Newborn Screening for Neurodevelopmental Disorders May Exacerbate Health Disparities.

Newborn screening (NBS) began in the early 1960s with screening for phenylketonuria on blood collected on filter paper. The number of conditions included in NBS programs expanded significantly with the adoption of tandem mass spectrometry. The recommended uniform screening panel provides national guidance and has reduced state variability. Universality and uniformity have been supported to promote equity. Recently, a number of researchers have suggested expanding NBS to include genomic sequencing to identify all genetic disorders in newborns. This has been specifically suggested for genes that increase the risk for neurodevelopmental disorders (NDDs), with the presumption that early identification in the newborn period would reduce disabilities. We offer arguments to show that genomic sequencing of newborns for NDDs risks exacerbating disparities. First, the diagnosis of NDD requires clinical expertise, and both genetic and neurodevelopmental expertise are in short supply, leading to disparities in access to timely follow-up. Second, therapies for children with NDDs are insufficient to meet their needs. Increasing early identification for those at risk who may never manifest developmental delays could shift limited resources to those children whose parents are more poised to advocate, worsening disparities in access to services. Rather, we suggest an alternative: genomic sequencing of all children with diagnosed NDDs. This focused strategy would have the potential to target genomic sequencing at children who manifest NDDs across diverse populations which could better improve our understanding of contributory genes to NDDs.

How Race, Ethnicity, and Social Determinants of Health Are Reported in Three European Pediatric Journals.

OBJECTIVES: To evaluate how geography, ethnicity, ancestry, and race or religion (GEAR) and social determinants of health (SDOH) data are reported and discussed in 3 European pediatric journals and to compare practices between European and American journals. STUDY DESIGN: A retrospective analysis of all original articles that enrolled children (<18 years old) published from January through June 2021 in 3 European pediatric journals: Archives of Disease in Childhood, European Journal of Pediatrics, and Acta Paediatrica. We categorized SDOH using the 5 domains as outlined by the US Healthy People 2030 framework. For each article, we recorded whether GEAR and SDOH were reported in the results and interpreted in the discussion sections. We then compared these European data by χ2 tests with data from 3 US pediatric journals. RESULTS: Of the 320 studied articles, 64 (20%) and 80 (25%) reported GEAR and SDOH data in the results sections, respectively. Of those articles, 32 (50%) and 53 (66.3%) studies interpreted the GEAR and SDOH data in their discussion sections, respectively. On average, articles reported factors from 1.2 GEAR and 1.9 SDOH categories with great variability in the variables collected and data groupings. Articles published in European journals were less likely to report GEAR and SDOH than articles published in US journals (P < .001 for both). CONCLUSIONS: Articles published in European pediatric journals did not commonly report either GEAR or SDOH, and there was wide variation in how data were collected and reported. Harmonization of categories will allow for more accurate interstudy comparisons.

The philosopher as partner: an introduction to the scholarship of Robert M. Veatch.

A diverse group of scholars reflect on the scholarship of Robert M. Veatch, the breadth of which is unmatched in modern day bioethics. Essays were written by both philosophers and clinician-philosophers, by contemporaries and mentees. They span the breadth of Bob's work and include analyses of his ideas about death, dying and organ transplantation, human experimentation and research ethics, disability, equality and justice, the doctor-patient relationship, the history of bioethics, as well as his pedagogical approach to teaching bioethics to clinicians across the health care spectrum. Recognition of Bob's influence in the modern field of bioethics and the challenges that persist are clearly identified.

The Use of Race, Ethnicity, and Social Determinants of Health in Three Pediatrics Journals.

OBJECTIVE: To evaluate how race, ethnicity, and social determinants of health (SDOH) are reported and discussed in 3 pediatrics journals. STUDY DESIGN: Bibliometric analysis of original articles that enrolled children as participants between January-June 2021 published in The Journal of Pediatrics, Pediatrics, and JAMA Pediatrics. We recorded in aggregate the inclusion of race, ethnicity, and SDOH data from the methods, results, and discussion sections of each article. We then used χ2 analyses and t tests to compare recording and use of race, ethnicity, and SDOH data on a number of factors. RESULTS: A total of 317 original articles were included with 200 (63.1%) conducted in the US. Researchers presented 116 unique race and ethnicity categories. US studies reported race significantly more frequently than international studies (166/200, 83.0% vs 29/117, 24.8% P < .001), yet only 24.7% (41/166) of US and 10.3% (3/29) of international studies that reported these data interpreted their significance and linked such to their study findings. US federal funding influenced reporting of race and ethnicity but not interpretation. Less than one-half of all studies reported SDOH (147/317, 46.4%), and very few that reported SDOH interpreted the data to study findings in both the US (18/106, 17.0%) and internationally (3/41, 7.3%). CONCLUSION: Race, ethnicity, and SDOH data are reported without consistent categories, and their significance is not often explained in both US and international articles. Researchers should be more intentional about how and why they collect, report, and interpret these data to help identify health disparities and highlight health inequities.

