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Quantifying Kinematic Tremor in an NGLY1-Deficient Individual: A Case Study

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Case Reports in Clinical Medicine 2024年第1期13卷 25-36页

作者： Brock Futrell Christopher Alexander Malaya Dacia Martinez Diaz Christian Alfaro Hannah Elizabeth Gustafson Subhalakshmi Chandrasekaran Rhea Mohan Phatak Bernhard Suter Charles shannon Layne Department of Health and Human Performance University of Houston Houston USA Center for Neuromotor and Biomechanics Research University of Houston Houston USA Department of Computer Science and Engineering Texas A&M University College Station USA Blue Bird Circle Rett Center Texas Children’s Hospital Houston USA Baylor College of Medicine Houston USA Center for NeuroEngineering and Cognitive Science University of Houston Houston USA

NGLY1 Deficiency is an autosomal recessive congenital disorder that has been identified in less than 100 individuals. Most individuals with NGLY1 Deficiency display hyperkinetic movement disorders, including choreiform, athetoid, dystonic myoclonic, dyskinetic, and dysmetric movements. Developing a consistent and concise consensus on the classification and evaluation of tremors is essential to forward the research and treatment of tremors. It has also been reported that some individuals with NGLY1 Deficiency demonstrate tremor, but such tremor has never been formally investigated. The primary objective of this study is to determine if an individual with NGLY1 Deficiency demonstrates an identifiable tremor during a series of arm movements and, if so, describe the frequency and power characteristics of that tremor. Arm movement kinematics were obtained using a 16-camera Vicon system, and time series trajectory waveforms for three planes of a marker placed on the hand were developed. Custom MATLAB scripts were utilized to compute Fast Fourier Transformations of the data within the identified waveform segments. A mean frequency of 2.30 Hz (SD = 1.05) with a mean power of 5.02 |P1(f)| (SD = 4.63) suggests that our participant’s kinematic data did display a persistent tremor in both hands across all tasks and movement planes. Analyses of the reaching hand and the non-reaching hand suggest the participant displayed an action tremor in both postural and intention (kinetic) tremors. Future directions should include assessing additional individuals with NGLY1 Deficiency to determine if the tremor is a distinguishable disorder behavior. Additionally, evaluating other anatomical sites, such as the elbow, head, and lower limbs, would provide further insights into the characteristics of this tremor.

关键词： Tremor NGLY Fast Fourier Transformation Reaching Hyperkinetic Movement

Particle elasticity influences polymeric artificial antigen presenting cell effectiveness in vivo via CD8+T cell activation,macrophage uptake,and the protein corona

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Nano Research 2024年第10期17卷 9052-9064页

作者： Savannah E.Est-Witte Sydney R.shannon Dennis H.Gong Kaitlyn G.Calabresi Jawaun J.Harris Kaitlyn Storm Edwin J.Yoo Ariel Isser Vivek P.Jani Natalie K.Livingston Mary O.Omotoso Kelly Rhodes Elana Ben-Akiva Randall A.Meyer Zoe T.Hsieh Simone Sidoli Stephany Y.Tzeng Jonathan P.Schneck Jordan J.Green Translational Tissue Engineering Center Institute for NanobiotechnologyDepartment of Biomedical EngineeringJohns Hopkins School of Medicine400 N BroadwayBaltimoreMD 21231USA Institute for Cell Engineering Department of PathologyJohns Hopkins School of Medicine733 N BroadwayBaltimoreMD 21205USA Department of Biochemistry Albert Einstein College of MedicineThe BronxNew York CityNY 10461USA

Adoptive cell therapy(ACT)is an immunotherapy strategy for cancer that has seen widespread clinical *** ACT,patient-derived lymphocytes are stimulated with the antigen of interest ex vivo,proliferated,then returned to the patient to initiate an antigen-specific antitumor *** effective,this process is resource-intensive and logistically impossible for many *** artificial antigen presenting cells(aAPCs)offer a potential“off-the-shelf”alternative to ex vivo *** particulate aAPCs perform well in vitro,they have had limited success in vivo due to poor bioavailability after *** to bioavailability include rapid clearance,unfavorable biodistribution,and inadequate interactions with CD8+T cells at sites of *** properties such as elasticity have been shown to vastly impact the bioavailability and particle-cell interactions,but this has yet to be investigated in the context of aAPCs for in vivo T-cell *** literature likewise indicates that biomaterial properties,especially elasticity,can modulate T-cell activation in *** the goal of creating a more biomimetic,next-generation particulate aAPC,we developed a poly(ethylene)glycol hydrogel particle platform with tunable elasticity to investigate the impact of elasticity on antigen-specific T cell activation for in vivo adoptive *** this knowledge,we were able to gain more precise control over in vivo T cell activation and investigate possible mechanisms including the effects of aAPC elasticity on T cell binding,macrophage uptake,and the protein corona.

