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1. Contemporary Outcomes in Tetralogy of Fallot With Absent Pulmonary Valve After Fetal Diagnosis

Author

Chelliah, Anjali, Moon‐Grady, Anita J, Peyvandi, Shabnam, Chiu, Joanne S, Bost, James E, Schidlow, David, Carroll, Sheila J, Davey, Brooke, Divanovic, Allison, Hornberger, Lisa, Howley, Lisa W, Kavanaugh‐McHugh, Ann, Kovalchin, John P, Levasseur, Stephanie M, Lindblade, Christopher L, Morris, Shaine A, Ngwezi, Deliwe, Pruetz, Jay D, Puchalski, Michael D, Rychik, Jack, Samai, Cyrus, Tacy, Theresa A, Tworetzky, Wayne, Vernon, Margaret M, Yeh, Jay, and Donofrio, Mary T

Subjects
Infant Mortality, Pediatric, Lung, Genetics, Clinical Research, Perinatal Period - Conditions Originating in Perinatal Period, Reproductive health and childbirth, Good Health and Well Being, Canada, Echocardiography, Doppler, Color, Fetal Death, Fetal Heart, Humans, Predictive Value of Tests, Prognosis, Pulmonary Valve, Retrospective Studies, Risk Assessment, Risk Factors, Tetralogy of Fallot, Ultrasonography, Prenatal, United States, Ventricular Dysfunction, Left, Ventricular Dysfunction, Right, congenital heart disease, fetal cardiology, fetal echocardiography, prenatal diagnosis, tetralogy of Fallot with absent pulmonary valve, Cardiorespiratory Medicine and Haematology
Abstract

Background Tetralogy of Fallot with absent pulmonary valve is associated with high mortality, but it remains difficult to predict outcomes prenatally. We aimed to identify risk factors for mortality in a large multicenter cohort. Methods and Results Fetal echocardiograms and clinical data from 19 centers over a 10-year period were collected. Primary outcome measures included fetal demise and overall mortality. Of 100 fetuses, pregnancy termination/postnatal nonintervention was elected in 22. Of 78 with intention to treat, 7 (9%) died in utero and 21 (27%) died postnatally. With median follow-up of 32.9 months, no deaths occurred after 13 months. Of 80 fetuses with genetic testing, 46% had chromosomal abnormalities, with 22q11.2 deletion in 35%. On last fetal echocardiogram, at a median of 34.6 weeks, left ventricular dysfunction independently predicted fetal demise (odds ratio [OR], 7.4; 95% CI 1.3, 43.0; P=0.026). Right ventricular dysfunction independently predicted overall mortality in multivariate analysis (OR, 7.9; 95% CI 2.1-30.0; P=0.002). Earlier gestational age at delivery, mediastinal shift, left ventricular/right ventricular dilation, left ventricular dysfunction, tricuspid regurgitation, and Doppler abnormalities were associated with fetal and postnatal mortality, although few tended to progress throughout gestation on serial evaluation. Pulmonary artery diameters did not correlate with outcomes. Conclusions Perinatal mortality in tetralogy of Fallot with absent pulmonary valve remains high, with overall survival of 64% in fetuses with intention to treat. Right ventricular dysfunction independently predicts overall mortality. Left ventricular dysfunction predicts fetal mortality and may influence prenatal management and delivery planning. Mediastinal shift may reflect secondary effects of airway obstruction and abnormal lung development and is associated with increased mortality.

Published
2021

2. Risk Factors for Mortality and Circulatory Outcome Among Neonates Prenatally Diagnosed With Ebstein Anomaly or Tricuspid Valve Dysplasia: A Multicenter Study

Author

Freud, Lindsay R, McElhinney, Doff B, Kalish, Brian T, Escobar‐Diaz, Maria C, Komarlu, Rukmini, Puchalski, Michael D, Jaeggi, Edgar T, Szwast, Anita L, Freire, Grace, Levasseur, Stéphanie M, Kavanaugh‐McHugh, Ann, Michelfelder, Erik C, Moon‐Grady, Anita J, Donofrio, Mary T, Howley, Lisa W, Tierney, Elif Seda Selamet, Cuneo, Bettina F, Morris, Shaine A, Pruetz, Jay D, van der Velde, Mary E, Kovalchin, John P, Ikemba, Catherine M, Vernon, Margaret M, Samai, Cyrus, Satou, Gary M, Gotteiner, Nina L, Phoon, Colin K, Silverman, Norman H, and Tworetzky, Wayne

