Developmental vascular remodeling defects and postnatal kidney failure in mice lacking Gpr116 (Adgrf5) and Eltd1 (Adgrl4).
GPR116 (ADGRF5) and ELTD1 (ADGRL4) belong to different subfamilies of the adhesion G-protein-coupled receptor group but are both expressed in endothelial cells. We therefore analyzed their functions in mice lacking these receptors. While loss of GPR116 or ELTD1 alone had no obvious effect on cardiov...
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| Format: | Article |
| Language: | English |
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Public Library of Science (PLoS)
2017-01-01
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| Series: | PLoS ONE |
| Online Access: | https://journals.plos.org/plosone/article/file?id=10.1371/journal.pone.0183166&type=printable |
| _version_ | 1826578977106427904 |
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| author | Shun Lu Shuya Liu Astrid Wietelmann Baktybek Kojonazarov Ann Atzberger Cong Tang Ralph Theo Schermuly Hermann-Josef Gröne Stefan Offermanns |
| author_facet | Shun Lu Shuya Liu Astrid Wietelmann Baktybek Kojonazarov Ann Atzberger Cong Tang Ralph Theo Schermuly Hermann-Josef Gröne Stefan Offermanns |
| author_sort | Shun Lu |
| collection | DOAJ |
| description | GPR116 (ADGRF5) and ELTD1 (ADGRL4) belong to different subfamilies of the adhesion G-protein-coupled receptor group but are both expressed in endothelial cells. We therefore analyzed their functions in mice lacking these receptors. While loss of GPR116 or ELTD1 alone had no obvious effect on cardiovascular or kidney function, mice lacking both, GPR116 and ELTD1, showed malformations of the aortic arch arteries and the cardiac outflow tract leading to perinatal lethality in about 50% of the mutants. In addition to cardiovascular malformations, surviving mice developed renal thrombotic microangiopathy as well as hemolysis and splenomegaly, and their lifespan was significantly reduced. Loss of GPR116 and ELTD1 specifically in endothelial cells or neural crest-derived cells did not recapitulate any of the phenotypes observed in GPR116-ELTD1 double deficient mice, indicating that loss of GPR116 and ELTD1 expressed by other cells accounts for the observed cardiovascular and renal defects. |
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| format | Article |
| id | doaj.art-e8e326545b03427e8fba2dba3f7837c8 |
| institution | Directory Open Access Journal |
| issn | 1932-6203 |
| language | English |
| last_indexed | 2025-03-14T14:10:57Z |
| publishDate | 2017-01-01 |
| publisher | Public Library of Science (PLoS) |
| record_format | Article |
| series | PLoS ONE |
| spelling | doaj.art-e8e326545b03427e8fba2dba3f7837c82025-02-27T05:37:17ZengPublic Library of Science (PLoS)PLoS ONE1932-62032017-01-01128e018316610.1371/journal.pone.0183166Developmental vascular remodeling defects and postnatal kidney failure in mice lacking Gpr116 (Adgrf5) and Eltd1 (Adgrl4).Shun LuShuya LiuAstrid WietelmannBaktybek KojonazarovAnn AtzbergerCong TangRalph Theo SchermulyHermann-Josef GröneStefan OffermannsGPR116 (ADGRF5) and ELTD1 (ADGRL4) belong to different subfamilies of the adhesion G-protein-coupled receptor group but are both expressed in endothelial cells. We therefore analyzed their functions in mice lacking these receptors. While loss of GPR116 or ELTD1 alone had no obvious effect on cardiovascular or kidney function, mice lacking both, GPR116 and ELTD1, showed malformations of the aortic arch arteries and the cardiac outflow tract leading to perinatal lethality in about 50% of the mutants. In addition to cardiovascular malformations, surviving mice developed renal thrombotic microangiopathy as well as hemolysis and splenomegaly, and their lifespan was significantly reduced. Loss of GPR116 and ELTD1 specifically in endothelial cells or neural crest-derived cells did not recapitulate any of the phenotypes observed in GPR116-ELTD1 double deficient mice, indicating that loss of GPR116 and ELTD1 expressed by other cells accounts for the observed cardiovascular and renal defects.https://journals.plos.org/plosone/article/file?id=10.1371/journal.pone.0183166&type=printable |
| spellingShingle | Shun Lu Shuya Liu Astrid Wietelmann Baktybek Kojonazarov Ann Atzberger Cong Tang Ralph Theo Schermuly Hermann-Josef Gröne Stefan Offermanns Developmental vascular remodeling defects and postnatal kidney failure in mice lacking Gpr116 (Adgrf5) and Eltd1 (Adgrl4). PLoS ONE |
| title | Developmental vascular remodeling defects and postnatal kidney failure in mice lacking Gpr116 (Adgrf5) and Eltd1 (Adgrl4). |
| title_full | Developmental vascular remodeling defects and postnatal kidney failure in mice lacking Gpr116 (Adgrf5) and Eltd1 (Adgrl4). |
| title_fullStr | Developmental vascular remodeling defects and postnatal kidney failure in mice lacking Gpr116 (Adgrf5) and Eltd1 (Adgrl4). |
| title_full_unstemmed | Developmental vascular remodeling defects and postnatal kidney failure in mice lacking Gpr116 (Adgrf5) and Eltd1 (Adgrl4). |
| title_short | Developmental vascular remodeling defects and postnatal kidney failure in mice lacking Gpr116 (Adgrf5) and Eltd1 (Adgrl4). |
| title_sort | developmental vascular remodeling defects and postnatal kidney failure in mice lacking gpr116 adgrf5 and eltd1 adgrl4 |
| url | https://journals.plos.org/plosone/article/file?id=10.1371/journal.pone.0183166&type=printable |
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