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Yang Kwon, Jae Sung Ahn, Sang Ryong Jeon, Jeong Hoon Kim, Chang Jin Kim, Jung Kyo Lee, Byung Duk Kwun, Do Hee Lee and Sun Young Kim

Object. The authors evaluated whether gamma knife radiosurgery (GKS) could be a causative factor in intratumoral bleeding in meningiomas.

Methods. Gamma knife radiosurgery was used in the treatment of 173 meningiomas during a 10-year period. Four patients suffered post-GKS intratumoral hemorrhage. The course in these patients was reviewed.

Four of 173 patients suffered an intratumoral hemorrhage during a follow-up period of 1 to 8 years. The risk of intratumoral bleeding after GKS for meningioma was 2.3%. Intracystic hemorrhage occurred in two patients 1 and 5 years, respectively, after radiosurgery. In the other two cases intratumoral bleeding occurred 2 and 8 years, respectively, after radiosurgery. Histological examination in three cases found no specific findings related to the postradiosurgical changes.

Conclusions. Because the reported risk of spontaneous intratumoral bleeding in meningiomas is 1.3 to 2.7%, the incidence in this series was not unduly high. Radiosurgery itself could not be shown to be a significant factor in the development of the intratumoral bleeding.

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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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Eun Jung Lee, Jeong Hoon Kim, Eun Suk Park, Young-Hoon Kim, Jae Koo Lee, Seok Ho Hong, Young Hyun Cho and Chang Jin Kim

OBJECTIVE

Advances in neuroimaging techniques have led to the increased detection of asymptomatic intracranial meningiomas (IMs). Despite several studies on the natural history of IMs, a comprehensive evaluation method for estimating the growth potential of these tumors, based on the relative weight of each risk factor, has not been developed. The aim of this study was to develop a weighted scoring system that estimates the risk of rapid tumor growth to aid treatment decision making.

METHODS

The authors performed a retrospective analysis of 232 patients with presumed IM who had been prospectively followed up in the absence of treatment from 1997 to 2013. Tumor volume was measured by imaging at each follow-up visit, and the growth rate was determined by regression analysis. Predictors of rapid tumor growth (defined as ≥ 2 cm3/year) were identified using a logistic regression model; each factor was awarded a score based on its own coefficient value. The probability (P) of rapid tumor growth was estimated using the following formula:

FD1

RESULTS

Fifty-nine tumors (25.4%) showed rapid growth. Tumor size (OR per cm3 1.07, p = 0.000), absence of calcification (OR 3.87, p = 0.004), peritumoral edema (OR 2.74, p = 0.025), and hyperintense or isointense signal on T2-weighted MRI (OR 3.76, p = 0.049) were predictors of tumor growth rate. In the Asan Intracranial Meningioma Scoring System (AIMSS), tumor size was categorized into 3 groups of < 2.5 cm, ≥ 2.5 to < 4.0 cm, and ≥ 4.0 cm in diameter and awarded a score of 0, 3, and 6, respectively; the parameters of calcification and peritumoral edema were categorized into 2 groups based on their presence or absence and given a score of 0 or 2 and 1 or 0, respectively; and the signal on T2-weighted MRI was categorized into 2 groups of hypointense and hyperintense/isointense and given a score of 0 or 2, respectively. The risk of rapid tumor growth was estimated to be < 10% when the total score was 0–2, 10%–50% when the total score was 3–6, and ≥ 50% when the total score was 7–11 (Hosmer-Lemeshow goodness-of-fit test, p = 0.9958). The area under the receiver operating characteristic curve was 0.86.

CONCLUSIONS

The authors suggest a weighted scoring system (AIMSS) that predicts the specific probability of rapid tumor growth for patients with untreated IM. This scoring system will aid treatment decision making in clinical settings by screening out patients at high risk for rapid tumor growth.

