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Pseudogout, an unconventional imitator of the lumbosacral spine with associated chronic epidural hematoma: illustrative cases

Stephen Jaffee, Seung Won Jeong, Rocco Dabecco, Shahed Elhamdani, and Alexander Yu

BACKGROUND

Pseudogout, or calcium pyrophosphate dihydrate (CPPD) disease, is an inflammatory joint disease that most commonly involves the joints of the knees, ankles, and wrists. Pseudogout has also been known to involve the spine, especially the atlanto-occipital joint of the cervical spine, but there is limited documentation of its involvement in the lumbar spine. Though the atypical presentation of spinal pseudogout with findings consistent with discitis and epidural abscess has been documented, its presentation with associated chronic spinal epidural hematoma is a rare entity.

OBSERVATIONS

The authors present two separate cases of pseudogout involvement of the lumbar spine, one case presenting with a clinical and radiographic picture consistent with discitis and epidural abscess and the other with radiographic and operative findings consistent with a chronic epidural hematoma.

LESSONS

This case series demonstrates rare and atypical presentations of pseudogout within the lumbosacral spine.

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Meta-analysis of the surgical outcomes of symptomatic moyamoya disease in adults

Jin Pyeong Jeon, Jeong Eun Kim, Won-Sang Cho, Jae Seung Bang, Young-Je Son, and Chang Wan Oh

OBJECTIVE

The purpose of this study was to evaluate treatment outcomes of future stroke prevention, perioperative complications, and angiographic revascularization in adults with symptomatic moyamoya disease (MMD) according to treatment modalities and surgical techniques.

METHODS

A systemic literature review was performed based on searches of the PubMed, Embase, and Cochrane Central databases. A fixed-effects model was used in cases of heterogeneity less than 50%. Publication bias was determined by Begg’s funnel plot, Egger’s test of the intercept, and the Begg and Mazumdar rank correlation test.

RESULTS

Eleven articles were included in the meta-analysis. Bypass surgery significantly decreased the future stroke events compared with conservative treatments in adult MMD (odds ratio [OR] 0.301, p < 0.001). Direct bypass showed better future stroke prevention than indirect bypass (OR 0.494, p = 0.028). There was no meaningful difference in perioperative complications between direct and indirect bypass (OR 0.665, p = 0.176). Direct bypass was associated with better angiographic outcomes than indirect bypass (OR 6.832, p < 0.001).

CONCLUSIONS

Bypass surgery can be effective in preventing future stoke events in adults with MMD. Direct bypass seems to provide better risk reduction with respect to stroke than indirect bypass in these patients.

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Angiographic findings of in-stent intimal hyperplasia after stent-assisted coil embolization: are they permanent findings?

Young Soo Kim, Sang Won Lee, Jeong A Yeom, Chang Hyo Yoon, and Seung Kug Baik

OBJECT

Stent-assisted coil embolization for the treatment of intracranial aneurysms has been used widely. This study aimed to investigate the effect of stent implantation in the nonatherosclerotic parent artery with cerebral aneurysms. The authors evaluated luminal changes and the related factors following stent-assisted coil embolization.

METHODS

This study included 97 patients harboring a total of 99 unruptured aneurysms of the distal internal carotid artery (ICA) who underwent single-stent implantation and more than 1 session of conventional angiography during follow-up (midterm follow-up only, n = 70; midterm and long-term follow-up, n = 29) between January 2009 and April 2014. The luminal narrowing point was measured using a local thickness map (ImageJ plug-in).

RESULTS

Stent-assisted coil embolization caused dynamic luminal narrowing of approximately 82% of the parent artery diameter on average after 8 months, which was reversed to 91% after 25 months. In addition, luminal narrowing greater than 40% was noticed in 2 (7%) of the 29 patients who experienced spontaneous reversion without additional management during follow-up. Most luminal narrowing changes seen were diffuse.

CONCLUSIONS

Luminal narrowing after aneurysm stent-assisted coil embolization is a dynamic process and appears to be a spontaneously reversible event. Routine management of luminal narrowing may not cause adverse events that require additional treatment.

