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.

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.

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.

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.

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..

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.

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.

Pediatric Shared Decision-Making for Simple and Complex Decisions: Findings From a Delphi Panel.

OBJECTIVE: To develop recommendations for pediatric shared decision-making (SDM). METHODS: We conducted a Delphi method study from 2020 to 2021 with an international panel (n = 21) of clinicians, researchers, and parents with expertise in pediatric SDM. We conducted semistructured interviews to identify the key processes of pediatric SDM. We coded the interviews using content analysis and developed a questionnaire on the potential processes of pediatric SDM. Using a Likert scale, panelists evaluated each process twice, once for simple decisions and once for complex decisions. Panelists were provided with a summary of the results and evaluated each process again. The processes that were agreed on for simple and complex decisions were reported as "fundamental processes." The processes that were agreed on for complex decisions were reported as "additional processes." RESULTS: A total of 79 recommendations were developed, including 29 fundamental processes and 14 additional processes for complex decisions. A recurring theme was the importance of personalizing the decision-making process. For example, the panel recommended that physicians should assess the family and child's desired roles in the decision-making process, assess their desired level of directiveness, and elicit and clarify their values, preferences, and goals. The panel also disagreed on several subprocesses, such as how to determine the child's role and the appropriate level of directiveness. CONCLUSIONS: An international expert panel developed recommendations for pediatric SDM for both simple and complex decisions. The recommendations highlight the importance of personalizing the decision-making process.

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.

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.

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.

Critical decision-making in neonatology and pediatrics: the I-P-O framework.

Critical decision-making in neonatology and other areas of pediatrics often carries with it a complex and difficult ethical component. For any treatment under consideration, the impermissible-permissible-obligatory (I-P-O) spectrum provides a useful framework for determining how to proceed. Any proposed treatment can be located along this spectrum, and identified as either ethically impermissible, permissible, or obligatory. Treatments determined to be ethically impermissible should not be made available by physicians. Those deemed ethically permissible should be explained to parents, commonly with a specific recommendation. Informed parents should then be free to choose from among permissible options. Potential treatments deemed ethically obligatory should be provided to the patient, even in the face of parental objection. The fundamental ethical work in neonatology and pediatrics is determining where on the I-P-O spectrum a treatment under consideration should be located. This should be determined by the prognosis for the patient with and without the treatment, the feasibility of providing the treatment, and consideration of all relevant rights and obligations. Location on the line is dynamic, and clinicians should be open to movement of a given treatment along the spectrum as new information, particularly regarding effectiveness, toxicity, and/or alternatives, becomes available. This framework provides a structure for ethical conversation and decision-making related to a specific patient, as well as in the formation of institutional and national guidelines.

Gene and Stem Cell Therapies for Fetal Care: A Review.

Importance: Gene and stem cell therapies have become viable therapeutic options for many postnatal disorders. For select conditions, prenatal application would provide improved outcomes. The fetal state allows for several theoretical advantages over postnatal therapy, including immune immaturity and cellular niche accessibility. Observations: Advances in prenatal diagnostic accuracy and surgical precision, as well as improvements in stem cell and gene therapy methods, have made prenatal gene and stem cell therapy realistic. Studies in mouse models and early human trials demonstrate the feasibility of these approaches. Additional efforts are under way to streamline fetal applications of stem cell and gene therapy while carefully considering best ethical practice and following established regulatory pathways. Conclusions and Relevance: Fetal stem cell and gene therapy bring important therapeutic opportunities for select disorders that present in the fetal and neonatal periods. While this field is in its infancy, these therapies are starting to be available clinically, and clinicians should be aware of their benefits and challenges.

Historical Perspectives: Shared Decision Making in the NICU.

The ethical dilemmas and predominant frameworks surrounding decision making for critically ill newborns have evolved substantially over the last 40 years. A shared decision-making approach is now favored, involving an exchange of information between parents and clinicians that emphasizes parental values and preferences, resulting in a personalized approach to decision making. In this review, we summarize the history of clinical decision making with a focus on the NICU, highlight different models of decision making, describe the advantages and current limitations of shared decision making, and discuss the ongoing and future challenges of decision making in the NICU amidst medical innovations and emerging technologies.

Variable management strategies for NEC totalis: a national survey.

BACKGROUND/OBJECTIVES: Necrotizing enterocolitis (NEC) is a serious disease linked to prematurity. A variant, NEC totalis, is associated with nearly 100% mortality. There is wide variation in counseling practices for NEC totalis. Our objectives are to determine what treatment options, if any, are offered to families, and which factors influence these decisions. METHODS: An anonymous survey was distributed to members of the AAP Sections on Neonatal-Perinatal Medicine and Pediatric Surgery. Data were analyzed utilizing chi-square tests and Spearman correlations, where applicable. RESULTS: In the setting of NEC totalis, 90% of the 378 respondents viewed offering life-sustaining interventions (LSI) as ethically permissible and 87% felt that transfer to another center willing to provide LSI should be considered; however, only 43% reported offering LSI to families. CONCLUSIONS: Management of NEC totalis remains challenging and significant practice variability persists. Most respondents do not offer ongoing medical/surgical management, despite believing it is an ethically permissible option.