Research

• Stillbirth Overview
  • Introduction
  • Summary of key points
• Stillbirth and perinatal mortality statistics
  • Risk factors for stillbirth
  • Reduced fetal movements and stillbirth
• Causes and risk factors for stillbirth
• Unexplained stillbirth
• Investigation, audit and classification of stillbirth
  • Stillbirth audit and classification in Australia and New Zealand
  • Stillbirth investigation protocols - current evidence
  • PSANZ Stillbirth investigation protocol
  • Investigation of stillbirth in Australia
  • Costs of stillbirth investigations
• Autopsy examination
  • Value of autopsy
  • Autopsy quality
  • Decline in autopsy rates
  • Reasons for the decline
• Information and communication about perinatal autopsy
  • Pilot data autopsy communication and information and planned study
  • Support for parents
• References

Stillbirth Overview

Introduction

The purpose of this overview is to provide general information to enhance knowledge, understanding and awareness of stillbirth in Australia and New Zealand on the causes, risk factors, investigation, audit and classification and bereavement support for parents in the setting of maternity hospital care. This overview forms the basis of a more comprehensive report based on a systematic literature review which is currently being undertaken by ANZSA with funding provided by the Stillbirth Foundation Australia and the Department of Health and Ageing. The final report from the review will conclude by summarising the implications for practice and research toward the reduction of stillbirth in Australia and New Zealand. In addition to this review, a number of studies are currently underway which address current gaps in our understanding of stillbirth. For a summary of these studies please refer to the ANZSA stillbirth studies registry.

We hope you find this overview helpful and we welcome your feedback.

This overview was initially prepared by:
Vicki Flenady, Tomasina Stacey, Liz Davis, Emma Kirkwood, Ros Richardson, Yee Khong, Adrian Charles.
Last edited October 20th 2008.
 

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Summary of key points

While infant mortality rates have declined in Australia over the past two decades there has been no reduction in the rate of stillbirth. Stillbirths account for 70% of perinatal deaths. Over 2,000 babies are stillborn in Australia and New Zealand every year occurring at a rate of approximately 7 out of every 1000 births; approximately 6 babies each day.

Stillbirth rates for Indigenous women are over twice that of non-Indigenous women.

Major risk factors for stillbirth (many of which are modifiable) are: overweight and obesity, advanced maternal age (over 35), maternal smoking, and primiparity. Prolonged pregnancy is also a modifiable risk factor for antepartum stillbirth. The presence of fetal growth restriction in approximately 40-50% of unexplained stillbirth is an important consideration for future prevention strategies.

The main categories of stillbirth according to the Perinatal Society of Australia Perinatal Death Classification (PSANZ-PDC) system are: for singleton pregnancies, accounting for over 70% of stillbirths: Unexplained antepartum death (28%); Congenital abnormality (20%); Maternal conditions (13%); and Spontaneous preterm (10%). In multiple pregnancies (contributing 84% to the total): Specific perinatal conditions (mainly twin-twin transfusion) (35%); Spontaneous preterm (24%); Unexplained antepartum death (15%); and Congenital abnormality (11%).

It is estimated that unexplained stillbirth is now 10 times that of SIDS  500 unexplained stillbirths each year compared to 50 SIDS deaths. The high proportion of unexplained stillbirth is a major barrier to further reduction in the rates of stillbirth. The lack of diagnosis leaves little clues for parents and care providers struggling with decisions about future pregnancies.

Many stillbirths are not fully investigated and therefore important factors to further understand why the baby died may be missed. The recommended investigations within the PSANZ stillbirth protocol should be followed in all cases of stillbirth. Exclusion of the presence of infection is an important component of this protocol.

Autopsy and placental pathological examination remain the gold standard investigation for stillbirth. Parents who have a stillborn baby should be offered the option of an autopsy and given sufficient information (both written and oral) to enable them to make the decision which is right for them.

Stillbirth is devastating for families and one which is often accompanied by long lasting grief and loss of normal family functioning; the psychosocial burden of stillbirth to the family and the broader community should not be underestimated. While high level evidence on the best model of support for parents following a perinatal death is lacking, maternity hospitals need to ensure that mechanisms (including training and support for clinical staff and processes to ensure optimal transition to community support) are put in place to meet the individual needs of parents and families.

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Stillbirth and perinatal mortality statistics

For perinatal reports from across Australia and New Zealand please click here.

Globally, over 3 million babies are stillborn every year with the vast majority occurring in developing countries (1). While less frequent in developed countries (<1% of births), the large contribution of stillbirth to overall perinatal deaths combined with static or increasing rates over the past decade (2), means stillbirth remains a major public health problem in these settings.

