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J J Kurinczuk

Publications and source records attributed to J J Kurinczuk.

At least 19 recordsLinked to original sources

Who best to make the assessment? Professionals' and families' classifications of gross motor function in cerebral palsy are highly consistent.

AIM: To determine the reliability of family assessment for the Gross Motor Function Classification System (GMFCS) for children with cerebral palsy in the UK. METHODS: Families of a complete geographically defined population of children with cerebral palsy between 6 and 12 years old were identified from the 4Child epidemiological database. Postal surveys were conducted with the families and any of the child's health professionals that were nominated by the families. RESULTS: Families of 129/314 eligible children took part in the study (41%). The indices of agreement and reliability between families and professionals were also equivalent to those observed between the professionals (kappa = 0.5, ICC > or = 0.9). Reliability coefficients were higher when more of the professionals classified children using direct observation rather than only reviewing their clinical records. CONCLUSIONS: Despite excellent reliability, families and professionals did not always agree exactly on a child's GMFCS level. Classifications may differ due to children's varying performance in different environments, in which case families will almost certainly know their children's ability in a broader range of settings. The indices of reliability of family assessment for the GMFCS meet the recommended criteria for use with individuals and groups, indicating that the method is suitable for use in research studies and clinical practice.

Cerebral Palsy↗

The association between information and communication technology exposure and physical activity, musculoskeletal and visual symptoms and socio-economic status in 5-year-olds.

BACKGROUND: Increasing use of computers by children has raised concerns over the potential impact on their cognitive, social, educational, visual and physical development. Despite this concern, there are no large-scale studies relating the use of computers to specific health indicators in children as they reach school age. METHODS: A cross-sectional analysis of 1600 5-year-old Western Australian children participating in a longitudinal cohort study was conducted to ascertain their computer use, other activities (watching television and videos, playing electronic games, reading and looking at books, drawing on paper and moderate to vigorous physical activity), and specific health indicators. RESULTS: More than half (56%) of the children used computers each week. Computer use was significantly related to TV viewing (OR 1.97 weekday) and electronic game use (console games OR 2.48 weekday, 1.81 weekend; hand-held games OR 1.88 weekend) and negatively associated with vigorous physical activity on weekends (OR 0.72). Computer use was also significantly related to socio-economic indicators such as the mother being older (40+ years, OR 1.70 weekend, 1.73 weekday), tertiary educated (OR 1.63 weekend) and studying (OR 1.52 weekend, 1.41 weekday). Almost 1% children were reported to have complained of tired or sore muscles, and 2.2% had complained of tired or sore eyes, after watching television or using a computer. CONCLUSION: A substantial proportion of 5-year-old Western Australian children are using computers. Computer use was related to other sedentary activities and less vigorous activity. While musculoskeletal and vision problems are not widespread, their presence and the sedentary nature of computer use is of public health concern.

Child Behavior↗

Child or family assessed measures of activity performance and participation for children with cerebral palsy: a structured review.

BACKGROUND: There is a need to measure children's 'activity performance and participation' as defined in the World Health Organization's International Classification of Functioning, Disability and Health for Children and Youth (WHO ICF). The aim of this review is to identify instruments that are suitable for use in postal surveys with families of children with cerebral palsy. METHODS: We conducted a structured review of instruments that use child or family self-assessment of 'activity performance and participation'. The review involved a systematic search for instruments using multiple published sources. Appraisal of the instruments used the predefined criteria of appropriateness, validity, reliability, responsiveness, precision, interpretability, acceptability and feasibility. RESULTS: There are relatively few child or family assessed instruments appropriate for measuring children's activities and participation. Seven instruments were identified that could potentially be administered by mail. The Assessment of Life Habits for Children (LIFE-H) was the most appropriate instrument as assessed by its content but the reliability of child or family self-assessment is not known. If the LIFE-H were shown to be a reliable self-report measure then the LIFE-H would be the recommended choice. Currently, the Activities Scale for Kids and the condition-specific Lifestyle Assessment Questionnaire for cerebral palsy (LAQ-CP) provide the broadest description of what and how frequently children with cerebral palsy perform a range of activities and thereby indicate participation. The LAQ-CP also provides additional contextual information on the impact of any disability on the participation of the family unit. CONCLUSION: There remains much scope for developing valid and reliable self-assessed measures corresponding to the WHO ICF dimensions of activities and participation.

Activities of Daily Living↗

Congenital cystic adenomatoid malformation: accuracy of prenatal diagnosis, prevalence and outcome in a general population.

