International Fetal Cardiac Intervention Registry: A Worldwide Collaborative Description and Preliminary Outcomes

Anita J Moon-Grady; Shaine A Morris; Michael Belfort; Ramen Chmait; Joanna Dangel; Roland Devlieger; Stephen Emery; Michele Frommelt; Alberto Galindo; Sarah Gelehrter; Ulrich Gembruch; Sofia Grinenco; Mounira Habli; Ulrike Herberg; Edgar Jaeggi; Mark Kilby; Eftichia Kontopoulos; Pablo Marantz; Owen Miller; Lucas Otaño; Carlos Pedra; Simone Pedra; Jay Pruetz; Ruben Quintero; Greg Ryan; Gurleen Sharland; John Simpson; Emanuel Vlastos; Wayne Tworetzky; Louise Wilkins-Haug; Dick Oepkes; International Fetal Cardiac Intervention Registry

doi:10.1016/j.jacc.2015.05.037

International Fetal Cardiac Intervention Registry: A Worldwide Collaborative Description and Preliminary Outcomes

Anita J Moon-Grady, Shaine A Morris, Michael Belfort, Ramen Chmait, Joanna Dangel, Roland Devlieger, Stephen Emery, Michele Frommelt, Alberto Galindo, Sarah Gelehrter, Ulrich Gembruch, Sofia Grinenco, Mounira Habli, Ulrike Herberg, Edgar Jaeggi, Mark Kilby, Eftichia Kontopoulos, Pablo Marantz, Owen Miller, Lucas OtañoCarlos Pedra, Simone Pedra, Jay Pruetz, Ruben Quintero, Greg Ryan, Gurleen Sharland, John Simpson, Emanuel Vlastos, Wayne Tworetzky, Louise Wilkins-Haug, Dick Oepkes, International Fetal Cardiac Intervention Registry

Metabolism and Systems Science

Research output: Contribution to journal › Article › peer-review

72 Citations (Scopus)

Abstract

BACKGROUND: Invasive fetal cardiac intervention (FCI) has been reported in single-institution series, promoting technical and physiologic success.

OBJECTIVES: This study describes the creation of an international registry of cases presenting for FCI, intended to compile technical and outcome data from a multicenter cohort.

METHODS: For this initial analysis, the entire database of the International Fetal Cardiac Intervention Registry (IFCIR) was queried for details of diagnoses, procedures, and outcomes. Maternal-fetal dyads from January 2001 through June 2014 were included.

RESULTS: Eighteen institutions submitted data by data harvest. Of 370 cases entered, 245 underwent FCI: 100 aortic valvuloplasties from a previous single-center report (excluded from additional reporting here), an additional 86 aortic and 16 pulmonary valvuloplasties, 37 atrial septal cases, and 6 unclassified cases. FCI did not appear to affect overall survival to hospital discharge. Among live-born infants with a fetal diagnosis of aortic stenosis/evolving hypoplastic left heart syndrome, more than twice as many were discharged with biventricular circulation after successful FCI versus those meeting institutional criteria but without any or successful FCI (42.8% vs. 19.4%, respectively). When fetal deaths were counted as treatment failures, the percentages were similar: biventricular circulation at discharge was 31.3% versus 18.5% for those discharged with univentricular palliation. Survival to discharge for live-born fetuses with atrial restriction was similar to that of those undergoing technically successful versus unsuccessful FCI (63.6% vs. 46.7%, respectively), although criteria for diagnosis were nonuniform.

CONCLUSIONS: We describe the contents of the IFCIR and present post-natal data to suggest potential benefit to fetal therapy among pregnancies considered for possible intervention and support proposals for additional work.

Original language	English
Pages (from-to)	388-99
Number of pages	12
Journal	Journal of the American College of Cardiology
Volume	66
Issue number	4
DOIs	https://doi.org/10.1016/j.jacc.2015.05.037
Publication status	Published - 28 Jul 2015

Keywords

Female
Fetal Diseases
Fetal Therapies
Heart Defects, Congenital
Humans
Infant, Newborn
International Cooperation
Pregnancy
Pregnancy Outcome
Registries

Access to Document

10.1016/j.jacc.2015.05.037

Cite this

Moon-Grady, A. J., Morris, S. A., Belfort, M., Chmait, R., Dangel, J., Devlieger, R., Emery, S., Frommelt, M., Galindo, A., Gelehrter, S., Gembruch, U., Grinenco, S., Habli, M., Herberg, U., Jaeggi, E., Kilby, M., Kontopoulos, E., Marantz, P., Miller, O., ... International Fetal Cardiac Intervention Registry (2015). International Fetal Cardiac Intervention Registry: A Worldwide Collaborative Description and Preliminary Outcomes. Journal of the American College of Cardiology, 66(4), 388-99. https://doi.org/10.1016/j.jacc.2015.05.037

