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Select item 104404331.

Brain cell type specific proteomics approach to discover pathological mechanisms in the childhood CNS disorder mucolipidosis type IV

Madison Sangster, Sanjid Shahriar, Zachary Niziolek, Maria Carla Carisi, Michael Lewandowski, Bogdan Budnik, Yulia Grishchuk

Front Mol Neurosci. 2023; 16: 1215425. Published online 2023 Aug 7. doi: 10.3389/fnmol.2023.1215425

PMCID:: PMC10440433

Abstract Article PDF–3.4M

Select item 98838052.

Exosomal release through TRPML1-mediated lysosomal exocytosis is required for adipogenesis

Mi Seong Kim, Shmuel Muallem, Sung Hyun Kim, Kang Beom Kwon, Min Seuk Kim

Biochem Biophys Res Commun. Author manuscript; available in PMC 2023 Jan 28.

Published in final edited form as: Biochem Biophys Res Commun. 2019 Mar 12; 510(3): 409–415. Published online 2019 Jan 31. doi: 10.1016/j.bbrc.2019.01.115

PMCID:: PMC9883805

Abstract Article PDF–1.2M

Select item 98699313.

Impaired autophagic flux and dedifferentiation in podocytes lacking Asah1 gene: Role of lysosomal TRPML1 channel

Guangbi Li, Dandan Huang, Yao Zou, Jason Kidd, Todd W.B. Gehr, Ningjun Li, Joseph K. Ritter, Pin-Lan Li

Biochim Biophys Acta Mol Cell Res. Author manuscript; available in PMC 2023 Jan 23.

Published in final edited form as: Biochim Biophys Acta Mol Cell Res. 2023 Jan; 1870(1): 119386. Published online 2022 Oct 24. doi: 10.1016/j.bbamcr.2022.119386

PMCID:: PMC9869931

Abstract Article PDF–1.7M

Select item 98615944.

TRPML1-Induced Lysosomal Ca²⁺ Signals Activate AQP2 Translocation and Water Flux in Renal Collecting Duct Cells

Simona Ida Scorza, Serena Milano, Ilenia Saponara, Maira Certini, Roberta De Zio, Maria Grazia Mola, Giuseppe Procino, Monica Carmosino, Francesco Moccia, Maria Svelto, Andrea Gerbino

Int J Mol Sci. 2023 Jan; 24(2): 1647. Published online 2023 Jan 13. doi: 10.3390/ijms24021647

PMCID:: PMC9861594

Abstract Article PDF–7.5M

Select item 96739245.

Targeting MCOLN1/TRPML1 channels to protect against ischemia-reperfusion injury by restoring the inhibited autophagic flux in cardiomyocytes

Zhongheng Sui, Meng-Meng Wang, Yanhong Xing, Jiansong Qi, Wuyang Wang

Autophagy. 2022; 18(12): 3053–3055. Published online 2022 May 6. doi: 10.1080/15548627.2022.2072657

PMCID:: PMC9673924

Abstract Article PDF–761K

Select item 88865306.

LAMTOR1 inhibition of TRPML1‐dependent lysosomal calcium release regulates dendritic lysosome trafficking and hippocampal neuronal function

Jiandong Sun, Yan Liu, Xiaoning Hao, Weiju Lin, Wenyue Su, Emerald Chiang, Michel Baudry, Xiaoning Bi

EMBO J. 2022 Mar 1; 41(5): e108119. Published online 2022 Jan 31. doi: 10.15252/embj.2021108119

PMCID:: PMC8886530

Abstract Article PDF–4.8M

Select item 87602767.

