Abstract
BackgroundSpina bifida is a disorder of the cerebrospinal fluid system associated with failure of neural tube closure in the fetus. Reproductive history studies of mothers with spina bifida offspring have often been conducted shortly after the affected child's birth. In this study, a large group of community-based mothers were studied after most had completed their families. The aims were to present a more comprehensive reproductive history and to test several hypotheses regarding the nature of spina bifida.MethodsData from 271 mothers was collected by interview 18.3 mean years after the affected child's birth. Data analysis was by χ-square, Fisher exact test and t test with a p value less than 0.05 considered significant.ResultsFemales made up 56.5% of affected offspring (probands) and 53.1% of unaffected offspring. The spina bifida and anencephaly recurrence rate was 4.0%. The twinning rate was 8.6/1000 live births. 24.4% of mothers had a history of spontaneous abortion and the rate varied by pregnancy order from 87 to 185/1000 live births. Duration of pregnancies subsequent to probands was shorter for female than male probands. Mean birth weight of probands with high lesions exceeded those with low lesions. A spontaneous abortion preceded female probands more often than males as compared to live births. Affected males with high lesions conceived by white mothers were at greater risk to be spontaneously aborted. Previous inter-gestational interval for mothers with no history of spontaneous abortion was longer for probands than unaffected offspring but not for mothers with a history of spontaneous abortion.ConclusionOverall, and for every major subgroup of these mothers, more affected and unaffected female than male offspring were born. Differences by gender and lesion level among probands and between probands and unaffected offspring were consistent with an etiology of unknown genetic factors, hormonal and/or immune system factors.
Highlights
Spina bifida is a disorder of the cerebrospinal fluid system associated with failure of neural tube closure in the fetus
Spina bifida is a disorder of the cerebrospinal fluid system resulting from a failure of neural tube closure in the fetus and associated with accompanying deformities leading to hydrocephalus
These hypotheses were: 1) the occurrence of twins is related to the incidence of spina bifida [4,5]; 2) a preceding spontaneous abortion is associated with the presence of spina bifida in the following pregnancy [6]; 3) spina bifida offspring are associated with tissue remaining from a previously aborted conceptus [7,8] ; 4) more males than females affected with spina bifida are lost prenatally resulting in a preponderance of affected females at birth [9,10]
Summary
Spina bifida is a disorder of the cerebrospinal fluid system associated with failure of neural tube closure in the fetus. Reproductive history studies of mothers with spina bifida offspring have often been conducted shortly after the affected child's birth. The first was to describe and analyze variables related to the reproductive history of mothers who had given birth to a child with spina bifida (affected). The second purpose was to use the findings to test four hypotheses regarding the nature of spina bifida These hypotheses were: 1) the occurrence of twins is related to the incidence of spina bifida [4,5]; 2) a preceding spontaneous abortion is associated with the presence of spina bifida in the following pregnancy [6]; 3) spina bifida offspring are associated with tissue remaining from a previously aborted conceptus [7,8] ; 4) more males than females affected with spina bifida are lost prenatally resulting in a preponderance of affected females at birth [9,10]
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