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Chih-Chen Chang, Hong-Shiu Chang and Cheng Hong Toh

The authors report a case in which intravitreous silicone oil migrated into the ventricles. They note that intraventricular silicone oil can be misdiagnosed as intraventricular hemorrhage and neurosurgeons should be aware of this possibility.

This 58-year-old woman with a history of Type II diabetic mellitus and retinal detachment (resulting from diabetic retinopathy), which had been treated with intravitreous silicone tamponade, presented with dizziness and headache approximately 10 years after the intravitreous silicone treatment. Over the next 6 years she underwent 2 non–contrast-enhanced brain CT studies and 1 MRI study for evaluation of her symptoms. On CT scan, extension of the intraocular silicone along the optic nerve was evident. Two hyperdense nodules were observed freely floating in the right lateral and fourth ventricles, remaining in the nondependent portion of ventricles in both supine and prone positions. On T2-weighted MRI, the left orbital content and the intraventricular nodules all demonstrated chemical shift artifacts typically associated with silicone. The imaging findings were characteristic for intraventricular silicone after silicone oil tamponade. The patient's dizziness and headache were treated symptomatically and she was followed up at the outpatient department.

Migration of intravitreous silicone oil into the cerebral ventricles is a rare complication. Intraventricular silicone oil can mimic intraventricular hemorrhage. Radiographically, intraventricular silicone oil can be distinguished from hemorrhage as silicone oil tends to stay in the nondependent portion of the ventricle. Chemical shift artifacts on MRI may help establishing the diagnosis of intraventricular silicone oil. Currently, there is no consensus on surgical removal of intraventricular silicone oil, and in the majority of cases reported in the literature, the patients were asymptomatic.

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Chen-Yu Ding, Han-Pei Cai, Hong-Liang Ge, Liang-Hong Yu, Yuan-Xiang Lin and De-Zhi Kang

OBJECTIVE

The relationship between lipoprotein-associated phospholipase A2 (Lp-PLA2) and various cardiovascular and cerebrovascular diseases is inconsistent. However, the connection between Lp-PLA2 level and delayed cerebral ischemia (DCI) following aneurysmal subarachnoid hemorrhage (aSAH) remains unclear. The objective of this study was to investigate the relationships between the Lp-PLA2 levels in the early stages of aSAH and the occurrence of DCI.

METHODS

The authors evaluated 114 patients with aSAH who were enrolled into a prospective observational cohort study. Serum Lp-PLA2 level at admission (D0), on the first morning (D1), and on the second morning of hospitalization (D2) were determined using commercial enzyme-linked immunosorbent assay kits. The relationship between Lp-PLA2 levels and DCI was analyzed.

RESULTS

Forty-three patients with aSAH (37.72%) experienced DCI. Mean serum Lp-PLA2 level decreased from 183.06 ± 61.36 μg/L at D0 (D0 vs D1, p = 0.303), to 175.32 ± 51.49 μg/L at D1 and 167.24 ± 54.10 μg/L at D2 (D0 vs D2, p = 0.040). The Lp-PLA2 level changes (D0-D1 and D0-D2) were comparable between patients with and without DCI. Multivariate model analysis revealed Lp-PLA2 level (D0) > 200 μg/L was a more significant factor of DCI compared with Lp-PLA2 (D1) and Lp-PLA2 (D2), and was a strong predictor of DCI (odds ratio [OR] 6.24, 95% confidence interval [CI] 2.05–18.94, p = 0.001) after controlling for World Federation of Neurosurgical Societies (WFNS) grade (OR 3.35, 95% CI 1.18–9.51, p = 0.023) and modified Fisher grade (OR 6.07, 95% CI 2.03–18.14, p = 0.001). WFNS grade (area under the curve [AUC] = 0.792), modified Fisher grade (AUC = 0.731), and Lp-PLA2 level (D0; AUC = 0.710) were all strong predictors of DCI. The predictive powers of WFNS grade, modified Fisher grade, and Lp-PLA2 (D0) were comparable (WFNS grade vs Lp-PLA2: p = 0.233; modified Fisher grade vs Lp-PLA2: p = 0.771). The poor-grade patients with Lp-PLA2 (D0) > 200 μg/L had significantly worse DCI survival rate than poor-grade patients with Lp-PLA2 (D0) ≤ 200 μg/L (p < 0.001).