Subjects
Pediatric, Perinatal Period - Conditions Originating in Perinatal Period, Lung, Cardiovascular, Heart Disease, Infant Mortality, Clinical Research, Good Health and Well Being, Blood Flow Velocity, Ebstein Anomaly, Echocardiography, Female, Heart Valve Diseases, Hospital Mortality, Humans, Infant, Newborn, Logistic Models, Male, Perinatal Mortality, Prenatal Diagnosis, Retrospective Studies, Risk Factors, Tricuspid Valve, congenital heart disease, Ebstein anomaly, mortality, neonate, outcome, palliation, right ventricle, tricuspid regurgitation, Cardiorespiratory Medicine and Haematology
Abstract

Background In a recent multicenter study of perinatal outcome in fetuses with Ebstein anomaly or tricuspid valve dysplasia, we found that one third of live-born patients died before hospital discharge. We sought to further describe postnatal management strategies and to define risk factors for neonatal mortality and circulatory outcome at discharge. Methods and Results This 23-center, retrospective study from 2005 to 2011 included 243 fetuses with Ebstein anomaly or tricuspid valve dysplasia. Among live-born patients, clinical and echocardiographic factors were evaluated for association with neonatal mortality and palliated versus biventricular circulation at discharge. Of 176 live-born patients, 7 received comfort care, 11 died

Published
2020

3. Outcomes and Predictors of Perinatal Mortality in Fetuses With Ebstein Anomaly or Tricuspid Valve Dysplasia in the Current Era

Author

Freud, Lindsay R, Escobar-Diaz, Maria C, Kalish, Brian T, Komarlu, Rukmini, Puchalski, Michael D, Jaeggi, Edgar T, Szwast, Anita L, Freire, Grace, Levasseur, Stéphanie M, Kavanaugh-McHugh, Ann, Michelfelder, Erik C, Moon-Grady, Anita J, Donofrio, Mary T, Howley, Lisa W, Tierney, Elif Seda Selamet, Cuneo, Bettina F, Morris, Shaine A, Pruetz, Jay D, van der Velde, Mary E, Kovalchin, John P, Ikemba, Catherine M, Vernon, Margaret M, Samai, Cyrus, Satou, Gary M, Gotteiner, Nina L, Phoon, Colin K, Silverman, Norman H, McElhinney, Doff B, and Tworetzky, Wayne

Subjects
Paediatrics, Reproductive Medicine, Biomedical and Clinical Sciences, Cardiovascular Medicine and Haematology, Perinatal Period - Conditions Originating in Perinatal Period, Pediatric, Infant Mortality, Lung, Clinical Research, Preterm, Low Birth Weight and Health of the Newborn, Congenital Structural Anomalies, Reproductive health and childbirth, Good Health and Well Being, Abortion, Eugenic, Adult, Birth Weight, Cardiac Catheterization, Cardiac Surgical Procedures, Down Syndrome, Ebstein Anomaly, Female, Gestational Age, Heart Defects, Congenital, Hospital Mortality, Humans, Infant, Newborn, Infant, Premature, Infant, Premature, Diseases, Male, Palliative Care, Pericardial Effusion, Pregnancy, Pregnancy Outcome, Retrospective Studies, Risk Factors, Treatment Outcome, Tricuspid Valve, Tricuspid Valve Insufficiency, Ultrasonography, Prenatal, Young Adult, Ebstein anomaly, echocardiography, heart defects, congenital, mortality, tricuspid valve insufficiency, heart defects, congenital, Cardiorespiratory Medicine and Haematology, Clinical Sciences, Public Health and Health Services, Cardiovascular System & Hematology, Cardiovascular medicine and haematology, Clinical sciences, Sports science and exercise
Abstract

BackgroundEbstein anomaly and tricuspid valve dysplasia are rare congenital tricuspid valve malformations associated with high perinatal mortality. The literature consists of small, single-center case series spanning several decades. We performed a multicenter study to assess the outcomes and factors associated with mortality after fetal diagnosis in the current era.Methods and resultsFetuses diagnosed with Ebstein anomaly and tricuspid valve dysplasia from 2005 to 2011 were included from 23 centers. The primary outcome was perinatal mortality, defined as fetal demise or death before neonatal discharge. Of 243 fetuses diagnosed at a mean gestational age of 27±6 weeks, there were 11 lost to follow-up (5%), 15 terminations (6%), and 41 demises (17%). In the live-born cohort of 176 live-born patients, 56 (32%) died before discharge, yielding an overall perinatal mortality of 45%. Independent predictors of mortality at the time of diagnosis were gestational age

