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Select item 113616421.

FOXC1 and FOXC2 regulate growth plate chondrocyte maturation towards hypertrophy in the embryonic mouse limb skeleton

Asra Almubarak, Qiuwan Zhang, Cheng-Hai Zhang, Noor Abdelwahab, Tsutomu Kume, Andrew B. Lassar, Fred B. Berry

Development. 2024 Aug 15; 151(16): dev202798. Published online 2024 Aug 22. doi: 10.1242/dev.202798

PMCID:: PMC11361642

Abstract Article

Select item 113189952.

Characterization of Novel PHEX Variants in X-linked Hypophosphatemic Rickets and Genotype-PHEX Activity Correlation

Huixiao Wu, Hui Ying, Wanyi Zhao, Yan Sun, Yanzhou Wang, Xinyu Chen, Guimei Li, Yangyang Yao, Shuo Xu, Tianyou Li, Li Fang, Xiaoqing Sun, Ning Wang, Jin Xu, Qingbo Guan, Weibo Xia, Li Wang, Ling Gao, Jiajun Zhao, Chao Xu

J Clin Endocrinol Metab. 2024 Sep; 109(9): 2242–2255. Published online 2024 Mar 5. doi: 10.1210/clinem/dgae120

PMCID:: PMC11318995

Abstract Article

Select item 108393423.

A PAI‐1 antagonist ameliorates hypophosphatemia in the Hyp vitamin D‐resistant rickets model mouse

Cheng Qian, Nobuaki Ito, Kunikazu Tsuji, Shingo Sato, Katsushi Kikuchi, Toshitaka Yoshii, Toshio Miyata, Yoshinori Asou

FEBS Open Bio. 2024 Feb; 14(2): 290–299. Published online 2023 Dec 25. doi: 10.1002/2211-5463.13745

PMCID:: PMC10839342

Abstract Article PDF–914K

Select item 107326784.

Impaired Growth Plate Maturation in XLH Is due to Both Excess FGF23 and Decreased 1,25-Dihydroxyvitamin D Signaling

Prem Swaroop Yadav, Margaret M Kobelski, Janaina S Martins, Tao Tao, Eva S Liu, Marie B Demay

Endocrinology. 2024 Jan; 165(1): bqad186. Published online 2023 Dec 8. doi: 10.1210/endocr/bqad186

PMCID:: PMC10732678

Abstract Article

Select item 105623825.

Sclerostin antibody improves alveolar bone quality in the Hyp mouse model of X-linked hypophosphatemia (XLH)

Kelsey A. Carpenter, Delia O. Alkhatib, Bryan A. Dulion, Elizabeth Guirado, Shreya Patel, Yinghua Chen, Anne George, Ryan D. Ross

Int J Oral Sci. 2023; 15: 47. Published online 2023 Oct 10. doi: 10.1038/s41368-023-00252-1

PMCID:: PMC10562382

Abstract Article PDF–2.4M

Select item 105442166.

Impaired 1,25-dihydroxyvitamin D₃ action underlies enthesopathy development in the Hyp mouse model of X-linked hypophosphatemia

Rakshya Rana, Jiana T. Baker, Melissa Sorsby, Supriya Jagga, Shreya Venkat, Shaza Almardini, Eva S. Liu

JCI Insight. 2023 Sep 8; 8(17): e163259. Published online 2023 Sep 8. doi: 10.1172/jci.insight.163259

PMCID:: PMC10544216

Abstract Article PDF–35M

Select item 105299697.

Contributions of increased osteopontin and hypophosphatemia to dentoalveolar defects in osteomalacic Hyp mice

Fatma F Mohamed, Betty Hoac, Aonjittra Phanrungsuwan, Michelle H. Tan, Priscila Alves Giovani, Sana Ghiba, Monzur Murshed, Brian L Foster, Marc D McKee

Bone. Author manuscript; available in PMC 2024 Nov 1.

Published in final edited form as: Bone. 2023 Nov; 176: 116886. Published online 2023 Aug 25. doi: 10.1016/j.bone.2023.116886

PMCID:: PMC10529969

Abstract Article PDF–5.1M

Select item 97147888.

