Advance Care Planning and Parent-Reported End-of-Life Outcomes in the Neonatal Intensive Care Unit.

OBJECTIVE: This study aimed to evaluate the impact of advance care planning (ACP) on parent-reported end-of-life (EOL) outcomes in the neonatal intensive care unit (NICU). STUDY DESIGN: Single-center, cross-sectional mixed-methods survey study of bereaved parents who experienced the death of a child in the Boston Children's Hospital NICU between 2010 and 2021. Logistic regression, chi-square test, Fisher's exact test, and Wilcoxon rank-sum test were used to evaluate the relationship between ACP and parent-reported EOL outcomes. Qualitative content was analyzed through inductive coding. RESULTS: A total of 40/146 (27%) of eligible parents responded to our survey. There was a significant association between ACP and improved EOL care processes and parental satisfaction with communication. Parents with ACP were more likely to report goal-concordant care and higher levels of perceived shared decision-making. Qualitatively, emerging themes in parents' descriptions of goal-concordant care included misaligned expectations and communication. Emerging themes for parental preparedness included infant symptoms, logistical aspects, impact on parents, and degrees of preparedness. For decisional regret, the primary theme was reevaluating supports. CONCLUSION: ACP is associated with improved EOL care outcomes and parents qualitatively conceptualize goal-concordant care, preparedness for their child's death, and decisional regret in nuanced ways. Families should have the opportunity to participate in ACP discussions that meet their unique communication and decision-making needs. KEY POINTS: · ACP is associated with improved parent-reported end-of-life outcomes.. · Parents conceptualize end-of-life care outcomes in nuanced ways.. · Families should have the opportunity to participate in ACP discussions that meet their unique needs..

Parent Preferences and Experiences in Advance Care Planning in the Neonatal Intensive Care Unit.

OBJECTIVE: Our objective was to evaluate the preferences and experiences of bereaved parents around advance care planning (ACP) in the neonatal intensive care unit (NICU). STUDY DESIGN: Single-center cross-sectional survey of bereaved parents who experienced the death of a child in the Boston Children's Hospital NICU between 2010 and 2021 was carried out. Chi-square, Fisher's exact, Fisher Freeman Halton, and Wilcoxin rank sum tests were used to evaluate differences between parents who did and did not receive ACP. RESULTS: Out of eligible parents, 40 out of 146 (27%) responded to our survey. Most parents (31 out of 33, 94%) rated ACP as being very important and 27 out of 33 (82%) reported having ACP discussions during their child's admission. Parents preferred initial ACP discussions to occur early in their child's illness trajectory with members of the primary NICU team, with most parents' experiences aligning with these preferences. CONCLUSION: Parents value ACP discussions suggesting a further role for ACP in the NICU. KEY POINTS: · NICU parents value and participate in advance care planning discussions. · Parents prefer advance care planning with members of the primary NICU, specialty, and palliative care teams. · Parents prefer advance care planning early in their child's illness trajectory.

A Qualitative Study of Parental Perspectives on Prenatal Counseling at Extreme Prematurity.

OBJECTIVE: To determine parental preferred language, terminology, and approach after prenatal counseling for an anticipated extremely preterm delivery. STUDY DESIGN: Pregnant persons (and their partners) admitted at 220/7-256/7 weeks of estimated gestation participated in post antenatal-counseling semistructured interviews to explore preferred language and decision-making approaches of their antenatal counseling session. Interviews were audio-recorded and transcribed, and thematic analysis of the data was performed. RESULTS: Thirty-nine interviews were conducted representing 28 total prenatal consults. Analysis identified 2 overarching themes impacting the whole counseling experience: the need for reassurance and compassionate communication, while parents traveled along a dynamic decision-making journey they described as fluid and ever-changing. Related themes included the following: (1) Finding Balance: parents reported the importance of balancing positivity and negativity as well as tailoring the amount of information, (2) The Unspoken: parents described assumptions and inferences surrounding language, resuscitation options, and values that can cloud the counseling process, (3) Making the Intangible Tangible: parents reported the importance of varied communication strategies, for example, visuals to better anticipate and prepare, and (4) Team Synergism: Parents expressed desire for communication and consistency among and between teams, which increased trust. CONCLUSIONS: Parents facing extremely premature delivery generally did not report remembering specific terminology used during prenatal consultation but rather how the language and counseling approach made them feel and affected the decision-making process. These findings have implications for further research and educational intervention design to improve clinicians' counseling practices to better reflect parental preferences and ultimately improve counseling outcomes.