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Surgical treatment of symptomatic Rathke cleft cysts: clinical features and results with special attention to recurrence

Jeong Eun Kim, Jeong Hoon Kim, Oh Lyong Kim, Sun Ha Paek, Dong Gyu Kim, Je G. Chi, and Hee-Won Jung

Object. Rathke cleft cysts (RCCs) are rarely symptomatic. The purpose of this study was to clarify the clinical, neuroimaging, surgical, and pathological features of symptomatic RCCs with special attention to their recurrence.

Methods. This retrospective study involved 53 patients with pathologically confirmed symptomatic RCCs. There were 28 female and 25 male patients, ranging in age from 11 to 68 years (mean 37 years). Common clinical presentations included headache, visual impairment, and endocrine disturbance. The most common endocrine disturbances were hyperprolactinemia and diabetes insipidus. Most of these improved or were resolved after surgery, with the exception of diabetes insipidus and panhypopituitarism. The cysts were intrasellar with suprasellar extension in 33 patients, and ranged in size from 5 to 40 mm (mean 17 mm). In the 50 magnetic resonance (MR) images that were reviewed, the signal intensities were quite variable. Fourteen MR images demonstrated enhancement after an injection of Gd. Intraoperatively, the cyst contents were found to be yellowish (18 patients [37%]) and mucoid (25 patients [51%]). Pathological examinations revealed a pseudostratified columnar epithelium in 26 patients (49%). Abundant squamous metaplasia and a stratified squamous epithelium were also found in 12 patients (23%). Follow-up MR images revealed cyst recurrences that required a repeated operation in six patients. Statistically significant risk factors for a recurrence included enhancement of the lesion on MR images (p = 0.017), the extent of cyst removal (p = 0.012), and the presence of squamous epithelium (p = 0.008).

Conclusions. Rathke cleft cysts are associated with a variety of clinical presentations and sometimes confusing intraoperative and pathological findings. Close postoperative observation with neuroimaging and neuroophthalmological assessment is necessary, especially after a partial removal, as in cases with squamous metaplasia.

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Novel diffusion tractography methodology using Kalman filter prediction to improve preoperative benefit-risk analysis in pediatric epilepsy surgery

Min-Hee Lee, Nolan B. O’Hara, Hirotaka Motoi, Aimee F. Luat, Csaba Juhász, Sandeep Sood, Eishi Asano, and Jeong-Won Jeong

OBJECTIVE

In this study the authors investigated the clinical reliability of diffusion weighted imaging maximum a posteriori probability (DWI-MAP) analysis with Kalman filter prediction in pediatric epilepsy surgery. This approach can yield a suggested resection margin as a dynamic variable based on preoperative DWI-MAP pathways. The authors sought to determine how well the suggested margin would have maximized occurrence of postoperative seizure freedom (benefit) and minimized occurrence of postoperative neurological deficits (risk).

METHODS

The study included 77 pediatric patients with drug-resistant focal epilepsy (age 10.0 ± 4.9 years) who underwent resection of their presumed epileptogenic zone. In preoperative DWI tractography from the resected hemisphere, 9 axonal pathways, Ci=1–9, were identified using DWI-MAP as follows: C1–3 supporting face, hand, and leg motor areas; C4 connecting Broca’s and Wernicke’s areas; C5–8 connecting Broca’s, Wernicke’s, parietal, and premotor areas; and C9 connecting the occipital lobe and lateral geniculate nucleus. For each Ci, the resection margin, di, was measured by the minimal Euclidean distance between the voxels of Ci and the resection boundary determined by spatially coregistered postoperative MRI. If Ci was resected, di was assumed to be negative (calculated as –1 × average Euclidean distance between every voxel inside the resected Ci volume, ri). Kalman filter prediction was then used to estimate an optimal resection margin, d*i, to balance benefit and risk by approximating the relationship between di and ri. Finally, the authors defined the preservation zone of Ci that can balance the probability of benefit and risk by expanding the cortical area of Ci up to d*i on the 3D cortical surface.

