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Movement Disorders : Official Journal... Jul 2022Mutations in the GBA gene cause Gaucher's disease (GD) and constitute the most frequent genetic risk factor for idiopathic Parkinson's disease (iPD). Nonmanifesting... (Review)
Review
BACKGROUND
Mutations in the GBA gene cause Gaucher's disease (GD) and constitute the most frequent genetic risk factor for idiopathic Parkinson's disease (iPD). Nonmanifesting carriers of GBA mutations/variants (GBA-NMC) constitute a potential PD preclinical population, whereas PD patients carrying some GBA mutations/variants (GBA-PD) have a higher risk of a more aggressive disease course. Different neuroimaging techniques are emerging as potential biomarkers in PD and have been used to study GBA-associated parkinsonism.
OBJECTIVE
The aim is to critically review studies applying neuroimaging to GBA-associated parkinsonism.
METHODS
Literature search was performed using PubMed and EMBASE databases (last search February 7, 2022). Studies reporting neuroimaging findings in GBA-PD, GD with and without parkinsonism, and GBA-NMC were included.
RESULTS
Thirty-five studies were included. In longitudinal studies, GBA-PD patients show a more aggressive disease than iPD at both structural magnetic resonance imaging and 123-fluoropropylcarbomethoxyiodophenylnortropane single-photon emission computed tomography. Fluorodeoxyglucose-positron emission tomography and brain perfusion studies reported a greater cortical involvement in GBA-PD compared to iPD. Overall, contrasting evidence is available regarding GBA-NMC for imaging and clinical findings, although subtle differences have been reported compared with healthy controls with no mutations.
CONCLUSIONS
Although results must be interpreted with caution due to limitations of the studies, in line with previous clinical observations, GBA-PD showed a more aggressive disease progression in neuroimaging longitudinal studies compared to iPD. Cognitive impairment, a "clinical signature" of GBA-PD, seems to find its neuroimaging correlate in the greater cortical burden displayed by these patients as compared to iPD. © 2022 The Authors. Movement Disorders published by Wiley Periodicals LLC on behalf of International Parkinson and Movement Disorder Society.
Topics: Gaucher Disease; Glucosylceramidase; Humans; Neuroimaging; Parkinson Disease; Parkinsonian Disorders
PubMed: 35521899
DOI: 10.1002/mds.29047 -
Neurosurgical Review Jun 2023Deep-seated unruptured AVMs located in the thalamus, basal ganglia, or brainstem have a higher risk of hemorrhage compared to superficial AVMs and surgical resection is... (Meta-Analysis)
Meta-Analysis Review
Deep-seated unruptured AVMs located in the thalamus, basal ganglia, or brainstem have a higher risk of hemorrhage compared to superficial AVMs and surgical resection is more challenging. Our systematic review and meta-analysis provide a comprehensive summary of the stereotactic radiosurgery (SRS) outcomes for deep-seated AVMs. This study follows the guidelines set forth by the Preferred Reporting Items for Systematic Reviews and Meta-Analysis (PRISMA) Statement. We conducted a systematic search in December 2022 for all reports of deep-seated arteriovenous malformations treated with SRS. Thirty-four studies (2508 patients) were included. The mean obliteration rate in brainstem AVM was 67% (95% CI: 0.60-0.73), with significant inter-study heterogeneity (tau = 0.0113, I = 67%, chi = 55.33, df = 16, p-value < 0.01). The mean obliteration rate in basal ganglia/thalamus AVM was 65% (95% CI: 0.58-0.72) with significant inter-study heterogeneity (tau = 0.0150, I = 78%, chi = 81.79, df = 15, p-value < 0.01). The presence of deep draining veins (p-value: 0.02) and marginal radiation dose (p-value: 0.04) were positively correlated with obliteration rate in brainstem AVMs. The mean incidence of hemorrhage after treatment was 7% for the brainstem and 9% for basal ganglia/thalamus AVMs (95% CI: 0.05-0.09 and 95% CI: 0.05-0.12, respectively). The meta-regression analysis demonstrated a significant positive correlation (p-value < 0.001) between post-operative hemorrhagic events and several factors, including ruptured lesion, previous surgery, and Ponce C classification in basal ganglia/thalamus AVMs. The present study found that radiosurgery appears to be a safe and effective modality in treating brainstem, thalamus, and basal ganglia AVMs, as evidenced by satisfactory rates of lesion obliteration and post-surgical hemorrhage.
