Evidence map›Paper›PMID 25052401›Full record

Trial reportCirculation2014

Fetal aortic valvuloplasty for evolving hypoplastic left heart syndrome: postnatal outcomes of the first 100 patients.

Lindsay R Freud, Doff B McElhinney, Audrey C Marshall, Gerald R Marx, Kevin G Friedman, Pedro J del Nido, Sitaram M Emani, Terra Lafranchi, Virginia Silva, Louise E Wilkins-Haug and 3 more

Registry-linked trialOpen access · bronzeAbstract readClinical Trial
PubMed Full text at PMC Publisher
In one paragraph

Trial report in Circulation, 2014. The graph could read no effect estimate from its abstract, so it casts no vote on the map. It is linked to trial NCT07542509 (Digital diagnoSis of Cardiac sOUNd in peDiatric Patients [DI-SOUND Study]), which is not on this map. Cited by 55 papers, 2 of them syntheses that pooled it.

0numbers the graph read from it
0cells of the map it votes in
55citing papers in PubMed, 2 pooled it
12.2field-weighted citation impact, top 1% of its field
1 · What the graph read from it

What it found

Each row is one number read from the abstract, on the scale the paper reported it, with its interval. Left of the dashed line favours the treatment, right favours the comparator. Under each row is the sentence it came from. New to these charts? A ten-minute tutorial.

The abstract states no effect estimate the extractor could read, or names no intervention and outcome on the map, so this paper lights no cell and moves no belief. It is still indexed, cited and linked below.

2 · The registry

The trial behind it

Trials whose registry record cites this paper, or whose number appears in the abstract. A trial that started after this paper was published is citing it as background, not reporting it.

NCT07542509 recruitingnot on this mapstarted 2024, after this paper: background citation

Digital diagnoSis of Cardiac sOUNd in peDiatric Patients [DI-SOUND Study]

TypeobservationalSponsorIRCCS Azienda Ospedaliero-Universitaria di BolognaRan2024 to 2027Enrolled1,000ConditionsCardiac Disease, Auscultation of Heart, Machine Learning
3 · Its place in the literature

Who cites it

55 citing papers in PubMed, 2 syntheses or guidelines pooled it, 202 citations in OpenAlex.

  1. Pooled it
  2. Pooled it
  3. Article
  4. Is There a Unified Etiology of Hypoplastic Left Heart Syndrome? Evaluating Genetic, Structural, and Hemodynamic Models of Disease Initiation.Pathophysiology : the official journal of the International Society for Pathophysiology · 2026
    Review
  5. Current practice and challenges in fetal cardiac intervention.Frontiers in cardiovascular medicine · 2026
    Review
  6. Article
  7. Article
  8. Review
  9. Postinterventional fetal aortic regurgitation: prevalence, outcome and effects on fetal circulation in large single-center cohort.Ultrasound in obstetrics & gynecology : the official journal of the International Society of Ultrasound in Obstetrics and Gynecology · 2025
    Article
  10. Catheter-Based Fetal Cardiac Interventions.Journal of cardiovascular development and disease · 2024
    Review
  11. Review
  12. Article
  13. Advances in Fetal Cardiac Imaging and Intervention.CJC pediatric and congenital heart disease · 2024
    Review
  14. Article
  15. Molecular Pathways and Animal Models of Hypoplastic Left Heart Syndrome.Advances in experimental medicine and biology · 2024
    Review
  16. Article
  17. Review
  18. Article
  19. Review
  20. Article
4 · The record

Corrections and comments

5 · Who and what money

Authors and funding

13 authors at 2 institutions in 1 country.

