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1.  Selective surgical management of neural tube malformations. 
Archives of Disease in Childhood  1985;60(5):415-419.
The physical characteristics of 49 children with spina bifida cystica, survivors of a group subjected to selection for early surgery are compared with 39 children alive from an earlier unselected series, born in the 1960s, and reviewed retrospectively. Sixteen children were also studied in whom the initial decision not to operate had been followed by survival and subsequent treatment. Children selected for initial surgery have a significantly lower mortality than those not selected and their mobility at 5 to 7 years of age is better, although only marginally so compared with the unselected group. Selection does not decrease the need for shunt treatment of the associated hydrocephalus. None of those not initially selected for surgery have normal faecal or urinary continence, whereas 35% of the selected in group have normal continence and urinary tracts. Children treated immediately have significantly higher degrees of intelligence than both the unselectively treated and those whose treatment was delayed but a fifth of the latter group were intellectually normal. There were only small differences in intelligence between children given delayed treatment and those unselectively treated, suggesting that postponing surgery does not necessarily have a deleterious effect on ability.
PMCID: PMC1777339  PMID: 4015145
2.  Some sources of stress found in mothers of spina bifida children. 
The scores on a malaise inventory for 51 mothers of spina bifida children born between 1956 and 1962 in South Wales revealed significantly higher scores when the child was incontinent, had a severe locomotor disability, had an IQ less than 80, and was attending a special school rather than a normal school. No difference in scores was found between the sexes or for size of family, and only a slight difference occurred between the malaise scores of mothers of mildly and moderately handicapped children.
PMCID: PMC478883  PMID: 124188
3.  Natural History of Spina Bifida Cystica and Cranium Bifidum Cysticum 
Archives of Disease in Childhood  1971;46(246):127-138.
The 425 cases of spina bifida cystica and cranium bifidum cysticum born in a population of 850,000 between 1956 and 1962 (with an incidence of 4·12 per 1000 births) were followed. Most did not receive the modern treatment for the condition. Follow-up in 1968 showed that 25% were stillborn, 13% died during the first week of perinatal causes, a further 47% died mostly of the complications of the condition, and 15% are still alive. Life table survival figures suggest for the liveborns a 12·8% life expectancy to the 11th birthday. The series included 18 cases of meningocele (4·2% of the total), diagnosed largely by exclusion; all are surviving and are largely free from physical and mental handicap. 26 cases of encephalocele include 8 survivors who are all retarded but not much crippled. Of the 381 myeloceles, 37 survive who though not much affected mentally are severely handicapped physically with limb paralysis and incontinence, the pattern of which was largely determined by the level of the lesion. There is a close relation between degree of hydrocephalus and severity of mental retardation. Boys surviving show better performance than girls. The incontinent, whether treated or untreated, show no more emotional disturbance than the continent.
This series provides a `baseline' allowing modern and surgical treatment to be evaluated. 60% of the total (or 85% of those liveborn) should be regarded as potentially salvageable. Nearly all cases should probably have the `open' lesion closed as a surgical emergency, but thereafter no further procedure should be undertaken on those with severe hydrocephalus, established renal damage, persistent intracranial infection, or severe kyphosis, or if other severe malformations are present.
PMCID: PMC1647472  PMID: 4930541

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