Cilia kinases in skeletal development and homeostasis

Kód výsledku v IS VaVaI

<a href="https://www.isvavai.cz/riv?ss=detail&h=RIV%2F67985904%3A_____%2F22%3A00556562" target="_blank" >RIV/67985904:_____/22:00556562 - isvavai.cz</a>

Nalezeny alternativní kódy

RIV/00159816:_____/22:00077559 RIV/00216224:14110/22:00125089

Výsledek na webu

<a href="https://anatomypubs.onlinelibrary.wiley.com/doi/10.1002/dvdy.426" target="_blank" >https://anatomypubs.onlinelibrary.wiley.com/doi/10.1002/dvdy.426</a>

DOI - Digital Object Identifier

<a href="http://dx.doi.org/10.1002/dvdy.426" target="_blank" >10.1002/dvdy.426</a>

Jazyk výsledku

angličtina

Název v původním jazyce

Cilia kinases in skeletal development and homeostasis

Popis výsledku v původním jazyce

Primary cilia are dynamic compartments that regulate multiple aspects of cellular signaling. The production, maintenance, and function of cilia involve more than 1000 genes in mammals, and their mutations disrupt the ciliary signaling which manifests in a plethora of pathological conditions-the ciliopathies. Skeletal ciliopathies are genetic disorders affecting the development and homeostasis of the skeleton, and encompass a broad spectrum of pathologies ranging from isolated polydactyly to lethal syndromic dysplasias. The recent advances in forward genetics allowed for the identification of novel regulators of skeletogenesis, and revealed a growing list of ciliary proteins that are critical for signaling pathways implicated in bone physiology. Among these, a group of protein kinases involved in cilia assembly, maintenance, signaling, and disassembly has emerged. In this review, we summarize the functions of cilia kinases in skeletal development and disease, and discuss the available and upcoming treatment options.

Název v anglickém jazyce

Cilia kinases in skeletal development and homeostasis

Popis výsledku anglicky

Primary cilia are dynamic compartments that regulate multiple aspects of cellular signaling. The production, maintenance, and function of cilia involve more than 1000 genes in mammals, and their mutations disrupt the ciliary signaling which manifests in a plethora of pathological conditions-the ciliopathies. Skeletal ciliopathies are genetic disorders affecting the development and homeostasis of the skeleton, and encompass a broad spectrum of pathologies ranging from isolated polydactyly to lethal syndromic dysplasias. The recent advances in forward genetics allowed for the identification of novel regulators of skeletogenesis, and revealed a growing list of ciliary proteins that are critical for signaling pathways implicated in bone physiology. Among these, a group of protein kinases involved in cilia assembly, maintenance, signaling, and disassembly has emerged. In this review, we summarize the functions of cilia kinases in skeletal development and disease, and discuss the available and upcoming treatment options.

Druh

J_imp - Článek v periodiku v databázi Web of Science

CEP obor

—

OECD FORD obor

10605 - Developmental biology

Projekt

Výsledek vznikl pri realizaci vícero projektů. Více informací v záložce Projekty.

Návaznosti

I - Institucionalni podpora na dlouhodoby koncepcni rozvoj vyzkumne organizace

Rok uplatnění

2022

Kód důvěrnosti údajů

S - Úplné a pravdivé údaje o projektu nepodléhají ochraně podle zvláštních právních předpisů

Název periodika

Developmental Dynamics

ISSN

1058-8388

e-ISSN

1097-0177

Svazek periodika

251

Číslo periodika v rámci svazku

4

Stát vydavatele periodika

US - Spojené státy americké

Počet stran výsledku

32

Strana od-do

577-608

Kód UT WoS článku

000708091000001

EID výsledku v databázi Scopus

2-s2.0-85117131883