Infant Sleep Hazards and the Risk of Sudden Unexpected Death in Infancy.

OBJECTIVE: To examine the effects of infant sofa-sleeping, recent use by caregivers of alcohol, cannabis, and/or other drugs, and bed type and pillows, on the risk of sudden unexpected death in infancy (SUDI) in New Zealand. STUDY DESIGN: A nationwide prospective case-control study was implemented between March 2012 and February 2015. Data were collected during interviews with parents/caregivers. "Hazards" were defined as infant exposure to 1 or more of sofa-sleeping and recent use by caregivers of alcohol, cannabis, and other drugs. The interaction of hazards with tobacco smoking in pregnancy and bed sharing, including for very young infants, and the difference in risk for Maori and non-Maori infants, also were assessed. RESULTS: The study enrolled 132 cases and 258 controls. SUDI risk increased with infant sofa-sleeping (imputed aOR [IaOR] 24.22, 95% CI 1.65-356.40) and with hazards (IaOR 3.35, 95% CI 1.40-8.01). The SUDI risk from the combination of tobacco smoking in pregnancy and bed sharing (IaOR 29.0, 95% CI 10.10-83.33) increased with the addition of 1 or more hazards (IaOR 148.24, 95% CI 15.72-1398), and infants younger than 3 months appeared to be at greater risk (IaOR 450.61, 95% CI 26.84-7593.14). CONCLUSIONS: Tobacco smoking in pregnancy and bed sharing remain the greatest SUDI risks for infants and risk increases further in the presence of sofa-sleeping or recent caregiver use of alcohol and/or cannabis and other drugs. Continued implementation of effective, appropriate programs for smoking cessation, safe sleep, and supplying safe sleep beds is required to reduce New Zealand SUDI rates and SUDI disparity among Maori.

Late stillbirth post mortem examination in New Zealand: Maternal decision-making.

BACKGROUND: For parents who experience stillbirth, knowing the cause of their baby's death is important. A post mortem examination is the gold standard investigation, but little is known about what may influence parents' decisions to accept or decline. AIM: We aimed to identify factors influencing maternal decision-making about post mortem examination after late stillbirth. METHODS: In the New Zealand Multicentre Stillbirth Study, 169 women with singleton pregnancies, no known abnormality at recruitment, and late stillbirth (≥28weeks gestation), from seven health regions were interviewed within six weeks of birth. The purpose of this paper was to explore factors related to post mortem examination decision-making and the reasons for declining. We asked women if they would make the same decision again. RESULTS: Maternal decision to decline a post mortem (70/169, 41.4%) was more common among women of Maori (adjusted odds ratio (aOR) 4.99 95% confidence interval (CI) 1.70-14.64) and Pacific (aOR 3.94 95% CI 1.47-10.54) ethnicity compared to European, and parity two or more (aOR 2.95 95% CI 1.14-7.62) compared to primiparous. The main reason for declining was that women 'did not want baby to be cut'. Ten percent (7/70) who declined said they would not make this decision again. No woman who consented regretted her decision. CONCLUSION: Ethnic differences observed in women's post mortem decision-making should be further explored in future studies. Providing information of the effect of post mortem on the baby's body and the possible emotional benefits of a post mortem may assist women faced with this decision in the future.

Rethinking placental pathology in the PSANZ classification of unexplained stillbirth at term.

BACKGROUND: Approximately 30 per cent of stillbirths are currently classified 'unexplained' using the Perinatal Society of Australia and New Zealand (PSANZ) classification system in New Zealand. This unexplained category includes deaths with placental pathology even though the importance of placental pathology and its causal relationship to stillbirth is well described. AIMS: To determine whether unexplained stillbirths in New Zealand classified using PSANZ criteria can be more usefully classified based on placental pathology. METHODS: Audit of the classification of cause of death among 'unexplained antepartum death' at term by perinatal pathologist review of postmortem and/or placental pathology reports using the current PSANZ Perinatal Death Classification (PDC)10 classification and a proposed 'significant placental pathology' subclassification. The main outcome measure was a change in cause of death from unexplained term stillbirth to an alternative PSANZ classification or to significant placental pathology subcategory. RESULTS: In total, 177 unexplained stillbirths with a postmortem and/or placental pathology report in New Zealand between 2007 and 2013 inclusive were reviewed. Twenty-three cases (13%) had significant placental pathology that could have been a direct cause of the stillbirth. A further seven cases (4%) were misclassified and could be better classified within another PDC category. CONCLUSIONS: A classification system incorporating placental pathologies which are recognised by the current literature to be causative of stillbirth would better describe stillbirths at term in New Zealand. This would benefit parental counselling and follow-up in subsequent pregnancies. A standard approach to reporting placental pathology would benefit clinicians. Education on placental pathology for clinicians working with parents experiencing stillbirth and multidisciplinary approach to classification is also recommended.

