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  1. Article ; Online: Expanding neonatal ECMO criteria: When is the premature neonate too premature.

    Wild, K Taylor / Burgos, Carmen Mesas / Rintoul, Natalie E

    Seminars in fetal & neonatal medicine

    2022  Volume 27, Issue 6, Page(s) 101403

    Abstract: Extracorporeal membrane oxygenation (ECMO) is a universally accepted and life-saving therapy for neonates with respiratory or cardiac failure that is refractory to maximal medical management. Early studies found unacceptable risks of mortality and ... ...

    Abstract Extracorporeal membrane oxygenation (ECMO) is a universally accepted and life-saving therapy for neonates with respiratory or cardiac failure that is refractory to maximal medical management. Early studies found unacceptable risks of mortality and morbidities such as intracranial hemorrhage among premature and low birthweight neonates, leading to widely accepted ECMO inclusion criteria of gestational age ≥34 weeks and birthweight >2 kg. Although contemporary data is lacking, the most recent literature demonstrates increased survival and decreased rates of intracranial hemorrhage in premature neonates who are supported with ECMO. As such, it seems like the right time to push the boundaries of ECMO on a case-by-case basis beginning with neonates 32-34 weeks GA in large volume centers with careful neurodevelopmental follow-up to better inform practices changes on this select population.
    MeSH term(s) Infant, Newborn ; Female ; Humans ; Infant ; Extracorporeal Membrane Oxygenation ; Birth Weight ; Infant, Newborn, Diseases ; Intracranial Hemorrhages/diagnostic imaging ; Intracranial Hemorrhages/therapy ; Gestational Age ; Premature Birth ; Retrospective Studies
    Language English
    Publishing date 2022-11-17
    Publishing country Netherlands
    Document type Journal Article ; Review
    ZDB-ID 2174416-6
    ISSN 1878-0946 ; 1744-165X
    ISSN (online) 1878-0946
    ISSN 1744-165X
    DOI 10.1016/j.siny.2022.101403
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  2. Article ; Online: Prenatal management of congenital diaphragmatic hernia.

    Danzer, Enrico / Rintoul, Natalie E / van Meurs, Krisa P / Deprest, Jan

    Seminars in fetal & neonatal medicine

    2022  Volume 27, Issue 6, Page(s) 101406

    Abstract: Recently, two randomized controlled, prospective trials, the Tracheal Occlusion to Accelerate Lung Growth (TOTAL) trials, reported the outcomes on fetal endoluminal tracheal occlusion (FETO) for isolated left congenital diaphragmatic hernia (CDH). FETO ... ...

    Abstract Recently, two randomized controlled, prospective trials, the Tracheal Occlusion to Accelerate Lung Growth (TOTAL) trials, reported the outcomes on fetal endoluminal tracheal occlusion (FETO) for isolated left congenital diaphragmatic hernia (CDH). FETO significantly improved outcomes for severe hypoplasia. The effect in moderate cases, where the balloon was inserted later in pregnancy, did not reach significance. In a pooled analysis investigating the effect of the heterogeneity of the treatment effect by the time point of occlusion and severity, the difference may be explained by a difference in the duration of occlusion. Nevertheless, FETO carries a significant risk of preterm birth. The primary objective of this review is to provide an overview of the rationale for fetal intervention in CDH and the results of the randomized trials. The secondary objective is to discuss the technical aspects of FETO. Finally, recent developments of potential alternative fetal approaches will be highlighted.
    MeSH term(s) Female ; Humans ; Infant, Newborn ; Pregnancy ; Fetoscopy ; Hernias, Diaphragmatic, Congenital/surgery ; Premature Birth ; Prospective Studies ; Trachea/surgery ; Randomized Controlled Trials as Topic
    Language English
    Publishing date 2022-11-17
    Publishing country Netherlands
    Document type Journal Article ; Review
    ZDB-ID 2174416-6
    ISSN 1878-0946 ; 1744-165X
    ISSN (online) 1878-0946
    ISSN 1744-165X
    DOI 10.1016/j.siny.2022.101406
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  3. Article ; Online: Delivery Room Resuscitation of Infants with Congenital Diaphragmatic Hernia: Lessons Learned through Video Review.

