SorCS2 binds progranulin to regulate motor neuron development

J 2023

SorCS2 binds progranulin to regulate motor neuron development

THOMASEN, Pernille Bogetofte, Alena SALASOVA, Kasper KJAER-SORENSEN, Lucie WOLOSZCZUKOVA, Josef LAVICKÝ et. al.

Basic information

Original name

SorCS2 binds progranulin to regulate motor neuron development

Authors

THOMASEN, Pernille Bogetofte, Alena SALASOVA, Kasper KJAER-SORENSEN, Lucie WOLOSZCZUKOVA, Josef LAVICKÝ (203 Czech Republic, belonging to the institution), Hande LOGIN, Jeppe TRANBERG-JENSEN, Sergio ALMEIDA, Sander BEEL, Michaela KAVKOVÁ (203 Czech Republic, belonging to the institution), Per QVIST, Mads KJOLBY, Peter Lund OVESEN, Stella NOLTE, Benedicte VESTERGAARD, Andreea-Cornelia UDREA, Lene Niemann NEJSUM, Moses V CHAO, Van Damme PHILIP, Jan KŘIVÁNEK (203 Czech Republic, belonging to the institution), Jeremy DASEN, Claus OXVIG and Anders NYKJAER

Edition

Cell Reports, CAMBRIDGE, Cell Press, 2023, 2211-1247

Other information

Language

English

Type of outcome

Článek v odborném periodiku

Field of Study

10605 Developmental biology

Country of publisher

United States of America

Confidentiality degree

není předmětem státního či obchodního tajemství

References:

URL

Impact factor

Impact factor: 8.800 in 2022

RIV identification code

RIV/00216224:14110/23:00132520

Organization unit

Faculty of Medicine

DOI

http://dx.doi.org/10.1016/j.celrep.2023.113333

UT WoS

001104229200001

Keywords in English

motor neurons; SorCS2; progranulin; neurotrophic signaling; VPS10p-D receptors; neurodevelopment; nerve injury; light-sheet microscopy; image segmentation; zebrafish

Abstract

V originále

Motor neuron (MN) development and nerve regeneration requires orchestrated action of a vast number of molecules. Here, we identify SorCS2 as a progranulin (PGRN) receptor that is required for MN diversification and axon outgrowth in zebrafish and mice. In zebrafish, SorCS2 knockdown also affects neuromuscular junction morphology and fish motility. In mice, SorCS2 and PGRN are co-expressed by newborn MNs from embryonic day 9.5 until adulthood. Using cell-fate tracing and nerve segmentation, we find that SorCS2 deficiency perturbs cell-fate decisions of brachial MNs accompanied by innervation deficits of posterior nerves. Additionally, adult SorCS2 knockout mice display slower motor nerve regeneration. Interestingly, primitive macrophages express high levels of PGRN, and their interaction with SorCS2-positive motor axon is required during axon pathfinding. We further show that SorCS2 binds PGRN to control its secretion, signaling, and conversion into granulins. We propose that PGRN-SorCS2 signaling controls MN development and regeneration in vertebrates.

Links

GA23-06160S, research and development project

Name: Fluktuace mikroprostředí kmenových buněk jako zdroj tkáňové adaptability ve zdraví a nemoci

Investor: Czech Science Foundation, Fluctuation of the stem cell niche as a source of tissue adaptability in health and disease

LM2023050, research and development project

Name: Národní infrastruktura pro biologické a medicínské zobrazování

Investor: Ministry of Education, Youth and Sports of the CR, Czech BioImaging: National research infrastructure for biological and medical imaging

90250, large research infrastructures

Name: Czech-BioImaging III

Citovat

THOMASEN, Pernille Bogetofte, Alena SALASOVA, Kasper KJAER-SORENSEN, Lucie WOLOSZCZUKOVA, Josef LAVICKÝ, Hande LOGIN, Jeppe TRANBERG-JENSEN, Sergio ALMEIDA, Sander BEEL, Michaela KAVKOVÁ, Per QVIST, Mads KJOLBY, Peter Lund OVESEN, Stella NOLTE, Benedicte VESTERGAARD, Andreea-Cornelia UDREA, Lene Niemann NEJSUM, Moses V CHAO, Van Damme PHILIP, Jan KŘIVÁNEK, Jeremy DASEN, Claus OXVIG and Anders NYKJAER. SorCS2 binds progranulin to regulate motor neuron development. Cell Reports. CAMBRIDGE: Cell Press, 2023, vol. 42, No 11, p. 1-34. ISSN 2211-1247. Available from: https://dx.doi.org/10.1016/j.celrep.2023.113333.

