Structural and Biochemical Consequences of Disease-Causing Mutations in the Ankyrin Repeat Domain of the Human TRPV4 Channel

Inada, Hitoshi; Procko, Erik; Sotomayor, Marcos; Gaudet, Rachelle

doi:10.1021/bi300279b

Title: Structural and Biochemical Consequences of Disease-Causing Mutations in the Ankyrin Repeat Domain of the Human TRPV4 Channel

Authors:

Inada, Hitoshi ^[1]; Procko, Erik ^[1]; Sotomayor, Marcos ^[2]; Gaudet, Rachelle ^[1]

Department of Molecular and Cellular Biology, Harvard University, 52 Oxford Street, Cambridge, Massachusetts 02138, United States
Department of Molecular and Cellular Biology, Harvard University, 52 Oxford Street, Cambridge, Massachusetts 02138, United States, Howard Hughes Medical Institute and Department of Neurobiology, Harvard Medical School, Boston, Massachusetts 02115, United States

Publication Date:: Wed Jul 25 00:00:00 EDT 2012

Sponsoring Org.:: USDOE

OSTI Identifier:: 1123365

Resource Type:: Published Article

Journal Name:: Biochemistry

Additional Journal Information:: Journal Name: Biochemistry Journal Volume: 51 Journal Issue: 31; Journal ID: ISSN 0006-2960

Publisher:: American Chemical Society

Country of Publication:: United States

Language:: English

Citation Formats


                    Inada, Hitoshi, Procko, Erik, Sotomayor, Marcos, and Gaudet, Rachelle. Structural and Biochemical Consequences of Disease-Causing Mutations in the Ankyrin Repeat Domain of the Human TRPV4 Channel.  United States: N. p., 2012. 
Web.  doi:10.1021/bi300279b.

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                    Inada, Hitoshi, Procko, Erik, Sotomayor, Marcos, & Gaudet, Rachelle. Structural and Biochemical Consequences of Disease-Causing Mutations in the Ankyrin Repeat Domain of the Human TRPV4 Channel.  United States.  https://doi.org/10.1021/bi300279b

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                    Inada, Hitoshi, Procko, Erik, Sotomayor, Marcos, and Gaudet, Rachelle. Wed .  
"Structural and Biochemical Consequences of Disease-Causing Mutations in the Ankyrin Repeat Domain of the Human TRPV4 Channel".  United States.  https://doi.org/10.1021/bi300279b.

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@article{osti_1123365,

  title        = {Structural and Biochemical Consequences of Disease-Causing Mutations in the Ankyrin Repeat Domain of the Human TRPV4 Channel},

  author       = {Inada, Hitoshi and Procko, Erik and Sotomayor, Marcos and Gaudet, Rachelle},

  abstractNote = {},

  doi          = {10.1021/bi300279b},

  journal      = {Biochemistry},

  number       = 31,

  volume       = 51,

  place        = {United States},

  year         = {Wed Jul 25 00:00:00 EDT 2012},

  month        = {Wed Jul 25 00:00:00 EDT 2012}

}

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https://doi.org/10.1021/bi300279b

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Works referenced in this record:

A Transient Receptor Potential Vanilloid 4-Dependent Mechanism of Hyperalgesia Is Engaged by Concerted Action of Inflammatory Mediators
journal, April 2006

Alessandri-Haber, N.
Journal of Neuroscience, Vol. 26, Issue 14
DOI: 10.1523/JNEUROSCI.5385-05.2006

Scapuloperoneal spinal muscular atrophy and CMT2C are allelic disorders caused by alterations in TRPV4
journal, December 2009

Deng, Han-Xiang; Klein, Christopher J.; Yan, Jianhua
Nature Genetics, Vol. 42, Issue 2
DOI: 10.1038/ng.509

Folding landscapes of ankyrin repeat proteins: experiments meet theory
journal, February 2008

Barrick, Doug; Ferreiro, Diego U.; Komives, Elizabeth A.
Current Opinion in Structural Biology, Vol. 18, Issue 1
DOI: 10.1016/j.sbi.2007.12.004

TRPV4-Mediated Calcium Influx Regulates Terminal Differentiation of Osteoclasts
journal, September 2008

Masuyama, Ritsuko; Vriens, Joris; Voets, Thomas
Cell Metabolism, Vol. 8, Issue 3
DOI: 10.1016/j.cmet.2008.08.002