RESULTS

In the whole group (n = 77), nonresection of the preoperative preservation zone (i.e., actual resection margin d*i greater than the Kalman filter–defined d*i) accurately predicted the absence of postoperative motor (d*1–3: 0.93 at seizure-free probability of 0.80), language (d*4–8: 0.91 at seizure-free probability of 0.81), and visual deficits (d*9: 0.90 at seizure-free probability of 0.75), suggesting that the preservation of preoperative Ci within d*i supports a balance between postoperative functional deficit and seizure freedom. The subsequent subgroup analyses found that preservation of preoperative Ci =1–4,9 within d*i =1–4,9 may provide accurate deficit predictions independent of age and seizure frequency, suggesting that the DWI-based surgical margin can be effective for surgical planning even in young children and across a range of epilepsy severity.

CONCLUSIONS

Integrating DWI-MAP analysis with Kalman filter prediction may help guide epilepsy surgery by visualizing the margins of the eloquent white matter pathways to be preserved.

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Prediction of postoperative deficits using an improved diffusion-weighted imaging maximum a posteriori probability analysis in pediatric epilepsy surgery

Min-Hee Lee, Nolan B. O’Hara, Yasuo Nakai, Aimee F. Luat, Csaba Juhasz, Sandeep Sood, Eishi Asano, and Jeong-Won Jeong

OBJECTIVE

This study is aimed at improving the clinical utility of diffusion-weighted imaging maximum a posteriori probability (DWI-MAP) analysis, which has been reported to be useful for predicting postoperative motor, language, and visual field deficits in pediatric epilepsy surgery. The authors determined the additive value of a new clustering mapping method in which average direct-flip distance (ADFD) reclassifies the outliers of original DWI-MAP streamlines by referring to their minimum distances to the exemplar streamlines (i.e., medoids).

METHODS

The authors studied 40 children with drug-resistant focal epilepsy (mean age 8.7 ± 4.8 years) who had undergone resection of the presumed epileptogenic zone and had five categories of postoperative deficits (i.e., hemiparesis involving the face, hand, and/or leg; dysphasia requiring speech therapy; and/or visual field cut). In pre- and postoperative images of the resected hemisphere, DWI-MAP identified a total of nine streamline pathways: C1 = face motor area, C2 = hand motor area, C3 = leg motor area, C4 = Broca’s area–Wernicke’s area, C5 = premotor area–Broca’s area, C6 = premotor area–Wernicke’s area, C7 = parietal area–Wernicke’s area, C8 = premotor area–parietal area, and C9 = occipital lobe–lateral geniculate nucleus. For each streamline of the identified pathway, the minimal ADFD to the nine exemplars corrected the pathway membership. Binary logistic regression analysis was employed to determine how accurately two fractional predictors, Δ1–9 (postoperative volume change of C1–9) and γ1–9 (preoperatively planned volume of C1–9 resected), predicted postoperative motor, language, and visual deficits.

RESULTS

The addition of ADFD to DWI-MAP analysis improved the sensitivity and specificity of regression models for predicting postoperative motor, language, and visual deficits by 28% for Δ1–3 (from 0.62 to 0.79), 13% for Δ4–8 (from 0.69 to 0.78), 13% for Δ9 (from 0.77 to 0.87), 7% for γ1–3 (from 0.81 to 0.87), 1% for γ4–8 (from 0.86 to 0.87), and 24% for γ9 (from 0.75 to 0.93). Preservation of the eloquent pathways defined by preoperative DWI-MAP analysis with ADFD (up to 97% of C1–4,9) prevented postoperative motor, language, and visual deficits with sensitivity and specificity ranging from 88% to 100%.

CONCLUSIONS

The present study suggests that postoperative functional outcome substantially differs according to the extent of resected white matter encompassing eloquent cortex as determined by preoperative DWI-MAP analysis. The preservation of preoperative DWI-MAP–defined pathways may be crucial to prevent postoperative deficits. The improved DWI-MAP analysis may provide a complementary noninvasive tool capable of guiding the surgical margin to minimize the risk of postoperative deficits for children.

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Clinical features and treatment outcomes of the spinal arteriovenous fistulas and malformations

Clinical article

Won-Sang Cho, Ki-Jeong Kim, O-Ki Kwon, Chi Heon Kim, Jiha Kim, Moon Hee Han, and Chun Kee Chung

Object

Spinal vascular diseases, such as spinal dural arteriovenous fistulas (DAVFs), perimedullary arteriovenous fistulas (AVFs), and spinal arteriovenous malformations (AVMs), are very rare. The authors analyzed the features and treatment outcomes of these conditions.

