Medicine and Health Sciences Commons^™

Final Results From A Phase 2b Randomized, Placebo-Controlled Clinical Trial Of Rbx2660: A Microbiota-Based Drug For The Prevention Of Recurrent Clostridioides Difficile Infection, Erik R Dubberke, Robert Orenstein, Sahil Khanna, Beth Guthmueller, Christine Lee

2020-Current year OA Pubs

INTRODUCTION: Effective treatments for recurrent Clostridioides difficile infection (rCDI) are urgently needed. RBX2660 is an investigational microbiota-based live biotherapeutic to reduce CDI recurrence following standard-of-care antibiotic treatment in individuals with rCDI. Here we report the final safety data through 24 months of follow-up as well as final efficacy data, reflecting alignment of the pre-specified statistical analysis plan definitions with the data presented.

METHODS: The PUNCH CD2 clinical trial was a prospective, multicenter, randomized, double-blinded, placebo-controlled, three-arm phase 2b study conducted to evaluate the efficacy and safety of RBX2660 for the reduction of rCDI compared to placebo. Eligible patients were at …

Missed Opportunities In The Diagnosis Of Tuberculosis Meningitis, Niamh Simmons, Margaret A Olsen, Joanna Buss, Thomas C Bailey, Carlos Mejia-Chew

2020-Current year OA Pubs

BACKGROUND: Tuberculosis meningitis (TBM) has high mortality and morbidity. Diagnostic delays can impact TBM outcomes. We aimed to estimate the number of potentially missed opportunities (MOs) to diagnose TBM and determine its impact on 90-day mortality.

METHODS: This is a retrospective cohort of adult patients with a central nervous system (CNS) TB

RESULTS: Of 893 patients with TBM, median age at diagnosis was 50 years (interquartile range, 37-64), 61.3% were male, and 35.2% had Medicaid as primary payer. Overall, 407 (45.6%) had a prior hospital or ED visit with an MO code. In-hospital 90-day mortality was not different between those …

Primary Central Nervous System Germ Cell Tumors In Children And Young Adults: A Review Of Controversies In Diagnostic And Treatment Approach, Kee Kiat Yeo, Sumanth Nagabushan, Girish Dhall, Mohamed S Abdelbaki

2020-Current year OA Pubs

Primary central nervous system (CNS) germ cell tumors (GCT) are a rare heterogenous group of cancers, arising most commonly in the second decade of life. Through several clinical trials conducted around the world by various groups, the treatment approach for CNS GCT has advanced substantially with generally improved overall outcomes. In recent years, the goal of clinical trials has been focused on reduction of the radiotherapy burden and minimization of long-term toxicity. This review summarizes the current diagnostic and treatment regimens for CNS GCT, examines the controversies associated with these approaches, gaps in contemporary knowledge, and underscores the challenges we …

Myostatin And Follistatin As Monitoring And Prognostic Biomarkers In Dysferlinopathy, Ursula Moore, Alan Pestronk, Et Al.

2020-Current year OA Pubs

Myostatin is a myokine which acts upon skeletal muscle to inhibit growth and regeneration. Myostatin is endogenously antagonised by follistatin. This study assessed serum myostatin and follistatin concentrations as monitoring or prognostic biomarkers in dysferlinopathy, an autosomal recessively inherited muscular dystrophy. Myostatin was quantified twice with a three-year interval in 76 patients with dysferlinopathy and 38 controls. Follistatin was quantified in 62 of these patients at the same timepoints, and in 31 controls. Correlations with motor function, muscle fat fraction and contractile cross-sectional area were performed. A regression model was used to account for confounding variables. Baseline myostatin, but not …

Final Findings From The Control Trial: Strategies To Reduce The Incidence And Severity Of Neratinib-Associated Diarrhea In Patients With Her2-Positive Early-Stage Breast Cancer, Arlene Chan, Ron Bose, Et Al.

