A Narrative Review of the Association between Prematurity and Attention-Deficit/Hyperactivity Disorder and Accompanying Inequities across the Life-Course.

Preterm birth is associated with an increased risk of neurodevelopmental and neurobehavioral impairments including attention-deficit/hyperactivity disorder (ADHD), the most common neurobehavioral disorder of childhood. In this narrative review, we examine the known associations between prematurity and ADHD and highlight the impact of both prematurity and ADHD on multiple domains across the pediatric life-course. We develop a framework for understanding the health services journey of individuals with ADHD to access appropriate services and treatments for ADHD, the "ADHD Care Cascade". We then discuss the many racial and ethnic inequities that affect the risk of preterm birth as well as the steps along the "ADHD Care Cascade". By using a life-course approach, we highlight the ways in which inequities are layered over time to magnify the neurodevelopmental impact of preterm birth on the most vulnerable children across the life-course.

Advancing Understanding of Inequities in Rare Disease Genomics.

PURPOSE: Advances in genomic research have facilitated rare disease diagnosis for thousands of individuals. Unfortunately, the benefits of advanced genetic diagnostic technology are not distributed equitably among the population, as has been seen in many other health care contexts. Quantifying and describing inequities in genetic diagnostic yield is inherently challenging due to barriers to both clinical and research genetic testing. We therefore present an implementation protocol developed to expand access to our rare disease genomic research study and to further understand existing inequities. METHODS AND FINDINGS: The Rare Genomes Project (RGP) at the Broad Institute of MIT and Harvard offers research genome sequencing to individuals with rare disease who remain genetically undiagnosed through direct interaction with the individual or family. This presents an opportunity for diagnosis beyond the clinical context, thus eliminating many barriers to access. An initial goal of RGP was to equalize access to genomic sequencing by decoupling testing access from proximity to a major medical center and physician referral. However, study participants over the initial 3 years of this project were predominantly white and well resourced. To further understand and address the lack of diversity within RGP, we developed a novel protocol embedded within the larger RGP study, in an approach informed by an implementation science framework. The aims of this protocol were: (1) to diversify recruitment and enrollment within RGP; (2) understand the process and context of implementing genomic medicine for rare disease diagnosis; and (3) investigate the value of a diagnosis for underserved populations. IMPLICATIONS: Improved understanding of existing inequities and potential strategies to address them are needed to advance equity in rare disease genetic diagnosis and research. In addition to the moral imperative of equity in genomic medicine, this approach is critical in order to fully understand the genomic underpinnings of rare disease.

Advancing Understanding of Inequities in Rare Disease Genomics.

Purpose: Advances in genomic research have led to the diagnosis of rare, early-onset diseases for thousands of individuals. Unfortunately, the benefits of advanced genetic diagnostic technology are not distributed equitably among the population, as has been seen in many other healthcare contexts. Even quantifying and describing inequities in genetic diagnostic yield is challenging due to variation in referrals to clinical genetics practices and other barriers to clinical genetic testing. Methods: The Rare Genomes Project (RGP) at the Broad Institute of MIT and Harvard offers research genome sequencing to individuals with rare disease who remain genetically undiagnosed through direct interaction with the individual or family. This presents an opportunity for diagnosis beyond the clinical context, thus eliminating many barriers to access. Findings: An initial goal of RGP was to equalize access to genomic sequencing by decoupling testing access from proximity to a major medical center and physician referral. However, our study participants are overwhelmingly non-disadvantaged, as evidenced by their access to specialist care and genetic testing prior to RGP enrollment, and are also predominantly white. Implications: We therefore describe our novel initiative to diversify RGP enrollment in order to advance equity in rare disease genetic diagnosis and research. In addition to the moral imperative of medical equity, this is also critical in order to fully understand the genomic underpinnings of rare disease. We utilize a mixed methods approach to understand the priorities and values of underrepresented communities, existing disparities, and the obstacles to addressing them: all of which is necessary to promote equity in future genomic medicine initiatives.

Racial Inequity in High-Risk Infant Follow-Up Among Extremely Low Birth Weight Infants.

