PPV, and 80% NPV for the prediction of severe angiographic moyamoya disease.

CONCLUSIONS

Pathological hemispheres in these children with moyamoya disease demonstrated increased MTT, TTP, and CBV and decreased CBF. The authors’ results suggest that preoperative CT perfusion may, with high sensitivity, be useful in deciphering perfusion mismatch in brain tissue in children with moyamoya disease. More severe angiographic disease displays a more distinct correlation, allowing surgeons to recognize when to intervene in these patients.

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Julius Griauzde, Vijay M. Ravindra, Neeraj Chaudhary, Joseph J. Gemmete, Marcus D. Mazur, Christopher D. Roark, William T. Couldwell, Min S. Park, Philipp Taussky and Aditya S. Pandey

OBJECTIVE

Flow-diverting devices have been used for the treatment of complex intracranial vascular pathology with success, but the role of these devices in treating iatrogenic intracranial vascular injuries has yet to be clearly defined. Here, the authors report their bi-institutional experience with the use of the Pipeline embolization device (PED) for the treatment of iatrogenic intracranial vascular injuries.

METHODS

The authors reviewed a retrospective cohort of patients with iatrogenic injuries to the intracranial vasculature that were treated with the PED between 2012 and 2016. Data collection included demographic data, indications for treatment, number and sizes of PEDs used, and immediate and follow-up angiographic and clinical outcomes.

RESULTS

Four patients with a mean age of 47.5 years (range 18–63 years) underwent PED placement for iatrogenic vessel injuries. In 3 patients, the intracranial internal carotid artery (ICA) was injured during transnasal tumor resection. In 1 patient, a basilar apex injury occurred during endoscopic third ventriculostomy. Three patients had a pseudoaneurysm as a result of vessel injury, and 1 patient had frank ICA laceration and extravasation. All 3 pseudoaneurysms were successfully treated with PED deployment. The ICA laceration was refractory to PED placement, and the vessel was subsequently occluded endovascularly. All 4 patients had a good clinical outcome (modified Rankin Scale score of 0 or 1).

CONCLUSIONS

The use of the PED is feasible in the management of iatrogenic pseudoaneurysms of the intracranial vasculature. In cases of frank vessel perforation, an alternative strategy such as covered stent placement should be considered. Endovascular or surgical vessel occlusion remains the definitive treatment in cases of refractory hemorrhage.

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Vijay M. Ravindra, Michael Karsy, Arianna Lanpher, Robert J. Bollo, Julius Griauzde, R. Michael Scott, William T. Couldwell and Edward R. Smith

OBJECTIVE

Comprehensive multicenter data on the surgical treatment of pediatric cerebrovascular malformations (CVMs) in the US are lacking. The goal of this study was to identify national trends in patient demographics and assess the effect of hospital case volume on outcomes.

METHODS

Admissions for CVMs (1997–2012) were identified from the nationwide Kids’ Inpatient Database. Admissions with and without craniotomy were reviewed separately. Patients were categorized by whether they were treated at low-, medium-, or high-volume centers (< 10, 10–40, > 40 cases/year, respectively). A generalized linear model was used to evaluate the association of hospital pediatric CVM case volume and clinical variables assessing outcomes.

RESULTS

Among the 9655 patients, 1828 underwent craniotomy and 7827 did not. Patient age and race differed in the two groups, as did the rate of private medical payers. High-volume hospitals had fewer nonroutine discharges (11.2% [high] vs 16.4% [medium] vs 22.3% [low], p = 0.0001). For admissions requiring craniotomy, total charges ($106,282 [high] vs $126,215 [medium] vs $134,978 [low], p < 0.001) and complication rates (0.09% [high] vs 0.11% [medium] vs 0.16% [low], p = 0.001) were lower in high-volume centers.

CONCLUSIONS

This study revealed that further investigation may be needed regarding barriers to surgical treatment of pediatric CVMs. The authors found that surgical treatment of pediatric CVM at high-volume centers is associated with significantly fewer complications, better dispositions, and lower costs, but for noncraniotomy patients, low-volume centers had lower rates of complications and death and lower costs. These findings may support the consideration of appropriate referral of CVM patients requiring surgery or with intracranial hemorrhage toward high-volume, specialized centers.

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Vijay M. Ravindra, Robert J. Bollo, Ilyas M. Eli, Julius Griauzde, Arianna Lanpher, Jennifer Klein, Huirong Zhu, Douglas L. Brockmeyer, John R. W. Kestle, William T. Couldwell, R. Michael Scott and Edward Smith

OBJECTIVE

Large experiences with the treatment of pediatric arteriovenous malformations (AVMs) remain relatively rare, with limited data on presentation, treatment, and long-term functional outcomes. Because of the expected long lifespan of children, caregivers are especially interested in outcome measures that assess quality of life. The authors’ intention was to describe the long-term functional outcomes of pediatric patients who undergo AVM surgery and to identify predictors of sustained neurological deficits.

METHODS

The authors analyzed a 21-year retrospective cohort of pediatric patients with intracranial AVMs treated with microsurgery at two institutions. The primary outcome was a persistent neurological deficit at last follow-up. Secondary outcome measures included modified Rankin Scale (mRS) score and independent living.

RESULTS

Overall, 97 patients (mean age 11.1 ± 4.5 years; 56% female) were treated surgically for intracranial AVMs (mean follow-up 77.5 months). Sixty-four patients (66%) presented with hemorrhage, and 45 patients (46%) had neurological deficits at presentation. Radiologically, 39% of lesions were Spetzler-Martin grade II. Thirty-seven patients (38%) with persistent neurological deficits at last follow-up were compared with those without deficits; there were no differences in patient age, presenting Glasgow Coma Scale score, AVM size, surgical blood loss, or duration of follow-up. Multivariate analysis demonstrated that a focal neurological deficit on presentation, AVM size > 3 cm, and lesions in eloquent cortex were independent predictors of persistent neurological deficits at long-term follow-up. Overall, 92% of the children had an mRS score ≤ 2 on long-term follow-up.

CONCLUSIONS

Pediatric patients with AVMs treated with microsurgical resection have good functional and radiological outcomes. There is a high rate (38%) of persistent neurological deficits, which were independently predicted by preoperative deficits, AVMs > 3 cm, and lesions located in eloquent cortex. This information can be useful in counseling families on the likelihood of long-term neurological deficits after cerebral AVM surgery.