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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.
Karolina Storesund Francine Amon Anne Steen‐Hansen Shayesteh Haghighatpanah Ida Larsson 《火与材料》2021,45(1):181-190
The aim of this exploratory study has been to investigate the fire properties and environmental aspects of different upholstery material combinations, mainly for domestic applications. An analysis of the sustainability and circularity of selected textiles, along with lifecycle assessment, is used to qualitatively evaluate materials from an environmental perspective. The cone calorimeter was the primary tool used to screen 20 different material combinations from a fire performance perspective. It was found that textile covers of conventional fibres such as wool, cotton and polyester, can be improved by blending them with fire resistant speciality fibres. A new three‐dimensional web structure has been examined as an alternative padding material, showing preliminary promising fire properties with regard to ignition time, heat release rates and smoke production. 相似文献
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7.
《International Journal of Hydrogen Energy》2022,47(2):1083-1091
α-Ni(OH)2 is a promising candidate of the currently commercialized β-Ni(OH)2 due to its higher theoretical discharge capacity in alkaline solution; however, its instability and poor conductivity plague the practical application. Herein, we propose α-Ni(OH)2 with Co doping and spherical structure to strengthen the stability and enhance the conductivity and use it as the cathode for nickel-metal hydride batteries. Studies show that proper Co doping promotes the electrochemical reaction between the active materials and the electrolyte due to the spherical α-Ni(OH)2 with enlarged interlayer distance and abundant hole channels, as well as high conductivity of Co, therefore, the obtained spherical α-Ni(OH)2 with 7 mol% Co doping delivers significantly improved discharge capability, which is 384.6 mAh g?1 at 70 mA g?1 (0.2 C), increased by 54.3 mAh g?1 compared with pure α-Ni(OH)2, and at a high current of 5 C, it still gives 269.4 mAh g?1, in contrast 218.5 mA g?1 for the pure α-Ni(OH)2. Besides, the cycling stability of the α-Ni(OH)2 with 7 mol% Co doping maintains 340 cycles at a capacity retention of 80% (1C), which is extended 110 cycles in contrast to the pure α-Ni(OH)2. These results provide the underpinning platform of α-Ni(OH)2 for battery applications with high discharge ability and cycle life. 相似文献
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.
《International Journal of Hydrogen Energy》2022,47(29):13887-13899
We propose a self-sustaining power supply system consisting of a “Hybrid Energy Storage System (HESS)” and renewable energy sources to ensure a stable supply of high-quality power in remote islands. The configuration of the self-sustaining power supply system that can utilize renewable energy sources effectively on remote islands where the installation area is limited is investigated. It is found that it is important to select renewable energy sources whose output power curve is close to the load curve to improve the efficiency of the system. The operation methods that can increase the cost-effectiveness of the self-sustaining power supply system are also investigated. It is clarified that it is important for increasing the cost effectiveness of the self-sustaining power supply system to operate the HESS with a smaller capacity of its components by setting upper limits on the output power of the renewable energy sources and cutting the infrequent generated power. 相似文献