BACKGROUND AND OBJECTIVES: High-risk infant follow-up programs (HRIFs) are a recommended standard of care for all extremely low birth weight (ELBW) infants to help mitigate known risks to long-term health and development. However, participation is variable, with known racial and ethnic inequities, though hospital-level drivers of inequity remain unknown. We conducted a study using a large, multicenter cohort of ELBW infants to explore within- and between-hospital inequities in HRIF participation. METHODS: Vermont Oxford Network collected data on 19 503 ELBW infants born between 2006 and 2017 at 58 US hospitals participating in the ELBW Follow-up Project. Primary outcome was evaluation in HRIF at 18 to 24 months' corrected age. The primary predictor was infant race and ethnicity, defined as maternal race (non-Hispanic white, non-Hispanic Black, Hispanic, Asian American, Native American, other). We used generalized linear mixed models to test within- and between-hospital variation and inequities in HRIF participation. RESULTS: Among the 19 503 infants, 44.7% (interquartile range 31.1-63.3) were seen in HRIF. Twenty six percent of the total variation in HRIF participation rates was due to between-hospital variation. In adjusted models, Black infants had significantly lower odds of HRIF participation compared with white infants (adjusted odds ratio, 0.73; 95% confidence interval, 0.64-0.83). The within-hospital effect of race varied significantly between hospitals. CONCLUSIONS: There are significant racial inequities in HRIF participation, with notable variation within and between hospitals. Further study is needed to identify potential hospital-level targets for interventions to reduce this inequity.

Defining an Infant's Race and Ethnicity: A Systematic Review.

CONTEXT: Infant race and ethnicity are used ubiquitously in research and reporting, though inconsistent approaches to data collection and definitions yield variable results. The consistency of these data has an impact on reported findings and outcomes. OBJECTIVE: To systematically review and examine concordance among differing race and ethnicity data collection techniques presented in perinatal health care literature. DATA SOURCES: PubMed, CINAHL, and Ovid were searched on June 17, 2021. STUDY SELECTION: English language articles published between 1980 and 2021 were included if they reported on the United States' infant population and compared 2 or more methods of capturing race and/or ethnicity. DATA EXTRACTION: Two authors independently evaluated articles for inclusion and quality, with disagreements resolved by a third reviewer. RESULTS: Our initial search identified 4329 unique citations. Forty articles passed title/abstract review and were reviewed in full text. Nineteen were considered relevant and assessed for quality and bias, from which 12 studies were ultimately included. Discordance in infant race and ethnicity data were common among multiple data collection methods, including those frequently used in perinatal health outcomes research. Infants of color and those born to racially and/or ethnically discordant parents were the most likely to be misclassified across data sources. LIMITATIONS: Studies were heterogeneous in methodology and populations of study and data could not be compiled for analysis. CONCLUSIONS: Racial and ethnic misclassification of infants leads to inaccurate measurement and reporting of infant morbidity and mortality, often underestimating burden in minoritized populations while overestimating it in the non-Hispanic/Latinx white population.

A mixed methods study of perceptions of bias among neonatal intensive care unit staff.

BACKGROUND: Inequity in neonatology may be potentiated within neonatal intensive care units (NICUs) by the effects of bias. Addressing bias can lead to improved, more equitable care. Understanding perceptions of bias can inform targeted interventions to reduce the impact of bias. We conducted a mixed methods study to characterize the perceptions of bias among NICU staff. METHODS: Surveys were distributed to all staff (N = 245) in a single academic Level IV NICU. Respondents rated the impact of bias on their own and others' behaviors on 5-point Likert scales and answered one open-ended question. Kruskal-Wallis test (KWT) and Levene's test were used for quantitative analysis and thematic analysis was used for qualitative analysis. RESULTS: We received 178 responses. More respondents agreed that bias had a greater impact on others' vs. their own behaviors (KWT p < 0.05). Respondents agreed that behaviors were influenced more by implicit than explicit biases (KWT p < 0.05). Qualitative analysis resulted in nine unique themes. CONCLUSIONS: Staff perceive a high impact of bias across different domains with increased perceived impact of implicit vs. explicit bias. Staff perceive a greater impact of others' biases vs. their own. Mixed methods studies can help identify unique, unit-responsive approaches to reduce bias. IMPACT: Healthcare staff have awareness of bias and its impact on their behaviors with patients, families, and staff. Healthcare staff believe that implicit bias impacts their behaviors more than explicit bias, and that they have less bias than others. Healthcare staff have ideas for strategies and approaches to mitigate the impact of bias. Mixed method studies are effective ways of understanding environment-specific perceptions of bias, and contextual assets and barriers when creating interventions to reduce bias and improve equity. Generating interventions to reduce the impact of bias in healthcare requires a context-specific understanding of perceptions of bias among staff.

