Introduction

Sections of this chapter have been reproduced with permission from Pillay et al.1 and Cupit et al.2 These are Open Access articles distributed in accordance with the terms of the Creative Commons Attribution (CC BY 4.0) licence, which permits others to distribute, remix, adapt and build upon this work, for commercial use, provided the original work is properly cited. See: https://creativecommons.org/licenses/by/4.0/. The text below includes minor additions and formatting changes to the original text.

Background

The structure of neonatal units in England

In England care for sick neonates is supported by NHS Specialised Commissioning. It is delivered by neonatal units clustered in and managed through 10 neonatal operational delivery networks. These neonatal units are designated either as neonatal intensive care units (NICU), local neonatal units (LNUs) or special care baby units (SCBU) based on their resources, staffing and capabilities.3,4 NICU are tertiary units colocated within maternity services that have specialist obstetric and feto-maternal medicine services. NICU have resources and staff to provide tertiary level care for sick babies with varying complexities of clinical conditions, and across all gestational ages at birth. LNU provide care for neonates in their local catchment area; these are not configured to provide long-term intensive care, but can provide emergency care, and short-term intensive support for sick babies. They are considered non-tertiary units. In general, they care for those who do not demand the very highest intensity of neonatal care. SCBU provide care, ideally close to the family home, usually for those born at > 32 weeks gestation. They provide only the lowest intensity of neonatal care.

Clinical profile of very preterm babies

When compared with their extremely preterm counterparts born between 23⁺⁰ and 26⁺⁶ weeks gestation (hereafter referred to as born at 23–26 weeks), very preterm babies born between 27⁺⁰ and 31⁺⁶ weeks gestation (hereafter referred to as born at 27–31 weeks gestation) tend to have a more favourable prognosis. The effects of immaturity per se are often less profound in the group born at 27–31 weeks gestation compared to those on the threshold of viability, born at 23–26 weeks.5 Unless considered non-viable in the delivery suite, babies born at 27–31 weeks gestation are always admitted to a neonatal unit for care, as they are too immature for immediate transfer home or for routine care in a postnatal ward. They are a heterogeneous group; birth in this range is associated with varying but substantial adverse outcomes and differing mortality for each week of gestational age.5 Many studies that have included babies born at 27–31 weeks gestation were conducted many years ago and were designed to clarify the effects of very low birthweight rather than prematurity on outcomes.

Very preterm babies have shown a relatively greater increase in survival over the past two decades compared with less mature babies.5 A gradient of risk, with rates of mortality and morbidity increasing from birth at 31 to 27 weeks gestation, has been observed.5 These place increasing demands on neonatal services and have likely led to changes in the profile of neonatal morbidity, hospitalisation and outcome, the details of which have not yet been captured for UK cohorts.

Very preterm babies: where are they born and cared for in England?

For babies born extremely preterm, at 23–26 weeks gestation, management in a NICU or high-volume neonatal unit confers significant benefit for survival to discharge;6 this has shaped policy for these babies in England.7 Similar findings have been observed in other parts of the world.8–12 In contrast, there is limited evidence to guide the location of care for the next most vulnerable group of very preterm babies, born at 27–31 weeks gestation. Current policy indicates that they can be born and cared for in either NICU or LNU setting, but their care pathways are undefined, and their birth and care is spread, often arbitrarily, across the two settings. In 2013 neonatal data analysis unit (NDAU) data for England shows (unpublished), ~48% were born into maternity services colocated with NICU, and ~41% with LNU. Currently, this translates to care across 43 NICU and 75 LNU in England. Those inadvertently born into maternity services attached to SCBU are transferred to NICU or LNU for ongoing care. In a narrative review of the impact of place of birth and care for this cohort of babies,13 there appears to be a lack of clarity and consensus not just in England, but globally, on the best place of birth and care for very preterm babies.

