DLG1 functions upstream of SDCCAG3 and IFT20 to control ciliary targeting of polycystin-2

Csenge K. Rezi; Mariam G. Aslanyan; Gaurav D. Diwan; Tao Cheng; Mohamed Chamlali; Katrin Junger; Zeinab Anvarian; Esben Lorentzen; Kleo B. Pauly; Yasmin Afshar-Bahadori; Eduardo F.A. Fernandes; Feng Qian; Sébastien Tosi; Søren T. Christensen; Stine F. Pedersen; Kristian Strømgaard; Robert B. Russell; Jeffrey H. Miner; Moe R. Mahjoub; Karsten Boldt; Ronald Roepman; Lotte B. Pedersen

doi:10.1038/s44319-024-00170-1

DLG1 functions upstream of SDCCAG3 and IFT20 to control ciliary targeting of polycystin-2

Csenge K. Rezi, Mariam G. Aslanyan, Gaurav D. Diwan, Tao Cheng, Mohamed Chamlali, Katrin Junger, Zeinab Anvarian, Esben Lorentzen, Kleo B. Pauly, Yasmin Afshar-Bahadori, Eduardo F.A. Fernandes, Feng Qian, Sébastien Tosi, Søren T. Christensen, Stine F. Pedersen, Kristian Strømgaard, Robert B. Russell, Jeffrey H. Miner, Moe R. Mahjoub, Karsten BoldtRonald Roepman, Lotte B. Pedersen^*

^*Corresponding author for this work

Department of Molecular Biology and Genetics - Protein Science

Research output: Contribution to journal/Conference contribution in journal/Contribution to newspaper › Journal article › Research › peer-review

Abstract

(Figure presented.) The Scribble polarity complex protein DLG1 regulates ciliary length and protein composition in kidney epithelial cells. A Congenital Anomalies of the Kidney and Urinary Tract (CAKUT)-associated DLG1 variant may be associated with ciliary dysfunction. Loss of DLG1 causes ciliary elongation in kidney epithelial cells. Loss of DLG1 impairs targeting of SDCCAG3, IFT20 and PC2 to the primary cilium of kidney epithelial cells. The CAKUT-associated p.T489R DLG1 fails to rescue ciliary defects of Dlg1^-/- cells, indicating a possible ciliary involvement in CAKUT disease etiology.

Original language	English
Journal	EMBO Reports
Volume	25
Issue	7
Pages (from-to)	3040-3063
Number of pages	24
ISSN	1469-221X
DOIs	https://doi.org/10.1038/s44319-024-00170-1
Publication status	Published - Jul 2024

Keywords

DLG1
IFT20
Polycystin-2
Primary Cilia
SDCCAG3

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Rezi, C. K., Aslanyan, M. G., Diwan, G. D., Cheng, T., Chamlali, M., Junger, K., Anvarian, Z., Lorentzen, E., Pauly, K. B., Afshar-Bahadori, Y., Fernandes, E. F. A., Qian, F., Tosi, S., Christensen, S. T., Pedersen, S. F., Strømgaard, K., Russell, R. B., Miner, J. H., Mahjoub, M. R., ... Pedersen, L. B. (2024). DLG1 functions upstream of SDCCAG3 and IFT20 to control ciliary targeting of polycystin-2. EMBO Reports, 25(7), 3040-3063. https://doi.org/10.1038/s44319-024-00170-1

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title = "DLG1 functions upstream of SDCCAG3 and IFT20 to control ciliary targeting of polycystin-2",

abstract = "(Figure presented.) The Scribble polarity complex protein DLG1 regulates ciliary length and protein composition in kidney epithelial cells. A Congenital Anomalies of the Kidney and Urinary Tract (CAKUT)-associated DLG1 variant may be associated with ciliary dysfunction. Loss of DLG1 causes ciliary elongation in kidney epithelial cells. Loss of DLG1 impairs targeting of SDCCAG3, IFT20 and PC2 to the primary cilium of kidney epithelial cells. The CAKUT-associated p.T489R DLG1 fails to rescue ciliary defects of Dlg1-/- cells, indicating a possible ciliary involvement in CAKUT disease etiology.",