Rare diseases, common barriers: disparities in pediatric clinical genetics outcomes.

BACKGROUND: Identifying a precise genetic diagnosis can improve outcomes for individuals with rare disease, though the resources required to do so may impede access and exacerbate healthcare disparities leading to inequitable care. Our objective was therefore to determine the effect of multiple sociodemographic factors on the yield of the diagnostic evaluation for genetics outpatients. METHODS: This is a retrospective cohort study from 2017 to 2019 of outpatient genetics referrals at a pediatric academic tertiary care center. Exposures included: primary language, insurance type, and neighborhood resources (via the Childhood Opportunity Index, COI). The primary outcome was identification of a genetic diagnosis within 2 years of the initial clinic visit. RESULTS: COI quintile was not significantly associated with the odds of diagnosis but was significantly associated with clinic attendance, with lower neighborhood resources leading to incomplete referrals. Limited English proficiency was associated with a higher odds of diagnosis, though at an older age. Public insurance was associated with increased access to genetic testing. CONCLUSIONS: Lower neighborhood resources are negatively associated with clinic attendance. Our findings further suggest delays in care and a referral bias for more severe phenotypes among families with limited English proficiency. Improved access to clinical genetics is needed to improve diagnostic equity. IMPACT: The resources required to identify a genetic diagnosis may impede access and exacerbate healthcare disparities leading to inequitable care. In an analysis of pediatric outpatient genetics referrals, we observed a significant association between neighborhood resources and clinic attendance but not diagnostic yield for those attending, and a higher diagnostic yield for families with limited English proficiency, suggesting referral bias for more severe phenotypes. Thus, the primary barrier to finding a genetic diagnosis was initiation of care, not the ensuing diagnostic odyssey. Further research efforts should be directed at increasing access to clinical genetics evaluations for children with rare disease.

Implications of racial/ethnic perinatal health inequities on long-term neurodevelopmental outcomes and health services utilization.

Infants born preterm and with low birth weight have increased risk for neurodevelopmental challenges later in life compared to term-born peers. These include functional motor impairment, cognitive and speech delays, neurobehavioral disorders, and atypical social development. There are well-documented inequities in the population distributions of preterm birth and associated short-term morbidities by race, ethnicity, language, and nativity. Far less is known about how these inequities affect long-term outcomes, though the impact of unequal access to post-discharge support services for preterm infants raises concerns about widening gaps in health, development, and functioning. In this review, we describe what is currently known about the impact of race, ethnicity, nativity, and language on long-term outcomes. We provide a framework for understanding inequities in social, political, and historical context. And we offer guidance for next steps to delineate mechanistic pathways and to identify interventions to eliminate inequities in long-term neurodevelopmental outcomes through research, intervention, and advocacy.

Plugging the Leaky Pipeline: The Role of Peer Mentorship for Increasing Diversity.

I arrived to my shift early, nervous about caring for critically ill patients as a first-year fellow. I sat in the workroom alone, paralyzed, not sure how to preround despite being months into fellowship. The senior fellow appeared minutes before sign-out; fresh, knowledgeable, and calm, despite her busy night and lack of sleep. She asked me how I was doing. With tears in my eyes, my emotions poured out. I explained that I felt lost, unsure of myself, my place, and my knowledge. For the first time, I confessed out loud, "I don't think I'm supposed to be here. I have no idea what I'm doing." I could trust her in a way I couldn't trust others. She was like me: othered by her identity, minoritized by society. Though different from my own, her identity allowed her to understand my own experiences. We were different from one another and we were also the same. Unlike the senior faculty, it was safe to talk to her. And, unlike my other cofellows, there was a kinship between us in our otherness. She looked at me, closed the door, and shared words of strength that I needed to hear. I belonged. I was more than enough. She shared that the pressure I was experiencing was common among systematically minoritized individuals; she too had felt it before. Sitting with her, I was finally seen, supported, and comforted. As a peer mentor from a minoritized background, she provided a sense of security and belonging that had not been provided in my training and was distinct from the support of senior faculty.

Addressing bias and knowledge gaps regarding race and ethnicity in neonatology manuscript review.