Deliveries at extreme prematurity: outcomes, approaches, institutional variation, and uncertainty.

PURPOSE OF REVIEW: Medical decision making and practices surrounding extremely premature birth remains challenging for parents and care providers alike. Recently, concerns have been raised regarding wide practice variation, selection bias, and the limitations of outcomes data in this population. The purpose of this review is to summarize the recent literature relevant to deliveries at extreme prematurity with a focus on outcomes, approaches, and institutional variation. RECENT FINDINGS: Newer data suggest that evidence-based clinical guidelines and protocols for both pregnant women and infants at extreme prematurity are emerging and may improve care and outcomes at lower gestational ages. It has also been recently shown that wide practice variation, selection bias, and methodological limitations of outcomes data reporting with respect to deliveries at extreme prematurity exist. SUMMARY: Counseling at extreme prematurity should prioritize eliciting parental values and preferences with the goal of personalized shared decision-making. Providers should recognize limitations in counseling families at extreme prematurity, including selection bias, institutional variation, outcomes inaccuracies, prognostic uncertainty, and implicit biases. Standardized reporting of perinatal outcomes should be developed to help alleviate current outcomes misrepresentations and improve counseling at extreme prematurity. Education for providers in advanced communication skills is needed when counseling at extreme prematurity.

Assessing Ethics Knowledge: Development of a Test of Ethics Knowledge in Neonatology.

OBJECTIVE: To develop and validate the Test of Ethics Knowledge in Neonatology (TEK-Neo) with good internal consistency reliability, item performance, and construct validity that reliably assesses interprofessional staff and trainee knowledge of neonatal ethics. STUDY DESIGN: We adapted a published test of ethics knowledge for use in neonatology. The novel instrument had 46 true/false questions distributed among 7 domains of neonatal ethics: ethical principles, professionalism, genetic testing, beginning of life/viability, end of life, informed permission/decision making, and research ethics. Content and correct answers were derived from published statements and guidelines. We administered the voluntary, anonymous test via e-mailed link to 103 participants, including medical students, neonatology fellows, neonatologists, neonatology nurses, and pediatric ethicists. After item reduction, we examined psychometric properties of the resulting 36-item test and assessed overall sample performance. RESULTS: The overall response rate was 27% (103 of 380). The test demonstrated good internal reliability (Cronbach α = 0.66), with a mean score of 28.5 ± 3.4 out of the maximum 36. Participants with formal ethics training performed better than those without (30.3 ± 2.9 vs 28.1 ± 3.5; P = .01). Performance improved significantly with higher levels of medical/ethical training among the 5 groups: medical students, 25.9 ± 3.7; neonatal nurses/practitioners, 27.7 ± 2.7; neonatologists, 28.8 ± 3.7; neonatology fellows, 29.8 ± 2.9; and clinical ethicists, 33.0 ± 1.9 (P < .0001). CONCLUSIONS: The TEK-Neo reliably assesses knowledge of neonatal ethics among interprofessional staff and trainees in neonatology. This novel tool discriminates between learners with different levels of expertise and can be used interprofessionally to assess individual and group performance, track milestone progression, and address curricular gaps in neonatal ethics.

Do-Not-Resuscitate Orders in the Neonatal ICU: Experiences and Beliefs Among Staff.

OBJECTIVES: Studies in adult patients have shown that do-not-resuscitate orders are often associated with decreased medical intervention. In neonatology, this phenomenon has not been investigated, and how do-not-resuscitate orders potentially affect clinical care is unknown. DESIGN: Retrospective medical record data review and staff survey responses about neonatal ICU do-not-resuscitate orders. SETTING: Four academic neonatal ICUs. SUBJECTS: Clinical staff members working in each neonatal ICU. INTERVENTIONS: Survey response collection and analysis. MEASUREMENTS AND MAIN RESULTS: Participating neonatal ICUs had 14-48 beds and 120-870 admissions/yr. Frequency range of do-not-resuscitate orders was 3-11 per year. Two-hundred fifty-seven surveys were completed (46% response). Fifty-nine percent of respondents were nurses; 20% were physicians. Over the 5-year period, 44% and 17% had discussed a do-not-resuscitate order one to five times and greater than or equal to 6 times, respectively. Fifty-seven percent and 22% had cared for one to five and greater than or equal to 6 patients with do-not-resuscitate orders, respectively. Neonatologists, trainees, and nurse practitioners were more likely to report receiving training in discussing do-not-resuscitate orders or caring for such patients compared with registered nurses and respiratory therapists (p < 0.001). Forty-one percent of respondents reported caring for an infant in whom interventions had been withheld after a do-not-resuscitate order had been placed without discussing the specific withholding with the family. Twenty-seven percent had taken care of an infant in whom interventions had been withdrawn under the same circumstances. Participants with previous experiences withholding or withdrawing interventions were more likely to agree that these actions are appropriate (p < 0.001). CONCLUSIONS: Most neonatal ICU staff report experience with do-not-resuscitate orders; however, many, particularly nurses and respiratory therapists, report no training in this area. Variable beliefs with respect to withholding and withdrawing care for patients with do-not-resuscitate orders exist among staff. Because neonatal ICU patients with do-not-resuscitate orders may ultimately survive, withholding or withdrawing interventions may have long-lasting effects, which may or may not coincide with familial intentions.