2020-Current year OA Pubs

BACKGROUND: Neratinib is an irreversible pan-HER tyrosine kinase inhibitor approved for HER2-positive early-stage and metastatic breast cancer. Diarrhea is the most frequent side effect and the most common reason for early discontinuation. The phase II CONTROL trial investigated antidiarrheal prophylaxis or neratinib dose escalation (DE) for prevention of diarrhea. We present complete study results including final data for two DE strategies.

METHODS: Patients who completed trastuzumab-based adjuvant therapy received neratinib 240 mg/day for 1 year. Early cohorts investigated mandatory prophylaxis with loperamide, then additional budesonide or colestipol. Final cohorts assessed neratinib DE over the first 2 (DE1) or 4 weeks …

Relationship Between Electrode Position And Temporal Modulation Sensitivity In Cochlear Implant Users: Are Close Electrodes Always Better?, Ning Zhou, Xuyang Shi, Omkar Dixit, Jill B Firszt, Timothy A Holden

2020-Current year OA Pubs

Temporal modulation sensitivity has been studied extensively for cochlear implant (CI) users due to its strong correlation to speech recognition outcomes. Previous studies reported that temporal modulation detection thresholds (MDTs) vary across the tonotopic axis and attributed this variation to patchy neural survival. However, correlates of neural health identified in animal models depend on electrode position in humans. Nonetheless, the relationship between MDT and electrode location has not been explored. We tested 13 ears for the effect of distance on modulation sensitivity, specifically targeting the question of whether electrodes closer to the modiolus are universally beneficial. Participants in this study …

Lgmdd1 Natural History And Phenotypic Spectrum: Implications For Clinical Trials, Andrew R Findlay, Sarah E Robinson, Stephanie Poelker, Michelle Seiffert, Rocio Bengoechea, Conrad C Weihl

2020-Current year OA Pubs

OBJECTIVE: To delineate the full phenotypic spectrum and characterize the natural history of limb girdle muscular dystrophy type D1 (LGMDD1).

METHODS: We extracted age at clinical events of interest contributing to LGMDD1 disease burden via a systematic literature and chart review. Manual muscle testing and quantitative dynamometry data were used to estimate annualized rates of change. We also conducted a cross-sectional observational study using previously validated patient-reported outcome assessments (ACTIVLIM, PROMIS-57) and a new LGMDD1 questionnaire. Some individuals underwent repeat ACTIVLIM and LGMDD1 questionnaire assessments at 1.5 and 2.5 years.

RESULTS: A total of 122 LGMDD1 patients were included from …

Early Intervention And Intensive Management Of Patients With Diabetes, Cardiorenal, And Metabolic Diseases, Yehuda Handelsman, Samuel Klein, Et Al.

2020-Current year OA Pubs

Increasing rates of obesity and diabetes have driven corresponding increases in related cardiorenal and metabolic diseases. In many patients, these conditions occur together, further increasing morbidity and mortality risks to the individual. Yet all too often, the risk factors for these disorders are not addressed promptly in clinical practice, leading to irreversible pathologic progression. To address this gap, we convened a Task Force of experts in cardiology, nephrology, endocrinology, and primary care to develop recommendations for early identification and intervention in obesity, diabetes, and other cardiorenal and metabolic diseases. The recommendations include screening and diagnosis, early interventions with lifestyle, and …

The Diagnosis Of Severe Combined Immunodeficiency: Implementation Of The Pidtc 2022 Definitions, Christopher C Dvorak, Jeffrey J Bednarski, Et Al.

2020-Current year OA Pubs

BACKGROUND: Shearer et al in 2014 articulated well-defined criteria for the diagnosis and classification of severe combined immunodeficiency (SCID) as part of the Primary Immune Deficiency Treatment Consortium's (PIDTC's) prospective and retrospective studies of SCID.

OBJECTIVE: Because of the advent of newborn screening for SCID and expanded availability of genetic sequencing, revision of the PIDTC 2014 Criteria was needed.