Structural Analyses of the Ankyrin Repeat Domain of TRPV6 and Related TRPV Ion Channels ^† ^, ^‡
journal, February 2008

Phelps, Christopher B.; Huang, Robert J.; Lishko, Polina V.
Biochemistry, Vol. 47, Issue 8
DOI: 10.1021/bi702109w

Transient receptor potential channelopathies
journal, February 2010

Nilius, Bernd; Owsianik, Grzegorz
Pflügers Archiv - European Journal of Physiology, Vol. 460, Issue 2
DOI: 10.1007/s00424-010-0788-2

Regions of I B that are critical for its inhibition of NF- B{middle dot}DNA interaction fold upon binding to NF- B
journal, December 2006

Truhlar, S. M. E.; Torpey, J. W.; Komives, E. A.
Proceedings of the National Academy of Sciences, Vol. 103, Issue 50
DOI: 10.1073/pnas.0605794103

TRPV4-pathy, a novel channelopathy affecting diverse systems
journal, May 2010

Dai, Jin; Cho, Tae-Joon; Unger, Sheila
Journal of Human Genetics, Vol. 55, Issue 7
DOI: 10.1038/jhg.2010.37

The TRPV4 Cation Channel Mediates Stretch-evoked Ca2+ Influx and ATP Release in Primary Urothelial Cell Cultures
journal, August 2009

Mochizuki, Tsutomu; Sokabe, Takaaki; Araki, Isao
Journal of Biological Chemistry, Vol. 284, Issue 32
DOI: 10.1074/jbc.M109.020206

TRPV4 mutations and cytotoxic hypercalcemia in axonal Charcot-Marie-Tooth neuropathies
journal, February 2011

Klein, C. J.; Shi, Y.; Fecto, F.
Neurology, Vol. 76, Issue 10
DOI: 10.1212/WNL.0b013e31820f2de3

Scalable molecular dynamics with NAMD
journal, January 2005

Phillips, James C.; Braun, Rosemary; Wang, Wei
Journal of Computational Chemistry, Vol. 26, Issue 16, p. 1781-1802
DOI: 10.1002/jcc.20289

Spherically averaged phased translation function and its applications to search of molecules and fragments in the macromolecular electron density maps
journal, August 2000

Vagin, A.; Isupov, M.
Acta Crystallographica Section A Foundations of Crystallography, Vol. 56, Issue s1
DOI: 10.1107/S0108767300029081

A single N-terminal cysteine in TRPV1 determines activation by pungent compounds from onion and garlic
journal, February 2008

Salazar, Héctor; Llorente, Itzel; Jara-Oseguera, Andrés
Nature Neuroscience, Vol. 11, Issue 3
DOI: 10.1038/nn2056

Heat-evoked Activation of TRPV4 Channels in a HEK293 Cell Expression System and in Native Mouse Aorta Endothelial Cells
journal, September 2002

Watanabe, Hiroyuki; Vriens, Joris; Suh, Suk H.
Journal of Biological Chemistry, Vol. 277, Issue 49
DOI: 10.1074/jbc.M208277200

Structure and function of TRPV1
journal, June 2005

Tominaga, Makoto; Tominaga, Tomoko
Pflügers Archiv - European Journal of Physiology, Vol. 451, Issue 1
DOI: 10.1007/s00424-005-1457-8

A thermodynamic framework for understanding temperature sensing by transient receptor potential (TRP) channels
journal, November 2011

Clapham, D. E.; Miller, C.
Proceedings of the National Academy of Sciences, Vol. 108, Issue 49
DOI: 10.1073/pnas.1117485108

A Modular PIP2 Binding Site as a Determinant of Capsaicin Receptor Sensitivity
journal, May 2003

Prescott, E. D.
Science, Vol. 300, Issue 5623
DOI: 10.1126/science.1083646

Mutations in the Gene Encoding the Calcium-Permeable Ion Channel TRPV4 Produce Spondylometaphyseal Dysplasia, Kozlowski Type and Metatropic Dysplasia
journal, March 2009

Krakow, Deborah; Vriens, Joris; Camacho, Natalia
The American Journal of Human Genetics, Vol. 84, Issue 3
DOI: 10.1016/j.ajhg.2009.01.021

All-Atom Empirical Potential for Molecular Modeling and Dynamics Studies of Proteins ^†
journal, April 1998

