Return of the lesion: a meta-analysis of 1134 angiographically cured pediatric arteriovenous malformations

2021 ◽  
pp. 1-8
Author(s):  
David C. Lauzier ◽  
Ananth K. Vellimana ◽  
Arindam R. Chatterjee ◽  
Joshua W. Osbun ◽  
Christopher J. Moran ◽  
...  
Keyword(s):  
Arteriovenous Malformations ◽  
Pediatric Patients ◽  
Web Of Science ◽  
Meta Analysis ◽  
Study Cohort ◽  
Fisher Exact Test ◽  
Exact Test ◽  
Risk Of Rupture ◽  
Brain Avms ◽  
Prospective Database

OBJECTIVE Brain arteriovenous malformations (AVMs) carry a risk of rupture and subsequent morbidity or mortality unless fully treated. AVMs in pediatric patients are known to occasionally recur after obliteration. The objective of this study was to characterize the risk of AVM recurrence following angiographically confirmed obliteration in children. METHODS Consecutive pediatric AVMs treated at a single center were identified from a prospective database. Patients with angiographically confirmed AVM obliteration following treatment were included in this study. Associations between AVM recurrence and patient or procedural factors were characterized using the two-tailed Fisher exact test or Mann-Whitney U-test. A literature search was conducted using PubMed, Scopus, Embase, and the Clarivate Web of Science with defined search criteria, and eligible studies were included alongside this study cohort in a meta-analysis. Rates of AVM recurrence following obliteration were pooled across studies with a random-effects model and reported with 95% confidence intervals (CIs). RESULTS Recurrence after angiographic confirmation of AVM obliteration was observed in 10.4% (7/67) of pediatric AVMs treated at the authors’ center. Patients with recurrent AVMs were significantly younger than those without recurrence (p = 0.002). In the meta-analysis, which included 1134 patients across 24 studies, the rate of recurrence was 4.8% (95% CI 3.0%–6.7%). The rate of AVM recurrence following radiosurgery was 0.7% (95% CI 0%–1.6%), which was significantly lower than the 8.5% rate (95% CI 5.0%–12.0%) following microsurgery. CONCLUSIONS Recurrence of obliterated brain AVMs is common in children. Recurrence is more common in young children and following microsurgery.

scholarly journals The factors associated with hemorrhagic presentation in children with untreated brain arteriovenous malformation: a meta-analysis

2019 ◽  
Vol 23 (3) ◽  
pp. 343-354 ◽  
Author(s):  
Xiaolin Ai ◽  
Zengpanpan Ye ◽  
Jianguo Xu ◽  
Chao You ◽  
Yan Jiang
Keyword(s):  
Pediatric Patients ◽  
Meta Analysis ◽  
Predictive Ability ◽  
Venous Drainage ◽  
Brain Arteriovenous Malformation ◽  
Factors Associated ◽  
Risk Of Rupture ◽  
Brain Avms ◽  
Vein Stenosis ◽  
Deep Location

OBJECTIVERupture of arteriovenous malformations (AVMs) would result in high mortality and prevalence of disability in pediatric patients. Decisions regarding the treatment of AVMs need to weigh the risk of rupture over the course of their natural history against the possibility of creating a lesion during treatment. Multiple factors have been proposed to predict hemorrhagic presentation of pediatric patients with AVMs. The aim of this meta-analysis was to evaluate the predictors of hemorrhagic presentation in pediatric patients with AVMs.METHODSThe authors searched the PubMed and EMBASE databases. Studies reporting the predictors of hemorrhagic presentation in children with untreated brain AVMs were included. The predictive ability of identified predictors was assessed by odds ratios (ORs) and 95% confidence intervals (CIs).RESULTSA higher risk of hemorrhagic presentation was found in AVMs with smaller size (< 3 cm, OR 2.97, 95% CI 1.94–4.54, p < 0.00001), deep venous drainage (OR 2.28, 95% CI 1.55–3.36, p < 0.0001), a single draining vein (OR 2.23, 95% CI 1.27–3.92, p = 0.005), a single feeder (OR 3.72, 95% CI 1.31–10.62, p = 0.01), a deep location (OR 1.82, 95% CI 1.22–2.72, p = 0.004), an infratentorial location (OR 2.25, 95% CI 1.19–4.26, p = 0.01), and diffuse morphology (OR 8.94, 95% CI 3.01–26.55, p < 0.0001). In addition, the AVMs with draining vein ectasia (OR 0.35, 95% CI 0.13–0.97, p = 0.04) and high Spetzler-Martin (SM) grade (OR 0.53, 95% CI 0.36–0.78, p = 0.001) had a lower risk of hemorrhagic presentation in pediatric patients.CONCLUSIONSSmaller AVMs, deep venous drainage, a single draining vein, a single feeder, deep/infratentorial location, diffuse morphology, and high SM grade were identified as positive predictors for hemorrhagic presentation. Particularly, patients with diffuse AVMs have a higher risk of hemorrhagic presentation than other factors and may need active treatments. However, factors such as age, sex, draining vein stenosis, and associated aneurysms were not associated with hemorrhagic presentation.

