PubMed HealthSearch

Biomedical subjects

J P Bound

Publications and source records attributed to J P Bound.

11 recordsLinked to original sources

Down's syndrome: prevalence and ionising radiation in an area of north west England 1957-91.

OBJECTIVE: To analyse the prevalence of Down's syndrome in a specific, geographical area and seek to explain variations with particular reference to ionising radiation. DESIGN: Cases were ascertained by one paediatrician as part of a prospective survey of major congenital malformations in children born to residents of an area of Lancashire between 1957 and 1991. Temporal changes in prevalence rates were detected by a grid search technique using Poisson log linear models. These models were also used to determine the association between prevalence and ionising radiation from atomic fall out. SETTING: The Fylde district of Lancashire in the north west of England. PATIENTS: There were 167 cases, including five stillbirths and eight terminations, among 124,015 total births in a population which increased from about 250,000 to over 300,000 during the study period. MAIN RESULTS: There was significant increase in the prevalence of all cases conceived in 1963 and 1964, and a lesser peak in 1958 which did not quite reach statistical significance. There was no evidence that the increased prevalence in 1963-64 was a result of changes in the maternal age distribution in the population. Babies of mothers aged 35 years and over accounted for more of the variation, especially in 1958 when their increase was significant. There was a highly significant association between prevalence and radiation from fallout produced by atmospheric testing of atomic weapons. The 1963-64 peak coincided with the maximum estimated radiation dose. The lesser peak in 1958 also coincided with increased exposure to radiation from fallout, possibly enhanced by ground deposits after a fire at the Windscale reactor in October 1957. CONCLUSION: This study provides further support for low dose ionising radiation as one aetiological factor in Down's syndrome.

Dose-Response Relationship, Drug

Neural tube defects, maternal cohorts, and age: a pointer to aetiology.

The effects of maternal year of birth and age on the declining prevalence of neural tube defects after 1972-3 were examined using 403 cases ascertained in a prospective study in the Fylde of Lancashire during 1957-89. Matched case-control data were analysed using conditional logistic regression analysis. The risk of an anencephalic baby was significantly greater for older mothers, but changes in the maternal age distribution in the population did not appear to be relevant to the recent decline in prevalence. Antenatal diagnosis and termination of pregnancy was the major cause. Mothers born before 1950 were at significantly greater risk of producing a baby with spina bifida or cranium bifidum. We suggest that abandonment of mercury as a therapeutic agent for infants in the early 1950s is a possible factor in the current decline of these malformations.

Adult

Congenital malformations in the Fylde region of Lancashire, England 1957-1973.

This paper uses data collected by a consultant paediatrician to examine variations in the prevalence of neural tube and cardiovascular malformations within the Fylde region of North West England. Results at the district scale indicate contrasts in the geographical distributions of the two classes of malformation and these are then further assessed via a case-control study which standardises for factors such as date of conception, age of mother and parity. The results of this study suggest that there were wards in Blackpool and Fleetwood with unusually high prevalences of neural tube defects. Further research is being undertaken to identify the causes of these concentrations.

Case-Control Studies

Seasonal prevalence of major congenital malformations in the Fylde of Lancashire 1957-1981.

The seasonal prevalence of major congenital malformations was studied in a prospective survey of 88,449 children born in the circumscribed Fylde of Lancashire to residents there over 25 years. Ascertainment was thought to be as complete as was practically possible because cases were recorded daily by one, and for 17 years the only, paediatrician and a very high rate of necropsies was maintained. The number of malformations were classified by month of maternal last menstrual period and seasonal variation was assessed by three statistical models. Neural tube defects showed a significant seasonal variation in month of last menstrual period but not in month of birth. From May 1956 to April 1968, when the prevalence of neural tube defects was high (5.5 per 1000 total births), conceptions were significantly more common in December to May. For anencephaly alone the figures were not significant, but spina bifida and cranium bifidum were more common in March to May. From May 1968 to April 1981, when the prevalence of neural tube defects fell below the national average, the significant variations disappeared. Seasonality for spina bifida and cranium bifidum was seen only in "singles" (cases with no other major lesion), but for anencephaly it was seen only in "multiples" (cases with other lesions). The three types of cardiac septal defect and persistent ductus each showed a higher prevalence of conceptions at some time during May to October. In contrast the commonest group of cyanotic cases showed no such pattern but with greater numbers in winter. There was evidence of a seasonal variation for bilateral renal agenesis and for vesicoureteric reflux as ascertained. Seasonal prevalence in an aetiological factor for certain malformations of the central nervous system, cardiac and urinary systems.

Causality

Congenital malformations associated with anencephaly in the Fylde peninsula of Lancashire.

The necropsy reports of 174 cases of anencephaly, born in the Fylde peninsula of Lancashire between 1957 and 1980, have been analysed for the presence of other malformations. The results were compared with a similar previous series from Bristol, though the Bristol study differed both in time (1948 to 1975) and in the fact that it was hospital based and, unlike the present study, did not achieve near complete ascertainment. Of the Lancashire anencephalics, 24% had other malformations, a significantly lower rate than in the Bristol series. There was a much higher rate of renal and urinary tract defects in the Bristol series, and a higher rate of cardiovascular defects in the Lancashire series. The distribution of associated malformations differed in the two areas, possibly representing different patterns of aetiological heterogeneity. The iniencephaly rate was so much lower in Lancashire as to suggest an artefact, perhaps owing to the lack of a precise definition of the condition.

Abnormalities, Multiple

The incidence of anencephalus in the Fylde peninsula 1956-76 and changes in water hardness.

In an area which had a high incidence of anencephalus, 3.2 per 1000 births, there was a significant drop to 1.3 per 1000, below the national average of 1.7 to 1.8, among conceptions after 1967. In the northern part of the area this drop was greater in summer than winter conceptions, providing additional evidence of a different process underlying case occurrences in the north of the area from that in the south. In 1957-61 there were significantly more births of anencephalic babies in North Fylde than in South Fylde, but both areas were supplied with soft water. From 1962 to 1969 the water changed from soft to slightly hard. Soft water does not appear to be a primary aetiological factor in anencephalus, but hard water may mitigate the effect of other factors.

Anencephaly

Incidence of congenital heart disease in Blackpool 1957-1971.

In the 15 years from 1957 to 1971, there were 57 979 births in a circumscribed population in North-West England. The requirements for a valid survey of the incidence of congenital heart disease were largely met. The necropsy rate for all stillbirths and neonatal deaths in the population was 93 per cent. Diagnosis of the type of heart lesion was by necropsy in 50 per cent, cardiac catheterisation in 19 per cent, operation in 5 per cent, and clinical observations only in 26 per cent. The incidence of congenital heart disease was 6-8 per 1000 total births and 5-9 per 1000 live births. The overall incidence has remained unchanged over the 15 years, but there were trends suggesting an increase in the incidence of uncomplicated ventricular septal defect and of endocardial cushion defect and a decrease in the incidence of ventricular septal defect with right ventricular outflow obstruction and of hypoplastic left heart. We have observed seasonal variations in total incidence and in incidence of some common individual lesions.

Child, Preschool