Lindsay R FreudFrom the Departments of Cardiology (L.R.F., D.B.M., A.C.M., G.R.M., K.G.F., T.L., J.E.L., W.T.) and Cardiac Surgery (P.J.d.N., S.M.E.), Boston Children's Hospital, Boston, MA; and the Departments of Obstetrics and Gynecology (V.S., L.E.W.-H.) and Radiology (C.B.B.), Brigham and Women's Hospital; Harvard Medical School, Boston, MA. lindsay.freud@cardio.chboston.org.
Doff B McElhinneyFrom the Departments of Cardiology (L.R.F., D.B.M., A.C.M., G.R.M., K.G.F., T.L., J.E.L., W.T.) and Cardiac Surgery (P.J.d.N., S.M.E.), Boston Children's Hospital, Boston, MA; and the Departments of Obstetrics and Gynecology (V.S., L.E.W.-H.) and Radiology (C.B.B.), Brigham and Women's Hospital; Harvard Medical School, Boston, MA.
Audrey C MarshallFrom the Departments of Cardiology (L.R.F., D.B.M., A.C.M., G.R.M., K.G.F., T.L., J.E.L., W.T.) and Cardiac Surgery (P.J.d.N., S.M.E.), Boston Children's Hospital, Boston, MA; and the Departments of Obstetrics and Gynecology (V.S., L.E.W.-H.) and Radiology (C.B.B.), Brigham and Women's Hospital; Harvard Medical School, Boston, MA.
Gerald R MarxFrom the Departments of Cardiology (L.R.F., D.B.M., A.C.M., G.R.M., K.G.F., T.L., J.E.L., W.T.) and Cardiac Surgery (P.J.d.N., S.M.E.), Boston Children's Hospital, Boston, MA; and the Departments of Obstetrics and Gynecology (V.S., L.E.W.-H.) and Radiology (C.B.B.), Brigham and Women's Hospital; Harvard Medical School, Boston, MA.
Kevin G FriedmanFrom the Departments of Cardiology (L.R.F., D.B.M., A.C.M., G.R.M., K.G.F., T.L., J.E.L., W.T.) and Cardiac Surgery (P.J.d.N., S.M.E.), Boston Children's Hospital, Boston, MA; and the Departments of Obstetrics and Gynecology (V.S., L.E.W.-H.) and Radiology (C.B.B.), Brigham and Women's Hospital; Harvard Medical School, Boston, MA.
Pedro J del NidoFrom the Departments of Cardiology (L.R.F., D.B.M., A.C.M., G.R.M., K.G.F., T.L., J.E.L., W.T.) and Cardiac Surgery (P.J.d.N., S.M.E.), Boston Children's Hospital, Boston, MA; and the Departments of Obstetrics and Gynecology (V.S., L.E.W.-H.) and Radiology (C.B.B.), Brigham and Women's Hospital; Harvard Medical School, Boston, MA.
Sitaram M EmaniFrom the Departments of Cardiology (L.R.F., D.B.M., A.C.M., G.R.M., K.G.F., T.L., J.E.L., W.T.) and Cardiac Surgery (P.J.d.N., S.M.E.), Boston Children's Hospital, Boston, MA; and the Departments of Obstetrics and Gynecology (V.S., L.E.W.-H.) and Radiology (C.B.B.), Brigham and Women's Hospital; Harvard Medical School, Boston, MA.
Terra LafranchiFrom the Departments of Cardiology (L.R.F., D.B.M., A.C.M., G.R.M., K.G.F., T.L., J.E.L., W.T.) and Cardiac Surgery (P.J.d.N., S.M.E.), Boston Children's Hospital, Boston, MA; and the Departments of Obstetrics and Gynecology (V.S., L.E.W.-H.) and Radiology (C.B.B.), Brigham and Women's Hospital; Harvard Medical School, Boston, MA.
Virginia SilvaFrom the Departments of Cardiology (L.R.F., D.B.M., A.C.M., G.R.M., K.G.F., T.L., J.E.L., W.T.) and Cardiac Surgery (P.J.d.N., S.M.E.), Boston Children's Hospital, Boston, MA; and the Departments of Obstetrics and Gynecology (V.S., L.E.W.-H.) and Radiology (C.B.B.), Brigham and Women's Hospital; Harvard Medical School, Boston, MA.
Louise E Wilkins-HaugFrom the Departments of Cardiology (L.R.F., D.B.M., A.C.M., G.R.M., K.G.F., T.L., J.E.L., W.T.) and Cardiac Surgery (P.J.d.N., S.M.E.), Boston Children's Hospital, Boston, MA; and the Departments of Obstetrics and Gynecology (V.S., L.E.W.-H.) and Radiology (C.B.B.), Brigham and Women's Hospital; Harvard Medical School, Boston, MA.
Carol B BensonFrom the Departments of Cardiology (L.R.F., D.B.M., A.C.M., G.R.M., K.G.F., T.L., J.E.L., W.T.) and Cardiac Surgery (P.J.d.N., S.M.E.), Boston Children's Hospital, Boston, MA; and the Departments of Obstetrics and Gynecology (V.S., L.E.W.-H.) and Radiology (C.B.B.), Brigham and Women's Hospital; Harvard Medical School, Boston, MA.
James E LockFrom the Departments of Cardiology (L.R.F., D.B.M., A.C.M., G.R.M., K.G.F., T.L., J.E.L., W.T.) and Cardiac Surgery (P.J.d.N., S.M.E.), Boston Children's Hospital, Boston, MA; and the Departments of Obstetrics and Gynecology (V.S., L.E.W.-H.) and Radiology (C.B.B.), Brigham and Women's Hospital; Harvard Medical School, Boston, MA.
Wayne TworetzkyFrom the Departments of Cardiology (L.R.F., D.B.M., A.C.M., G.R.M., K.G.F., T.L., J.E.L., W.T.) and Cardiac Surgery (P.J.d.N., S.M.E.), Boston Children's Hospital, Boston, MA; and the Departments of Obstetrics and Gynecology (V.S., L.E.W.-H.) and Radiology (C.B.B.), Brigham and Women's Hospital; Harvard Medical School, Boston, MA.
Harvard University · USBrigham and Women's Hospital · US