METHODS: We developed and tested updated PIDTC 2022 SCID Definitions by analyzing 379 patients proposed for prospective enrollment into Protocol 6901, focusing on the ability to distinguish patients with various SCID subtypes.

RESULTS: According to PIDTC 2022 Definitions, 18 of 353 …

Human Cardiac Pericytes Are Susceptible To Sars-Cov-2 Infection, Brittany D Brumback, Oleksandr Dmytrenko, Ashley N Robinson, Adam L Bailey, Pan Ma, Jing Liu, Stephanie C Hicks, Sherwin Ng, Gang Li, David M Zhang, Catherine E Lipovsky, Chieh-Yu Lin, Michael S Diamond, Kory J Lavine, Stacey L Rentschler

2020-Current year OA Pubs

COVID-19 is associated with serious cardiovascular complications, with incompletely understood mechanism(s). Pericytes have key functions in supporting endothelial cells and maintaining vascular integrity. We demonstrate that human cardiac pericytes are permissive to SARS-CoV-2 infection in organotypic slice and primary cell cultures. Viral entry into pericytes is mediated by endosomal proteases, and infection leads to up-regulation of inflammatory markers, vasoactive mediators, and nuclear factor kappa-B-dependent cell death. Furthermore, we present evidence of cardiac pericyte infection in COVID-19 myocarditis patients. These data demonstrate that human cardiac pericytes are susceptible to SARS-CoV-2 infection and suggest a role for pericyte infection in COVID-19.

Extending Outbreak Investigation With Machine Learning And Graph Theory: Benefits Of New Tools With Application To A Nosocomial Outbreak Of A Multidrug-Resistant Organism, Andrew Atkinson, Benjamin Ellenberger, Vanja Piezzi, Tanja Kaspar, Luisa Salazar-Vizcaya, Olga Endrich, Alexander B Leichtle, Jonas Marschall

2020-Current year OA Pubs

OBJECTIVE: From January 1, 2018, until July 31, 2020, our hospital network experienced an outbreak of vancomycin-resistant enterococci (VRE). The goal of our study was to improve existing processes by applying machine-learning and graph-theoretical methods to a nosocomial outbreak investigation.

METHODS: We assembled medical records generated during the first 2 years of the outbreak period (January 2018 through December 2019). We identified risk factors for VRE colonization using standard statistical methods, and we extended these with a decision-tree machine-learning approach. We then elicited possible transmission pathways by detecting commonalities between VRE cases using a graph theoretical network analysis approach.

RESULTS: …

Late-Nc Staging In Routine Neuropathologic Diagnosis: An Update, Peter T Nelson, Katherine E Schwetye, Et Al.

2020-Current year OA Pubs

An international consensus report in 2019 recommended a classification system for limbic-predominant age-related TDP-43 encephalopathy neuropathologic changes (LATE-NC). The suggested neuropathologic staging system and nomenclature have proven useful for autopsy practice and dementia research. However, some issues remain unresolved, such as cases with unusual features that do not fit with current diagnostic categories. The goal of this report is to update the neuropathologic criteria for the diagnosis and staging of LATE-NC, based primarily on published data. We provide practical suggestions about how to integrate available genetic information and comorbid pathologies [e.g., Alzheimer's disease neuropathologic changes (ADNC) and Lewy body disease]. …

Dexamethasone To Prevent Everolimus-Induced Stomatitis (Alliance Mist Trial: A221701), Kathryn J Ruddy, David Zahrieh, Jun He, Blake Waechter, Julianne L Holleran, Lionel D Lewis, Selina Chow, Jan Beumer, Matthias Weiss, Nikolaos Trikalinos, Bryan Faller, Maryam Lustberg, Hope S Rugo, Charles Loprinzi

