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

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.

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Rituximab for Eosinophilic Granulomatosis with Polyangiitis: a Systematic Review of Observational Studies

Rheumatology ◽

10.1093/rheumatology/keab046 ◽

2021 ◽

Author(s):

Vincenzo G Menditto ◽

Giulia Rossetti ◽

Diletta Olivari ◽

Alessia Angeletti ◽

Marco Rocchi ◽

...

Keyword(s):

Systematic Review ◽

Cohort Studies ◽

Granulomatosis With Polyangiitis ◽

Case Reports ◽

Case Series ◽

Biologic Agents ◽

Cochrane Library ◽

Eosinophilic Granulomatosis With Polyangiitis ◽

Extensive Literature ◽

Eosinophilic Granulomatosis

Abstract Objective to analyze the available evidence about the use of rituximab (RTX) and other biologic agents in Eosinophilic Granulomatosis with Polyangiitis (EGPA) patients and to provide useful findings to inform the design of future, reliable clinical trials. Methods A systematic review was performed. A systematic search was conducted in PubMed/MEDLINE, Scopus, Web of Science and the Cochrane library databases and an extensive literature search on other biologic agents. Results 45 papers pertinent to our questions were found: 16 retrospective cohort studies, 8 case series, 3 prospective cohort studies and 18 single case reports, for a total of 368 EGPA patients. More than 80% of evaluable patients achieved complete or partial remission with a tendency towards a higher rate of complete response in pANCA positive subgroup. Conclusion Although the majority of the evaluable EGPA patients treated with RTX appears to achieve complete remission, we strongly believe that a number of sources of heterogeneity impairs a clear interpretation of results and limits their transferability in clinical practice. Differences in design, enrollment criteria, outcome definition and measurement make a comparison among data obtained from studies on RTX and other biologic agents unreliable.

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Z-drugs and Falls: A Focused Review of the Literature

The Senior Care Pharmacist ◽

10.4140/tcp.n.2020.549 ◽

2020 ◽

Vol 35 (12) ◽

pp. 549-554

Author(s):

Nicole Ryba ◽

Rebecca Rainess

Keyword(s):

Primary Outcome ◽

Case Reports ◽

Inappropriate Medication ◽

Case Series ◽

Cochrane Library ◽

Inpatient Setting ◽

Care Hospital ◽

Review Of The Literature ◽

Increased Risk ◽

Risk Of Falls

OBJECTIVE: To provide a focused review of the literature related to the association between exposure to Z-drugs and the risk of falls, especially in the older population. DATA SOURCES: A literature search was conducted using Medline, PsychInfo, and the Cochrane Library database for all clinical trials, case series, and case reports published in English up to May 2020. The search terms used consisted of each Z-drug, including "zolpidem," "zopiclone," "eszopiclone," and "zaleplon," matched with "falls." STUDY SELECTION: The search yielded 295 studies. After review of abstracts, content and references were reviewed, and duplicates removed, a total of 9 articles met inclusion of exposure to at least 1 Z-drug and a primary outcome of falls. DATA EXTRACTION: The American Geriatrics Society 2019 Beers Criteria Update for Potentially Inappropriate Medication Use in Older Adults recommends to avoid using nonbenzodiazepine hypnotics in this patient population because of the risk of adverse events. DATA SYNTHESIS: A majority of the literature suggests an increased risk of falls with exposure to Z-drug use, especially zolpidem. Eight trials examined falls as a primary outcome in non-elderly (n=3) and elderly (n=5) patients in different settings, mostly in an inpatient setting (nursing facility or acute care hospital). CONCLUSION: Exposure to Z-drugs, especially zolpidem, should be evaluated and counseled on continuously as these medications put patients at an increased risk for falls and other complications.