keywords = "DLG1, IFT20, Polycystin-2, Primary Cilia, SDCCAG3",

author = "Rezi, {Csenge K.} and Aslanyan, {Mariam G.} and Diwan, {Gaurav D.} and Tao Cheng and Mohamed Chamlali and Katrin Junger and Zeinab Anvarian and Esben Lorentzen and Pauly, {Kleo B.} and Yasmin Afshar-Bahadori and Fernandes, {Eduardo F.A.} and Feng Qian and S{\'e}bastien Tosi and Christensen, {S{\o}ren T.} and Pedersen, {Stine F.} and Kristian Str{\o}mgaard and Russell, {Robert B.} and Miner, {Jeffrey H.} and Mahjoub, {Moe R.} and Karsten Boldt and Ronald Roepman and Pedersen, {Lotte B.}",

year = "2024",

month = jul,

doi = "10.1038/s44319-024-00170-1",

language = "English",

volume = "25",

pages = "3040--3063",

journal = "EMBO Reports",

issn = "1469-221X",

publisher = "Wiley-Blackwell Publishing Ltd.",

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}

Rezi, CK, Aslanyan, MG, Diwan, GD, Cheng, T, Chamlali, M, Junger, K, Anvarian, Z, Lorentzen, E, Pauly, KB, Afshar-Bahadori, Y, Fernandes, EFA, Qian, F, Tosi, S, Christensen, ST, Pedersen, SF, Strømgaard, K, Russell, RB, Miner, JH, Mahjoub, MR, Boldt, K, Roepman, R & Pedersen, LB 2024, 'DLG1 functions upstream of SDCCAG3 and IFT20 to control ciliary targeting of polycystin-2', EMBO Reports, vol. 25, no. 7, pp. 3040-3063. https://doi.org/10.1038/s44319-024-00170-1

TY - JOUR

T1 - DLG1 functions upstream of SDCCAG3 and IFT20 to control ciliary targeting of polycystin-2

AU - Rezi, Csenge K.

AU - Aslanyan, Mariam G.

AU - Diwan, Gaurav D.

AU - Cheng, Tao

AU - Chamlali, Mohamed

AU - Junger, Katrin

AU - Anvarian, Zeinab

AU - Lorentzen, Esben

AU - Pauly, Kleo B.

AU - Afshar-Bahadori, Yasmin

AU - Fernandes, Eduardo F.A.

AU - Qian, Feng

AU - Tosi, Sébastien

AU - Christensen, Søren T.

AU - Pedersen, Stine F.

AU - Strømgaard, Kristian

AU - Russell, Robert B.

AU - Miner, Jeffrey H.

AU - Mahjoub, Moe R.

AU - Boldt, Karsten

AU - Roepman, Ronald

AU - Pedersen, Lotte B.

PY - 2024/7

Y1 - 2024/7

N2 - (Figure presented.) The Scribble polarity complex protein DLG1 regulates ciliary length and protein composition in kidney epithelial cells. A Congenital Anomalies of the Kidney and Urinary Tract (CAKUT)-associated DLG1 variant may be associated with ciliary dysfunction. Loss of DLG1 causes ciliary elongation in kidney epithelial cells. Loss of DLG1 impairs targeting of SDCCAG3, IFT20 and PC2 to the primary cilium of kidney epithelial cells. The CAKUT-associated p.T489R DLG1 fails to rescue ciliary defects of Dlg1-/- cells, indicating a possible ciliary involvement in CAKUT disease etiology.

AB - (Figure presented.) The Scribble polarity complex protein DLG1 regulates ciliary length and protein composition in kidney epithelial cells. A Congenital Anomalies of the Kidney and Urinary Tract (CAKUT)-associated DLG1 variant may be associated with ciliary dysfunction. Loss of DLG1 causes ciliary elongation in kidney epithelial cells. Loss of DLG1 impairs targeting of SDCCAG3, IFT20 and PC2 to the primary cilium of kidney epithelial cells. The CAKUT-associated p.T489R DLG1 fails to rescue ciliary defects of Dlg1-/- cells, indicating a possible ciliary involvement in CAKUT disease etiology.

KW - DLG1

KW - IFT20

KW - Polycystin-2

KW - Primary Cilia

KW - SDCCAG3

UR - http://www.scopus.com/inward/record.url?scp=85195398049&partnerID=8YFLogxK

U2 - 10.1038/s44319-024-00170-1

DO - 10.1038/s44319-024-00170-1

M3 - Journal article

C2 - 38849673

AN - SCOPUS:85195398049

SN - 1469-221X

VL - 25

SP - 3040

EP - 3063

JO - EMBO Reports

JF - EMBO Reports

IS - 7

ER -

DLG1 functions upstream of SDCCAG3 and IFT20 to control ciliary targeting of polycystin-2

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Keywords

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