scholarly journals Prevalence and characteristics of brain arteriovenous malformations in hereditary hemorrhagic telangiectasia: a systematic review and meta-analysis

2017 ◽  
Vol 127 (2) ◽  
pp. 302-310 ◽  
Author(s):  
Waleed Brinjikji ◽  
Vivek N. Iyer ◽  
Christopher P. Wood ◽  
Giuseppe Lanzino
Keyword(s):  
Systematic Review ◽  
Hereditary Hemorrhagic Telangiectasia ◽  
Arteriovenous Malformations ◽  
Clinical Presentation ◽  
Meta Analysis ◽  
Prevalence Rates ◽  
Brain Avm ◽  
Martin Grade ◽  
Cerebral Avms ◽  
Brain Avms

OBJECTIVEPatients with hereditary hemorrhagic telangiectasia (HHT) are known to suffer from high rates of cerebral arteriovenous malformations (AVMs). The authors performed a systematic review and meta-analysis of the literature examining prevalence rates, characteristics, and clinical presentation of cerebral AVMs in the HHT population.METHODSTo identify studies on AVM prevalence and characteristics in the HHT population, 4 databases (MEDLINE, EMBASE, Scopus and Web of Science) were searched by a reference librarian with over 30 years experience in systematic reviews and meta-analysis. The search period was January 1, 1990–March 2016. The following search terms were used: hereditary hemorrhagic telangiectasia, Osler-Weber-Rendu syndrome, AVM, brain AVM, arteriovenous malformation, arteriovenous fistula, prevalence, and epidemiology. The authors identified studies that examined the prevalence rates, characteristics, and clinical presentation of cerebral AVMs in patients with HHT. They assessed overall AVM prevalence rates as well as prevalence rates by age, sex, HHT type, and country/region. They also systematically reviewed the characteristics of AVMs, including rupture status, location, clinical presentation, angioarchitecture, and Spetzler-Martin grade. Data were analyzed using a random-effects meta-analysis model.RESULTSThirty-nine studies were included in this meta-analysis. Thirty studies examined brain AVM prevalence rates in various HHT patient populations, and 18 studies examined AVM clinical and angiographic characteristics (9 studies examined both prevalence rates and AVM characteristics). The prevalence of brain AVMs in HHT patients was 10.4% (95% CI 7.9%–13.0%) with no significant difference between males (8.5%, 95% CI 4.9%–12.0%) and females (11.0%, 95% CI 5.9%–16.1%). Patients with HHT Type 1 (HHT1) had a significantly higher brain AVM prevalence (13.4%, 95% CI 9.5%–17.4%) compared with those with HHT Type 2 (HHT2) (2.4%, 95% CI 1.0%–3.8%) (p < 0.0001). In 55.2% (95% CI 38.3%–72.1%) of cases, the AVMs were symptomatic. Spetzler-Martin grade was 2 or less in 86.9% (95% CI 67.5%–95.2%) of patients.CONCLUSIONSThe prevalence of brain AVMs in the HHT population is about 10%. HHT1 patients are significantly more likely to have brain AVMs than HHT2 patients. Most AVMs in the HHT population are symptomatic. The Spetzler-Martin grade for these lesions is 2 or less in nearly 90% of patients.

