Characterization of


Journal

Science translational medicine
ISSN: 1946-6242
Titre abrégé: Sci Transl Med
Pays: United States
ID NLM: 101505086

Informations de publication

Date de publication:
09 09 2020
Historique:
received: 24 04 2019
revised: 31 12 2019
accepted: 14 08 2020
entrez: 10 9 2020
pubmed: 11 9 2020
medline: 24 6 2021
Statut: ppublish

Résumé

Primary lymphedema is caused by developmental and functional defects of the lymphatic vascular system that result in accumulation of protein-rich fluid in tissues, resulting in edema. The 28 currently known genes causing primary lymphedema can explain <30% of cases. Angiopoietin 1 (ANGPT1) and ANGPT2 function via the TIE1-TIE2 (tyrosine kinase with immunoglobulin-like and epidermal growth factor-like domains 1 and 2) receptor complex and α5β1 integrin to form an endothelial cell signaling pathway that is critical for blood and lymphatic vessel formation and remodeling during embryonic development, as well as for homeostasis of the mature vasculature. By screening a cohort of 543 individuals affected by primary lymphedema, we identified one heterozygous de novo

Identifiants

pubmed: 32908006
pii: 12/560/eaax8013
doi: 10.1126/scitranslmed.aax8013
pii:
doi:

Substances chimiques

ANGPT2 protein, human 0
Angiopoietin-1 0
Angiopoietin-2 0
Receptor, TIE-2 EC 2.7.10.1

Types de publication

Journal Article Research Support, Non-U.S. Gov't

Langues

eng

Sous-ensembles de citation

IM

Informations de copyright

Copyright © 2020 The Authors, some rights reserved; exclusive licensee American Association for the Advancement of Science. No claim to original U.S. Government Works.

Auteurs

Veli-Matti Leppänen (VM)

Wihuri Research Institute, Biomedicum Helsinki, Haartmaninkatu 8, 00290 Helsinki, Finland. miikka.vikkula@uclouvain.be kari.alitalo@helsinki.fi.
Translational Cancer Medicine Program, Faculty of Medicine and Helsinki Institute of Life Science, 00014 University of Helsinki, Finland.

Pascal Brouillard (P)

Human Molecular Genetics, de Duve Institute, University of Louvain, 1200 Brussels, Belgium. miikka.vikkula@uclouvain.be kari.alitalo@helsinki.fi.

Emilia A Korhonen (EA)

Wihuri Research Institute, Biomedicum Helsinki, Haartmaninkatu 8, 00290 Helsinki, Finland.

Tuomas Sipilä (T)

Wihuri Research Institute, Biomedicum Helsinki, Haartmaninkatu 8, 00290 Helsinki, Finland.

Sawan Kumar Jha (SK)

Translational Cancer Medicine Program, Faculty of Medicine and Helsinki Institute of Life Science, 00014 University of Helsinki, Finland.

Nicole Revencu (N)

Center for Human Genetics, Cliniques universitaires Saint-Luc, University of Louvain, 1200 Brussels, Belgium.

Veerle Labarque (V)

Centre for Molecular and Vascular Biology, University of Leuven, 3000 Leuven, Belgium.

Elodie Fastré (E)

Human Molecular Genetics, de Duve Institute, University of Louvain, 1200 Brussels, Belgium.

Matthieu Schlögel (M)

Human Molecular Genetics, de Duve Institute, University of Louvain, 1200 Brussels, Belgium.

Marie Ravoet (M)

Center for Human Genetics, Cliniques universitaires Saint-Luc, University of Louvain, 1200 Brussels, Belgium.

Amihood Singer (A)

Barzilai Medical Center, 78306 Ashekelon, Israel.

Claudia Luzzatto (C)

Padova University Hospital, 35128 Padova, Italy.

Donatella Angelone (D)

A. Gemelli Hospital, 00168 Roma, Italy.

Giovanni Crichiutti (G)

Azienda Ospedaliero-Universitaria Santa Maria della Misericordia, 33100 Udine, Italy.

Angela D'Elia (A)

Azienda Ospedaliero-Universitaria Santa Maria della Misericordia, 33100 Udine, Italy.

Jaakko Kuurne (J)

Wihuri Research Institute, Biomedicum Helsinki, Haartmaninkatu 8, 00290 Helsinki, Finland.

Harri Elamaa (H)

Oulu Centre for Cell-Matrix Research, Faculty of Biochemistry and Molecular Medicine, Biocenter Oulu, University of Oulu, 90220 Oulu, Finland.

Gou Young Koh (GY)

Center for Vascular Research, Institute of Basic Science (IBS), 34141 Daejeon, Republic of Korea.
Graduate School of Medical Science and Engineering, Korea Advanced Institute of Science and Technology (KAIST), 34141 Daejeon, Republic of Korea.

Pipsa Saharinen (P)

Wihuri Research Institute, Biomedicum Helsinki, Haartmaninkatu 8, 00290 Helsinki, Finland.
Translational Cancer Medicine Program, Faculty of Medicine and Helsinki Institute of Life Science, 00014 University of Helsinki, Finland.

Miikka Vikkula (M)

Human Molecular Genetics, de Duve Institute, University of Louvain, 1200 Brussels, Belgium. miikka.vikkula@uclouvain.be kari.alitalo@helsinki.fi.
Walloon Excellence in Lifesciences and Biotechnology (WELBIO), University of Louvain, 1200 Brussels, Belgium.

Kari Alitalo (K)

Wihuri Research Institute, Biomedicum Helsinki, Haartmaninkatu 8, 00290 Helsinki, Finland. miikka.vikkula@uclouvain.be kari.alitalo@helsinki.fi.
Translational Cancer Medicine Program, Faculty of Medicine and Helsinki Institute of Life Science, 00014 University of Helsinki, Finland.

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Classifications MeSH