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1.
Sialidosis, caused by a genetic deficiency of the lysosomal sialidase gene (NEU1), is a systemic disease involving various tissues and organs, including the nervous system. Understanding the neurological dysfunction and pathology associated with sialidosis remains a challenge, partially due to the lack of a human model system. In this study, we have generated two types of induced pluripotent stem cells (iPSCs) with sialidosis-specific NEU1G227R and NEU1V275A/R347Q mutations (sialidosis-iPSCs), and further differentiated them into neural precursor cells (iNPCs). Characterization of NEU1G227R- and NEU1V275A/R347Q- mutated iNPCs derived from sialidosis-iPSCs (sialidosis-iNPCs) validated that sialidosis-iNPCs faithfully recapitulate key disease-specific phenotypes, including reduced NEU1 activity and impaired lysosomal and autophagic function. In particular, these cells showed defective differentiation into oligodendrocytes and astrocytes, while their neuronal differentiation was not notably affected. Importantly, we found that the phenotypic defects of sialidosis-iNPCs, such as impaired differentiation capacity, could be effectively rescued by the induction of autophagy with rapamycin. Our results demonstrate the first use of a sialidosis-iNPC model with NEU1G227R- and NEU1V275A/R347Q- mutation(s) to study the neurological defects of sialidosis, particularly those related to a defective autophagy–lysosome pathway, and may help accelerate the development of new drugs and therapeutics to combat sialidosis and other LSDs. 相似文献
2.
Yujing Zhang Pin Liu Kumara Raja Kandula Wenru Li Yanfeng Qin Haibo Zhang Guangzu Zhang 《Journal of the European Ceramic Society》2021,41(1):360-367
The applications of antiferroelectric (AFE) materials in miniaturized and integrated electronic devices are limited by their low energy density. To address the above issue, the antiferroelectricity of the reinforced material was designed to improve its AFE-ferroelectric (FE) phase transition under electric fields. In this present study, the composition of Zr4+ (0.72 Å) and Ti4+ (0.605 Å) at B-site of Pb0.97La0.02(ZrxSn0.05Ti0.95-x)O3 ceramics with orthogonal reflections are synthesized via the tape-casting method. These ceramics are modified to enhance their antiferroelectricity by reducing their tolerance factor. A recoverable energy storage density Wrec 12.1 J/cm3 was obtained for x = 0.93 under 376 kV/cm, which is superior value than reported until now in lead-based energy storage systems. Moreover, the discharge energy density can reach 10.23 J/cm3, and 90 % of which can be released within 5.66 μs. This work provides a new window and potential materials for further industrialization of pulse power capacitors. 相似文献
3.
Novel lead-free (1-x)Ba0·9Ca0·1Ti0·9Zr0·1O3-xSrNb2O6 ceramics were synthesized via a two-step high energy ball milling process. The evolution of microstructural properties, phase transformation, and energy storage characteristics was comprehensively investigated to assess the applicability of material in multi-layered ceramic capacitors. The substitution of SrNb2O6 (SNO) in Ba0·9Ca0·1Ti0·9Zr0·1O3 (BTCZ) has resulted in substantial improvement in materials density along with a small increase in the grain size of the synthesized ceramic. A thorough microstructural investigation indicates an excellent dispersibility and compatibility between BTCZ and SNO phases. With an increase in SNO substitution, a transition from typical ferroelectric to relaxor ferroelectric has been observed, which has led to a significantly slimmer ferroelectric loop along with frequency dispersive dielectric properties. The optimized composition (i.e., x = 0.10) exhibits an ultra-high recoverable energy density of 2.68 J/cm3 along with a moderately high energy efficiency of 83.4%. Further, SNO substituted samples have also shown an enhancement in breakdown strength. The improvement in energy storage performance and breakdown strength of SNO substituted BTCZ composites are mainly attributed to relatively homogeneous grain morphology, optimum grain size, microstructural density, and improved grain boundary interface. 相似文献
4.
为推进病历数字化发展,并确保其信息的安全性,将以HIS电子病历系统为基础,采用安信数字签名技术和PKI或PMI系统搭建相信并加以任用的授权服务,经过针对实际的PKC和CA的确认、委托与管控构建整体的数字签名平台,完成电子病历数字签名功能设计,以加强电子文件的完整性、真实性和不可抵赖性。最后以某医院的XML结构化的电子病历系统为基础进行项目实施,完成了医护人员通过HIS的快速身份认证,和准确地数字签名。 相似文献
5.