Lung emphysema and impaired macrophage elastase clearance in mucolipin 3 deficient mice

Barbara Spix, Elisabeth S. Butz, Cheng-Chang Chen, Anna Scotto Rosato, Rachel Tang, Aicha Jeridi, Veronika Kudrina, Eva Plesch, Philipp Wartenberg, Elisabeth Arlt, Daria Briukhovetska, Meshal Ansari, Gizem Günes Günsel, Thomas M. Conlon, Amanda Wyatt, Sandra Wetzel, Daniel Teupser, Lesca M. Holdt, Fabien Ectors, Ingrid Boekhoff, Ulrich Boehm, Jaime García-Añoveros, Paul Saftig, Martin Giera, Sebastian Kobold, Herbert B. Schiller, Susanna Zierler, Thomas Gudermann, Christian Wahl-Schott, Franz Bracher, Ali Önder Yildirim, Martin Biel, Christian Grimm

Nat Commun. 2022; 13: 318. Published online 2022 Jan 14. doi: 10.1038/s41467-021-27860-x

PMCID:: PMC8760276

Abstract Article PDF–3.8M

Select item 81639858.

Regulation of TRPML1 channel activity and inflammatory exosome release by endogenously produced reactive oxygen species in mouse podocytes

Guangbi Li, Dandan Huang, Ningjun Li, Joseph K. Ritter, Pin-Lan Li

Redox Biol. 2021 Jul; 43: 102013. Published online 2021 May 16. doi: 10.1016/j.redox.2021.102013

PMCID:: PMC8163985

Abstract Article PDF–7.2M

Select item 81187129.

Lipid-gated monovalent ion fluxes regulate endocytic traffic and support immune surveillance

Spencer A. Freeman, Stefan Uderhardt, Amra Saric, Richard F. Collins, Catherine M. Buckley, Sivakami Mylvaganam, Parastoo Boroumand, Jonathan Plumb, Ronald N. Germain, Dejian Ren, Sergio Grinstein

Science. Author manuscript; available in PMC 2021 May 13.

Published in final edited form as: Science. 2020 Jan 17; 367(6475): 301–305. Published online 2019 Dec 5. doi: 10.1126/science.aaw9544

PMCID:: PMC8118712

Abstract Article PDF–1.5M

Select item 777012210.

Abnormal podocyte TRPML1 channel activity and exosome release in mice with podocyte-specific Asah1 gene deletion

Guangbi Li, Dandan Huang, Owais M. Bhat, Justin L. Poklis, Aolin Zhang, Yao Zou, Jason Kidd, Todd W.B. Gehr, Pin-Lan Li

Biochim Biophys Acta Mol Cell Biol Lipids. Author manuscript; available in PMC 2022 Feb 1.

Published in final edited form as: Biochim Biophys Acta Mol Cell Biol Lipids. 2021 Feb; 1866(2): 158856. Published online 2020 Nov 19. doi: 10.1016/j.bbalip.2020.158856

PMCID:: PMC7770122

Abstract Article PDF–2.1M

Select item 772019311.

The intracellular Ca²⁺ release channel TRPML1 regulates lower urinary tract smooth muscle contractility

Caoimhin S. Griffin, Michael G. Alvarado, Evan Yamasaki, Bernard T. Drumm, Vivek Krishnan, Sher Ali, Eleanor M. Nagle, Kenton M. Sanders, Scott Earley

Proc Natl Acad Sci U S A. 2020 Dec 1; 117(48): 30775–30786. Published online 2020 Nov 16. doi: 10.1073/pnas.2016959117

PMCID:: PMC7720193

Abstract Article PDF–1.8M

Select item 740632812.

Early evidence of delayed oligodendrocyte maturation in the mouse model of mucolipidosis type IV

Molly Mepyans, Livia Andrzejczuk, Jahree Sosa, Sierra Smith, Shawn Herron, Samantha DeRosa, Susan A. Slaugenhaupt, Albert Misko, Yulia Grishchuk, Kirill Kiselyov

Dis Model Mech. 2020 Jul 1; 13(7): dmm044230. Published online 2020 Jul 30. doi: 10.1242/dmm.044230

PMCID:: PMC7406328

Abstract Article

Select item 739786013.

TRPML1 Channels Initiate Ca²⁺ Sparks in Vascular Smooth Muscle Cells

Pratish Thakore, Harry A. T. Pritchard, Caoimhin S. Griffin, Evan Yamasaki, Bernard T. Drumm, Conor Lane, Kenton M. Sanders, Yumei Feng Earley, Scott Earley

Sci Signal. Author manuscript; available in PMC 2020 Dec 23.