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Z-drugs and Falls: A Focused Review of the Literature

The Senior Care Pharmacist ◽

10.4140/tcp.n.2020.549. ◽

2020 ◽

Vol 35 (12) ◽

pp. 549-554

Author(s):

Nicole Ryba ◽

Rebecca Rainess

Keyword(s):

Primary Outcome ◽

Case Reports ◽

Inappropriate Medication ◽

Case Series ◽

Cochrane Library ◽

Inpatient Setting ◽

Care Hospital ◽

Review Of The Literature ◽

Increased Risk ◽

Risk Of Falls

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A Systematic Review of the Literature on Early Vocalizations and Babbling Patterns in Young Children

Communication Disorders Quarterly ◽

10.1177/1525740118760215 ◽

2018 ◽

Vol 40 (1) ◽

pp. 3-14 ◽

Cited By ~ 12

Author(s):

Lydia Morgan ◽

Yvonne E. Wren

Keyword(s):

Systematic Review ◽

Case Series ◽

Speech Development ◽

Cochrane Library ◽

Bibliographic Databases ◽

Review Of The Literature ◽

Phonological Acquisition ◽

Future Studies ◽

The Cochrane Library ◽

Children's Speech

Children’s speech development begins in infancy. The pattern of this development has been explored in studies over a number of years using a range of research methodology and approaches to investigation. A systematic review of the existing literature was carried out to determine the collective contribution of this literature to our understanding of early vocalizations and babbling through the period 9 to 18 months. Eight bibliographic databases were searched as well as the Cochrane library. Thirteen studies were identified for inclusion, which were mostly longitudinal observational case series. The review identified progressive increases in the complexity and volume of infants’ early vocalizations through the period. It also found a broad order of phonological acquisition. Although the studies in this review demonstrated marked individual variation, the review provides indicative patterns of development which can be used as a basis to explore relationships with later speech development in future studies.

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Repurposing of drugs for Covid-19: a systematic review and meta-analysis

10.1101/2020.06.07.20124677 ◽

2020 ◽

Cited By ~ 1

Author(s):

Pinky Kotecha ◽

Alexander Light ◽

Enrico Checcucci ◽

Daniele Amparore ◽

Cristian Fiori ◽

...

Keyword(s):

Systematic Review ◽

Clinical Trials ◽

Adverse Events ◽

Clinical Improvement ◽

Case Reports ◽

Meta Analysis ◽

Case Series ◽

Cochrane Library ◽

Control Group ◽

Significant Difference

AbstractObjectiveThe aim of this systematic review is to evaluate the data currently available regarding the repurposing of different drugs for Covid-19 treatment. Participants with suspected or diagnosed Covid-19 will be included. The interventions being considered are drugs being repurposed, and comparators will include standard of care treatment or placebo.MethodsWe searched Ovid-MEDLINE, EMBASE, Cochrane library, clinical trial registration site in the UK(NIHR), Europe (clinicaltrialsregister.eu), US (ClinicalTrials.gov) and internationally (isrctn.com), and reviewed the reference lists of articles for eligible articles published up to April 22, 2020. All studies in English that evaluated the efficacy of the listed drugs were included. Cochrane RoB 2.0 and ROBINS-I tool were used to assess study quality. This systematic review adheres to the PRISMA guidelines. The protocol is available at PROSPERO (CRD42020180915).ResultsFrom 708 identified studies or clinical trials, 16 studies and 16 case reports met our eligibility criteria. Of these, 6 were randomized controlled trials (763 patients), 7 cohort studies (321 patients) and 3 case series (191 patients). Chloroquine (CQ) had a 100% discharge rate compared to 50% with lopinavir-ritonavir at day 14, however a trial has recommended against a high dosage due to cardiotoxic events. Hydroxychloroquine (HCQ) has shown no significant improvement in negative seroconversion rate which is also seen in our meta-analysis (p=0.68). Adverse events with HCQ have a significant difference compared to the control group (p=0.001). Lopinavir-ritonavir has shown no improvement in time to clinical improvement which is seen in our meta-analyses (p=0.1). Remdesivir has shown no significant improvement in time to clinical improvement but this trial had insufficient power.DiscussionDue to the paucity in evidence, it is difficult to establish the efficacy of these drugs in the treatment of Covid-19 as currently there is no significant clinical effectiveness of the repurposed drugs. Further large clinical trials are required to achieve more reliable findings. A risk-benefit analysis is required on an individual basis to weigh out the potential improvement in clinical outcome and viral load reduction compared to the risks of the adverse events. (1-16)