In Australia for the year 2011, based on data from the National Perinatal Statistics Unit derived from State and Territory perinatal data collections (3) there were 301,810 births, and 2,992 perinatal deaths giving a perinatal mortality rate (PMR) of 9.9 per 1000 births. The perinatal mortality comprised of 2,220 fetal deaths, giving a fetal death rate (FDR) of 7.4 per 1000 births, and 772 neonatal deaths, giving a neonatal death rate (NDR) of 2.6 per 1000 livebirths (of 299,588 total livebirths). The PMR of babies born to Aboriginal or Torres Strait Islander mothers remains almost twice that of babies born to other mothers (18.6 versus 9.4) (3).

In New Zealand in 2004, there were 58,723 births and 666 perinatal deaths, giving a PMR of 11.2 per 1000 (8.5 and 3.4/1000 for fetal and neonatal death rates respectively) (4).

Differences in definitions and reporting processes across regions within Australia and New Zealand (ANZ) make comparisons of perinatal mortality rates difficult, and it is hoped that these differences will be addressed by the various reporting agencies. The NPSU report Australias Mothers and Babies also included the PMR for Australia calculated according to the Australian Bureau of Statistics (ABS) which is based on death certificate data for the same year. This rate was reported as 8.5 per 1000 (FDR 5.4, NDR 3.1). This difference is thought to be due to the differences in definition and reporting processes by the two sources with better ascertainment achieved by the perinatal data collections. At a round table meeting of ANZSA and NPSU in November 2007 a decision was made to include only stillbirth rates derived from the perinatal collections in the annual NPSU report Australias Mothers and Babies.

According to ABS data, the PMR in Australia declined by nearly two-thirds over the period from 1973 - 2000 from 23 per 1000 to the current rate of approximately 8 per 1000 (5). The fall in the neonatal death component (a 75% reduction from 12.6 to 3 per 1000) was greater than the fetal death reduction which fell by 50% from 11 to 5 per 1000 births. Fetal death after the onset of labour has decreased by two-thirds. Antepartum deaths decreased to a lesser extent (46%) (5) and currently make up approximately 65% of all fetal deaths (6). These patterns are similar to developed countries and have resulted in a focus of attention towards reducing the antepartum stillbirth rate.
 

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Causes and risk factors for stillbirth

The reduction in the PMR seen in earlier periods was largely due to improved intrapartum care resulting in less deaths from intrapartum asphyxia and birth trauma, and also a reduction in deaths from isoimmunisation and diabetes (7, 8). Congenital abnormality, unexplained fetal death and spontaneous preterm births have now emerged as the leading causes of perinatal death in developed country settings (9-12).

While different classifications systems make comparisons of the causes of stillbirth across and within countries difficult, the main reported causes of stillbirth internationally currently include: congenital abnormality, abruption, hypertension, diabetes and other maternal conditions, and maternal/fetal infection (8).

In approximately 30-50% of stillbirths, a cause of death is not identified (8, 13-15).

Data from a current ANZSA NHMRC funded study of 3,500 stillbirths over the period 2000-2003 in three States of Australia (Queensland, Victoria and Western Australia) demonstrates the main causes of death in an Australian population (15). In this large study, according to the Perinatal Society of Australia Perinatal Death Classification (PSANZ-PDC), the four major categories of stillbirth in singleton pregnancies, accounting for over 70% of stillbirths, were: Unexplained antepartum death (28%); Congenital abnormality (20%); Maternal conditions (13%); and Spontaneous preterm (10%). In multiple preganancies the main categories (contributing 84% to the total) were: Specific perinatal conditions (mainly twin-twin transfusion) (35%); Spontaneous preterm (24%); Unexplained antepartum death (15%); and Congenital abnormality (11%) (15).

Infection is acknowledged as an important contributor to stillbirth even when clinical markers for infection may not exist (16, 17). Although there are increasing research data to support the importance of infection in stillbirth, due to the lack of routine investigation for infection in many stillbirths, the magnitude of the contribution of infection is currently not well understood (16) and very few stillbirths in Australia are currently attributed to infection.

In pregnancy, thrombophilic disorders are associated with an increased risk of venous thromboembolism (VTE), pre-eclampsia, placental abruption, early and late fetal demise, recurrent pregnancy loss and fetal growth restriction (18, 19). While there is limited data on the strength of the association between inherited thrombophilic disorders and adverse pregnancy outcome such as stillbirth and controversy remains, recent reviews have demonstrated a statistically significant increase in the risk of stillbirth associated with: APC resistance(18, 20); Factor V Leiden mutation (18, 20-22); Protein C deficiency(20); Protein S deficiency (18, 20, 21); Prothrombin G20210 mutation (20, 21); and MTHFR (20). One review also demonstrated statistically significant associations with these thrombophilic conditions and pre-eclampsia which was strengthened in the analysis for severe pre-eclampsia (20). Maternal thrombophilic disorders are a potentially important cause of stillbirth and, as investigation for these conditions are often not routinely performed, it may be responsible for a proportion of unexplained stillbirth. Ideally the identification of thrombophilia following an apparently unexplained stillbirth would result in intervention in future pregnancies to reduce the risk (23).