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Min A. Yoon, Eunhee Kim, Bae-Ju Kwon, Jeong Eun Kim, Hyun-Seung Kang, Jae Hyo Park, Chul-Ho Sohn, Ji-Hoon Kim and Dong Hoon Lee

Object

Reinforcement of aneurysms with additional wrapping is an alternative procedure if the aneurysm cannot be completely clipped. Wrapping with muslin (cotton gauze) rarely incites foreign body inflammatory reactions. In this study, the authors describe the clinical and radiological features of muslinomas or muslin-induced foreign body reactions that can develop after treatment of intracranial aneurysms.

Methods

Over a 3-year period, 5 patients with muslinomas underwent treatment at the authors' institution. All patients underwent aneursym clipping and wrapping, and were subsequently readmitted with acute or subacute neurological symptoms. Clinical and imaging features on diffusion weighted MR images and cerebral angiography images were retrospectively reviewed. The patients' clinical course and follow-up imaging studies were also evaluated.

Results

In all 5 cases, muslinomas were seen as rim-enhancing inflammatory masses around the clipped aneurysms with perilesional edema visible on MR images at the time of clinical deterioration. The MR images also demonstrated adhesive arachnoiditis with a sterile intracranial abscess in 3 patients, optic neuropathy in 2, parent artery narrowing in 2, and a resultant acute ischemic infarction in 1 patient. Follow-up imaging revealed resolution of both the perilesional edema and adhesive arachnoiditis but no significant changes in the muslinomas. All patients underwent conservative management and fully recovered, but during the follow-up period, 2 patients experienced clinical and radiological relapses.

Conclusions

When a patient with a history of wrapping of an aneurysm presents with acute neurological symptoms and an enhancing intracranial mass in the region of the surgical site on MR imaging, a muslin-induced foreign body inflammatory reaction should be considered in the differential diagnosis, and careful clinical and radiological follow-up is advised.

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Ji Hoon Phi, Sun Ha Paek, Hyun-Tai Chung, Sang Soon Jeong, Chul-Kee Park, Hee-Won Jung and Dong Gyu Kim

Object

The current study was undertaken to evaluate the tumor control rate and functional outcome after Gamma Knife surgery (GKS) in patients with a trigeminal schwannoma. The conditions associated with the development of cranial neuropathies after radiosurgery were scrutinized.

Methods

The authors reviewed the clinical records and radiological data in 22 consecutive patients who received GKS for a trigeminal schwannoma. The median tumor volume was 4.1 ml (0.2–12.0 ml), and the mean tumor margin dose was 13.3 ± 1.3 Gy at an isodose line of 49.9 ± 0.6% (mean ± standard deviation). The median clinical follow-up period was 46 months (range 24–89 months), and the median length of imaging follow-up was 37 months (range 24–79 months).

Results

Tumor growth control was achieved in 21 (95%) of the 22 patients. Facial pain responded best to radio-surgery, with two thirds of patients showing improvement. However, only one third of patients with facial hypesthesia improved. Six patients (27%) experienced new or worsening cranial neuropathies after GKS. Ten patients (46%) showed tumor expansion after radiosurgery, and nine of these also showed central enhancement loss. Loss of central enhancement, tumor expansion, and a tumor in a cavernous sinus were found to be significantly related to the emergence of cranial neuropathies.

Conclusions

The use of GKS to treat trigeminal schwannoma resulted in a high rate of tumor control and functional improvement. Cranial neuropathies are bothersome complications of radiosurgery, and tumor expansion in a cavernous sinus after radiosurgery appears to be the proximate cause of the complication. Loss of central enhancement could be used as a warning sign of cranial neuropathies, and for this vigilant patient monitoring is required.

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Yang Kwon, Sang Ryong Jeon, Jeong Hoon Kim, Jung Kyo Lee, Dong Sook Ra, Dong Joon Lee and Byung Duk Kwun

Object. The authors sought to analyze causes for treatment failure following gamma knife radiosurgery (GKS) for intracranial arteriovenous malformations (AVMs), in cases in which the nidus could still be observed on angiography 3 years postsurgery.