The main driver for NHS neonatal care pathways for the very preterm baby born in the UK comes from gestation-specific analyses for extremely preterm babies born at 23–26 weeks gestation.6 There are currently no data on the optimal location of birth and care for preterm babies born at 27–31 weeks gestation, by designation of unit or any other characteristic, although there is limited evidence that mortality and the most prevalent morbidity outcomes for very preterm births, namely necrotising enterocolitis (NEC), retinopathy of prematurity (ROP) and bronchopulmonary dysplasia (BPD), are similar between LNU and NICU.11 However, it is not known whether location of care makes a difference to gestation-specific outcomes. While the type of care provided by LNU and NICU overlaps considerably, the complexity of problems that can be managed in each differs.5 We do not know if some babies, such as those who are more preterm or sick, may benefit from care in NICU, which manage the most complex conditions and complications, or whether others, less sick or more mature, may benefit more from care in LNU, focused on lower intensity care, closer to home.

At present, the chief determinant of where a pregnant woman receives antenatal care and gives birth between 27 and 31 weeks gestation, and therefore where her baby receives initial care, is the hospital where she chose to book for antenatal care. This may be in a maternity service colocated with NICU, LNU or SCBU. This is usually the centre closest to her home and is not necessarily determined by the anticipated degree of illness or complexity of care required for her or her baby. For most women at the time of booking for pregnancy care, the risk of pregnancy complications and/or preterm delivery is unpredictable. Where an antenatal reason for continued care in a maternity centre colocated with NICU is noted, and/or there is an anticipated need for NICU support for the baby, these pregnant women are triaged in utero for appropriate ongoing care.

National Health Service England service specifications for neonatal critical care (intensive, high-dependency and special care) E08/S/a7 outlined for LNU that anticipated singleton births before 26⁺⁶ weeks gestation, multiple births before 27⁺⁶ weeks gestation and births where the estimated birthweight is < 800 g should be transferred to maternity services colocated with NICU. Babies born critically ill requiring ongoing intensive care for more than 48 hours should be discussed with NICU. In many cases this has led to transfer to NICU. These recommendations have been consolidated in the Neonatal Critical Care Review,14 and a recent national neonatology report15 has explored this further. Bed/cot capacity for care of both mother and baby are major determinants for place of care for these babies. Where all these conditions are met, LNU may triage babies to NICU in the antenatal period (in utero) or in the postnatal period (ex utero), per protocol, as dictated by the NHS service specifications.

Healthcare burden of very preterm births

These babies accounted for approximately 12% of all viable preterm births in England in 2013 (6242/51,0001). They utilise over 304,893 bed-days/year, constituting over a third of all bed-days offered to babies in neonatal units in England in 2013 and 2014.16 This is more than twice the neonatal unit bed-days utilised by babies born at 23–26 weeks gestation, and 4–5 times the numbers of babies born at 23–26 weeks gestation in England. Their mortality in neonatal care in England was 30 per 1000 neonatal admissions (NDAU unpublished data, 181 deaths/5991 admissions in 2014) and their infant mortality, 26/1000 live births.17 They spend a median of 44 days in neonatal care and are discharged at a median of 36.4 weeks post conceptional age.18 Survivors at 31 weeks gestation utilise a median of 34 hospital bed-days for their initial neonatal care, while those at 27 weeks gestation, a median of 79 days (for those born at 27 weeks gestation).19,20

Economic cost of care

Preterm births (< 37 weeks gestation) generate a substantial healthcare cost, initially during the neonatal hospital stay, and subsequently through increased risks of long-term neurodevelopmental, cognitive, behavioural and physical health problems.21–23 The overall cost of care for preterm babies in England and Wales was estimated at £2.946 billion in 2006.24 At that time, the initial hospitalisation costs for preterm babies < 33 weeks gestation was £57,726 [95% confidence interval (CI) 28,779 to 94,868]25 per baby. Current estimates of the costs associated with births between 27 and 31 weeks gestation are lacking for England. Recent literature suggests an inverse relationship between healthcare costs and gestational age.