Risk factors for stillbirth include: smoking, obesity, advanced maternal age (over 35) and primparity emerged as the most important risk factors for stillbirth (2). Prolonged pregnancy is also a modifiable risk factor for antepartum stillbirth, which affects about 1 per 1000 on-going pregnancies at 41 weeks, 1 in 500 at 42 weeks and 1 in 200 at 43 weeks. These risks can be avoided by routine induction post-dates (2).

Other reported factors which may increase the risk of stillbirth include: previous miscarriage,stillbirth or perinatal death and previous caesarean section (2), lower socioeconomic status (SES), alcohol and drug use (8, 24). In the USA, black women are at increased risk of stillbirth (8), as are Indigenous women in Australia where the risk is over twice that of non-Indigenous women (25).

Contributing factors relating to care (also called sub-optimal, avoidable or suspected preventable factors) have been reported in approximately 30-50% of perinatal deaths and therefore also require consideration as part of routine review of perinatal deaths by hospital committees. The report of the inquiry into Obstetrics and Gynaecology Services at the King Edward Memorial Hospital (26) highlighted the importance of clinical audit of perinatal deaths as part of ongoing clinical practice improvement.

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Reduced fetal movements and stillbirth

Regular fetal movement is widely accepted as a sign of fetal wellbeing. Almost all pregnant women perceive fetal movements and engage in self-screening by reporting their concerns of decreased fetal movements (DFM). Maternal reporting of DFM is a common pregnancy phenomenon occurring in between 4 - 15 percent of pregnancies in the third trimester . Pregnancies in which the woman consistently reports regular fetal movements have very low morbidity and mortality and conversely those with maternal reporting of DFM have been associated with stillbirth, fetal growth restriction, fetal distress, preterm birth, oligohydramnios and fetal abnormality mainly affecting the neurological or musculoskeletal system (27)

Fetal movement monitoring has been proposed as a screening tool to detect pregnancies at increased risk of late gestation sudden unexpected fetal death. This proposition is supported by the observation that as many as 50% of women perceive a gradual reduction of fetal movements several days before an intrauterine death (28-31).

However, there is little agreement as to what constitutes normal fetal movements (32-34) and the best methods for detection and management of a fetus at risk on the basis of DFM remains controversial (33). The use of maternal perception of DFM as screening tool is further complicated by the wide variation in the amount of fetal movements perceived by the women with reports ranging from 4-94% of movements identified by ultrasound.

In the late 1970s and early 1980s, formal fetal movement counting, which involves providing instructions to the woman about how to count and record movements, was a routine part of antenatal care in many settings. This practice reduced dramatically following the report, in 1989, of the single randomised controlled trial which concluded that formal monitoring of fetal movements did not reduce antepartum stillbirths and placed an increased burden on hospital resources (35). Current guidelines in the United Kingdom, the US or Norway do not recommended formal fetal movement counting. However, this remains somewhat controversial and others continue to recommend fetal movement counting as a part of routine antenatal care (28, 36, 37).

The NICE Guidelines in the UK, while not supporting formal fetal movement counting, do recommend that all women reporting DFM should be assessed for fetal wellbeing. However, no guidance is provided on what constitutes appropriate assessment. There is currently no randomised controlled trials evidence on any aspect of the initial evaluation or further management of pregnancies with decreased fetal movements. As a consequence, wide variation in management between populations, institutions and practitioners within single institutions is apparent 6;9;13;16;26;27;35-37 (+ ref Heazell) (38). The Cochrane systematic review on the topic (including the only available trial mentioned above) acknowledges the lack of evidence to inform practice and recommends further research to determine methods of detection of DFM and optimal management strategies (39).

A recent survey of Obstetricians in Australia and New Zealand (40) showed wide variation in practices in the detection and management of women with decreased fetal movement reflecting clinical uncertainty which stems from the current lack of high quality evidence. Approximately 30% of responders use Kick Charts as a part of routine care.

We are currently embarking on a large-scale prospective study across hospitals in Australia and New Zealand for raising DFM awareness using a mobile phone tool for pregnant women. See 'My Baby's Movements: a stepped wedge cluster randomised controlled trial of decreased fetal movement awareness to reduce stillbirth' on our Research Projects page.

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Unexplained stillbirth

With decreasing perinatal mortality rates, the relative contribution of antepartum fetal death where no cause for the death can be identified [unexplained antepartum fetal deaths (UAFD)] appears to be on the increase (11). The rates of UAFD reported internationally differ according to the definition used however, it is estimated at 1-2 per 1000 births (41). The wide variation in the reported contribution of unexplained stillbirth from 15% (42) to 71% (11) has been attributed to the classification system (11, 43) and the thoroughness of investigation and definition employed (41, 44). Lower rates of UAFD have been attributed to more stringent diagnostic criteria used, specifically autopsy and placental pathology examination (29).