    Wild, K Taylor / Rintoul, Natalie / Hedrick, Holly L / Heimall, Lauren / Soorikian, Leane / Foglia, Elizabeth E / Ades, Anne M / Herrick, Heidi M

    Fetal diagnosis and therapy

    2024  

    Abstract: Introduction: Delivery room (DR) interventions for infants with congenital diaphragmatic hernia (CDH) are not well described. This study sought to describe timing and order of DR interventions and identify system factors impacting CDH DR resuscitations ... ...

    Abstract Introduction: Delivery room (DR) interventions for infants with congenital diaphragmatic hernia (CDH) are not well described. This study sought to describe timing and order of DR interventions and identify system factors impacting CDH DR resuscitations using a human factors framework.
    Methods: Single center observational study of video recorded CDH DR resuscitations documenting timing and order of interventions. The team used the Systems Engineering Initiative for Patient Safety (SEIPS) model to identify system factors impacting DR resuscitations and time to invasive ventilation.
    Results: We analyzed 31 video recorded CDH resuscitations. We observed variability in timing and order of resuscitation tasks. The 'Internal Environment' and 'Tasks' components of the SEIPS model were prominent factors affecting resuscitation efficiency; significant room and bed spatial constraints exist, and nurses have a significant task burden. Additionally, endotracheal tube preparation was a prominent barrier to timely invasive ventilation.
    Conclusion: Video review revealed variation in event timing and order during CDH resuscitations. Standardization of room set-up, equipment, and event order and reallocation of tasks facilitate more efficient intubation and ventilation, representing targets for CDH DR improvement initiatives. This work emphasizes the utility of rigorous human factors review to identify areas for improvement during DR resuscitation.
    Language English
    Publishing date 2024-03-26
    Publishing country Switzerland
    Document type Journal Article
    ZDB-ID 1066460-9
    ISSN 1421-9964 ; 1015-3837
    ISSN (online) 1421-9964
    ISSN 1015-3837
    DOI 10.1159/000538536
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  4. Article ; Online: Reconsidering ECMO in Premature Neonates.

    Wild, K Taylor / Hedrick, Holly L / Rintoul, Natalie E

    Fetal diagnosis and therapy

    2020  Volume 47, Issue 12, Page(s) 927–932

    Abstract: Extracorporeal membrane oxygenation (ECMO) is a life-saving intervention for neonates with respiratory failure or congenital cardiac disease refractory to maximal medical management. Early studies showed high rates of mortality and morbidities among ... ...

    Abstract Extracorporeal membrane oxygenation (ECMO) is a life-saving intervention for neonates with respiratory failure or congenital cardiac disease refractory to maximal medical management. Early studies showed high rates of mortality and morbidities among preterm and low birthweight (BW) neonates, leading to widely accepted ECMO inclusion criteria of gestational age (GA) ≥34 weeks and BW >2 kg. In recent years, publications involving neonates of 32-34 weeks GA have reported improved survival and decreased intracranial hemorrhage. As such, ECMO should be considered on a case-by-case basis in premature neonates as long as the risks are understood.
    MeSH term(s) Extracorporeal Membrane Oxygenation ; Gestational Age ; Hernias, Diaphragmatic, Congenital ; Humans ; Infant ; Infant, Low Birth Weight ; Infant, Newborn ; Retrospective Studies
    Language English
    Publishing date 2020-09-01
    Publishing country Switzerland
    Document type Journal Article ; Review
    ZDB-ID 1066460-9
    ISSN 1421-9964 ; 1015-3837
    ISSN (online) 1421-9964
    ISSN 1015-3837
    DOI 10.1159/000509243
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  5. Article ; Online: Congenital Diaphragmatic Hernia Patients with Left Heart Hypoplasia and Left Ventricular Dysfunction Have Highest Odds of Mortality.

    Fraga, María V / Hedrick, Holly L / Rintoul, Natalie E / Wang, Yan / Ash, Devon / Flohr, Sabrina J / Mathew, Leny / Reynolds, Tom / Engelman, Jenny L / Avitabile, Catherine M

    The Journal of pediatrics

    2024  , Page(s) 114061

    Abstract: Objectives: To describe the scope of left ventricular (LV) dysfunction and left heart hypoplasia (LHH) in infants with congenital diaphragmatic hernia (CDH), to determine associations with CDH severity, and to evaluate the odds of extracorporeal ... ...