MacKerell, A. D.; Bashford, D.; Bellott, M.
The Journal of Physical Chemistry B, Vol. 102, Issue 18
DOI: 10.1021/jp973084f

Anandamide and arachidonic acid use epoxyeicosatrienoic acids to activate TRPV4 channels
journal, July 2003

Watanabe, Hiroyuki; Vriens, Joris; Prenen, Jean
Nature, Vol. 424, Issue 6947
DOI: 10.1038/nature01807

The Ankyrin Repeats of TRPV1 Bind Multiple Ligands and Modulate Channel Sensitivity
journal, June 2007

Lishko, Polina V.; Procko, Erik; Jin, Xiangshu
Neuron, Vol. 54, Issue 6
DOI: 10.1016/j.neuron.2007.05.027

Biophysical characterization of the free IκBα ankyrin repeat domain in solution
journal, July 2004

Croy, Carrie Hughes; Bergqvist, Simon; Huxford, Tom
Protein Science, Vol. 13, Issue 7
DOI: 10.1110/ps.04731004

TRPV4 as osmosensor: a transgenic approach
journal, June 2005

Liedtke, Wolfgang
Pflügers Archiv - European Journal of Physiology, Vol. 451, Issue 1
DOI: 10.1007/s00424-005-1449-8

TRPV4 related skeletal dysplasias: a phenotypic spectrum highlighted byclinical, radiographic, and molecular studies in 21 new families
journal, January 2011

Andreucci, Elena; Aftimos, Salim; Alcausin, Melanie
Orphanet Journal of Rare Diseases, Vol. 6, Issue 1
DOI: 10.1186/1750-1172-6-37

Structural determinant of TRPV1 desensitization interacts with calmodulin
journal, June 2003

Numazaki, M.; Tominaga, T.; Takeuchi, K.
Proceedings of the National Academy of Sciences, Vol. 100, Issue 13
DOI: 10.1073/pnas.1337252100

Vanilloid Receptor–Related Osmotically Activated Channel (VR-OAC), a Candidate Vertebrate Osmoreceptor
journal, October 2000

Liedtke, Wolfgang; Choe, Yong; Martí-Renom, Marc A.
Cell, Vol. 103, Issue 3
DOI: 10.1016/S0092-8674(00)00143-4

Crystal structure of the human TRPV2 channel ankyrin repeat domain
journal, September 2006

McCleverty, Clare J.; Koesema, Eric; Patapoutian, Ardem
Protein Science, Vol. 15, Issue 9
DOI: 10.1110/ps.062357206

Extending the treatment of backbone energetics in protein force fields: Limitations of gas-phase quantum mechanics in reproducing protein conformational distributions in molecular dynamics simulations
journal, August 2004

Mackerell, Alexander D.; Feig, Michael; Brooks, Charles L.
Journal of Computational Chemistry, Vol. 25, Issue 11
DOI: 10.1002/jcc.20065

TRPV4-mediated channelopathies
journal, August 2010

Verma, Pratibha; Kumar, Ashutosh; Goswami, Chandan
Channels, Vol. 4, Issue 4
DOI: 10.4161/chan.4.4.12905

Differential Regulation of TRPV1, TRPV3, and TRPV4 Sensitivity through a Conserved Binding Site on the Ankyrin Repeat Domain
journal, October 2009

Phelps, Christopher B.; Wang, Ruiqi R.; Choo, Shelly S.
Journal of Biological Chemistry, Vol. 285, Issue 1
DOI: 10.1074/jbc.M109.052548

Gain-of-function mutations in TRPV4 cause autosomal dominant brachyolmia
journal, June 2008

Rock, Matthew J.; Prenen, Jean; Funari, Vincent A.
Nature Genetics, Vol. 40, Issue 8
DOI: 10.1038/ng.166

International Union of Basic and Clinical Pharmacology. LXXVI. Current Progress in the Mammalian TRP Ion Channel Family
journal, August 2010

Wu, Long-Jun; Sweet, Tara-Beth; Clapham, David E.
Pharmacological Reviews, Vol. 62, Issue 3
DOI: 10.1124/pr.110.002725

Ca2+/Calmodulin Modulates TRPV1 Activation by Capsaicin
journal, December 2003

Rosenbaum, Tamara; Gordon-Shaag, Ariela; Munari, Mika
Journal of General Physiology, Vol. 123, Issue 1
DOI: 10.1085/jgp.200308906