Topics: Humans; Treatment Outcome; Follow-Up Studies; Radiosurgery; Intracranial Arteriovenous Malformations; Postoperative Hemorrhage; Basal Ganglia; Brain Stem; Thalamus; Retrospective Studies
PubMed: 37358733
DOI: 10.1007/s10143-023-02059-4 -
Neuro-oncology Practice Aug 2021Large vestibular schwannomas (VS) pose a treatment challenge for both microsurgery (MS) and stereotactic radiosurgery (SRS). Technical developments have allowed for... (Review)
Review
BACKGROUND
Large vestibular schwannomas (VS) pose a treatment challenge for both microsurgery (MS) and stereotactic radiosurgery (SRS). Technical developments have allowed for safer irradiation of large tumors. It remains unclear if SRS can achieve appropriate tumor control and acceptable cranial nerve toxicities. In this study, we assess outcomes of irradiation for large VS.
METHODS
PubMed MEDLINE, EMBASE, Web of Science, and Cochrane were searched for all the studies assessing SRS outcome in large VS. Primary endpoints included clinical and radiographic tumor control, need for salvage surgery, serviceable hearing, cranial nerve V and VII impairment, presence of hydrocephalus requiring shunting, and presence of vertigo/dizziness.
RESULTS
Twenty-two studies were identified that met selection criteria for analysis from an initial pool of 1272 reports. They were evaluated according to treatment protocol: 1) single-dose SRS (13 studies, 483 patients), 2) combination of MS and SRS (7 studies, 182 patients), and 3) fractionated SRS (3 studies, 82 patients). Tumor control was achieved in 89%, 94%, and 91% of patients, respectively. Odds ratios (ORs) of post- over pretreatment serviceable hearing were 0.42 ( < .01), 0.47 ( = .05), and 0.60 ( = .22); for facial nerve impairment, these ORs were 1.08 ( = .69), 3.45 ( = .28), and 0.87 ( = .71), respectively.
CONCLUSIONS
The management of large VS remains challenging. All treatment modalities resulted in high tumor control rates and worsening of pretreatment hearing. None, however, caused significant facial nerve impairment, suggesting that management strategies incorporating focal irradiation can be successful.
PubMed: 34277019
DOI: 10.1093/nop/npab011 -
The Laryngoscope Sep 2021Cerebellopontine angle (CPA) and internal auditory canal (IAC) lipomas are rare, benign tumors comprising 0.08% of all intracranial tumors and can be mistaken for other,... (Comparative Study)
Comparative Study
OBJECTIVES/HYPOTHESIS
Cerebellopontine angle (CPA) and internal auditory canal (IAC) lipomas are rare, benign tumors comprising 0.08% of all intracranial tumors and can be mistaken for other, more common lesions of the CPA/IAC such as vestibular schwannoma. The purpose of this study was to review the literature and assess the evolution of CPA/IAC lipoma diagnosis and management. In addition, we present 17 new lipomas, matching the largest known case series of this rare tumor.
STUDY DESIGN
Retrospective case series and systematic review.
METHODS
Systematic review of the literature was performed using PubMed and Google Scholar. References from identified articles were also reviewed to identify potential additional manuscripts. Manuscripts and abstracts were reviewed to identify unique cases. For the case series, the charts of all CPA/IAC lipoma patients seen at a single institution from 2006-2019 were manually reviewed. Logistic regression and chi-squared analysis were performed where appropriate.