Methods. Four hundred fifteen patients with AVMs were treated with GKS between April 1990 and March 2000. The mean margin dose was 23.6 Gy (range 10–25 Gy), and the mean nidus volume was 5.3 cm3 (range 0.4–41.7 cm3). The KULA treatment planning system and conventional subtraction angiography were used in treatment planning.

One hundred twenty-three of these 415 patients underwent follow-up angiography after GKS. After 3 years the nidus was totally obliterated in 98 patients (80%) and partial obliteration was noted in the remaining 25.

There were several reasons why complete obliteration was not achieved in all cases: inadequate nidus definition in four patients, changes in the size and location of the nidus in five patients due to recanalization after embolization or reexpansion after hematoma reabsorption, a large AVM volume in five patients, a suboptimal radiation dose to the thalamic and basal ganglia in eight patients, and radioresistance in three patients with an intranidal fistula.

Conclusions. The causes of failed GKS for treatment of AVMs seen on 3-year follow-up angiograms include inadequate nidus definition, large nidus volume, suboptimal radiation dose, recanalization/reexpansion, and radioresistance associated with an intranidal fistula.

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Eun Suk Park, Do Hoon Kwon, Jun Bum Park, Do Hee Lee, Young Hyun Cho, Jeong Hoon Kim and Chang Jin Kim

Object

Brain metastases from hepatocellular carcinoma (HCC) are rare, and the evidence of the effectiveness of Gamma Knife surgery (GKS) in this disease is lacking. The authors report their institutional experience with GKS in patients with brain metastases from HCCs.

Methods

The authors retrospectively reviewed the medical records of 73 consecutive patients who had a combined total of 141 brain metastases arising from HCCs and were treated with GKS. Sixty-four (87.7%) patients were male, and the mean age of the patients was 52.5 years (range 30–79 years). The mean tumor volume was 7.35 cm3 (range 0.19–33.7 cm3). The median margin dose prescribed was 23 Gy (range 15–32 Gy). Univariate and multivariate survival analyses were performed to identify possible prognostic factors of outcomes.

Results

The estimated rate of local tumor control was 79.6% at 3 months after GKS. The median overall survival time after GKS was 16 weeks. The actuarial survival rates were 76.7%, 58.9%, and 26.0% at 4, 12, and 24 weeks after GKS, respectively. In the univariate analysis, an age of ≤ 65 years, Child-Pugh Class A (pertaining to liver function), high Karnofsky Performance Scale score (≥ 70), and low Radiation Therapy Oncology Group recursive partitioning analysis class (I or II) were positively associated with the survival times of patients. No statistically significant variable was identified in the multivariate analysis.

Conclusions

Although survival was extremely poor in patients with brain metastases from HCCs, GKS showed acceptable local tumor control at 3 months after the treatment. The authors suggest that GKS represents a noninvasive approach that may provide a valuable option for treating patients with brain metastases from HCCs.

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Yang Kwon, Jun Seok Bae, Jae Myung Kim, Do Hee Lee, Soon Young Kim, Jae Sung Ahn, Jeong Hoon Kim, Chang Jin Kim, Byung Duk Kwun and Jung Kyo Lee

✓ Tumors involving the optic nerve (optic glioma, optic nerve sheath meningioma) are benign but difficult to treat. Gamma knife surgery (GKS) may be a useful treatment. The authors present data obtained in three such cases and record the effects of GKS.