Funding

RESEARCH METHODS IN PEDIATRICS HEART DISEASET32HL007572 · NHLBI · CHILDREN'S HOSPITAL BOSTON · PI Sarah D DE FERRANTI, Jane W. Newburger · 1985 to 2026
$13.0M
NHLBI NIH HHS T32 HL007572NHLBI NIH HHS T32-HL757229
6 · The paper itself

Abstract

backgroundFetal aortic valvuloplasty can be performed for severe midgestation aortic stenosis in an attempt to prevent progression to hypoplastic left heart syndrome (HLHS). A subset of patients has achieved a biventricular (BV) circulation after fetal aortic valvuloplasty. The postnatal outcomes and survival of the BV patients, in comparison with those managed as HLHS, have not been reported. METHODS AND

resultsWe included 100 patients who underwent fetal aortic valvuloplasty for severe midgestation aortic stenosis with evolving HLHS from March 2000 to January 2013. Patients were categorized based on postnatal management as BV or HLHS. Clinical records were reviewed. Eighty-eight fetuses were live-born, and 38 had a BV circulation (31 from birth, 7 converted after initial univentricular palliation). Left-sided structures, namely aortic and mitral valve sizes and left ventricular volume, were significantly larger in the BV group at the time of birth (P<0.01). After a median follow-up of 5.4 years, freedom from cardiac death among all BV patients was 96±4% at 5 years and 84±12% at 10 years, which was better than HLHS patients (log-rank P=0.04). There was no cardiac mortality in patients with a BV circulation from birth. All but 1 of the BV patients required postnatal intervention; 42% underwent aortic or mitral valve replacement. On the most recent echocardiogram, the median left ventricular end-diastolic volume z score was +1.7 (range, -1.3 to +8.2), and 80% had normal ejection fraction.

conclusionsShort- and intermediate-term survival among patients who underwent fetal aortic valvuloplasty and achieved a BV circulation postnatally is encouraging. However, morbidity still exists, and ongoing assessment is warranted.

Indexed as

Aortic Valve StenosisBalloon ValvuloplastyCoronary CirculationDisease ProgressionFemaleFetal DiseasesFollow-Up StudiesGestational AgeHumansHypoplastic Left Heart SyndromeInfant, NewbornKaplan-Meier EstimatePregnancyPregnancy OutcomeUltrasonography, Prenatalaortic valve stenosisballoon valvuloplastyfetal heartheart defects, congenitalhypoplastic left heart syndromepediatricstreatment outcome

Identifiers

PMID25052401
PMCPMC4299861
OpenAlexW2006917139

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Registered trials

Read under generation 80e0d062 · epoch 390. Bibliography from PubMed, PubMed Central and OpenAlex; grants from NIH RePORTER; trial links from ClinicalTrials.gov; estimates, votes and beliefs from the OpenQuestion graph.