Vascular Anomalies Associated with Esophageal Atresia and Tracheoesophageal Fistula

Objective To report the incidence of congenital vascular anomalies in a cohort of patients with esophageal atresia (EA) and tracheoesophageal fistula (TEF) while describing the clinical presentation, diagnosis, and consequences, and to evaluate the diagnostic value of esophagram in diagnosing an abe...

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Veröffentlicht in:The Journal of pediatrics 2015-05, Vol.166 (5), p.1140-1144.e2
Hauptverfasser: Berthet, Stéphanie, MD, Tenisch, Estelle, MD, Miron, Marie Claude, MD, Alami, Nassiba, MD, Timmons, Jennifer, MD, Aspirot, Ann, MD, Faure, Christophe, MD
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container_end_page 1144.e2
container_issue 5
container_start_page 1140
container_title The Journal of pediatrics
container_volume 166
creator Berthet, Stéphanie, MD
Tenisch, Estelle, MD
Miron, Marie Claude, MD
Alami, Nassiba, MD
Timmons, Jennifer, MD
Aspirot, Ann, MD
Faure, Christophe, MD
description Objective To report the incidence of congenital vascular anomalies in a cohort of patients with esophageal atresia (EA) and tracheoesophageal fistula (TEF) while describing the clinical presentation, diagnosis, and consequences, and to evaluate the diagnostic value of esophagram in diagnosing an aberrant right subclavian artery (ARSA). Methods All patients born with EA/TEF between 2005 and 2013 were studied. Preoperative echocardiography reports, surgical descriptions of primary esophageal repair, and esophagrams were reviewed retrospectively. Results Of the 76 children born with EA/TEF included in this study, 14 (18%) had a vascular malformation. The incidence of a right aortic arch (RAA) was 6% (5 of 76), and that of an aberrant right subclavian artery (ARSA) was 12% (9 of 76). RAA was diagnosed in the neonatal period by echocardiography (4 of 5) or surgery (1 of 5), and ARSA was diagnosed by echocardiography (7 of 9) or later on the esophagram (2 of 9). Respiratory and/or digestive symptoms occurred in 9 of the 14 patients with vascular malformation. Both long-gap EA and severe cardiac malformations necessitating surgery were significantly associated with vascular anomalies ( P  2 weeks) use of a nasogastric tube is considered.
doi_str_mv 10.1016/j.jpeds.2015.01.038
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Methods All patients born with EA/TEF between 2005 and 2013 were studied. Preoperative echocardiography reports, surgical descriptions of primary esophageal repair, and esophagrams were reviewed retrospectively. Results Of the 76 children born with EA/TEF included in this study, 14 (18%) had a vascular malformation. The incidence of a right aortic arch (RAA) was 6% (5 of 76), and that of an aberrant right subclavian artery (ARSA) was 12% (9 of 76). RAA was diagnosed in the neonatal period by echocardiography (4 of 5) or surgery (1 of 5), and ARSA was diagnosed by echocardiography (7 of 9) or later on the esophagram (2 of 9). Respiratory and/or digestive symptoms occurred in 9 of the 14 patients with vascular malformation. Both long-gap EA and severe cardiac malformations necessitating surgery were significantly associated with vascular anomalies ( P  &lt; .05). The sensitivity of the esophagram for diagnosing ARSA was 66%, the specificity was 98%, the negative predictive value was 95%, and the positive predictive value was 85%. Conclusion ARSA and RAA have an incidence of 12% and 6% respectively, in patients with EA/TEF. A computed tomography angioscan is recommended to rule out such malformations when stenting of the esophagus is indicated, before esophageal replacement surgery, and when prolonged (&gt;2 weeks) use of a nasogastric tube is considered.