Marigold J Thorburn

Validity of the Ten Questions screen for childhood disability: Results from population-based studies in Bangladesh, Jamaica, and Pakistan

An international study to validate the Ten Questions screen for serious childhood disability was undertaken in communities in Bangladesh, Jamaica, and Pakistan, where community workers screened more than 22,000 children ages 2–9 years. All children who screened positive, as well as random samples of those who screened negative, were referred for clinical evaluations. Applying comparable diagnostic criteria, the sensitivity of the screen for serious cognitive, motor, and seizure disabilities is acceptable (80–100%) in all three populations, whereas the positive predictive values range from 3 to 15%. These results confirm the usefulness of the Ten Questions as a low-cost and rapid screen for these disabilities, although not for vision and hearing disabilities, in populations where few affected children have previously been identified and treated. They also show that the value of the Ten Questions for identifying disability in underserved populations is limited to that of a screen; more thorough evaluations of children screened positive are necessary to distinguish true- from false-positive results and to identify the nature of the disability if present.

Identification of childhood disability in Jamaica: the ten question screen.

This is the first in a series of papers that report the testing of two instruments for the identification and assessment of childhood disability by community workers (CWs) in Third World countries. It is part of the International Epidemiologic Study on Childhood Disability. The Ten Question Screen (TQ) was used as the main instrument to identify disability in a two stage population-based survey of 5478 children aged 2-9 years in Clarendon, Jamaica. In the second stage, TQ positive and 8% of the screen negative controls were professionally assessed by a doctor and a psychologist using standard criteria based on the main classification system of the ICIDH. Sensitivity of the TQ as a whole varied in different strata of the group and amongst different disabilities, from perfect in girls under 6 years, fits and motor disabilities and for serious disability in all group except boys over 5 years with cognitive disability. Specificity was good but the false positive rate was unacceptably high at 74%. It was concluded, firstly, that the validation of a simple questionnaire of perceptions of behaviour against objective measurements of impairments was perhaps not fair to the TQ. In spite of this, the TQ would be a very useful instrument in collecting disability data or for identifying people in need of rehabilitation help, if a way of reducing false positives could be found.

Population-based studies of childhood disability in developing countries: rationale and study design.

Infant mortality level has been used for decades to indicate the health and socioeconomic status of populations. Given the relative availability of necessary data, this indicator has proved most viable. As the more glaring aspects of underdevelopment fade into history in some countries, other health status indices should be considered. Childhood disability is proposed as 1 such indicator. Disability is neither subsumed by nor necessarily correlated with declining infant mortality, and may thus be used to help identify solutions for both short- and longterm problems. To employ this indicator, one needs to know how various types of disabilities are spread through populations as well as the correlated risk factors. Methodology must be developed which is capable of rapidly identifying cases and assessing risk factors. A 2-stage method, comprised of screening and clinical evaluation, is described.

Categories, classes and criteria in childhood disability — experience from a survey in Jamaica

This paper reports a new classification and criteria for disabilities and handicaps used in a survey of childhood disability in Jamaica. Part of the International Classification of Impairments, Disabilities and Handicaps was used, with an alternative classification for disabilities similar to that of impairment, and with a complete set of criteria for levels of severity. For handicaps, the set proposed in the WHO manual, Training Disabled People in the Community, was used. No difficulties were encountered in the use of definitions or severity criteria. The inter-rater reliability for disability as a whole by community workers was 79% (kappa statistic 0.58), and for the physician and psychologist 90 and 100%. The handicap classification was easy to use but there was some confusion with resulting poor inter-rater reliability for some questions. This could be corrected by clarification of the meaning of the questions during training. We recommend use of this classification and criteria as being simple and realistic for surveys of childhood disability conducted by community workers.

A Comparison of Efficacy of the Key Informant and Community Survey Methods in the Identification of Childhood Disability in Jamaica

A comparison of the efficacy of the key informant and the community survey methods for identifying children with disability was carried out in the Jamaican component of an international epidemiological study of childhood disability. Approximately 130 key informants were exposed to a 2-day workshop giving information on signs of disability, aspects of the project, and available services. Questionnaires were given to enable the informants to refer children and they were reminded 6 months later. In the survey method, eight community workers completed a house-to-house survey of all families and administered the 10-question screen with probes on 5475 children, 2 to 9 years old. Seventeen referrals were made by the key informants; of these, two were found to have disabilities. Of the 821 children who tested positive on the 10-question screen in the house-to-house survey, 193 had disabilities. We concluded that the key informant method would not be a satisfactory way of identifying cases of childhood disability.

