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1.  Prevalence, prenatal diagnosis and clinical features of oculo-auriculo-vertebral spectrum: a registry-based study in Europe 
European Journal of Human Genetics  2014;22(8):1026-1033.
Oculo-auriculo-vertebral spectrum is a complex developmental disorder characterised mainly by anomalies of the ear, hemifacial microsomia, epibulbar dermoids and vertebral anomalies. The aetiology is largely unknown, and the epidemiological data are limited and inconsistent. We present the largest population-based epidemiological study to date, using data provided by the large network of congenital anomalies registries in Europe. The study population included infants diagnosed with oculo-auriculo-vertebral spectrum during the 1990–2009 period from 34 registries active in 16 European countries. Of the 355 infants diagnosed with oculo-auriculo-vertebral spectrum, there were 95.8% (340/355) live born, 0.8% (3/355) fetal deaths, 3.4% (12/355) terminations of pregnancy for fetal anomaly and 1.5% (5/340) neonatal deaths. In 18.9%, there was prenatal detection of anomaly/anomalies associated with oculo-auriculo-vertebral spectrum, 69.7% were diagnosed at birth, 3.9% in the first week of life and 6.1% within 1 year of life. Microtia (88.8%), hemifacial microsomia (49.0%) and ear tags (44.4%) were the most frequent anomalies, followed by atresia/stenosis of external auditory canal (25.1%), diverse vertebral (24.3%) and eye (24.3%) anomalies. There was a high rate (69.5%) of associated anomalies of other organs/systems. The most common were congenital heart defects present in 27.8% of patients. The prevalence of oculo-auriculo-vertebral spectrum, defined as microtia/ear anomalies and at least one major characteristic anomaly, was 3.8 per 100 000 births. Twinning, assisted reproductive techniques and maternal pre-pregnancy diabetes were confirmed as risk factors. The high rate of different associated anomalies points to the need of performing an early ultrasound screening in all infants born with this disorder.
PMCID: PMC4350601  PMID: 24398798
oculo-auriculo-vertebral spectrum; epidemiology; congenital anomalies; Europe
2.  Mothers’ perspectives on the perinatal loss of a co-twin: a qualitative study 
There is a growing body of literature exploring the emotional impact of perinatal loss upon parents but only limited research focussing specifically on the views and experiences of parents who have experienced a loss from a twin or higher order pregnancy. We undertook a qualitative study to provide an in-depth understanding of the experiences of mothers who have had a loss from a twin pregnancy and subsequently continued visiting hospital whilst their surviving twin was cared for.
A qualitative study involving semi-structured interviews. Mothers were recruited from a Neonatal Intensive Care Unit and Fetal Medicine department. Fourteen interviews were carried out with mothers who had experienced a loss in pregnancy or the neonatal period and had a surviving twin on the neonatal unit. Data were analysed using a generative thematic approach.
The analysis identified three key themes in the accounts mothers gave of their experiences: the status of ‘special’; the importance of trust; and control and empowerment. Where the surviving co-twin remained in hospital for many weeks, mothers described the emotional support of health professionals as crucial to their wellbeing. Few mothers sought formal bereavement support, instead they kept their grief ‘on hold’ in order to support their surviving baby. Due to the trauma of their loss, mothers reflected that they had been unable to make informed decisions, in particular in relation to the funeral of their deceased baby.
Our study highlighted that there are a specific set of issues for mothers who have lost a baby from a twin pregnancy. Relatively small changes to practice however, made a significant difference to wellbeing during their time in hospital with a surviving twin. Findings from this research will provide insight into the needs of bereaved mothers, will inform healthcare planning and the development of care packages.
Electronic supplementary material
The online version of this article (doi:10.1186/s12884-015-0579-z) contains supplementary material, which is available to authorized users.
PMCID: PMC4488109  PMID: 26134115
Bereavement; Multiple pregnancy; Twins; Grief; Trauma
3.  An Evaluation of the Implementation of Maternal Obesity Pathways of Care: A Mixed Methods Study with Data Integration 
PLoS ONE  2015;10(5):e0127122.
Maternal obesity has multiple associated risks and requires substantial intervention. This research evaluated the implementation of maternal obesity care pathways from multiple stakeholder perspectives.
Study Design
A simultaneous mixed methods model with data integration was used. Three component studies were given equal priority. 1: Semi-structured qualitative interviews explored obese pregnant women’s experiences of being on the pathways. 2: A quantitative and qualitative postal survey explored healthcare professionals’ experiences of delivering the pathways. 3: A case note audit quantitatively assessed pathway compliance. Data were integrated using following a thread and convergence coding matrix methods to search for agreement and disagreement between studies.
Study 1: Four themes were identified: women’s overall (positive and negative) views of the pathways; knowledge and understanding of the pathways; views on clinical and weight management advice and support; and views on the information leaflet. Key results included positive views of receiving additional clinical care, negative experiences of risk communication, and weight management support was considered a priority. Study 2: Healthcare professionals felt the pathways were worthwhile, facilitated good practice, and increased confidence. Training was consistently identified as being required. Healthcare professionals predominantly focussed on women’s response to sensitive obesity communication. Study 3: There was good compliance with antenatal clinical interventions. However, there was poor compliance with public health and postnatal interventions. There were some strong areas of agreement between component studies which can inform future development of the pathways. However, disagreement between studies included a lack of shared priorities between healthcare professionals and women, different perspectives on communication issues, and different perspectives on women’s prioritisation of weight management.
