Parent Perspectives on Defining Neonatal Serious Illness

Abstract

Objectives:

We hoped to understand (1) how parents of seriously ill neonatal intensive care unit patients conceptualize neonatal serious illness, and (2) how parent and physician perspectives on neonatal serious illness might differ.

Design:

This was a prospective survey study.

Setting/Subjects:

Parent members of the Courageous Parents Network.

Measurements:

We circulated a modified version of a previously developed survey. Participants were presented with a list of potential definition components and asked to rank components by importance and suggest modifications to the definition as needed. Thematic analysis of free-text responses was conducted to identify key themes in parent responses

Results:

Eighty-eight percent of parent participants agreed or strongly agreed with our working definition of neonatal serious illness. Parents agreed with the content of the definition but suggested using different language (especially less jargon) when using the definition to inform conversations with parents.

Conclusions:

The majority of parents surveyed for this study supported our definition of neonatal serious illness, which suggests that it may be useful for clinical and research applications. At the same time, parent responses revealed important differences in perceptions about serious illness between parents and physicians. In addition, parents will likely use any definition of neonatal serious illness differently than clinicians. We therefore propose that our definition be used to identify neonates with serious illness for the purposes of research and clinical care, but should not be used verbatim for communication with parents.

Introduction

Parents of seriously ill pediatric patients possess unique perspectives on serious illness that are underrepresented in the literature.¹ Given that research related to defining neonatal serious illness is an emerging field, parent perspectives on such a definition are not yet known. Earlier work has demonstrated that a definition of serious illness is needed to advance both clinical care for seriously ill patients and research intended to improve such care.^2,3 As a first step, and based solely on physician perspectives, we previously defined neonatal serious illness as:

“1) carries a high risk of short term mortality OR lifelong medical complexity with probable shortened lifespan, 2) may involve substantial prognostic uncertainty (especially in regard to neurodevelopment) that complicates medical decision-making, and 3) significantly impacts the patient and family's life now or in the future with strain related to treatments and care.”²

There is evidence to suggest that parents and physicians perceive serious illness differently. Families often doubt physicians' ability to prognosticate, which is further reinforced when patient outcomes do not align with provider predictions.^4,5 Parents of neonatal intensive care unit (NICU) patients and neonatologists may have discordant views about a child's illness severity. When compared with clinicians, parents are often more tolerant of the prospect of severe disability in their seriously ill infant and even in the face of severe disability remain more likely to want all reasonable measures to prevent death.⁶ This discordance often causes tension and likely impacts all parties' perceptions about neonatal serious illness.⁷

Given the importance of parental perspectives and the need for robust research that incorporates their points of view, this study aimed to elicit parent viewpoints on neonatal serious illness. We hoped to understand (1) how parents of seriously ill NICU patients conceptualize neonatal serious illness, and (2) how parent and physician perspectives on neonatal serious illness might differ. By integrating the voices of parents impacted by neonatal serious illness, we hope to strengthen our conceptual definition of neonatal serious illness to meaningfully improve care for these infants and their parents.

Methods

To further refine our definition of neonatal serious illness, we created a survey for parents of seriously ill neonates, modeled after our previous survey for physicians with expertise in neonatology and pediatric palliative care.² The modified survey was circulated to parents of seriously ill NICU graduates via e-mails sent by the Courageous Parents Network, a nonprofit organization and educational platform that orients, empowers, and accompanies families and providers caring for children with serious illness, to member parents.⁸ Because the Courageous Parents Network includes parents of seriously ill children with and without a history of NICU hospitalization, potential parent participants were screened to exclude families without NICU experience. This project was designated as exempt human research by the Institutional Review Board at the Icahn School of Medicine at Mount Sinai.

Demographic data for study participants were solicited. Participants were asked to rank a list of 15 definition components (such as “carries poor prognosis” or “creates caregiver stress,” see Table 2) according to how important each would be to include in a definition of neonatal serious illness (with 1 being most important and 15 being least important). Participants were presented with our previously published definition of neonatal serious illness² and were asked to rank, on a 5-point Likert scale, the extent to which they agreed with the definition and to suggest modifications, if indicated. Ranking of components were compared for physicians² and parents using Mann–Whitney U tests.