Gender and race/ethnicity differences in living kidney donor demographics: Preference or disparity?

In the United States, women are over-represented and Blacks are under-represented as living kidney donors. A traditional bioethics approach would state that as long as living donors believe that the benefits of participation outweigh the risks and harms (beneficence) and they give a voluntary and informed consent, then the demographics reflect a mere difference in preferences. Such an analysis, however, ignores the social, economic and cultural determinants as well as various forms of structural discrimination (e.g., racism, sexism) that may imply that the distribution is less voluntary than may appear initially. The distribution also raises justice concerns regarding the fair recruitment and selection of living donors. We examine the differences in living kidney donor demographics using a vulnerabilities analysis and argue that these gender and racial differences may not reflect mere preferences, but rather, serious justice concerns that need to be addressed at both the individual and systems level.

Reopening K-12 Schools in the Era of Coronavirus Disease 2019: Review of State-Level Guidance Addressing Equity Concerns.

OBJECTIVE: To determine how state guidance documents address equity concerns in K-12 schools during the coronavirus disease 2019 pandemic. STUDY DESIGN: Using online searches, we collected state-level documents from all 50 states and the District of Columbia discussing reopening plans for K-12 schools in the 2020-2021 academic year. We examined whether these documents explicitly mentioned equity as a concern, as well as if and how they addressed the following equity issues: food insecurity and child nutrition, homelessness or temporary housing, lack of access to Internet/technology, students with disabilities or special needs, English-language learners, students involved with or on the verge of involvement with the Department of Children and Family Services or an equivalent agency, mental health support, students/staff at greater risk of severe illness from severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2) infection, and students/staff living with someone at greater risk of severe illness from SARS-CoV-2 infection. RESULTS: Forty-four of 51 states (86%) explicitly mentioned equity as a concern or guiding principle. At least 90% of states offered guidance for 7 equity issues. Fewer than 75% of states addressed homelessness or temporary housing, students involved with or on the verge of involvement with Department of Children and Family Services or an equivalent agency, and students/staff living with someone at greater risk of severe illness from SARS-CoV-2 infection. CONCLUSIONS: Wide variability exists in state-level guidance to help K-12 schools develop reopening plans that protect those who are most vulnerable to learning loss or reduced access to basic needs. Interpretation and implementation by local educational agencies will need to be assessed.

Reflections on Charlie Gard and the Best Interests Standard From Both Sides of the Atlantic Ocean.

Charlie Gard (August 4, 2016, to July 28, 2017) was an infant in the United Kingdom who was diagnosed with an encephalopathic form of mitochondrial DNA depletion syndrome caused by a mutation in the RRM2B gene. Charlie's parents raised £1.3 million (∼$1.6 million US) on a crowdfunding platform to travel to New York to pursue experimental nucleoside bypass treatment, which was being used to treat a myopathic form of mitochondrial DNA depletion syndrome caused by mutations in a different gene (TK2). The case made international headlines about what was in Charlie's best interest. In the medical ethics community, it raised the question of whether best interest serves as a guidance principle (a principle that provides substantive directions as to how decisions are to be made), an intervention principle (a principle specifying the conditions under which third parties are to intervene), both guidance and intervention, or neither. I show that the United Kingdom uses best interest as both guidance and intervention, and the United States uses best interest for neither. This explains why the decision to withdraw the ventilator without attempting nucleoside bypass treatment was the correct decision in the United Kingdom and why the opposite conclusion would have been reached in the United States.

Health Care Surrogacy Laws Do Not Adequately Address the Needs of Minors.

A couple and their five-year-old daughter are in a car accident. The parents are not expected to survive. The child is transported to a children's hospital, and urgent treatment decisions must be made. Whom should the attending physician approach to make decisions for the child? When such cases arise in, for example, the hospitals where we work, the social worker or chaplain is instructed to use the Illinois Health Care Surrogacy Act as a guidepost to identify a decision-maker. But in our state and the country overall, the limitations of such statutes leave hospital workers to make a judgment call among friends, family, and clergy who may come forward. While surrogate decision-making statutes comprehensively address surrogate decision-makers for adults, a patchwork of laws-permanency statutes, kinship provider statutes, standby guardianship statutes, and, in some cases, surrogate decision-making statutes-provide variable decision-making pathways for children.

African-Americans with End Stage Renal Disease in the Early Years of Kidney Transplantation.