The differences between healthcare professionals’ and women’s priorities and perspectives are important factors to consider when developing care pathways. Shared perspectives could help facilitate more effective implementation of the pathway interventions that have poor compliance.
PMCID: PMC4446303  PMID: 26018338
4.  Holt Oram syndrome: a registry-based study in Europe 
Holt-Oram syndrome (HOS) is an autosomal dominant disorder characterised by upper limb anomalies and congenital heart defects. We present epidemiological and clinical aspects of HOS patients using data from EUROCAT (European Surveillance of Congenital Anomalies) registries.
The study was based on data collected during 1990–2011 by 34 registries. The registries are population-based and use multiple sources of information to collect data on all types of birth using standardized definitions, methodology and coding. Diagnostic criteria for inclusion in the study were the presence of radial ray abnormalities and congenital heart disease (CHD), or the presence of either radial ray anomaly or CHD, with family history of HOS.
A total of 73 cases of HOS were identified, including 11 (15.1%) TOPFA and 62 (84.9%) LB. Out of 73 HOS cases, 30.8% (20/65) were suspected prenatally, 55.4% (36/65) at birth, 10.7% (7/65) in the first week of life, and 3.1% (2/65) in the first year of life. The prenatal detection rate was 39.2% (20/51), with no significant change over the study period. In 55% (11/20) of prenatally detected cases, parents decided to terminate pregnancy. Thumb anomalies were reported in all cases. Agenesis/hypoplasia of radius was present in 49.2% (30/61), ulnar aplasia/hypoplasia in 24.6% (15/61) and humerus hypoplasia/phocomelia in 42.6% (26/61) of patients. Congenital heart defects (CHD) were recorded in 78.7% (48/61) of patients. Isolated septal defects were present in 54.2 (26/48), while 25% (12/48) of patients had complex/severe CHD. The mean prevalence of HOS diagnosed prenatally or in the early years of life in European registries was 0.7 per 100,000 births or 1:135,615 births.
HOS is a rare genetic condition showing regional variation in its prevalence. It is often missed prenatally, in spite of the existence of major structural anomalies. When discovered, parents in 45% (9/20) of cases opt for the continuation of pregnancy. Although a quarter of patients have severe CHD, the overall first week survival is very good, which is important information for counselling purposes.
PMCID: PMC4245183  PMID: 25344219
Holt Oram syndrome; Congenital anomalies; Prenatal diagnosis; Epidemiology; Europe
5.  Interventions to change maternity healthcare professionals’ behaviours to promote weight-related support for obese pregnant women: a systematic review 
There has been a rapid increase in the publication of guidelines for managing obesity and weight gain during pregnancy over the past five years. Healthcare professionals have identified multiple barriers to this area of practice, including the need to improve their communication skills, beliefs that pregnant women will have negative reactions to weight-related discussions, and a lack of weight management knowledge. This systematic review aimed to identify: the effectiveness of interventions in changing healthcare professionals’ practice relating to maternal obesity or weight management during pregnancy; and which behaviour change techniques and modes of intervention delivery have been used in interventions to date.
The search strategy included searching electronic databases, trial registers, and citation searching. Inclusion criteria were intervention studies targeted towards changing healthcare professionals’ practice in relation to maternal obesity or weight management. The searches identified 3,608 studies. However, no eligible completed studies were identified. One registered Canadian randomised controlled trial was identified. The trial includes a training intervention for family physicians with the aim of supporting adherence to gestational weight gain guidelines. The trial had not yet commenced therefore no effectiveness data were available.
The current focus of maternal obesity and weight management research is targeted towards changing pregnant women’s behaviours. These interventions do not address the multiple healthcare professionals’ barriers to maternal obesity and weight management practice. Further research is required to identify the most effective approaches to support healthcare professionals to implement maternal obesity and weight management guidelines into practice.
Electronic supplementary material
The online version of this article (doi:10.1186/s13012-014-0097-9) contains supplementary material, which is available to authorized users.
PMCID: PMC4244067  PMID: 25091032
Systematic review; Healthcare professionals; Behaviour change; Obesity; Pregnancy; Weight management
6.  Twenty-year trends in the prevalence of Down syndrome and other trisomies in Europe: impact of maternal age and prenatal screening 
This study examines trends and geographical differences in total and live birth prevalence of trisomies 21, 18 and 13 with regard to increasing maternal age and prenatal diagnosis in Europe. Twenty-one population-based EUROCAT registries covering 6.1 million births between 1990 and 2009 participated. Trisomy cases included live births, fetal deaths from 20 weeks gestational age and terminations of pregnancy for fetal anomaly. We present correction to 20 weeks gestational age (ie, correcting early terminations for the probability of fetal survival to 20 weeks) to allow for artefactual screening-related differences in total prevalence. Poisson regression was used. The proportion of births in the population to mothers aged 35+ years in the participating registries increased from 13% in 1990 to 19% in 2009. Total prevalence per 10 000 births was 22.0 (95% CI 21.7–22.4) for trisomy 21, 5.0 (95% CI 4.8–5.1) for trisomy 18 and 2.0 (95% CI 1.9–2.2) for trisomy 13; live birth prevalence was 11.2 (95% CI 10.9–11.5) for trisomy 21, 1.04 (95% CI 0.96–1.12) for trisomy 18 and 0.48 (95% CI 0.43–0.54) for trisomy 13. There was an increase in total and total corrected prevalence of all three trisomies over time, mainly explained by increasing maternal age. Live birth prevalence remained stable over time. For trisomy 21, there was a three-fold variation in live birth prevalence between countries. The rise in maternal age has led to an increase in the number of trisomy-affected pregnancies in Europe. Live birth prevalence has remained stable overall. Differences in prenatal screening and termination between countries lead to wide variation in live birth prevalence.