Published
2015

5. Contributors

Author

Abman, Steven H., primary, Allegaert, Karel, additional, Arya, Bhawna, additional, Askenazi, David, additional, Azhibekov, Timur, additional, Back, Stephen A., additional, Baldwin, H. Scott, additional, Ballard, Roberta A., additional, Bancalari, Eduardo, additional, Bates, Carlton M., additional, Batra, Maneesh, additional, Bayart, Cheryl B., additional, Bellus, Gary A., additional, Benedetti, Thomas J., additional, Benjamin, John T., additional, Bennett, James T., additional, Berry, Gerard T., additional, Binenbaum, Gil, additional, Boos, Markus D., additional, Bouchard, Maryse, additional, Brandling-Bennett, Heather A., additional, Broughton, Darcy E., additional, Brown, Zane, additional, Campbell, Katherine H., additional, Carmichael, Suzan L., additional, Carter, Brian S., additional, Cederbaum, Stephen, additional, Chabra, Shilpi, additional, Chang, Justine, additional, Cheng, Edith Y., additional, Chisholm, Karen M., additional, Christensen, Robert D., additional, Chun, Terrence, additional, Claure, Nelson, additional, Clyman, Ronald I., additional, Colaizy, Tarah T., additional, Cortezzo, DonnaMaria E., additional, Cotten, C. Michael, additional, Cunningham, Michael L., additional, de Alba Campomanes, Alejandra G., additional, Dees, Ellen, additional, DeMauro, Sara B., additional, Denne, Scott C., additional, Deschmann, Emöke, additional, Cecilia, Carolina, additional, DiBlasi, Robert M., additional, Dimmitt, Reed A., additional, DiVall, Sara A., additional, Djahangirian, Orchid, additional, Doherty, Dan, additional, Eichenwald, Eric C., additional, Engen, Rachel, additional, Engmann, Cyril, additional, Evans, Jacquelyn R., additional, Evans, Kelly N., additional, Farmer, Diana L., additional, Fechner, Patricia Y., additional, Ferrieri, Patricia, additional, Finer, Neil N., additional, Fleishman, Rachel A., additional, Fleiss, Bobbi, additional, Flynn, Joseph T., additional, Flynn-O'Brien, Katherine T., additional, Frey, Mark R., additional, Furman, Lydia, additional, Gallagher, Renata C., additional, Gauda, Estelle B., additional, Gleason, Christine A., additional, Goldberg, Michael J., additional, Goldin, Adam B., additional, Gospe, Sidney M., additional, Gressens, Pierre, additional, Gupta, Deepti, additional, Guttentag, Susan H., additional, Haldeman-Englert, Chad R., additional, Hansen, Thomas N., additional, Hing, Anne V., additional, Hingorani, Sangeeta, additional, Hintz, Susan R., additional, Hirose, Shinjiro, additional, Hodson, W. Alan, additional, Hoppe, Kara K., additional, Hostetter, Margaret K., additional, Huang, Benjamin, additional, Huang, Sarah Bauer, additional, Inder, Terrie E., additional, Inoita, Cristian, additional, Jackson, J. Craig, additional, Jain, Deepak, additional, Jain, Lucky, additional, Javid, Patrick J., additional, Josephson, Cassandra D., additional, Jungheim, Emily S., additional, Juul, Sandra E., additional, Katheria, Anup, additional, Keller, Benjamin A., additional, Keller, Roberta L., additional, Kelly, Thomas F., additional, Khorsand, Kate, additional, Kim, Grace, additional, Kinsella, John P., additional, Koves, Ildiko H., additional, Lam, Christina, additional, Lane, Erin R., additional, Lantos, John D., additional, Ledbetter, Daniel J., additional, Lee, Ben, additional, Levy, Harvey