Cavernous Malformations of the Optic Nerve and Optic Pathway: A Case Series and Systematic Review of the Literature

2021 ◽  
Author(s):  
Visish M Srinivasan ◽  
Stefan W Koester ◽  
Michele S Wang ◽  
Redi Rahmani ◽  
Kevin L Ma ◽  
...  
Keyword(s):  
Systematic Review ◽  
Optic Nerve ◽  
Single Case ◽  
Case Reports ◽  
Case Series ◽  
Senior Author ◽  
Optic Pathway ◽  
Review Of The Literature ◽  
Cavernous Malformations ◽  
Single Center Experience

Abstract BACKGROUND Although rare, cavernous malformations (CMs) of the optic nerve and anterior optic pathway (optic pathway cavernous malformations [OPCMs]) can occur, as described in several single case reports in the literature. OBJECTIVE To describe the technical aspects of microsurgical management of CMs of the optic pathway on the basis of an extensive single-center experience and review of the literature. METHODS A systematic literature review was performed to augment an earlier review, using PRISMA (Preferred Reporting Items for Systematic Reviews and Meta-Analyses) guidelines. In addition, an institutional database was searched for all patients undergoing surgical resection of OPCMs. Patient information, surgical technique, and clinical and radiographic outcomes were assessed. RESULTS Since the previous report, 14 CMs were resected at this institution or by the senior author at another institution. In addition, 34 cases were identified in the literature since the systematic review in 2015, including some earlier cases that were not discussed in the previous report. Most OPCMs were resected via pterional, orbital-pterional, and orbitozygomatic craniotomies. Visual outcomes were similar to those in earlier reports, with 70% of patients reporting stable to normal vision postoperatively. CONCLUSION OPCMs can occur throughout the anterior visual pathway and may cause significant symptoms. Surgery is feasible and should be considered for OPCMs presenting to a surface of the nerve. Favorable results can be obtained with resection, although optimal results are obtained with patients who present with milder symptoms without longstanding damage to the optic apparatus.

Thrombotic microangiopathy in the course of catastrophic antiphospholipid syndrome successfully treated with eculizumab: case report and systematic review of the literature

Lupus ◽  
2020 ◽  
Vol 29 (6) ◽  
pp. 631-639 ◽  
Author(s):  
Marta Skoczynska ◽  
Mark A Crowther ◽  
Malgorzata Chowaniec ◽  
Monika Ponikowska ◽  
Shruti Chaturvedi ◽  
...  
Keyword(s):  
Systematic Review ◽  
Case Report ◽  
Antiphospholipid Syndrome ◽  
Lupus Erythematosus ◽  
Single Case ◽  
Case Reports ◽  
Review Of The Literature ◽  
Catastrophic Antiphospholipid Syndrome ◽  
Total Remission

Objective The purpose of this study was to characterize the role of eculizumab, a monoclonal antibody against the terminal complement component C5, in patients with catastrophic antiphospholipid syndrome (CAPS). Methods We present a case report of a patient with systemic lupus erythematosus (SLE) and CAPS treated with eculizumab, as well as results of a systematic review of the literature. Results Including our patient, we identified 11 case reports of patients with CAPS treated with eculizumab. All of them had partial or total remission of symptoms. Conclusion Data on eculizumab efficacy in CAPS are promising but are limited to single case reports. More studies are needed to develop evidence-based recommendations for eculizumab use in CAPS.

Diagnostic Criteria for Odontogenic Sinusitis: A Systematic Review

2020 ◽  
pp. 194589242097676
Author(s):  
Fabiana Allevi ◽  
Gian Luca Fadda ◽  
Cecilia Rosso ◽  
Federica Martino ◽  
Carlotta Pipolo ◽  
...  
Keyword(s):  
Systematic Review ◽  
Quality Assessment ◽  
Diagnostic Criteria ◽  
Single Case ◽  
Case Reports ◽  
Significant Proportion ◽  
Case Series ◽  
Assessment Tools ◽  
Cochrane Library ◽  
Odontogenic Sinusitis