The TRPV4 cation channel: A molecule linking skin temperature and barrier function
journal, November 2010

Sokabe, Takaaki; Tominaga, Makoto
Communicative & Integrative Biology, Vol. 3, Issue 6
DOI: 10.4161/cib.3.6.13461

Mutant TRPV4-mediated Toxicity Is Linked to Increased Constitutive Function in Axonal Neuropathies
journal, May 2011

Fecto, Faisal; Shi, Yong; Huda, Rafiq
Journal of Biological Chemistry, Vol. 286, Issue 19
DOI: 10.1074/jbc.M111.237685

Effects of Body Temperature on Neural Activity in the Hippocampus: Regulation of Resting Membrane Potentials by Transient Receptor Potential Vanilloid 4
journal, February 2007

Shibasaki, K.; Suzuki, M.; Mizuno, A.
Journal of Neuroscience, Vol. 27, Issue 7
DOI: 10.1523/JNEUROSCI.4284-06.2007

Mutations in TRPV4 cause Charcot-Marie-Tooth disease type 2C
journal, December 2009

Landouré, Guida; Zdebik, Anselm A.; Martinez, Tara L.
Nature Genetics, Vol. 42, Issue 2
DOI: 10.1038/ng.512

Steered molecular dynamics and mechanical functions of proteins
journal, April 2001

Isralewitz, Barry; Gao, Mu; Schulten, Klaus
Current Opinion in Structural Biology, Vol. 11, Issue 2
DOI: 10.1016/S0959-440X(00)00194-9

Dominant TRPV4 mutations in nonlethal and lethal metatropic dysplasia
journal, May 2010

Camacho, Natalia; Krakow, Deborah; Johnykutty, Sharlin
American Journal of Medical Genetics Part A, Vol. 152A, Issue 5
DOI: 10.1002/ajmg.a.33392

Increased Basal Activity Is a Key Determinant in the Severity of Human Skeletal Dysplasia Caused by TRPV4 Mutations
journal, May 2011

Loukin, Stephen; Su, Zhenwei; Kung, Ching
PLoS ONE, Vol. 6, Issue 5
DOI: 10.1371/journal.pone.0019533

Novel and recurrent TRPV4 mutations and their association with distinct phenotypes within the TRPV4 dysplasia family
journal, June 2010

Dai, J.; Kim, O. -H.; Cho, T. -J.
Journal of Medical Genetics, Vol. 47, Issue 10
DOI: 10.1136/jmg.2009.075358

Dominant mutations in the cation channel gene transient receptor potential vanilloid 4 cause an unusual spectrum of neuropathies
journal, May 2010

Zimoń, Magdalena; Baets, Jonathan; Auer-Grumbach, Michaela
Brain, Vol. 133, Issue 6
DOI: 10.1093/brain/awq109

International Union of Pharmacology. XLIII. Compendium of Voltage-Gated Ion Channels: Transient Receptor Potential Channels
journal, December 2003

Clapham, David E.; Montell, Craig; Schultz, Guenter
Pharmacological Reviews, Vol. 55, Issue 4
DOI: 10.1124/pr.55.4.6

Localization of the PIP2 Sensor of TRPV1 Ion Channels
journal, March 2011

Ufret-Vincenty, Carmen A.; Klein, Rebecca M.; Hua, Li
Journal of Biological Chemistry, Vol. 286, Issue 11
DOI: 10.1074/jbc.M110.192526

Thermosensation and pain
journal, January 2004

Tominaga, Makoto; Caterina, Michael J.
Journal of Neurobiology, Vol. 61, Issue 1
DOI: 10.1002/neu.20079

[20] Processing of X-ray diffraction data collected in oscillation mode
book, January 1997

Otwinowski, Zbyszek; Minor, Wladek
Macromolecular Crystallography Part A, 307-326
DOI: 10.1016/S0076-6879(97)76066-X

Homo- and heteromeric assembly of TRP channel subunits
journal, June 2005

Schaefer, Michael
Pflügers Archiv - European Journal of Physiology, Vol. 451, Issue 1
DOI: 10.1007/s00424-005-1467-6