OBJECTIVES: Most available data regarding accuracy of prenatal diagnosis, prevalence and outcome of congenital cystic adenomatoid malformation (CCAM) are derived largely from tertiary referral centres and may not reflect general population rates. We aimed to describe the accuracy of prenatal diagnosis, ascertain the population prevalence and post-natal outcome for cases of suspected CCAM. METHODS: Retrospective collection of prenatal and paediatric data for cases of suspected CCAM notified to the Trent Congenital Anomalies Register 1997 to 2001. RESULTS: Thirty-seven cases of CCAM were suspected prenatally. Twenty-one cases were confirmed post-natally as having a CCAM (positive predictive value 57%). Eighteen of the 21 cases were delivered at term as live births, 15 of which have undergone successful surgery to date. Thirteen of the 37 cases had apparently resolved by delivery. Three further cases were subsequently found to be cases of lung sequestration or lobar emphysema. Five cases of CCAM were detected after delivery (sensitivity of prenatal detection 81%). The population prevalence at delivery was 9.0 per 1,00,000 total births. Five confirmed cases of CCAM developed hydrops, three required in utero intervention and delivered as live births at term, one was terminated and one died in utero. The overall mortality in the confirmed cases of CCAM was 23% of which the majority were terminations of pregnancy. CONCLUSIONS: Problems of diagnostic accuracy and apparent resolution of CCAM render counselling difficult, although our data suggest that the prognosis is better than others have reported. Confirmation of the diagnosis in the neonatal period is vital in order to obtain the true population prevalence figures and to interpret outcome data.

Adult↗

Antenatal detection of a single umbilical artery: does it matter?

The presence of a single umbilical artery is recognised as a soft marker for congenital anomalies, aneuploidy, earlier delivery and low birthweight. Most of the available data are derived from case series or highly selected populations and are therefore likely to be unrepresentative. In this retrospective case-comparison study, we firstly aimed to determine the incidence of a single umbilical artery in an unselected population and secondly to examine the clinical significance of this soft marker. Over a 40-month period, 107 cases were identified from a cohort of 35 066 births giving an incidence of 3.1 per 1000 total births and late pregnancy losses. The antenatal detection rate was only 30%. Compared to fetuses with normal cord vasculature, fetuses with a single umbilical artery were more likely to be delivered at an earlier gestation and to weigh less, were 1.7 times more likely to be delivered by a Caesarean section and 19% of the cases had a congenital anomaly. The perinatal mortality was 49.0 per 1000 total births, which was 6 times higher than the background hospital rate. The presence of a single umbilical artery is associated with a poorer perinatal outcome compared to that in fetuses with three vessels in the cord. Unfortunately, the antenatal detection rate is poor. Recognising the importance of this soft marker in counselling and management of pregnancies should provide the stimulus to improve detection rates.

Abnormalities, Multiple↗

Congenital hypothyroidism in Western Australia 1981-1998.

OBJECTIVE: Newborn screening for congenital hypothyroidism began in Western Australia in 1981. We describe the epidemiology, including trends over time. METHODOLOGY: Population-based registers and databases were used to identify cases of congenital hypothyroidism in 1981-1998 (n = 126), a random sample of controls (n = 1260), and descriptive data on cases and controls. The prevalence of congenital hypothyroidism per 10 000 live births was calculated, and a case-control analysis conducted. RESULTS: The prevalence of congenital hypothyroidism was 1.74 per 10 000 in the period 1981-1987, compared with 3.54 per 10 000 between 1988 and 1998. Infants identified with congenital hypothyroidism were significantly more likely to have other birth defects (particularly heart defects), be female, have a birthweight heavier than 4500 g, and be born either preterm or after 41 weeks gestation. No cases had cerebral palsy or intellectual disability. CONCLUSIONS: Population-based registers and linked databases can provide very useful information for evaluating screening programmes, and extending our knowledge of the epidemiology of congenital hypothyroidism.

Abnormalities, Multiple↗

A confidential enquiry into cases of neonatal encephalopathy.