Background Odontogenic sinusitis affects a significant proportion of patients with paranasal sinus infections. Nevertheless, no shared diagnostic criteria for this condition have yet been implemented and published studies differ in their definition of the disease. Objective The present systematic review of the literature was undertaken to characterize and analyze the different diagnostic criteria currently employed for odontogenic sinusitis. Methods Systematic searches for studies published between 2009 and 2019 were performed in Medline, Embase, Web of Science, Cochrane Library, and ClinicalTrials.gov databases. Search criteria were designed to identify all studies focusing, even partially, on odontogenic sinusitis. Human original studies except single case reports published in the English, French, German, Spanish, or Italian language were included. We removed duplicate abstracts and conducted full-text reads, data extraction, and quality assessment procedures (using the Oxford Centre for Evidence-based Medicine levels of evidence and National Heart Lung and Blood Institute Study Quality Assessment Tools). We reviewed articles for diagnostic criteria, both in terms of definition and etiology identification. Results Among 1,000 unique citations, 63 studies were deemed eligible. Most articles (n = 45) were retrospective case series; a single randomized clinical trial was available. Only 49 studies reported diagnostic criteria, yet relied marginally on published guidelines (n = 10 articles) for identifying sinusitis, often choosing instead to develop their own clinical (n = 15 articles), endoscopic (n = 12 articles), and/or radiologic (n = 30 articles) criteria. For odontogenic focus identification, 14 papers required a multidisciplinary evaluation, 11 papers required a time relationship between dental procedures and sinusitis, 24 papers required oroscopy and/or dental evaluation, and 53 papers required computed tomography. Conclusions Current diagnostic criteria for odontogenic sinusitis are extremely heterogeneous. Establishing shared diagnostic criteria aimed at defining both sinusitis and related odontogenic foci would spur collaboration between investigators and support more comprehensive outcomes evaluations together with a better understanding of treatment options.

scholarly journals Othello syndrome in Parkinson’s disease: a systematic review and report of a case series

2021 ◽  
Author(s):  
Giovanna De Michele ◽  
Gianluigi Rosario Palmieri ◽  
Chiara Pane ◽  
Carmen Diletta Paola Dello Iacovo ◽  
Sandra Perillo ◽  
...  
Keyword(s):  
Systematic Review ◽  
Parkinson’S Disease ◽  
Parkinson's Disease ◽  
Single Case ◽  
Case Reports ◽  
Case Series ◽  
Visual Hallucinations ◽  
Levodopa Dose ◽  
Cross Sectional ◽  
Pubmed Database

Abstract Introduction Psychosis in Parkinson’s disease (PD) is common and consists of hallucinations, illusions, and delusions. Among the latter, delusional jealousy, also named Othello syndrome (OS), might impair the quality of life of both patients and their partners. We aimed to perform a systematic review and report a series of PD patients presenting with OS. Methods A systematic review research was performed in PubMed database, excluding non-English articles, single case reports, reviews and neuropathology articles, comments, and articles concerning OS associated with deep brain stimulation (DBS) and levodopa-carbidopa intestinal gel infusion. We also described eleven PD patients (9 M and 2 F) with OS, identified in a cohort of consecutive 153 patients, comparing them with eleven matched no OS (nOS) PD subjects taken from the same cohort. Results We included eight articles (four case series and four cross-sectional studies). OS resulted more common among males than females. We did not find higher levodopa dose and levodopa equivalent dose for dopamine agonists and for all anti-parkinsonian drugs in our OS group. In our case series, OS patients showed visual hallucinations (p=0.001) and a trend to have depression (p=0.080) more frequently than nOS ones. Conclusions OS is not a rare disorder in PD, probably due not only to abnormal dopaminergic stimulation but also to serotonergic dysfunction in biologically predisposed subjects. Visual hallucinations and other concomitant psychiatric diseases, in particular depression, might represent a risk factor for the OS development.

Post-Thrombotic Syndrome (PTS) in Children: A Systematic Review of Prevalence, Validity of Outcome Measures, and Prognostic Factors.