L., additional, Levy, Ofer, additional, Lewin, Mark B., additional, Lewis, David B., additional, Lin, P. Ling, additional, Lin, Tiffany Fangtse, additional, Lorch, Scott A., additional, Maheshwari, Akhil, additional, Maltepe, Emin, additional, Marsh, Ketzela J., additional, Martin, Richard J., additional, Mayock, Dennis E., additional, McAdams, Ryan Michael, additional, McAleer, Irene, additional, McElroy, Steven J., additional, McNelis, Kera M., additional, McQuillen, Patrick, additional, Meadow, William L., additional, Merguerian, Paul A., additional, Merjaneh, Lina, additional, Merritt, J. Lawrence, additional, Mezger, Valerie, additional, Michaels, Marian G., additional, Miller, Steven P., additional, Mohan, Sowmya S., additional, Mollen, Thomas J., additional, Moore, Thomas R., additional, Murray, Jeffrey C., additional, Murray, Karen F., additional, Nandi-Munshi, Debika, additional, Natarajan, Niranjana, additional, Neil, Jeffrey J., additional, Ness, Kathryn D., additional, Neu, Josef, additional, Siu-Ying, Angel, additional, Noori, Shahab, additional, O'Mahony, Lila, additional, Palma, Jonathan P., additional, Paneth, Nigel, additional, Parker, Thomas A., additional, Patel, Ravi Mangal, additional, Penn, Anna A., additional, Pettker, Christian M., additional, Peyvandi, Shabnam, additional, Pihoker, Cate, additional, Plosa, Erin, additional, Poindexter, Brenda B., additional, Posencheg, Michael A., additional, Reinking, Benjamin E., additional, Rice-Townsend, Samuel, additional, Richards, Morgan K., additional, Richardson, C. Peter, additional, Richardson, Kelsey, additional, Riggle, Kevin M., additional, Robbins, Elizabeth, additional, Rollins, Mark D., additional, Rosen, Mark A., additional, Rowe, Courtney K., additional, Sahai, Inderneel, additional, Saitta, Sulagna C., additional, Salehi, Parisa, additional, Sanchez, Pablo, additional, Saxonhouse, Matthew A., additional, Schanler, Richard J., additional, Schleiss, Mark R., additional, Scholz, Thomas, additional, Schwaderer, Andrew L., additional, Selewski, David, additional, Sellers, Zachary M., additional, Seri, Istvan, additional, Shnorhavorian, Margarett, additional, Sibley, Eric, additional, Sidbury, Robert, additional, Simmons, Rebecca, additional, Smith, Caitlin, additional, Sola-Visner, Martha C., additional, Srinivasan, Lakshmi, additional, Steinhorn, Robin H., additional, Stevenson, David K., additional, Stolp, Helen, additional, Taplin, Craig, additional, Tarczy-Hornoch, Peter, additional, Taylor, James A., additional, Thomas, Janet A., additional, Thompson, Tracy, additional, Tiller, George E., additional, Torres, Benjamin A., additional, Traudt, Christopher Michael, additional, van den Anker, John N., additional, Vernon, Margaret M., additional, Vohr, Betty, additional, Walker, Valencia P., additional, Wallen, Linda D., additional, Wallenstein, Matthew B., additional, Wang, Peter (Zhan Tao), additional, Warady, Bradley A., additional, Ward, Robert M., additional, Watchko, Jon F., additional, Wehbi, Elias, additional, Weitkamp, Joern-Hendrik, additional, Werny, David, additional, White, Klane K., additional, Willig, Laurel, additional, Woodrum, David, additional, Woodward, George A., additional, Wright, Clyde J., additional, Wright, Jeffrey A., additional, Yonekawa, Karyn, additional, and Zackai, Elaine H., additional