Because participants were instructed not to assign a rank for any component that they did not feel was important to include in a definition, component rankings were analyzed twice: first with any missing rankings excluded from the analysis, and a second time with missing rankings assigned a value of “16” to imply least important.

Parents provided free-text responses to the question, “If you don't feel the above definition comprehensively defines neonatal serious illness, what additions, changes, or deletions would you make to better conceptually define neonatal serious illness?” Thematic analysis, modeled after work carried out by Braun and Clarke, was then conducted by two authors (K.F.G. and G.N.R.) to identify key themes in parent responses and add additional depth to our understanding of neonatal serious illness.⁹

Results

Survey responses

Characteristics of the parent study population (N = 52) are presented in Table 1. Nearly all participants were White, non-Hispanic women. For the majority of participants, their child's NICU hospitalization was between two weeks and two months duration. The most frequently reported reasons for NICU hospitalization were prematurity, neurologic issues, and genetic conditions. Parent ranking of the importance of definition components is given in Table 2. When results were analyzed with a ranking of “16” inserted for any missing rankings, rankings did not change and all significant p-values remained significant. Eighty-eight percent of parent participants agreed or strongly agreed with our working definition of neonatal serious illness.

Table 1.

Participant Demographics and Characteristics

	n (%)
Sex, female (N = 52)	50 (96.2)
Age, years (N = 52)
<24	1 (1.9)
25–34	6 (11.5)
35–44	28 (53.8)
45–54	10 (19.2)
55–64	5 (9.6)
>65	2 (3.8)
Geographic location (N = 52)
Northeast	19 (36.5)
Southeast	5 (9.6)
Midwest	13 (25)
West	9 (17.3)
Southwest	2 (3.8)
International	4 (7.7)
Race (N = 51)
White	47 (92.2)
Black	2 (3.9)
Asian	1 (2.0)
Native American	0
Pacific islander	0
Other	1 (2)
Ethnicity (N = 50)
Non-Hispanic	48 (96)
Hispanic	2 (4)
Child's diagnosis category in NICU (N = 52)
Prematurity	23 (44.2)
Cardiac	6 (11.8)
Pulmonary	7 (13.5)
Neurologic	21 (40.4)
Genetic	24 (46.4)
Metabolic	3 (5.8)
Intestinal	4 (7.7)
Liver	0
Renal	9 (1.9)
Other	4 (7.7)
Length of NICU stay (N = 52)
<1 week	1 (1.9)
1–2 weeks	5 (9.6)
2 weeks–1 month	11 (21.2)
1–2 months	14 926.9)
2–3 months	7 (13.5)
3–4 months	3 (5.8)
4–5 months	8 (15.4)
5–6 months	0
>6 months	3 (5.8)

NICU, neonatal intensive care unit.

Table 2.

Parent Ranking of Importance of Definition Components (N = 52)

Component	Median ^a	IQR	Minimum	Maximum	% Responded
High risk of short-term mortality	1	2	1	13	92.3
Cannot be “cured”	3	6	1	15	82.6
Results in shortened lifespan	3	4	2	14	88.5
Negatively impacts QOL, now or in the future	4	3	1	14	96.1
Negatively impacts daily function, now or in the future	5	2	2	15	94.2
Has/will have burdensome symptoms	7	4	3	10	90.4
Has/will have burdensome treatments	7	4	3	10	86.5
Creates caregiver stress	8	4	2	13	80.8
Creates stress for other family members	11	4	1	15	80.8
Negatively impacts family QOL	10	5	1	14	84.6
Associated with poor neurodevelopmental outcome	9	6	3	12	92.3
Associated with future lack of independence	11	3	3	14	88.5
Associated with future need for long-term care	11	6	1	14	84.6
Associated with future technology dependence	14	3	6	14	82.7
Other	15	0	4	15	61.5
Extent to which participant agreed with definition	4	1	0	4	98

Participants ranked components on a 15-point Likert scale with 1 being most important to include in a definition and 15 being least important to include.