PMCID: PMC3522199  PMID: 22713804
trisomy 21; trisomy 18; trisomy 13; trends; maternal age; prenatal screening
7.  The Role of Maternal Stress in Early Pregnancy in the Aetiology of Gastroschisis: An Incident Case Control Study  
PLoS ONE  2013;8(11):e80103.
The incidence of gastroschisis, a congenital anomaly where the infant abdominal wall is defective and intestines protrude from the abdominal cavity, is increasing in many countries. The role of maternal stress in some adverse birth outcomes is now well established. We tested the hypothesis that major stressful life events in the first trimester are risk factors for gastroschisis, and social support protective, in a case-control study in the United Kingdom.
Gastroschisis cases and three controls per case (matched for maternal age) were identified at routine 18-20 week fetal anomaly ultrasound scan, in 2007-2010. Face to face questionnaire interviews were carried out during the antenatal period (median 24 weeks gestation) asking about serious stressful events and social support in the first trimester. Data were analysed using conditional logistic regression.
Two or more stressful life events in the first trimester (adjusted OR 4.9; 95% CI 1.2-19.4), and moving address in the first trimester (aOR 4.9; 95% CI 1.7-13.9) were strongly associated with risk of gastroschisis, independent of behavioural risk factors including smoking, alcohol, and poor diet. Perceived availability of social support was not associated with reduced risk of gastroschisis (aOR 0.8; 95% CI 0.2-3.1).
Stressful maternal life events in the first trimester of pregnancy including change of address were strongly associated with a substantial increase in the risk of gastroschisis, independent of stress related high risk behaviours such as smoking, alcohol consumption and poor diet. This suggests that stress pathways are involved in the aetiology of gastroschisis.
PMCID: PMC3832654  PMID: 24260340
8.  Rare chromosome abnormalities, prevalence and prenatal diagnosis rates from population-based congenital anomaly registers in Europe 
The aim of this study is to quantify the prevalence and types of rare chromosome abnormalities (RCAs) in Europe for 2000–2006 inclusive, and to describe prenatal diagnosis rates and pregnancy outcome. Data held by the European Surveillance of Congenital Anomalies database were analysed on all the cases from 16 population-based registries in 11 European countries diagnosed prenatally or before 1 year of age, and delivered between 2000 and 2006. Cases were all unbalanced chromosome abnormalities and included live births, fetal deaths from 20 weeks gestation and terminations of pregnancy for fetal anomaly. There were 10 323 cases with a chromosome abnormality, giving a total birth prevalence rate of 43.8/10 000 births. Of these, 7335 cases had trisomy 21,18 or 13, giving individual prevalence rates of 23.0, 5.9 and 2.3/10 000 births, respectively (53, 13 and 5% of all reported chromosome errors, respectively). In all, 473 cases (5%) had a sex chromosome trisomy, and 778 (8%) had 45,X, giving prevalence rates of 2.0 and 3.3/10 000 births, respectively. There were 1 737 RCA cases (17%), giving a prevalence of 7.4/10 000 births. These included triploidy, other trisomies, marker chromosomes, unbalanced translocations, deletions and duplications. There was a wide variation between the registers in both the overall prenatal diagnosis rate of RCA, an average of 65% (range 5–92%) and the prevalence of RCA (range 2.4–12.9/10 000 births). In all, 49% were liveborn. The data provide the prevalence of families currently requiring specialised genetic counselling services in the perinatal period for these conditions and, for some, long-term care.
PMCID: PMC3330224  PMID: 22234154
rare chromosome; prevalence; prenatal diagnosis; Europe
9.  No improvement in socioeconomic inequalities in birthweight and preterm birth over four decades: a population-based cohort study 
BMC Public Health  2013;13:345.
Birthweight and gestational age are associated with socioeconomic deprivation, but the evidence in relation to temporal changes in these associations is sparse. We investigated changes in the associations between socioeconomic status (SES) and birthweight and gestational age in Newcastle upon Tyne, North of England, during 1961–2000.
We used population-based data from hospital neonatal records on all singleton births to mothers resident in Newcastle (births with complete covariate information n = 113,182). We used linear regression to analyse the associations between neighbourhood SES and birthweight over the entire 40-year period and by decade, and logistic regression for associations with low birthweight (LBW) and preterm birth, adjusting for potential confounders.
There was a significant interaction between SES and decade of birth for birthweight (p = 0.028) and preterm birth (p < 0.001). Socioeconomic gradients were similar in each decade for birthweight outcomes, but for preterm birth, socioeconomic disparities were more evident in the later decades [for 1961–70, odds ratio (OR) was 1.1, 95% CI 0.9, 1.3, for the most deprived versus the least deprived quartile, while for 1991–2000, the corresponding OR was 1.5, 95% CI 1.3, 1.7]. In each decade, there was a significant decrease in birthweight adjusted for gestational age for the most deprived compared to the least deprived SES group [1961–1970: –113.4 g (95% CI–133.0, –93.8); 1991–2000: –97.5 g (95% CI–113.0, –82.0)], while there was a significant increase in birthweight in each SES group over time.