A recent shift in public attention to racism, racial disparities, and health equity have resulted in an abundance of calls for relevant papers and publications in academic journals. Peer-review for such articles may be susceptible to bias, as subject matter expertise in the evaluation of social constructs, like race, is variable. From the perspective of researchers focused on neonatal health equity, we share our positive and negative experiences in peer-review, provide relevant publicly available data regarding addressing bias in peer-review from 12 neonatology-focused journals, and give recommendations to address bias and knowledge gaps in the peer review process of health equity research.

Effect of a Novel Mindfulness Curriculum on Burnout During Pediatric Internship: A Cluster Randomized Clinical Trial.

IMPORTANCE: Mindfulness curricula can improve physician burnout, but implementation during residency presents challenges. OBJECTIVE: To examine whether a novel mindfulness curriculum implemented in the first 6 months of internship reduces burnout. DESIGN, SETTING, AND PARTICIPANTS: This pragmatic, multicenter, stratified cluster randomized clinical trial of a mindfulness curriculum randomized 340 pediatric interns to the intervention or control arm within program pairs generated based on program size and region. Fifteen US pediatric training programs participated from June 14, 2017, to February 28, 2019. INTERVENTIONS: The intervention included 7 hour-long sessions of a monthly mindfulness curriculum (Mindfulness Intervention for New Interns) and a monthly mindfulness refresher implemented during internship. The active control arm included monthly 1-hour social lunches. MAIN OUTCOMES AND MEASURES: The primary outcome was emotional exhaustion (EE) as measured by the Maslach Burnout Inventory 9-question EE subscale (range, 7-63; higher scores correspond to greater perceived burnout). Secondary outcomes were depersonalization, personal accomplishment, and burnout. The study assessed mindfulness with the Five Facet Mindfulness Questionnaire and empathy with the Interpersonal Reactivity Index subscales of perspective taking and empathetic concern. Surveys were implemented at baseline, month 6, and month 15. RESULTS: Of the 365 interns invited to participate, 340 (93.2%; 255 [75.0%] female; 51 [15.0%] 30 years or older) completed surveys at baseline; 273 (74.8%) also participated at month 6 and 195 (53.4%) at month 15. Participants included 194 (57.1%) in the Mindfulness Intervention for New Interns and 146 (42.9%) in the control arm. Analyses were adjusted for baseline outcome measures. Both arms' EE scores were higher at 6 and 15 months than at baseline, but EE did not significantly differ by arm in multivariable analyses (6 months: 35.4 vs 32.4; adjusted difference, 3.03; 95% CI, -0.14 to 6.21; 15 months: 33.8 vs 32.9; adjusted difference, 1.42; 95% CI, -2.42 to 5.27). None of the 6 secondary outcomes significantly differed by arm at month 6 or month 15. CONCLUSIONS AND RELEVANCE: A novel mindfulness curriculum did not significantly affect EE, burnout, empathy, or mindfulness immediately or 9 months after curriculum implementation. These findings diverge from prior nonrandomized studies of mindfulness interventions, emphasizing the importance of rigorous study design and suggesting that additional study is needed to develop evidence-based methods to reduce trainee burnout. TRIAL REGISTRATION: ClinicalTrials.gov Identifier: NCT03148626.

Antiracism in the Field of Neonatology: A Foundation and Concrete Approaches.

Neonatal patients and families from historically marginalized and discriminated communities have long been documented to have differential access to health care, disparate health care, and as a result, inequitable health outcomes. Fundamental to these processes is an understanding of what race and ethnicity represent for patients and how different levels of racism act as social determinants of health. The NICU presents a unique opportunity to intervene with regard to the detrimental ways in which structural, institutional, interpersonal, and internalized racism affect the health of newborn infants. The aim of this article is to provide neonatal clinicians with a foundational understanding of race, racism, and antiracism within medicine, as well as concrete ways in which health care professionals in the field of neonatology can contribute to antiracism and health equity in their professional careers.

Race, language, and neighborhood predict high-risk preterm Infant Follow Up Program participation.

OBJECTIVES: Infant Follow Up Programs (IFUPs) provide developmental surveillance for preterm infants after hospital discharge but participation is variable. We hypothesized that infants born to Black mothers, non-English speaking mothers, and mothers who live in "Very Low" Child Opportunity Index (COI) neighborhoods would have decreased odds of IFUP participation. STUDY DESIGN: There were 477 infants eligible for IFUP between 1/1/2015 and 6/6/2017 from a single large academic Level III NICU. Primary outcome was at least one visit to IFUP. We used multivariable logistic regression to identify factors associated with IFUP participation. RESULT: Two hundred infants (41.9%) participated in IFUP. Odds of participation was lower for Black compared to white race (aOR 0.43, p = 0.03), "Very Low" COI compared to "Very High" (aOR 0.39, p = 0.02) and primary non-English speaking (aOR 0.29, p = 0.01). CONCLUSION: We identified disparities in IFUP participation. Further study is needed to understand underlying mechanisms to develop targeted interventions for reducing inequities.