CONCLUSIONS

The serum level of Lp-PLA2 was significantly elevated in patients with DCI, and decreased within the first 2 days after admission. Lp-PLA2 in the early stages of aSAH might be a novel predictive biomarker for the occurrence of DCI.

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Xu Bang-Zong, Pan Hong-Xue, Li Ke-Ming, Chen Xi-Jin, Tian Ying-Dei, Li Yong-Lin and Liu Jian

✓ A biomembrane was developed from pig peritoneum treated with 0.65% glutaraldehyde. This was evaluated for use as a dural substitute in an animal model and in a patient population. After being treated with the glutaraldehyde solution, the biomembrane lost its antigenicity while its collagen underwent an irreversible cross-linking reaction, causing it to become a stable nonviable polymer resistant to absorption by the host. The biomembrane was used experimentally in 43 procedures on 20 dogs and was applied clinically in 614 patients. The results demonstrated that it is an acceptable material for the repair of dural defects, with the following advantages: 1) it is nontoxic to the body and brain tissues, with minimal tissue reaction; 2) its biophysical properties facilitate watertight closure with sutures; 3) its distensibility makes it suitable for decompressive surgical dural repair; and 4) its visceral surface is extremely smooth, causing virtually no adhesions with the brain tissue while the outer surface readily heals with the subcutaneous tissue.

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Bo Wu, Hong Zhu, Weidong Liu and Longyi Chen

Radiation-induced cranial fasciitis is a rare complication of radiotherapy, especially in an intradiploic location. The authors report such a case of cranial fasciitis in a 13-year-old girl previously subjected to cranial radiotherapy for a recurrent cerebellar medulloblastoma. The patient had undergone a gross-total removal of a medulloblastoma followed by no radiation therapy at the age of 10 years. The tumor recurred at the original site 2 years later, warranting a repeat operation with a gross-total tumor removal and subsequent radiation therapy. The follow-up MRI sequence demonstrated no abnormal appearance for 1 year, until a new enhancing mass was found within the occipital bone adjacent to the prior bone window. Following its resection, the new lesion was histologically identified as cranial fasciitis. Differential diagnosis of a well-circumscribed bone lesion should include cranial fasciitis, especially in young children with radiotherapy for a previous intracranial malignancy. Radiotherapy should be considered among the inciting factors in the development of cranial fasciitis. The osteolytic lesions of cranial fasciitis, although nontumoral and self-limited in duration, should be eligible candidates for early, total resection to avoid potential intracranial expansion.

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Jian-Bin Chen, Ding Lei, Min He, Hong Sun, Yi Liu, Heng Zhang, Chao You and Liang-Xue Zhou

OBJECT

The present study aimed to clarify the incidence and clinical features of disease progression in adult moyamoya disease (MMD) patients with Graves disease (GD) for better management of these patients.

METHODS

During the past 18 years, 320 adult Chinese patients at West China Hospital were diagnosed with MMD, and 29 were also diagnosed with GD. A total of 170 patients (25 with GD; 145 without GD) were included in this study and were followed up. The mean follow-up was 106.4 ± 48.6 months (range 6–216 months). The progression of the occlusive lesions in the major intracranial arteries was measured using cerebral angiography and was evaluated according to Suzuki's angiographic staging. Information about cerebrovascular strokes was obtained from the records of patients' recent clinical visits. Both angiographic progression and strokes were analyzed to estimate the incidences of angiographic progression and strokes using Kaplan-Meier analysis. A multivariate logistic regression model was used to test the effects of sex, age at MMD onset, disease type, strokes, and GD on the onset of MMD progression during follow-up.