Published in final edited form as: Sci Signal. 2020 Jun 23; 13(637): eaba1015. Published online 2020 Jun 23. doi: 10.1126/scisignal.aba1015

PMCID:: PMC7397860

Abstract Article PDF–1.2M

Select item 736096714.

NAADP‐regulated two‐pore channels drive phagocytosis through endo‐lysosomal Ca²⁺ nanodomains, calcineurin and dynamin

Lianne C Davis, Anthony J Morgan, Antony Galione

EMBO J. 2020 Jul 15; 39(14): e104058. Published online 2020 Jun 8. doi: 10.15252/embj.2019104058

PMCID:: PMC7360967

Abstract Article PDF–4.9M

Select item 722702815.

Mouse Models of Inherited Retinal Degeneration with Photoreceptor Cell Loss

Gayle B. Collin, Navdeep Gogna, Bo Chang, Nattaya Damkham, Jai Pinkney, Lillian F. Hyde, Lisa Stone, Jürgen K. Naggert, Patsy M. Nishina, Mark P. Krebs

Cells. 2020 Apr; 9(4): 931. Published online 2020 Apr 10. doi: 10.3390/cells9040931

PMCID:: PMC7227028

Abstract Article PDF–5.4M

Select item 699223316.

Abnormal LAMP1 glycosylation may play a role in Niemann-Pick disease, type C pathology

Niamh X. Cawley, Caitlin Sojka, Antony Cougnoux, Anna T. Lyons, Elena‐Raluca Nicoli, Christopher A. Wassif, Forbes D. Porter

PLoS One. 2020; 15(1): e0227829. Published online 2020 Jan 30. doi: 10.1371/journal.pone.0227829

PMCID:: PMC6992233

Abstract Article PDF–2.9M

Select item 697842317.

A lysosomal K⁺ channel regulates large particle phagocytosis by facilitating lysosome Ca²⁺ release

Xue Sun, Mengnan Xu, Qi Cao, Peng Huang, Xiaojuan Zhu, Xian-Ping Dong

Sci Rep. 2020; 10: 1038. Published online 2020 Jan 23. doi: 10.1038/s41598-020-57874-2

PMCID:: PMC6978423

Abstract Article PDF–3.7M

Select item 691583118.

Fingolimod phosphate inhibits astrocyte inflammatory activity in mucolipidosis IV

Laura D Weinstock, Amanda M Furness, Shawn S Herron, Sierra S Smith, Sitara B Sankar, Samantha G DeRosa, Dadi Gao, Molly E Mepyans, Anna Scotto Rosato, Diego L Medina, Ayelet Vardi, Natalia S Ferreira, Soo Min Cho, Anthony H Futerman, Susan A Slaugenhaupt, Levi B Wood, Yulia Grishchuk

Hum Mol Genet. 2018 Aug 1; 27(15): 2725–2738. Published online 2018 May 16. doi: 10.1093/hmg/ddy182

PMCID:: PMC6915831

Abstract Article PDF–2.2M

Select item 686412019.

Association of luteal cell degeneration and progesterone deficiency with lysosomal storage disorder mucolipidosis type IV in Mcoln1^−/− mouse model

Zidao Wang, Ahmed E El Zowalaty, Yuehuan Li, Christian L Andersen, Xiaoqin Ye

Biol Reprod. 2019 Oct; 101(4): 782–790. Published online 2019 Jul 18. doi: 10.1093/biolre/ioz126

PMCID:: PMC6864120

Abstract Article PDF–1.9M

Select item 643482920.

ZnT3 expression levels are down-regulated in the brain of Mcoln1 knockout mice

Jonathan Chacon, Lauren Rosas, Math P. Cuajungco

Mol Brain. 2019; 12: 24. Published online 2019 Mar 26. doi: 10.1186/s13041-019-0446-3

PMCID:: PMC6434829

Abstract Article PDF–687K

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