In this issue of Pediatrics, Fuller et al1  provide compelling evidence that both the fathers and mothers of children born with major congenital anomalies have a higher risk of premature mortality. The increased paternal mortality is the new revelation. The authors looked at time to death and cause of death after the birth of an infant with major congenital anomalies. Data were harvested from multiple national registries in Denmark by using a robust methodologic approach.

The authors found an increased risk of both paternal and maternal mortality due to cardiovascular, endocrine, and metabolic causes. Behavioral health needs, including for substance and alcohol abuse, were higher. Distressingly, mothers of affected infants had an increased risk of death from unintentional injuries, violence, and other trauma. Notably, these are largely preventable causes of morbidity and mortality.

The authors’ analytic approach does not support drawing causal inferences related to circumstances arising beyond the newborn period, limiting the ability to generalize the conclusion that parenting an infant with complex medical needs can lead to increased premature mortality. However, it is generally known that family caregivers of children with complex needs experience significant stress. The authors appropriately cite evidence that increased stressors borne by family caregivers of children and youth with special health care needs (CYSHCN) are associated with increased risk of poor health as well as negative social and economic outcomes. Self-reported outcomes in the US National Survey of Children’s Health (2018–2019) reveal that 53% of mothers of CYSHCN with more complex needs who are the primary caregiver in the household and 45% of fathers who are the primary caregiver in the home report that their physical and/or mental health is not “excellent or very good.”2 

The recommendation to include assessment of the health risks and wellness of fathers, in addition to the mother, is of vital importance and represents an important contribution to the evidence in support of advancing comprehensive, family-centered care. We are now confronted with important questions regarding the most reliable, equitable, and sustainable approach by which this recommendation might be implemented. For example, who should be accountable for thorough, longitudinal screening of both parents? How do those doing the screening get trained, and what instrument might be employed? Should this screening be linked with ongoing screening of the CYSHCN by team members of the patient- and family-centered medical home?3,4  How will the results of these screens be interpreted, and who will be responsible for following-up? Should the care plan of the CYSHCN include referrals for assessment of the health and wellness of the parents? What are implications for integrating care across providers of different family members? For care models in which the medical home team follows the entire family, such as family medicine or medicine-pediatrics, operationalizing this may be more straightforward. In care models that are limited to children and adolescents, this may be more challenging. To inform this more holistic approach, we must draw on frameworks that support person- and family-centered care coordination and integration.5,6  Performance measurement will be essential.7 

The limitations of this study need to be acknowledged. First, Denmark is a small country with a predominantly white population with access to universal health care. In fact, associated parental mortality may be more significant in countries with diverse populations, poorer economies, and/or reduced access to health care. Second, the study period spans 30 years (1986–2015), a period that has seen significant improvements in health care and management for individuals with congenital anomalies and rare genetic conditions.

Although this study is primarily focused on children with congenital anomalies, one wonders about its implications for a broader spectrum of conditions, including neurodevelopmental disorders, autism,8  Down syndrome,9  and disorders identified through newborn screening, in which long-term parental anxiety plays a significant role, even in conditions with excellent outcomes.10 

The results of the study support a holistic, family-oriented approach to the management and care of patients with congenital anomalies and potentially other rare genetic conditions that is long overdue and must inform current standards of care. It is reasonable to advocate for screening the health and wellness of the caregivers of CYSHCN, irrespective of the etiology of those special health care needs. In addition, we must note the impact of disparities based on race, ethnicity, and language for some CYSHCN and their families who are at an increased risk of receiving insufficient care coordination and of consequent poor outcomes.11,12  To ensure achievement of equitable outcomes, we must implement robust measurement, enabling stratification, if not risk adjustment, of performance metrics. If we are committed to optimizing the health outcomes of all persons, the family-centered care model must include assessment of the needs and strengths of all caregivers in the family. If this is not done, are we truly providing care that is family centered?

Opinions expressed in these commentaries are those of the authors and not necessarily those of the American Academy of Pediatrics or its Committees.

FUNDING: No external funding.

COMPANION PAPER: A companion to this article can be found online at www.pediatrics.org/cgi/doi/10.1542/peds.2020-028571.

CYSHCN

children and youth with special health care needs

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Competing Interests

POTENTIAL CONFLICT OF INTEREST: The authors have indicated they have no potential conflicts of interest to disclose.

FINANCIAL DISCLOSURE: The authors have indicated they have no financial relationships relevant to this article to disclose.