Fetal megacystis: Institutional experience and outcomes.

BACKGROUND: Fetal megacystis is a sonographic feature that may be indicative of several underlying pathologies. Despite advances in diagnosis and management, the overall prognosis of affected fetuses remains poor and about 50% of such pregnancies are terminated. AIMS: To define the frequency, management, survival and renal outcomes of fetal megacystis over nine years at Wellington Hospital, New Zealand. MATERIALS AND METHODS: A nine-year retrospective review of fetuses with an antenatal diagnosis of megacystis was undertaken. RESULTS: Sixteen cases were identified (nine live births, five terminations and two perinatal deaths). This gives an observed frequency of one per 940 fetuses scanned. Two-thirds of the live births have survived and been followed for a mean of 5.3 years. None have required renal dialysis or transplantation to date. CONCLUSIONS: The current series contributes to our knowledge of fetal megacystis and helps to inform antenatal counselling. Improved prognostic criteria are urgently required to accurately differentiate between fetuses with favourable versus poor outcomes.

Valved shunt as a treatment for obstructive uropathy: does pressure make a difference?

PURPOSE: A valved ventriculo-peritoneal shunt (V-P shunt) as a vesico-amniotic shunt (V-A shunt) preserves the filling/emptying cycle and normal bladder development in fetal lambs with bladder outlet obstruction. The optimal pressure for such shunts is unknown. MATERIALS AND METHODS: We created obstructive uropathy in 60-day gestation fetal lambs. A V-A shunt was placed 3 weeks later, using a low-pressure (Group L: 15-54 mmH2O) or a high-pressure (Group H: 95-150 mmH2O) V-P shunt. We included non-shunted (obstructive uropathy, Group O) and control lambs (Group C). All were delivered at 130 days. Bladder volumes, bladder thickness, renal and bladder histology were compared. RESULTS: Seventeen lambs had an obstructive uropathy created. Five Group L (four survived), four Group H (three survived) and five Group O survived. Body weight and crown-to-rump lengths of the three groups were not significantly different. Group H lambs had a dilated urachus, urinary ascites and severe ureteral dilatation similar to Group O lambs. There were four Group C lambs. Bladder volume was 10, 15 and 1,150 ml in Group H, 115 ± 67.9 ml in Group L, 128 ± 99.8 ml in Group O and 24.5 ± 3.84 ml in Group C. Unlike Group O lambs, Group L did not have urinary ascites, urinomas or renal dysplasia. CONCLUSION: Low-pressure shunts preserved both bladder volume and renal development. High-pressure shunts did neither.

Gliosis in neonatal SUDI cases.

AIM: To review sudden unexpected infant deaths (SUDI) in the first 28 days of life referred to a Coronial Perinatal Forensic Pathology Service over a 10-year period from 2000 to 2009. METHODS: Cases were collected from mortuary records, and a retrospective review of autopsy reports and other available infant records was undertaken. RESULTS: Twenty-four neonatal SUDI were reviewed. For eight infants, a diagnosis was made at autopsy. For the remaining 16 infants, 14 (87.5%) were bedsharing at the time of death. Maori infants and those living in deprived neighbourhoods were over-represented. Only two infants were preterm, and four were growth-restricted. At post-mortem, white matter gliosis was found in 10 of the 16 (62.5%) unexplained SUDI cases. CONCLUSION: Sudden unexpected infant deaths occur in the first month of life in association with bedsharing. Gliosis may be an important associated risk factor, and its presence indicates a previous insult of prenatal onset.