    Abstract Objectives: To describe the scope of left ventricular (LV) dysfunction and left heart hypoplasia (LHH) in infants with congenital diaphragmatic hernia (CDH), to determine associations with CDH severity, and to evaluate the odds of extracorporeal membrane oxygenation (ECMO) and death with categories of left heart disease.
    Study design: Demographic and clinical variables were collected from a single-center, retrospective cohort of CDH patients from January 2017 through May 2022. Quantitative measures of LV function and LHH were prospectively performed on initial echocardiograms. LHH was defined as ≥2 of the following: Z-score ≤ -2 of any left heart structure or LV end-diastolic volume <3 mL. LV dysfunction was defined as shortening fraction <28%, ejection fraction <60%, or global longitudinal strain <20%. The exposure was operationalized as a four-group categorical variable (LV dysfunction +/-, LHH +/-). Logistic regression models evaluated associations with ECMO and death, adjusting for CDH severity.
    Results: One-hundred and eight-two patients (80.8% left CDH, 63.2% liver herniation, 23.6% ECMO, 12.1% mortality) were included. Twenty percent demonstrated normal LV function and no LHH (LV dysfunction-/LHH-), 37% normal LV function with LHH (LV dysfunction-/LHH+), 14% LV dysfunction without LHH (LV dysfunction+/LHH-), and 28% both LV dysfunction and LHH (LV dysfunction+/LHH+). There was a dose-response effect between increasing severity of left heart disease, ECMO use, and mortality. LV dysfunction+/LHH+ infants had the highest odds of ECMO use and death, after adjustment for CDH severity [OR (95% CI); 1.76 (1.20,2.62) for ECMO, 2.76 (1.63, 5.17) for death].
    Conclusions: In our large single-center cohort, CDH patients with LV dysfunction+/LHH+ had the highest risk of ECMO use and death.
    Language English
    Publishing date 2024-04-16
    Publishing country United States
    Document type Journal Article
    ZDB-ID 3102-1
    ISSN 1097-6833 ; 0022-3476
    ISSN (online) 1097-6833
    ISSN 0022-3476
    DOI 10.1016/j.jpeds.2024.114061
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  6. Article ; Online: High rate of extreme thrombocytosis indicates bone marrow hyperactivity and splenic dysfunction among congenital diaphragmatic hernia patients.

    Gu, Hannah / Devine, Matthew / Hedrick, Holly L / Rintoul, Natalie E / Thom, Christopher S

    Platelets

    2021  Volume 33, Issue 5, Page(s) 787–789

    Abstract: Pediatric extreme thrombocytosis (EXT, platelet count > 1000 x ... ...

    Abstract Pediatric extreme thrombocytosis (EXT, platelet count > 1000 x 10
    MeSH term(s) Bone Marrow ; Child ; Hernias, Diaphragmatic, Congenital/complications ; Humans ; Infant ; Platelet Count ; Retrospective Studies ; Thrombocytosis/etiology
    Language English
    Publishing date 2021-10-26
    Publishing country England
    Document type Journal Article
    ZDB-ID 1034283-7
    ISSN 1369-1635 ; 0953-7104
    ISSN (online) 1369-1635
    ISSN 0953-7104
    DOI 10.1080/09537104.2021.1994546
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    Zs.A 2888: Show issues Location:
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  7. Article ; Online: Genomic Contributors to Esophageal Atresia and Tracheoesophageal Fistula: A 12 Year Retrospective Review.

    Wild, K Taylor / Conlin, Laura / Blair, Justin / Manfredi, Michael / Hamilton, Thomas E / Muir, Amanda / Zackai, Elaine H / Nace, Gary / Partridge, Emily A / Devine, Matthew / Reynolds, Tom / Rintoul, Natalie E / Hedrick, Holly L / Spinner, Nancy / Krantz, Ian D

    The Journal of pediatrics

    2024  Volume 271, Page(s) 114060

    Abstract: ... hospital. For each infant, demographic information, prenatal and postnatal history, and genetic testing ...