RESULTS

During follow-up, the incidence of disease progression in MMD patients with GD was significantly higher than in patients without GD (40.0% vs 20.7%, respectively; p = 0.036). The interval between initial diagnosis and disease progression was significantly shorter in MMD patients with GD than in patients without GD (p = 0.041). Disease progression occurred in both unilateral MMD and bilateral MMD, but the interval before disease progression in patients with unilateral disease was significantly longer than in patients with bilateral disease (p = 0.021). The incidence of strokes in MMD patients with GD was significantly higher than in patients without GD (48% vs 26.2%, respectively; p = 0.027). The Kaplan-Meier survival curve showed significant differences in the incidence of disease progression (p = 0.038, log-rank test) and strokes (p = 0.031, log-rank test) between MMD patients with GD and those without GD. Multivariate analysis suggested that GD may contribute to disease progression in MMD (OR 5.97, 95% CI 1.24–33.76, p = 0.043).

CONCLUSIONS

The incidence of disease progression in MMD patients with GD was significantly higher than that in MMD patients without GD, and GD may contribute to disease progression in MMD patients. The incidence of strokes was significantly higher in MMD patients with GD than in patients without GD. Management guidelines for MMD patients with GD should be developed.

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Jian-Bin Chen, Yi Liu, Liang-Xue Zhou, Hong Sun, Min He and Chao You

OBJECT

This study explored whether there were differences between the autoimmune disease prevalence rates in unilateral and bilateral moyamoya disease (MMD).

METHODS

The authors performed a retrospective review of data obtained from the medical records of their hospital, analyzing and comparing the clinical characteristics and prevalence rates of all autoimmune diseases that were associated with unilateral and bilateral MMD in their hospital from January 1995 to October 2014.

RESULTS

Three hundred sixteen patients with bilateral MMD and 68 with unilateral MMD were identified. The results indicated that patients with unilateral MMD were more likely to be female than were patients with bilateral MMD (67.6% vs 51.3%, p = 0.014, odds ratio [OR] 1.99). Overall, non-autoimmune comorbidities tended to be more prevalent in the unilateral MMD cases than in the bilateral MMD cases (17.6% vs 9.8%, p = 0.063, OR 1.97, chi-square test). Autoimmune thyroid disease and other autoimmune diseases also tended to be more prevalent in the unilateral MMD cases than in the bilateral MMD cases (19.1% vs 10.8%, p = 0.056, OR 1.96 and 8.8% vs 3.5%, p = 0.092, OR 2.77, respectively, chi-square test). The overall autoimmune disease prevalence in the unilateral MMD cases was significantly higher than in the bilateral MMD cases (26.5% vs 13.6%, p = 0.008, OR 2.29, 95% CI 1.22–4.28, chi-square test). Multiple logistic regression analysis showed that autoimmune disease was more likely to be associated with unilateral than with bilateral MMD (p = 0.039, OR 10.91, 95% CI 1.13–105.25).

CONCLUSIONS

This study indicated a higher overall autoimmune disease prevalence in unilateral than in bilateral MMD. Unilateral MMD may be more associated with autoimmune disease than bilateral MMD. Different pathogenetic mechanisms may underlie moyamoya vessel formation in unilateral and bilateral MMD.

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Xiao-Qiang Wang, Cheng-Chuan Jiang, Lin Zhao, Ye Gong, Jie Hu and Hong Chen

Object

High-grade meningiomas in childhood are rare, and their clinical features are unknown. The objective of this study was to determine the clinical characteristics and prognosis of childhood high-grade meningiomas.

Methods

Twenty-three patients with childhood high-grade meningiomas were treated at the Huashan Hospital. Clinical data were collected, tumor samples were reexamined, and prognoses were attained through follow-up visits and telephone interviews. Survival probability was calculated using the Kaplan-Meier method. A 2-sided probability level of 0.05 was chosen for statistical significance.

Results

The series included 18 males and 5 females (mean age 12.1 years). The most common symptoms were headache and vomiting (43%). Three patients had accompanying neurofibromatosis Type II (NF2). The high-grade meningioma cases with NF2 had larger tumor diameters than those without NF2 (p = 0.010). The skull base was the most common tumor site (39%). Complete resections were achieved in 11 patients after their initial operations. Adjuvant radiation therapy was performed in 9 cases. Follow-up evaluations were performed for 20 patients (mean follow-up 70 months). Ten patients experienced recurrences, 2 patients had lung metastases, and 7 patients died of the recurrence. The extent of surgery was significantly related to progression-free survival (PFS; p = 0.038). A negative progesterone receptor combined with strongly positive Bcl-2 immunoreactivity was significantly related to PFS (p = 0.001) and overall survival (p = 0.002). The MIB-1 labeling index was significantly related to overall survival (p = 0.018), whereas postoperative radiation therapy was not significantly related to PFS (p = 0.087) and overall survival (p = 0.40).