Mean [standard deviation (SD)] initial hospitalisation costs from Canada for example26 were estimated at $101,757 ($61,788), $82,719 ($55,321), $59,428 ($44,395), $45,229 ($35,330) and $35,699 ($30,822) for babies born at 27, 28, 29, 30 and 31 weeks gestation, respectively (2017 Canadian $). In Finland, costs were estimated to range from €88,188 per infant born at 26–27 weeks gestation, €56,588 per infant born at 28–29 weeks gestation and €35,147 per infant born at 30–31 weeks gestation (2008 Euro €).21

National Health Service hospital costs are high in England, with specialised neonatal intensive care ranging from £776 to £1810 per day and high-dependency care from £709 to £1243 per day, over the past 5 years1,27,28 depending on the neonatal network in which care is delivered. With increased survival in very preterm babies, the costs of their neonatal and childhood course are unknown. Specifically, for LNU and NICU in England, it is unknown whether care is delivered more cost-effectively in one or the other type of unit. Cost-effective healthcare delivery is a key priority in the NHS; this information is essential in any attempt to optimise neonatal services for very preterm babies, born at 27–31 weeks gestation in England.

Limited healthcare resources mean that alongside assessments of clinical effectiveness, consideration also needs to be given to the cost-effectiveness of alternative care settings for babies born at 27–31 weeks gestation. Survival following preterm birth has improved, and hospital care is costly. The 2021–2 daily cost for neonatal intensive care in the NHS was around 50% higher than for neonatal high- dependency care (£1810 vs. £1243) reflecting the more specialist, intensive and costly management.28 It is crucial therefore that the appropriate setting and level of care is identified for babies born at 27–31 weeks gestation. If care for less sick preterm babies is unnecessarily provided within NICU, there is an opportunity cost in terms of the health that is foregone by not using these resources in a more appropriate way. Equally, it is important to ensure that sicker preterm babies are not under-supported in LNU, as this could potentially result in prolonged intensive care and ultimately greater morbidity and mortality. At the time of writing and to the best of our knowledge, there have been no previously published analyses assessing the cost-effectiveness of different neonatal care settings for preterm births at any gestational age.

Parent perspectives on place of birth, care and transfers between units

The experience of giving birth to a preterm baby, and parenting a baby receiving neonatal care, can be extremely stressful and often has a negative psychological impact on parents.29,30 Parents experience moral distress related to caring for a sick baby, and stress associated with separation from their baby, loss of parental role,31 inconsistent staffing,32 transfers, and lack of communication and/or involvement in daily decision-making.33–35 Studies have documented the importance of parental (especially maternal) participation in caring for their baby;36 satisfaction has been associated with involvement in decision-making processes, respect and empathy from staff, attentive communication, and continuity of treatment and care.37,38 Within highly medicalised neonatal care facilities, and relevant to preterm babies born at 27–31 weeks gestation, ‘parenthood’ may be disrupted, with control no longer belonging to parents, and often significant parental trauma.39,40

Much of the qualitative research on experiences of preterm delivery and neonatal care has focused on transfers of babies between different levels of neonatal unit, within or between hospitals. Transfers occur not only in response to babies’ clinical needs, but may occur to maximise cot utilisation across neonatal networks.41 Although there is a lack of definitive evidence regarding the psychological impact of transfers on clinical outcomes for neonates,42 it is recognised that transfers are stressful and emotional for parents,43,44 create uncertainties, often separate parents from their baby, and disrupt parenthood.45 Effective communication between staff and parents about transfer processes can help parents feel more comfortable and in control of transfer decisions,43,46 but parents have reported feeling excluded from such decisions.42,46 Maternal stress has been related to maladaptive parenting and to long-term developmental problems in the child.32,47 There are minimal data on parental perspectives on place of care in the UK, and none specifically in relation to unit designation in very preterm babies.

Staff perspectives on care and place of care of very preterm babies

Staff perspectives are important to understand, to drive optimisation of care for this cohort. Staff also experience moral distress in caring for sick preterm babies, and organisational infrastructure is an important facilitator to enable nurses to develop trusting relationships with parents.48 Several studies have focused on nurses’ support for maternal closeness and attachment, which are important for both mothers and babies.49,50 Challenges for nurses include balancing the importance of facilitating and promoting closeness with the baby’s ability to handle stimulation,50 and managing the work involved in maximising parents’ involvement while also attending to the baby’s biomedical needs.51 Studies including both parent and staff perspectives have found that these may be at variance. In the context of very preterm babies, staff perspectives on place of birth and care have not been described.