Data from the current NHMRC grant has revealed a UAFD rate of 2.0 per 1 000 births (15). This rate is almost ten times the current rate of Sudden Infant Death Syndrome (SIDS) (45).

Using the number of ongoing pregnancies as the denominator, the risk of unexplained antepartum fetal death is shown to increase in late pregnancy (2, 46). Data from our current NHMRC grant has demonstrated that the contribution of UAFD increased after 23 weeks gestation reaching a maximum of 60% in term singleton stillbirths (37-41 weeks) and 49% at 35-36 weeks for stillbirth in a multiple pregnancy (47).

Several studies have been conducted both nationally and internationally to identify risk factors for UAFD. However, due to small numbers, differing methods including definitions and classification of fetal deaths, there is limited agreement on the reported risk factors. Factors which have been reported include: cigarette smoking in pregnancy, overweight or obesity (29); advanced maternal age (29, 48); low maternal age (31); advanced maternal age (29, 48, 49) duplicated?; primiparity and multiparity>3 (48); low socioeconomic status (29, 31, 48, 49); subclinical infection (31, 50); fetal growth restriction (11, 29, 49, 51, 52); sub-optimal antenatal care (11); polyhydramnios, cord problems, antenatal fetal distress (53, 54); rural origin, private accommodation status; higher systolic blood pressure (55); low blood pressure (56), and previous caesarean section (57).

The presence of fetal growth restriction in approximately 40-50% of unexplained stillbirth (13, 14) suggests the potential for prevention if these "at risk" infants can be detected in the antenatal period where appropriate intervention may be undertaken to reduce the risk.

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Investigation, audit and classification of stillbirth

Determining the causes of stillbirth is an essential part of quality care for parents and families. The purposes of investigations are to provide an explanation for the death of their baby to the parents and family who need to know what went wrong to relieve suffering, to enable appropriate counselling about recurrence risk, and to inform the management of future pregnancies. An accurate cause of death may assist in the parents grieving process by providing an explanation for the death and other information on the circumstances surrounding the death which may alleviate feelings of guilt (41, 46, 48, 52, 58). Even when a cause of death is not identified exclusion of some possible causes is valuable as a reassurance to parents and to inform the planning and management of future pregnancies (8).

Stillbirth audit and classification in Australia and New Zealand

The PSANZ-PMG has developed clinical practice guidelines for audit of perinatal deaths (23) which were disseminated across ANZ in 2005. The guidelines recommend comprehensive non-selective investigations for stillbirths and detailed instructions on the application of the clinical classification system for stillbirth and neonatal deaths. Due to the often complex nature of causation of stillbirth, the clinico-pathological correlation is best accomplished by discussion of a multidisciplinary team (11, 16, 23, 59-64) which includes obstetricians, paediatricians, neonatologists, perinatal pathologists, geneticists, midwives, and neonatal nurses. Therefore, the PSANZ guideline recommends review of all perinatal deaths by a multidisciplinary team.

Stillbirths first became notifiable in Scotland in 1940 (65), and in 1954 the classification developed by Sir Dugald Baird and his colleagues in Aberdeen for the purpose of audit and surveillance was published (66). Subsequently, numerous systems have emerged. In a recent search, we identified 33 new systems (7, 11, 41, 42, 66-94) and a further 12 modifications of these systems (11, 95-105) for the classification of stillbirths causes and associated conditions and/or suboptimal care

The majority were designed for both stillbirths and neonatal deaths, however three systems have been designed specifically for stillbirths (42, 83, 88), the remainder also included neonatal deaths and two systems included post-neonatal deaths; one up to hospital discharge (74) and the other up to twelve months of age (81). While it is important to analyse the causes of perinatal death according to its components of stillbirth and neonatal death (106), a system specifically designed to incorporate both groups enables interpretation of differences in the rates and causation across regions arising from variation in definition, reporting and registration practices for perinatal deaths (106).

In acknowledging the need and for a systematic approach to audit and review of perinatal deaths in Australia and New Zealand (ANZ), PSANZ endorsed the establishment of the Perinatal Mortality Group (PSANZ-PMG) (107) in March 2003. In the absence of an available classification system which suited the needs of this group, a new classification system (PSANZ Perinatal Death Classification (PSANZ-PDC) (69) was developed based on those used in Queensland and South Australia which were based on the Whitfield system (70).

The purpose of the classification is to determine the single most important obstetric factor which leads to the chain of events which resulted in the death and in doing so aims to identify avoidable stillbirth. The classification has been shown to have a high level of clinical agreement (108) and results in approximately 30% unexplained antepartum deaths (10) which has been suggested as a measure for an acceptable classification system (41). In a recent evaluation of six systems across seven countries, the PSANZ-PDC performed well (109).