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Hyun-Seung Kang, Youn-Joo Moon, Young-Yim Kim, Woong-Yang Park, Ae Kyung Park, Kyu-Chang Wang, Jeong Eun Kim, Ji Hoon Phi, Ji Yeoun Lee and Seung-Ki Kim

Object

Moyamoya disease (MMD) is a cerebrovascular occlusive disease affecting bilateral internal carotid termini. Smooth-muscle cells are one of the major cell types involved in this disease process. The characteristics of circulating smooth-muscle progenitor cells (SPCs) in MMD are poorly understood. The authors purified SPCs from the peripheral blood of patients with MMD and sought to identify differentially expressed genes (DEGs) in SPCs from these patients.

Methods

The authors cultured and isolated SPCs from the peripheral blood of patients with MMD (n = 25) and healthy control volunteers (n = 22). After confirmation of the cellular phenotype, RNA was extracted from the cells and DEGs were identified using a commercially available gene chip. Real-time quantitative reverse transcription polymerase chain reaction was performed to confirm the putative pathogenetic DEGs.

Results

The SPC-type outgrowth cells in patients with MMD invariably showed a hill-and-valley appearance under microscopic examination, and demonstrated high α–smooth muscle actin, myosin heavy chain, and calponin expression (96.5% ± 2.1%, 42.8% ± 18.6%, and 87.1% ± 8.2%, respectively), and minimal CD31 expression (less than 1%) on fluorescence-activated cell sorter analysis. The SPCs in the MMD group tended to make more irregularly arranged and thickened tubules on the tube formation assay. In the SPCs from patients with MMD, 286 genes (124 upregulated and 162 downregulated) were differentially expressed; they were related to cell adhesion, cell migration, immune response, and vascular development.

Conclusions

With adequate culture conditions, SPCs could be established from the peripheral blood of patients with MMD. These cells showed specific DEGs compared with healthy control volunteers. This study provides a novel experimental cell model for further research of MMD.

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Seong-Cheol Park, Seung-Ki Kim, Byung-Kyu Cho, Hyun Jib Kim, Jeong Eun Kim, Ji Hoon Phi, In-One Kim and Kyu-Chang Wang

Object

Sinus pericranii (SP) is a rare venous varix in an extracranial location connected to the intracranial venous system. The aim of this retrospective study was to report on 16 pediatric cases of SP with consideration of the preoperative evaluation of surgical risk.

Methods

The study population consisted of 10 patients who had undergone surgery for SP and 6 patients with concomitant craniosynostosis and SP. The mean age of the patients at presentation was 3.7 years. To identify characteristics of SP with high operative risk, 8 cases in this report and 11 previously reported cases of SP with sufficient information were categorized on the basis of the number and size of SP, the number and size of transcranial channels, the venous drainage type, and the amount of blood loss. Hemorrhage amounts were classified into 3 grades based on the description of intraoperative blood loss.

Results

Sinus pericranii not associated with craniosynostosis were resected without any postoperative morbidity. Sinus pericranii associated with craniosynostosis were preserved. After craniofacial reconstruction, 2 cases of SP with craniosynostosis regressed, completely in one patient and partially in another. These 2 patients with SP were confirmed to have compromised intracranial sinus before craniofacial reconstruction. Among a total of 19 patients, multiplicity or size (> 6 cm) of SP (p = 0.036) and multiplicity (> 3) or size (> 3 mm) of transcranial channels (p = 0.004) was associated with more severe hemorrhage grade. Sinus pericranii with peripheral venous drainage (drainer type) was not associated with hemorrhage grade after classification into 3 grades (p = 0.192). However, all 3 cases of SP with massive Grade 3 hemorrhage were the drainer type. Hemorrhage grade was correlated with the number of risk factors for SP (r = 0.793, p < 0.001).

Conclusions

Three risk factors of SP and the presence of compromised intracranial sinus are markers for highrisk SP. “Squeezed-out sinus syndrome” is suggested as a concept for SP associated with compromised intracranial sinus, mainly caused by craniosynostosis. Sinus pericranii in squeezed-out sinus syndrome probably serves as a crucial alternative to venous drainage of the brain with intracranial venous compromise. Conservative treatment for such patients with SP is recommended.