Blood ◽  
2009 ◽  
Vol 114 (22) ◽  
pp. 2978-2978 ◽  
Author(s):  
Neil A Goldenberg ◽  
Susan Kahn ◽  
Mark A. Crowther ◽  
Gili Kenet ◽  
Ulrike Nowak-Gottl ◽  
...  
Keyword(s):  
Systematic Review ◽  
Prognostic Factors ◽  
Cohort Studies ◽  
Conflicts Of Interest ◽  
Single Case ◽  
Case Reports ◽  
Case Series ◽  
Cross Sectional ◽  
Medline Search ◽  
Post Thrombotic Syndrome

Abstract 2978 Poster Board II-949 BACKGROUND: Post-thrombotic syndrome (PTS) is a condition of chronic venous insufficiency following deep venous thrombosis (DVT) that affects both adults and children. Nevertheless, investigation of PTS in children has lagged behind that in adults. OBJECTIVE: This systematic review was undertaken to summarize evidence from the pediatric literature on the prevalence of PTS, the validity of proposed measures of PTS, and prognostic factors for PTS in children. METHODS: A comprehensive Medline search was performed employing the following terms: “[pediatric OR children] AND [post-thrombotic syndrome OR post-phlebitic syndrome]”. Single case reports, narrative reviews, and commentaries were excluded. RESULTS: Seven case series and cross-sectional studies, nine registries and cohort studies, and one uncontrolled clinical trial were identified that met inclusion criteria; these studies reported on a total of 1316 children with DVT (Table 1). The prevalence of PTS differed substantially between observational and non-observational studies: 15% of 1042 children versus 46% of 274 children, respectively. No pediatric studies have evaluated quality of life (QOL) associated with PTS. Two reports from a single-institutional cohort have identified elevated levels of factor VIII and D-dimer and non-use of thrombolytic therapies as potentially prognostic of PTS in children, particularly among those with veno-occlusive thrombi; these are the only reports employing a PTS outcome measure that has been validated in children (the Manco-Johnson instrument). CONCLUSIONS: Overall, high-quality evidence on pediatric PTS is lacking. Collaborative prospective cohort studies and trials that use validated measures of pediatric PTS are needed to assess the incidence of, prognostic factors for, and QOL impact of PTS in children. Disclosures: No relevant conflicts of interest to declare.

scholarly journals Sunitinib Treatment for Advanced Paraganglioma: Case Report of a Novel SDHD Gene Mutation Variant and Systematic Review of the Literature

2021 ◽  
Vol 11 ◽  
Author(s):  
Franz Sesti ◽  
Tiziana Feola ◽  
Giulia Puliani ◽  
Roberta Centello ◽  
Valentina Di Vito ◽  
...  
Keyword(s):  
Systematic Review ◽  
Novel Mutation ◽  
Case Reports ◽  
Therapeutic Option ◽  
Case Series ◽  
Cochrane Library ◽  
Neuroendocrine Neoplasms ◽  
Antiangiogenic Agents ◽  
Review Of The Literature ◽  
Sunitinib Treatment

BackgroundParagangliomas (PGLs) are neuroendocrine neoplasms arising from chromaffin cells of sympathetic or parasympathetic paraganglia. Systemic therapies have been used only in metastatic PGLs. Antiangiogenic agents, such as sunitinib, could be a viable therapeutic choice in the subgroup of patients with SDH-positive PGLs. We describe the case of a man with Familial Paraganglioma Syndrome type 1 (FPGL) related to a novel mutation in SDHD gene treated with sunitinib. Furthermore, we performed a systematic review of the literature aimed to address the following question: is sunitinib treatment effective in patients with advanced/progressive/metastatic PGL?MethodsWe performed a data search using MEDLINE, Cochrane Library, and Scopus between April 2019 and September 2020. We included studies reporting data on clinical or biological characteristics, or clinical outcomes of patients with PGLs treated with sunitinib.ResultsThe search leaded to the selection of 25 publications. Data from case reports and case series showed that disease control rate (DCR = stable disease + partial response + complete response) was achieved in 34.7% of cases under sunitinib treatment. In 39% of patients DCR was followed by progressive disease (PD) or tumor relapse, 26.1% patients showed PD. Data from clinical trials showed that DCR was 83%, and the median progression free survival was 13.4 months.DiscussionData from the present literature review suggested that sunitinib could be a viable therapeutic option in advanced/progressive/metastatic inoperable PGLs. However, further trials on the efficacy of sunitinib in FPGL and sporadic PGL are needed.

