Serum endostatin levels are associated with diffusion capacity and with tuberous sclerosis- associated lymphangioleiomyomatosis
Endostatin is a naturally occurring collagen fragment with anti-angiogenic properties. We investigated the association between serum endostatin levels and DLCO in a cohort of patients with lymphangioleiomyomatosis (LAM). Associations of endostatin levels to clinical features of LAM were explored usi...
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| Published in | Orphanet journal of rare diseases Vol. 14; no. 1; pp. 72 - 5 |
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| Main Authors | , , , , , , , , , , , , |
| Format | Journal Article |
| Language | English |
| Published |
London
BioMed Central
29.03.2019
BioMed Central Ltd BMC |
| Subjects | |
| Online Access | Get full text |
| ISSN | 1750-1172 1750-1172 |
| DOI | 10.1186/s13023-019-1050-4 |
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| Abstract | Endostatin is a naturally occurring collagen fragment with anti-angiogenic properties. We investigated the association between serum endostatin levels and DLCO in a cohort of patients with lymphangioleiomyomatosis (LAM). Associations of endostatin levels to clinical features of LAM were explored using logistic regression models. Endostatin levels were associated with DLCO and were higher in subjects with TSC-associated LAM compared to sporadic LAM. These data suggest that endostatin could be a predictive biomarker of decline in DLCO and that germline mutational inactivation of the TSC1 or TSC2 gene is associated with higher endostatin levels. These findings could offer novel insights into the pathogenesis of LAM. |
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| AbstractList | Endostatin is a naturally occurring collagen fragment with anti-angiogenic properties. We investigated the association between serum endostatin levels and DLCO in a cohort of patients with lymphangioleiomyomatosis (LAM). Associations of endostatin levels to clinical features of LAM were explored using logistic regression models. Endostatin levels were associated with DLCO and were higher in subjects with TSC-associated LAM compared to sporadic LAM. These data suggest that endostatin could be a predictive biomarker of decline in DLCO and that germline mutational inactivation of the TSC1 or TSC2 gene is associated with higher endostatin levels. These findings could offer novel insights into the pathogenesis of LAM. Endostatin is a naturally occurring collagen fragment with anti-angiogenic properties. We investigated the association between serum endostatin levels and DLCO in a cohort of patients with lymphangioleiomyomatosis (LAM). Associations of endostatin levels to clinical features of LAM were explored using logistic regression models. Endostatin levels were associated with DLCO and were higher in subjects with TSC-associated LAM compared to sporadic LAM. These data suggest that endostatin could be a predictive biomarker of decline in DLCO and that germline mutational inactivation of the TSC1 or TSC2 gene is associated with higher endostatin levels. These findings could offer novel insights into the pathogenesis of LAM. Keywords: LAM, TSC, Endostatin, DLCO, Isolated decrease Endostatin is a naturally occurring collagen fragment with anti-angiogenic properties. We investigated the association between serum endostatin levels and DLCO in a cohort of patients with lymphangioleiomyomatosis (LAM). Associations of endostatin levels to clinical features of LAM were explored using logistic regression models. Endostatin levels were associated with DLCO and were higher in subjects with TSC-associated LAM compared to sporadic LAM. These data suggest that endostatin could be a predictive biomarker of decline in DLCO and that germline mutational inactivation of the TSC1 or TSC2 gene is associated with higher endostatin levels. These findings could offer novel insights into the pathogenesis of LAM.Endostatin is a naturally occurring collagen fragment with anti-angiogenic properties. We investigated the association between serum endostatin levels and DLCO in a cohort of patients with lymphangioleiomyomatosis (LAM). Associations of endostatin levels to clinical features of LAM were explored using logistic regression models. Endostatin levels were associated with DLCO and were higher in subjects with TSC-associated LAM compared to sporadic LAM. These data suggest that endostatin could be a predictive biomarker of decline in DLCO and that germline mutational inactivation of the TSC1 or TSC2 gene is associated with higher endostatin levels. These findings could offer novel insights into the pathogenesis of LAM. Abstract Endostatin is a naturally occurring collagen fragment with anti-angiogenic properties. We investigated the association between serum endostatin levels and DLCO in a cohort of patients with lymphangioleiomyomatosis (LAM). Associations of endostatin levels to clinical features of LAM were explored using logistic regression models. Endostatin levels were associated with DLCO and were higher in subjects with TSC-associated LAM compared to sporadic LAM. These data suggest that endostatin could be a predictive biomarker of decline in DLCO and that germline mutational inactivation of the TSC1 or TSC2 gene is associated with higher endostatin levels. These findings could offer novel insights into the pathogenesis of LAM. |
