This new publication of ours has been an interesting process, Annie Janvier and I wrote it in collaboration with other parent representatives, Barb Farlow, who we have collaborated with previously, a mother of a little girl who had trisomy 13, and Rebecca Pearce and Jason Baardsnes, who had twins at 25 weeks, one of whom died. As many of you will know, Annie and I are also parent “representatives”, our little treasure Violette (whose hand is at the top of this blog) was born at 24 weeks gestation.

Janvier A, Farlow B, Baardsnes J, Pearce R, Barrington KJ. Measuring and communicating meaningful outcomes in neonatology: A family perspective. Semin Perinatol. 2016 In this article we discuss the outcomes that are commonly measured in neonatology, both in research, and in quality control, and for individual assessments, and ask if we are measuring the right things.

We wanted to be thoughtful and provocative (wanting to provoke change, not just strong reactions), and I think we got it right, let me know if you agree:

Dichotomous and continuous outcomes : we discuss how things which are in reality continuous outcomes, such as lung injury, or developmental delay, have been dichotomized into yes/no, good/bad outcomes, mostly for the convenience of researchers. It makes little sense to label someone who stays in oxygen until 36 weeks and 2 days as having BPD, whereas if the oxygen was weaned 3 days earlier they would not have had BPD. What is important is the impact of that lung injury on the life of the baby.

Even more problematic is combining artificially dichotomized outcomes especially combining them with death (death or BPD, for example) as if coming out of oxygen later were in some way equivalent to being dead.

Of course combining multiple artificially dichotomized outcomes is even more problematic, death or NDI being the most important example, being dead, or having a serious hearing deficit requiring hearing aids, or having a major visual problem, or being stiff on one side, or having a developmental delay which puts you below -2 SD of the population mean, are often combined, as if it was self evident that each one of those adverse outcomes were equally important. We note :

When two outcomes have potentially very different values to patients, they should be discussed separately.

So outcome data combining outcomes which are of very different individual value are of little use. Discussing the risk of death separately to the potential long-term consequences if the infant survives, may give a very different response, to the “80% risk of death or NDI”.

In the article we also discuss other important issues such as “Confusing screening tests with disabilities”, the Bayley Scales of Infant Development should be considered to be a screening test, a way of evaluating current development, screening for delays, and plugging kids into intervention programs if necessary, but NOT used as a way of defining who is OK, and who has an impairment; we know the Bayleys are very poor at prediction of longer term intellectual problems, and I know of no data that low Bayley scores are related to poor quality of life among NICU graduates.

Some outcomes which have a huge impact on families are, in contrast, minimized in neonatal outcome studies: feeding difficulties and need for home gavage feeding for example, are rarely quantified in studies but may be very disruptive for families.

We also discuss the importance of resilience, or “coping”, how many families can adapt to non-optimal outcomes, and can even benefit from them, how resilience has been undervalued in neonatal research:

… a family with a child who has severe cerebral palsy, for example, will find meaning and hope in the life of their infant, even if they wish it was otherwise. The same can be said about families of children who have home ventilation, Down syndrome trisomy 13 or 18. [for each of these examples we give references] Adaptation and coping have been measured in older patients, but neonatal research is lagging behind. Several studies have noted that the adverse impact of a child with neurological or developmental difficulties decreases markedly over the years^. A recent study of families with children who have intellectual disabilities was almost universally positive about the impacts of having such a child on themselves and their family. Interventions aimed at families with poor resilience predictors could improve neonatal and parental outcomes.

We end the article with some recommendations which I reproduce below in shortened form.

Recommendations for Clinicians

1. The caregiver should themselves be informed regarding the condition, the risks, and the potential impacts of the condition for children.

2. Avoid conflating neonatal outcomes: Remember that for a parent, death is not the same as disability. Clinicians should speak about the risk of death, and then of disability in survivors.

3. Children are more than a diagnosis : Parents should not only be informed of potential adverse conditions or diagnoses. They should know what these diagnoses mean to children and families, in a practical sense.

4. Personalize the information: The information should be adapted to the needs and wishes of the family, and the precise nature of the infant’s situation. Many recommendations for antenatal counseling in extreme prematurity demand that information be standardized and that providers describe all the possible complications of prematurity.

5. Provide balanced information: Parents should be given an opportunity to ponder what a diagnosis means for them, for their family, for their future, they should be told about the positives as well as the negatives.

6. Provide information relative to follow-up programs : Parents are often not well informed of the reasons for systematic follow-up. Many parents think this is essential to identify potential problems their child may have…

7. Discuss real-life outcomes: Providers should have regular interactions with NICU parents about the life trajectory of their child: what is the next step—remove the tube, remove the CPAP, full feeds, transition to oral feeds, oximetry, etc…

8. Normality and normativity: Avoid the term “normal” when describing babies after NICU discharge. Their tests and imaging may be “normal” or “abnormal,” above or below average; but for a family—especially after the NICU experience—these babies are extraordinary.

9. Empower: Parents of all NICU children should be able to feel like good parents—doing a good job caring for their child. In the NICU, they should be congratulated when they hold their baby, visit, provide breast milk, read to their child, ask questions, etc. .

10. Inform NICU families of parental support groups or associations that can offer them experiential information about the child/family experience

11. Words are important : Know the name of the baby and avoid labels: neonates are not their condition or their outcome. The way we describe babies can have an impact on how parents see their children.

Recommendations for researchers

1. Research projects should focus on primary outcomes that are of importance to families. Families should be involved in planning research, and in determining which outcomes should be investigated…

2.Decreasing the impacts of neonatal critical illness is a vitally important task, given the improved survival of infants with so many different conditions. But unless we investigate which outcomes are truly most important to our patients and their families, we will not be able to progress further.