scholarly journals Dabigatran Reversal With Idarucizumab and In-Hospital Mortality in Intracranial Hemorrhage: A Systematic Review of Real-Life Data From Case Reports and Case Series

2021 ◽  
Vol 12 ◽  
Author(s):  
Senta Frol ◽  
Dimitrios Sagris ◽  
Mišo Šabovič ◽  
George Ntaios ◽  
Janja Pretnar Oblak
Keyword(s):  
Systematic Review ◽  
Mortality Rate ◽  
Hospital Mortality ◽  
Intracranial Hemorrhage ◽  
Single Case ◽  
Case Reports ◽  
Anticoagulation Therapy ◽  
Real Life ◽  
Case Series ◽  
Real Life Data

Background: Intracranial hemorrhage is a severe and possibly fatal consequence of anticoagulation therapy. Idarucizumab is used in dabigatran-treated patients suffering from intracranial hemorrhage (ICH) to reverse the anticoagulant effect of dabigatran. Systematic review of real-life mortality in these patients is missing.Objectives: A review of all published dabigatran-related ICH cases treated with idarucizumab was performed. We aimed to estimate in-hospital mortality rate in these patients.Method: We searched PubMed and Scopus for all published cases of ICH in idarucizumab/dabigatran-treated patients until May 15, 2021. The assessed outcome was in-hospital mortality.Results: We identified six eligible studies (case series) with 386 patients and 54 single case reports. In-hospital mortality rate was 11.4% in the case series and 9.7% in the case reports.Conclusions: Our analysis provides clinically relevant quantitative data regarding in-hospital mortality in idarucizumab/dabigatran-treated patients with ICH, which is estimated to be 9.7–11.4%.

Cannabis-associated arteritis

VASA ◽  
2010 ◽  
Vol 39 (1) ◽  
pp. 43-53 ◽  
Author(s):  
Grotenhermen
Keyword(s):  
Systematic Review ◽  
Case Reports ◽  
Case Series ◽  
Cardiovascular Effects ◽  
Causative Factor ◽  
Cannabis Use ◽  
Thromboangiitis Obliterans ◽  
Pathological Features ◽  
Scientific Support ◽  
Clinical And Pathological Features

Background: To investigate the hypothesis that cases of arteritis similar to thromboangiitis obliterans (TAO) and associated with the use of cannabis were caused by cannabis or THC (dronabinol), or that cannabis use is a co-factor of TAO. Patients and methods: A systematic review on case reports and the literature on so-called cannabis arteritis, TAO, and cardiovascular effects of cannabinoids was conducted. Results: Fifteen reports with 57 cases of an arteritis associated with the use of cannabis and two additional case series of TAO, in which some patients also used cannabis, were identified. Clinical and pathological features of cannabis-associated arteritis do not differ from TAO and the major risk factor of TAO, tobacco use, was present in most, if not in all of these cases. The proposed pathophysiological mechanisms for the development of an arteritis by cannabis use are not substantiated. Conclusions: The hypothesis of cannabis being a causative factor or co-factor of TAO or an arteritis similar to TAO is not supported by the available evidence. The use of the term “cannabis arteritis” should be avoided until or unless more convincing scientific support is forthcoming.