Methods

Data from 64 patients were retrospectively reviewed. There were 33 spinal DAVFs (1 patient had 2 lesions), 20 perimedullary AVFs, and 12 spinal AVMs. Clinical features, radiological findings, treatment results, and clinical outcomes were evaluated according to the diseases, subtypes, and treatment modalities. The median duration of follow-up was 20, 42, and 56 months for spinal DAVFs, perimedullary AVFs, and spinal AVMs, respectively.

Results

Spinal DAVFs showed faster progression of symptoms (median 5, 12, and 36 months for spinal DAVFs, perimedullary AVFs, and spinal AVMs, respectively) and worse neurological status at diagnosis (poor neurological status in 56%, 65%, and 33%, respectively). On MRI, signal voids were demonstrated in all except 1 spinal DAVF. At the last follow-up, 94% of spinal DAVFs, 68% of perimedullary AVFs, and 50% of spinal AVMs were completely obliterated. Favorable clinical outcomes were achieved in 91%, 95%, and 58%, respectively. In detail, the majority (78%) of spinal DAVFs were embolized, resulting in complete obliteration in 92% and favorable clinical outcomes in 92%. Most Type IVa and IVb perimedullary AVFs were surgically treated (71% and 88%), with complete obliterations of 86% and 71%, and favorable clinical outcomes in 100% and 86%, respectively. All Type IVc lesions were embolized with a low cure rate of 40%; however, clinical outcomes were satisfactory. Spinal AVMs were generally embolized (67%), and only glomus-type lesions attained a satisfactory cure rate (80%) and clinical outcome (100%).

Conclusions

Embolization produced satisfactory outcomes in spinal DAVFs and glomus-type spinal AVMs. Surgery is advantageous in Type IVa and IVb perimedullary AVFs. Palliative embolization can be effective in Type IVc perimedullary AVFs and juvenile spinal AVMs.

Free access

Delayed ischemic events with low-dose prasugrel medication for stent-assisted coil embolization in intracranial aneurysm patients

Hyun Ho Choi, Heui Seung Lee, Sung Ho Lee, Kangmin Kim, Won-Sang Cho, Jeong Eun Kim, and Hyun-Seung Kang

OBJECTIVE

Much emphasis has been put on the use of antiplatelet medication for the prevention of ischemic events in the treatment of cerebral aneurysms with stent assistance. In this regard, the effectiveness and safety of a low-dose prasugrel regimen during the periprocedural period was recently reported. The purpose of this study was to present the outcomes of patients on low-dose prasugrel regimens during the follow-up period after stent-assisted coil embolization (SACE) of cerebral aneurysms.

METHODS

For the 396 consecutive patients undergoing SACE procedures, low-dose prasugrel therapy (5 mg of prasugrel and 100 mg of aspirin) was recommended for 3 months after the endovascular treatment. The authors performed a retrospective review of a single-center experience focusing on delayed ischemic events beyond 1 month after treatment. The mean follow-up period was 24.6 ± 11.3 months.

RESULTS

In this cohort of patients on a low-dose prasugrel regimen, cerebral infarction occurred in 1 patient (0.3%, 95% CI 0%–1.8%) beyond 1 month after SACE. No intracranial hemorrhage occurred. Overall ischemic events occurred in 14 patients (3.5%, 95% CI 2.1%–5.9%), all within 6 months of the coiling procedure. All patients had transient symptoms. The events occurred within 2 months after cessation of prasugrel in 11 patients (78.6%). Prasugrel maintenance for 6 months was found to result in lower ischemic events compared with maintenance for 3 months.

CONCLUSIONS

For patients undergoing SACE, a low-dose prasugrel regimen was a safe and reliable treatment option for the prevention of delayed ischemic events. Transient ischemic events often occurred within 2 months of stopping prasugrel medication.

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Clinical and neuroimaging outcome of cerebral arteriovenous malformations after Gamma Knife surgery: analysis of the radiation injury rate depending on the arteriovenous malformation volume

Jung Ho Han, Dong Gyu Kim, Hyun-Tai Chung, Chul-Kee Park, Sun Ha Paek, Jeong Eun Kim, Hee-Won Jung, and Dae Hee Han

Object

In this paper the authors analyzed the clinical and neuroimaging outcomes of patients with cerebral arteriovenous malformations (AVMs) after Gamma Knife surgery (GKS), focusing on the analysis of the radiation injury rate depending on the AVM volume.