</description><identifier>ISSN: 0022-3476</identifier><identifier>EISSN: 1097-6833</identifier><identifier>DOI: 10.1016/j.jpeds.2015.01.038</identifier><identifier>PMID: 25720367</identifier><language>eng</language><publisher>United States: Elsevier Inc</publisher><subject>Aorta, Thoracic - abnormalities ; Child ; Child, Preschool ; Echocardiography ; Esophageal Atresia - complications ; Esophagus - abnormalities ; Female ; Humans ; Male ; Pediatrics ; Predictive Value of Tests ; Reproducibility of Results ; Retrospective Studies ; Sensitivity and Specificity ; Subclavian Artery - pathology ; Tracheoesophageal Fistula - complications ; Vascular Malformations - complications</subject><ispartof>The Journal of pediatrics, 2015-05, Vol.166 (5), p.1140-1144.e2</ispartof><rights>Elsevier Inc.</rights><rights>2015 Elsevier Inc.</rights><rights>Copyright © 2015 Elsevier Inc. All rights reserved.</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><citedby>FETCH-LOGICAL-c414t-cb6256b70cc802582f7ff9312996ac5b07ce7b61040721ed2256030a683889a23</citedby><cites>FETCH-LOGICAL-c414t-cb6256b70cc802582f7ff9312996ac5b07ce7b61040721ed2256030a683889a23</cites></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><linktohtml>$$Uhttps://www.sciencedirect.com/science/article/pii/S0022347615000852$$EHTML$$P50$$Gelsevier$$H</linktohtml><link.rule.ids>314,776,780,3537,27903,27904,65309</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/25720367$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Berthet, Stéphanie, MD</creatorcontrib><creatorcontrib>Tenisch, Estelle, MD</creatorcontrib><creatorcontrib>Miron, Marie Claude, MD</creatorcontrib><creatorcontrib>Alami, Nassiba, MD</creatorcontrib><creatorcontrib>Timmons, Jennifer, MD</creatorcontrib><creatorcontrib>Aspirot, Ann, MD</creatorcontrib><creatorcontrib>Faure, Christophe, MD</creatorcontrib><title>Vascular Anomalies Associated with Esophageal Atresia and Tracheoesophageal Fistula</title><title>The Journal of pediatrics</title><addtitle>J Pediatr</addtitle><description>Objective To report the incidence of congenital vascular anomalies in a cohort of patients with esophageal atresia (EA) and tracheoesophageal fistula (TEF) while describing the clinical presentation, diagnosis, and consequences, and to evaluate the diagnostic value of esophagram in diagnosing an aberrant right subclavian artery (ARSA). Methods All patients born with EA/TEF between 2005 and 2013 were studied. Preoperative echocardiography reports, surgical descriptions of primary esophageal repair, and esophagrams were reviewed retrospectively. Results Of the 76 children born with EA/TEF included in this study, 14 (18%) had a vascular malformation. The incidence of a right aortic arch (RAA) was 6% (5 of 76), and that of an aberrant right subclavian artery (ARSA) was 12% (9 of 76). RAA was diagnosed in the neonatal period by echocardiography (4 of 5) or surgery (1 of 5), and ARSA was diagnosed by echocardiography (7 of 9) or later on the esophagram (2 of 9). Respiratory and/or digestive symptoms occurred in 9 of the 14 patients with vascular malformation. Both long-gap EA and severe cardiac malformations necessitating surgery were significantly associated with vascular anomalies ( P  &lt; .05). The sensitivity of the esophagram for diagnosing ARSA was 66%, the specificity was 98%, the negative predictive value was 95%, and the positive predictive value was 85%. Conclusion ARSA and RAA have an incidence of 12% and 6% respectively, in patients with EA/TEF. A computed tomography angioscan is recommended to rule out such malformations when stenting of the esophagus is indicated, before esophageal replacement surgery, and when prolonged (&gt;2 weeks) use of a nasogastric tube is considered.