scholarly journals Nearly Half of TP53 Germline Variants Predicted To Be Pathogenic in Patients With Osteosarcoma Are De Novo: A Report From the Children’s Oncology Group

2020 ◽  
pp. 1187-1195
Author(s):  
Brandon J. Diessner ◽  
Nathan Pankratz ◽  
Anthony J. Hooten ◽  
Lisa Mirabello ◽  
Aaron L. Sarver ◽  
...  
Keyword(s):  
Pediatric Patients ◽  
Rare Variants ◽  
De Novo ◽  
Phase 1 ◽  
Control Measures ◽  
Fisher Exact Test ◽  
Exact Test ◽  
Whole Exome ◽  
History Of ◽  
De Novo Variant

PURPOSE To ascertain the prevalence of recurrent de novo variants among 240 pediatric patients with osteosarcoma (OS; age < 20 years) unselected for family history of cancer. METHODS The identification of de novo variants was implemented in 2 phases. In the first, we identified genes with a rare (minor allele frequency < 0.01) de novo variant in > 1 of the 95 case-parent trios examined by whole-exome sequencing (WES) who passed quality control measures. In phase 2, 145 additional patients with OS were evaluated by targeted sequencing to identify rare de novo variants in genes nominated from phase 1. Recurrent rare variants identified from phase 1 and 2 were verified as either de novo or inherited by Sanger sequencing of affected patients and their parents. Categorical and continuous data were analyzed using Fisher exact test and t tests, respectively. RESULTS Among 95 case-parent trios who underwent WES, we observed 61 de novo variants in 60 genes among 47 patients, with TP53 identified as the only gene with a pathogenic or likely pathogenic (P/LP) de novo variant in more than one case-parent trio. Among all 240 patients with OS, 13 (5.4%) harbored a P/LP TP53 germline variant, of which 6 (46.2%) were confirmed to be de novo. CONCLUSION Apart from TP53, we did not observe any other recurrent de novo P/LP variants in the case-parent trios, suggesting that new mutations in other genes are not a frequent cause of pediatric OS. That nearly half of P/LP TP53 variants in our sample were de novo suggests universal screening for germline TP53 P/LP variants among pediatric patients with OS should be considered.

scholarly journals Diagnostic Test Accuracy of New Generation Tympanic Thermometry in Children: A Systematic Review and Meta-analysis

2020 ◽  
Author(s):  
Dan Shi ◽  
Liyuan Zhang ◽  
Haixia Li
Keyword(s):  
Pediatric Patients ◽  
Web Of Science ◽  
Meta Analysis ◽  
Internal Validity ◽  
Youden Index ◽  
Test Accuracy ◽  
Optimal Cutoff ◽  
High Diagnostic Accuracy ◽  
New Generation ◽  
Review Manager

Abstract Purposes: The purposes of this review were to compare sensitivity and specificity of the new generation tympanic thermometry under different cutoffs and to give the optimal cutoff. Methods: Articles were derived from a systematic search in PubMed, Web of science, and Embase, and were assessed for internal validity by QUADAS-Ⅱ. The figure of risk of bias was drew by Review Manager 5.3 and data were synthesized by MetaDisc 1.4. Results: Nineteen diagnostic studies, involving 4,639 pediatric patients, were included. The cut-offs varied from 37.0℃ to 38.0℃ among these studies. The cut-off 37.8 was with the highest sROC AUC (0.97) and Youden Index (0.83) and was deemed to be the optimal cutoff. Conclusion: New Generation Tympanic Thermometry is with high diagnostic accuracy in pediatric patients and can be an alternative for rectal measurements.

scholarly journals International multicenter cohort study of pediatric brain arteriovenous malformations. Part 1: Predictors of hemorrhagic presentation