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625 EFFICACY OF MYOTOMY IN PATIENTS WITH SIGMOID MEGAESOPHAGUS: A SYSTEMATIC REVIEW AND META-ANALYSIS

Diseases of the Esophagus ◽

10.1093/dote/doab052.625 ◽

2021 ◽

Vol 34 (Supplement_1) ◽

Author(s):

Marina Orlandini ◽

Maria Carolina Serafim ◽

Letícia Datrino ◽

Clara Santos ◽

Luca Tristão ◽

...

Keyword(s):

Systematic Review ◽

Heller Myotomy ◽

Selection Process ◽

Case Reports ◽

Meta Analysis ◽

Case Series ◽

Cochrane Library ◽

Cross Sectional ◽

Manual Search ◽

The Mean

Abstract Megaesophagus progress to sigmoid megaesophagus (SM) in 10–15% of patients, presenting tortuosity and sigmoid colon aspect. Esophagectomy is the choice treatment but is associated with high complications and mortality rates. To avoid the esophagectomy inherent morbidity, several authors recommend Heller myotomy (HM) with pull-down technique for SM, mainly for patients with comorbidities and the elderly. This systematic review and meta-analysis is the first to analyze the effectiveness of HM for treating SM. Methods A systematic review was conducted in PubMed, Embase, Cochrane Library Central, Lilacs (BVS), and manual search of references. Inclusion criteria were: a) clinical trials, cohort studies, case series; b) patients with SM and esophageal diameter ≥ 6 cm; and c) patients undergoing primary myotomy. The exclusion criteria were: a) reviews, case reports, cross-sectional studies, editorials, letters, congress abstracts, full-text unavailability; b) animal studies, c) previous surgical treatment for achalasia; and d) pediatric studies. There were no restrictions on language or date of publication, and no filters were applied for the selection process. Random model and a 95% confidence interval (CI) were used. Results Sixteen articles were selected, encompassing 231 patients. The mean age ranged from 36 to 61 years old, and the mean follow-up ranged from 16 to 109 months. The analyzed outcomes include mortality, complications (pneumonia, pneumothorax, gastroesophageal reflux), need for reintervention (remyotomy, dilation and esophagectomy), and results classified as ‘good’ and ‘excellent’. Mortality rate was 0.035 (CI: 0.017–0.07; p < 0.01). Complications rate was 0.08 (CI: 0.04–0.153; p = 0.01). Need for retreatment rate was 0.161 (CI: 0.053–0.399; p < 0.01). Probability of good or excellent outcomes after myotomy was 0.762 (CI: 0.693–0.819; p < 0,01). Conclusion Heller myotomy is an option for avoiding esophagectomy in achalasia, with a low morbimortality rate and good results. It is effective for most patients but will fail in a minority of patients and demand retreatment, be it a remyotomy, endoscopic treatment or esophagectomy.

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The Use of Milrinone in Patients with Delayed Cerebral Ischemia Following Subarachnoid Hemorrhage: A Systematic Review

Canadian Journal of Neurological Sciences / Journal Canadien des Sciences Neurologiques ◽

10.1017/cjn.2016.316 ◽

2016 ◽

Vol 44 (2) ◽

pp. 152-160 ◽

Cited By ~ 14

Author(s):

Marcelo Lannes ◽

Frederick Zeiler ◽

Céline Guichon ◽

Jeanne Teitelbaum

Keyword(s):