Spatial metabolomics reveals upregulation of several pyrophosphate-producing pathways in cortical bone of Hyp mice

Achim Buck, Verena M. Prade, Thomas Kunzke, Reinhold G. Erben, Axel Walch

JCI Insight. 2022 Oct 24; 7(20): e162138. Published online 2022 Oct 24. doi: 10.1172/jci.insight.162138

PMCID:: PMC9714788

Abstract Article PDF–9.6M

Select item 96742179.

SOX9 and SOX10 control fluid homeostasis in the inner ear for hearing through independent and cooperative mechanisms

Irene Y. Y. Szeto, Daniel K. H. Chu, Peikai Chen, Ka Chi Chu, Tiffany Y. K. Au, Keith K. H. Leung, Yong-Heng Huang, Sarah L. Wynn, Angel C. Y. Mak, Ying-Shing Chan, Wood Yee Chan, Ralf Jauch, Bernd Fritzsch, Mai Har Sham, Robin Lovell-Badge, Kathryn S. E. Cheah

Proc Natl Acad Sci U S A. 2022 Nov 15; 119(46): e2122121119. Published online 2022 Nov 7. doi: 10.1073/pnas.2122121119

PMCID:: PMC9674217

Abstract Article PDF–2.7M

Select item 940725310.

PHEX^L222P Mutation Increases Phex Expression in a New ENU Mouse Model for XLH Disease

Carole El Hakam, Alexis Parenté, Fabienne Baraige, Laetitia Magnol, Lionel Forestier, Florent Di Meo, Véronique Blanquet

Genes (Basel) 2022 Aug; 13(8): 1356. Published online 2022 Jul 28. doi: 10.3390/genes13081356

PMCID:: PMC9407253

Abstract Article PDF–11M

Select item 938150411.

Spatiotemporal single-cell regulatory atlas reveals neural crest lineage diversification and cellular function during tooth morphogenesis

Junjun Jing, Jifan Feng, Yuan Yuan, Tingwei Guo, Jie Lei, Fei Pei, Thach-Vu Ho, Yang Chai

Nat Commun. 2022; 13: 4803. Published online 2022 Aug 16. doi: 10.1038/s41467-022-32490-y

PMCID:: PMC9381504

Abstract Article PDF–6.6M

Select item 916306212.

Function of PHEX mutations p.Glu145* and p.Trp749Arg in families with X-linked hypophosphatemic rickets by the negative regulation mechanism on FGF23 promoter transcription

Yu-mian Gan, Yan-ping Zhang, Dan-dan Ruan, Jian-bin Huang, Yao-bin Zhu, Xin-fu Lin, Xiao-ping Xiao, Qiong Cheng, Zhen-bo Geng, Li-sheng Liao, Fa-qiang Tang, Jie-wei Luo

Cell Death Dis. 2022 Jun; 13(6): 518. Published online 2022 Jun 2. doi: 10.1038/s41419-022-04969-5

PMCID:: PMC9163062

Abstract Article PDF–3.8M

Select item 868820013.

Conductive hearing loss in the Hyp mouse model of X-linked hypophosphatemia is accompanied by hypomineralization of the auditory ossicles

Maximilian M. Delsmann, Richard Seist, Julian Stürznickel, Felix N. Schmidt, Amer Mansour, Margaret M. Kobelski, Gabriel Broocks, Jonathan Peichl, Ralf Oheim, Mark Praetorius, Thorsten Schinke, Michael Amling, Marie B. Demay, Konstantina M. Stankovic, Tim Rolvien

J Bone Miner Res. Author manuscript; available in PMC 2022 Dec 1.

Published in final edited form as: J Bone Miner Res. 2021 Dec; 36(12): 2317–2328. Published online 2021 Oct 4. doi: 10.1002/jbmr.4443

PMCID:: PMC8688200

Abstract Article PDF–2.2M

Select item 867124914.

Sclerostin antibody improves phosphate metabolism hormones, bone formation rates, and bone mass in adult Hyp mice

Kelsey A. Carpenter, Reid Davison, Shruti Shakthivel, Kyle D. Anderson, Frank C. Ko, Ryan D. Ross

Bone. Author manuscript; available in PMC 2023 Jan 1.

Published in final edited form as: Bone. 2022 Jan; 154: 116201. Published online 2021 Sep 16. doi: 10.1016/j.bone.2021.116201

PMCID:: PMC8671249

Abstract Article PDF–1.4M

Select item 831502915.

