Papers by Svetlana Glinianaia
ABSTRACT Background The measurement of quality of life (QoL) has become increasingly important in... more ABSTRACT Background The measurement of quality of life (QoL) has become increasingly important in clinical practice. For children with chronic illnesses, QoL is an important dimension of assessment owing to advances in medical care and an increase in survival rates for many conditions. However, there is limited research on QoL in children born with congenital anomalies. Gastroschisis, a congenital anomaly of the abdominal wall, has a good prognosis overall but requires surgical correction and repeated hospitalisation. The aim of this pilot study was: (1) to test whether the KIDSCREEN-52 questionnaire could be used to measure QoL in children born with gastroschisis; and (2) to assess self-reported QoL in these children. Methods The KIDSCREEN-52 is a validated QoL measure developed and normed across 13 European countries as a self-report questionnaire for healthy and chronically ill children/adolescents (8–18 years) covering ten domains. Children (8–12 years) born with gastroschisis, identified from the Northern Congenital Abnormality Survey, were invited to complete the questionnaire, the relevance of the questions was discussed through cognitive interviewing with the children and their parents, and their QoL scores were compared to age-matched UK norms. Results Ten children (six females, four males, mean age 9.6 years) and ten parents completed the questionnaire. Children and parents found it a good measure of QoL with further explanation needed on only a few items. Children reported finding it a helpful tool in exploring their feelings and that it covered areas important to them. Parents were positive acknowledging that all priority areas were represented and that it was straightforward for their children to complete. All ten children described their health as good/very good or excellent. Seven said that they were happy with the way they are, eight were seldom/never worried about the way they looked and eight said they would not like to change anything about their body. In six domains (physical wellbeing; moods and emotions; self-perception; relationships with parents; social support and peers; school environment) children with gastroschisis had similar QoL scores, in three domains (psychological wellbeing; autonomy; social acceptance) their QoL scores were significantly better (p < 0.05) and in one domain (financial resources) significantly lower (p < 0.01) than in the reference group. Conclusion This study has demonstrated that the KIDSCREEN questionnaire has adequate face validity as a measure of QoL in these children, is acceptable to both children and parents, and suggests that children with gastroschisis have similar, or better, QoL scores than other children. Acknowledgements This abstract was first presented as a poster at Perinatal Medicine 2014, 9–11 June 2014, Harrogate International Centre, Harrogate, UK, and published as: Rankin J, Jardine J, Glinianaia SV, Borrill H, McConachie E, Embleton ND. Self-reported quality of life in 8-12 year old children born with gastroschisis: a pilot study. Archives of Disease in Childhood Fetal & Neonatal Ed. 2014;99(Suppl 1):A52.
Paediatric and perinatal epidemiology, 2000
Using data from the Northern Congenital Abnormality Survey, we describe trends in the prevalence ... more Using data from the Northern Congenital Abnormality Survey, we describe trends in the prevalence of neural tube defects (NTDs), maternal age-specific prevalence, outcome of pregnancy and the sensitivity of antenatal diagnosis of NTD-affected pregnancies in the Northern Region during 1984-96. This population-based sample consisted of 934 NTDs: there were 403 (43.1%) with anencephaly, 472 (50.5%) with spina bifida and 59 (6.3%) with encephalocele. The total prevalence at birth was 17.9 per 10,000 births and terminations (95% confidence interval [CI] = 16.7, 19.0) with a significant reduction over time (chi(2) for trend = 7.2; P = 0.007). The overall birth prevalence was 5.6 per 10,000 births [95% CI = 5.0, 6.3]; there was also a significant reduction in birth prevalence with time (chi(2) for trend = 68.3; P < 0.0001). Maternal age-specific prevalence rates decreased with increasing age. The proportion of NTD pregnancies terminated increased from 60.3% (325 cases) during 1984-90 to ...