Published
2018
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10. Contributors

Author

Cawley, Peter J., primary, Choueiter, Nadine F., additional, Choy, Raylene M., additional, Conwell, Jeffrey A., additional, Hattendorf, Brandy, additional, Joffe, Denise, additional, Johnston, Troy, additional, Kemna, Mariska, additional, Lester, Joel, additional, Lewin, Mark B., additional, Likes, Maggie L., additional, Owens, David S., additional, Schultz, Amy H., additional, Soriano, Brian D., additional, Stout, Karen, additional, and Vernon, Margaret M., additional

Published
2012
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11. Contributors

Author

Abman, Steven H., primary, Ahmed, Amina, additional, Allen, Marilee C., additional, Askenazi, David, additional, Back, Stephen A., additional, Baldwin, H. Scott, additional, Ballard, Roberta A., additional, Bancalari, Eduardo, additional, Bates, Carlton M., additional, Batisky, Donald L., additional, Baumgart, Stephen, additional, Benedetti, Thomas J., additional, Berry, Gerard T., additional, Bianchi, Diana W., additional, Binenbaum, Gil, additional, Bollepalli, Sureka, additional, Bonifacio, Sonia L., additional, Cairo, Mitchell S., additional, Campbell, Katherine H., additional, Caplan, Michael, additional, Cederbaum, Stephen, additional, Chandra, Sudhish, additional, Chen, Ming, additional, Claure, Nelson, additional, Clyman, Ronald I., additional, Cohen, Bernard A., additional, Cole, F. Sessions, additional, Copelovitch, Lawrence, additional, Cunningham, Michael, additional, de Alba Campomanes, Alejandra G., additional, Dees, Ellen, additional, Denne, Scott C., additional, Devaskar, Sherin U., additional, DiBlasi, Robert M., additional, Dimmitt, Reed A., additional, Eichenwald, Eric C., additional, Eisenstein, Eli M., additional, Evans, Jacquelyn R., additional, Evans, Kelly, additional, Farmer, Diana L., additional, Ferrieri, Patricia, additional, Ferriero, Donna M., additional, Finer, Neil N., additional, Fraga, Maria Victoria, additional, Furman, Lydia, additional, Furth, Susan, additional, Gauda, Estelle B., additional, Glader, Bertil, additional, Gleason, Christine A., additional, Goldberg, Michael J., additional, Gonzalez, Fernando, additional, Gopalani, Sameer, additional, Grant, P. Ellen, additional, Greene, Carol L., additional, Guevara-Gallardo, Salvador, additional, Guignard, Jean-Pierre, additional, Guttentag, Susan, additional, Haldeman-Englert, Chad R., additional, Hansen, Thomas, additional, Hing, Anne V., additional, Hohimer, A. Roger, additional, Hostetter, Margaret K., additional, Hull, Andrew D., additional, Jackson, J. Craig, additional, Jain, Lucky, additional, Jain, Vandana, additional, Janjua, Halima Saadia, additional, Juul, Sandra E., additional, Kalkunte, Satyan, additional, Kaplan, Bernard S., additional, Keller, Roberta L., additional, Kelly, Thomas F., additional, Kern, Steven E., additional, Kerkar, Nanda, additional, Kinsella, John P., additional, Kirsch, Roxanne, additional, Kleinman, Monica E., additional, Klitzner, Thomas S., additional, Lambert, Sarah M., additional, Lantos, John D., additional, Leone, Tina A., additional, Leppert, Mary, additional, Levy, Harvey L., additional, Lewin, Mark, additional, Lin-Su, Karen, additional, L. Loh, Mignon, additional, Lorch, Scott A., additional, Lugo, Ralph A., additional, Mai, Volker, additional, Marino, Bradley S., additional, Markovitz, Barry, additional, Marquard, Kerri, additional, Martin, Camilia R., additional, Martin, Richard J., additional, Matthay, Katherine K., additional, Matthews, Dana C., additional, Mayock, Dennis E., additional, Meadow, William L., additional, Menon, Ram K., additional, Mercuri, Eugenio, additional, Mohan, Sowmya S., additional, Moley, Kelle, additional, Mollen, Thomas J., additional, Moore, Jeremy P., additional, Moore, Thomas R., additional, Munson, David A., additional, Murray, Jeffrey C., additional, Neu, Josef, additional, New, Maria I., additional, Nguyen-Vermillion, Annie, additional, Niklas, Victoria, additional, Nimkarn, Saroj, additional, Padbury, James F., additional, Pane, Marika, additional, Paneth, Nigel, additional, Parker, Thomas A., additional, Patterson, Janna C., additional, Pettker, Christian M., additional, Plawner, Lauren L., additional, Poenaru, Dan, additional, Poindexter, Brenda B., additional, Posencheg, Michael A., additional, Prabhu, Sanjay P., additional, Püttgen, Katherine B., additional, Quinn, Graham E., additional, Raju, Tonse N.K., additional, Ramos, Gladys A., additional, Reinking, Benjamin E., additional, Richardson, C. Peter, additional, Rimoin, David L., additional, Robbins, Elizabeth, additional, Robertson, Richard L., additional, Rollins, Mark D., additional, Rose, Susan R., additional, Rosen, Mark A., additional, Rubin, Lewis P., additional, Sahai, Inderneel, additional, Saitta, Sulagna C., additional, Sánchez, Pablo J., additional, Satou, Gary M., additional, Schanler, Richard J., additional, Scher, Mark S., additional, Schleiss, Mark R., additional, Scholz, Thomas D., additional, Schwaderer, Andrew L., additional, Seri, Istvan, additional, Sharma, Surendra, additional, Shereck, Evan B., additional, Sibley, Eric, additional, Signore, Caroline, additional, Simmons, Rebecca, additional, Smith, Jeffrey B., additional, Smith, Lorie B., additional, Song, Clara, additional, Steinhorn, Robin H., additional, Suchy, Frederick J., additional, Sulyok, Endre, additional, Tarczy-Hornoch, Peter, additional, Taylor, George A., additional, Taylor, James A., additional, Thomas, Janet A., additional, Tiller, George E., additional, Tran, Mark M., additional, Trautman, Michael Stone, additional, Upperman, Jeffrey S., additional, van de Ven, Carmella, additional, Vernon, Margaret M., additional, Allan Walker, W., additional, Wallen, Linda D., additional, Waller, Sarah A., additional, Warady, Bradley A., additional, Ward, Robert M., additional, Watchko, Jon F., additional, Wernovsky, Gil, additional, White, Klane K., additional, Williams, Calvin B., additional, Woodrum, David, additional, Woodward, George A., additional, Wright, Dakara Rucker, additional, Wright, Jeffrey A., additional, Wright, Linda L., additional, Young, Christopher M., additional, Young, Guy, additional, Zackai, Elaine H., additional, and Zderic, Stephen A., additional