Conclusions

Childhood high-grade meningioma is a rare tumor type. Childhood high-grade meningioma has a male predominance and the basilar region is the most common tumor location. Patients with these tumors have high recurrence and mortality rates. The extent of resection is an important prognosis factor. A negative progesterone receptor combined with a strongly positive Bcl-2 immunoreaction might predict cancer recurrence. The MIB-1 labeling index correlates with the prognosis, and an MIB-1 labeling index > 3% increases the risk of recurrence in childhood high-grade meningioma. More cases should be collected, and longer follow-up periods should be obtained, to evaluate the effects of postoperative radiation therapy in childhood high-grade meningioma.

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Hong-Qi Zhang, Ling-Qiang Chen, Shao-Hua Liu, Di Zhao and Chao-Feng Guo

Object

The object of this study was to evaluate the efficacy and safety of posterior decompression with kyphosis correction for thoracic myelopathy due to ossification of the ligamentum flavum (OLF) and ossification of the posterior longitudinal ligament (OPLL) at the same level.

Methods

Between January 2003 and December 2005, 11 patients (8 men and 3 women) with thoracic myelopathy due to OLF and OPLL at the same level underwent posterior decompressive laminectomy and excision of OLF. Posterior instrumentation was also performed for stabilization of the spine and reducing the thoracic kyphosis angle by approximately 5–15° (kyphosis correction), and spinal fusion was performed in all cases. The follow-up period ranged from 2 to 4 years (mean 2.8 years). The outcomes were evaluated using a recovery scale based on the Japanese Orthopaedic Association classification. The score of each patient was calculated before surgery, 1 year after surgery, and at the final follow-up visit.

Results

After surgery, the thoracic kyphosis in the stabilization area was reduced from 30.0 ± 4.02° to 20.8 ± 2.14° on average. The mean score on the Japanese Orthopaedic Association scale improved from 3.5 ± 1.69 preoperatively to 8.5 ± 1.63 at the final follow-up, with a recovery rate of 68.0%. The results were good in 9 patients and fair in 2 patients. Postoperative MR imaging showed that the spinal cord was shifted posteriorly and decompressed completely in all cases. Myelopathy was not aggravated in any case after surgery.

Conclusions

A considerable degree of neurological recovery was observed after posterior decompression and kyphosis correction. The procedure is easy to perform with a low risk of postoperative paralysis. The authors therefore suggest that the procedure is useful for patients whose spinal cords are severely impinged by OLF and OPLL at the same level.

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Cheng-Hong Toh, Yao-Liang Chen, Ho-Fai Wong, Kuo-Chen Wei, Shu-Hang Ng and Yung-Liang Wan

✓ Rosai—Dorfman disease (RDD) is an idiopathic proliferation of histiocytes that affects the lymph nodes. Central nervous system involvement in the absence of nodal disease is extremely rare. On neuroimaging studies, intracranial RDD appears as solitary or multiple well-circumscribed, dura-based lesions. The authors report on two cases of RDD with locally aggressive features including dural sinus invasion, which to their knowledge has never before been described.

A 60-year-old woman presented with progressive dizziness and vertigo that had lasted for 1 week. Cranial computerized tomography and magnetic resonance imaging revealed an extraaxial homogeneous lobulated enhancing mass involving the right occipital lobe and the right cerebellar hemisphere. Invasion of the right transverse sinus was identified on a cerebral digital subtraction angiogram. A 59-year-old man with no prior medical illness experienced progressive weakness of both upper extremities and a partial complex seizure. Magnetic resonance imaging of his brain revealed a well-circumscribed enhancing mass in the left frontal lobe with extension to the right frontal lobe and invasion of the superior sagittal sinus. Both patients underwent resection of their brain masses. Pathological studies identified the disease as RDD in both patients.