Methods

Between 1997 and 2004, 277 consecutive patients with cerebral AVMs were treated with GKS. Of these patients, 218 were followed up for ≥ 2 years. The mean age was 31 ± 15 years, the median AVM volume was 3.4 cm3 (range 0.17–35.2 cm3), the median marginal dose was 18.0 Gy (range 10.0–25.0 Gy), and the mean follow-up duration was 44 ± 20 months. The authors reduced the prescription dose by various amounts, depending on the AVM volume and location as prescribed in the classic guideline to avoid irreversible radiation injuries.

Results

The angiographic obliteration rate was 66.4% overall, and it was 81.7, 53.1, and 12.5% for small, medium, and large AVMs, respectively. The overall annual bleeding rate was 1.9%. The annual bleeding rate was 0.44 and 4.64% for small and large AVMs, respectively. Approximately 20% of the patients showed severe postradiosurgery imaging (PRI) changes. The rate of PRI change was 11.4, 33.3, and 9.5% for small, medium, and large AVM volume groups, respectively, and a permanent radiation injury developed in 5.1% of patients.

Conclusions

By using the reduced dose from what is usually prescribed, the authors were able to obtain outcomes in small AVMs that were comparable to the outcomes described in previous reports. However, medium AVMs appear to still be at risk for adverse radiation effects. Last, in large AVMs, the authors were able to attain a tolerable rate of radiation injury; however, the clinical outcomes were quite disappointing following administration of a reduced dose of GKS for large AVMs.

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Clinical and radiological outcomes of proactive Gamma Knife surgery for asymptomatic meningiomas compared with the natural course without intervention

Kyung Hwan Kim, So Jeong Kang, Jung-Won Choi, Doo-Sik Kong, Ho Jun Seol, Do-Hyun Nam, and Jung-Il Lee

OBJECTIVE

This study aimed to verify the effect of proactive Gamma Knife surgery (GKS) in the treatment of asymptomatic meningioma compared with the natural course without any therapeutic intervention.

METHODS

From January 2006 to May 2017, 354 patients newly diagnosed with asymptomatic meningioma were reviewed and categorized into GKS (n = 153) and observation (n = 201) groups. Clinical and radiological progression rates were examined, and changes in volume were analyzed.

RESULTS

Clinical progression (i.e., clinician-judged progression), combining symptomatic progression (n = 43) and clinician-judged increase in size using images routinely acquired (n = 34), occurred in 4 patients (2.6%) and 73 patients (36.3%) in the GKS and observation groups, respectively (p < 0.001). The clinical progression-free survival (PFS) rates in the GKS and observation groups were 98.7% and 64.6%, respectively, at 5 years (p < 0.001), and 92.9% and 42.7%, respectively, at 10 years (p < 0.001). The radiological tumor control rate was 94.1% in the GKS group, and radiological progression was noted in 141 patients (70.1%) in the observation group. The radiological PFS rates in the GKS and observation groups were 94.4% and 38.5%, respectively, at 5 years (p < 0.001), and 88.5% and 7.9%, respectively, at 10 years (p < 0.001). Young age, absence of calcification, peritumoral edema, and high T2 signal intensity were correlated with clinical progression in the observation group. Volumetric analysis showed that untreated tumors gradually increased in size. However, GKS-treated tumors shrank gradually, although transient volume expansion was observed in the first 6 months. Adverse events developed in 26 of the 195 GKS-treated patients (13.3%), including 1 (0.5%) major event requiring microsurgery due to severe edema after GKS. Peritumoral edema was related to the development of adverse events (p = 0.004).

CONCLUSIONS

Asymptomatic meningioma is a benign disease; however, nearly two-thirds of patients experience tumor growth and one-third of untreated patients eventually require neurosurgical interventions during watchful waiting. GKS can control tumors clinically and radiologically with high probability. Although the risk of transient adverse events exists, proactive GKS may be a reasonable treatment option when there are no comorbidities limiting life expectancy.