    Abstract Objective: To evaluate genetic testing utilization and diagnostic yield in infants with esophageal atresia (EA)/tracheoesophageal fistula (TEF) over the past 12 years to inform future practices and individualize prognostication and management.
    Study design: A retrospective cohort study was performed for all infants with EA or EA/TEF hospitalized between January 2011 and January 2023 at a quaternary children's hospital. For each infant, demographic information, prenatal and postnatal history, and genetic testing were reviewed.
    Results: There were 212 infants who were classified as follows: 1) complex/syndromic with EA/TEF plus an additional major anatomic anomaly (n = 114, of which 74 met VACTERL criteria); 2) isolated/nonsyndromic EA/TEF (n = 88) and 3) isolated/nonsyndromic EA (n = 10). A range of genetic tests were sent with varying diagnostic rates including karyotype analysis in 12 (all with complex/syndromic phenotypes and all positive), chromosomal microarray analysis in 189 (114 of whom were complex/syndromic with an overall diagnostic rate of 3/189), single gene testing for CHD7 in 18 (4 positive), and exome analysis in 37 complex/syndromic patients (8 positive).
    Conclusions: EA/TEF with and without additional anomalies is genetically heterogeneous with a broad range of associated phenotypes. While the genetic etiology of EA/TEF with or without VACTERL remains largely unknown, genome wide testing (exome or genome) including copy number analysis is recommended over chromosomal microarray testing. We anticipate that expanded genetic/genomic testing modalities such as RNA sequencing and tissue specific molecular testing are needed in this cohort to improve our understanding of the genomic contributors to EA/TEF.
    Language English
    Publishing date 2024-04-18
    Publishing country United States
    Document type Journal Article
    ZDB-ID 3102-1
    ISSN 1097-6833 ; 0022-3476
    ISSN (online) 1097-6833
    ISSN 0022-3476
    DOI 10.1016/j.jpeds.2024.114060
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  8. Article ; Online: Single-Center Comparison of Outcomes Following Cardiac Surgery in Low Birth Weight and Standard Birth Weight Neonates.

    Kim, Minso / Okunowo, Oluwatimilehin / Ades, Anne M / Fuller, Stephanie / Rintoul, Natalie E / Naim, Maryam Y

    The Journal of pediatrics

    2021  Volume 238, Page(s) 161–167.e1

    Abstract: Objective: To compare outcomes between low birth weight (LBW; <2.5 kg) and standard birth weight neonates undergoing cardiac surgery.: Study design: A single-center retrospective study of neonates undergoing cardiac surgery with cardiopulmonary ... ...

    Abstract Objective: To compare outcomes between low birth weight (LBW; <2.5 kg) and standard birth weight neonates undergoing cardiac surgery.
    Study design: A single-center retrospective study of neonates undergoing cardiac surgery with cardiopulmonary bypass from 2012 to 2018. LBW neonates were 1:2 propensity score-matched to standard birth weight neonates (n = 93 to n = 186) using clinical characteristics. The primary and secondary outcomes were survival to hospital discharge and postoperative complications, respectively. After matching, regression analyses were conducted to compare outcomes.
    Results: The LBW group had a higher proportion of premature neonates than the standard birth weight group (60% vs 8%; P < .01) and were less likely to survive to hospital discharge (88% vs 95%; OR, 0.39; 95% CI, 0.15-0.97). There was no difference in unplanned cardiac reoperations or catheter-based interventions, cardiac arrest, extracorporeal membrane oxygenation, infection, and end-organ complications between the groups. Among LBW infants, survival was improved at weight >2 kg.
    Conclusions: LBW is a risk factor for decreased survival. LBW neonates weighing >2 kg have survival comparable to those weighing >2.5 kg.
    MeSH term(s) Birth Weight ; Cardiac Surgical Procedures ; Heart Defects, Congenital/surgery ; Humans ; Infant ; Infant, Low Birth Weight ; Infant, Newborn ; Retrospective Studies ; Treatment Outcome
    Language English
    Publishing date 2021-06-30
    Publishing country United States
    Document type Journal Article ; Comment
    ZDB-ID 3102-1
    ISSN 1097-6833 ; 0022-3476
    ISSN (online) 1097-6833
    ISSN 0022-3476
    DOI 10.1016/j.jpeds.2021.06.059
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  9. Article ; Online: The Delivery Room Resuscitation of Infants with Congenital Diaphragmatic Hernia Treated with Fetoscopic Endoluminal Tracheal Occlusion: Beyond the Balloon.

    Wild, K Taylor / Rintoul, Natalie E / Ades, Anne M / Gebb, Juliana S / Moldenhauer, Julie S / Mathew, Leny / Flohr, Sabrina / Bostwick, Anna / Reynolds, Tom / Ruiz, Ryan L / Javia, Luv R / Nelson, Olivia / Peranteau, William H / Partridge, Emily A / Adzick, N Scott / Hedrick, Holly L

    Fetal diagnosis and therapy

    2024  Volume 51, Issue 2, Page(s) 184–190

    Abstract: ... The neonatal clinical team must be skilled in managing the unique postnatal physiology inherent to FETO ...

