Low-density lipoprotein receptor-related protein 1(LRP1)is a multifunctional endocytic receptor whose dysfunction is linked to developmental dysplasia of the hip,osteoporosis and osteoarthritis.Our work addresses the ...Low-density lipoprotein receptor-related protein 1(LRP1)is a multifunctional endocytic receptor whose dysfunction is linked to developmental dysplasia of the hip,osteoporosis and osteoarthritis.Our work addresses the critical question of how these skeletal pathologies emerge.Here,we show the abundant expression of LRP1 in skeletal progenitor cells at mouse embryonic stage E10.5 and onwards,especially in the perichondrium,the stem cell layer surrounding developing limbs essential for bone formation.Lrp1 deficiency in these stem cells causes joint fusion,malformation of cartilage/bone template and markedly delayed or lack of primary ossification.展开更多
This review summarizes the rapporteur report on tropical cyclone(TC)intensity change from the operational perspective,as presented to the 10th International Workshop on TCs(IWTC-10)held in Bali,Indonesia,from Dec.5–9...This review summarizes the rapporteur report on tropical cyclone(TC)intensity change from the operational perspective,as presented to the 10th International Workshop on TCs(IWTC-10)held in Bali,Indonesia,from Dec.5–9,2022.The accuracy of TC intensity forecasts issued by operational forecast centers depends on three aspects:real-time observations,TC dynamical model forecast guidance,and techniques and methods used by forecasters.The rapporteur report covers the progress made over the past four years(2018–2021)in all three aspects.This review focuses on the progress of dynamical model forecast guidance.The companion paper(Part II)summarizes the advance from operational centers.The dynamical model forecast guidance continues to be the main factor leading to the improvement of operational TC intensity forecasts.Here,we describe recent advances and developments of major operational regional dynamical TC models and their intensity forecast performance,including HWRF,HMON,COAMPS-TC,Met Office Regional Model,CMA-TYM,and newly developed HAFS.The performance of global dynamical models,including NOAA's GFS,Met Office Global Model(MOGM),JMA's GSM,and IFS(ECMWF),has also been improved in recent years due to their increased horizontal and vertical resolution as well as improved data assimilation systems.Recent challenging cases of rapid intensification are presented and discussed.展开更多
基金The Andor dragonfly Spinning Disk microscope in the CCI was funded by the BBSRC(BB/R01390X/1)This work was supported by the ministry of education of the Kingdom of Saudi Arabia(to M.Alhashmi)+6 种基金Libyan Ministry of Higher Education and Scientific Research and ECMage(to A.M.E.Gremida)Qatar National Research Fund(to N.A.Al-Maslamani)European Union’s Horizon 2020 research and innovation programme under the Marie Skłodowska-Curie grant agreement(860635 to M.Antonaci and A.Kerr)BBSRC Grants(BB/T00715X/1 to S.K.Maharana and G.N.WheelerBB/X000907/1 to D.A.Turner)Versus Arthritis Career Development Fellowship(21447 to K.Yamamoto)Versus Arthritis Bridging Fellowship(23137 to K.Yamamoto).
文摘Low-density lipoprotein receptor-related protein 1(LRP1)is a multifunctional endocytic receptor whose dysfunction is linked to developmental dysplasia of the hip,osteoporosis and osteoarthritis.Our work addresses the critical question of how these skeletal pathologies emerge.Here,we show the abundant expression of LRP1 in skeletal progenitor cells at mouse embryonic stage E10.5 and onwards,especially in the perichondrium,the stem cell layer surrounding developing limbs essential for bone formation.Lrp1 deficiency in these stem cells causes joint fusion,malformation of cartilage/bone template and markedly delayed or lack of primary ossification.
文摘This review summarizes the rapporteur report on tropical cyclone(TC)intensity change from the operational perspective,as presented to the 10th International Workshop on TCs(IWTC-10)held in Bali,Indonesia,from Dec.5–9,2022.The accuracy of TC intensity forecasts issued by operational forecast centers depends on three aspects:real-time observations,TC dynamical model forecast guidance,and techniques and methods used by forecasters.The rapporteur report covers the progress made over the past four years(2018–2021)in all three aspects.This review focuses on the progress of dynamical model forecast guidance.The companion paper(Part II)summarizes the advance from operational centers.The dynamical model forecast guidance continues to be the main factor leading to the improvement of operational TC intensity forecasts.Here,we describe recent advances and developments of major operational regional dynamical TC models and their intensity forecast performance,including HWRF,HMON,COAMPS-TC,Met Office Regional Model,CMA-TYM,and newly developed HAFS.The performance of global dynamical models,including NOAA's GFS,Met Office Global Model(MOGM),JMA's GSM,and IFS(ECMWF),has also been improved in recent years due to their increased horizontal and vertical resolution as well as improved data assimilation systems.Recent challenging cases of rapid intensification are presented and discussed.