In the past, thinking of carrying electronic devices inside our bodies was only posed by non-real scenarios. The emergence of insertable devices has changed this. Since this technology is still in its initial development stages, few studies have investigated factors that influence its acceptance. This paper analyzes the predictors of the intention to use non-medical insertable devices in two Latin American contexts. We used partial least squares structural equation modeling to examine whether six constructs predicted intention to use insertable devices. A questionnaire was administered to undergraduate students located in Colombia and Chile (n = 672). We also examined whether these predictors influenced intention differently for both of them. Four common constructs significantly and positively influenced both Chilean and Colombian respondents to use insertable devices (hedonic motivation, habit, performance expectancy, and social influence). Also, the habit has a complementary mediating effect on the relationship between social influence and behavioral intention. By contrast, effort expectations were a positive and significant predictor, but only among Chilean respondents. Findings suggest that when technologies are emerging, well-known predictors of intention (e.g., performance and effort expectations) are less influential than predictors related to self-efficacy (e.g., habit and hedonic motivation). The use of insertable devices has a significant impact on society. Thus, a better understanding of what motivates their use has implications for both academia and industry. 相似文献
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8.
《International Journal of Hydrogen Energy》2022,47(3):1752-1764
Subsurface hydrogen (H2) storage in geological formations is of growing interest for decarbonization. However, there is a knowledge gap in understanding the multiphase flow involved in this process, which can have a significant impact on the recovery performance of H2. Therefore, a full-compositional modeling study was conducted to analyze potential issues and to understand the fundamental hydrodynamic mechanisms of H2 storage. We performed a range of 2D vertical simulations at the decametre scale with a very fine cell size (0.1 m) to observe the detailed flow behaviour of H2 with carbon dioxide (CO2) as cushion gas in various flow regimes. Issues such as viscous instability, capillary bypassing, gas trapping and gravity segregation are analysed here. To generalize our calculations, we have validated and applied the scaling theory in the context of subsurface H2 storage. Since this study is focused on the hydrodynamic behaviour, three dimensionless groups, including aspect factor, capillary/viscous ratio and gravity/viscous ratio were identified to correlate recovery performance between various scales in a fixed heterogeneous system. It was found that H2 could infiltrate the cushion gas in the proximity of the injectors, meaning that CO2 is not displaced away from the injectors in a piston-like fashion. As a result, the purity of the back produced H2 is much degraded, particularly in a viscous-dominated scenario. On the other hand, the injected H2 mostly accumulates at the top forming a highly restricted mixing zone with CO2 in the gravity-dominated case. The recovery performance is therefore much improved in this case. Although the gas distribution can be significantly altered by capillary forces leading to bypassed zones, the recovery performance of H2 is hardly influenced. This is because the back-produced H2 recovery is not dependent on the sweep efficiency of the gas. H2 can be back produced following the same paths which were formed during injection. 相似文献
9.
《International Journal of Hydrogen Energy》2022,47(16):9829-9834
A recent commentary by Santhosh and Ravindran on our paper (Int. J. Hydrogen Energy 2014, 39:10,606) demonstrated that the interaction between H2 and MXene (Sc2C and Ti2C) phases are not Kubas-type and should be of weak physisorption, and thus made a conclusion that 2D Sc2C and Ti2C are not suitable for practical hydrogen storage applications. In this responses, we recalculated hydrogen adsorption on 2D Sc2C and Ti2C by using different exchange-correlation functionals. And based on the calculated results, bare MXenes (especially the Ti2C) are suitable as hydrogen storage materials at temperatures of several tens degrees lower than room temperature. And the hydrogen adsorptions on the MXenes terminated with oxygen group were also investigated. Among the Ti2C, Sc2C and their oxygen-functional counterparts, the binding energy of H2 on Sc2CO2 surface is the closest to the ideal range of 0.16–0.42 eV/H2 at ambient conditions, and thus the Sc2C with oxygen group is expected to be more suitable as hydrogen storage materials. 相似文献
10.
张延盛 《电信工程技术与标准化》2022,35(5):7-10,26
随着通信技术的快速发展及智能设备的普及,网络规模、网元数量的不断增长,通信设备巡检任务成倍增加,给巡检系统的存储带来了巨大挑战,传统文件存储方式已无法满足要求。通过研究分布式对象存储,基于MinIO设计实现了一种新的巡检系统存储方案,可有效提升系统存储效率和可扩展性。该方案已经在江苏移动实际应用,支撑常态化巡检工作,对于网络运维及管理有较好的参考意义。 相似文献