Socioeconomic inequalities did not narrow over the four decades for birthweight and widened for preterm birth. Mean birthweight adjusted for gestational age increased in all socioeconomic groups, suggesting an overall increase in fetal growth.
PMCID: PMC3651338  PMID: 23587186
Low birthweight; Preterm birth; Socioeconomic status; Townsend deprivation score; Temporal trends
10.  Existing maternal obesity guidelines may increase inequalities between ethnic groups: a national epidemiological study of 502,474 births in England 
Asians are at increased risk of morbidity at a lower body mass index (BMI) than European Whites, particularly relating to metabolic risk. UK maternal obesity guidelines use general population BMI criteria to define obesity, which do not represent the risk of morbidity among Asian populations. This study compares incidence of first trimester obesity using Asian-specific and general population BMI criteria.
A retrospective epidemiological study of 502,474 births between 1995 and 2007, from 34 maternity units across England. Data analyses included a comparison of trends over time between ethnic groups using Asian-specific and general population BMI criteria. Logistic regression estimated odds ratios for first trimester obesity among ethnic groups following adjustment for population demographics.
Black and South Asian women have a higher incidence of first trimester obesity compared with White women. This is most pronounced for Pakistani women following adjustment for population structure (OR 2.19, 95% C.I. 2.08, 2.31). There is a twofold increase in the proportion of South Asian women classified as obese when using the Asian-specific BMI criteria rather than general population BMI criteria. The incidence of obesity among Black women is increasing at the most rapid rate over time (p=0.01).
The twofold increase in maternal obesity among South Asians when using Asian-specific BMI criteria highlights inequalities among pregnant women. A large proportion of South Asian women are potentially being wrongly assigned to low risk care using current UK guidelines to classify obesity and determine care requirements. Further research is required to identify if there is any improvement in pregnancy outcomes if Asian-specific BMI criteria are utilised in the clinical management of maternal obesity to ensure the best quality of care is provided for women irrespective of ethnicity.
PMCID: PMC3554430  PMID: 23249162
Obesity; Pregnancy; Epidemiology; Inequalities; Ethnic group; Asian; Guidelines; Body Mass Index (BMI)
11.  Brief intervention to reduce risky drinking in pregnancy: study protocol for a randomized controlled trial 
Trials  2012;13:174.
Risky drinking in pregnancy by UK women is likely to result in many alcohol-exposed pregnancies. Studies from the USA suggest that brief intervention has promise for alcohol risk reduction in antenatal care. However, further research is needed to establish whether this evidence from the USA is applicable to the UK. This pilot study aims to investigate whether pregnant women can be recruited and retained in a randomized controlled trial of brief intervention aimed at reducing risky drinking in women receiving antenatal care.
The trial will rehearse the parallel-group, non-blinded design and procedures of a subsequent definitive trial. Over 8 months, women aged 18 years and over (target number 2,742) attending their booking appointment with a community midwife (n = 31) in north-east England will be screened for alcohol consumption using the consumption questions of the Alcohol Use Disorders Identification Test (AUDIT-C). Those screening positive, without a history of substance use or alcohol dependence, with no pregnancy complication, and able to give informed consent, will be invited to participate in the trial (target number 120). Midwives will be randomized in a 1:1 ratio to deliver either treatment as usual (control) or structured brief advice and referral for a 20-minute motivational interviewing session with an alcohol health worker (intervention). As well as demographic and health information, baseline measures will include two 7-day time line follow-back questionnaires and the EuroQoL EQ-5D-3 L questionnaire. Measures will be repeated in telephone follow-ups in the third trimester and at 6 months post-partum, when a questionnaire on use of National Health Service and social care resources will also be completed. Information on pregnancy outcomes and stillbirths will be accessed from central health service records before the follow-ups. Primary outcomes will be rates of eligibility, recruitment, intervention delivery, and retention in the study population, to inform power calculations for a definitive trial. The health-economics component will establish how cost-effectiveness will be assessed, and examine which data on health service resource use should be collected in a main trial. Participants’ views on instruments and procedures will be sought to confirm their acceptability.
The study will produce a full trial protocol with robust sample-size calculations to extend evidence on effectiveness of screening and brief intervention.
Trial Registration
Current Controlled Trials ISRCTN43218782
PMCID: PMC3543230  PMID: 23006975
Pregnancy; Alcohol; Screening; Brief intervention; Trial; Midwife; Motivational interviewing; Public health
12.  Particulate matter exposure during pregnancy is associated with birth weight, but not gestational age, 1962-1992: a cohort study 
Environmental Health  2012;11:13.
Exposure to air pollutants is suggested to adversely affect fetal growth, but the evidence remains inconsistent in relation to specific outcomes and exposure windows.
Using birth records from the two major maternity hospitals in Newcastle upon Tyne in northern England between 1961 and 1992, we constructed a database of all births to mothers resident within the city. Weekly black smoke exposure levels from routine data recorded at 20 air pollution monitoring stations were obtained and individual exposures were estimated via a two-stage modeling strategy, incorporating temporally and spatially varying covariates. Regression analyses, including 88,679 births, assessed potential associations between exposure to black smoke and birth weight, gestational age and birth weight standardized for gestational age and sex.
