Lasting Legacy: Maternal Perspectives of Perinatal Palliative Care

Abstract

Background:

Many of the leading causes of infant mortality are diagnosed prenatally, presenting providers with the ability to present perinatal palliative care planning as an option.

Objective:

Our study adds to the literature both by describing infant interaction with the health care system and by gaining deeper understanding of the maternal experience after being offered perinatal palliative care.

Methods:

The study was conducted at a public university-based medical center in the Midwest. Phase 1 consisted of a retrospective review of electronic medical records of 27 mother–infant pairs offered perinatal palliative care, 18 of whom elected to develop a perinatal palliative care. Phase 2 consisted of a focus group and interviews of seven of the mothers.

Results:

In the initial phase of this study, results revealed differences regarding the infant's end-of-life trajectory, including location of death, number of invasive procedures, and death in the setting of withholding versus withdrawing life-sustaining treatment. Highlighting that without a perinatal palliative care plan in place, the default treatment for infants with prenatally diagnosed life-limiting conditions is likely to be invasive and painful with often times minimal likelihood of long-term survival. Analysis of interview and focus group data revealed three themes: care, choice, and legacy.

Conclusion:

The authors used their experience with the health care system to draw implications for practice from the focus group and interview data, which care can serve to promote women feeling cared for and cared about, as well as promote opportunities for hope during a fragile pregnancy.

Introduction

While infant mortality in the United States has declined over the last decade from 6.14 to 5.96 per 1000 live births, the leading causes of infant mortality are congenital malformations, deformations, and chromosomal abnormalities, which account for ∼21% of all infant deaths.¹ Many of these conditions are now commonly diagnosed prenatally, which gives providers the ability to present perinatal palliative care (PPC) planning as an option.

Palliative care for infants was first described in 1982² and over the past three decades, the concept has evolved. A 2012 review by Balaguer et al. identified 101 studies on neonatal palliative care, with 25% of those studies focusing on perinatal palliative care.³ Existing literature focuses primarily on the composition of the palliative care team,⁴ illustration of a palliative care plan,⁵ or description of the patient population served.^6,7 Few studies have described the infant interaction with the health care system after birth^6,8 and although there has been a recent increase in literature involving parental viewpoints of perinatal palliative care,^9,10 the recent review by Rocha Catania et al.¹¹ confirms that perinatal palliative care is a growing area of scholarship. Our study adds to the literature both by describing infant interaction with the health care system and by gaining a deeper understanding of the maternal experience among mother's who contemplated perinatal palliative care during pregnancy from which important implications for practice are described.

Methods

The study was conducted at a public university-based medical center in the Midwestern USA. Study approval was obtained from the Institutional Review Board. At our institution, mothers, whose fetus has a prenatally diagnosed life-limiting condition, including, but not limited to, chromosomal abnormalities, renal anomalies, central nervous system anomalies, cardiac malformations, or other severe structural anomalies, receive care through the interprofessional Fetal Diagnosis and Treatment Center (FDTC). Referrals to the perinatal palliative care program (PPCP) are made following FDTC discussion and group consensus. The palliative care plan is entered into the maternal and fetal electronic medical record. During the study period most plans included do not attempt resuscitation (DNAR) orders with plan for exclusively comfort care, which pediatric hospitalists provided on the obstetric ward until either death or discharge to home with hospice care. For families who chose intensive care, the PPC team remained involved to offer concurrent care in the neonatal intensive care unit (NICU).

Phase 1 of this study consisted of a retrospective review of electronic medical records by the principal investigator (P.I.) between 1/2011-9/2014 to determine how many mothers of infants with a life-limiting condition were offered perinatal palliative care. Twenty-seven infants were identified (Fig. 1). Descriptive statistics (i.e., median, percentage) were used to describe the patient populations, continuous data were analyzed using the Mann–Whitney U test, and categorical data were analyzed with Fischer's exact test.

FIG. 1.

Infant identification and inclusion. FDTC, Fetal Diagnosis and Treatment Center; NICU, neonatal intensive care unit; PPCP, perinatal palliative care program.

Phase 2 of this study were focus group and interviews to gain deeper understanding of maternal experience after being offered PPC (Fig. 2).

FIG. 2.

Focus group flowchart. P.I., principal investigator.