Antenatal care, identification of suboptimal fetal growth and risk of late stillbirth: findings from the Auckland Stillbirth Study.

INTRODUCTION: Stillbirth remains an important public health problem in Australia and New Zealand. The role that antenatal care plays in the prevention of stillbirth in high-income countries is unclear. METHODS: Cases were women with a singleton, late stillbirth without congenital abnormality, booked to deliver in the Auckland region and born between July 2006 and June 2009. Two controls with ongoing pregnancies were randomly selected at the same gestation at which the stillbirth occurred. Data were collected through interview-administered questionnaires and from antenatal records. RESULTS: One hundred and fifty five of 215 (72%) cases and 310 of 429 (72%) controls consented to take part in the study. Accessing <50% of recommended antenatal visits was associated with a more than twofold increase in late stillbirth (adjusted odds ratio, aOR, 2.68; 95% CI, 1.04-6.90) compared with accessing the recommended number of visits. Small-for-gestational-age (SGA) babies that had not been identified as SGA prior to birth were significantly more at risk of being stillborn (aOR, 9.46; 95% CI, 1.98-45.13) compared with SGA babies that were identified as such in the antenatal period. No relationship was found between type or model of maternity care provider at booking and late stillbirth risk. DISCUSSION: This study reinforces the importance of regular antenatal care attendance. Identification of SGA may be one way by which antenatal care reduces stillbirth.

Relationship between obesity, ethnicity and risk of late stillbirth: a case control study.

BACKGROUND: In high income countries there has been little improvement in stillbirth rates over the past two decades. Previous studies have indicated an ethnic disparity in the rate of stillbirths. This study aimed to determine whether maternal ethnicity is independently associated with late stillbirth in New Zealand. METHODS: Cases were women with a singleton, late stillbirth (≥ 28 weeks' gestation) without congenital abnormality, born between July 2006 and June 2009 in Auckland, New Zealand. Two controls with ongoing pregnancies were randomly selected at the same gestation at which the stillbirth occurred. Women were interviewed in the first few weeks following stillbirth, or at the equivalent gestation for controls. Detailed demographic data were recorded. The study was powered to detect an odds ratio of 2, with a power of 80% at the 5% level of significance, given a prevalence of the risk factor of 20%. A multivariable regression model was developed which adjusted for known risk factors for stillbirth, as well as significant risk factors identified in the current study, and adjusted odds ratios and 95% confidence intervals were calculated. RESULTS: 155/215 (72%) cases and 310/429 (72%) controls consented. Pacific ethnicity, overweight and obesity, grandmultiparity, not being married, not being in paid work, social deprivation, exposure to tobacco smoke and use of recreational drugs were associated with an increased risk of late stillbirth in univariable analysis. Maternal overweight and obesity, nulliparity, grandmultiparity, not being married and not being in paid work were independently associated with late stillbirth in multivariable analysis, whereas Pacific ethnicity was no longer significant (adjusted Odds Ratio 0.99; 0.51-1.91). CONCLUSIONS: Pacific ethnicity was not found to be an independent risk factor for late stillbirth in this New Zealand study. The disparity in stillbirth rates between Pacific and European women can be attributed to confounding factors such as maternal obesity and high parity.

Maternal perception of fetal activity and late stillbirth risk: findings from the Auckland Stillbirth Study.