Counselling variation among physicians regarding intestinal transplant for short bowel syndrome.

BACKGROUND: Intestinal transplant in infants with severe short bowel syndrome (SBS) is an emerging therapy, yet without sufficient long-term data or established guidelines, resulting in possible variation in practice. OBJECTIVES: To assess current attitudes and counselling practices among physicians regarding intestinal transplant in infants with SBS, and to determine whether counselling and management vary between subspecialists or centres. METHODS: A national sample of practicing paediatric surgeons and neonatologists was surveyed via the American Academy of Paediatrics listserves. Results were analysed by physician subspecialty and again by presence or absence of intestinal transplant at respondent's centre. RESULTS: The survey was completed by 433 respondents, consisting of 363 neonatologists and 70 paediatric surgeons. Fifty-seven respondents (13.2%) practiced at a centre that performed intestinal transplants in children. The vast majority of respondents (91% for preterm, 95% for term neonates) felt that maintaining a neonate with SBS on total parenteral nutrition for intestinal transplant was ethically optional (neither impermissible nor obligatory), and that parents should be given an informed choice whether to pursue that option. However, only 33% indicated they often/always offer intestinal transplant as a treatment option to families in this situation. CONCLUSIONS: There is a marked disparity between individual physicians' beliefs regarding the acceptability of intestinal transplant for severe SBS and their reported practice. Wide variability exists among physicians with respect to their knowledge, beliefs and practice regarding severe SBS, raising concerns about transparency and justice. Survival data prior to transplant, currently unavailable, are essential to rational decision making and informed parental permission.

Lowering the Age of Consent for Vaccination to Promote Pediatric Vaccination: It's Worth a Shot.

This paper challenges historically preconceived notions surrounding a minor's ability to make medical decisions, arguing that federal health law should be reformed to allow minors with capacity as young as age 12 to consent to their own Centers for Diseases Control and Prevention (CDC)-approved COVID-19 vaccinations. This proposal aligns with and expands upon current exceptions to limitations on adolescent decision-making. This analysis reviews the historic and current anti-vaccination sentiment, examines legal precedence and rationale, outlines supporting ethical arguments regarding adolescent decision-making, and offers rebuttals to anticipated ethical counterarguments.

How Can Newborn Toxicology Testing Be More Equitable? An Interactive Ethics Workshop.

Introduction: Practice variation in newborn toxicology testing during the birth hospitalization exists across institutions and legal jurisdictions. While testing can provide benefits, indiscriminate testing has been shown to perpetuate health care inequities. In the backdrop of an opioid epidemic and a charged medicolegal landscape, this workshop guides participants to reexamine newborn toxicology testing through a shared ethical lens. Methods: We conducted a live, 90-minute workshop in English at an international pediatric conference. Physicians, residents, and fellows participated in large- and small-group breakout sessions to learn relevant clinical and bioethical frameworks, share their own local context and expertise, and explore ethical applications through case-based discussions. We administered two anonymous online follow-up surveys to assess self-perceived impact on participant knowledge, behavior, and clinical practice. Results: Seven facilitators and 45 individuals participated in the workshop. Eighteen participants completed survey 1 immediately following workshop conclusion, and six participants completed survey 2 after 3 months had elapsed. Immediately following the workshop, 94% of respondents reported that they had been introduced to a new idea, and 82% were considering practice change. A low response rate to survey 2 limited interpretation, but some respondents reported self-perceived change following workshop attendance. Discussion: This workshop facilitated conversation between physician participants on a complex pediatric health care inequity issue using an ethical framework.