There is little consistency in the use of instruments for measuring self-reported quality of life... more There is little consistency in the use of instruments for measuring self-reported quality of life (QoL) in young children. To systematically review studies of self-reported QoL in children aged &amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;lt;12 years with congenital health conditions, and to examine the agreement between self- and proxy-reports. Literature databases (MEDLINE, EMBASE, Web of Science, PsychINFO) were systematically searched, reference lists of eligible studies were scanned. We included studies published in English between January 1989 and June 2013 which used validated instruments to assess self-reported QoL in children aged &amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;lt;12 years with a distinct congenital health condition identified in early infancy. We extracted data on study design, objective, sample characteristics, QoL assessment instrument, statistical techniques and results. From 403 full-text articles assessed for eligibility, 50 studies underwent detailed review, and 37 were included in a narrative synthesis. Children&amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;#39;s self-reported QoL was assessed by using a variety of generic and/or condition-specific instruments, with the Pediatric Quality of Life Inventory being the most frequently used (25% [9 studies]). Regardless of the condition or the instrument used, children often reported QoL similar to the reference population, except for lower scores in the physical functioning/health domain. There were differences between younger and older age groups according to QoL domain. The child&amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;#39;s perception of QoL differed from that of his or her parents, in particular for subjective domains such as emotional functioning, and these differences were age related. The main limitation of the review resulted from the lack of published studies on self-reported QoL in young children, in particular, lacking both self-reports and proxy reports. Existing studies demonstrated wide variability in the QoL instruments used and approaches to statistical analyses, lack of information about the formation of the study sample (response rate; comparison of responders and nonresponders) and low sample sizes in the age group of interest. The reviewed studies demonstrated that, even for younger children, both child and parent perspectives are essential to understanding the impact of a condition on a child&amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;amp;#39;s QoL.
BackgroundWomen with pre-gestational diabetes (PGDM) are at increased risk of pregnancy affected ... more BackgroundWomen with pre-gestational diabetes (PGDM) are at increased risk of pregnancy affected by a congenital anomaly. National guidance recommends enhanced antenatal ultrasound screening for cardiovascular defects.AimTo compare antenatal detection of congenital anomaly, and outcomes of affected pregnancies, in women with and without PGDM.MethodsWe linked data for 1996–2008 from the Northern Diabetes in Pregnancy and Northern Congenital Abnormality Surveys in the
ABSTRACT Aim To describe the prevalence, pregnancy outcome and survival of higher order multiple ... more ABSTRACT Aim To describe the prevalence, pregnancy outcome and survival of higher order multiple pregnancies. Methods Epidemiological analysis of data prospectively notified to the Northern Survey of Twin and Multiple Pregnancy (NorSTAMP), the only population-based multiple pregnancy register in the UK, during 1998–2011. Results There were a total of 160 higher order pregnancies (495 fetuses) notified to NorSTAMP during the 14 years comprising 148 (92.5%) triplet pregnancies, 10 (6.3%) quadruplets, one (0.6%) quintuplet and one (0.6%) sextuplet pregnancy. The prevalence of triplet maternities decreased from 4.2 per 10,000 maternities in 1998–2004 to 1.9 per 10,000 maternities in 2005–2011 (RR = 0.45, 95% CI 0.31–0.64, p &lt; 0.0001). From a total of 444 triplets, there were 71 spontaneous fetal losses (&lt;24 weeks), 13 feticides or terminations of pregnancy and 340 live births. There were 20 stillbirths (17 antepartum, three intrapartum) and 19 infant deaths (nine early neonatal, five late neonatal and five postneonatal). The overall extended perinatal mortality rate (EPMR, stillbirths and neonatal deaths) for triplets was 94.4 per 1000 total births and the infant mortality rate was 55.9 per 1000 live births. The EPMR decreased from 102.5 per 1000 total births in 1998–2004 to 77.6 per 1000 in 2005–2011 (RR = 0.76, 95% CI 0.37–1.57, p = 0.56). Infant mortality decreased from 77.6 per 1000 live births to 9.3 per 1000 over the same periods (RR = 0.12; 95% CI 0.02–0.88, p = 0.01). Conclusion Triplet rates have significantly decreased during the study period, most likely due to changes in assisted reproductive policies. Mortality outcomes, particularly infant mortality, have improved.