Published
2012
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16. Colaboradores

Author

Abman, Steven H., Allegaert, Karel, Arya, Bhawna, Askenazi, David, Azhibekov, Timur, Back, Stephen A., Baldwin, H. Scott, Ballard, Roberta A., Bancalari, Eduardo, Bates, Carlton M., Batra, Maneesh, Bayart, Cheryl B., Bellus, Gary A., Benedetti, Thomas J., Benjamin, John T., Bennett, James T., Berry, Gerard T., Binenbaum, Gil, Boos, Markus D., Bouchard, Maryse, Brandling-Bennett, Heather A., Broughton, Darcy E., Brown, Zane, Campbell, Katherine H., Carmichael, Suzan L., Carter, Brian S., Cederbaum, Stephen, Chabra, Shilpi, Chang, Justine, Cheng, Edith Y., Chisholm, Karen M., Christensen, Robert D., Chun, Terrence, Claure, Nelson, Clyman, Ronald I., Colaizy, Tarah T., Cortezzo, DonnaMaria E., Cotten, C. Michael, Cunningham, Michael L., Campomanes, Alejandra G. de Alba, Dees, Ellen, DeMauro, Sara B., Denne, Scott C., Deschmann, Emöke, DiBlasi, Carolina Cecilia, DiBlasi, Robert M., Dimmitt, Reed A., DiVall, Sara A., Djahangirian, Orchid, Doherty, Dan, Eichenwald, Eric C., Engen, Rachel, Engmann, Cyril, Evans, Jacquelyn R., Evans, Kelly N., Farmer, Diana L., Fechner, Patricia Y., Ferrieri, Patricia, Finer, Neil N., Fleishman, Rachel A., Fleiss, Bobbi, Flynn, Joseph T., Flynn-O’Brien, Katherine T., Frey, Mark R., Furman, Lydia, Gallagher, Renata C., Gauda, Estelle B., Gleason, Christine A., Goldberg, Michael J., Goldin, Adam B., Gospe, Sidney M., Jr., Gressens, Pierre, Gupta, Deepti, Guttentag, Susan H., Haldeman-Englert, Chad R., Hansen, Thomas N., Hing, Anne V., Hingorani, Sangeeta, Hintz, Susan R., Hirose, Shinjiro, Hodson, W. Alan, Hoppe, Kara K., Hostetter, Margaret K., Huang, Benjamin, Huang, Sarah Bauer, Inder, Terrie E., Inoita, Cristian, Jackson, J. Craig, Jain, Deepak, Jain, Lucky, Javid, Patrick J., Josephson, Cassandra D., Jungheim, Emily S., Juul, Sandra E., Katheria, Anup, Keller, Benjamin A., Keller, Roberta L., Kelly, Thomas F., Khorsand, Kate, Kim, Grace, Kinsella, John P., Koves, Ildiko H., Lam, Christina, Lane, Erin R., Lantos, John D., Ledbetter, Daniel J., Lee, Ben, Levy, Harvey L., Levy, Ofer, Lewin, Mark B., Lewis, David B., Lin, P. Ling, Lin, Tiffany Fangtse, Lorch, Scott A., Maheshwari, Akhil, Maltepe, Emin, Marsh, Ketzela J., Martin, Richard J., Mayock, Dennis E., McAdams, Ryan Michael, McAleer, Irene, McElroy, Steven J., McNelis, Kera M., McQuillen, Patrick, Meadow, William L., Merguerian, Paul A., Merjaneh, Lina, Merritt, J. Lawrence, II, Mezger, Valerie, Michaels, Marian G., Miller, Steven P., Mohan, Sowmya S., Mollen, Thomas J., Moore, Thomas R., Murray, Jeffrey C., Murray, Karen F., Nandi-Munshi, Debika, Natarajan, Niranjana, Neil, Jeffrey J., Ness, Kathryn D., Neu, Josef, Nip, Angel