Prenatal Recognition of Hepatopulmonary Fusion in Right-Sided Congenital Diaphragmatic Hernia for Successful Operative Planning
Introduction: Hepatopulmonary fusion (HPF) is a rare anomaly specifically associated with right-sided congenital diaphragmatic hernia (CDH). Fewer than 50 cases of HPF have been reported, most at the time of surgery or postmortem with an associated high morbidity and mortality rate. Prenatal diagnos...
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Veröffentlicht in: | Fetal diagnosis and therapy 2023-02, Vol.49 (11-12), p.451-458 |
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creator | Terp, Kristy L. Roberts, Bailey K. Alonso, Dailen Pevsner Crum, Rachel M. Crombleholme, Timothy Karakas, S. Pinar Alkhoury, Fuad |
description | Introduction: Hepatopulmonary fusion (HPF) is a rare anomaly specifically associated with right-sided congenital diaphragmatic hernia (CDH). Fewer than 50 cases of HPF have been reported, most at the time of surgery or postmortem with an associated high morbidity and mortality rate. Prenatal diagnosis and optimal management of these rare cases have not been established. Case Presentation: We present a case of HPF diagnosed prenatally by fetal ultrasound and magnetic resonance imaging allowing postnatal and intraoperative planning leading to successful HPF separation and repair of the CDH. Discussion: The prenatal recognition allowed the use of preoperative computed tomography angiogram and right cardiac catheterization to plan the surgical approach. Intraoperative ultrasound confirmed abnormal vasculature associated with the HPF facilitating a successful complete HPF separation and CDH repair was performed. The patient survived and continues to do well on long-term follow-up. Prenatal recognition may help reduce the high morbidity and mortality associated with HPF. |
doi_str_mv | 10.1159/000527802 |
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Pinar ; Alkhoury, Fuad</creator><creatorcontrib>Terp, Kristy L. ; Roberts, Bailey K. ; Alonso, Dailen ; Pevsner Crum, Rachel M. ; Crombleholme, Timothy ; Karakas, S. Pinar ; Alkhoury, Fuad</creatorcontrib><description>Introduction: Hepatopulmonary fusion (HPF) is a rare anomaly specifically associated with right-sided congenital diaphragmatic hernia (CDH). Fewer than 50 cases of HPF have been reported, most at the time of surgery or postmortem with an associated high morbidity and mortality rate. Prenatal diagnosis and optimal management of these rare cases have not been established. Case Presentation: We present a case of HPF diagnosed prenatally by fetal ultrasound and magnetic resonance imaging allowing postnatal and intraoperative planning leading to successful HPF separation and repair of the CDH. Discussion: The prenatal recognition allowed the use of preoperative computed tomography angiogram and right cardiac catheterization to plan the surgical approach. Intraoperative ultrasound confirmed abnormal vasculature associated with the HPF facilitating a successful complete HPF separation and CDH repair was performed. The patient survived and continues to do well on long-term follow-up. Prenatal recognition may help reduce the high morbidity and mortality associated with HPF.</description><identifier>ISSN: 1015-3837</identifier><identifier>EISSN: 1421-9964</identifier><identifier>DOI: 10.1159/000527802</identifier><identifier>PMID: 36623503</identifier><language>eng</language><publisher>Basel, Switzerland: S. Karger AG</publisher><subject>Birth defects ; Causes of ; Diagnosis ; Diaphragm ; Hernia ; Methods ; Pediatric research ; Prenatal Diagnosis</subject><ispartof>Fetal diagnosis and therapy, 2023-02, Vol.49 (11-12), p.451-458</ispartof><rights>2023 S. Karger AG, Basel</rights><rights>2023 S. Karger AG, Basel.</rights><rights>COPYRIGHT 2023 S. Karger AG</rights><lds50>peer_reviewed</lds50><woscitedreferencessubscribed>false</woscitedreferencessubscribed><orcidid>0000-0002-5055-9262 ; 0000-0003-4339-6829</orcidid></display><links><openurl>$$Topenurl_article</openurl><openurlfulltext>$$Topenurlfull_article</openurlfulltext><thumbnail>$$Tsyndetics_thumb_exl</thumbnail><link.rule.ids>314,776,780,2423,27901,27902</link.rule.ids><backlink>$$Uhttps://www.ncbi.nlm.nih.gov/pubmed/36623503$$D View this record in MEDLINE/PubMed$$Hfree_for_read</backlink></links><search><creatorcontrib>Terp, Kristy L.