2017 ◽  
Vol 19 (2) ◽  
pp. 127-135 ◽  
Author(s):  
Dale Ding ◽  
Robert M. Starke ◽  
Hideyuki Kano ◽  
David Mathieu ◽  
Paul P. Huang ◽  
...  
Keyword(s):  
Cohort Study ◽  
Arteriovenous Malformations ◽  
Pediatric Patients ◽  
Management Strategies ◽  
Multivariate Logistic Regression Analysis ◽  
Venous Drainage ◽  
Study Cohort ◽  
Multicenter Cohort Study ◽  
Brain Arteriovenous Malformations ◽  
Occipital Lobes

OBJECTIVE Brain arteriovenous malformations (AVMs) are the most common cause of spontaneous intracranial hemorrhage in pediatric patients (age < 18 years). Since the cumulative lifetime risk of AVM hemorrhage is considerable in children, an improved understanding of the risk factors influencing hemorrhagic presentation may aid in the management of pediatric AVMs. The aims of this first of a 2-part multicenter, retrospective cohort study are to evaluate the incidence and determine the predictors of hemorrhagic presentation in pediatric AVM patients. METHODS The authors analyzed pooled AVM radiosurgery data from 7 institutions participating in the International Gamma Knife Research Foundation (IGKRF). Patients younger than 18 years at the time of radiosurgery and who had at least 12 months of follow-up were included in the study cohort. Patient and AVM characteristics were compared between unruptured and ruptured pediatric AVMs. RESULTS A total of 357 pediatric patients were eligible for analysis, including 112 patients in the unruptured and 245 patients in the ruptured AVM cohorts (69% incidence of hemorrhagic presentation). The annual hemorrhage rate prior to radiosurgery was 6.3%. Hemorrhagic presentation was significantly more common in deep locations (basal ganglia, thalamus, and brainstem) than in cortical locations (frontal, temporal, parietal, and occipital lobes) (76% vs 62%, p = 0.006). Among the factors found to be significantly associated with hemorrhagic presentation in the multivariate logistic regression analysis, deep venous drainage (OR 3.2, p < 0.001) was the strongest independent predictor, followed by female sex (OR 1.7, p = 0.042) and smaller AVM volume (OR 1.1, p < 0.001). CONCLUSIONS Unruptured and ruptured pediatric AVMs have significantly different patient and nidal features. Pediatric AVM patients who possess 1 or more of these high-risk features may be candidates for relatively more aggressive management strategies.

scholarly journals Diagnostic Test Accuracy of New Generation Tympanic Thermometry in Children under Different Cutoffs: A Systematic Review and Meta-analysis

2020 ◽  
Author(s):  
Dan Shi ◽  
Liyuan Zhang ◽  
Haixia Li
Keyword(s):  
Diagnostic Accuracy ◽  
Pediatric Patients ◽  
Web Of Science ◽  
Meta Analysis ◽  
Internal Validity ◽  
Youden Index ◽  
Test Accuracy ◽  
Optimal Cutoff ◽  
New Generation ◽  
Review Manager

Abstract Purposes: To identify and quantify studies reporting the diagnostic accuracy of the new generation of infrared tympanic thermometer (IRTT) in children and to compare the sensitivity and specificity of IRTT under different cutoffs and give the optimal cutoff. Methods: Articles were derived from a systematic search in PubMed, Web of Science Core Collection, and Embase, and were assessed for internal validity by the Quality Assessment of Diagnostic Accuracy Studies-2　（QUADAS-Ⅱ）. The figure of risk of bias was created by Review Manager 5.3 and data were synthesized by MetaDisc 1.4. Results: Nineteen diagnostic studies, involving 4,639 pediatric patients, were included. The cut-offs varied from 37.0℃ to 38.0℃ among these studies. The cut-off 37.8℃ was with the highest sROC AUC (0.97) and Youden Index (0.83) and was deemed to be the optimal cutoff. Conclusion: The optimal cutoff for infrared tympanic thermometers is 37.8℃. New Generation Tympanic Thermometry is with high diagnostic accuracy in pediatric patients and can be an alternative for fever screening in children.