Researcher S.O.V. led the focus groups and interviews. She is currently Associate Professor in the School of Nursing at her home institution. She has extensive experience in qualitative research and is published in numerous peer-reviewed journals.^12–14 To begin the focus group and interview sessions, the consent form was thoroughly reviewed by the researcher allowing time for questions. Each participant received a folder, which included a consent form, demographic form, Complicated Grief Questionnaire,¹⁵ mental health resources, and an honorarium.

The focus group and interview text analysis were simultaneous and continuous. Each member of the research team independently read through the typed transcripts to get a sense of the whole. Following this review, members of the research team met to discuss the coding scheme, extract common themes, and identify relevant quotations. From this analysis, researchers used an inductive approach to combine the codes into three major themes related to the maternal experience of receiving a life-limiting diagnosis during pregnancy.

Results

Data from the initial phase of this study were obtained from maternal and infant electronic medical records and are summarized in Tables 1 and 2. For mothers, there was a statistically significant difference between the groups in the proportion of mothers who were receiving medical assistance. There were no statistically significant differences between the infant groups. Results revealed differences regarding the infant's health care system interaction after birth, summarized in Table 3. Thirty-three percent of those invited agreed to participate in the focus group/interviews. Analysis of the focus group/interview data revealed that five study participants completed the Complicated Grief Questionnaire,¹² none of whom met criteria for a complicated grief diagnosis. Qualitative data analysis revealed three themes: care, choice, and legacy.

Table 1.

Infant Demographics

	PPCP accepted (n = 18)	PPCP declined (n = 9)	p
Gestational age, median (range)	35^2/7 (20^3/7–40 ^6/7)	38 ^1/7 (29^1/7–39^5/7)	0.90
Birthweight, median (range)	2.89 (1.64–3.40)	1.96 (1.05–2.93)	0.21
Infant diagnosis (n)
Chromosomal abnormalities	3	3
Skeletal dysplasia	6	0
Central nervous system malformation	4	0
Severe cardiac anomalies	2	1
Severe renal anomalies	1	1
Hydrops fetalis	1	1
Complex congenital diaphragmatic hernia	0	2
Metabolic	1	0
Severe airway anomalies	0	1

PPCP, perinatal palliative care program.

Table 2.

Maternal Demographics

	Phase 1 PPCP mothers (n = 17)^a	Phase 1 no PPCP mothers (n = 9)	p
Age, median (range)	32 (20–38)	28 (24–34)	0.91
Relationship status, n (%)
Married/partnered	15 (88)	8 (89)	1.0
Single	2 (12)	1 (11)	1.0
Race, n (%)
White, non-Hispanic	14 (82)	4 (44)	0.16
Of color	3 (18)	4 (44)
Unknown	—	1 (11)
Medical assistance recipient, n (%)	5 (29)	7 (78)	0.038
	Phase 2 mothers (n = 7)	All mothers (n = 26)^a	p
Age, median (range)	33 (28–33)	31.5 (20–38)	0.46
Relationship status, n (%)			0.28
Married/Partnered	5 (71)	23 (34)
Single	2 (29)	3 (12)
Race, n (%)			0.64
White, non-Hispanic	6 (86)	18 (70)
Of color	1 (14)	7 (27)
Unknown	—	1 (3)
Medical assistance recipient, n (%)	2 (29)	12 (44)	0.55

Data for one of the mothers were not available.

Table 3.

Infant Interaction with Health Care System

	PPCP accepted (n = 18)	PPCP declined (n = 9)	p
ICU days, median (range)	0 (0–6)	1 (1–34)	0.001
Aggressive life-sustaining treatment provided, n (%)	1 (6)	8 (89)	<0.001
Withdrawal from life-sustaining treatment, n (%)	1 (6)	5 (56)	0.04
Median invasive procedures per patient^a, median (range)	0 (0–4)	3 (0–10)	0.008
Days from DNAR to death, median (range)	0 (0–1)	3.5 (1–12)	0.03
Death in the NICU, n (%)	2 (11)	9 (100)	<0.001
Death following discontinuation of CPR, n (%)	1 (6)	5 (56)	0.04

Includes: intubation, umbilical catheter placement, needle decompression, chest tube placement, percutaneously inserted central catheter line placement, therapeutic hypothermia, pericardial tap, extracorporeal membrane oxygenation, ex utero intrapartum treatment procedure. Does not include peripheral venous catheter, nasogastric/orogastric tube placement.

DNAR; ICU, intensive care unit; NICU, neonatal intensive care unit.