The influence of social determinants of health on the genetic diagnostic odyssey: who remains undiagnosed, why, and to what effect?

Although Mendelian genetic disorders are individually rare, they are collectively more common and contribute disproportionately to pediatric morbidity and mortality. Remarkable advances in the past decade have led to identification of the precise genetic variants responsible for many of these conditions. Confirming the molecular diagnosis through genetic testing allows for individualized treatment plans in addition to ending the diagnostic odyssey, which not only halts further unnecessary testing but may also result in immense psychological benefit, leading to improved quality of life. However, ensuring equitable application of these advances in genomic technology has been challenging. Though prior studies have revealed disparities in testing for genetic predisposition to cancer in adults, little is known about the prevalence and nature of disparities in diagnostic testing in the pediatric rare disease population. While it seems logical that those with impaired access to healthcare would be less likely to receive the genetic testing needed to end their odyssey, few studies have addressed this question directly and the potential impact on health outcomes. This review synthesizes the available evidence regarding disparities in pediatric genetic diagnosis, defining the need for further, prospective studies with the ultimate goal of delivering precision medicine to all who stand to benefit. IMPACT: Social determinants of health are known to contribute to inequality in outcomes, though the impact on pediatric rare disease patients is not fully understood. Diagnostic genetic testing is a powerful tool, though it may not be available to all in need. This article represents the first effort, to our knowledge, to evaluate the existing literature regarding disparities in genetic testing for pediatric rare disease diagnosis and identify gaps in care.

COVID-19: neonatal-perinatal perspectives.

The coronavirus disease 2019 (COVID-19) pandemic, resulting from infection with severe acute respiratory syndrome coronavirus 2 (SARS-CoV-2), has caused severe and widespread illness in adults, including pregnant women, while rarely infecting neonates. An incomplete understanding of disease pathogenesis and viral spread has resulted in evolving guidelines to reduce transmission from infected mothers to neonates. Fortunately, the risk of neonatal infection via perinatal/postnatal transmission is low when recommended precautions are followed. However, the psychosocial implications of these practices and racial/ethnic disparities highlighted by this pandemic must also be addressed when caring for mothers and their newborns. This review provides a comprehensive overview of neonatal-perinatal perspectives of COVID-19, ranging from the basic science of infection and recommendations for care of pregnant women and neonates to important psychosocial, ethical, and racial/ethnic topics emerging as a result of both the pandemic and the response of the healthcare community to the care of infected individuals.

MINdI: Mindfulness Instruction for New Interns.

Introduction: Trainee burnout has reached epidemic proportions and is increasing among physicians compared to non-health care professionals. Burnout is associated with depression and lower empathy, poor patient adherence to medical plans, and early physician retirement. Mindfulness is the quality of being nonjudgmental and present and has been shown to decrease physician burnout. Implementation of mindfulness curricula may decrease trainee burnout. Methods: Using Kern's six-step approach, we developed an easy-to-implement, facilitator-friendly mindfulness curriculum for pediatric interns. Curricular sessions were held monthly during preexisting 1-hour didactics over 6 months, facilitated by individuals without mindfulness experience. Learners were assessed on knowledge, attitudes, and behavior with postintervention surveys during a pilot in 2016. Qualitative data were used for curricular improvement resulting in the published curriculum. Results: Postcurriculum surveys from our pilot revealed that 69% of interns reported a more positive attitude toward mindfulness, while 62% reported having (1) greater knowledge about evidence supporting mindfulness, (2) improved knowledge on how to apply mindfulness techniques, and (3) the belief that techniques they had learned positively impacted their lives. Thirty-three percent endorsed using mindfulness techniques more frequently than they had prior to the start of the mindfulness curriculum. Discussion: Our novel curriculum provided longitudinal mindfulness training that meaningfully impacted trainee knowledge, behaviors, and attitudes. The curricular structure overcame the need for local topic experts and was feasible to implement within the confines of our complex program structure. Ongoing work will determine the impact of our curriculum on objective measures of burnout, empathy, and mindfulness.