QOL, quality of life.

Component comparisons between parents and physicians

Using our previously published data for physician rankings,² we explored which components differed in importance between physicians and parents. “Burdensome symptoms” (parent median = 7, physician median = 6, p = 0.013), “poor neurodevelopmental outcome” (parent median = 9, physician median = 5, p = 0.001), and “future dependence on technology” (parent median = 14, physician median = 9, p = 0.018) were ranked as significantly more important for physicians than for parents, whereas parents ranked “caregiver stress” (parent median = 8, physician median = 10, p = 0.011) as more important. When results were analyzed with a ranking of “16” inserted for any missing rankings, all significant p-values remained significant.

Thematic analysis

Five key themes emerged from participant free-text responses: financial strain, prognostication, hope, definition challenges, life of value (Table 3). Parents also discussed the importance of “protecting hope” when clinicians speak with families. In discussing challenges and potential pitfalls of a definition for neonatal serious illness, one parent participant suggested that parents may need a definition that is different or modified from the one clinicians use among themselves. Implicit in the comment was the idea that parents anticipate that a definition for neonatal serious illness would be used largely as a communication tool for clinicians to share news of serious illness with parents. This suggestion was echoed in other parent comments that discussed how a purely clinical definition with grim implications might land with a parent who “has hope and [is] focused on fighting for the health of their child.”

Table 3.

Key Themes from Parent Free-Text Responses

Theme	Quote 1	Quote 2
Financial strain	I think taking into consideration the financial strain a child's medical complexity will cause might be important as well. A child with asthma and a child with spina bifida may both be dependent on technology but the costs borne by their families are very different	The financial impact on the family is severe due to parents not being able to work as much due to caregiving needs AND massive co-pays for hospital, prescriptions, equipment, etc.
Prognostication	NICU professionals are very poor prognosticators of life post-NICU	I would say possible, not probable, shortened lifespan. Many children who are medically complex are at risk of a shortened life span but it's not necessarily probable
Hope	If this is a definition given to an overwhelmed new parent, the tone (while necessarily serious) really leaves no room for hope	“High risk of death in the short term” & “probable shortened lifespan”—I agree this is true but difficult to hear as the parent of a NICU baby—how can this be addressed without knocking the wind out of a parent that has hope and focused on fighting for the health of their child.
Definition challenges	My biggest concern is a definition patients and families can understand as it relates to their NICU journey … Being mindful of a definition that clarifies categorical significance for doctors but also caregivers may require a simplified definition care providers can use to assist families in understanding their child's illness	Before a proper prognosis/diagnosis can be determined, I believe you should look deeply beyond NICU professionals … I believe that there ^is^ such a thing as neonatal serious illness and palliative care is absolutely necessary for very severely neurologically impaired infants, but a lot more research needs to be done focusing on post-NICU outcomes before creating this standard
Life of Value	I am just a bit concerned that neonatal serious illness does not become conflated with a life not worth living. My child is living with a serious illness and has been since his NICU stay. It has impacted his life and our life as a family, but he is nonetheless living an amazing life. Impacts on the child and the family are often multidimensional, and many parents find new meaning and purpose through their child's illness journey	While she does have global delays, she has quite a good quality of life and as one GI specialist described it: she is “thriving.” She goes to school, she consistently shows progress in her developmental evaluations, she laughs all the time and love to play games (which really cracks her and all those around her up)

Parents agreed with the content of the definition but suggested using different language (especially less jargon) when using the definition to inform conversations with parents. One parent spoke about wanting a definition that clinicians can use to “assist families in understanding their child's illness.”

Discussion

Our survey study results indicate that our definition of neonatal serious illness resonated with parents of NICU graduates with serious illness. The majority of parents either agreed or strongly agreed with our definition. Given these findings, our conceptual definition of neonatal serious illness may be most useful for clinicians hoping to prospectively identify infants in need of additional supports such as palliative care consultation and for researchers aiming to improve clinical care for such patients.