关键词： adoptive cell therapy artificial antigen presenting cells particle drug delivery elasticity protein corona poly(ethylene glycol)

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Come as you are

Come as you are

A Progressive Deployment Strategy for Cooperative Adaptive Cruise Control to Improve Trafc Dynamics

作者： (1951-)roof Judith

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International Journal of Automation and computing 2014年第1期11卷 10-18页

作者： Georges M.Arnaout shannon Bowling Department of Engineering Management and Systems Engineering Old Dominion University Department of Electrical Engineering Technology Bluefeld State College

Cooperative adaptive cruise control(CACC)vehicles are intelligent vehicles that use vehicular ad hoc networks(VANETs)to share trafc information in real *** studies have shown that CACC could have an impact on increasing highway capacities at high market *** reaching a high CACC market penetration level is not occurring in the near future,this study presents a progressive deployment approach that demonstrates to have a great potential of reducing trafc congestions at low CACC penetration *** a previously developed microscopic trafc simulation model of a freeway with an on-ramp—created to induce perturbations and trigger stop-and-go trafc,the CACC system s efect on the trafc performance is *** results show signifcance and indicate the potential of CACC systems to improve trafc characteristics which can be used to reduce trafc *** study shows that the impact of CACC is positive and not only limited to a high market *** giving CACC vehicles priority access to high-occupancy vehicle(HOV)lanes,the highway capacity could be signifcantly improved with a CACC penetration as low as 20%.

关键词： Intelligent transportation systems telematics vehicular adhoc networks cooperative adaptive cruise control trafc simulation agent-based trafc simulation

Direct and in direct effects of light pollution on the performance of an herbivorous insect

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Insect Science 2019年第4期26卷 770-776页

作者： Kylee Grenis shannon M. Murphy Department of Biological Sciences University of Denver Denver Colorado USA

Light pollution is a global disturbance with resounding impacts on a wide variety of organisms, but our understanding of these impacts is restricted to relatively few higher vertebrate species. We tested the direct effects of light pollution on herbivore performance as well as indirect effects mediated by host plant quality. We found that artificial light from streetlights alters plant toughness. Additionally, we found evidence of both direct and indirect effects of light pollution on the performance of an herbivorous insect, which indicates that streetlights can have cascading impacts on multiple trophic levels. Our novel findings suggest that light pollution can alter plant-insect interactions and thus may have important community-wide consequences.

关键词： anthropogenic disturbance continuous photoperiod light pollution Lepidoptera

A bayesian optimisation methodology for the inverse derivation of viscoplasticity model constants in high strain-rate simulations

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Defence Technology（防务技术） 2022年第9期18卷 1563-1577页

作者： shannon Ryan Julian Berk Santu Rana Brodie McDonald Svetha Venkatesh Applied Artificial Intelligence Institute(A^(2)I^(2)) Deakin University75 Pigdons RdWaurn PondsVIC3216Australia Defence Science and Technology Group 506 Lorimer StreetFishermans BendVIC3207Australia

We present an inverse methodology for deriving viscoplasticity constitutive model parameters for use in explicit finite element simulations of dynamic processes using functional experiments, i.e., those which provide value beyond that of constitutive model development. The developed methodology utilises Bayesian optimisation to minimise the error between experimental measurements and numerical simulations performed in LS-DYNA. We demonstrate the optimisation methodology using high hardness armour steels across three types of experiments that induce a wide range of loading conditions: ballistic penetration, rod-on-anvil, and near-field blast deformation. By utilising such a broad range of conditions for the optimisation, the resulting constitutive model parameters are generalised, i.e., applicable across the range of loading conditions encompassed the by those experiments(e.g., stress states, plastic strain magnitudes, strain rates, etc.). Model constants identified using this methodology are demonstrated to provide a generalisable model with superior predictive accuracy than those derived from conventional mechanical characterisation experiments or optimised from a single experimental condition.

关键词： Constitutive modelling Finite element Bayesian optimisation Finite element model updating

Who＇s your neighbor？ Acoustic cues to individual identity in red squirrel Tamiasciurus hudsonicus rattle calls

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Current Zoology 2012年第5期58卷 758-764页

作者： shannon M. DIGWEED Drew RENDALL Teana IMBEAU Departments of Psychology and Biological Science Grant MacEwan University Edmonton Alberta T5J 2P2 Canada Department of Psychology University of Lethbridge Lethbridge Alberta T1K 3M4 Canada Department of Biological Science Grant MacEwan University Edmonton Alberta T5J 2P2 Canada