Within this classification, fetal deaths prior to the onset of labour or membrane rupture where no cause for the death was able to be determined and, following review of all relevant available clinical information are assigned to the category of Unexplained antepartum death. However, unlike unexplained infant death (Sudden Infant Death Syndrome), the Unexplained antepartum death category is not dependent on the level of investigation and does not include the requirement for an autopsy examination. This may result in misclassification and overestimation of the contribution of UAFD (29).

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Stillbirth investigation protocols - current evidence

Formal protocols are intended to guide caregivers in stillbirth investigation, to ensure the quality and consistency of the investigation which will enable the validity of comparisons of trends across regions and over time. A recent systematic review of protocols, or component of protocols, for determining the causes of stillbirth by Corabian and Scott (110) identified five formal protocols currently in use, including the PSANZ stillbirth protocol (23). A comparison of the components of these protocols has identified some variation.

However, all protocols recommended placental pathology and autopsy. In the review of individual studies to determine appropriate investigations, the authors identified 53 studies potentially meeting the inclusion criteria. Of these, 46 were excluded due to lack of information in the published reports to adequately assess eligibility, poor methodological quality, or the definition of stillbirths which did not meet the inclusion criteria, leaving seven included studies (111-117). All included studies evaluated autopsy and placental pathology.

The results confirmed the value of these investigations. In the five retrospective studies (113-115, 117, 118) autopsy findings were shown to: confirm clinical findings in 29% to 89% of cases; change the diagnosis in 10% to 38%; and provide additional information in 4 to 24%. Despite this, the cause of death remained unexplained in up to 40% of stillbirths. In the two prospective studies (111, 116) placental pathology was shown to confirm clinical or autopsy findings or both in up to 75% of cases and were diagnostic in 23% to 46%.

The authors of this review concluded that, while autopsy and placental pathology were valuable, due to a lack of high quality data on the value of other investigations, "no formal scientific judgement could be made on which is the most appropriate protocol for stillbirth investigations or which components should be considered for the most relevant and efficient investigative protocol"(110).

Despite methodological concerns raised by Corabian and Scott, in recent years several studies on stillbirth protocols have consistently demonstrated that sufficient valuable information is obtained to justify the use of comprehensive investigation protocols (9, 61, 114, 119-123). While results for individual components of the investigation protocols across studies are conflicting, all studies have reported that formal protocols provide useful information (either a cause of death or additional valuable information for future pregnancy counselling) in approximately 30-50% of stillbirths.

However, these studies also report a substantial proportion of stillbirths remaining unexplained, ranging from 60% (61) to 36% (124). Differences in the components of the protocols, definition of stillbirth, the classification systems, and study design (population or hospital based) render comparisons and interpretation of the findings of these studies problematic. The two largest studies to date come from the Wisconsin Stillbirth Surveillance Program (61) and Incerpi et al (124). The WiSSP is a regional referral program for investigation of stillbirth which has reported prospectively collected population based data study on investigation and causes of death in over 1000 stillbirths (defined as gestation >20 weeks or over 375gms birthweight). This series has shown that using a non-selective approach to the investigation of fetal causes of death improved the yield of helpful information in 50% of stillbirths. The investigators reported that all individual components of the protocol yielded critical information, whereby exclusion of any single component would result in missed diagnoses in 2% (family history) to 15% (autopsy and placental pathology) of cases. However, the focus of the WiSSP protocol is fetal investigation and the extent to which maternal investigation (eg for infection, or other medical conditions) is undertaken is unclear apart from the Kleihauer-Betke test for maternal-fetal haemorrhage.

The other study (124), a retrospective study of over 700 stillbirths at a single institution, showed that while the use of a comprehensive protocol resulted in a high yield from autopsy and placental pathology, other investigations including the Kleihauer-Betke and maternal screening for congenital infection was low. The finding of low yield for the Kleihauer-Betke test is in conflict with other reports (122, 125). The overall autopsy rate in this series was 60%, and the proportion of unexplained stillbirth was 36%. A lower number of unexplained fetal deaths was reported for stillbirths with an autopsy than those without (31% v 44%). A hospital based study by Lim et al examined the value of a selective approach to investigation based on the presence of certain obvious causes of death (123). In this small series of 55 stillbirths with an autopsy rate of only 28%, the investigators reported that 38% of cases remained unexplained, similar to that of the non-selective approach.

There is currently no available data on the value of either a selective or non-selective protocol for investigation of stillbirths in an Australian setting. A large-scale prospective study is currently under way across hospitals in Australia and New Zealand. See 'ANZSA Investigating Causes of Stillbirth: a prospective cohort study examining use and effectiveness of a comprehensive investigation protocol' on our Research Projects page.
 

