European network on Noonan syndrome and related disorders (NSEuroNet)

RASopathies constitute the most common family of non-chromosomal disorders affecting development. This group of rare diseases includes an increasing number of conditions characterized by reduced growth, heart defects, cognitive deficits, dysmorphisms and variable predisposition to malignancies. RASopathies share upregulated RAS-MAPK and/or PI3K-AKT-mTOR signaling as pathogenetic mechanism. The planned work, which is the natural follow-up of a previously funded E-Rare project, is directed to resolve the functional consequences of a large panel of uncharacterized mutations, generate in vitro and in vivo gene/mutation-specific models, characterize novel circuits modulating RAS signaling, explore the pathophysiology of key complications of these diseases, and identify novel disease genes. Work will also be directed to populate a dedicated database that will be used for clinically oriented studies to help clinicians towards a timely diagnosis and a more effective patient care.

Producten

Titel: Inflammatory response in hematopoietic stem and progenitor cells triggered by activating SHP2 mutations evokes blood defects.
Auteur: Solman M, Blokzijl-Franke S, Piques F, Yan C, Yang Q, Strullu M, Kamel SM, Ak P, Bakkers J, Langenau DM, Cavé H, den Hertog J
Magazine: eLife
Begin- en eindpagina: 3040-3040
Titel: Expanding the molecular spectrum of pathogenic SHOC2 variants underlying Mazzanti syndrome
Auteur: Motta M*, Solman M*, Bonnard AA, Kuechler A, Pantaleoni F, Priolo M, Chandramouli B, Coppola S, Pizzi S, Zara E, Ferilli M, Kayserili H, Onesimo R, Leoni C, Brinkmann J, Vial Y, Kamphausen S, Thomas-Teinturier C, Guimier A, Cordeddu V, Mazzanti L, Zampino G, Chillemi G, Zenker M, Cavé H, den Hertog J, Tartaglia M
Magazine: Human Molecular Genetics
Begin- en eindpagina: 2766-2778
Titel: Modeling (not so) rare developmental disorders associated with mutations in the protein-tyrosine phosphatase SHP2.
Auteur: Solman M, Woutersen DTJ, den Hertog J.
Magazine: Frontiers in Cell and Developmental biology
Begin- en eindpagina: 641-641
Titel: Targeting Oncogenic Src Homology 2 Domain-Containing Phosphatase 2 (SHP2) by Inhibiting Its Protein-Protein Interactions
Auteur: Bobone S, Pannone L, Biondi B, Solman M, Flex E, Canale VC, Calligari P, De Faveri C, Gandini T, Quercioli A, Torini G, Venditti M, Lauri A, Fasano G, Hoeksma J, Santucci V, Cattani G, Bocedi A, Carpentieri G, Tirelli V, Sanchez M, Peggion C, Formaggio F, den Hertog J, Martinelli S, Bocchinfuso G, Tartaglia M, Stella L.
Magazine: Journal of Medical Chemistry
Begin- en eindpagina: 15973-15990
Titel: The seventh international RASopathies symposium: Pathways to a cure-expanding knowledge, enhancing research, and therapeutic discovery.
Auteur: Kontaridis MI, Roberts AE, Schill L, Schoyer L, Stronach B, Andelfinger G, Aoki Y, Axelrad ME, Bakker A, Bennett AM, Broniscer A, Castel P, Chang CA, Cyganek L, Das TK, den Hertog J, Galperin E, Garg S, Gelb BD, Gordon K, Green T, Gripp KW, Itkin M, Kiuru M, Korf BR, Livingstone JR, López-Juárez A, Magoulas PL, Mansour S, Milner T, Parker E, Pierpont EI, Plouffe K, Rauen KA, Shankar SP, Smith SB, Stevenson DA, Tartaglia M, Van R, Wagner ME, Ware SM, Zenker M.
Magazine: American Journal of Medical Genetics
Begin- en eindpagina: 1915-1927

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Kenmerken

Projectnummer:
463002003
Looptijd: 100%
2020
2023
Gerelateerde programma's:
Gerelateerde subsidieronde:
Projectleider en penvoerder:
J den Hertog
Verantwoordelijke organisatie:
Hubrecht Institute