Gestational Carrier Pregnancies: Legal and Ethical Considerations for Pediatricians.

This case explores the legal and ethical considerations for pediatricians surrounding gestational carrier pregnancies in the United States. Because of high success rates for assisted reproduction, state laws supporting same-sex adoption and surrogacy, and established legal precedents, gestational carrier pregnancies are increasingly common. The case presented involves a gestational carrier in preterm labor at 30 weeks' gestation with malpositioned twins who declines a cesarean delivery. Three commentaries are presented. The first highlights the importance of understanding the ethical implications of gestational carrier pregnancies in prenatal counseling. The second commentary emphasizes the pregnant person's right to autonomy and bodily integrity, and discusses considerations in surrogacy pregnancies, including the authority to authorize a cesarean delivery, valid informed consent, and decision-making for neonates. The third commentary discusses autonomy, emphasizing the importance of contracts in surrogacy pregnancies, and suggests that, in the case of a conflict between the gestational carrier and the intended parent(s), the gestational carrier's preference should be decisive regarding medical care during pregnancy. These discussions highlight key concepts for ethically informed and family-centered care in gestational carrier pregnancies and deliveries.

Advice to Clinicians From Expectant Parents at Extreme Prematurity: A Multimethod Study.

BACKGROUND AND OBJECTIVES: Despite recommendations for patient-centered counseling on extreme prematurity, clinicians often miss opportunities to communicate in a way that facilitates parental knowledge, decision-making, and emotional support. In this study, we aimed to determine empirical, parent-derived recommendations and advice for clinicians counseling on extreme prematurity. METHODS: Pregnant women (and their partners) admitted at 22 0/7 to 25 6/7 weeks' estimated gestation participated in postantenatal counseling semi-structured interviews or questionnaires to explore parental preferences in the counseling process, including advice to clinicians. Thematic analysis was performed. RESULTS: A total of 39 interviews and 47 questionnaires, representing 62 total prenatal consultations, were completed. Thematic analysis of participants' advice to clinicians from both interview and questionnaire data resulted in 14 parent-derived recommendations to clinicians who counsel expectant parents at extreme prematurity. Parental recommendations related to compassionately engaging, supporting, and communicating with families, as well as aligning teams and following up. CONCLUSIONS: We present an empirical parent-derived, family-centered, and practical approach for clinicians counseling on extreme prematurity. Future studies should include a more diverse patient population and assess the impact of these recommendations on the counseling process and outcomes.

Collaboration between Maternal-Fetal Medicine and Neonatology When Counseling at Extreme Prematurity.

Enhanced communication between maternal-fetal medicine (MFM)/obstetrics and neonatology regarding counseling at extreme prematurity remains an essential element of prenatal consultations. Together, the obstetrician and neonatologist can collaborate to provide timely and synergistic information to affected couples during a dynamic period, combining their expertise to elucidate values and formulate a plan that best supports the pregnant person and partner's goals. Such collaboration can help resolve differing perspectives between specialties, minimize redundancy and inconsistencies, and mitigate the impact of clinician bias. Best practices for joint-specialty collaboration include a precounseling clinician huddle, contemporaneous counseling by MFM specialists/obstetricians and neonatologists with the expectant parents or individualized sequential counseling if preferred by the couple, and a postcounseling clinician debrief. This approach can help establish a trusting relationship with families facing possible extremely preterm delivery and optimize the overall counseling experience. Future efforts focused on education and research, including a standardized approach to educational curricula among fellowship programs, should be emphasized.

Vitelline vascular remnant causing intestinal obstruction in a patient with TARP syndrome.

BACKGROUND: TARP syndrome, characterized by talipes equinovarus, atrial septal defect, Robin sequence, and persistent left superior vena cava, is an X-linked recessive condition caused by deleterious variants in RBM10. Vitelline vascular remnants (VVR) are a rare vitelline duct anomaly with approximately 26 cases previously reported. There are no previously reported cases of VVRs in patients with TARP syndrome. CASE: We present a male neonate diagnosed with TARP syndrome via trio whole exome sequencing who had classic features of this syndrome, although his course was additionally complicated by feeding intolerance with multiple episodes of abdominal distension. Serial imaging and contrast studies of the upper GI tract and small bowel demonstrated small bowel obstruction of unclear etiology. Given the poor prognosis associated with this condition, life-sustaining measures were withdrawn, and he passed away at 38 days of age. On autopsy, a VVR was unexpectedly identified with proximal bowel dilation, explaining his feeding intolerance. CONCLUSIONS: We highlight the importance of full post-mortem examination in understanding the complete spectrum of manifestations of genetic syndromes and provide a review of the literature.