Possible X-autosomal translocation in a girl with gonadal dysgenesis

By screening patients with primary amenorrhoea with chromosome studies, many anomalies of the sex chromosomes are detected (Jacobs et al., 1961; Clavero, 1964; Philip, Sele, and Trolle, 1965b; Shearman, 1968; Thorburn and Pathak, 1970). These range from classical Turners syndrome with 45,XO, mosaicism, 46,XY, apparently normal 46,XX karyotypes, and structural anomalies including deletions, isochromosomes, and fragments. During such a programme over the past 5 years (Thorburn and Pathak, 1970) we found a patient who appears to have a translocation which could be interpreted as either a t(C;C) or a t(X;C). We have been able to find only two similar cases previously reported (Mann et al., 1965; Neuhauser and Back, 1966), and such an abnormality is not mentioned in comprehensive series or reviews of sex chromosome abnormalities (Court Brown et al., 1964; Ferguson-Smith, 1965; Jacobs, 1969). In this paper we present the detailed clinical and cytogenetic findings in this patient and discussion of the possibility of X autosomal translocation. This patient was included in a series of cases of primary amenorrhoea previously reported (Thorburn and Pathak, 1970).

Chromosome studies in 101 mentally handicapped Jamaican children

Sex chromatin surveys have shown that inmates of institutions for the mentally retarded show an excess of sex chromosome abnormalities over newborns (Court Brown and Smith, 1969). The frequency of chromosomal abnormalities depends on the selection of the patients and the severity of retardation, autosomal abnormalities being commoner in the more severely retarded and sex chromosomal abnormalities in the mildly retarded. In fact the incidence of sex chromosomal abnormalities in the severely retarded approaches that in newborn populations (Court Brown and Smith, 1969). For these reasons, it was felt worth while to examine children at an institution for mentally retarded in Jamaica. Initially it was hoped that there might be a high yield of sex chromosomal abnormalities from such a survey. The incidence of mental retardation in Jamaica is not known. As might be expected in a country where the infant mortality rate is still in the thirties, and where malnutrition and infection are still potent causes of death (Thorburn and Hayes, 1968), facilities for the mentally handicapped are extremely limited. Custodial care is provided by the government for the severely handicapped, and at the mental hospital for the more ambulant but disturbed patients. Recently a small school for children with multiple handicaps due to rubella was opened by the Salvation Army. These institutions house approximately 120 children. A voluntary organization, the Jamaica Association for Mentally Handicapped Children (JAMHC), provides a home for 35 boys and the only school in the island for retarded children. A survey was carried out on the children attending this institution.

Service needs of children with disabilities in Jamaica.

We investigated the service needs of children attending a medical assessment as part of a two stage survey of 2 to 9-year-old children in mid and south Clarendon, Jamaica. Parents were asked about symptoms relating to six different disabilities: visual, hearing, speech, motor, cognitive and fits. Following medical and psychological assessment, a diagnosis of mild, moderate, severe or no disability was made. For children with disabilities, the frequencies of five possible types of intervention recommended by the physician were analysed and related to the prevalence of the six disabilities in the parish. To estimate the needs of the Jamaican child population the figures were extrapolated based on an estimate of 1 million children under the age of 15 years. These needs were then compared with places in existing services. Of the disabled children, 62% needed special education, 29.5% needed community-based services, 21% needed spectacles, 21% needed specialist referral, and 6% required medical treatment. Although the vast majority of these needs are not met, many more could be met in the community if existing health and education personnel are trained in basic techniques of screening and assessment.

The pathology of sickle cell anaemia in Jamaican adults over 30

Abstract The pathological findings at autopsy in 7 patients over the age of 30 with sickle cell anaemia are described and discussed. The 3 youngest died of well recognized complications of sickle cell anaemia. The 4 older patients, all over 50, had been remarkably free of direct complications and died of diseases which could have been unrelated to sickle cell anaemia. Cirrhosis and chronic nephritis were not seen. Pulmonary vascular disease was seen in 2 patients but its significance in relation to sickle cell anaemia has not yet been established in Jamaican patients.

Chromosomal rearrangements in three generations of a Jamaican family

Chromosomal rearrangements are described in a three-generation family whose members were all normal but for the propositus, who had phenotypic abnormalities suggestive of Patau’s syndrome. The rearran