</creatorcontrib><creatorcontrib>Roberts, Bailey K.</creatorcontrib><creatorcontrib>Alonso, Dailen</creatorcontrib><creatorcontrib>Pevsner Crum, Rachel M.</creatorcontrib><creatorcontrib>Crombleholme, Timothy</creatorcontrib><creatorcontrib>Karakas, S. Pinar</creatorcontrib><creatorcontrib>Alkhoury, Fuad</creatorcontrib><title>Prenatal Recognition of Hepatopulmonary Fusion in Right-Sided Congenital Diaphragmatic Hernia for Successful Operative Planning</title><title>Fetal diagnosis and therapy</title><addtitle>Fetal Diagn Ther</addtitle><description>Introduction: Hepatopulmonary fusion (HPF) is a rare anomaly specifically associated with right-sided congenital diaphragmatic hernia (CDH). Fewer than 50 cases of HPF have been reported, most at the time of surgery or postmortem with an associated high morbidity and mortality rate. Prenatal diagnosis and optimal management of these rare cases have not been established. Case Presentation: We present a case of HPF diagnosed prenatally by fetal ultrasound and magnetic resonance imaging allowing postnatal and intraoperative planning leading to successful HPF separation and repair of the CDH. Discussion: The prenatal recognition allowed the use of preoperative computed tomography angiogram and right cardiac catheterization to plan the surgical approach. Intraoperative ultrasound confirmed abnormal vasculature associated with the HPF facilitating a successful complete HPF separation and CDH repair was performed. The patient survived and continues to do well on long-term follow-up. Prenatal recognition may help reduce the high morbidity and mortality associated with HPF.</description><subject>Birth defects</subject><subject>Causes of</subject><subject>Diagnosis</subject><subject>Diaphragm</subject><subject>Hernia</subject><subject>Methods</subject><subject>Pediatric research</subject><subject>Prenatal Diagnosis</subject><issn>1015-3837</issn><issn>1421-9964</issn><fulltext>true</fulltext><rsrctype>article</rsrctype><creationdate>2023</creationdate><recordtype>article</recordtype><recordid>eNpt0d9r1TAUB_AgipvTB99FAgNhD9X8aJv2cdx5N2Gwsc3ncpqe9EbbpCTtwKf96-ZSd1HwKQfO55uH8yXkPWefOS_qL4yxQqiKiRfkmOeCZ3Vd5i_TzHiRyUqqI_Imxh-JVUqWr8mRLEshCyaPydNtQAczDPQOte-dna131Bt6hRPMflqG0TsIv-h2ifuNdfTO9rs5u7cddnTjXY8plPIXFqZdgH6E2eoUD84CNT7Q-0VrjNEsA72ZMKT1I9LbAZyzrn9LXhkYIr77856Q79uvD5ur7Prm8tvm_DrTUrA5KzRXZZcbVdQMsEbeCkAQmGusAdsOWixlnueq07lCYAbavFJVYbRKSoM8Iafrvz0M2Fhn_BxAjzbq5lzJWghRFzKpT3-pHcIw76Iflv1R4r_wbIU6-BgDmmYKdkx3ajhr9p00h06S_bjaaWlH7A7yuYQEPqzgJ4QewwEc8qf_XW8vHlbRTJ2RvwHkwp1O</recordid><startdate>20230201</startdate><enddate>20230201</enddate><creator>Terp, Kristy L.</creator><creator>Roberts, Bailey K.</creator><creator>Alonso, Dailen</creator><creator>Pevsner Crum, Rachel M.</creator><creator>Crombleholme, Timothy</creator><creator>Karakas, S. 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Case Presentation: We present a case of HPF diagnosed prenatally by fetal ultrasound and magnetic resonance imaging allowing postnatal and intraoperative planning leading to successful HPF separation and repair of the CDH. Discussion: The prenatal recognition allowed the use of preoperative computed tomography angiogram and right cardiac catheterization to plan the surgical approach. Intraoperative ultrasound confirmed abnormal vasculature associated with the HPF facilitating a successful complete HPF separation and CDH repair was performed. The patient survived and continues to do well on long-term follow-up. Prenatal recognition may help reduce the high morbidity and mortality associated with HPF.</abstract><cop>Basel, Switzerland</cop><pub>S. Karger AG</pub><pmid>36623503</pmid><doi>10.1159/000527802</doi><tpages>8</tpages><orcidid>https://orcid.org/0000-0002-5055-9262</orcidid><orcidid>https://orcid.org/0000-0003-4339-6829</orcidid></addata></record> |
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source | Karger Journal Archive Collection; Karger Journals; Alma/SFX Local Collection |
subjects | Birth defects Causes of Diagnosis Diaphragm Hernia Methods Pediatric research Prenatal Diagnosis |
title | Prenatal Recognition of Hepatopulmonary Fusion in Right-Sided Congenital Diaphragmatic Hernia for Successful Operative Planning |
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