Pub Date : 2021-09-01DOI: 10.1097/jova.0000000000000016
Aqsa Shakoor, June K Wu, Ajit Muley, Christopher Kitajewski, Joseph D McCarron, Noa Shapiro-Franklin, Rozelle Corda, Sophia Chrisomalis-Dring, Paul J Chai, Carrie J Shawber
Objectives: Chylothorax following cardiac surgery for congenital cardiac anomalies is a complication associated with severe morbidities and mortality. We hypothesize that there are intrinsic defects in the lymphatics of congenital cardiac patients.
Methods: Postsurgical chylothorax lymphatic endothelial cells (pcLECs) (n = 10) were isolated from the chylous fluid from congenital cardiac defect patients, and characterized by fluorescent-activated cell sorting, immunofluorescent staining, and quantitative RT-PCR. Results were compared to normal human dermal lymphatic endothelial cells (HdLECs). pcLECs (n = 3) and HdLECs were xenografted into immunocompromised mice. Implants and postoperative chylothorax patient's pulmonary tissues were characterized by immunostaining for lymphatic endothelial proteins.
Results: pcLECs expressed endothelial markers VECADHERIN, CD31, VEGFR2, lymphatic endothelial markers PROX1, podoplanin, VEGFR3, and progenitor endothelial markers CD90 and CD146. However, pcLECs had key differences relative to HdLECs, including altered expression and mislocalization of junctional proteins (VECADHERIN and CD31), and essential endothelial proteins, VEGFR2, VEGFR3, and PROX1. When xenografted in mice, pcLECs formed dilated lymphatic channels with poor cell-cell association. Similar to congenital lymphatic anomalies, the pulmonary lymphatics were dilated in a patient who developed postoperative chylothorax after cardiac surgery.
Conclusions: Recent studies have shown that some postoperative chylothoraces in congenital cardiac anomalies are associated with anatomical lymphatic defects. We found that pcLECs have defects in expression and localization of proteins necessary to maintain lymphatic specification and function. This pcLEC phenotype is similar to that observed in lymphatic endothelial cells from congenital lymphatic anomalies. Co-existence of lymphatic anomalies should be considered as a feature of congenital cardiac anomalies.
{"title":"Lymphatic Endothelial Cell Defects in Congenital Cardiac Patients With Postoperative Chylothorax.","authors":"Aqsa Shakoor, June K Wu, Ajit Muley, Christopher Kitajewski, Joseph D McCarron, Noa Shapiro-Franklin, Rozelle Corda, Sophia Chrisomalis-Dring, Paul J Chai, Carrie J Shawber","doi":"10.1097/jova.0000000000000016","DOIUrl":"https://doi.org/10.1097/jova.0000000000000016","url":null,"abstract":"<p><strong>Objectives: </strong>Chylothorax following cardiac surgery for congenital cardiac anomalies is a complication associated with severe morbidities and mortality. We hypothesize that there are intrinsic defects in the lymphatics of congenital cardiac patients.</p><p><strong>Methods: </strong>Postsurgical chylothorax lymphatic endothelial cells (pcLECs) (n = 10) were isolated from the chylous fluid from congenital cardiac defect patients, and characterized by fluorescent-activated cell sorting, immunofluorescent staining, and quantitative RT-PCR. Results were compared to normal human dermal lymphatic endothelial cells (HdLECs). pcLECs (n = 3) and HdLECs were xenografted into immunocompromised mice. Implants and postoperative chylothorax patient's pulmonary tissues were characterized by immunostaining for lymphatic endothelial proteins.</p><p><strong>Results: </strong>pcLECs expressed endothelial markers VECADHERIN, CD31, VEGFR2, lymphatic endothelial markers PROX1, podoplanin, VEGFR3, and progenitor endothelial markers CD90 and CD146. However, pcLECs had key differences relative to HdLECs, including altered expression and mislocalization of junctional proteins (VECADHERIN and CD31), and essential endothelial proteins, VEGFR2, VEGFR3, and PROX1. When xenografted in mice, pcLECs formed dilated lymphatic channels with poor cell-cell association. Similar to congenital lymphatic anomalies, the pulmonary lymphatics were dilated in a patient who developed postoperative chylothorax after cardiac surgery.