2020-Current year OA Pubs

mTOR inhibitors such as everolimus may cause oral stomatitis, often a dose-limiting toxicity. Prior clinical research has suggested that a dexamethasone mouth rinse might help prevent and/or treat this. Alliance A221701 was a randomized phase III trial of patients initiating 10 mg daily oral everolimus that compared dexamethasone mouthwash taken preventively (initial dexamethasone group) versus therapeutically (initial placebo group) to assess two coprimary endpoints: the incidence of mTOR inhibitor-associated stomatitis (mIAS), and the area under the curve (AUC) of mIAS-associated pain over an 8-week treatment period. A Fisher's exact test was used to compare the incidences while a Wilcoxon rank-sum …

Clinical Importance Of Clonal Hematopoiesis In Metastatic Gastrointestinal Tract Cancers, Bill H Diplas, Kelly Bolton, Et Al.

2020-Current year OA Pubs

IMPORTANCE: Clonal hematopoiesis (CH) has been associated with development of atherosclerosis and leukemia and worse survival among patients with cancer; however, the association with cancer therapy efficacy, in particular immune checkpoint blockade (ICB), and toxicity has not yet been established. Given the widespread use of ICB and the critical role hematopoietic stem cell-derived lymphocytes play in mediating antitumor responses, CH may be associated with therapeutic efficacy and hematologic toxicity.

OBJECTIVE: To determine the association between CH and outcomes, hematologic toxicity, and therapeutic efficacy in patients with metastatic gastrointestinal tract cancers being treated with systemic therapy, both in the first-line metastatic …

Fixed-Dose Combination Therapy-Based Protocol Compared With Free Pill Combination Protocol: Results Of A Cluster Randomized Trial, Olutobi A Sanuade, Mark D Huffman, Et Al.

2020-Current year OA Pubs

Fixed-dose combination (FDC) therapy is recommended for hypertension management in Nigeria based on randomized trials at the individual level. This cluster-randomized trial evaluates effectiveness and safety of a treatment protocol that used two-drug FDC therapy as the second and third steps for hypertension control compared with a protocol that used free pill combinations. From January 2021 to June 2021, 60 primary healthcare centers in the Federal Capital Territory of Nigeria were randomized to a protocol using FDC therapy as second and third steps compared with a protocol that used the same medications in free pill combination therapy for these steps. …

Real-World Covid-19 Vaccine Effectiveness Against The Omicron Ba.2 Variant In A Sars-Cov-2 Infection-Naive Population, Jonathan J Lau, Chao Wu, Gaya K Amarasinghe, Et Al.

2020-Current year OA Pubs

The SARS-CoV-2 Omicron variant has demonstrated enhanced transmissibility and escape of vaccine-derived immunity. Although first-generation vaccines remain effective against severe disease and death, robust evidence on vaccine effectiveness (VE) against all Omicron infections, irrespective of symptoms, remains sparse. We used a community-wide serosurvey with 5,310 subjects to estimate how vaccination histories modulated risk of infection in infection-naive Hong Kong during a large wave of Omicron BA.2 epidemic in January-July 2022. We estimated that Omicron infected 45% (41-48%) of the local population. Three and four doses of BNT162b2 or CoronaVac were effective against Omicron infection 7 days after vaccination (VE of …

Navigating Care For Rare Diseases: Caregiver And Patient Advice For Families And Clinicians Managing Care For Vascular Malformations, Anna M Kerr, Christine Bereitschaft, Kayla M Duty, Bryan A Sisk

2020-Current year OA Pubs

OBJECTIVE: Families affected by rare diseases face many challenges finding adequate care and often report poor communication with clinicians. In the current study, we explore patient and caregiver advice for families and clinicians in the context of complex vascular malformations (VMs), a condition that is frequently misunderstood and misdiagnosed.

METHODS: We performed semi-structured interviews with 21 adult patients with complex VMs and 24 caregivers of children with VMs. We analyzed the transcripts using thematic analysis.