Osteocytes but not osteoblasts directly build mineralized bone structures

Ke Wang, Yinshi Ren, Shuxian Lin, Yan Jing, Chi Ma, Jun Wang, X. Baozhi Yuan, Xianglong Han, Hu Zhao, Zheng Wang, Minghao Zheng, Yin Xiao, Lin Chen, Bjorn Reino Olsen, Jian Q. Feng

Int J Biol Sci. 2021; 17(10): 2430–2448. Published online 2021 Jun 11. doi: 10.7150/ijbs.61012

PMCID:: PMC8315029

Abstract Article PDF–8.5M

Select item 825603616.

Stat3 loss in mesenchymal progenitors causes Job syndrome–like skeletal defects by reducing Wnt/β-catenin signaling

Prem Swaroop Yadav, Shuhao Feng, Qian Cong, Hanjun Kim, Yuchen Liu, Yingzi Yang

Proc Natl Acad Sci U S A. 2021 Jun 29; 118(26): e2020100118. Published online 2021 Jun 25. doi: 10.1073/pnas.2020100118

PMCID:: PMC8256036

Abstract Article PDF–3.8M

Select item 815893017.

Impaired 1,25 dihydroxyvitamin D₃ action and hypophosphatemia underlie the altered lacuno-canalicular remodeling observed in the Hyp mouse model of XLH

Ye Yuan, Supriya Jagga, Janaina S. Martins, Rakshya Rana, Paola Divieti Pajevic, Eva S. Liu

PLoS One. 2021; 16(5): e0252348. Published online 2021 May 27. doi: 10.1371/journal.pone.0252348

PMCID:: PMC8158930

Abstract Article PDF–3.5M

Select item 796977618.

Renal Dnase1 expression is regulated by FGF23 but loss of Dnase1 does not alter renal phosphate handling

Daniela Egli-Spichtig, Martin Y. H. Zhang, Alfred Li, Eva Maria Pastor Arroyo, Nati Hernando, Carsten A. Wagner, Wenhan Chang, Farzana Perwad

Sci Rep. 2021; 11: 6175. Published online 2021 Mar 17. doi: 10.1038/s41598-021-84735-3

PMCID:: PMC7969776

Abstract Article PDF–2.1M

Select item 793877019.

BraInMap elucidates the macromolecular connectivity landscape of mammalian brain

Reza Pourhaghighi, Peter E. A. Ash, Sadhna Phanse, Florian Goebels, Lucas Z. M. Hu, Siwei Chen, Yingying Zhang, Shayne D. Wierbowski, Samantha Boudeau, Mohamed T. Moutaoufik, Ramy H. Malty, Edyta Malolepsza, Kalliopi Tsafou, Aparna Nathan, Graham Cromar, Hongbo Guo, Ali Al Abdullatif, Daniel J. Apicco, Lindsay A. Becker, Aaron D. Gitler, Stefan M. Pulst, Ahmed Youssef, Ryan Hekman, Pierre C. Havugimana, Carl A. White, Benjamin C. Blum, Antonia Ratti, Camron D. Bryant, John Parkinson, Kasper Lage, Mohan Babu, Haiyuan Yu, Gary D. Bader, Benjamin Wolozin, Andrew Emili

Cell Syst. Author manuscript; available in PMC 2021 Apr 22.

Published in final edited form as: Cell Syst. 2020 Apr 22; 10(4): 333–350.e14. doi: 10.1016/j.cels.2020.03.003

Correction in:: Cell Syst. 2020 Aug 26; 11(2): 208.

PMCID:: PMC7938770

Abstract Article PDF–2.0M

Select item 793033620.

Impact of Early Conventional Treatment on Adult Bone and Joints in a Murine Model of X-Linked Hypophosphatemia

Axelle Cauliez, Volha V. Zhukouskaya, Stéphane Hilliquin, Jérémy Sadoine, Lotfi Slimani, Corinne Miceli-Richard, Karine Briot, Agnès Linglart, Catherine Chaussain, Claire Bardet

Front Cell Dev Biol. 2020; 8: 591417. Published online 2021 Feb 18. doi: 10.3389/fcell.2020.591417

PMCID:: PMC7930336

Abstract Article PDF–12M

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