OBJECTIVES: To assess the quality of care and timing of possible asphyxial events for infants with neonatal encephalopathy; to compare the quality of care findings with those relating to the deaths from the Confidential Enquiry into Stillbirths and Deaths in Infancy (CESDI); and to assess whether the confidential enquiry method is a useful clinical governance tool for investigating morbidity. DESIGN: Independent, anonymised, multidisciplinary case reviews. SETTING: Trent Health Region, UK. PATIENTS: All cases of grade II and III neonatal encephalopathy born in 1997, excluding those due to congenital malformation, inborn error of metabolism, or infection. All CESDI deaths thought to have resulted from intrapartum asphyxia in 1996 and 1997. MAIN MEASURES: Quality of care provided, timing of possible asphyxial episodes, and the source and timing of episodes of suboptimal care. RESULTS: Significant or major episodes of suboptimal care were identified for 64% of the encephalopathy cases and 75% of the deaths. An average of 2.8 and 2.5 episodes of suboptimal care were identified for the deaths and encephalopathy cases respectively. Over 90% of episodes involved the care provided by health professionals. Results were fed directly back to the units concerned on request and changes in practice have been reported. CONCLUSIONS: The findings were very similar for the encephalopathy cases and the deaths. We have demonstrated that with minor adaptations the CESDI process can be applied to serious cases of morbidity. However, explicit quality standards, control data, and a more formal mechanism for the implementation of findings would strengthen the confidential enquiry process as part of clinical governance.

Asphyxia↗

Developmental outcomes of newborn encephalopathy in the term infant.

Newborn encephalopathy is a clinically defined condition of abnormal neurological behaviours in the newborn period. Though most cases have their origin in the preconceptional and antepartum period, newborn encephalopathy represents a crucial link between intrapartum events and permanent neurological problems in the child. The birth prevalence of newborn encephalopathy ranges from 1.8 to 7.7 per 1000 term live births according to the definition used and the population to which it is applied. Few studies have investigated the outcomes of newborn encephalopathy other than for cases solely attributed to intrapartum hypoxia. These adverse outcomes range from death to cerebral palsy, intellectual disability, and less severe neurological disabilities such as learning and behavioural problems. Outcomes following newborn encephalopathy may vary from country to country with 9.1% of affected babies dying in the newborn period in Western Australia and 10.1% manifesting cerebral palsy by the age of two. These compare to a case fatality of 30.5% in Kathmandu and a cerebral palsy rate of 14.5% by one year of age. The study by Robertson et al which followed children with hypoxic ischaemic encephalopathy found an incidence of impairment of 16% among survivors assessed at 8 years with 42% requiring school resource room help or special classes. This review emphasises the great need for comprehensive clinical and educational assessment as these infants approach school entry to enable appropriate educational provisions to be made.

Brain Damage, Chronic↗

Case-control study of leatherwork and male infertility.

OBJECTIVES: To test the hypothesis that leatherwork is associated with male infertility mediated through the development of oligozoospermia. The basis of any association was postulated, at the outset, to be with exposure to the solvents used in leatherwork. METHODS: All new referrals with infertility presenting in Leicestershire hospital clinics between November 1988 and September 1992 and Kettering District General Hospital from August 1990 were eligible to participate; 88.5% agreed to be interviewed. Exposure to leatherwork and work with solvents was defined by job title. Comparisons were made with fertile controls and in an analysis within men from infertile couples with oligozoospermia as the primary outcome. Effects on sperm motility and deformity were investigated secondarily. Analyses used logistic regression for binary outcomes and multilevel modelling for continuous outcomes. RESULTS: 1906 men were interviewed. Compared with the fertile controls the men from infertile couples were 1.10 times (95% confidence interval (95% CI) 0.46 to 2.63; p=0.99) more likely to be leatherworkers and 1.73 times (95% CI 1.26 to 2.38; p<0.001) more likely to work with solvents. Compared with other men, leatherworkers were 1.20 times (95% CI 0.43 to 3.33; p=0.73) more likely to present with oligozoospermia and 1.65 times (95% CI 0.37 to 7.30; p=0.51) more likely to present with teratozoospermia. Being a leatherworker was associated with only a 6% reduction in sperm concentration; motility and deformity were similarly unaffected by this exposure. Work with solvents did not statistically, nor clinically, increase the risk of oligozoospermia, teratozoospermia, or asthenozoospermia. CONCLUSIONS: There was little evidence to support the hypothesis that leatherwork is associated with an increased risk of presenting with infertility or oligozoospermia. There was limited evidence that leatherwork is a risk factor for teratozoospermia. Workers with solvents were at an increased risk of presenting with infertility, although this was not mediated through effects on standard measures of semen quality; this finding merits further investigation.

Adult↗

Trends in the frequency and predictive value of reporting high grade abnormalities in cervical smears.