</p><p><strong>Conclusions: </strong>Recent studies have shown that some postoperative chylothoraces in congenital cardiac anomalies are associated with anatomical lymphatic defects. We found that pcLECs have defects in expression and localization of proteins necessary to maintain lymphatic specification and function. This pcLEC phenotype is similar to that observed in lymphatic endothelial cells from congenital lymphatic anomalies. Co-existence of lymphatic anomalies should be considered as a feature of congenital cardiac anomalies.</p>","PeriodicalId":74008,"journal":{"name":"Journal of vascular anomalies","volume":"2 3","pages":""},"PeriodicalIF":0.0,"publicationDate":"2021-09-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"https://www.ncbi.nlm.nih.gov/pmc/articles/PMC8478352/pdf/","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"39495375","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"OA","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2021-08-27DOI: 10.1097/jova.0000000000000022
S. P. Koh, H. Brasch, J. Patel, N. Bockett, Erin Paterson, P. Davis, Swee T. Tan
{"title":"Expression of Cathepsins B, D, and G in Hypertrophic Port-wine Stain","authors":"S. P. Koh, H. Brasch, J. Patel, N. Bockett, Erin Paterson, P. Davis, Swee T. Tan","doi":"10.1097/jova.0000000000000022","DOIUrl":"https://doi.org/10.1097/jova.0000000000000022","url":null,"abstract":"","PeriodicalId":74008,"journal":{"name":"Journal of vascular anomalies","volume":"104 1","pages":""},"PeriodicalIF":0.0,"publicationDate":"2021-08-27","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"78747584","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2021-08-19DOI: 10.1097/jova.0000000000000023
João Euzébio Encarnação Ferreira, Marina Vilela Chagas Ferreira, R. Zatz, G. Foronda, R. Gemperli, D. Goldenberg
{"title":"Safe Use of Propranolol in a Patient With PHACES Syndrome: A Case Report","authors":"João Euzébio Encarnação Ferreira, Marina Vilela Chagas Ferreira, R. Zatz, G. Foronda, R. Gemperli, D. Goldenberg","doi":"10.1097/jova.0000000000000023","DOIUrl":"https://doi.org/10.1097/jova.0000000000000023","url":null,"abstract":"","PeriodicalId":74008,"journal":{"name":"Journal of vascular anomalies","volume":"05 1","pages":""},"PeriodicalIF":0.0,"publicationDate":"2021-08-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"83641709","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2021-08-05DOI: 10.1097/jova.0000000000000021
F. Worede, E. Granquist, A. Srinivasan, S. Vatsky, A. Cahill
{"title":"Direct Nidal Ethyl Vinyl Alcohol Copolymer Embolization With Flow Reduction Techniques as a Primary Treatment for Intraosseous Mandibular Arteriolovenous Malformations","authors":"F. Worede, E. Granquist, A. Srinivasan, S. Vatsky, A. Cahill","doi":"10.1097/jova.0000000000000021","DOIUrl":"https://doi.org/10.1097/jova.0000000000000021","url":null,"abstract":"","PeriodicalId":74008,"journal":{"name":"Journal of vascular anomalies","volume":"3 1","pages":""},"PeriodicalIF":0.0,"publicationDate":"2021-08-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"89218813","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2021-08-05DOI: 10.1097/jova.0000000000000020
Sam Siljee, A. Gower, H. Brasch, J. Patel, N. Bockett, T. Itinteang, S. Tan
{"title":"Expression of Angiotensin II Receptor 2 in Microcystic Lymphatic Malformation","authors":"Sam Siljee, A. Gower, H. Brasch, J. Patel, N. Bockett, T. Itinteang, S. Tan","doi":"10.1097/jova.0000000000000020","DOIUrl":"https://doi.org/10.1097/jova.0000000000000020","url":null,"abstract":"","PeriodicalId":74008,"journal":{"name":"Journal of vascular anomalies","volume":"43 1","pages":""},"PeriodicalIF":0.0,"publicationDate":"2021-08-05","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"85236946","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2021-07-26DOI: 10.1097/jova.0000000000000019