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PSANZ Stillbirth investigation protocol

The PSANZ guidelines recommend a comprehensive non-selective stillbirth investigation protocol. While it is acknowledged that compliance with the protocol may depend on the particular circumstances of the death (e.g. family wishes and access to services), using a selective investigative approach may result in missed important diagnoses (61), therefore the guideline recommends that clinicians adopt the non-selective approach as the standard and debate the relative merits of not following this approach on an individual case basis. Unfortunately, due to a lack of information about the value of many of the recommend investigations, these investigations are largely based on consensus of the guideline development working party and not evidence. As a result, there is some clinical controversy about the recommendations and unwillingness by clinicians to follow the recommendations. Further research is urgently need to determine the best approach to investigation of stillbirth.

Investigation of stillbirth in Australia

While no information is available on the yield of investigations for stillbirth in ANZ, preliminary data from the current ongoing NHMRC study on almost 200 stillbirths provides some indication of current practices in investigation. In three states of Australia over the period 2000-2003 (pre dissemination of the PSANZ guidelines), a low rate for a number of the currently recommended core investigation for stillbirths is evident as follows: autopsy 45% (54% for unexplained stillbirths); placental histopathology 45%; vaginal cultures 5%; amniocentesis for culture <1%, amniocentesis for chromosomal analysis <2%; Kleihauer-Betke 60%; external examination of the baby by clinician 60%; Clinical photographs 30%; Glycosolated haemoglobin (HBA1c) 40%; maternal full blood count and blood group and antibodies 75%, thrombophilia screening 20% (126). The extent to which this apparently low level of investigation is artificially inflating the number of unexplained stillbirths in Australia is not known.

Costs of stillbirth investigations

There is very limited information currently available to enable consideration of the cost benefit of stillbirth investigation protocols, or their individual components. Michalski et al (120) reported a cost consequence analysis from the Wisconsin Stillbirth Service Program (WiSSP) (61) which showed the real cost of a comprehensive stillbirth protocol was $1450USD per assessment or approximately $12USD per cared-for pregnancy. In this series of almost 1500 stillbirths, new relevant information was identified in 51% of stillbirths as a result of the protocol. The authors concluded that the comprehensive protocol should become a part of routine antenatal care. Another study (123) estimated that a selective approach to investigation of stillbirths would reduce the costs of investigation in 30% of cases without compromising the yield of investigation. There is currently no data available on costs to enable consideration of the cost benefit of stillbirth investigations in an Australian or New Zealand setting.

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Autopsy examination

Value of autopsy

The autopsy examination is the gold standard for identification of the cause of perinatal death (127); diagnostic utility of the perinatal autopsy has been repeatedly demonstrated (128). One comprehensive review of 27 studies found perinatal autopsy to change diagnosis or provide additional findings in 22-76% of cases (128). In another review of 53 studies across a range of health care settings, diagnostic errors detected at perinatal autopsy showed a median error rate in pre-autopsy diagnosis of 23.5% for major errors (clinically missed diagnoses involving a principal underlying disease or primary cause of death) and 9% for Class 1 errors (major error that, had they been detected during life, "would", "could", "possibly" or "might" have affected patient prognosis or outcome).

The recent systematic review of stillbirth investigations (110) found that while the evidence base for many recommended investigations is in doubt, autopsy and placental pathology make a valuable diagnostic contribution. The reviewers concluded that data derived from the review may be "helpful in counselling parents who are considering the difficult decision of whether or not to consent to a post-mortem examination following a stillbirth" (110). In addition to the value of information obtained at autopsy in the planning and management of future pregnancies (129) autopsy may have long-term psychological implications for parents. Parents, mothers in particular, often attribute the death of their child to their own actions (130, 131). Autopsy findings may give reassurance that they are not to blame and assist in grief integation (130). Some parents may regret the decision to have an autopsy, particularly when no cause of death is identified, and some may find comfort in an altruistic perspective where no cause is found after autopsy examination (131).

A study in WA by Dr Buccilli and Charles confirmed the findings of the value of the perinatal autopsy in an Australian context (132). In this study, a total 98 of the 167 stillbirths (58.7%) had an autopsy performed. Examination of the quality of autopsy, using a modified scoring system (117), revealed that in 25% the autopsy confirmed clinical diagnoses; in 21% the autopsy findings changed the diagnosis; in 31% some extra information to the original diagnosis was identified; and in 23% the findings were inconclusive. A more recent study from Western Australia of 1619 fetal deaths revealed 49% having had complete autopsy investigation. Following autopsy 22% of these deaths were explained and a further 18% were identified as having fetal growth restriction. In addition, of the 42% of deaths which were unexplained on the death certificate, 65% were explained after autopsy investigation (44). Based on these findings, it is evident that the cause of death recorded on perinatal death certificates without an autopsy are often misleading and therefore may jeopardise the quality of research and audit activities and subsequent public health policy based on these data (118, 133-135).