INTRODUCTION: The first successful kidney transplant in humans was performed in 1954. In the following 25 years, the biomedical, ethical, and social implications of kidney transplantation were widely discussed by both healthcare professionals and the public. Issues relating to race, however, were not commonly addressed, representing a "blind spot" regarding racial disparities in access and health outcomes. METHODS: Through primary sources in the medical literature and lay press, this paper explores the racial dynamics of kidney transplantation in the 1950-1970s in the United States as the procedure grew from an experimental procedure to the standard of care for patients in end-stage renal disease (ESRD). RESULTS & DISCUSSION: An extensive search of the medical literature found very few papers about ESRD, dialysis, or renal transplant that mentioned the race of the patients before 1975. While the search did not reveal whether race was explicitly used in determining patient access to dialysis or transplant, the scant data that exist show that African-Americans disproportionately developed ESRD and were underrepresented in these early treatment populations. Transplant outcome data in the United States failed to include race demographics until the late 1970s. The Social Security Act of 1972 (PL 92-603) extended Medicare coverage to almost all Americans with ESRD and led to a rapid increase in both dialysis and kidney transplantation for African-Americans in ESRD, but disparities persist today.

Respecting Choice in Definitions of Death.

The definition of death was clearer one hundred years ago than it is today. People were declared dead if diagnosed with permanent cessation of both cardio-circulatory function and respiratory function. But the definition has been muddled by the development of new technologies and interventions-first by cardiopulmonary resuscitation and ventilators, which were introduced in the mid-twentieth century, and now by extracorporeal membrane oxygenation, which creates the ability to keep oxygenated blood circulating, with or without a beating heart or functioning lungs. In Defining Death: The Case for Choice, Robert Veatch and I argue that the definition of death should focus on "what change in a human being is so fundamental that we can say the individual is no longer with us as a member of the human community bearing rights such as the right not to be killed." We assert that this decision is a normative issue about which different stakeholders may believe that different changes are fundamental, and we therefore propose that the optimal policy solution may be to allow stakeholders to choose their own definition within a reasonable range of options. There are three caveats that need to be highlighted regarding this approach.

The Role of Gender in Publication in The Journal of Pediatrics 2015-2016: Equal Reviews, Unequal Opportunities.

OBJECTIVES: To examine whether the gender of corresponding authors, reviewers, and editors led to differential publication recommendations and outcomes for original research articles and invited editorials submitted to The Journal of Pediatrics in 2015 and 2016. STUDY DESIGN: Names of corresponding authors, reviewers, editors, and editorial writers in The Journal of Pediatrics databases for 2015-2016 were analyzed to determine gender using computer algorithms and Internet searches. Reviewer recommendations and final editor dispositions were stratified by their gender and the gender of the corresponding authors. RESULTS: Of 3729 original manuscripts, 54.3% had female corresponding authors. Women were the associate editor (40.2% of submissions), guest editor (34.8%), or primary reviewer (37.4%), with no gender difference in editor or reviewer assignments for submissions by female vs male corresponding authors. There were no outcome differences by author gender for manuscripts overseen by female (P = .71) or male (P = .62) editors nor recommendation differences by female (P = .18) or male (P = .71) reviewers. Female editors had a lower acceptance rate overall than male editors (20.1% vs 25.6%; P < .001). Women were statistically less likely to accept and complete the invitation to peer review original articles (34.0%; 2295 of 6743) compared with men (40.0%; 3930 of 9823; P < .001). Women wrote 33 of 107 editorials (30.8%). CONCLUSIONS: There were no differences in reviewer recommendations or editor decisions for original research articles based on corresponding author gender. However, women had fewer opportunities to serve as peer reviewers and editorial writers than would be expected given their representation as academic pediatric faculty.

Ethical Analysis and Policy Recommendations Regarding Domino Liver Transplantation.

Due to the widening gap between supply and demand, patients who need a liver transplant due to metabolic disease may be asked to serve as domino liver donors-to have their native liver transplanted into another candidate. We here analyze the ethical problems surrounding informed consent for the implant and explant procedures in transplant candidates who will serve as domino donors, using the case of a child with maple syrup urine disease. We discuss the need for 2 distinct consent processes separated in time to ensure that potential domino donors (or their surrogates) give a truly voluntary consent. We propose a Domino Donor Advocate-based on the concept of the independent living donor advocate to help the patient and/or his or her surrogates consider the risks, benefits and alternatives. Finally, we evaluate the Organ Procurement and Transplantation Network policy regarding "therapeutic organ donation" and propose several modifications to ensure that the decision by the potential domino donor (and/or his or her surrogate) is voluntary and informed.

Impact of the kidney allocation system on young pediatric recipients.