Siu-Ying, Noori, Shahab, O’Mahony, Lila, Palma, Jonathan P., Paneth, Nigel, Parker, Thomas A., Patel, Ravi Mangal, Penn, Anna A., Pettker, Christian M., Peyvandi, Shabnam, Pihoker, Cate, Plosa, Erin, Poindexter, Brenda B., Posencheg, Michael A., Reinking, Benjamin E., Rice-Townsend, Samuel, Richards, Morgan K., Richardson, C. Peter, Richardson, Kelsey, Riggle, Kevin M., Robbins, Elizabeth, Rollins, Mark D., Rosen, Mark A., Rowe, Courtney K., Sahai, Inderneel, Saitta, Sulagna C., Salehi, Parisa, Sanchez, Pablo, Saxonhouse, Matthew A., Schanler, Richard J., Schleiss, Mark R., Scholz, Thomas, Schwaderer, Andrew L., Selewski, David, Sellers, Zachary M., Seri, Istvan, Shnorhavorian, Margarett, Sibley, Eric, Sidbury, Robert, Simmons, Rebecca, Smith, Caitlin, Sola-Visner, Martha C., Srinivasan, Lakshmi, Steinhorn, Robin H., Stevenson, David K., Stolp, Helen, Taplin, Craig, Tarczy-Hornoch, Peter, Taylor, James A., Thomas, Janet A., Thompson, Tracy, Tiller, George E., Torres, Benjamin A., Traudt, Christopher Michael, van den Anker, John N., Vernon, Margaret M., Vohr, Betty, Walker, Valencia P., Wallen, Linda D., Wallenstein, Matthew B., Wang, Peter (Zhan Tao), Warady, Bradley A., Ward, Robert M., Watchko, Jon F., Wehbi, Elias, Weitkamp, Joern-Hendrik, Werny, David, White, Klane K., Willig, Laurel, Woodrum, David, Woodward, George A., Wright, Clyde J., Wright, Jeffrey A., Yonekawa, Karyn, and Zackai, Elaine H.

Published
2019
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19. Outcomes and Predictors of Perinatal Mortality in Fetuses With Ebstein Anomaly or Tricuspid Valve Dysplasia in the Current Era: A Multicenter Study.

Author

Freud, Lindsay R, Freud, Lindsay R, Escobar-Diaz, Maria C, Kalish, Brian T, Komarlu, Rukmini, Puchalski, Michael D, Jaeggi, Edgar T, Szwast, Anita L, Freire, Grace, Levasseur, Stéphanie M, Kavanaugh-McHugh, Ann, Michelfelder, Erik C, Moon-Grady, Anita J, Donofrio, Mary T, Howley, Lisa W, Tierney, Elif Seda Selamet, Cuneo, Bettina F, Morris, Shaine A, Pruetz, Jay D, van der Velde, Mary E, Kovalchin, John P, Ikemba, Catherine M, Vernon, Margaret M, Samai, Cyrus, Satou, Gary M, Gotteiner, Nina L, Phoon, Colin K, Silverman, Norman H, McElhinney, Doff B, Tworetzky, Wayne, Freud, Lindsay R, Freud, Lindsay R, Escobar-Diaz, Maria C, Kalish, Brian T, Komarlu, Rukmini, Puchalski, Michael D, Jaeggi, Edgar T, Szwast, Anita L, Freire, Grace, Levasseur, Stéphanie M, Kavanaugh-McHugh, Ann, Michelfelder, Erik C, Moon-Grady, Anita J, Donofrio, Mary T, Howley, Lisa W, Tierney, Elif Seda Selamet, Cuneo, Bettina F, Morris, Shaine A, Pruetz, Jay D, van der Velde, Mary E, Kovalchin, John P, Ikemba, Catherine M, Vernon, Margaret M, Samai, Cyrus, Satou, Gary M, Gotteiner, Nina L, Phoon, Colin K, Silverman, Norman H, McElhinney, Doff B, and Tworetzky, Wayne