TRP channels as cellular sensors
journal, December 2003

Clapham, David E.
Nature, Vol. 426, Issue 6966
DOI: 10.1038/nature02196

Phaser crystallographic software
journal, July 2007

McCoy, Airlie J.; Grosse-Kunstleve, Ralf W.; Adams, Paul D.
Journal of Applied Crystallography, Vol. 40, Issue 4
DOI: 10.1107/S0021889807021206

VMD: Visual molecular dynamics
journal, February 1996

Humphrey, William; Dalke, Andrew; Schulten, Klaus
Journal of Molecular Graphics, Vol. 14, Issue 1
DOI: 10.1016/0263-7855(96)00018-5

Heat-Evoked Activation of the Ion Channel, TRPV4
journal, August 2002

Güler, Ali Deniz; Lee, Hyosang; Iida, Tohko
The Journal of Neuroscience, Vol. 22, Issue 15
DOI: 10.1523/JNEUROSCI.22-15-06408.2002

Structure of the N-terminal Ankyrin Repeat Domain of the TRPV2 Ion Channel
journal, September 2006

Jin, Xiangshu; Touhey, Jason; Gaudet, Rachelle
Journal of Biological Chemistry, Vol. 281, Issue 35
DOI: 10.1074/jbc.C600153200

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Title: Structural and Biochemical Consequences of Disease-Causing Mutations in the Ankyrin Repeat Domain of the Human TRPV4 Channel

Citation Formats

A Transient Receptor Potential Vanilloid 4-Dependent Mechanism of Hyperalgesia Is Engaged by Concerted Action of Inflammatory Mediators journal, April 2006

Scapuloperoneal spinal muscular atrophy and CMT2C are allelic disorders caused by alterations in TRPV4 journal, December 2009

Folding landscapes of ankyrin repeat proteins: experiments meet theory journal, February 2008

TRPV4-Mediated Calcium Influx Regulates Terminal Differentiation of Osteoclasts journal, September 2008

Structural Analyses of the Ankyrin Repeat Domain of TRPV6 and Related TRPV Ion Channels † , ‡ journal, February 2008

Transient receptor potential channelopathies journal, February 2010

Regions of I B that are critical for its inhibition of NF- B{middle dot}DNA interaction fold upon binding to NF- B journal, December 2006

TRPV4-pathy, a novel channelopathy affecting diverse systems journal, May 2010

The TRPV4 Cation Channel Mediates Stretch-evoked Ca2+ Influx and ATP Release in Primary Urothelial Cell Cultures journal, August 2009

TRPV4 mutations and cytotoxic hypercalcemia in axonal Charcot-Marie-Tooth neuropathies journal, February 2011

Scalable molecular dynamics with NAMD journal, January 2005

Spherically averaged phased translation function and its applications to search of molecules and fragments in the macromolecular electron density maps journal, August 2000

A single N-terminal cysteine in TRPV1 determines activation by pungent compounds from onion and garlic journal, February 2008

Heat-evoked Activation of TRPV4 Channels in a HEK293 Cell Expression System and in Native Mouse Aorta Endothelial Cells journal, September 2002

Structure and function of TRPV1 journal, June 2005

A thermodynamic framework for understanding temperature sensing by transient receptor potential (TRP) channels journal, November 2011

A Modular PIP2 Binding Site as a Determinant of Capsaicin Receptor Sensitivity journal, May 2003

Mutations in the Gene Encoding the Calcium-Permeable Ion Channel TRPV4 Produce Spondylometaphyseal Dysplasia, Kozlowski Type and Metatropic Dysplasia journal, March 2009

All-Atom Empirical Potential for Molecular Modeling and Dynamics Studies of Proteins † journal, April 1998

Anandamide and arachidonic acid use epoxyeicosatrienoic acids to activate TRPV4 channels journal, July 2003

The Ankyrin Repeats of TRPV1 Bind Multiple Ligands and Modulate Channel Sensitivity journal, June 2007

Biophysical characterization of the free IκBα ankyrin repeat domain in solution journal, July 2004

TRPV4 as osmosensor: a transgenic approach journal, June 2005

TRPV4 related skeletal dysplasias: a phenotypic spectrum highlighted byclinical, radiographic, and molecular studies in 21 new families journal, January 2011

Structural determinant of TRPV1 desensitization interacts with calmodulin journal, June 2003