@article{edf65bf7cb414b0d91eef057a5c2d76e,

title = "International Fetal Cardiac Intervention Registry: A Worldwide Collaborative Description and Preliminary Outcomes",

abstract = "BACKGROUND: Invasive fetal cardiac intervention (FCI) has been reported in single-institution series, promoting technical and physiologic success.OBJECTIVES: This study describes the creation of an international registry of cases presenting for FCI, intended to compile technical and outcome data from a multicenter cohort.METHODS: For this initial analysis, the entire database of the International Fetal Cardiac Intervention Registry (IFCIR) was queried for details of diagnoses, procedures, and outcomes. Maternal-fetal dyads from January 2001 through June 2014 were included.RESULTS: Eighteen institutions submitted data by data harvest. Of 370 cases entered, 245 underwent FCI: 100 aortic valvuloplasties from a previous single-center report (excluded from additional reporting here), an additional 86 aortic and 16 pulmonary valvuloplasties, 37 atrial septal cases, and 6 unclassified cases. FCI did not appear to affect overall survival to hospital discharge. Among live-born infants with a fetal diagnosis of aortic stenosis/evolving hypoplastic left heart syndrome, more than twice as many were discharged with biventricular circulation after successful FCI versus those meeting institutional criteria but without any or successful FCI (42.8% vs. 19.4%, respectively). When fetal deaths were counted as treatment failures, the percentages were similar: biventricular circulation at discharge was 31.3% versus 18.5% for those discharged with univentricular palliation. Survival to discharge for live-born fetuses with atrial restriction was similar to that of those undergoing technically successful versus unsuccessful FCI (63.6% vs. 46.7%, respectively), although criteria for diagnosis were nonuniform.CONCLUSIONS: We describe the contents of the IFCIR and present post-natal data to suggest potential benefit to fetal therapy among pregnancies considered for possible intervention and support proposals for additional work.",

keywords = "Female, Fetal Diseases, Fetal Therapies, Heart Defects, Congenital, Humans, Infant, Newborn, International Cooperation, Pregnancy, Pregnancy Outcome, Registries",

author = "Moon-Grady, {Anita J} and Morris, {Shaine A} and Michael Belfort and Ramen Chmait and Joanna Dangel and Roland Devlieger and Stephen Emery and Michele Frommelt and Alberto Galindo and Sarah Gelehrter and Ulrich Gembruch and Sofia Grinenco and Mounira Habli and Ulrike Herberg and Edgar Jaeggi and Mark Kilby and Eftichia Kontopoulos and Pablo Marantz and Owen Miller and Lucas Ota{\~n}o and Carlos Pedra and Simone Pedra and Jay Pruetz and Ruben Quintero and Greg Ryan and Gurleen Sharland and John Simpson and Emanuel Vlastos and Wayne Tworetzky and Louise Wilkins-Haug and Dick Oepkes and {International Fetal Cardiac Intervention Registry}",

year = "2015",

month = jul,

day = "28",

doi = "10.1016/j.jacc.2015.05.037",

language = "English",

volume = "66",

pages = "388--99",

journal = "Journal of the American College of Cardiology",

issn = "0735-1097",

publisher = "Elsevier",

number = "4",

}

Moon-Grady, AJ, Morris, SA, Belfort, M, Chmait, R, Dangel, J, Devlieger, R, Emery, S, Frommelt, M, Galindo, A, Gelehrter, S, Gembruch, U, Grinenco, S, Habli, M, Herberg, U, Jaeggi, E, Kilby, M, Kontopoulos, E, Marantz, P, Miller, O, Otaño, L, Pedra, C, Pedra, S, Pruetz, J, Quintero, R, Ryan, G, Sharland, G, Simpson, J, Vlastos, E, Tworetzky, W, Wilkins-Haug, L, Oepkes, D & International Fetal Cardiac Intervention Registry 2015, 'International Fetal Cardiac Intervention Registry: A Worldwide Collaborative Description and Preliminary Outcomes', Journal of the American College of Cardiology, vol. 66, no. 4, pp. 388-99. https://doi.org/10.1016/j.jacc.2015.05.037

