Geographical variation in anophthalmia and microphthalmia in England, 1988-94

BMJ 1998;317:905-910 ( 3 October )

Papers

Editorial by Mariman

H Dolk, senior lecturer, A Busby, research fellow, B G Armstrong, senior lecturer in medical statistics, P H Walls, senior computing scientist.

Environmental Epidemiology Unit, Department of Public Health and Policy, London School of Hygiene and Tropical Medicine, London WC1E 7HT

Correspondence to: Dr Dolk h.dolk{at}lshtm.ac.uk

Objective: To investigate the geographical variation and clustering of congenital anophthalmia and microphthalmia inEngland, in response to media reports ofclusters.
Design: Comparison of pattern of residence at birth of cases of anophthalmia and microphthalmia in England in 1988-94,notified to a special register, with pattern of residence of allbirths. Three groups studied included all cases, all severe cases,and all severe cases of unknownaetiology.
Outcome measures: Prevalence rates of anophthalmia and microphthalmia by region and district, and by ward populationdensity and socioeconomic deprivation index of enumeration districtgrouped into fifths. Clustering expressed as the tendency forthe three nearest neighbours of a case to be more likely to becases than expected by chance, or for there to be more cases withincircles of fixed radius of a case than expected bychance.
Results: The overall prevalence of anophthalmia and microphthalmia was 1.0 per 10 000 births. Regional and districtvariation in prevalence did not reach statistical significance.Prevalence was higher in rural than urban areas: the relativerisk in the group of wards of lowest population density comparedwith the most densely populated group was 1.79 (95% confidenceinterval 1.15 to 2.81) for all cases and 2.37 (1.38 to 4.08) forsevere cases. There was no evidence of a trend in risk with socioeconomicdeprivation. There was very little evidence of localisedclustering.
Conclusions: There is very little evidence to support the presence of strongly localised environmental exposures causingclusters of children to be born with anophthalmia or microphthalmia.The excess risk in rural areas requires furtherinvestigation.

Key messages

Clusters of anophthalmia and microphthalmia in England have been alleged in the media, with hypothesised links to environmental exposure such as pesticides
To answer concerns about clustering a register has been established of all cases of anophthalmia and microphthalmia born in England in 1988-94
There is no large regional or district variation in prevalence
Rural areas have a roughly twofold excess in prevalence, which requires further confirmation and investigation
There is very little evidence for localised clustering in England in 1988-94

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