Perinatal-lethal nonimmune fetal hydrops attributed to MECOM-associated bone marrow failure.

Pathogenic variants in MECOM, a gene critical to the self-renewal and proliferation of hematopoietic stem cells, are known to cause a rare bone marrow failure syndrome associated with amegakaryocytic thrombocytopenia and bilateral radioulnar synostosis known as RUSAT2. However, the spectrum of disease seen with causal variants in MECOM is broad, ranging from mildly affected adults to fetal loss. We report two cases of infants born preterm who presented at birth with symptoms of bone marrow failure including severe anemia, hydrops, and petechial hemorrhages; radioulnar synostosis was not observed in either patient, and, unfortunately, neither infant survived. In both cases, genomic sequencing revealed de novo variants in MECOM considered to be responsible for their severe presentations. These cases add to the growing body of literature that describe MECOM-associated disease, particularly MECOM as a cause of fetal hydrops due to bone marrow failure in utero. Furthermore, they support the use of a broad sequencing approach for perinatal diagnosis, as MECOM is absent from available targeted gene panels for hydrops, and highlight the importance of postmortem genomic investigation.

Decision making at extreme prematurity: Innovation in clinician education.

Decision-making at extreme prematurity remains ethically and practically challenging and can result in parental and clinician distress. It is vital that clinicians learn the necessary skills integral to counseling and decision-making with families in these situations. A pedagogical approach to teaching counseling should incorporate adult learning theory, emphasize multidisciplinary team in-situ simulation that links to counseling clinicians' daily practice, and includes critical reflection, debriefing, and program assessment. Multiple educational strategies that train clinicians in advanced communication and decision-making offer promising results to optimize antenatal counseling and shared decision-making for families facing possible delivery at extreme prematurity. Continued process evaluation and innovation in these educational domains are needed while also assessing the effect on patient-centered outcomes.

Infant mode of death in the neonatal intensive care unit: A systematic scoping review.

OBJECTIVE: To characterize literature that describes infant mode of death and to clarify how limitation of life-sustaining treatment (LST) is defined and rationalized. STUDY DESIGN: Eligible studies were peer-reviewed, English-language, and included number of infant deaths by mode out of all infant deaths in the NICU and/or delivery room. RESULT: 58 included studies were primarily published in the last two decades from North American and European centers. There was variation in rates of infant mode of death by study, with some showing an increase in deaths following limitation of LST over time. Limitation of LST was defined by the intervention withheld/withdrawn, the relationship between the two practices, and prior frameworks. Themes for limiting LST included diagnoses, low predicted survival and/or quality of life, futility, and suffering. CONCLUSION: Limitation of LST is a common infant mode of death, although rates, study definitions, and clinical rationale for this practice are variable.

Moral equivalence theory in neonatology.

This article explores the ethical concept of "the equivalence thesis" (ET), or the idea that withdrawing and withholding life sustaining treatments are morally equivalent practices, within neonatology. We review the historical origins, theory, and clinical rationale behind ET, and provide an analysis of how ET relates to literature that describes neonatal mode of death and healthcare professional and parent attitudes towards end-of-life care. While ET may serve as an ethical tool to optimize resource allocation in theory, its clinical utility is limited given the complexity of end-of-life care decisions.

The Case for Advance Care Planning in the NICU.

Many NICUs have reached an inflection point where infant deaths following limitation of life-sustaining treatments outnumber those following unsuccessful resuscitations, and many infants who survive continue to require intensive supports. Families of such infants with serious illness may benefit from a standardized, process-oriented approach in decisional-support. Advance care planning (ACP), or communication that supports patients, or their surrogate decision-makers, in sharing values, goals, and preferences for future medical care, is recognized as a valuable strategy in supporting adults with serious and chronic illness. Although the role of ACP in older children and adolescents is evolving, its utility has not been systematically explored in the perinatal context. This article builds a case for formalizing a role for ACP in the NICU by defining ACP and appraising recent outcomes research, reviewing the current state of parental needs for decisional support and serious illness in the NICU, and describing how ACP may address current limitations in primary and specialty pediatric palliative care and challenges for decision-making in the newborn period.