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Abstract

Objective

To determine the effect, if any, of prenatal intervention in fetuses with congenital diaphragmatic hernia (CDH) on the likelihood of attempted trial of resuscitation (TOR) or withdrawal of life-sustaining measures (WLSM) in the postnatal neonatal intensive care environment.

Study Design

Four hypothetical patients with either left- or right-sided CDH were devised using prognostic survival data from the literature, depending on whether they received fetoscopic endoluminal tracheal occlusion (FETO) or expectant management in the prenatal setting. Healthcare providers from the CDH Study Group (CDHSG) listserv were surveyed to report their likelihood of offering a trial of resuscitation and obliging a parental request for WLSM for each patient. Response data were analyzed using a Wilcoxon rank sum test, and statistical significance was assumed at p < 0.01.

Results

39 complete responses (rate = 19%) were comprised primarily of physicians in pediatric surgery and neonatology, with other healthcare roles and specialties represented. A left-sided CDH patient with prenatal intervention was significantly favored for a TOR when compared to a patient without prenatal intervention (69% vs 11%; p < 0.001). Additionally, there was a lower rate of obliging a parental request for WLSM in the patient with prenatal intervention compared to a patient without (19% vs 64%; p < 0.001). In contrast, there was no observed effect of prenatal intervention in patients with right-sided hernias. In a scenario of simultaneous resource utilization, the patient with a left-sided CDH who underwent prenatal intervention was given priority for a TOR.

Conclusions

To our knowledge, this is the first description of the potential impact of prenatal intervention on postnatal decision-making, using the exemplar case of CDH. These findings have profound implications for maternal-fetal surgery providers and patients, raising concerns of equity and justice and providing preliminary empirical evidence for the fetus as a patient in the era of modern prenatal intervention.

Keywords

congenital diaphragmatic hernia clinical decision-making tracheal occlusion life-sustaining treatment

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References

Walsh

Adzick

. Fetal surgical intervention. Am J Perinatol 2000; 17(06): 277–284. https://doi.org/10.1055/s-2000-13439

Evans

Harrison

. Modern fetal surgery—a historical review of the happenings that shaped modern fetal surgery and its practices. Transl Pediatr 2021; 10(5): 1401–1417. https://doi.org/10.21037/tp-20-114

Coughlin

Gupta

Ebanks

, et al. Incidence and outcomes of patients with congenital diaphragmatic hernia and pulmonary sequestration. J Pediatr Surg 2021; 56(6): 1126–1129. https://doi.org/10.1016/j.jpedsurg.2021.02.032

Tsao

Allison

Harting

, et al. Congenital diaphragmatic hernia in the preterm infant. Surgery 2010; 148(2): 404–410. https://doi.org/10.1016/j.surg.2010.03.018

Graziano

. Cardiac anomalies in patients with congenital diaphragmatic hernia and their prognosis: a report from the Congenital Diaphragmatic Hernia Study Group. J Pediatr Surg 2005; 40(6): 1045–1050. https://doi.org/10.1016/j.jpedsurg.2005.03.025

Gupta

Harting

. Congenital diaphragmatic hernia-associated pulmonary hypertension. Semin Perinatol 2020; 44(1): 151167. https://doi.org/10.1053/j.semperi.2019.07.006

Montalva

Antounians

Zani

. Pulmonary hypertension secondary to congenital diaphragmatic hernia: factors and pathways involved in pulmonary vascular remodeling. Pediatr Res 2019; 85(6): 754–768. https://doi.org/10.1038/s41390-019-0345-4

Madenci

Church

Gajarski

, et al.

Pulmonary hypertension in patients with congenital diaphragmatic hernia: does lung size matter?

Eur J Pediatr Surg 2018; 28(06): 508–514. https://doi.org/10.1055/s-0037-1607291

Russo

Eastwood

Keijzer

, et al. Lung size and liver herniation predict need for extracorporeal membrane oxygenation but not pulmonary hypertension in isolated congenital diaphragmatic hernia: systematic review and meta-analysis. Ultrasound Obstet Gynecol 2017; 49(6): 704–713. https://doi.org/10.1002/uog.16000

10.

Cordier

Russo

Deprest

, et al. Prenatal diagnosis, imaging, and prognosis in Congenital Diaphragmatic Hernia. Semin Perinatol 2020; 44(1): 51163. https://doi.org/10.1053/j.semperi.2019.07.002

11.

Deprest

Benachi

Gratacos

, et al. Randomized trial of fetal surgery for moderate left diaphragmatic Hernia. N Engl J Med 2021; 385(2): 119–129. https://doi.org/10.1056/NEJMoa2026983

12.

Deprest

Nicolaides

Benachi

, et al. Randomized trial of fetal surgery for severe left diaphragmatic Hernia. N Engl J Med 2021; 385(2): 107–118. https://doi.org/10.1056/NEJMoa2027030

13.

Vergote

Russo

Basurto

, et al.

Have the results of the TOTAL‐trials changed the attitude and practice of maternal‐fetal medicine specialists?

Prenat Diagn 2023; 43(8): 1018–1027. https://doi.org/10.1002/pd.6403

14.

Pertierra Cortada

Clotet Caba

Hadley

, et al.

Do FETO CDH survivors need the same follow-up program as non-FETO patients?

Eur J Pediatr 2023; 182(7): 3243–3255. https://doi.org/10.1007/s00431-023-04977-3

15.