Abstract

BackgroundEbstein anomaly and tricuspid valve dysplasia are rare congenital tricuspid valve malformations associated with high perinatal mortality. The literature consists of small, single-center case series spanning several decades. We performed a multicenter study to assess the outcomes and factors associated with mortality after fetal diagnosis in the current era.Methods and resultsFetuses diagnosed with Ebstein anomaly and tricuspid valve dysplasia from 2005 to 2011 were included from 23 centers. The primary outcome was perinatal mortality, defined as fetal demise or death before neonatal discharge. Of 243 fetuses diagnosed at a mean gestational age of 27±6 weeks, there were 11 lost to follow-up (5%), 15 terminations (6%), and 41 demises (17%). In the live-born cohort of 176 live-born patients, 56 (32%) died before discharge, yielding an overall perinatal mortality of 45%. Independent predictors of mortality at the time of diagnosis were gestational age <32 weeks (odds ratio, 8.6; 95% confidence interval, 3.5-21.0; P<0.001), tricuspid valve annulus diameter z-score (odds ratio, 1.3; 95% confidence interval, 1.1-1.5; P<0.001), pulmonary regurgitation (odds ratio, 2.9; 95% confidence interval, 1.4-6.2; P<0.001), and a pericardial effusion (odds ratio, 2.5; 95% confidence interval, 1.1-6.0; P=0.04). Nonsurvivors were more likely to have pulmonary regurgitation at any gestational age (61% versus 34%; P<0.001), and lower gestational age and weight at birth (35 versus 37 weeks; 2.5 versus 3.0 kg; both P<0.001).ConclusionIn this large, contemporary series of fetuses with Ebstein anomaly and tricuspid valve dysplasia, perinatal mortality remained high. Fetuses with pulmonary regurgitation, indicating circular shunt physiology, are a high-risk cohort and may benefit from more innovative therapeutic approaches to improve survival.

Published
2015

20. Abstract 17203: Postnatal Management of Fetuses With Ebstein Anomaly or Tricuspid Valve Dysplasia in the Current Era: A Multi-center Study

Author

Freud, Lindsay R, primary, Kalish, Brian T, additional, Escobar-Diaz, Maria C, additional, Komarlu, Rukmini, additional, Puchalski, Michael D, additional, Jaeggi, Edgar T, additional, Szwast, Anita L, additional, Freire, Grace, additional, Levasseur, Stephanie M, additional, Kavanaugh-McHugh, Ann, additional, Michelfelder, Erik C, additional, Moon-Grady, Anita J, additional, Donofrio, Mary T, additional, Howley, Lisa W, additional, Selamet Tierney, Elif Seda, additional, Cuneo, Bettina F, additional, Morris, Shaine A, additional, Pruetz, Jay D, additional, van der Velde, Mary E, additional, Kovalchin, John P, additional, Ikemba, Catherine M, additional, Vernon, Margaret M, additional, Samai, Cyrus, additional, Satou, Gary M, additional, Gotteiner, Nina L, additional, Phoon, Colin K, additional, Silverman, Norman H, additional, McElhinney, Doff B, additional, and Tworetzky, Wayne, additional

Published
2015
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24. Contributors

Author

Cawley, Peter J., Choueiter, Nadine F., Choy, Raylene M., Conwell, Jeffrey A., Hattendorf, Brandy, Joffe, Denise, Johnston, Troy, Kemna, Mariska, Lester, Joel, Lewin, Mark B., Likes, Maggie L., Owens, David S., Schultz, Amy H., Soriano, Brian D., Stout, Karen, and Vernon, Margaret M.

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