Although our study supports use of our proposed definition, it also reveals important differences in perceptions of neonatal serious illness between parents and physicians. When comparing component rankings, we found that physicians weighted components relating to long-term patient outcome such as “future dependence on technology” and “poor neurodevelopmental outcome” as significantly more important than did parents. Historically, physicians have relatively low tolerance for such outcomes compared with parents, and are more likely to view disability as representing a lesser quality of life.^6,10 Many parents endorse positive experiences raising a child with disabilities and do not want to be pushed to think of neurodevelopmental impairment as “an inherently negative outcome.”^11,12 Parent free-text responses in our survey echoed these concerns, and emphasized the importance of not equating neonatal serious illness with a life not worth living.

Parents in our cohort also endorsed sentiments that neonatologists are poor prognosticators and may underestimate a child's long-term outcome or overemphasize potential challenges, which may further complicate parent views on discussions of patient outcomes.¹³ Parents of children at high risk for neurodevelopmental impairment report that conversations with physicians frequently focus on long-term outcomes.¹⁴ Our work suggests that this may be reflective of physician and not parent preferences.

Parents rated “caregiver stress” as a more important component of neonatal serious illness than physicians. Given the significant mental health burden (including post-traumatic stress disorder) in parents of former NICU graduates, this is perhaps not surprising.^15–17 Although it has been previously established that parents of seriously ill NICU patients face significant stressors both during and after NICU hospitalization, much work remains to determine how best to support these families longitudinally.¹⁷ Parent free-text responses underscored component rankings and particularly emphasized the financial strain of neonatal serious illness as a key stressor, which has been previously described but is not often discussed by clinicians.¹⁸ Such stress was included in our original physician-generated definition.

The emphasis on caregiver stress and financial strain in parent free-text responses highlights their importance, although these elements may be effects of neonatal serious illness more than inherent in its definition. This should signal to clinicians the need to discuss this often underacknowledged component of neonatal serious illness.

Parents articulated the importance of including “hope” in discussions of neonatal serious illness. They voiced the need for physicians to include strengths-based information in discussions with parents and to avoid assumptions that life with serious illness is not worth living. The importance of hope is well established in the literature, and our results provide another example of its critical role for parents of neonates with serious illness. Our data reinforces the need for enhanced clinician awareness of the value of fostering hope.^4,19

Taken together, our findings suggest that our definition of neonatal serious illness (1) strongly resonates with parents and (2) will be useful in prospectively identifying patients who could benefit from additional family support and/or for the purposes of research. Clinicians and researchers can confidently use our definition to identify seriously ill neonates with the knowledge that this definition is meaningful to both physicians and parents.

Where our definition may fall short is meeting the communication needs of parents. Our data indicate that parents anticipate using a definition of neonatal serious illness differently than physicians. Parents envisioned the definition being useful as a communication tool when clinicians share concern for serious illness. They highlighted the importance of using simple language infused with hope to communicate that raising a child with serious illness can bring joy and meaning. We propose that such a definition might take the form of a VitalTalk-inspired “headline.”²⁰ For example, to disclose concerns that a patient has neonatal serious illness, a provider could say, “Based on what we're seeing, I'm worried that your baby is showing signs that they have serious illness. What this means is that they may face significant health challenges even after the NICU, which will likely impact your whole family and may cause stress. I also want you to know that even with these challenges, many parents find joy and meaning in raising children with serious illness and there is absolutely room for hope about the future.” This approach uses commonly practiced communication skills that are still relatively underutilized in pediatric contexts and incorporates both our novel definition and parent preferences in relation to discussion of neonatal serious illness. Ultimately, clinicians can pair our definition of neonatal serious illness with communication best practices to discuss serious illness with parents and help them plan for the future.