RESULTS
A total of 219 unique lipomas have been reported in the literature, including 17 presented in this study. Surgical management has been performed in 46% of cases and has been conducted less often in recent decades, likely due to improved radiographic diagnostic capabilities and understanding of surgical outcomes. Surgical management is associated with worse neurologic outcomes (P = .002) and has become less common in recent decades. Although growth is unlikely, it has been demonstrated in patients into their 30s.
CONCLUSIONS
Accurate radiographic diagnosis is imperative for appropriate patient management, as CPA/IAC lipomas should typically be managed through observation and serial imaging whereas vestibular schwannomas and other CPA/IAC lesions may require microsurgical or radiosurgical intervention depending on growth and symptomatology. Laryngoscope, 131:2081-2087, 2021.
Topics: Adolescent; Adult; Aged; Brain Neoplasms; Cerebellopontine Angle; Child; Child, Preschool; Diagnosis, Differential; Disease Management; Ear Canal; Female; Humans; Lipoma; Logistic Models; Magnetic Resonance Imaging; Male; Meniere Disease; Microsurgery; Middle Aged; Neuroma, Acoustic; Outcome Assessment, Health Care; Radiography; Radiosurgery; Retrospective Studies; Tomography, X-Ray Computed; Young Adult
PubMed: 33567134
DOI: 10.1002/lary.29434 -
World Neurosurgery Feb 2023Tympanojugular paragangliomas (TJPs) are slow-growing tumors arising within the middle ear or jugular foramen. The development of modified skull base approaches and the... (Meta-Analysis)
Meta-Analysis Review
OBJECTIVE
Tympanojugular paragangliomas (TJPs) are slow-growing tumors arising within the middle ear or jugular foramen. The development of modified skull base approaches and the increasing use of stereotactic radiosurgery have provided more modern techniques in the management of TJPs. Several factors dictating approach selection, and resulting clinical outcomes have been inconsistently described.
METHODS
A systematic review of the literature describing modern management of complex TJPs was performed and summarized. A random-effects meta-analysis was performed to describe the rate of tumor control, complications, and symptom improvement in patients undergoing radiosurgery or surgical resection.
RESULTS
Nineteen studies were identified with a total of 852 TJP patients. A minority (153 patients) underwent radiosurgery while 699 underwent surgery. On meta-analysis, there was a 3.5% (95% confidence interval [CI]: 0.5%-6.4%) tumor growth rate following radiosurgery and 3.9% (95% confidence interval [CI]: 1.8%-6.0%) recurrence rate in surgical resection, with no significant moderator effect between the 2 groups (P = 0.9046). Complication rate for radiosurgery was 7.6% (95% CI: 2.8%-12.4%), differing significantly from surgical complication rates of 29.6% (95% CI: 17.1-42.0%, P = 0.0418).
CONCLUSIONS
Stereotactic radiosurgery and surgical resection for TJPs have similar rates of tumor recurrence. Radiation is associated with less risk and lower morbidity, yet there is comparably modest reduction of the tumor size. In sum, the data suggest that radiosurgery is a reasonable management option for patients with minimal symptoms who are high risk for surgery. Microsurgical resection should be reserved for patients with lower cranial neuropathies or those who have failed radiation treatment.
Topics: Humans; Glomus Jugulare Tumor; Neoplasm Recurrence, Local; Paraganglioma; Cranial Nerve Diseases; Radiosurgery; Treatment Outcome; Retrospective Studies
PubMed: 36400356
DOI: 10.1016/j.wneu.2022.11.037 -
Lung Cancer (Amsterdam, Netherlands) Aug 2023Stereotactic body radiation therapy (SBRT) is the standard of care for inoperable early stage non-small cell lung cancer (NSCLC). Use of image guided thermal ablation... (Meta-Analysis)
Meta-Analysis Review
RATIONALE
Stereotactic body radiation therapy (SBRT) is the standard of care for inoperable early stage non-small cell lung cancer (NSCLC). Use of image guided thermal ablation (IGTA; including microwave ablation [MWA] and radiofrequency ablation [RFA]) has increased in NSCLC, however there are no studies comparing all three.