109 Vascular Composite Allotransplantation in Burn Reconstruction: Systematic Review and Meta-analysis

2020 ◽  
Vol 41 (Supplement_1) ◽  
pp. S72-S73
Author(s):  
Joshua Peterson ◽  
Pablo Padilla ◽  
Kelsey Gray ◽  
Ramón Zapata-Sirvent ◽  
William Norbury ◽  
...  
Keyword(s):  
Risk Factors ◽  
Systematic Review ◽  
Upper Extremity ◽  
Meta Analysis ◽  
Senior Author ◽  
Fisher Exact Test ◽  
Burn Patients ◽  
Exact Test ◽  
Relative Safety ◽  
Significant Difference

Abstract Introduction The first vascular composite allotransplantation (VCA) was performed in 1998, with the first post-burn VCA in 2003. Since then, 21 burn patients have been recipients of VCAs. Controversy exists regarding safety of VCA in burn patients due to high levels of immunologic sensitization attributed to use of cadaver allograft. We seek to understand unique considerations of VCA in burn reconstruction and its relative safety compared to VCA in non-burn reconstruction. Methods A systematic review of VCA in burn reconstruction was conducted using the PubMed, Media and EBSCO database to retrieve all articles and news stories studying VCA from Jan. 2000 to May 2019. Only articles containing composite tissue allotransplantation, VCA, and reconstructive surgery were included. Articles that did not report outcomes or patient information were excluded. Reported cases were grouped according to injury and tissue (face vs. upper extremity) with mortality as a dependent variable. Statistical analysis was performed with the Fisher exact test. Results Of 63 articles obtained, 21 met criteria. After secondary review by the senior author, 6 more articles were included. To date, 147 VCAs are reported in 112 patients, including 40 face VCAs and 107 upper extremity VCAs. Of these, 30 (20%) were performed for post-burn deformities, and 117 (80%) for non-burn deformities. A total of 21 post-burn patients received VCAs with 4 subsequent deaths (19%). Post-burn recipients of upper extremity VCAs had significantly higher mortality than non-burn recipients of the same (p = 0.01), but there was no significant difference in face VCA mortality (p = 1.0) or overall mortality (p = 0.06) between groups. Conclusions VCA is a forefront procedure employed worldwide for reconstruction of devastating injuries. Presently, burn patients are at greater risk of mortality after upper extremity VCA, but not face VCA. Burn patients will benefit from focused investigation of unique post-burn risk factors of VCA mortality. Applicability of Research to Practice Disparities between post-burn and non-burn VCA outcomes suggest the presence of unique risk factors in burn patients. Investigation of unique risk factors will improve safety of VCA for burn patients.

Do the morphological characteristics of arteriovenous malformations affect the results of radiosurgery?

2004 ◽  
Vol 101 (3) ◽  
pp. 393-401 ◽  
Author(s):  
Gregory J. Zipfel ◽  
Patrick Bradshaw ◽  
Frank J. Bova ◽  
William A. Friedman
Keyword(s):  
Strong Correlation ◽  
Arteriovenous Malformations ◽  
Ct Scanning ◽  
Morphological Features ◽  
Fisher Exact Test ◽  
Repeated Treatment ◽  
Content Type ◽  
Exact Test ◽  
Presenting Symptoms ◽  
Fine Print