Our study has important limitations. Our study population was largely White and female and therefore is not representative of all parents impacted by neonatal serious illness. For example, earlier work has demonstrated that fathers may experience NICU hospitalization differently and therefore the diminished presence of paternal voices in this work may limit its generalizability.^21,22 In addition, it is well-documented that parents who come from minoritized backgrounds face disparate care in the NICU with worse outcomes for minoritized infants.^23,24 Parents who are subject to racial bias and discrimination may therefore see neonatal serious illness differently. Without their voices we cannot understand their perspectives. Parents who responded to our survey may differ in important ways from parents who chose not to respond. In addition, parents who opt to be involved with an organization such as Courageous Parents Network may differ from parents who do not join such a group. We cannot know how other parents might respond to our survey. We did not collect data relating to parental bereavement or length of time between NICU stay and survey completion. Both of these factors may impact parent perspectives on neonatal serious illness, which may change over time. Rank ordering of definition components may not provide sufficient insight into parental perceptions of neonatal serious illness. The range for multiple components was as wide as 1–15 (although interquartile ranges are much narrower), suggesting considerable variability in parent perceptions, which must be considered when interpreting our findings. Finally, we instructed participants not to rank any components that they felt were unimportant; this may have added avoidable complexity to our analysis and to the interpretation of our results.

Given these limitations, we hope that the thematic analysis of free-text responses adds depth and breadth to the present study and acknowledge that further work will be needed to build upon the foundational data presented here. In addition, although free-text responses are not as comprehensive as qualitative interviews, they enabled parents to provide additional insight into their perceptions resulting in the identification of key themes.²⁵ Future work should include qualitative data collection, including semistructured interviews, to build on the data that we have captured to further enrich our understanding of parent perspectives on neonatal serious illness.

Conclusion

The majority of parents surveyed for this study supported our definition of neonatal serious illness, which suggests that this definition may be useful for clinical and research applications. At the same time, parent responses revealed important differences in perceptions about serious illness between parents and physicians. In addition, parents will likely use a definition of neonatal serious illness differently than clinicians. We therefore propose that our definition be used to identify neonates with serious illness for the purposes of research and clinical care, but should not be used verbatim for communication with parents. Prospective work is needed to understand how our definition performs in real time, as well as to further explore how our definition can be used by clinicians to inform their conversations with parents, and how it may be operationalized in research studies with parent participants.

Footnotes

Acknowledgments

The authors gratefully acknowledge Courageous Parents Network for its support of this research and the parent members of Courageous Parents Network for their thoughtful responses to our survey. We truly appreciate their time, candor, and willing engagement in research.

Authors' Contributions

K.F.G. conceived of the study, acquired and interpreted data, drafted the original article, and approved the final version of the article. A.S.W. conceived of the study, analyzed the data, critically revised the article, and approved the final version of the article. G.N.R. made substantial contributions to the conception of this work, analyzed the data, critically revised the article, and approved the final version of the article. C.A.F. made substantial contributions to the conception of this work, critically revised the article, and approved the final version of the article. All authors agree to be accountable for all aspects of this work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved.

Funding Information

K.F.G.'s work is funded by the National Palliative Care Research Center Kornfeld Scholar's Award and by the Mount Sinai Distinguished Scholar Award.

Author Disclosure Statement

No competing financial interests exist.

Supplementary Material

References

Lord

. Parent perspective and response to challenges and priorities for pediatric palliative care research. J Pain Symptom Manage, 2019; 58(5):e9–e10; doi: 10.1016/j.jpainsymman.2019.08.007

Guttmann

, Weintraub

, Kelley

, et al. Defining neonatal serious illness. J Palliat Med, 2022; 25(11):1655–1660.

Kelley

, Bollens-Lund

. Identifying the population with serious illness: The “denominator” challenge. J Palliat Med, 2018; 21(S2):S7–S16; doi: 10.1089/jpm.2017.0548

Roscigno

, Savage

, Kavanaugh

, et al. Divergent views of hope influencing communications between parents and hospital providers. Qual Health Res, 2012; 22(9):1232–1246

Zier

, Burack

, Micco

, et al. Doubt and belief in physicians' ability to prognosticate during critical illness: The perspective of surrogate decision makers. Crit Care Med, 2008; 36(8):2341–2347.

Lam

, Wong

SPS

, Liu

FYB

, et al. Attitudes toward neonatal intensive care treatment of preterm infants with a high risk of developing long-term disabilities. Pediatrics, 2009; 123(6):1501–1508.