TY - JOUR

T1 - International Fetal Cardiac Intervention Registry

T2 - A Worldwide Collaborative Description and Preliminary Outcomes

AU - Moon-Grady, Anita J

AU - Morris, Shaine A

AU - Belfort, Michael

AU - Chmait, Ramen

AU - Dangel, Joanna

AU - Devlieger, Roland

AU - Emery, Stephen

AU - Frommelt, Michele

AU - Galindo, Alberto

AU - Gelehrter, Sarah

AU - Gembruch, Ulrich

AU - Grinenco, Sofia

AU - Habli, Mounira

AU - Herberg, Ulrike

AU - Jaeggi, Edgar

AU - Kilby, Mark

AU - Kontopoulos, Eftichia

AU - Marantz, Pablo

AU - Miller, Owen

AU - Otaño, Lucas

AU - Pedra, Carlos

AU - Pedra, Simone

AU - Pruetz, Jay

AU - Quintero, Ruben

AU - Ryan, Greg

AU - Sharland, Gurleen

AU - Simpson, John

AU - Vlastos, Emanuel

AU - Tworetzky, Wayne

AU - Wilkins-Haug, Louise

AU - Oepkes, Dick

AU - International Fetal Cardiac Intervention Registry

PY - 2015/7/28

Y1 - 2015/7/28

N2 - BACKGROUND: Invasive fetal cardiac intervention (FCI) has been reported in single-institution series, promoting technical and physiologic success.OBJECTIVES: This study describes the creation of an international registry of cases presenting for FCI, intended to compile technical and outcome data from a multicenter cohort.METHODS: For this initial analysis, the entire database of the International Fetal Cardiac Intervention Registry (IFCIR) was queried for details of diagnoses, procedures, and outcomes. Maternal-fetal dyads from January 2001 through June 2014 were included.RESULTS: Eighteen institutions submitted data by data harvest. Of 370 cases entered, 245 underwent FCI: 100 aortic valvuloplasties from a previous single-center report (excluded from additional reporting here), an additional 86 aortic and 16 pulmonary valvuloplasties, 37 atrial septal cases, and 6 unclassified cases. FCI did not appear to affect overall survival to hospital discharge. Among live-born infants with a fetal diagnosis of aortic stenosis/evolving hypoplastic left heart syndrome, more than twice as many were discharged with biventricular circulation after successful FCI versus those meeting institutional criteria but without any or successful FCI (42.8% vs. 19.4%, respectively). When fetal deaths were counted as treatment failures, the percentages were similar: biventricular circulation at discharge was 31.3% versus 18.5% for those discharged with univentricular palliation. Survival to discharge for live-born fetuses with atrial restriction was similar to that of those undergoing technically successful versus unsuccessful FCI (63.6% vs. 46.7%, respectively), although criteria for diagnosis were nonuniform.CONCLUSIONS: We describe the contents of the IFCIR and present post-natal data to suggest potential benefit to fetal therapy among pregnancies considered for possible intervention and support proposals for additional work.

AB - BACKGROUND: Invasive fetal cardiac intervention (FCI) has been reported in single-institution series, promoting technical and physiologic success.OBJECTIVES: This study describes the creation of an international registry of cases presenting for FCI, intended to compile technical and outcome data from a multicenter cohort.METHODS: For this initial analysis, the entire database of the International Fetal Cardiac Intervention Registry (IFCIR) was queried for details of diagnoses, procedures, and outcomes. Maternal-fetal dyads from January 2001 through June 2014 were included.RESULTS: Eighteen institutions submitted data by data harvest. Of 370 cases entered, 245 underwent FCI: 100 aortic valvuloplasties from a previous single-center report (excluded from additional reporting here), an additional 86 aortic and 16 pulmonary valvuloplasties, 37 atrial septal cases, and 6 unclassified cases. FCI did not appear to affect overall survival to hospital discharge. Among live-born infants with a fetal diagnosis of aortic stenosis/evolving hypoplastic left heart syndrome, more than twice as many were discharged with biventricular circulation after successful FCI versus those meeting institutional criteria but without any or successful FCI (42.8% vs. 19.4%, respectively). When fetal deaths were counted as treatment failures, the percentages were similar: biventricular circulation at discharge was 31.3% versus 18.5% for those discharged with univentricular palliation. Survival to discharge for live-born fetuses with atrial restriction was similar to that of those undergoing technically successful versus unsuccessful FCI (63.6% vs. 46.7%, respectively), although criteria for diagnosis were nonuniform.CONCLUSIONS: We describe the contents of the IFCIR and present post-natal data to suggest potential benefit to fetal therapy among pregnancies considered for possible intervention and support proposals for additional work.

KW - Female

KW - Fetal Diseases

KW - Fetal Therapies

KW - Heart Defects, Congenital

KW - Humans

KW - Infant, Newborn

KW - International Cooperation

KW - Pregnancy

KW - Pregnancy Outcome

KW - Registries

U2 - 10.1016/j.jacc.2015.05.037

DO - 10.1016/j.jacc.2015.05.037

M3 - Article

C2 - 26205597

SN - 0735-1097

VL - 66

SP - 388

EP - 399

JO - Journal of the American College of Cardiology

JF - Journal of the American College of Cardiology

IS - 4

ER -

International Fetal Cardiac Intervention Registry: A Worldwide Collaborative Description and Preliminary Outcomes

Abstract

Keywords

Access to Document

Fingerprint

Cite this