OBJECTIVE
To compare the efficacy of IGTA (including MWA and RFA) and SBRT for the treatment of NSCLC.
METHODS
Published literature databases were systematically searched for studies assessing MWA, RFA, or SBRT. Local tumor progression (LTP), disease-free survival (DFS), and overall survival (OS) were assessed with single-arm pooled analyses and meta-regressions in NSCLC patients and a stage IA subgroup. Study quality was assessed with a modified methodological index for non-randomized studies (MINORS) tool.
RESULTS
Forty IGTA study-arms (2,691 patients) and 215 SBRT study-arms (54,789 patients) were identified. LTP was lowest after SBRT at one and two years in single-arm pooled analyses (4% and 9% vs. 11% and 18%) and at one year in meta-regressions when compared to IGTA (OR = 0.2, 95%CI = 0.07-0.63). MWA patients had the highest DFS of all treatments in single-arm pooled analyses. In meta-regressions at two and three-years, DFS was significantly lower for RFA compared to MWA (OR = 0.26, 95%CI = 0.12-0.58; OR = 0.33, 95%CI = 0.16-0.66, respectively). OS was similar across modalities, timepoints, and analyses. Older age, male patients, larger tumors, retrospective studies, and non-Asian study region were also predictors of worse clinical outcomes. In high-quality studies (MINORS score ≥ 7), MWA patients had better clinical outcomes than the overall analysis. Stage IA MWA patients had lower LTP, higher OS, and generally lower DFS, compared to the main analysis of all NSCLC patients.
CONCLUSIONS
NSCLC patients had comparable outcomes after SBRT and MWA, which were better than those with RFA.
Topics: Humans; Male; Carcinoma, Non-Small-Cell Lung; Retrospective Studies; Treatment Outcome; Radiosurgery; Lung Neoplasms; Catheter Ablation; Liver Neoplasms
PubMed: 37321074
DOI: 10.1016/j.lungcan.2023.107259 -
World Neurosurgery Oct 2023To compare fractionated stereotactic radiotherapy (FSRT) with staged stereotactic radiosurgery (SSRS) in patients with brain metastases >2 cm without prior whole brain... (Review)
Review
Comparison of Staged Stereotactic Radiosurgery and Fractionated Stereotactic Radiotherapy in Patients with Brain Metastases > 2 cm without Prior Whole Brain Radiotherapy: A Systematic Review and Meta-Analysis.
OBJECTIVE
To compare fractionated stereotactic radiotherapy (FSRT) with staged stereotactic radiosurgery (SSRS) in patients with brain metastases >2 cm without prior whole brain radiotherapy.
METHODS
In this systematic review and meta-analysis, PubMed, Scopus, Web of Science, Embase, and Cochrane were searched to include studies that evaluated FSRT and/or SSRS for brain metastases >2 cm or 4 cm in adult patients with a known primary malignancy and no prior history of whole brain radiotherapy. Preferred Reporting Items for Systematic Reviews and Meta-Analyses guidelines were followed and an indirect random-effect meta-analyses was conducted to compare treatment outcomes between the two modalities.
RESULTS
A total of 10 studies were included, comprising 612 (778 metastases) and 250 patients (265 metastases) in the SSRS and FSRT groups, respectively. The SSRS group had significantly older patients (66.6 ± 17.51 years vs. 62.37 ± 37.89 years; P = 0.029) with lower rate of control of primary disease (11.59% vs. 78.7%, P < 0.00001), and more patients with Karnofsky performance status ≥70 at baseline (92.81% vs. 88.56%; P = 0.045). FSRT was associated with a statistically nonsignificant but clinically important lower 12-month overall survival (44.75% [95% confidence interval [CI]: 30.48%-59.95%] vs. 53.25% [95%CI: 45.15%-61.19%], P = 0.1615) and higher rate of salvage radiotherapy (18.18% [95%CI: 8.75%-34%] vs. 12.27% [95%CI: 5.98%-23.53%], P = 0.0841). Both groups had comparable rates of local tumor control, mortality, tumor progression, recurrence, neurological death, and 6-month overall survival.