| ArticleNumber | 72 |
| Audience | Academic |
| Author | Poli, Sergio El-Chemaly, Souheil Bagwe, Shefali Stump, Benjamin Rosas, Ivan Thiele, Elizabeth A. Shrestha, Shikshya Goldberg, Hilary J. Kidambi, Pranav Henske, Elizabeth P. Lamattina, Anthony M. Courtwright, Andrew Louis, Pierce H. |
| Author_xml | – sequence: 1 givenname: Anthony M. surname: Lamattina fullname: Lamattina, Anthony M. organization: Division of Pulmonary and Critical Care Medicine, Brigham and Women’s Hospital, Harvard Medical School – sequence: 2 givenname: Sergio surname: Poli fullname: Poli, Sergio organization: Division of Pulmonary and Critical Care Medicine, Brigham and Women’s Hospital, Harvard Medical School – sequence: 3 givenname: Pranav surname: Kidambi fullname: Kidambi, Pranav organization: Division of Pulmonary and Critical Care Medicine, Brigham and Women’s Hospital, Harvard Medical School – sequence: 4 givenname: Shefali surname: Bagwe fullname: Bagwe, Shefali organization: Division of Pulmonary and Critical Care Medicine, Brigham and Women’s Hospital, Harvard Medical School – sequence: 5 givenname: Andrew surname: Courtwright fullname: Courtwright, Andrew organization: Division of Pulmonary and Critical Care Medicine, Hospital of the University of Pennsylvania – sequence: 6 givenname: Pierce H. surname: Louis fullname: Louis, Pierce H. organization: Division of Pulmonary and Critical Care Medicine, Brigham and Women’s Hospital, Harvard Medical School – sequence: 7 givenname: Shikshya surname: Shrestha fullname: Shrestha, Shikshya organization: Division of Pulmonary and Critical Care Medicine, Brigham and Women’s Hospital, Harvard Medical School – sequence: 8 givenname: Benjamin surname: Stump fullname: Stump, Benjamin organization: Division of Pulmonary and Critical Care Medicine, Brigham and Women’s Hospital, Harvard Medical School – sequence: 9 givenname: Hilary J. surname: Goldberg fullname: Goldberg, Hilary J. organization: Division of Pulmonary and Critical Care Medicine, Brigham and Women’s Hospital, Harvard Medical School – sequence: 10 givenname: Elizabeth A. surname: Thiele fullname: Thiele, Elizabeth A. organization: Department of Neurology, Massachusetts General Hospital, Harvard Medical School – sequence: 11 givenname: Ivan surname: Rosas fullname: Rosas, Ivan organization: Division of Pulmonary and Critical Care Medicine, Brigham and Women’s Hospital, Harvard Medical School – sequence: 12 givenname: Elizabeth P. surname: Henske fullname: Henske, Elizabeth P. organization: Division of Pulmonary and Critical Care Medicine, Brigham and Women’s Hospital, Harvard Medical School – sequence: 13 givenname: Souheil surname: El-Chemaly fullname: El-Chemaly, Souheil email: sel-chemaly@bwh.harvard.edu organization: Division of Pulmonary and Critical Care Medicine, Brigham and Women’s Hospital, Harvard Medical School |
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| Cites_doi | 10.1183/09031936.00076209 10.1126/science.1260419 10.1016/j.pediatrneurol.2013.08.001 10.1186/1471-2105-12-77 10.1006/bbrc.1999.1342 10.1158/0008-5472.CAN-08-0295 10.1186/s13075-015-0756-5 10.1165/rcmb.2014-0224OC 10.1378/chest.10-0573 10.1016/S0092-8674(00)81848-6 10.1016/j.chest.2018.08.1029 |
| ContentType | Journal Article |
| Copyright | The Author(s). 2019 COPYRIGHT 2019 BioMed Central Ltd. 2019. This work is licensed under http://creativecommons.org/licenses/by/4.0/ (the “License”). Notwithstanding the ProQuest Terms and Conditions, you may use this content in accordance with the terms of the License. |
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| Keywords | LAM DLCO TSC Isolated decrease Endostatin |
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| Snippet | Endostatin is a naturally occurring collagen fragment with anti-angiogenic properties. We investigated the association between serum endostatin levels and DLCO... Abstract Endostatin is a naturally occurring collagen fragment with anti-angiogenic properties. We investigated the association between serum endostatin levels... |
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| StartPage | 72 |
| SubjectTerms | Adult Age Angiogenesis Angiogenesis inhibitors Biomarkers - blood Cohort Studies Collagen Disease DLCO Endostatin Endostatins - blood Endostatins - genetics Female Gene expression Gene Silencing Genes Germ-Line Mutation Human Genetics Humans Isolated decrease LAM Letter to the Editor Lymphangioleiomyomatosis - blood Lymphangioleiomyomatosis - complications Lymphangioleiomyomatosis - genetics Lymphangioleiomyomatosis - physiopathology Lymphatic diseases Medical research Medicine Medicine & Public Health Middle Aged Pharmacology/Toxicology Rare diseases Rare pulmonary diseases Regression analysis TSC TSC1 gene TSC2 gene Tuberous sclerosis Tuberous Sclerosis - complications Tuberous Sclerosis Complex 1 Protein - genetics Tuberous Sclerosis Complex 2 Protein - genetics Variables Vascular endothelial growth factor Womens health |
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| Title | Serum endostatin levels are associated with diffusion capacity and with tuberous sclerosis- associated lymphangioleiomyomatosis |
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