Perinatal autopsy is important for education, training and to ensure high standards in perinatal pathology (136-138)

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Autopsy quality

There is limited research on the quality of perinatal autopsies however, the available data suggests that approximately half may not reach minimum standards (62, 139). The post-mortem examination of an infant is very different to that performed on an adult (8, 16, 63, 64), and ideally should be performed by a paediatric pathologist. Pathologists with paediatric training find a higher incidence of causes of death in infants (112) and provide a much higher proportion of adequate reports (139, 140). The Confidential Enquiry into Stillbirth and Deaths in Infancy (CESDI) in the United Kingdom have reported that the causes of death based on perinatal/paediatric pathologists reports are infrequently revised (11). Cartlidge et al demonstrated an association with higher quality autopsy and the number of perinatal and infant deaths where the main cause of death was identified (112). Understandably, the ethics of approaching parents for consent where a quality post-mortem service is not available has been questioned (127). There are currently no guidelines for ANZ on quality and minimum standards for perinatal autopsies.

Decline in autopsy rates

An optimal perinatal autopsy rate of 75% is recommended by the Royal College of Pathologists (136). However, the perinatal autopsy rate has steadily declined over recent years in many regions. Perinatal autopsy rates vary across jurisdictions in Australia. The rates in our previous study across three States in 2000 were 39%, 48% and 70% overall, and 96%, 59% and 62% for unexplained antepartum deaths (10). In our current NHMRC funded population based study, the overall unexplained stillbirth autopsy rate in singletons over the period 2000-2003 was 45%; consistent across the three participating states. In Queensland, the autopsy rate for stillbirths has declined by 50% from the period 1997-2003 to the current rate of 30% (141). The recent perinatal autopsy rate in Tasmania was 7% (142).

Reasons for the decline

While research into the determinants of the decreasing perinatal autopsy rate is limited, consent is considered a major factor (137, 143, 144). Adverse publicity following the inquiries into autopsy practices in the United Kingdom and the NSW Institute of Forensic Pathology (129) are thought to have affected clinicians willingness to seek consent and parents acceptance of the procedure (130). Although the inquiries led to improved practices, complexity in the consent process also increased and this may be a deterrent to both clinicians and parents (145). Workforce shortages in clinical care settings and pathology are also limiting adequate autopsy rates (138).

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Information and communication about perinatal autopsy

All hospital perinatal autopsy examinations require written consent from the parents following informed discussion(138). However, consent for autopsy is difficult for both clinicians and parents. Parents face an apparently intrusive process and are required to understand detailed consent procedures in a state of grief and clinicians are reluctant to place further burden on the parents (138, 146). Parents may think an autopsy will mutilate their baby (131), that their baby has suffered enough or the parents may have religious objections (130). In most cases, the medical consultant will approach the parents for consent (131), however, midwives, nurses, social workers and pathologists often have a role in providing information to assist in decision-making (130, 131, 147).

Two surveys have examined attitudes of staff to perinatal autopsy (143, 148). Both studies indicate that the attitudes of staff and the experience level of staff exert important influences on consent to autopsy. The Australian survey of staff by Khong et al (143) showed that while obstetricians and neonatologists were not averse to seeking consent midwives and neonatal nurses were reluctant to do so. Obstetricians and neonatologists rated nurses and midwives as influential in parents decisions around autopsy. However, clinicians report feeling inadequate talking to parents about autopsy, and may avoid discussion due to compassion and their lack of confidence to provide care following a perinatal death (137, 143). Research into parents perspectives and experiences on perinatal autopsy is very limited. However, it does appear that parents may regret their decision about autopsy and that this may be related to inadequate information and communication at the time of consent (131).

Two small studies have reported the frequency of parents regret about the decision for autopsy on their baby or infant with conflicting results (130, 131). McHaffie et al, in interviews with parents after a neonatal death, reported that no parent expressed regret about the decision. However, in the survey by Rankin et al (130) of 148 women after pregnancy or infant loss (including miscarriage, termination, stillbirth, neonatal or post-neonatal death), approximately 10% regretted their decision; more parents who had declined an autopsy regretted the decision than parents who had agreed. This survey also showed that 80% of parents would agree to a post-mortem examination if asked (130). Therefore, clinician reluctance to seek consent due to compassionate reasons may be misplaced.

Other factors which may affect clinician willingness to approach parents for consent for paediatric and perinatal deaths include: lower gestation at death (143, 149); discipline and seniority of clinicians (148); ambivalence about the value of an autopsy (112); and lack of technical understanding of the autopsy procedure. Parents enduring powerful negative experiences such as the stillbirth of their baby may not recall information given to them (150) which may lead to negative feelings towards care providers for poor communication increasing parental distress. Staff need specific training to know how to respond appropriately to bereaved parents following stillbirth (151).