CONCLUSIONS
SSRS and FSRT were found to be comparable for treating brain metastases >2 cm not previously irradiated. Given the paucity of such studies, trials directly comparing the two treatment strategies are warranted to support these findings.
PubMed: 37543203
DOI: 10.1016/j.wneu.2023.07.143 -
Journal of Clinical Neuroscience :... Jul 2022Gamma knife radiosurgery (GKRS) has been deemed as the gold standard stereotactic radiosurgery (SRS) mode for the treatment of intracranial tumors, cerebrovascular... (Meta-Analysis)
Meta-Analysis Review
BACKGROUND
Gamma knife radiosurgery (GKRS) has been deemed as the gold standard stereotactic radiosurgery (SRS) mode for the treatment of intracranial tumors, cerebrovascular diseases and brain functional diseases. Our study was aimed to systematically evaluate the efficacy, safety, and complications of gamma knife radiosurgery for trigeminal schwannomas.
METHOD
We performed a systematic review and meta-analysis to analyze the clinical outcomes of patients with trigeminal schwannomas treated primarily or adjunctly with GKRS. We searched two databases, Pubmed and Embase, for studies published before January 1, 2021, using GKRS for trigeminal schwannomas. Studies reporting treatment of other schwannomas, or other forms of SRS such as linear accelerator and Cyberknife were excluded to reduce its heterogeneity.
RESULTS
Our search achieved 351 studies, of which 35 were assessed for full-text eligibility. 19 studies were included in the meta-analysis. 456 of 504 patients (0.94, 95% CI 0.91-0.96, I = 3.02%, p < 0.01) from 18 studies had local control, and 278 of 489 patients (0.63, 95%CI 0.48-0.78, I = 88.75%, p < 0.01) from 16 studies experienced tumor regression or disappearance. 231 of 499 patients (0.50, 95% CI 0.37-0.62; I = 83.89%, P < 0.01) from 17 studies had clinical symptoms improved. There was no significant difference in tumor control between those treated with GKRS as either primary treatment or adjuvant to surgery(p = 0.390).
CONCLUSION
GKRS is an efficacious primary and adjuvant method of treating trigeminal schwannomas, with reliable tumor control rates. Randomized controlled trials are needed to further and comprehensively evaluate the benefit-to-risk ratio of gamma knife radiosurgery.
Topics: Cranial Nerve Neoplasms; Follow-Up Studies; Humans; Neurilemmoma; Particle Accelerators; Radiosurgery; Retrospective Studies; Treatment Outcome
PubMed: 35561433
DOI: 10.1016/j.jocn.2022.05.001 -
Cureus Apr 2022Introduction The complex anatomy of the cavernous sinus confers a true challenge when it comes to safe tumor resection. Due to its non-invasive nature, stereotactic...