The kidney allocation system (KAS) altered pediatric candidate prioritization. We determined KAS's impact on pediatric kidney recipients by examining delayed graft function (DGF) rates from 2010 to 2016. A propensity score-matched pediatric recipients pre- and post-KAS. A semiparametric decomposition analysis estimated the contributions of KAS-related changes in donor characteristics and dialysis time on DGF rate. The unadjusted odds of DGF were 69% higher post-KAS for young (<10 years at listing) recipients (N = 1153, P = .02) but were not significantly increased for older pediatric (10-17 years at listing) recipients (N = 2624, P = .48). Post-KAS, young recipients received significantly fewer pediatric (<18 years) donor kidneys (21% vs 32%, P < .01) and had longer median pretransplant dialysis time (603 vs 435 days, P < .01). After propensity score matching, post-KAS status increased the odds of DGF in young recipients 71% (OR 1.71, 95% CI 1.01-2.46). In decomposition analysis, 24% of the higher DGF rate post-KAS was attributable to donor characteristics and 19% to increased recipient dialysis time. In a confirmatory survival analysis, DGF was associated with a 2.2 times higher risk of graft failure (aHR2.28, 95% CI 1.46-3.54). In conclusion, KAS may lead to worse graft survival outcomes in children. Allocation changes should be considered.

The 1966 Ciba Symposium on Transplantation Ethics: 50 Years Later.

In March 1966, the Ciba Foundation sponsored the first international, interdisciplinary symposium focused on ethical and legal issues in transplantation. The attendees included not only physicians and surgeons but also judges and legal scholars, a minister, and a science journalist. In this article, we will consider some of the topics in organ transplantation that were discussed by the attendees, what we have learned in the intervening half century, and the relevance of their discussions today. Specifically, we examine the definition of death and its implications for organ procurement, whether it is ethical and legal to "maim" a living individual for the benefit of another, how to ensure that the consent of the living donor is voluntary and informed, the case of identical twins, the question of whether ethically minors can serve as living donors, the health risks of living donation, the ethics and legality of an organ market, and the economic barriers to living donation. We show that many of the concerns discussed at the Ciba symposium remain highly relevant, and their discussions have helped to shape the ethical boundaries of organ transplantation today.

Age and equity in liver transplantation: An organ allocation model.

A moral liver allocation policy must be fair. We considered a 2-step, 2-principle allocation system called "age mapping." Its first principle, equal opportunity, ensures that candidates of all ages have an equal chance of getting an organ. Its second principle, prudential lifespan equity, allocates younger donor grafts to younger candidates and older donors to older candidates in order to increase the likelihood that all recipients achieve a "full lifespan." Data from 2476 candidates and 1371 consecutive adult liver transplantations (from 1999 to 2012) were used to determine whether age mapping can reduce the gap in years of life lost (YLL) between younger and older recipients. A parametric Weibull prognostic model was developed to estimate total life expectancy after transplantation using survival of the general population matched by sex and age as a reference. Life expectancy from birth was calculated by adding age at transplant and total life expectancy after transplantation. In multivariate analysis, recipient age, hepatitis C virus status, Model for End-Stage Liver Disease score at transplant of >30, and donor age were significantly related to prognosis after surgery (P < 0.05). The mean (and standard deviation) number of years of life from birth, calculated from the current allocation model, for various age groups were: recipients 18-47 years (n = 340) = 65.2 (3.3); 48-55 years (n = 387) = 72.7 (2.1); 56-61 years (n = 372) = 74.7 (1.7) and for recipients >61 years (n = 272) = 77.4 (1.4). The total number of YLL equaled 523 years. Redistributing liver grafts, using an age mapping algorithm, reduces the lifespan gap between younger and older candidates by 33% (from 12.3% to 8.3%) and achieves a 14% overall reduction of YLL (73 years) compared to baseline liver distribution. In conclusion, deliberately incorporating age into an allocation algorithm promotes fairness and increases efficiency.

Racial disparities in reaching the renal transplant waitlist: is geography as important as race?

BACKGROUND: In the United States, African Americans and whites differ in access to the deceased donor renal transplant waitlist. The extent to which racial disparities in waitlisting differ between United Network for Organ Sharing (UNOS) regions is understudied. METHODS: The US Renal Data System (USRDS) was linked with US census data to examine time from dialysis initiation to waitlisting for whites (n = 188,410) and African Americans (n = 144,335) using Cox proportional hazards across 11 UNOS regions, adjusting for potentially confounding individual, neighborhood, and state characteristics. RESULTS: Likelihood of waitlisting varies significantly by UNOS region, overall and by race. Additionally, African Americans face significantly lower likelihood of waitlisting compared to whites in all but two regions (1 and 6). Overall, 39% of African Americans with ESRD reside in Regions 3 and 4--regions with a large racial disparity and where African Americans comprise a large proportion of the ESRD population. In these regions, the African American-white disparity is an important contributor to their overall regional disparity. CONCLUSIONS: Race remains an important factor in time to transplant waitlist in the United States. Race contributes to overall regional disparities; however, the importance of race varies by UNOS region.