</description><subject>Aorta, Thoracic - abnormalities</subject><subject>Child</subject><subject>Child, Preschool</subject><subject>Echocardiography</subject><subject>Esophageal Atresia - complications</subject><subject>Esophagus - abnormalities</subject><subject>Female</subject><subject>Humans</subject><subject>Male</subject><subject>Pediatrics</subject><subject>Predictive Value of Tests</subject><subject>Reproducibility of Results</subject><subject>Retrospective Studies</subject><subject>Sensitivity and Specificity</subject><subject>Subclavian Artery - pathology</subject><subject>Tracheoesophageal Fistula - complications</subject><subject>Vascular Malformations - complications</subject><issn>0022-3476</issn><issn>1097-6833</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2015</creationdate><recordtype>article</recordtype><sourceid>EIF</sourceid><recordid>eNqFkcFu1DAQhi0EotvCEyChHLkkjO3ETg4graq2IFXqgYWr5TgT1iEbL54E1LfhWfpkeNnSSlw4-eDvn9F8P2OvOBQcuHo7FMMeOyoE8KoAXoCsn7AVh0bnqpbyKVsBCJHLUqsTdko0AEBTAjxnJ6LSAqTSK7b5Yskto43Zego7O3qkbE0UnLczdtlPP2-zCwr7rf2KdszWc0TyNrNTd_drE63bYsDH70tPcxr2gj3r7Uj48v49Y58vLzbnH_Lrm6uP5-vr3JW8nHPXKlGpVoNzNYiqFr3u-0Zy0TTKuqoF7VC3ikMJWnDsRKJBgk3X1XVjhTxjb45z9zF8X5Bms_PkcBzthGEhw5XWUktRVQmVR9TFQBSxN_vodzbeGg7moNMM5o9Oc9BpgJukM6Ve3y9Y2h12D5m__hLw7ghgOvOHx2jIeZwcdj6im00X_H8WvP8n70Y_eWfHb3iLNIQlTsmg4YaEAfPp0OihUF6lMutKyN9egZv2</recordid><startdate>20150501</startdate><enddate>20150501</enddate><creator>Berthet, Stéphanie, MD</creator><creator>Tenisch, Estelle, MD</creator><creator>Miron, Marie Claude, MD</creator><creator>Alami, Nassiba, MD</creator><creator>Timmons, Jennifer, MD</creator><creator>Aspirot, Ann, MD</creator><creator>Faure, Christophe, MD</creator><general>Elsevier Inc</general><scope>CGR</scope><scope>CUY</scope><scope>CVF</scope><scope>ECM</scope><scope>EIF</scope><scope>NPM</scope><scope>AAYXX</scope><scope>CITATION</scope><scope>7X8</scope></search><sort><creationdate>20150501</creationdate><title>Vascular Anomalies Associated with Esophageal Atresia and Tracheoesophageal Fistula</title><author>Berthet, Stéphanie, MD ; Tenisch, Estelle, MD ; Miron, Marie Claude, MD ; Alami, Nassiba, MD ; Timmons, Jennifer, MD ; Aspirot, Ann, MD ; Faure, Christophe, MD</author></sort><facets><frbrtype>5</frbrtype><frbrgroupid>cdi_FETCH-LOGICAL-c414t-cb6256b70cc802582f7ff9312996ac5b07ce7b61040721ed2256030a683889a23</frbrgroupid><rsrctype>articles</rsrctype><prefilter>articles</prefilter><language>eng</language><creationdate>2015</creationdate><topic>Aorta, Thoracic - abnormalities</topic><topic>Child</topic><topic>Child, Preschool</topic><topic>Echocardiography</topic><topic>Esophageal Atresia - complications</topic><topic>Esophagus - abnormalities</topic><topic>Female</topic><topic>Humans</topic><topic>Male</topic><topic>Pediatrics</topic><topic>Predictive Value of Tests</topic><topic>Reproducibility of Results</topic><topic>Retrospective Studies</topic><topic>Sensitivity and Specificity</topic><topic>Subclavian Artery - pathology</topic><topic>Tracheoesophageal Fistula - complications</topic><topic>Vascular Malformations - complications</topic><toplevel>peer_reviewed</toplevel><toplevel>online_resources</toplevel><creatorcontrib>Berthet, Stéphanie, MD</creatorcontrib><creatorcontrib>Tenisch, Estelle, MD</creatorcontrib><creatorcontrib>Miron, Marie