Vanilloid Receptor–Related Osmotically Activated Channel (VR-OAC), a Candidate Vertebrate Osmoreceptor journal, October 2000

Crystal structure of the human TRPV2 channel ankyrin repeat domain journal, September 2006

Extending the treatment of backbone energetics in protein force fields: Limitations of gas-phase quantum mechanics in reproducing protein conformational distributions in molecular dynamics simulations journal, August 2004

TRPV4-mediated channelopathies journal, August 2010

Differential Regulation of TRPV1, TRPV3, and TRPV4 Sensitivity through a Conserved Binding Site on the Ankyrin Repeat Domain journal, October 2009

Gain-of-function mutations in TRPV4 cause autosomal dominant brachyolmia journal, June 2008

International Union of Basic and Clinical Pharmacology. LXXVI. Current Progress in the Mammalian TRP Ion Channel Family journal, August 2010

Ca2+/Calmodulin Modulates TRPV1 Activation by Capsaicin journal, December 2003

The TRPV4 cation channel: A molecule linking skin temperature and barrier function journal, November 2010

Mutant TRPV4-mediated Toxicity Is Linked to Increased Constitutive Function in Axonal Neuropathies journal, May 2011

Effects of Body Temperature on Neural Activity in the Hippocampus: Regulation of Resting Membrane Potentials by Transient Receptor Potential Vanilloid 4 journal, February 2007

Mutations in TRPV4 cause Charcot-Marie-Tooth disease type 2C journal, December 2009

Steered molecular dynamics and mechanical functions of proteins journal, April 2001

Dominant TRPV4 mutations in nonlethal and lethal metatropic dysplasia journal, May 2010

Increased Basal Activity Is a Key Determinant in the Severity of Human Skeletal Dysplasia Caused by TRPV4 Mutations journal, May 2011

Novel and recurrent TRPV4 mutations and their association with distinct phenotypes within the TRPV4 dysplasia family journal, June 2010

Dominant mutations in the cation channel gene transient receptor potential vanilloid 4 cause an unusual spectrum of neuropathies journal, May 2010

International Union of Pharmacology. XLIII. Compendium of Voltage-Gated Ion Channels: Transient Receptor Potential Channels journal, December 2003

Localization of the PIP2 Sensor of TRPV1 Ion Channels journal, March 2011

Thermosensation and pain journal, January 2004

[20] Processing of X-ray diffraction data collected in oscillation mode book, January 1997

Homo- and heteromeric assembly of TRP channel subunits journal, June 2005

TRP channels as cellular sensors journal, December 2003

Phaser crystallographic software journal, July 2007

VMD: Visual molecular dynamics journal, February 1996

Heat-Evoked Activation of the Ion Channel, TRPV4 journal, August 2002

Structure of the N-terminal Ankyrin Repeat Domain of the TRPV2 Ion Channel journal, September 2006

A Transient Receptor Potential Vanilloid 4-Dependent Mechanism of Hyperalgesia Is Engaged by Concerted Action of Inflammatory Mediators
journal, April 2006

Scapuloperoneal spinal muscular atrophy and CMT2C are allelic disorders caused by alterations in TRPV4
journal, December 2009

Folding landscapes of ankyrin repeat proteins: experiments meet theory
journal, February 2008

TRPV4-Mediated Calcium Influx Regulates Terminal Differentiation of Osteoclasts
journal, September 2008

Structural Analyses of the Ankyrin Repeat Domain of TRPV6 and Related TRPV Ion Channels ^† ^, ^‡
journal, February 2008

Transient receptor potential channelopathies
journal, February 2010

Regions of I B that are critical for its inhibition of NF- B{middle dot}DNA interaction fold upon binding to NF- B
journal, December 2006

TRPV4-pathy, a novel channelopathy affecting diverse systems
journal, May 2010

The TRPV4 Cation Channel Mediates Stretch-evoked Ca2+ Influx and ATP Release in Primary Urothelial Cell Cultures
journal, August 2009

TRPV4 mutations and cytotoxic hypercalcemia in axonal Charcot-Marie-Tooth neuropathies
journal, February 2011

Scalable molecular dynamics with NAMD
journal, January 2005

Spherically averaged phased translation function and its applications to search of molecules and fragments in the macromolecular electron density maps
journal, August 2000

A single N-terminal cysteine in TRPV1 determines activation by pungent compounds from onion and garlic
journal, February 2008