BACKGROUND: The "organized approach" to cervical screening in Australia includes standardized quality assurance measures for laboratories. This study examines changes in the frequency and the positive predictive value of reporting severe abnormalities in cervical smears over a 3-year period as a guide to the effects of implementing these measures. METHODS: The results of screening in 6-month periods from January 1995 to December 1997 were determined. Biopsy follow-up for results in the high grade epithelial abnormality ("HGEA") and "inconclusive: possible HGEA" categories was obtained from the Western Australian Cervical Cytology Registry (CCR). RESULTS: Approximately 40,000 smears were examined in each 6-month period. The frequencies of reporting HGEA were 0.47%, 0.59%, 0.79%, 0.85%, and 0.84%, and 0.91% for the study periods (P < 0.001). For the inconclusive category, they were 0.24%, 0.18%, 0.24%, 0.31%, 0.38%, and 0.35% (P < 0.001). Biopsy follow-up was available for 83. 9%, 80.5%, 89.9%, 92.4%, 93.1%, and 90.3% of the HGEA results and for 78.6%, 71.7%, 80.5%, 75.0%, 87.1%, and 85.9% of the inconclusive results over the study periods. The yield of high grade lesions for the biopsied cases was 82.6%, 82.3%, 83.1%, 79.5%, 80.9%, and 79% for HGEA cases and 58.2%, 41.9%, 60.6%, 52.8%, 47.5%, and 54.1% for inconclusive cases. CONCLUSIONS: There was a doubling in the reporting of HGEA results, whereas the positive predictive value for biopsied cases remained at about 80%. Reporting rates for inconclusive: possible HGEA cases also doubled, but the yield of biopsy-proven, high grade lesions remained at about 50%. These changes occurred in the absence of ancillary testing and with targeted rescreening methods. A high rate of reporting HGEA, in combination with a high positive predictive value, is among the most important indicators of cervical cytology laboratory performance. Large improvements in results may occur using conventional methods of quality assurance. Cancer (Cancer Cytopathol)

Adenocarcinoma↗

Fetal growth and subsequent mental health problems in children aged 4 to 13 years.

To test the hypothesis that children with suboptimal fetal growth have significantly poorer mental health outcomes than those with optimal growth, a population random sample survey of children aged 4 to 16 years in Western Australia in 1993 was conducted. The Child Behavior Checklist (Achenbach 1991a) and the Teacher Report Form (Achenbach 1991b) were used to define mental health morbidity. Survey data for 1775 children aged 4 to 13 years were available for linkage with original birth information. The percentage of expected birthweight (PEBW) was used as the measure of fetal growth. Children below the 2nd centile of PEBW who had achieved only 57% to 72% of their expected birthweight given their gestation at delivery were at significant risk of a mental health morbidity (OR 2.9, 95% CI 1.18, 7.12). In addition, they were more likely to be rated as academically impaired (OR 6.0, 95% CI 2.25, 16.06) and to have poor general health (OR 5.1, 95% CI 1.69, 15.52).

Adolescent↗

Birth defects in children with newborn encephalopathy.

This study was designed to investigate birth defects found in association with newborn encephalopathy. All possible birth defects were ascertained in a population-based study of 276 term infants with moderate or severe encephalopathy and 564 unmatched term control infants. A strong association between birth defects and newborn encephalopathy was found with defects affecting 27.5% of children with encephalopathy and 4.3% of control children (odds ratio 8.55; 95% confidence interval 5.25 to 13.91;p<0.001). In 11.8% of infants with a birth defect the defect was not diagnosed until after the newborn period, illustrating one of the difficulties in attempting to exclude infants with birth defects from studies of newborn encephalopathy. The majority of defects (89%) were not specific anomalies of the CNS. In 36.8% of children with encephalopthy who had a birth defect, the defect was considered to be the probable cause of the encephalopathy. Infants with birth defects who had encephalopathy had a poorer prognosis than those without: they were twice as likely to die by the age of 2 years and three times more likely to have cerebral palsy. This study catalogues the spectrum of birth defects associated with newborn encephalopathy and illustrates the importance of their inclusion when investigating both the aetiology and outcome of this condition.

Abnormalities, Multiple↗

Bayesian statistics in medical research: an intuitive alternative to conventional data analysis.

Statistical analysis of both experimental and observational data is central to medical research. Unfortunately, the process of conventional statistical analysis is poorly understood by many medical scientists. This is due, in part, to the counter-intuitive nature of the basic tools of traditional (frequency-based) statistical inference. For example, the proper definition of a conventional 95% confidence interval is quite confusing. It is based upon the imaginary results of a series of hypothetical repetitions of the data generation process and subsequent analysis. Not surprisingly, this formal definition is often ignored and a 95% confidence interval is widely taken to represent a range of values that is associated with a 95% probability of containing the true value of the parameter being estimated. Working within the traditional framework of frequency-based statistics, this interpretation is fundamentally incorrect. It is perfectly valid, however, if one works within the framework of Bayesian statistics and assumes a 'prior distribution' that is uniform on the scale of the main outcome variable. This reflects a limited equivalence between conventional and Bayesian statistics that can be used to facilitate a simple Bayesian interpretation based on the results of a standard analysis. Such inferences provide direct and understandable answers to many important types of question in medical research. For example, they can be used to assist decision making based upon studies with unavoidably low statistical power, where non-significant results are all too often, and wrongly, interpreted as implying 'no effect'. They can also be used to overcome the confusion that can result when statistically significant effects are too small to be clinically relevant. This paper describes the theoretical basis of the Bayesian-based approach and illustrates its application with a practical example that investigates the prevalence of major cardiac defects in a cohort of children born using the assisted reproduction technique known as ICSI (intracytoplasmic sperm injection).