RESULTS: Participants advised patients and caregivers to advocate for care, address mental and emotional well-being, seek social support, and promote self-management and self-care. Participants advised …

Machine Learning Approximations To Predict Epigenetic Age Acceleration In Stroke Patients, Isabel Fernández-Pérez, Joan Jiménez-Balado, Uxue Lazcano, Eva Giralt-Steinhauer, Lucía Rey Álvarez, Elisa Cuadrado-Godia, Ana Rodríguez-Campello, Adrià Macias-Gómez, Antoni Suárez-Pérez, Anna Revert-Barberá, Isabel Estragués-Gázquez, Carolina Soriano-Tarraga, Jaume Roquer, Angel Ois, Jordi Jiménez-Conde

2020-Current year OA Pubs

Age acceleration (Age-A) is a useful tool that is able to predict a broad range of health outcomes. It is necessary to determine DNA methylation levels to estimate it, and it is known that Age-A is influenced by environmental, lifestyle, and vascular risk factors (VRF). The aim of this study is to estimate the contribution of these easily measurable factors to Age-A in patients with cerebrovascular disease (CVD), using different machine learning (ML) approximations, and try to find a more accessible model able to predict Age-A. We studied a CVD cohort of 952 patients with information about VRF, lifestyle habits, …

Autophagy Requirements For Eye Lens Differentiation And Transparency, Lisa Brennan, M Joseph Costello, J Fielding Hejtmancik, A Sue Menko, S Amer Riazuddin, Alan Shiels, Marc Kantorow

2020-Current year OA Pubs

Recent evidence points to autophagy as an essential cellular requirement for achieving the mature structure, homeostasis, and transparency of the lens. Collective evidence from multiple laboratories using chick, mouse, primate, and human model systems provides evidence that classic autophagy structures, ranging from double-membrane autophagosomes to single-membrane autolysosomes, are found throughout the lens in both undifferentiated lens epithelial cells and maturing lens fiber cells. Recently, key autophagy signaling pathways have been identified to initiate critical steps in the lens differentiation program, including the elimination of organelles to form the core lens organelle-free zone. Other recent studies using ex vivo lens culture …

Identification Of Bradycardia Following Remdesivir Administration Through The Us Food And Drug Administration American College Of Medical Toxicology Covid-19 Toxic Pharmacovigilance Project, Jason M Devgun, Rongmei Zhang, Jeffrey Brent, Paul Wax, Keith Burkhart, Alison Meyn, Sharan Campleman, Stephanie Abston, Kim Aldy, Toxicology Investigators Consortium Fact Study Group

2020-Current year OA Pubs

IMPORTANCE: The rapid spread and mortality associated with COVID-19 emphasized a need for surveillance system development to identify adverse events (AEs) to emerging therapeutics. Bradycardia is a remdesivir infusion-associated AE listed in the US Food and Drug Administration-approved prescribing information.

OBJECTIVE: To evaluate the magnitude and duration of bradycardic events following remdesivir administration.

DESIGN, SETTING, AND PARTICIPANTS: A multicenter cohort study of patients with recorded heart rate less than 60 beats per minute within 24 hours after administration of a remdesivir dose was conducted between November 23, 2020, and October 31, 2021. Participants included patients hospitalized with COVID-19 at 15 …

Dissecting Cell Identity Via Network Inference And In Silico Gene Perturbation, Kenji Kamimoto, Blerta Stringa, Christy M Hoffmann, Kunal Jindal, Lilianna Solnica-Krezel, Samantha A Morris

2020-Current year OA Pubs

Cell identity is governed by the complex regulation of gene expression, represented as gene-regulatory networks

Effect Of Histology On Stereotactic Body Radiotherapy For Non-Small Cell Lung Cancer Oligometastatic Pulmonary Lesions, Alexander J. Allen, Dominic A. Labella, Roman O. Kowalchuk, Michael R. Waters, Charles R. Kersh

2020-Current year OA Pubs

BACKGROUND: Stereotactic body radiotherapy (SBRT) is commonly used to provide targeted treatment to metastatic lung disease. Investigation is needed to understand the influence of histology on treatment outcomes. We report how tumor histology affects local control (LC) in a cohort of patients with non-small cell lung cancer (NSCLC) receiving SBRT for oligometastatic and recurrent pulmonary lesions.