Significant associations were seen between black smoke and both standardized and unstandardized birth weight, but not for gestational age when adjusted for potential confounders. Not all associations were linear. For an increase in whole pregnancy black smoke exposure, from the 1st (7.4 μg/m3) to the 25th (17.2 μg/m3), 50th (33.8 μg/m3), 75th (108.3 μg/m3), and 90th (180.8 μg/m3) percentiles, the adjusted estimated decreases in birth weight were 33 g (SE 1.05), 62 g (1.63), 98 g (2.26) and 109 g (2.44) respectively. A significant interaction was observed between socio-economic deprivation and black smoke on both standardized and unstandardized birth weight with increasing effects of black smoke in reducing birth weight seen with increasing socio-economic disadvantage.
The findings of this study progress the hypothesis that the association between black smoke and birth weight may be mediated through intrauterine growth restriction. The associations between black smoke and birth weight were of the same order of magnitude as those reported for passive smoking. These findings add to the growing evidence of the harmful effects of air pollution on birth outcomes.
PMCID: PMC3324390  PMID: 22404858
Black smoke; Particulate matter; Air pollution; Birth weight; Gestational age
13.  Association Between Maternal Exposure to Ambient Air Pollution and Congenital Heart Disease: A Register-based Spatiotemporal Analysis 
American Journal of Epidemiology  2010;173(2):171-182.
Recent studies have linked maternal exposure to air pollution with a range of adverse pregnancy outcomes. However, the available evidence linking this exposure to congenital anomalies is still limited and controversial. The present case-control study tested the hypothesis that maternal exposure to ambient black smoke and sulfur dioxide is a risk factor for the occurrence of congenital heart disease. The authors used registry-based data on congenital heart disease for the population of the northeast of England in 1985–1996. A 2-stage spatiotemporal model was developed to predict weekly black smoke and sulfur dioxide levels at each maternal place of residence. Controls were frequency-matched to cases by year of birth (control-to-case ratio of 4:1). Two sets of analyses were performed, using predicted mean values of exposure and 1,000 simulated scenarios of exposure. The analyses were adjusted for birth year, socioeconomic status, infant sex, season of conception, and degree of urbanity. The authors found a weak association between maternal exposure to black smoke and congenital malformations of cardiac chambers and connections only when using exposure as a continuous variable. When the authors used quartiles of exposure, odds ratios did not show a dose-response relation for consecutive quartiles. For sulfur dioxide, the results were not indicative of any association.
PMCID: PMC3011953  PMID: 21123851
air pollution; congenital abnormalities; heart defects, congenital; maternal exposure; sulfur dioxide; spatiotemporal analysis
14.  Ambient Air Pollution and Risk of Congenital Anomalies: A Systematic Review and Meta-analysis 
Environmental Health Perspectives  2010;119(5):598-606.
We systematically reviewed epidemiologic studies on ambient air pollution and congenital anomalies and conducted meta-analyses for a number of air pollutant–anomaly combinations.
Data sources and extraction
From bibliographic searches we extracted 10 original epidemiologic studies that examined the association between congenital anomaly risk and concentrations of air pollutants. Meta-analyses were conducted if at least four studies published risk estimates for the same pollutant and anomaly group. Summary risk estimates were calculated for a) risk at high versus low exposure level in each study and b) risk per unit increase in continuous pollutant concentration.
Data synthesis
Each individual study reported statistically significantly increased risks for some combinations of air pollutants and congenital anomalies, among many combinations tested. In meta-analyses, nitrogen dioxide (NO2) and sulfur dioxide (SO2) exposures were related to increases in risk of coarctation of the aorta [odds ratio (OR) per 10 ppb NO2 = 1.17; 95% confidence interval (CI), 1.00–1.36; OR per 1 ppb SO2 = 1.07; 95% CI, 1.01–1.13] and tetralogy of Fallot (OR per 10 ppb NO2 = 1.20; 95% CI, 1.02–1.42; OR per 1 ppb SO2 = 1.03; 95% CI, 1.01–1.05), and PM10 (particulate matter ≤ 10 μm) exposure was related to an increased risk of atrial septal defects (OR per 10 μg/m3 = 1.14; 95% CI, 1.01–1.28). Meta-analyses found no statistically significant increase in risk of other cardiac anomalies and oral clefts.
We found some evidence for an effect of ambient air pollutants on congenital cardiac anomaly risk. Improvements in the areas of exposure assessment, outcome harmonization, assessment of other congenital anomalies, and mechanistic knowledge are needed to advance this field.
PMCID: PMC3094408  PMID: 21131253
air pollution; congenital abnormalities; pregnancy
15.  Preconception Counseling in Women With Diabetes 
Diabetes Care  2009;33(3):586-588.
To investigate the association of preconception counseling with markers of care and maternal characteristics in women with pregestational diabetes.
The study includes data from a regional multi-center survey on 588 women with pregestational diabetes who delivered a singleton pregnancy between 2001 and 2004. Logistic regression was used to obtain crude and adjusted estimates of association.
Preconception counseling was associated with better glycemic control 3 months preconception (odds ratio 1.91, 95% CI 1.10–3.04) and in the first trimester (2.05, 1.39–3.03), higher preconception folic acid intake (4.88, 3.26–7.30), and reduced risk of adverse pregnancy outcome (P = 0.027). Uptake of preconception counseling was positively associated with type 1 diabetes (1.87, 1.14–3.07) and White British ethnicity (2.56, 1.17–5.6) and negatively with deprivation score (0.78, 0.70–0.87).
Efforts are needed to improve preconception counseling rates. Uptake is associated with maternal sociodemographic characteristics.