Systematic Review ◽

Subarachnoid Hemorrhage ◽

Cerebral Ischemia ◽

Case Reports ◽

Randomized Study ◽

Delayed Cerebral Ischemia ◽

Case Series ◽

Cochrane Library ◽

York Academy ◽

Randomized Controlled Studies

AbstractObjective:The purpose of this article is to provide a systematic review of the evidence supporting the use of milrinone for the management of delayed cerebral ischemia (DCI) following subarachnoid hemorrhage (SAH).Design:Primary outcomes were functional neurological status and the incidence of cerebral infarction. Search strategies adapted to the different databases were developed by a professional librarian. Medline, EMBASE, the Cochrane Library database, Web of Science, SCOPUS, BIOSIS, Global Health, Health Star, Open SIGLE, Google Scholar and the New York Academy of Medicine Gray Literature were searched as well as clinical trials databases and the proceedings of several scientific meetings. Quality of the evidence for these outcomes across studies was adjudicated using the GRADE Working Group criteria.Results:The search resulted in 284 citations after elimination of duplicates. Of those 9 conference proceedings and 15 studies met inclusion criteria and consisted of case reports, case series and two comparative studies: one non-randomized study with physiological outcomes only and a case series with historical controls. There was considerable variation in dosing and in co-interventions and no case control or randomized controlled studies were found.Conclusion:There is currently only very low quality evidence to support the use of milrinone to improve important outcomes in patients with delayed cerebral ischemia secondary to subarachnoid hemorrhage. Further research is needed to clarify the value and risks of this medication in patients with SAH.

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Clinical features, Diagnosis, and Treatment of COVID-19: A systematic review of case reports and case series

10.1101/2020.03.28.20046151 ◽

2020 ◽

Cited By ~ 2

Author(s):

Azin Tahvildari ◽

Mahta Arbabi ◽

Yeganeh Farsi ◽

Parnian Jamshidi ◽

Saba Hasanzadeh ◽

...

Keyword(s):

Systematic Review ◽

Case Reports ◽

Antiviral Treatment ◽

Surrogate Marker ◽

Case Series ◽

Neuraminidase Inhibitor ◽

Hiv Protease ◽

Treatment Modalities ◽

Cochrane Library ◽

Novel Coronavirus

AbstractObjectivesThe 2019 novel coronavirus (COVID-19) has been declared a public health emergency worldwide. The objective of this systematic review was to characterize the clinical, diagnostic, and treatment characteristics of patients presenting with COVID-19.MethodsWe conducted a structured search using PubMed/Medline, Embase, Web of Science and the Cochrane Library to collect both case reports and case series on COVID-19 published up to February 30, 2020.ResultsThirty-four articles were included analyzing a total of 99 patients with a mean age of 46.2 years. The most common presenting symptom in patients who tested positive for COVID-19 was fever, reported in up to 83% of patients from 76.4% of the analyzed studies. Other symptoms including rhinorrhea, dizziness, and chills were less frequently reported. Additionally, in studies which reported C-reactive protein (CRP) measurements (44%), a large majority of patients displayed an elevated CRP (73%). Progression to acute respiratory distress syndrome (ARDS) was the most common complication of patients testing positive for COVID-19 (33%). CT images displayed ground-glass opacification (GGO) patterns (80%) as well as bilateral lung involvement (71.0%). The most commonly used antiviral treatment modalities included, lopinavir (HIV protease inhibitor), arbidiol hydrochloride (influenza fusion inhibitor), and oseltamivir (neuraminidase inhibitor).ConclusionsDevelopment of ARDS may play a role in estimating disease progression and mortality risk. Early detection of elevations in serum CRP, combined with a clinical COVID-19 symptom presentation may be used as a surrogate marker for presence and severity of disease. There is a paucity of data surrounding the efficacy of treatments. There is currently not a well-established gold standard therapy for the treatment of diagnosed COVID-19. Further prospective investigations are necessary.

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

Operative Neurosurgery ◽

10.1093/ons/opab284 ◽

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.

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Diagnostic Criteria for Odontogenic Sinusitis: A Systematic Review

American Journal of Rhinology and Allergy ◽

10.1177/1945892420976766 ◽

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.

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