Claude, MD</creatorcontrib><creatorcontrib>Alami, Nassiba, MD</creatorcontrib><creatorcontrib>Timmons, Jennifer, MD</creatorcontrib><creatorcontrib>Aspirot, Ann, MD</creatorcontrib><creatorcontrib>Faure, Christophe, MD</creatorcontrib><collection>Medline</collection><collection>MEDLINE</collection><collection>MEDLINE (Ovid)</collection><collection>MEDLINE</collection><collection>MEDLINE</collection><collection>PubMed</collection><collection>CrossRef</collection><collection>MEDLINE - Academic</collection><jtitle>The Journal of pediatrics</jtitle></facets><delivery><delcategory>Remote Search Resource</delcategory><fulltext>fulltext</fulltext></delivery><addata><au>Berthet, Stéphanie, MD</au><au>Tenisch, Estelle, MD</au><au>Miron, Marie Claude, MD</au><au>Alami, Nassiba, MD</au><au>Timmons, Jennifer, MD</au><au>Aspirot, Ann, MD</au><au>Faure, Christophe, MD</au><format>journal</format><genre>article</genre><ristype>JOUR</ristype><atitle>Vascular Anomalies Associated with Esophageal Atresia and Tracheoesophageal Fistula</atitle><jtitle>The Journal of pediatrics</jtitle><addtitle>J Pediatr</addtitle><date>2015-05-01</date><risdate>2015</risdate><volume>166</volume><issue>5</issue><spage>1140</spage><epage>1144.e2</epage><pages>1140-1144.e2</pages><issn>0022-3476</issn><eissn>1097-6833</eissn><abstract>Objective To report the incidence of congenital vascular anomalies in a cohort of patients with esophageal atresia (EA) and tracheoesophageal fistula (TEF) while describing the clinical presentation, diagnosis, and consequences, and to evaluate the diagnostic value of esophagram in diagnosing an aberrant right subclavian artery (ARSA). Methods All patients born with EA/TEF between 2005 and 2013 were studied. Preoperative echocardiography reports, surgical descriptions of primary esophageal repair, and esophagrams were reviewed retrospectively. Results Of the 76 children born with EA/TEF included in this study, 14 (18%) had a vascular malformation. The incidence of a right aortic arch (RAA) was 6% (5 of 76), and that of an aberrant right subclavian artery (ARSA) was 12% (9 of 76). RAA was diagnosed in the neonatal period by echocardiography (4 of 5) or surgery (1 of 5), and ARSA was diagnosed by echocardiography (7 of 9) or later on the esophagram (2 of 9). Respiratory and/or digestive symptoms occurred in 9 of the 14 patients with vascular malformation. Both long-gap EA and severe cardiac malformations necessitating surgery were significantly associated with vascular anomalies ( P  &lt; .05). The sensitivity of the esophagram for diagnosing ARSA was 66%, the specificity was 98%, the negative predictive value was 95%, and the positive predictive value was 85%. Conclusion ARSA and RAA have an incidence of 12% and 6% respectively, in patients with EA/TEF. A computed tomography angioscan is recommended to rule out such malformations when stenting of the esophagus is indicated, before esophageal replacement surgery, and when prolonged (&gt;2 weeks) use of a nasogastric tube is considered.</abstract><cop>United States</cop><pub>Elsevier Inc</pub><pmid>25720367</pmid><doi>10.1016/j.jpeds.2015.01.038</doi></addata></record>
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subjects Aorta, Thoracic - abnormalities
Child
Child, Preschool
Echocardiography
Esophageal Atresia - complications
Esophagus - abnormalities
Female
Humans
Male
Pediatrics
Predictive Value of Tests
Reproducibility of Results
Retrospective Studies
Sensitivity and Specificity
Subclavian Artery - pathology
Tracheoesophageal Fistula - complications
Vascular Malformations - complications
title Vascular Anomalies Associated with Esophageal Atresia and Tracheoesophageal Fistula
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