BACKGROUND: Maternal perception of decreased fetal movements has been associated with adverse pregnancy outcomes, including stillbirth. Little is known about other aspects of perceived fetal activity. The objective of this study was to explore the relationship between maternal perception of fetal activity and late stillbirth (≥28 wk gestation) risk. METHODS: Participants were women with a singleton, late stillbirth without congenital abnormality, born between July 2006 and June 2009 in Auckland, New Zealand. Two control women with ongoing pregnancies were randomly selected at the same gestation at which the stillbirth occurred. Detailed demographic and fetal movement data were collected by way of interview in the first few weeks after the stillbirth, or at the equivalent gestation for control women. RESULTS: A total of 155/215 (72%) women who experienced a stillbirth and 310/429 (72%) control group women consented to participate in the study. Maternal perception of increased strength and frequency of fetal movements, fetal hiccups, and frequent vigorous fetal activity were all associated with a reduced risk of late stillbirth. In contrast, perception of decreased strength of fetal movement was associated with a more than twofold increased risk of late stillbirth (aOR: 2.37; 95% CI: 1.29-4.35). A single episode of vigorous fetal activity was associated with an almost sevenfold increase in late stillbirth risk (aOR: 6.81; 95% CI: 3.01-15.41) compared with no unusually vigorous activity. CONCLUSIONS: Our study suggests that maternal perception of increasing fetal activity throughout the last 3 months of pregnancy is a sign of fetal well-being, whereas perception of reduced fetal movements is associated with increased risk of late stillbirth.

The Auckland Stillbirth study, a case-control study exploring modifiable risk factors for third trimester stillbirth: methods and rationale.

BACKGROUND: In high-income countries, stillbirth rates have been static in recent decades. Unexplained stillbirths account for up to 50% of these deaths. METHODS: A case-control study was conducted in Auckland, New Zealand, from July 2006 to June 2009 to explore modifiable risk factors for late stillbirth (≥28 weeks of gestation). Eligible participants were women who had a singleton late stillbirth without a congenital abnormality. Two controls with ongoing pregnancies were randomly selected at the same gestation as each case. Data were collected through face-to-face interviews and from clinical records. RESULTS: A total of 155/215 (72%) cases and 310/429 (72%) controls consented to take part in the study. Women who had a late stillbirth were more likely to be of Pacific ethnicity and of parity ≥4 (OR = 1.7, 95% CI: 1.1-2.6 and 2.7, 95% CI: 1.4-5.3, respectively). The median gestational age at diagnosis of fetal death was 261 days (IQR 239-279), and the median gestation at which the controls were interviewed was 264.5 days (IQR 240-274) P = 0.48. 'Unexplained antepartum death' (n = 61, 39.4%) and 'fetal growth restriction' (n = 29, 18.7%) accounted for almost 60% of stillbirths. The post-mortem rate for all cases was 47% (73/155) and 43% (26/61) for those classified as 'unexplained antepartum death'. CONCLUSION: This study of risk factors for stillbirth is novel in that it used gestation-matched controls with ongoing pregnancies. Its detailed investigation into maternal health and behaviour during pregnancy has the potential to lead to a better understanding of modifiable risk factors for late stillbirth.

Simultaneous sudden unexpected death in infancy of twins: case report.

Simultaneous sudden infant death syndrome (SIDS) in twins is an extremely rare event. Some believe these are natural deaths, whereas others suggest they are unnatural. We report monozygotic twins dying at 12 weeks of age. Extensive investigation concluded that the cause of death was natural. Our case fulfilled the criteria of simultaneous SIDS in twins. However, thermal stress due to excessive thermal insulation, use of a pillow and placing twins in the same cot all represent a potentially unsafe sleeping environment. We recommend the term "simultaneous sudden unexpected death in infancy of twins" to describe such cases.

Forensic issues in cases of water birth fatalities.

Birth under water has become a widely disseminated technique that is promoted to improve the quality of labor. The case of a 42-week gestation male infant is reported who died of respiratory and multiorgan failure secondary to florid pneumonia and sepsis due Pseudomonas aeruginosa following a water birth. Other infants who have been delivered underwater have drowned or have had near-drowning episodes with significant hyponatremia and water intoxication. Local and disseminated sepsis has been reported, with respiratory distress, fevers, hypoxic brain damage, and seizures. There have also been episodes of cord rupture with hemorrhage. The postmortem investigation of such cases requires a complete autopsy of the infant, with examination of the placenta. Full details of the pregnancy and delivery and inspection of the birthing unit are also needed. A septic workup of the infant and placenta should be undertaken along with sampling of water from the birthing unit and microbiological swabbing of the equipment. Vitreous sodium levels may reveal electrolyte disturbances. While fatal cases appear rare, this may change if water births gain in popularity.