Katz

, Hynson

, Gillam

. Dissonance in views between parents and clinicians of children with serious illness: How can we bridge the gap?. J Paediatr Child Health, 2021; 57(9):1370–1375; doi: 10.1111/jpc.15612

Anonymous. Courageous Parents Network. 2021. Available from: https://courageousparentsnetwork.org [Last accessed: June 13, 2023].

Braun

, Clarke

. Using thematic analysis in psychology. Qual Res Psychol, 2006; 3(2):77–101.

10.

Kon

, Ackerson

, Lo

. Choices physicians would make if they were the parents of a child with hypoplastic left heart syndrome. Am J Cardiol, 2003; 91(12):1506–1509; doi: 10.1016/S0002-9149(03)00412-0

11.

Adams

, Tucker

, Clark

, et al. “Quality of life”: parent and neonatologist perspectives. J Perinatol, 2020; 40(12):1809–1820; doi: 10.1038/s41372-020-0654-9

12.

Taunt

, Hastings

. Positive impact of children with developmental disabilities on their families: A preliminary study. Educ Train Ment Retard Dev Disabil, 2002; 37(4):410–420.

13.

Guttmann

, Flibotte

, DeMauro

. Parental perspectives on diagnosis and prognosis of neonatal intensive care unit graduates with cerebral palsy. J Pediatr, 2018; 203:156–162.

14.

Guttmann

, Flibotte

, DeMauro

, et al. A mixed methods analysis of parental perspectives on diagnosis and prognosis of Neonatal Intensive Care Unit graduates with cerebral palsy. J Child Neurol, 2020; 35(5):336–343.

15.

Treyvaud

, Lee

, Doyle

, et al. Very preterm birth influences parental mental health and family outcomes seven years after birth. J Pediatr, 2014; 164(3):515–521.

16.

Pace

, Spittle

, Molesworth

CML

, et al. Evolution of depression and anxiety symptoms in parents of very preterm infants during the newborn period. JAMA Pediatr, 2016; 170(9):863–870.

17.

Williams

, Patel

, Stausmire

, et al. The neonatal intensive care unit: Environmental stressors and supports. Int J Environ Res Public Health, 2018; 15(1):60.

18.

Miller

, Pamela

, Seltzer et al.R Costs of neonatal medical complexity: Impact on new parent stress and decision-making. Am J Perinatol, 2022. [Epub ahead of print]; doi: 10.1055/a-1948-2580

19.

Rosenberg

, Arnold

, Schenker

. Holding hope for patients with serious illness. JAMA, 2021; 326(13):1259–1260; doi: 10.1001/jama.2021.14802

20.

Anonymous. VitalTalk. n.d. Available from: vitaltalkconversations[dot]com [Last accessed: March 2, 2023].

21.

Prouhet

, Gregory

, Russell

, et al. Fathers' stress in the neonatal intensive care unit. Adv Neonatal Care, 2018; 18(2):105–120.

22.

Arockiasamy

, Holsti

, Albersheim

. Fathers' experiences in the neonatal intensive care unit: A search for control. Pediatrics, 2008; 121(2):e215–e222.

23.

Witt

, Malcolm

, Colvin

, et al. Racism and quality of neonatal intensive care: Voices of Black mothers. Pediatrics, 2022; 150(3); doi: 10.1542/peds.2022-056971

24.

Ravi

, Iacob

, Profit

. Unequal care: Racial/ethnic disparities in neonatal intensive care delivery. Semin Perinatol, 2021; 45(4); doi: 10.1016/j.semperi.2021.151411

25.

O'Cathain

, Thomas

. “Any other comments?” Open questions on questionnaires—A bane or a bonus to research?. BMC Med Res Methodol, 2004; 4(25):1–7.

Supplementary Material

Please find the following supplemental material available below.

For Open Access articles published under a Creative Commons License, all supplemental material carries the same license as the article it is associated with.

For non-Open Access articles published, all supplemental material carries a non-exclusive license, and permission requests for re-use of supplemental material or any part of supplemental material shall be sent directly to the copyright owner as specified in the copyright notice associated with the article.

0.00 MB

0.08 MB