North American red squirrels Tamiasciurus hudsonicus often produce a loud territorial rattle call when conspecifics enter or invade a territory. Previous playback experiments suggest that the territorial rattle call may indicate an invader＇s identity as squirrels responded more intensely to calls played from strangers than to calls played from neighbors. This dear-enemy effect is well known in a variety of bird and mammal species and functions to reduce aggressive interactions between known neighbors. However, although previous experiments on red squirrels suggest some form of individual differentiation and thus recognition, detailed acoustic analysis of potential acoustic cues in rattle calls have not been conducted. If calls function to aid in conspecific identifi- cation in order to mitigate aggressive territorial interactions, we would expect that individual recognition cues would be acoustically represented. Our work provides a detailed analysis of acoustic cues to identity within rattle calls. A total of 225 calls across 32 in- dividual squirrels from Sheep River Provincial Park, Kananaskis, AB, Canada, were analyzed with discriminant function analysis for potential acoustic cues to individual identity. Initial analysis of all individuals revealed a reliable acoustic differentiation across individuals. A more detailed analysis of clusters of neighboring squirrels was performed and results again indicated a statistically significant likelihood that calls were assigned correctly to specific squirrels （55%-75% correctly assigned）; in other words squirrels have distinct voices that should allow for individual identification and discrimination by conspecifics [Current Zoology 58 （5）： 758-764, 2012].

关键词： Vocal communication Territory calls Red squirrel Dear-enemy Individual identity

Shark scavenging behavior in the presence of competition

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Current Zoology 2010年第1期56卷 100-108页

作者： shannon P.GERRY Andrea J.SCOTT Department of Biological Sciences University of Rhode Island Department of Biological Sciences Wellesley College

The distribution of organisms within a community can often be determined by the degree of plasticity or degree of specialization of resource acquisition. Resource acquisition is often based on the morphology of an organism, behavior, or a combination of both. Performance tests of feeding can identify the possible interactions that allow one species to better exploit a prey item. Scavenging behaviors in the presence or absence of a competitor were investigated by quantifying prey selection in a trophic generalist, spiny dogfish Squalus acanthias, and atrophic specialist, smooth-hounds Mustelus canis, in order to determine if each shark scavenged according to its jaw morphology. The diet of dogfish consists of small fishes, squid, ctenophores, and bi- valves; they are expected to be nonselective predators. Smooth-hounds primarily feed on crustaceans; therefore, they are predicted to select crabs over other prey types. Prey selection was quantified by ranking each prey item according to the order it was consumed. Dietary shifts were analyzed by comparing the percentage of each prey item selected during solitary versus competitive scavenging. When scavenging alone, dogfish prefer herring and squid, which are easily handled by the cutting dentition of dogfish. Dogfish shift their diet to include a greater number of prey types when scavenging with a competitor. Smooth-hounds scavenge on squid, herring, and shrimp when alone, but increase the number of crabs in the diet when scavenging competitively. Competition causes smooth-hounds to scavenge according to their jaw morphology and locomotor abilities, which enables them to feed on a specialized resource [Current Zoology 56 （1）： 100-108 2010].

关键词： Optimal foraging theory Prey selection Squalus acanthias Mustelus canis

Taxon sampling and the accuracy of phylogenetic analyses

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植物分类学报 2008年第3期46卷 239-257页

作者： Tracy A. HEATH shannon M. HEDTKE David M. HILLIS Section of lntegrative Biology and Center for Computational Biology and Bioinformatics One University Station C0930 The University of Texas Austin TX 78712 USA

Appropriate and extensive taxon sampling is one of the most important determinants of accurate phylogenetic estimation. In addition, accuracy of inferences about evolutionary processes obtained from phyloge-netic analyses is improved significantly by thorough taxon sampling efforts. Many recent efforts to improve phylogenetic estimates have focused instead on increasing sequence length or the number of overall characters in the analysis, and this often does have a beneficial effect on the accuracy of phylogenetic analyses. However, phylogenetic analyses of few taxa (but each represented by many characters) can be subject to strong systematic biases, which in turn produce high measures of repeatability (such as bootstrap proportions) in support of incor-rect or misleading phylogenetic results. Thus, it is important for phylogeneticists to consider both the sampling of taxa, as well as the sampling of characters, in designing phylogenetic studies. Taxon sampling also improves estimates of evolutionary parameters derived from phylogenetic trees, and is thus important for improved applica-tions of phylogenetic analyses. Analysis of sensitivity to taxon inclusion, the possible effects of long-branch attraction, and sensitivity of parameter estimation for model-based methods should be a part of any careful and thorough phylogenetic analysis. Furthermore, recent improvements in phylogenetic algorithms and in computa-tional power have removed many constraints on analyzing large, thoroughly sampled data sets. Thorough taxon sampling is thus one of the most practical ways to improve the accuracy of phylogenetic estimates, as well as the accuracy of biological inferences that are based on these phylogenetic trees.

关键词：动物物种分类方法取样方法