@article{2349460,
   author = {Thomasen, Pernille Bogetofte and Salasova, Alena and KjaerandSorensen, Kasper and Woloszczukova, Lucie and Lavický, Josef and Login, Hande and TranbergandJensen, Jeppe and Almeida, Sergio and Beel, Sander and Kavková, Michaela and Qvist, Per and Kjolby, Mads and Ovesen, Peter Lund and Nolte, Stella and Vestergaard, Benedicte and Udrea, AndreeaandCornelia and Nejsum, Lene Niemann and Chao, Moses V and Philip, Van Damme and Křivánek, Jan and Dasen, Jeremy and Oxvig, Claus and Nykjaer, Anders},
   article_location = {CAMBRIDGE},
   article_number = {11},
   doi = {http://dx.doi.org/10.1016/j.celrep.2023.113333},
   keywords = {motor neurons; SorCS2; progranulin; neurotrophic signaling; VPS10p-D receptors; neurodevelopment; nerve injury; light-sheet microscopy; image segmentation; zebrafish},
   language = {eng},
   issn = {2211-1247},
   journal = {Cell Reports},
   title = {SorCS2 binds progranulin to regulate motor neuron development},
   url = {https://www.sciencedirect.com/science/article/pii/S2211124723013451},
   volume = {42},
   year = {2023}
}

TY  - JOUR
ID  - 2349460
AU  - Thomasen, Pernille Bogetofte - Salasova, Alena - Kjaer-Sorensen, Kasper - Woloszczukova, Lucie - Lavický, Josef - Login, Hande - Tranberg-Jensen, Jeppe - Almeida, Sergio - Beel, Sander - Kavková, Michaela - Qvist, Per - Kjolby, Mads - Ovesen, Peter Lund - Nolte, Stella - Vestergaard, Benedicte - Udrea, Andreea-Cornelia - Nejsum, Lene Niemann - Chao, Moses V - Philip, Van Damme - Křivánek, Jan - Dasen, Jeremy - Oxvig, Claus - Nykjaer, Anders
PY  - 2023
TI  - SorCS2 binds progranulin to regulate motor neuron development
JF  - Cell Reports
VL  - 42
IS  - 11
SP  - 1-34
EP  - 1-34
PB  - Cell Press
SN  - 22111247
KW  - motor neurons
KW  - SorCS2
KW  - progranulin
KW  - neurotrophic signaling
KW  - VPS10p-D receptors
KW  - neurodevelopment
KW  - nerve injury
KW  - light-sheet microscopy
KW  - image segmentation
KW  - zebrafish
UR  - https://www.sciencedirect.com/science/article/pii/S2211124723013451
N2  - Motor neuron (MN) development and nerve regeneration requires orchestrated action of a vast number of molecules. Here, we identify SorCS2 as a progranulin (PGRN) receptor that is required for MN diversification and axon outgrowth in zebrafish and mice. In zebrafish, SorCS2 knockdown also affects neuromuscular junction morphology and fish motility. In mice, SorCS2 and PGRN are co-expressed by newborn MNs from embryonic day 9.5 until adulthood. Using cell-fate tracing and nerve segmentation, we find that SorCS2 deficiency perturbs cell-fate decisions of brachial MNs accompanied by innervation deficits of posterior nerves. Additionally, adult SorCS2 knockout mice display slower motor nerve regeneration. Interestingly, primitive macrophages express high levels of PGRN, and their interaction with SorCS2-positive motor axon is required during axon pathfinding. We further show that SorCS2 binds PGRN to control its secretion, signaling, and conversion into granulins. We propose that PGRN-SorCS2 signaling controls MN development and regeneration in vertebrates.
ER  -

THOMASEN, Pernille Bogetofte, Alena SALASOVA, Kasper KJAER-SORENSEN, Lucie WOLOSZCZUKOVA, Josef LAVICKÝ, Hande LOGIN, Jeppe TRANBERG-JENSEN, Sergio ALMEIDA, Sander BEEL, Michaela KAVKOVÁ, Per QVIST, Mads KJOLBY, Peter Lund OVESEN, Stella NOLTE, Benedicte VESTERGAARD, Andreea-Cornelia UDREA, Lene Niemann NEJSUM, Moses V CHAO, Van Damme PHILIP, Jan KŘIVÁNEK, Jeremy DASEN, Claus OXVIG and Anders NYKJAER. SorCS2 binds progranulin to regulate motor neuron development. \textit{Cell Reports}. CAMBRIDGE: Cell Press, 2023, vol.~42, No~11, p.~1-34. ISSN~2211-1247. Available from: https://dx.doi.org/10.1016/j.celrep.2023.113333.

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