Introduction The complex anatomy of the cavernous sinus confers a true challenge when it comes to safe tumor resection. Due to its non-invasive nature, stereotactic radiosurgery (SRS) is expected to have lower mortality and morbidity rates than microsurgery. The purpose of this study was to evaluate clinical results regarding visual symptoms after SRS for benign tumors invading the cavernous sinus. We also conducted a systematic literature review to provide a robust analysis regarding visual outcomes. Methods The study included 58 patients (43 women and 15 men; mean age: 52 years) with benign tumors invading the cavernous sinus (27 pituitary adenomas and 31 meningiomas) who underwent SRS with different platforms between August 2011 and December 2021. Of these, 26 patients underwent surgery before SRS, and the remaining 32 had SRS as first-line therapy. We identified symptoms involving cranial nerves (CN) II, III, IV, and VI in 38 patients at the time of SRS. We conducted a systematic review to identify all original studies assessing visual outcomes. We searched PubMed, the Latin American and Caribbean Health Sciences Literature index, and Google Scholar using the Medical Subject Heading search terms "radiosurgery" and "cavernous sinus" for valid studies published until January 31, 2022. Results Regarding pituitary adenomas, median tumor volume was 2.05 cc, 3.12 cc, and 2.39 cc for Gamma Knife (GK), CyberKnife (CK), and tomotherapy (Tomo), respectively. Median doses were 14 Gy for GK, 17 Gy for CK, and 15 Gy for Tomo. For meningiomas, median tumor volume was 10.2 cc, 2.62 cc, and 16.3 cc for GK, CK, and Tomo, respectively. The median dose was 14 Gy for GK, 14 Gy for CK, and 14.5 Gy for Tomo. The overall tumor control rate was 100% with a median follow-up of 33 months (range: 6-128 months). A reduction of >30% in total tumor size per the Response Evaluation Criteria in Solid Tumors (RECIST) classification was documented in seven patients (RECIST II; 12.1%), 51 patients (87.9%) had stable disease (RECIST III), and no increase in tumor volume was documented in any patient. Visual symptoms improved in 51.7% of patients. In the systematic review, the mean visual improvement was 36% (range: 25.8-42.5%). Conclusion SRS is an effective treatment for benign tumors invading the cavernous sinus. In this series, patients who underwent SRS as a primary treatment showed improvement in pre-existing cranial neuropathy and visual symptoms. Given the natural history of these tumors, which tend to grow and cause visual alternations, treating asymptomatic patients is a feasible approach worth considering for the appropriate patients.
PubMed: 35530870
DOI: 10.7759/cureus.23928 -
Journal of Neuro-oncology Oct 2023To perform a systematic review of literature specific to single-fraction stereotactic radiosurgery (SRS) for large vestibular schwannomas (VS), maximum... (Review)
Review
Single-fraction radiosurgery outcomes for large vestibular schwannomas in the upfront or post-surgical setting: a systematic review and International Stereotactic Radiosurgery Society (ISRS) Practice Guidelines.
PURPOSE
To perform a systematic review of literature specific to single-fraction stereotactic radiosurgery (SRS) for large vestibular schwannomas (VS), maximum diameter ≥ 2.5 cm and/or classified as Koos Grade IV, and to present consensus recommendations on behalf of the International Stereotactic Radiosurgery Society (ISRS).
METHODS
The Medline and Embase databases were used to apply the Preferred Reporting Items for Systematic Reviews and Meta-Analyses (PRISMA) approach. We considered eligible prospective and retrospective studies, written in the English language, reporting treatment outcomes for large VS; SRS for large post-operative tumors were analyzed in aggregate and separately.
RESULTS
19 of the 229 studies initially identified met the final inclusion criteria. Overall crude rate of tumor control was 89% (93.7% with no prior surgery vs 87.7% with prior surgery). Rates of salvage microsurgical resection, need for shunt, and additional SRS in all series versus those with no prior surgery were 9.6% vs 3.3%, 4.7% vs 6.4% and 1% vs 0.9%, respectively. Rates of facial palsy and hearing preservation in all series versus those with no prior surgery were 1.3% vs 3.4% and 34.2% vs 40.4%, respectively.
CONCLUSIONS
Upfront SRS resulted in high rates of tumor control with acceptable rates of facial palsy and hearing preservation as compared to the results in those series including patients with prior surgery (level C evidence). Therefore, although large VS are considered classic indication for microsurgical resection, upfront SRS can be considered in selected patients and we recommend a prescribed marginal dose from 11 to 13 Gy (level C evidence).
Topics: Humans; Radiosurgery; Retrospective Studies; Neuroma, Acoustic; Prospective Studies; Facial Paralysis; Treatment Outcome; Follow-Up Studies
PubMed: 37843727
DOI: 10.1007/s11060-023-04455-8