Bayes Theorem↗

Maternal thyroid disease: a risk factor for newborn encephalopathy in term infants.

Two previously published studies of term newborn encephalopathy showed that maternal thyroid disease to be a risk factor. From these studies we identified 13 case and three control mothers with thyroid disease and investigated them further. The majority of affected case mothers had idiopathic or autoimmune hypothyroidism. Compared with control mothers, case mothers had fewer thyroid function tests in pregnancy, were more likely to remain on the same dose of medication throughout pregnancy and to have experienced other pregnancy complications. The association between maternal thyroid disease and encephalopathy may be the result of a series of different causal pathways, some of which are suggested by our data.

Brain Diseases↗

Why is the placenta being ignored?

The relationship between the frequency of published recommended indications for placental pathological examination and the frequency of requests for such examination in a population-based study of term newborn encephalopathy was examined. Only 11.2% of placentas among 276 case infants and 0.7% of placentas among 564 term control infants were examined. Using the criteria set out in a consensus statement by the American College of Pathologists, all 276 cases fulfilled multiple maternal, fetal and placental indications for placental examination. Furthermore 43.3% of control infants fulfilled at least one criterion. Of the 25 case placentas that underwent pathological review, 16 were reported as having no diagnostic abnormality Six cases (24%) showed clinically important findings: four had evidence of infection, one had multiple chorangiomata and one had thrombosis and rupture of the umbilical vein. Of the three remaining placentas, one showed funisitis, one showed minor lymphohistiocytic villitis and one was from monochorionic twins. To our knowledge there are no agreed Australian guidelines for when a placenta should be submitted for pathological examination. We suggest that until guidelines based on properly designed studies are developed it may be appropriate to store all placentas for at least 72 hours. If the infant develops neurological symptoms or requires unexpected admission to a neonatal intensive care unit then placental examination may reveal important aetiological diagnostic and prognostic information.

Australia↗

Assessment of separate contributions to perinatal mortality of infertility history and treatment: a case-control analysis.

BACKGROUND: Few studies have described the perinatal risks associated with infertility, other than for infertility treated by in-vitro fertilisation or gamete intrafallopian transfer. The aim of this analysis was to estimate the risks of perinatal death associated with treated and untreated infertility. METHODS: A population-based case-control study of perinatal deaths was carried out in Leicestershire Health District over the period 1990-94, during which 60,922 babies were delivered. Of these, 567 perinatal deaths were associated with 542 women. 972 mothers were randomly selected as controls. Medical, obstetric, and social data were collected for cases and controls from the medical notes and interviews with the women. The relative risks of perinatal death associated with treated and untreated infertility before the index pregnancy were estimated as odds ratios by means of unconditional logistic regression analysis. FINDINGS: 65 (10%) of cases and 34 (3.5%) of the controls had infertility before the index pregnancy. History of infertility in the index pregnancy, irrespective of treatment, increased the risk of perinatal death (odds ratio 2.9 [95% CI 1.8-4.5]). The population attributable risk fraction for perinatal death related to infertility was 6.2% (3.4-9.0). 45 (54%) of the deaths, even in the untreated group, were associated with immaturity. Compared with women without infertility, women with untreated infertility were at increased risk of perinatal death (3.3 [1.6-6.8]). The risk of perinatal death associated with multiple births did not explain this finding. Similarly, treated infertility also increased the risk of perinatal death (2.7 [1.5-4.7]); the risks associated with multiple births explained some, but not all, of this excess. In Leicestershire, the overall underlying risk of a mother experiencing at least one perinatal death over the study was 9.0 per 1000 women. For women who experience infertility, this risk increases by about 18 per 1000 (6-30). INTERPRETATION: Counselling for women before any form of infertility treatment should include discussion of the risks of perinatal death. Our results would benefit from confirmation. However, we advocate that at antenatal booking a history of infertility, irrespective of treatment, should be sought, because these women have a significantly increased risk of perinatal death, particularly associated with prematurity.

Adult↗