PMCID: PMC2827513  PMID: 20040652
16.  Objectively measured physical activity during pregnancy: a study in obese and overweight women 
Obese and overweight women may benefit from increased physical activity (PA) during pregnancy. There is limited published data describing objectively measured PA in such women.
A longitudinal observational study of PA intensity, type and duration using objective and subjective measurement methods. Fifty five pregnant women with booking body mass index (BMI) ≥ 25 kg/m2 were recruited from a hospital ultrasound clinic in North East England. 26 (47%) were nulliparous and 22 (40%) were obese (BMI ≥ 30 kg/m2). PA was measured by accelerometry and self report questionnaire at 13 weeks, 26 weeks and/or 36 weeks gestation. Outcome measures were daily duration of light, moderate or vigorous activity assessed by accelerometry; calculated overall PA energy expenditure, (PAEE), and PAEE within four domains of activity based on self report.
At median 13 weeks gestation, women recorded a median 125 mins/day light activity and 35 mins/day moderate or vigorous activity (MVPA). 65% achieved the minimum recommended 30 mins/day MVPA. This proportion was maintained at 26 weeks (62%) and 36 weeks (71%). Women achieving more than 30 mins/day MVPA in the first trimester showed a significant reduction in duration of MVPA by the third trimester (11 mins/day, p = 0.003). Walking, swimming and floor exercises were the most commonly reported recreational activities but their contribution to estimated energy expenditure was small.
Overweight and obese pregnant women can achieve and maintain recommended levels of PA throughout pregnancy. Interventions to promote PA should target changes in habitual activities at work and at home, and in particular walking.
PMCID: PMC3001702  PMID: 21114834
17.  Physical activity in pregnancy: a qualitative study of the beliefs of overweight and obese pregnant women 
Whilst there has been increasing research interest in interventions which promote physical activity during pregnancy few studies have yielded detailed insights into the views and experiences of overweight and obese pregnant women themselves. The qualitative study described in this paper aimed to: (i) explore the views and experiences of overweight and obese pregnant women; and (ii) inform interventions which could promote the adoption of physical activity during pregnancy.
The study was framed by a combined Subtle Realism and Theory of Planned Behaviour (TPB) approach. This enabled us to examine the hypothetical pathway between beliefs and physical activity intentions within the context of day to day life. The study sample for the qualitative study was chosen by stratified, purposive sampling from a previous study of physical activity measurements in pregnancy. Research participants for the current study were recruited on the basis of Body Mass Index (BMI) at booking and parity. Semi-structured, in-depth interviews were conducted with 14 overweight and obese pregnant women. Data analysis was undertaken using a Framework Approach and was informed by TPB.
Healthy eating was often viewed as being of greater importance for the health of mother and baby than participation in physical activity. A commonly cited motivator for maintaining physical activity during pregnancy is an aid to reducing pregnancy-related weight gain. However, participants often described how they would wait until the postnatal period to try and lose weight. A wide range of barriers to physical activity during pregnancy were highlighted including both internal (physical and psychological) and external (work, family, time and environmental). The study participants also lacked access to consistent information, advice and support on the benefits of physical activity during pregnancy.
Interventions to encourage recommended levels of physical activity in pregnancy should be accompanied by accessible and consistent information about the positive effects for mother and baby. More research is required to examine how to overcome barriers to physical activity and to understand which interventions could be most effective for overweight/obese pregnant women. Midwives should be encouraged to do more to promote activity in pregnancy.
PMCID: PMC2879230  PMID: 20426815
18.  Residential mobility during pregnancy in the north of England 
Many epidemiological studies assign exposure to an individual's residence at a single time point, such as birth or death. This approach makes no allowance for migration and may result in exposure error, leading to reduced study power and biased risk estimates. Pregnancy outcomes are less susceptible to this bias, however data from North American populations indicate that pregnant women are a highly mobile group. We assessed mobility in pregnant women in the north of England using data from the Northern Congenital Abnormality Survey (NorCAS).
Data were extracted from NorCAS for 1985 to 2003. Eligible cases had a gestational age at delivery of ≥ 24 weeks (a viable delivery) (n = 11 559). We assessed mobility between booking appointment (average gestational age 13 weeks) and delivery for pregnancies where the address at booking appointment and delivery were known. The impacts on mobility of maternal age and area-level socio-economic indicators were explored using standard descriptive statistics. A sensitivity analysis and a small validation exercise were undertaken to assess the impact of missing data on the estimate of mobility.
Out of 7 919 eligible cases for whom addresses at booking and delivery were known, 705 (8.9% (95% CI 8.3 - 9.5)) moved between booking and delivery; the mean and median moving distance was 9.7 and 1.4 km respectively. Movers were significantly younger (25.4 versus 27.3 years, p < 0.01) and lived in more deprived areas (index of multiple deprivation score 38.3 versus 33.7, p < 0.01) than non movers.
Mobility in the north of England (9%) is considerably lower than that reported in North America and the only other study from the UK (23%). Consistent with other studies, mobility was related to maternal age and socio-economic status, and the majority of moves were over a relatively short distance. Although this population appears relatively stable, the mobility we have observed may still introduce misclassification or error into an exposure assessment relying solely on postcode at delivery, and migration should still therefore be considered a potential source of bias in future studies.
PMCID: PMC2784435  PMID: 19912662
19.  Quitting smoking and experience of smoking cessation interventions among UK Bangladeshi and Pakistani adults: the views of community members and health professionals 
To explore attitudes to quitting smoking and experience of smoking cessation among Bangladeshi and Pakistani ethnic minority communities.