Theme: Care

The participants in our study appreciated expert and personalized care from the interprofessional team. This level of care requires excellent team communication and comprehensive care for women who are offered perinatal palliative care planning. Medical providers should encourage the “normal” within the extraordinary for these patients and families and should advocate for memory making whenever possible. The participants in our study objected strongly to the language “incompatible with life” as a descriptor for their infant's condition. Certainly there are many ways to convey the likely death of a newborn without using terminology, which evokes such a negative response.

Theme: Choice

Participants also identified the lack of provided resources as a deficit in otherwise generally positive care experience. Many participants described learning of options for their child's care through online organizations, which were ultimately able to be supported by the medical team. It would be prudent for organizations providing high-risk obstetric care and/or neonatal consultation to have a list of resources available for families to reference. Medical teams should provide all medically appropriate options available, including termination, palliative care, and aggressive care early to support informed maternal decision making and autonomy.

Theme: Legacy

Interprofessional perinatal palliative care teams should promote legacy formation with focus on the infant, family, and larger community. These legacy opportunities are present from diagnosis, through pregnancy as well as after birth. It is beholden on us as the care team to identify and anticipate these opportunities for families, especially for those families who find meaning through opportunities, such as organ donation or research. Ultimately, the care these mothers received, the choices they made, and the experiences/memories come together to form a lasting legacy no matter how close together hello and goodbye might be.

The authors used their experience with the health care system to draw implications for practice from the focus group and interview data, presented in Table 4 along with selected supporting quotations.

Table 4.

Themes, Quotations and Implications for Practice

Theme	Supporting quotations	Implications for practice
Care	• The doctor…He had so much care and concern on his face and countenance and everything about it and asking our questions, taking the time. We never felt rushed; we never felt we were putting someone out. We felt so cared for, cared about (P2)• I have to commend the way the social worker handled us and spoke with us. And honestly, we were home probably a week, and she called to see how we were doing. To me, I felt like, we weren't just a number then. We weren't just a family that came in and took their [child] home to die. (P6)• Once they [health care team] understand that the parents do understand, but still are choosing life for their child and choosing to do as much as they possibly can to give him every change, is to then change their focus; to come alongside them. (P6)• Everyone made it seem so normal. Like, here, let's give her a bath. Of course we're going to give her a bath; that's what you do with a baby. Then of course we're going to hold her and of course your whole family is going to come in here… We can hold her and we can pass her around. We can talk and we can smile and we can take pictures, but we didn't know what to expect. They just so nonchalantly, this is what you do. (P1)• I had the same pediatrician for all four of my kids… he said, ‘I'm going to be that baby's doctor,’ so she was there. She wasn't on call or anything, but she was there for my daughter, and just knowing that because over the years we've built a relationship and that that doctor wanted that baby for a patient, that meant a lot, too (P3)• They're not incompatible and it [incompatible with life] does need to go away and doctors should never say that. It just shouldn't be said. It's dehumanizing. (P4)• At times, it was just too much information where it became daunting. (P6)	1. Provide detailed birth planning and ensure that the birth plan is accessible to all members of the team.2. Empower all members of the interdisciplinary team to support families as they process the diagnosis and care plan throughout the pregnancy3. Embrace flexibility when the care plan is changing and communicate this openly to parents.4. Embed routine newborn care as much as possible: bathing, handprints, family visits, pictures, etc.5. Use alternative phrasing and avoid stating that the diagnosis is incompatible with life.6. Be sensitive to, and try to limit reminders about normal newborn care provided within the same unit7. Schedule frequent visits so that questions can be answered frequently and information can be delivered in manageable amounts.
Choice	• I kept asking was there a way to connect with other families and they didn't really have anything. (P3)• She said, ‘Who's going to be your pediatrician?’ Well, that's something we never really thought about, because we didn't think we'd ever really need one, because we didn't know. It was one of those things that was such a back-burner thing. (P6)• We let them know we want to do everything we can, because we didn't know how close hello and goodbye would really be (P6)• When I got the quad test it was not because, well, something is wrong; I'm going to terminate. It was because I wanted to be the best mom I could. (P3)• For me what helped me make decisions was knowing in advance what my options were. (P4)• As a family we focus on quality, and not quantity of life. (P6)• We did work with a lot of specialists ahead of time, so that helped a lot. (P7)• This is God's choice not mine when this baby goes and I'm going to love this little girl. (P3)• I would say that was my saving grace, knowing in advance. I am a planner, I could plan it to a T and I could be prepared for it. (P5)	1. Provide resources early to families, particularly local and online parent support groups.2. Encourage parents to identify a primary care provider should the infant survive to hospital discharge.3. Provide all options (including termination, palliative care, and aggressive care) as well as subspecialty consultation early to support informed maternal decision making.
Legacy	• I have a [ultrasound] video of them playing together and I'll show her sister. (P2)• The anesthesiologist grabbed my camera and he took a video, which I never would have thought of, so we have her on video, alive. (P1)• They made me a plaster mold in the shape of a heart that's got her hands and feet on it… That is my most cherished possession in the entire world. I just thought that was amazing. (P7)• If they knew she was going to die, like they had determined there was nothing they could do, I wish they would have shown her to me sooner, because by the time I saw her, she was very discolored, like purple. Dark purple. I would've liked to see her when she was still pink. (P7)• That is something I'll never forget- they came to the hospital and this man had a box, a black box, and they put your baby in a box and they walk out. It's really something devastating… That was really tough. You get these images burned in your mind forever. (P2)• My brother-in-law took our pictures along with a Now I Lay Me Down to Sleep photographer and then he has signed up to do the same which I think is a really positive thing. (P5)• I was able to donate her heart valves, her liver cells and something else… I wish more people knew about that, too, that they can donate their baby's organs in most circumstances, because you want to be able to have that baby have this impact on other babies and families. (P3)	1. Support opportunities for whole family memory making both before and after delivery.2. Offer to provide tangible mementos of the infant, such as hand and foot molds, and locks of hair.3. Think outside of routine protocols to support memory making, such as providing opportunities for families to bring their infant home after death or alternative means of transporting the dead infant to the morgue.4. Encourage families to have someone available to take a video of the infant immediately after delivery.5. Assist families in identifying meaningful legacy opportunities, such as participation in research and breastmilk donation.