Sigurd Berger, R. Andersen, J. Jakobsen, O. Geier, A. Abildgaard, E. Dorenberg
{"title":"Precontrast Magnetic Resonance Imaging Findings and Assessment of Dynamic Contrast-Enhanced Magnetic Resonance Imaging in Low- and High-Flow Vascular Malformations","authors":"Sigurd Berger, R. Andersen, J. Jakobsen, O. Geier, A. Abildgaard, E. Dorenberg","doi":"10.1097/jova.0000000000000019","DOIUrl":"https://doi.org/10.1097/jova.0000000000000019","url":null,"abstract":"","PeriodicalId":74008,"journal":{"name":"Journal of vascular anomalies","volume":"100 1","pages":""},"PeriodicalIF":0.0,"publicationDate":"2021-07-26","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"77326623","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2021-07-19DOI: 10.1097/JOVA.0000000000000014
Alejandro Celis-Jiménez, A. Meza-Lopez, Carmen Leticia Santana-Cárdenas, Delma Gabriela Cortés-Barrios, Bertha Graciela Requena-Torres, Héctor Manuel Tiznado-García
{"title":"Management of Kasabach–Merritt Phenomenon With Transarterial Embolization in Patients With Kaposiform Hemangioendothelioma","authors":"Alejandro Celis-Jiménez, A. Meza-Lopez, Carmen Leticia Santana-Cárdenas, Delma Gabriela Cortés-Barrios, Bertha Graciela Requena-Torres, Héctor Manuel Tiznado-García","doi":"10.1097/JOVA.0000000000000014","DOIUrl":"https://doi.org/10.1097/JOVA.0000000000000014","url":null,"abstract":"","PeriodicalId":74008,"journal":{"name":"Journal of vascular anomalies","volume":"102 1","pages":""},"PeriodicalIF":0.0,"publicationDate":"2021-07-19","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"89929408","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2021-07-01DOI: 10.1097/jova.0000000000000018
D. Cooke, I. Frieden, K. Shimano
{"title":"Angiographic Evidence of Response to Trametinib Therapy for a Spinal Cord Arteriovenous Malformation","authors":"D. Cooke, I. Frieden, K. Shimano","doi":"10.1097/jova.0000000000000018","DOIUrl":"https://doi.org/10.1097/jova.0000000000000018","url":null,"abstract":"","PeriodicalId":74008,"journal":{"name":"Journal of vascular anomalies","volume":"5 1","pages":""},"PeriodicalIF":0.0,"publicationDate":"2021-07-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"87169603","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2021-06-01DOI: 10.1097/JOVA.0000000000000012
Julia K. Tanguay, H. Erdemir, Grace Onimoe
{"title":"Novel Treatment of a Diffuse Infantile Hepatic Hemangioma With a Selective Beta-1 Antagonist","authors":"Julia K. Tanguay, H. Erdemir, Grace Onimoe","doi":"10.1097/JOVA.0000000000000012","DOIUrl":"https://doi.org/10.1097/JOVA.0000000000000012","url":null,"abstract":"","PeriodicalId":74008,"journal":{"name":"Journal of vascular anomalies","volume":"149 1","pages":""},"PeriodicalIF":0.0,"publicationDate":"2021-06-01","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"88629153","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
Pub Date : 2020-04-08DOI: 10.1007/978-3-030-25624-1
Tara L. Rosenberg, David H. Darrow
{"title":"Vascular Anomalies: A Guide for the Hematologist/Oncologist","authors":"Tara L. Rosenberg, David H. Darrow","doi":"10.1007/978-3-030-25624-1","DOIUrl":"https://doi.org/10.1007/978-3-030-25624-1","url":null,"abstract":"","PeriodicalId":74008,"journal":{"name":"Journal of vascular anomalies","volume":"27 1","pages":""},"PeriodicalIF":0.0,"publicationDate":"2020-04-08","publicationTypes":"Journal Article","fieldsOfStudy":null,"isOpenAccess":false,"openAccessPdf":"","citationCount":null,"resultStr":null,"platform":"Semanticscholar","paperid":"87336775","PeriodicalName":null,"FirstCategoryId":null,"ListUrlMain":null,"RegionNum":0,"RegionCategory":"","ArticlePicture":[],"TitleCN":null,"AbstractTextCN":null,"PMCID":"","EPubDate":null,"PubModel":null,"JCR":null,"JCRName":null,"Score":null,"Total":0}
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