METHODS: Patients who received SBRT to recurrent or oligometastatic NSCLC pulmonary lesions from 2015-2019 at our institution were included in this retrospective cohort study. Minimum follow-up was 2 months. Kaplan-Meier (KM) analysis was performed to assess local progression-free survival (LPFS). Local failure cumulative incidence …

The Hepatic Mitochondrial Pyruvate Carrier As A Regulator Of Systemic Metabolism And A Therapeutic Target For Treating Metabolic Disease, Kyle S Mccommis, Brian N Finck

2020-Current year OA Pubs

Pyruvate sits at an important metabolic crossroads of intermediary metabolism. As a product of glycolysis in the cytosol, it must be transported into the mitochondrial matrix for the energy stored in this nutrient to be fully harnessed to generate ATP or to become the building block of new biomolecules. Given the requirement for mitochondrial import, it is not surprising that the mitochondrial pyruvate carrier (MPC) has emerged as a target for therapeutic intervention in a variety of diseases characterized by altered mitochondrial and intermediary metabolism. In this review, we focus on the role of the MPC and related metabolic pathways …

Exploration Of The Nuclear Proteomes In The Ciliate Oxytricha Trifallax, Michael W Lu, Leslie Y Beh, V Talya Yerlici, Wenwen Fang, Katarzyna Kulej, Benjamin A Garcia, Laura F Landweber

2020-Current year OA Pubs

Nuclear dimorphism is a fundamental feature of ciliated protozoa, which have separate somatic and germline genomes in two distinct organelles within a single cell. The transcriptionally active somatic genome, contained within the physically larger macronucleus, is both structurally and functionally different from the silent germline genome housed in the smaller micronucleus. This difference in genome architecture is particularly exaggerated in

Chronic Anti-Coagulation Therapy Reduced Mortality In Patients With High Cardiovascular Risk Early In Covid-19 Pandemic, Mohamed S Zaghloul, Momodou Jammeh, Andrew Gibson, Suhong Luo, Kelley Chadwick-Mansker, Qianjin Liu, Yan Yan, Mohamed A Zayed

2020-Current year OA Pubs

BACKGROUND: Coronavirus disease 2019 (COVID-19) is associated with provoked thrombo-inflammatory responses. Early in the COVID-19 pandemic this was thought to contribute to hypercoagulability and multi-organ system complications in infected patients. Limited studies have evaluated the impact of therapeutic anti-coagulation therapy (AC) in alleviating these risks in COVID-19 positive patients. Our study aimed to investigate whether long-term therapeutic AC can decrease the risk of multi-organ system complications (MOSC) including stroke, limb ischemia, gastrointestinal (GI) bleeding, in-hospital and intensive care unit death in COVID-19 positive patients hospitalized during the early phase of the pandemic in the United States.

METHODS: A retrospective analysis …

Neuroimaging Studies Of The Neural Correlates Of Heart Rate Variability: A Systematic Review, Patrycja S Matusik, Chuwen Zhong, Paweł T Matusik, Omar Alomar, Phyllis K Stein

2020-Current year OA Pubs

Direct and indirect links between brain regions and cardiac function have been reported. We performed a systematic literature review to summarize current knowledge regarding the associations of heart rate variability (HRV) and brain region morphology, activity and connectivity involved in autonomic control at rest in healthy subjects. Both positive and negative correlations of cortical thickness and gray matter volumes of brain structures with HRV were observed. The strongest were found for a cluster located within the cingulate cortex. A decline in HRV, as well as cortical thickness with increasing age, especially in the orbitofrontal cortex were noted. When associations of …