Quotations are attributed to the participant in parenthesis after the quotation. For example, (P1) indicates the comment was made by participant 1.

Discussion and Conclusion

Perinatal palliative care is a recent advancement in the field of palliative care medicine. In this sample, 67% of participants offered perinatal palliative care developed a care plan. Perhaps most striking is that for those families that developed a PPCP, 89% of deaths occurred in the setting of with-holding life-sustaining intervention. For those who did not, 72% ultimately chose to withdraw life-sustaining treatment for their infant, but the remaining 28% died in the setting of cardiopulmonary resuscitation. This study highlights that without a perinatal palliative care plan in place, the default treatment for infants with prenatally diagnosed life-limiting conditions is likely to be invasive. Despite routine comfort measures, including nonpharmacological, oral sucrose, and pain medications as needed, there are aspects of intensive care that are painful with often minimal likelihood of long-term survival. For those diagnoses where survival is not expected, PPCP should be an integral part of comprehensive prenatal collaboration, an approach to care, which is supported by the American Academy of Pediatrics policy statement: Noninitiation or Withdrawal of Intensive Care for High-Risk Newborns.¹⁶

Thus far, literature in this field has primarily focused on perinatal palliative care team composition, implementation, and scope. Our study adds important insight from those most intimately affected by the diagnosis of a fetal life-limiting diagnosis, the pregnant mother. Limitations of this study include that it is a single-center study with retrospective demographic data, which are limited due to inconsistency in documentation of ethnicity, religion, and education. Only mother–infant pairs with a perinatal palliative care plan or whose infant died in the NICU were included. This study does not include mother–infant pairs without a prenatal diagnosis or mother–infant pairs who declined perinatal palliative care where the infant survived to discharge. Additionally, we do not present data from significant others regarding their experience.

Empathic interprofessional care can serve to promote women feeling cared for and cared about as well as promote opportunities for hope during a fragile pregnancy. Decision making can be supported by care teams who are open minded, provide comprehensive choices, are able to anticipate memory making opportunities, and importantly avoid use of the term “incompatible with life.” Ultimately, the care and choices during pregnancy develop into a legacy these mothers have created for their child, a legacy which lasts beyond the final heartbeat.

Footnotes

Acknowledgments

This research was partially supported by a grant from the Benjamin Walker Hanson Neonatology Fund, part of the University of Minnesota Foundation. The authors would like to thank Sonja J. Meiers PhD, RN who provided expertise on focus group text analysis.

Funding Source: Ben's Buddies, Roseville, MN.

Author Disclosure Statement

No competing financial interests exist.

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