Heat-evoked Activation of TRPV4 Channels in a HEK293 Cell Expression System and in Native Mouse Aorta Endothelial Cells
journal, September 2002

Structure and function of TRPV1
journal, June 2005

A thermodynamic framework for understanding temperature sensing by transient receptor potential (TRP) channels
journal, November 2011

A Modular PIP2 Binding Site as a Determinant of Capsaicin Receptor Sensitivity
journal, May 2003

Mutations in the Gene Encoding the Calcium-Permeable Ion Channel TRPV4 Produce Spondylometaphyseal Dysplasia, Kozlowski Type and Metatropic Dysplasia
journal, March 2009

All-Atom Empirical Potential for Molecular Modeling and Dynamics Studies of Proteins ^†
journal, April 1998

Anandamide and arachidonic acid use epoxyeicosatrienoic acids to activate TRPV4 channels
journal, July 2003

The Ankyrin Repeats of TRPV1 Bind Multiple Ligands and Modulate Channel Sensitivity
journal, June 2007

Biophysical characterization of the free IκBα ankyrin repeat domain in solution
journal, July 2004

TRPV4 as osmosensor: a transgenic approach
journal, June 2005

TRPV4 related skeletal dysplasias: a phenotypic spectrum highlighted byclinical, radiographic, and molecular studies in 21 new families
journal, January 2011

Structural determinant of TRPV1 desensitization interacts with calmodulin
journal, June 2003

Vanilloid Receptor–Related Osmotically Activated Channel (VR-OAC), a Candidate Vertebrate Osmoreceptor
journal, October 2000

Crystal structure of the human TRPV2 channel ankyrin repeat domain
journal, September 2006

Extending the treatment of backbone energetics in protein force fields: Limitations of gas-phase quantum mechanics in reproducing protein conformational distributions in molecular dynamics simulations
journal, August 2004

TRPV4-mediated channelopathies
journal, August 2010

Differential Regulation of TRPV1, TRPV3, and TRPV4 Sensitivity through a Conserved Binding Site on the Ankyrin Repeat Domain
journal, October 2009

Gain-of-function mutations in TRPV4 cause autosomal dominant brachyolmia
journal, June 2008

International Union of Basic and Clinical Pharmacology. LXXVI. Current Progress in the Mammalian TRP Ion Channel Family
journal, August 2010

Ca2+/Calmodulin Modulates TRPV1 Activation by Capsaicin
journal, December 2003

The TRPV4 cation channel: A molecule linking skin temperature and barrier function
journal, November 2010

Mutant TRPV4-mediated Toxicity Is Linked to Increased Constitutive Function in Axonal Neuropathies
journal, May 2011

Effects of Body Temperature on Neural Activity in the Hippocampus: Regulation of Resting Membrane Potentials by Transient Receptor Potential Vanilloid 4
journal, February 2007

Mutations in TRPV4 cause Charcot-Marie-Tooth disease type 2C
journal, December 2009

Steered molecular dynamics and mechanical functions of proteins
journal, April 2001

Dominant TRPV4 mutations in nonlethal and lethal metatropic dysplasia
journal, May 2010

Increased Basal Activity Is a Key Determinant in the Severity of Human Skeletal Dysplasia Caused by TRPV4 Mutations
journal, May 2011

Novel and recurrent TRPV4 mutations and their association with distinct phenotypes within the TRPV4 dysplasia family
journal, June 2010

Dominant mutations in the cation channel gene transient receptor potential vanilloid 4 cause an unusual spectrum of neuropathies
journal, May 2010

International Union of Pharmacology. XLIII. Compendium of Voltage-Gated Ion Channels: Transient Receptor Potential Channels
journal, December 2003

Localization of the PIP2 Sensor of TRPV1 Ion Channels
journal, March 2011

Thermosensation and pain
journal, January 2004

[20] Processing of X-ray diffraction data collected in oscillation mode
book, January 1997

Homo- and heteromeric assembly of TRP channel subunits
journal, June 2005

TRP channels as cellular sensors
journal, December 2003

Phaser crystallographic software
journal, July 2007

VMD: Visual molecular dynamics
journal, February 1996

Heat-Evoked Activation of the Ion Channel, TRPV4
journal, August 2002

Structure of the N-terminal Ankyrin Repeat Domain of the TRPV2 Ion Channel
journal, September 2006