Drug Specificity And Affinity Are Encoded In The Probability Of Cryptic Pocket Opening In Myosin Motor Domains, Artur Meller, Jeffrey M Lotthammer, Louis G Smith, Borna Novak, Lindsey A Lee, Catherine C Kuhn, Lina Greenberg, Leslie A Leinwand, Michael J Greenberg, Gregory R Bowman

2020-Current year OA Pubs

The design of compounds that can discriminate between closely related target proteins remains a central challenge in drug discovery. Specific therapeutics targeting the highly conserved myosin motor family are urgently needed as mutations in at least six of its members cause numerous diseases. Allosteric modulators, like the myosin-II inhibitor blebbistatin, are a promising means to achieve specificity. However, it remains unclear why blebbistatin inhibits myosin-II motors with different potencies given that it binds at a highly conserved pocket that is always closed in blebbistatin-free experimental structures. We hypothesized that the probability of pocket opening is an important determinant of the …

Development Of De Novo Nonalcoholic Fatty Liver Disease Following Pancreatectomy, Vanisha Patel, Parth Shah, Daniel R. Ludwig, Chet W. Hammill, Motaz Ashkar

2020-Current year OA Pubs

De novo non-alcoholic fatty liver disease (NAFLD) after pancreatectomy is a recognized phenomenon; however, its pathophysiology is poorly understood. This study aimed to determine the incidence and identify peri-operative risk factors for the development of de novo NAFLD within various pancreatectomy groups. This single-center retrospective cohort study included patients who underwent pancreatectomy between 2000 and 2020. The incidence rate of de novo NAFLD and time to diagnosis were recorded across patients with malignant versus benign indications for pancreatectomy. The overall incidence of de novo NAFLD after pancreatectomy was 17.5% (24/136). Twenty-one percent (20/94) of patients with malignant indications for surgery …

Gold Nanoparticle-Enhanced X-Ray Microtomography Of The Rodent Reveals Region-Specific Cerebrospinal Fluid Circulation In The Brain, Shelei Pan, Peter H Yang, Dakota Defreitas, Sruthi Ramagiri, Peter O Bayguinov, Carl D Hacker, Abraham Z Snyder, Jackson Wilborn, Hengbo Huang, Gretchen M Koller, Dhvanii K Raval, Grace L Halupnik, Sanja Sviben, Rui Tang, Gabriel Haller, James D Quirk, James A J Fitzpatrick, Prabagaran Esakky, Jennifer M Strahle, Et Al.

2020-Current year OA Pubs

Cerebrospinal fluid (CSF) is essential for the development and function of the central nervous system (CNS). However, the brain and its interstitium have largely been thought of as a single entity through which CSF circulates, and it is not known whether specific cell populations within the CNS preferentially interact with the CSF. Here, we develop a technique for CSF tracking, gold nanoparticle-enhanced X-ray microtomography, to achieve micrometer-scale resolution visualization of CSF circulation patterns during development. Using this method and subsequent histological analysis in rodents, we identify previously uncharacterized CSF pathways from the subarachnoid space (particularly the basal cisterns) that mediate …

A Broad Characterization Of Glycogen Storage Disease Iv Patients: A Clinical, Genetic, And Histopathological Study, Matheus Vernet Machado Bressan Wilke, Bibiana Mello De Oliveira, Rodrigo Tzovenos Starosta, Marwan Shinawi, Liang Lu, Mai He, Yamin Ma, Janis Stoll, Carolina Fischinger Moura De Souza, Ana Cecilia Menezes De Siqueira, Sandra Maria Gonçalves Vieira, Carlos Thadeu Cerski, Lilia Farret Refosco, Ida Vanessa Doederlein Schwartz

2020-Current year OA Pubs

Glycogen storage disease type IV (GSD IV) is an ultra-rare autosomal recessive disease caused by variants in the