Herpetic Whitlow of the Hand in Infants

2020 ◽  
Author(s):  
Mohammad M. Al-Qattan ◽  
Nada G. AlQadri ◽  
Ghada AlHayaza
Keyword(s):  
Bacterial Infection ◽  
Case Reports ◽  
Case Series ◽  
Individual Case ◽  
Senior Author ◽  
Secondary Bacterial Infection ◽  
Clinical Appearance ◽  
History Of ◽  
Mode Of Transmission ◽  
Time Of Presentation

Abstract Introduction Herpetic whitlows in infants are rare. Previous authors only reported individual case reports. We present a case series of six infants. Materials and Methods This is a retrospective study of six cases of herpetic whitlows in infants seen by the senior author (MMA) over the past 23 years (1995–2017 inclusive). The following data were collected: age, sex, digit involved in the hand, mode of transmission, time of presentation to the author, clinical appearance, presence of secondary bacterial infection, presence of other lesions outside the hand, method of diagnosis, treatment, and outcome. Results All six infants initially presented with classic multiple vesicles of the digital pulp. In all cases, there was a history of active herpes labialis in the mother. Incision and drainage or deroofing of the vesicles (for diagnostic purposes) resulted in secondary bacterial infection. Conclusion The current report is the first series in the literature on herpetic whitlows in infants. We stress on the mode of transmission (from the mother) and establishing the diagnosis clinically. In these cases, no need for obtaining viral cultures or polymerase chain reaction; and no medications are required. Once the vesicles are disrupted, secondary bacterial infection is frequent and a combination of oral acyclovir and intravenous antibiotics will be required.

Mechanical thrombectomy in pediatric stroke: systematic review, individual patient data meta-analysis, and case series

2019 ◽  
Vol 24 (5) ◽  
pp. 558-571 ◽  
Author(s):  
Kartik Bhatia ◽  
Hans Kortman ◽  
Christopher Blair ◽  
Geoffrey Parker ◽  
David Brunacci ◽  
...  
Keyword(s):  
Systematic Review ◽  
Ischemic Stroke ◽  
Acute Ischemic Stroke ◽  
Mechanical Thrombectomy ◽  
Case Reports ◽  
Meta Analysis ◽  
Case Series ◽  
Neurological Outcomes

OBJECTIVEThe role of mechanical thrombectomy in pediatric acute ischemic stroke is uncertain, despite extensive evidence of benefit in adults. The existing literature consists of several recent small single-arm cohort studies, as well as multiple prior small case series and case reports. Published reports of pediatric cases have increased markedly since 2015, after the publication of the positive trials in adults. The recent AHA/ASA Scientific Statement on this issue was informed predominantly by pre-2015 case reports and identified several knowledge gaps, including how young a child may undergo thrombectomy. A repeat systematic review and meta-analysis is warranted to help guide therapeutic decisions and address gaps in knowledge.METHODSUsing PRISMA-IPD guidelines, the authors performed a systematic review of the literature from 1999 to April 2019 and individual patient data meta-analysis, with 2 independent reviewers. An additional series of 3 cases in adolescent males from one of the authors’ centers was also included. The primary outcomes were the rate of good long-term (mRS score 0–2 at final follow-up) and short-term (reduction in NIHSS score by ≥ 8 points or NIHSS score 0–1 at up to 24 hours post-thrombectomy) neurological outcomes following mechanical thrombectomy for acute ischemic stroke in patients < 18 years of age. The secondary outcome was the rate of successful angiographic recanalization (mTICI score 2b/3).RESULTSThe authors’ review yielded 113 cases of mechanical thrombectomy in 110 pediatric patients. Although complete follow-up data are not available for all patients, 87 of 96 (90.6%) had good long-term neurological outcomes (mRS score 0–2), 55 of 79 (69.6%) had good short-term neurological outcomes, and 86 of 98 (87.8%) had successful angiographic recanalization (mTICI score 2b/3). Death occurred in 2 patients and symptomatic intracranial hemorrhage in 1 patient. Sixteen published thrombectomy cases were identified in children < 5 years of age.CONCLUSIONSMechanical thrombectomy may be considered for acute ischemic stroke due to large vessel occlusion (ICA terminus, M1, basilar artery) in patients aged 1–18 years (Level C evidence; Class IIb recommendation). The existing evidence base is likely affected by selection and publication bias. A prospective multinational registry is recommended as the next investigative step.

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