Qualitative study using community participatory methods, purposeful sampling, interviews and focus groups, and a grounded approach to data generation and analysis.
Newcastle upon Tyne, UK, 2000–2002.
53 men and 20 women aged 18–80 years, including smokers, former smokers, and smokers' relatives, from the Bangladeshi and Pakistani communities; and eight health professionals working with these communities.
Motivation to quit was high but most attempts had failed. “Willpower” was the most common approach to quitting. For some, the holy month of Ramadan was used as an incentive, however few had been successful in quitting. Perceived barriers to success included being tempted by others, everyday stresses, and withdrawal symptoms. Few participants had sought advice from health services, or received cessation aids, such as nicotine replacement therapy (NRT) or buproprion. Family doctors were not viewed as accessible sources of advice on quitting. Health professionals and community members identified common barriers to accessing effective smoking cessation, including: language, religion and culture; negative attitudes to services; and lack of time and resources for professionals to develop necessary skills.
High levels of motivation do not seem to be matched by effective interventions or successful attempts to quit smoking among Bangladeshi and Pakistani adults in the UK. There is a need to adapt and test effective smoking cessation interventions to make them culturally acceptable to ethnic minority communities. UK tobacco control policies need to give special attention to the needs of ethnic minority groups.
PMCID: PMC2563973  PMID: 16614330
smoking cessation; qualitative research; South Asian; Pakistani; Bangladeshi
20.  Temporal changes in key maternal and fetal factors affecting birth outcomes: A 32-year population-based study in an industrial city 
The link between maternal factors and birth outcomes is well established. Substantial changes in society and medical care over time have influenced women's reproductive choices and health, subsequently affecting birth outcomes. The objective of this study was to describe temporal changes in key maternal and fetal factors affecting birth outcomes in Newcastle upon Tyne over three decades, 1961–1992.
For these descriptive analyses we used data from a population-based birth record database constructed for the historical cohort Particulate Matter and Perinatal Events Research (PAMPER) study. The PAMPER database was created using details from paper-based hospital delivery and neonatal records for all births during 1961–1992 to mothers resident in Newcastle (out of a total of 109,086 singleton births, 97,809 hospital births with relevant information). In addition to hospital records, we used other sources for data collection on births not included in the delivery and neonatal records, for death and stillbirth registrations and for validation.
The average family size decreased mainly due to a decline in the proportion of families with 3 or more children. The distribution of mean maternal ages in all and in primiparous women was lowest in the mid 1970s, corresponding to a peak in the proportion of teenage mothers. The proportion of older mothers declined until the late 1970s (from 16.5% to 3.4%) followed by a steady increase. Mean birthweight in all and term babies gradually increased from the mid 1970s. The increase in the percentage of preterm birth paralleled a two-fold increase in the percentage of caesarean section among preterm births during the last two decades. The gap between the most affluent and the most deprived groups of the population widened over the three decades.
Key maternal and fetal factors affecting birth outcomes, such as maternal age, parity, socioeconomic status, birthweight and gestational age, changed substantially during the 32-year period, from 1961 to 1992. The availability of accurate gestational age is extremely important for correct interpretation of trends in birthweight.
PMCID: PMC2542990  PMID: 18713457
21.  Geographic variation and localised clustering of congenital anomalies in Great Britain 
Environmental pollution as a cause of congenital anomalies is sometimes suspected because of clustering of anomalies in areas of higher exposure. This highlights questions around spatial heterogeneity (clustering) in congenital anomaly rates. If spatial variation is endemic, then any one specific cluster is less remarkable, though the presence of uncontrolled geographically clustered risk factors is suggested. If rates are relatively homogeneous across space other than around specific hazards, then evidence for these hazards causing the clusters is strengthened. We sought to estimate the extent of spatial heterogeneity in congenital anomaly rates in the United Kingdom.
The study population covered about one million births from five registers in Britain from 1991–1999. We estimated heterogeneity across four geographical levels: register area, hospital catchment, electoral ward, and enumeration district, using a negative binomial regression model. We also sought clusters using a circular scan statistic.
Congenital anomaly rates clearly varied across register areas and hospital catchments (p < 0.001), but not below this level (p > 0.2). Adjusting for socioeconomic deprivation and maternal age made little difference to the extent of geographical variation for most congenital anomaly subtypes. The two most significant circular clusters (of four ano-rectal atresias and six congenital heart diseases) contained two or more siblings.
The variation in rates between registers and hospital catchment area may have resulted in part from differences in case ascertainment, and this should be taken into account in geographical epidemiological studies of environmental exposures. The absence of evidence for variation below this level should be interpreted cautiously in view of the low power of general heterogeneity tests. Nevertheless, the data suggest that strong localised clusters in congenital anomalies are uncommon, so clusters around specific putative environmental hazards are remarkable when observed. Negative binomial models applied at successive hierarchical levels provide an approach of intermediate complexity to characterising geographical heterogeneity.
PMCID: PMC1939702  PMID: 17617898
22.  Does Particulate Air Pollution Contribute to Infant Death? A Systematic Review 
Environmental Health Perspectives  2004;112(14):1365-1370.