Object. The authors sought to determine which morphological features of arteriovenous malformations (AVMs) are statistically predictive of preradiosurgical hemorrhage, postradiosurgical hemorrhage, and neuroimaging-defined failure of radiosurgical treatment. In addition, correlation between computerized tomography (CT) scanning and angiography for the identification of AVM structures was investigated. Methods. Archived CT dosimetry and available angiographic and clinical data for 268 patients in whom AVMs were treated with linear accelerator radiosurgery were retrospectively reviewed. Many of the morphological features of AVMs, including location, volume, compact or diffuse nidus, neovascularity, ease of nidus identification, number of feeding arteries, location (deep or superficial) of feeding arteries, number of draining veins, deep or superficial venous drainage, venous stenoses, venous ectasias, and the presence of intranidal aneurysms, were analyzed. In addition, a number of patient and treatment factors, including patient age, presenting symptoms, radiation dose, repeated treatment, and radiological outcome, were subjected to multivariate analyses. Two hundred twenty-seven patients were treated with radiosurgery for the first time and 41 patients underwent repeated radiosurgery. Eighty-one patients presented with a history of AVM hemorrhage and 91 patients had AVMs in a periventricular location. Twenty-six patients (10%) experienced a hemorrhage following radiosurgery. Of the 268 patients, 81 (30%) experienced angiographically defined cures, and 37 (14%) experienced MR imaging-defined cures. Eighty-six patients (32%) experienced neuroimaging-defined treatment failure, and 64 underwent insufficiently long follow up. A larger AVM volume (odds ratio [OR] 0.349; p = 0.004) was associated with a decreased rate of pretreatment hemorrhage, whereas periventricular location (OR 6.358; p = 0.000) was associated with an increased rate of pretreatment hemorrhage. None of the analyzed factors was predictive of hemorrhage following radiosurgery. A higher radiosurgical dose was strongly correlated with neuroimaging-defined success (OR 3.743; p = 0.006), whereas a diffuse nidus structure (OR 0.246; p = 0.008) and associated neovascularity (OR 0.428; p = 0.048) were each associated with a lower neuroimaging-defined cure rate. A strong correlation between CT scanning and angiography was noted for both nidus structure (p = 0.000; Fisher exact test) and neovascularity (p = 0.002; Fisher exact test). Conclusions. Patients presenting with AVMs that are small or periventricular were at higher risk for experiencing hemorrhage. A higher radiosurgical dose correlated strongly with neuroimaging-defined success. Patients in whom the AVM had a diffuse structure or associated neovascularity were at higher risk for neuroimaging-defined failure of radiosurgery. A strong correlation between CT scanning and angiography in the assessment of AVM structure was demonstrated.

scholarly journals ASSOCIATION BETWEEN NUTRITIONAL STATUS AND THE OUTCOME OF PEDIATRIC PATIENT WITH DENGUE SHOCK SYNDROME

2020 ◽  
Vol 30 (1) ◽  
pp. 1
Author(s):  
Shidi Laras Pramudito ◽  
Dewi Ratna Sari ◽  
Ninik Asmaningsih Soemyarso
Keyword(s):  
Nutritional Status ◽  
General Hospital ◽  
Pediatric Patients ◽  
Dengue Infection ◽  
Secondary Data ◽  
Dengue Shock Syndrome ◽  
Observational Research ◽  
Fisher Exact Test ◽  
Chi Square ◽  
Exact Test

Background: The association between nutritional status and dengue infection is still considered controversial. Researches that study obesity and shock in pediatric patients with dengue hemmorhagic fever (DHF) has been widely carried out. However, researches that study the association between obesity and the outcome of patients with dengue shock syndrome (DSS) are still rarely done. Objective: The objective is to study the association of obesity in body mass index-for-age (BMI-for-age), gender, and age to the outcome of patients with dengue shock syndrome (recurrent shock or death). Material and method: The research was analytical retrospective observational research with a case-control design using secondary data from the medical records of pediatric patients with DSS in Dr. Soetomo General Hospital during 2015. The analyses used to test the association of obesity (BMI-for-age), gender, age and the outcome of patients with dengue shock syndrome (recurrent shock or death) were the Chi Square or Fisher Exact test. Result: The sample acquired was 60 data, consisting of 46 patients with DSS without recurrent shock or death and 14 pediatric patients with DSS experiencing recurrent shock or mortality. The frequency of boys and girls were found in equal numbers. Age group >5-10 years was found more common (45%). The results of the analyses test found no asssiation between gender (p=0.542), age (p=0.314), and obesity (p=1,000) and the outcome of pediatric patients with dengue shock syndrome. Conclusion: There were no association found between obesity, age, and gender to the outcomes of pediatric patients with DSS at Dr. Soetomo General Hospital.

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