The paucity of well conducted research on communication and consent for autopsy in stillbirths does not permit reliable conclusions to be drawn. Major limitations of the current research includes: small numbers studied; combining the populations of stillbirths and neonatal deaths; and the use of parent groups as the sampling frame which may result in an overestimate of negative outcomes and experiences of parents (152). The situation of a stillbirth (often an unexpected event in the antenatal period) differs significantly from that of a neonatal death (often death occurs after a period of time in an intensive care nursery). Therefore, it stands to reason that communication and information needs of parents are quite different and each are deserving of focused research.
 

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Pilot data autopsy communication and information and planned study

Pilot data on experiences and views of parents about autopsy consent were obtained through the consultation process to update the Perinatal Society of Australia and New Zealand (PSANZ) Mortality Audit Guidelines. The views and experiences of bereaved parents and their satisfaction with information provided to them around the time of their stillbirth were gathered by Stillbirth and Neonatal Death Support (SANDS) Queensland and the NSW Stillbirth Foundation. Seventeen parents (14 mothers and three fathers) identified through the SANDS newsletter and by word of mouth attended three focus groups. All parents experienced late gestation stillbirths. The majority had consented to an autopsy but the cause of death remained unexplained.

Parents frequently commented on their inability to make an informed decision about autopsy due to overwhelming shock and grief and their dependence on others in the decision making process. One parent did not understand that the investigation was optional, and some participants stated feeling dependent on professionals for their decision. Some parents felt it was the responsibility of the care provider to make the decision on their behalf, comparing this to other emergency type decisions which are made in their best interests (eg emergency caesarean section) and felt let down by the care provider. Others felt vulnerable due to perceptions of lack of objectivity from their care provider, resulting from possible concerns about clinical error, and would have preferred to discuss the option of autopsy with a third party.

The need to involve others such as their babys grandparents and friends in interpreting information was important to parents. All agreed that written, as well as verbal information, about an autopsy was important, but eight parents had no recollection of receiving written information. Three parents did not recall any conversation with their care providers about the option of autopsy. This appeared to be influenced by presenting the decision as urgent and giving inadequate information. Negative feelings towards autopsy included: fear of mutilation; limiting the available time to spend with the baby; and concerns that an autopsy could indicate some blame to the mother for "doing something wrong". Some parents said that the autopsy, even when it did not reveal a cause for the death, helped them in "moving forward". All parents who did not have an autopsy expressed regret or some doubt about their decision. No parent who had an autopsy performed expressed these feelings.

Parents frequently raised concerns about clinical staff seeming inadequate in coping with the event of stillbirth. They commented that some staff seemed "scared" and did not appear to have an understanding of how parents respond to loss of a baby. Many parents suggested the need for special training of health care providers in communicating with parents after a stillbirth. Satisfaction with information and counselling about autopsy appeared to be related to trust in the heath care provider and displaying compassion and understanding of what the parents were experiencing.

Current research to inform best practise on the information needs and consent practices for autopsy around the time of stillbirth is limited. Lack of appropriate care may have long term consequences for bereaved parents. These pilot data, despite being limited by possible volunteer bias, support the need to improve care in relation to autopsy consent.

A large-scale study, which is currently being piloted, aims to identify factors that contribute to the low autopsy consent rate for stillbirths and will provide robust information to develop information and educational materials that address the needs of parents and clinicians.

Acknowledgment: We wish to acknowledge the parents who participated in the focus groups.
 

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Support for parents

The death of an infant is among the most stressful of life events (153) and bereaved parents may experience acute emotional distress (153). Initial responses to an infants death include numbness, confusion, depersonalisation and disbelief and shock. This may be an adaptive mechanism to temporarily protect parents from the full impact of their babys death, however, mothers may continue to experience feelings of depersonalisation up to four years later (153). Bereaved fathers may deny the emotional impact of the experience (153), with up to 10% exhibiting "almost complete denial". Symptoms of depression may also continue for two years or more (153).

While grief after a stillbirth is a normal reaction, recent research has suggested that stillbirth may be associated with Post Traumatic Distress Disorder (PTSD) (154). Factors which have been reported to increase the risk of adverse psychological outcomes for parents after a perinatal death include: perceived inadequate social support, traumatic circumstances surrounding the death, difficulties in coping with a crisis in the past, problematic relationships in the nuclear family and the presence of other life crises. In addition, mothers report greater distress than do fathers.

A recent Cochrane review on support interventions for families after a perinatal death (155) concluded that there is insufficient evidence from randomised controlled trials to provide reliable information on which to base practice. While providing support for parents and families after a perinatal death is justified based on other types of research, there is insufficient evidence to confirm or refute the benefit of the different types of interventions aiming to provide such support.

This overview was initially prepared by :
Vicki Flenady, Tomasina Stacey, Liz Davis, Ros Richardson, Yee Khong, Adrian Charles.
Last edited October 20th 2008.
 

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