Risk factors for and timing of death of extremely preterm infants.

This study examined risk factors for timing and cause of death for extremely preterm infants > or = 23 weeks and < 28 weeks. There were 479 liveborn infants and 98 deaths reviewed over a ten-year period. Thirty-two deaths (33%) occurred on the first day of life and 72 (75%) in the first month of life. Lower gestation and intrauterine growth restriction were significant risk factors for death. Most deaths occurred in the first month of life and at the lowest gestation in the first week.

Placenta accreta in the first trimester of pregnancy: sonographic findings.

In a case of histologically confirmed placenta increta, decidual protrusion into the myometrium was observed sonographically at 6 weeks' gestation, corresponding to placental protrusion from a disrupted placental-uterine wall interface seen in the later part of the first and second trimester. It is hypothesized that the histologic finding of decidual scarcity in placenta accreta is not a cause but rather an end result of the recruitment of trophoblasts across the decidual-placental interface in a maternal attempt at healing and/or repair in the presence of uterine injury, disease, or malformation that accounts for the associated factors and course of the condition.

Juvenile Alpers disease.

BACKGROUND: Alpers disease is commonly associated with polymerase gamma deficiency and usually affects infants or young children. OBJECTIVE: To report a juvenile case of Alpers disease due to mutations in the polymerase gamma gene (POLG1). DESIGN: Clinical, pathologic, biochemical, and molecular analysis. SETTING: Tertiary care university hospital and academic institutions. PATIENT: A 17-year-old adolescent girl with intractable epilepsy and liver disease. MAIN OUTCOME MEASURES: Clinical course and pathologic, biochemical, and molecular features. RESULTS: Biochemical and pathologic evidence suggested a respiratory chain defect, which was confirmed by enzyme analysis of the liver. Mutational analysis of POLG1 showed 2 novel mutations: T851A and R1047W. CONCLUSION: The POLG1 mutations can cause juvenile and childhood Alpers disease.

Antenatal topographical assessment of placenta accreta with ultrasound.

The major risk of placenta accreta is severe haemorrhage when the placenta is separated at delivery. A case series of placenta accreta with antenatal sonographic topographical assessment of myometrial involvement is presented. The extent of myometrial involvement and the vascularity could be assessed by the observation of the extent of placental-uterine wall interface disruption and the vessels crossing the interface disruption sites. Such assessment results in strategic planning of management of the placenta at delivery with favourable pregnancy outcomes.

Evaluation of sonographic diagnostic criteria for placenta accreta.

PURPOSE: To compare the diagnostic value of reported sonographic criteria for placenta accreta and to develop a composite score system for antenatal evaluation. METHOD: Sixty-six women at risk for placenta accreta were examined for 9 cases of placenta accreta that were confirmed at delivery. The performance of previously reported gray-scale and Doppler sonographic criteria for the diagnosis of placenta accreta was analyzed individually. A composite score system was developed by combining selected sensitive or specific sonographic criteria, and its performance was evaluated. RESULTS: The criteria of obliteration of retroplacental clear space, a myometrial thickness of <1 mm, presence of vessels bridging placenta and uterine margin, disruption of the placental-uterine wall interface, and vessels crossing the sites of interface disruption showed a statistically significant association with placenta accreta. The disruption of the placental-uterine wall interface and the presence of vessels crossing these sites were the only 2 individual criteria that could distinguish placenta accreta from non-accreta, which could also be achieved by our composite score system using a cutoff value of 40, with a sensitivity of 89% and specificity of 98%. CONCLUSION: The diagnostic performance of the sonographic diagnostic criteria used in the diagnosis of placenta accreta varies, and a composite score system improves the overall accuracy.

The combination of bed sharing and maternal smoking leads to a greatly increased risk of sudden unexpected death in infancy: the New Zealand SUDI Nationwide Case Control Study.