    Abstract Introduction: Randomized controlled trials found that fetoscopic endoluminal tracheal occlusion (FETO) resulted in increased fetal lung volume and improved survival for infants with isolated, severe left-sided congenital diaphragmatic hernia (CDH). The delivery room resuscitation of these infants is particularly unique, and the specific delivery room events are largely unknown. The objective of this study was to compare the delivery room resuscitation of infants treated with FETO to standard of care (SOC) and describe lessons learned.
    Methods: Retrospective single-center cohort study of infants treated with FETO compared to infants who met FETO criteria during the same period but who received SOC.
    Results: FETO infants were more likely to be born prematurely with 8/12 infants born <35 weeks gestational age compared to 3/35 SOC infants. There were 5 infants who required emergent balloon removal (2 ex utero intrapartum treatment and 3 tracheoscopic removal on placental bypass with delayed cord clamping) and 7 with prenatal balloon removal. Surfactant was administered in 6/12 FETO (50%) infants compared to 2/35 (6%) in the SOC group. Extracorporeal membrane oxygenation use was lower at 25% and survival was higher at 92% compared to 60% and 71% in the SOC infants, respectively.
    Conclusion: The delivery room resuscitation of infants treated with FETO requires thoughtful preparation with an experienced multidisciplinary team. Given increased survival, FETO should be offered to infants with severe isolated left-sided CDH, but only in high-volume centers with the experience and capability of removing the balloon, emergently if needed. The neonatal clinical team must be skilled in managing the unique postnatal physiology inherent to FETO where effective interdisciplinary teamwork is essential. Empiric and immediate surfactant administration should be considered in all FETO infants to lavage thick airway secretions, particularly those delivered <48 h after balloon removal.
    MeSH term(s) Infant, Newborn ; Infant ; Female ; Humans ; Pregnancy ; Hernias, Diaphragmatic, Congenital/surgery ; Retrospective Studies ; Cohort Studies ; Delivery Rooms ; Balloon Occlusion/methods ; Placenta ; Fetoscopy/methods ; Trachea/surgery ; Surface-Active Agents
    Chemical Substances Surface-Active Agents
    Language English
    Publishing date 2024-01-10
    Publishing country Switzerland
    Document type Journal Article
    ZDB-ID 1066460-9
    ISSN 1421-9964 ; 1015-3837
    ISSN (online) 1421-9964
    ISSN 1015-3837
    DOI 10.1159/000536209
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  10. Article ; Online: Extracorporeal membrane oxygenation in infants with congenital diaphragmatic hernia.

    Grover, Theresa R / Rintoul, Natalie E / Hedrick, Holly L

    Seminars in perinatology

    2018  Volume 42, Issue 2, Page(s) 96–103

    Abstract: ... and mortality. CDH is the most common non-cardiac indication for neonatal ECMO. Prenatal and postnatal ...

    Abstract Congenital diaphragmatic hernia (CDH) is a severe congenital anomaly which impairs normal pulmonary development leading to acute and chronic respiratory failure, pulmonary hypoplasia, pulmonary hypertension, and mortality. CDH is the most common non-cardiac indication for neonatal ECMO. Prenatal and postnatal predictors of CDH severity aid in patient selection. Centers vary in preferred mode of ECMO and timing of CDH repair. Survivors of severe CDH with ECMO are at risk for long-term sequelae including neurodevelopmental delays.
    MeSH term(s) Developmental Disabilities/physiopathology ; Developmental Disabilities/prevention & control ; Extracorporeal Membrane Oxygenation ; Hernias, Diaphragmatic, Congenital/mortality ; Hernias, Diaphragmatic, Congenital/physiopathology ; Hernias, Diaphragmatic, Congenital/therapy ; Humans ; Infant ; Infant, Newborn ; Intensive Care, Neonatal ; Practice Guidelines as Topic ; Survival Rate
    Language English
    Publishing date 2018-01-12
    Publishing country United States
    Document type Journal Article ; Review
    ZDB-ID 752403-1
    ISSN 1558-075X ; 0146-0005
    ISSN (online) 1558-075X
    ISSN 0146-0005
    DOI 10.1053/j.semperi.2017.12.005
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