ALMS1 Regulates TGF-β Signaling and Morphology of Primary Cilia

Research output: Contribution to journal › Journal article › Research › peer-review

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ALMS1 Regulates TGF-β Signaling and Morphology of Primary Cilia. / Álvarez-Satta, María; Lago-Docampo, Mauro; Bea-Mascato, Brais; Solarat, Carlos; Castro-Sánchez, Sheila; Christensen, Søren T.; Valverde, Diana.

In: Frontiers in Cell and Developmental Biology, Vol. 9, 623829, 2021.

Research output: Contribution to journal › Journal article › Research › peer-review

Harvard

Álvarez-Satta, M, Lago-Docampo, M, Bea-Mascato, B, Solarat, C, Castro-Sánchez, S, Christensen, ST & Valverde, D 2021, 'ALMS1 Regulates TGF-β Signaling and Morphology of Primary Cilia', Frontiers in Cell and Developmental Biology, vol. 9, 623829. https://doi.org/10.3389/fcell.2021.623829

APA

Álvarez-Satta, M., Lago-Docampo, M., Bea-Mascato, B., Solarat, C., Castro-Sánchez, S., Christensen, S. T., & Valverde, D. (2021). ALMS1 Regulates TGF-β Signaling and Morphology of Primary Cilia. Frontiers in Cell and Developmental Biology, 9, [623829]. https://doi.org/10.3389/fcell.2021.623829

Vancouver

Álvarez-Satta M, Lago-Docampo M, Bea-Mascato B, Solarat C, Castro-Sánchez S, Christensen ST et al. ALMS1 Regulates TGF-β Signaling and Morphology of Primary Cilia. Frontiers in Cell and Developmental Biology. 2021;9. 623829. https://doi.org/10.3389/fcell.2021.623829

Author

Álvarez-Satta, María ; Lago-Docampo, Mauro ; Bea-Mascato, Brais ; Solarat, Carlos ; Castro-Sánchez, Sheila ; Christensen, Søren T. ; Valverde, Diana. / ALMS1 Regulates TGF-β Signaling and Morphology of Primary Cilia. In: Frontiers in Cell and Developmental Biology. 2021 ; Vol. 9.

Bibtex

@article{41f89cae07c5413a82d8845cd822d514,
title = "ALMS1 Regulates TGF-β Signaling and Morphology of Primary Cilia",
abstract = "In this study, we aimed to evaluate the role of ALMS1 in the morphology of primary cilia and regulation of cellular signaling using a knockdown model of the hTERT-RPE1 cell line. ALMS1 depletion resulted in the formation of longer cilia, which often displayed altered morphology as evidenced by extensive twisting and bending of the axoneme. Transforming growth factor beta/bone morphogenetic protein (TGF-β/BMP) signaling, which is regulated by primary cilia, was similarly affected by ALMS1 depletion as judged by reduced levels of TGFβ-1-mediated activation of SMAD2/3. These results provide novel information on the role of ALMS1 in the function of primary cilia and processing of cellular signaling, which when aberrantly regulated may underlie Alstr{\"o}m syndrome.",
keywords = "ALMS1, Alstr{\"o}m syndrome (AS), ciliary length, ciliary morphology, ciliopathies, hTERT RPE-1 cells, primary cilium, TGF-β/BMP signaling",
author = "Mar{\'i}a {\'A}lvarez-Satta and Mauro Lago-Docampo and Brais Bea-Mascato and Carlos Solarat and Sheila Castro-S{\'a}nchez and Christensen, {S{\o}ren T.} and Diana Valverde",
year = "2021",
doi = "10.3389/fcell.2021.623829",
language = "English",
volume = "9",
journal = "Frontiers in Cell and Developmental Biology",
issn = "2296-634X",
publisher = "Frontiers Media",

}

RIS

TY - JOUR

T1 - ALMS1 Regulates TGF-β Signaling and Morphology of Primary Cilia

AU - Álvarez-Satta, María

AU - Lago-Docampo, Mauro

AU - Bea-Mascato, Brais

AU - Solarat, Carlos

AU - Castro-Sánchez, Sheila

AU - Christensen, Søren T.

AU - Valverde, Diana

PY - 2021

Y1 - 2021

N2 - In this study, we aimed to evaluate the role of ALMS1 in the morphology of primary cilia and regulation of cellular signaling using a knockdown model of the hTERT-RPE1 cell line. ALMS1 depletion resulted in the formation of longer cilia, which often displayed altered morphology as evidenced by extensive twisting and bending of the axoneme. Transforming growth factor beta/bone morphogenetic protein (TGF-β/BMP) signaling, which is regulated by primary cilia, was similarly affected by ALMS1 depletion as judged by reduced levels of TGFβ-1-mediated activation of SMAD2/3. These results provide novel information on the role of ALMS1 in the function of primary cilia and processing of cellular signaling, which when aberrantly regulated may underlie Alström syndrome.

AB - In this study, we aimed to evaluate the role of ALMS1 in the morphology of primary cilia and regulation of cellular signaling using a knockdown model of the hTERT-RPE1 cell line. ALMS1 depletion resulted in the formation of longer cilia, which often displayed altered morphology as evidenced by extensive twisting and bending of the axoneme. Transforming growth factor beta/bone morphogenetic protein (TGF-β/BMP) signaling, which is regulated by primary cilia, was similarly affected by ALMS1 depletion as judged by reduced levels of TGFβ-1-mediated activation of SMAD2/3. These results provide novel information on the role of ALMS1 in the function of primary cilia and processing of cellular signaling, which when aberrantly regulated may underlie Alström syndrome.

KW - ALMS1

KW - Alström syndrome (AS)

KW - ciliary length

KW - ciliary morphology

KW - ciliopathies

KW - hTERT RPE-1 cells

KW - primary cilium

KW - TGF-β/BMP signaling

U2 - 10.3389/fcell.2021.623829

DO - 10.3389/fcell.2021.623829

M3 - Journal article

C2 - 33598462

AN - SCOPUS:85100867881

VL - 9

JO - Frontiers in Cell and Developmental Biology

JF - Frontiers in Cell and Developmental Biology

SN - 2296-634X

M1 - 623829

ER -

ID: 257601810