Background: Exposure to air pollutants is suggested to adversely affect fetal growth, but the evi... more Background: Exposure to air pollutants is suggested to adversely affect fetal growth, but the evidence remains inconsistent in relation to specific outcomes and exposure windows.
Background Twins have a much higher stillbirth rate than singletons with causes of intrauterine d... more Background Twins have a much higher stillbirth rate than singletons with causes of intrauterine death frequently unexplained. In the North of England the stillbirth rate in twins is higher than the national rate.AimTo audit antenatal surveillance of twin pregnancy resulting in antepartum stillbirth against regional consensus standards of care.MethodsA retrospective, case-note analysis of all antepartum stillbirths in twins delivered 2005–2009, identified from the population-based Northern Survey of twin and multiple pregnancy. Antenatal care was reviewed independently by two consultants.ResultsThere were 43 antepartum stillbirths in 38 pregnancies. 21 were DCDA, 14 MCDA, one MCMA and in two cases the chorionicity was unknown. Autopsy was performed in 18 (47%) pregnancies. Major causes of stillbirth were acute or chronic antepartum hypoxia (n=25) and twin-to-twin transfusion (TTTS n=14, 9 pregnancies). Case-note review clarified causes of death in 22 cases confirming TTTS in 8 pregna...
Archives of disease in childhood. Fetal and neonatal edition, 2014
Partial urorectal septum malformation (pURSM) sequence (or 'persistent cloaca') is a rare... more Partial urorectal septum malformation (pURSM) sequence (or 'persistent cloaca') is a rare congenital anomaly characterised by a joining of the urethral, anal, and genital openings into a single common channel. This study describes the epidemiology of pURSM sequence in England and Wales including prevalence, additional anomalies, and pregnancy outcomes. All cases of pURSM sequence prospectively notified to seven congenital anomaly registers in England and Wales during 1985-2010, whether delivered as live births, spontaneous fetal deaths (≥20 weeks' gestation), or elective terminations of pregnancy for fetal anomaly (TOPFA, any gestation), formed this population-based cohort. The risks of spontaneous fetal and infant death were examined by Kaplan-Meier analysis. Differences in prevalence over time, and between regions, were examined by multilevel Poisson regression. 117 cases were recorded among 4,251,241 total births. Six (5%) pregnancies resulted in spontaneous fetal dea...
Twin Research and Human Genetics, 2013
The population-based Northern Survey of Twin and Multiple Pregnancy (NorSTAMP, formerly the Multi... more The population-based Northern Survey of Twin and Multiple Pregnancy (NorSTAMP, formerly the Multiple Pregnancy Register) has collected data since 1998 on all multiple pregnancies in North of England (UK) from the earliest point of ascertainment in pregnancy. This paper updates recent developments to the NorSTAMP and presents some early mortality data from the first 10 years of data collection (1998-2007). Since 2005, mothers have been asked to give explicit consent for their identifiable data to be held by the survey, in line with changing guidance and legal frameworks for identifiable data. In 2009, regional standards of care for multiple pregnancies were developed, agreed, and disseminated. During 1998-2007, 4,865 twin maternities (pregnancies with at least one live birth or stillbirth) were registered, with an average twinning rate of 14.9 per 1,000 maternities. The overall stillbirth and neonatal mortality rates in twins were 18.0/1,000 births and 23.0/1,000 live births respectively. Stillbirth and neonatal mortality rates were significantly higher in monochorionic than dichorionic twins: 44.4 versus 12.2 per 1,000 births (relative risk [RR] 3.6, 95% Confidence Intervals [CI] 2.6-5.1), and 32.4 versus 21.4 per 1,000 live births (RR 1.5, 95% CI 1.04-2.2) respectively. There was no significant improvement during this period in either stillbirth or neonatal mortality rates in either chorionicity group. This population-based survey is an important source of data on multiple pregnancies, which allows monitoring of trends in multiple birth rates and pregnancy losses, providing essential information to support improvements in clinical care and for epidemiological research.
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Papers by Svetlana Glinianaia