Jancelewicz

Brindle

. Prediction tools in congenital diaphragmatic hernia. Semin Perinatol 2020; 44(1): 151165. https://doi.org/10.1053/j.semperi.2019.07.004

16.

Collin

Trinder

Minutillo

, et al. A modern era comparison of right versus left sided congenital diaphragmatic hernia outcomes. J Pediatr Surg 2016; 51(9): 1409–1413. https://doi.org/10.1016/j.jpedsurg.2016.05.001

17.

DeKoninck

Gomez

Sandaite

, et al. Right‐sided congenital diaphragmatic hernia in a decade of fetal surgery. BJOG Int J Obstet Gynaecol 2015; 122(7): 940–946. https://doi.org/10.1111/1471-0528.13065

18.

Janvier

Leblanc

Barrington

. The best-interest standard is not applied for neonatal resuscitation decisions. Pediatrics 2008; 121(5): 963–969. https://doi.org/10.1542/peds.2007-1520

19.

Janvier

Leblanc

Barrington

. Nobody likes premies: the relative value of patients’ lives. J Perinatol 2008; 28(12): 821–826. https://doi.org/10.1038/jp.2008.103

20.

Janvier

Mercurio

. Saving vs creating: perceptions of intensive care at different ages and the potential for injustice. J Perinatol 2013; 33(5): 333–335. https://doi.org/10.1038/jp.2012.134

21.

Marcello

Stefano

Lampron

, et al. The influence of family characteristics on perinatal decision making. Pediatrics 2011; 127(4): e934–e939. https://doi.org/10.1542/peds.2009-3010

22.

Minkoff

Berkowitz

. The myth of the precious baby. Obstet Gynecol 2005; 106(3): 607–609. https://doi.org/10.1097/01.AOG.0000174585.08884.59

23.

Zhong

Knobe

Feigenson

, et al. Age and disability biases in pediatric resuscitation among future physicians. Clin Pediatr (Phila) 2011; 50(11): 1001–1004. https://doi.org/10.1177/0009922811410053

24.

Strong

. The moral status of preembryos, Embryos, fetuses, and infants. J Med Philos 1997; 22(5): 457–478. https://doi.org/10.1093/jmp/22.5.457

25.

Brown

. The moral status of the fetus: implications of the somatic integration definition of human life. Bioethics 2021; 35(7): 672–679. https://doi.org/10.1111/bioe.12853

26.

DePresT

Toelen

Debyser

. The fetal patient – ethical aspects of fetal therapy.

27.

Rodrigues

HCML

Van Den Berg

Düwell

. Dotting the i’s and crossing the t’s: autonomy and/or beneficence? The ‘fetus as a patient’ in maternal–fetal surgery. J Med Ethics 2013; 39(4): 219–223. https://doi.org/10.1136/medethics-2012-100781

28.

Antiel

. Ethical challenges in the new world of maternal–fetal surgery. Semin Perinatol 2016; 40(4): 227–233. https://doi.org/10.1053/j.semperi.2015.12.012

29.

Lyerly

Little

Faden

. A Critique of the ‘Fetus as Patient. Am J Bioeth 2008; 8(7): 42–44. https://doi.org/10.1080/15265160802331678

30.

Goldblatt Hyatt

Wilpers

Bahtiyar

, et al. “I don’t have a telephone to the fetus”: clinicians’ conceptions of fetal patienthood in maternal-fetal surgery counseling. Soc Sci Med 2024; 342: 116525. https://doi.org/10.1016/j.socscimed.2023.116525

31.

Shao

Chen

Kozel

, et al. Postnatal myelomeningocele repair in the United States: rates and disparities before and after the management of myelomeningocele study trial. Neurosurgery 2023; 93(6): 1374–1382. https://doi.org/10.1227/neu.0000000000002604

32.

Wilpers

Lynn

Eichhorn

, et al. Understanding sociodemographic disparities in maternal-fetal surgery study participation. Fetal Diagn Ther 2022; 49(3): 125–137. https://doi.org/10.1159/000523867

33.

Fryer

Munoz

Rahangdale

, et al. Multiparous Black and latinx women face more barriers to prenatal care than white women. J Racial Ethn Health Disparities 2021; 8(1): 80–87. https://doi.org/10.1007/s40615-020-00759-x

34.

Brown-Madan

Williams

Langston

. Addressing implicit bias and health disparities in a level IV NICU. J Perinatol 2023; 43(12): 1494–1496. https://doi.org/10.1038/s41372-023-01736-y

35.

Johnson

Guillory

Imaizumi

. Health care disparities in high-risk neonates. Clin Perinatol 2023; 50(1): 67–80. https://doi.org/10.1016/j.clp.2022.11.008

36.

Murosko

Peña

Lorch

. Racial and ethnic disparities in outcomes after NICU discharge: an equity-focused model. Semin Perinatol 2022; 46(8): 151659. https://doi.org/10.1016/j.semperi.2022.151659

37.

Kahn

Cheng

Mitchell

. A framework for pursuing child health equity in pediatric practice. Pediatr Clin North Am 2023; 70(4): 629–638. https://doi.org/10.1016/j.pcl.2023.03.002

The effect of prenatal intervention on postnatal decision-making for infants born with congenital diaphragmatic hernia

Abstract

Objective

Study Design

Results

Conclusions

Keywords

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References