There is now substantial evidence that both short- and long-term increases in ambient air pollution are associated with increased mortality and morbidity in adults and children. Children’s health is particularly vulnerable to environmental pollution, and infant mortality is still a major contributor to childhood mortality. In this systematic review we summarize and evaluate the current level of epidemiologic evidence of an association between particulate air pollution and infant mortality. We identified relevant publications using database searches with a comprehensive list of search terms and other established search methods. We included articles in the review according to specified inclusion criteria. Fifteen studies met our inclusion criteria. Evidence of an association between particulate air pollution and infant mortality in general was inconsistent, being reported from locations with largely comparable pollution levels. There was some evidence that the strength of association with particulate matter differed by subgroups of infant mortality. It was more consistent for post-neonatal mortality due to respiratory causes and sudden infant death syndrome. Differential findings for various mortality subgroups within studies suggest a stronger association of particulate air pollution with some causes of infant death. Research is needed to confirm and clarify these links, using the most appropriate methodologies for exposure assessment and control of confounders.
PMCID: PMC1247561  PMID: 15471726
infant mortality; particulate air pollution; postneonatal respiratory mortality; sudden infant death syndrome; systematic review
23.  Understanding influences on smoking in Bangladeshi and Pakistani adults: community based, qualitative study 
BMJ : British Medical Journal  2003;326(7396):962.
To gain detailed understanding of influences on smoking behaviour in Bangladeshi and Pakistani communities in the United Kingdom to inform the development of effective and culturally acceptable smoking cessation interventions.
Qualitative study using community participatory methods, purposeful sampling, one to one interviews, focus groups, and a grounded approach to data generation and analysis.
Newcastle upon Tyne, during 2000-2.
87 men and 54 women aged 18-80 years, smokers and non-smokers, from the Bangladeshi and Pakistani communities.
Four dominant, highly inter-related themes had an important influence on smoking attitudes and behaviour: gender, age, religion, and tradition. Smoking was a widely accepted practice in Pakistani, and particularly Bangladeshi, men and was associated with socialising, sharing, and male identity. Among women, smoking was associated with stigma and shame. Smoking in women is often hidden from family members. Peer pressure was an important influence on smoking behaviour in younger people, who tended to hide their smoking from elders. There were varied and conflicting interpretations of how acceptable smoking is within the Muslim religion. Tradition, culture, and the family played an important role in nurturing and cultivating norms and values around smoking.
Although there are some culturally specific contexts for smoking behaviour in Bangladeshi and Pakistani adults—notably the influence of gender and religion—there are also strong similarities with white people, particularly among younger adults. Themes identified should help to inform the development and appropriate targeting of smoking cessation interventions.
What is already known on this topicSmoking is common among Bangladeshi and Pakistani men in Britain but rare among the womenSmoking is particularly common in Bangladeshi menSocioeconomic status is thought to influence smoking uptake in Bangladeshi menInfluences on smoking in South Asians in Britain are poorly understoodWhat this study addsSmoking among Pakistani and Bangladeshi men is strongly seen as socially acceptable—a “normal” part of being a manSmoking in Bangladeshi men is more deeply socially ingrained than in Pakistanis, contributing to group cohesion and identitySmoking in Bangladeshi and Pakistani women is associated with a strong sense of cultural taboo, stigma, and non-acceptanceIslam forbids addiction and intoxicants, but opinions differ on whether the Muslim religion allows smokingCulturally sensitive smoking cessation interventions for Bangladeshis and Pakistanis are needed
PMCID: PMC153853  PMID: 12727770
24.  Cross sectional survey of parents' experience and views of the postmortem examination 
BMJ : British Medical Journal  2002;324(7341):816-818.
To describe parents' experience and views of the postmortem examination after the loss of a baby.
Cross sectional survey.
Hospital with a dedicated bereavement counselling service, Newcastle upon Tyne.
258 women who had attended a bereavement counselling service at the Royal Victoria Infirmary, Newcastle upon Tyne, on at least one occasion after losing a baby during pregnancy or infancy, between October 1996 and October 2000.
Self completion postal questionnaire incorporating fixed choice and open ended questions.
Main outcome measures
Number of respondents who were asked if they would agree to a postmortem examination of their baby, and number who agreed to a postmortem examination; reasons for agreeing and not agreeing to a postmortem examination; quality of explanation received; number who regretted their decision to give or withhold consent for a postmortem examination.
166 (64%) respondents completed the questionnaire. Of these, 148 (89%) had been asked to agree to a postmortem examination on their baby and 120/148 of these respondents (81%) agreed, most of whom recognised benefits resulting from the examination. 101/117 (86%) respondents believed the findings had been explained appropriately. Nine (7%) of the 120 respondents who had agreed to a postmortem examination regretted their decision. Of the respondents who refused an examination, four (14%) had regrets about their decision.
Parents viewed the postmortem examination as a useful and necessary tool in helping to discover the reasons why their baby had died. Simplifying the language used to explain findings may further raise parents' understanding of the value of the postmortem examination and ensure that they are satisfied with it. Medical staff involved in consent for postmortem examinations should be fully trained in how to ask for parental consent, the postmortem examination procedure, and how to explain the findings.
What is already known on this topicCurrent literature relates mainly to health professionals' views of the postmortem examinationThe perceived benefits of having a postmortem examination relate mainly to improving understanding of the circumstances leading to the death of the babyWhat this study addsEvery family should be offered the opportunity for a postmortem examinationThe benefits and limitations of the postmortem examination should be explained so that expectations of the outcome are appropriateMedical concepts and terminology should be fully explained during follow up and families given the opportunity to ask questions at a later date if necessary
PMCID: PMC100790  PMID: 11934774

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