Rationale for OPTI-PREM

A study addressing these aspects of neonatal care meet the following current NHS neonatal service delivery needs:

1. Response to new evidence about outcomes associated with place of neonatal care for preterm infants: Improved survival outcomes for those born at 23–26 weeks gestation when managed in a NICU/high-volume neonatal unit6,11 must drive investigation of whether similar benefits exist for very preterm babies (born at 27–31 weeks gestation), who are also high risk for neonatal mortality, morbidity and long-term adverse outcomes, and about whom very little is known.
2. Configuration and optimisation of neonatal services: The potential benefits and challenges of centralisation of neonatal care and how best to optimise it are the focus of discussions in neonatal health service delivery in England. To fully inform this dialogue, data are required on place of birth and care for all very preterm babies, not only those born at ≤ 26 weeks gestation.
3. Optimising cost-effectiveness of neonatal care: Differences in facilities, resources and staffing means there are likely to be inherent differences between LNU and NICU in costs and in the way in which care is provided for babies with similar problems at similar gestational ages. The impact of such variation on short- or long-term outcomes and, as a result, accurate estimates of cost-effectiveness need to be explored in any attempt to optimise neonatal service delivery.
4. Partnerships between parents and clinicians: Increasing importance is being placed on service user involvement in decision-making within the NHS and this is now an expectation of the public. Partnership with parents in decision-making about their infants’ care following preterm birth is important, but their voices have not yet been effectively heard. Both parents and clinicians have important insights because of their experience in navigating this complex and sensitive healthcare setting.
5. The need for national guidance: Despite NHS specifications E08/S/a,7 there is substantial variation between units and neonatal networks in the way that care is provided for very preterm babies. Evidence-based national recommendations from professional bodies do not exist.

Very preterm babies are a vulnerable, heterogeneous group, with a high mortality rate in the neonatal period and infancy. Currently in the UK they may be cared for in either LNU or NICU, but their optimum place of birth and care has not been defined. Unlike their more immature counterparts, there are no gestation-specific data detailing their mortality and morbidity when managed in LNU compared to NICU. Healthcare costs for these babies are unknown and the opinions and perspectives of parents and staff relating to place of birth have not been investigated.

This NIHR HS&DR project therefore set out to establish whether the designation (type) of unit in which neonatal care is delivered influences outcomes for these babies, and whether benefits of NICU care in relation to survival and morbidities that have already been identified for babies born at 23–26 weeks gestation extend to those born up to 31 weeks gestation. It also explored whether it is safer and more cost-effective for very preterm babies to be cared for in LNU, and whether there is a set point within this range at which care can equitably be provided in either LNU or NICU. Perspectives of staff and parents in co-contributing to strategies for optimal care were included throughout.

Aim

The overarching aim was to optimise neonatal service delivery for babies born at 27⁺⁰ to 31⁺⁶ weeks gestation.

Objectives

Primary objective:

• For preterm babies born at 27–31 weeks gestation and admitted into a neonatal unit for care (population), does care in NICU (intervention), when compared to care in LNU (comparator), result in improved gestation-specific survival (primary outcome) up to 1 year of age, and reduced neonatal major morbidities (secondary outcomes)?

Secondary objectives:

• For preterm babies born at 27–31 weeks gestation and admitted into a neonatal unit for care
  1. are there key differences in clinical care provided in LNU and NICU, and are these associated with differences in outcomes?
  2. where is it most cost-effective to care for these babies from an NHS perspective (LNU or NICU)?
  3. what are parents’ perspectives regarding place of care, and how can these be used to guide decision-making about place of care?
  4. what are LNU and NICU clinicians’ perspectives regarding place of care, and how can these be used to guide decision-making about place of care?

The study findings were intended to form the basis of discussions with the British Association of Perinatal Medicine (BAPM), the Neonatal Clinical Reference Group (CRG), Neonatal Specialised Commissioning services and the national charity for babies born premature or sick (BLISS)52 as relevant stakeholders to

• help develop recommendations for the optimal, most cost-effective place of care for babies born at 27–31 weeks gestation. BAPM, BLISS, The Royal College of Paediatrics and Child Health (RCPCH) and the Neonatal CRG were aware of, and supportive of, this work.