BACKGROUND: Despite a major reduction in overall infant mortality, sudden unexpected death in infancy (SUDI) continues to be of concern in New Zealand, as the rate is high by international standards, and is even higher in indigenous Maori. AIM: To identify modifiable risk factors for SUDI. METHODS: A three-year (1 March 2012-28 February 2015) nationwide case-control study was conducted in New Zealand. RESULTS: There were 137 SUDI cases, giving a SUDI mortality rate of 0.76/1,000 live births. The rate for Maori was 1.41/1,000, Pacific 1.01/1,000 and non-Maori non-Pacific (predominantly European) 0.50/1,000. The parent(s) of 97% of the SUDI cases were interviewed. Six hundred and forty-nine controls were selected and 258 (40%) were interviewed. The two major risk factors for SUDI were: maternal smoking in pregnancy (adjusted OR=6.01, 95% CI=2.97, 12.15) and bed sharing (aOR=4.96, 95% CI=2.55, 9.64). There was a significant interaction (p=0.002) between bed sharing and antenatal maternal smoking. Infants exposed to both risk factors had a markedly increased risk of SUDI (aOR=32.8, 95% CI=11.2, 95.8) compared with infants not exposed to either risk factor. Infants not sharing the parental bedroom were also at increased risk of SUDI (aOR=2.77, 95% CI=1.45, 5.30). Just 21 cases over the three-year study were not exposed to smoking in pregnancy, bed sharing or front or side sleeping position. CONCLUSIONS: This study has shown that many of the risk factors that were identified in the original New Zealand Cot Death Study (1987-1989) are still relevant today. The combination of maternal smoking in pregnancy and bed sharing is extremely hazardous for infants. Furthermore, our findings indicate that the SUDI prevention messages are still applicable today and should be reinforced. SUDI mortality could be reduced to just seven p.a. in New Zealand (approximately one in 10,000 live births).

Classification of causes and associated conditions for stillbirths and neonatal deaths.

Accurate and consistent classification of causes and associated conditions for perinatal deaths is essential to inform strategies to reduce the five million which occur globally each year. With the majority of deaths occurring in low- and middle-income countries (LMICs), their needs must be prioritised. The aim of this paper is to review the classification of perinatal death, the contemporary classification systems including the World Health Organization's International Classification of Diseases - Perinatal Mortality (ICD-PM), and next steps. During the period from 2009 to 2014, a total of 81 new or modified classification systems were identified with the majority developed in high-income countries (HICs). Structure, definitions and rules and therefore data on causes vary widely and implementation is suboptimal. Whereas system testing is limited, none appears ideal. Several systems result in a high proportion of unexplained stillbirths, prompting HICs to use more detailed systems that require data unavailable in low-income countries. Some systems appear to perform well across these different settings. ICD-PM addresses some shortcomings of ICD-10 for perinatal deaths, but important limitations remain, especially for stillbirths. A global approach to classification is needed and seems feasible. The new ICD-PM system is an important step forward and improvements will be enhanced by wide-scale use and evaluation. Implementation requires national-level support and dedicated resources. Future research should focus on implementation strategies and evaluation methods, defining placental pathologies, and ways to engage parents in the process.

Does a large abdominal wall defect affect lung growth?

BACKGROUND/PURPOSE: Respiratory distress in babies with large abdominal wall defects suggests a relationship to decreased diaphragmatic movement. We evaluated pulmonary development in a fetal lamb gastroschisis model. METHODS: We created gastroschisis in 25 fetal lambs at 60days gestation (group A). Controls were 14 nonoperated lambs. (Group B) were all delivered at term. Lung volume, histology, and type 1 (AT1)/type 2 (AT2) cell ratios (AT1 ratio) were determined. We subdivided group A, comparing lambs with a large defect and scoliosis [group A (S)] with the remainder [group A (NS)]. RESULTS: Twenty-five lambs survived (11 fetuses in group A and 14 fetuses in group B). Lung volume in group A (S) was less than in group A (NS) (p<0.05). The AT1 ratio in group A was lower than in group B (p<0.01), without any difference in radial alveolar counts (RACs) or alveolar growth, and no association between scoliosis and alveolar differentiation. CONCLUSION: Gastroschisis in a sheep model reduces the AT1 ratio but not the RAC. Severe scoliosis affects lung volume but not the AT1 ratio, suggesting reduced diaphragmatic movement in fetuses with large abdominal defects.