FAMILIES OF TEENAGERS WITH DOWN'S SYNDROME: PARENT, CHILD AND SIBLING ADAPTATION

Transcription

1 Documentation for ESRC Data Archive FAMILIES OF TEENAGERS WITH DOWN'S SYNDROME: PARENT, CHILD AND SIBLING ADAPTATION A STUDY OF FAMILIES IN THE MANCHESTER DOWN'S SYNDROME COHORT DATA COLLECTED IN 1991; REPORT SUBMITTED 1994 PATRICIA SLOPER AND STEPHEN TURNER HESTER ADRIAN RESEARCH CENTRE UNIVERSITY OF MANCHESTER Manchester M13 9PL STUDY FUNDED BY THE ECONOMIC AND SOCIAL RESEARCH COUNCIL AWARD NO. R

2 Description of the Project This study is one of a series involving the Manchester Down's Syndrome Cohort families with a child with Down's syndrome living in the Greater Manchester area. One hundred and six families took part in the present study. The families were first recruited to the Cohort during the period , soon after the birth of the child with Down's syndrome. They have taken part in a series of studies both before and after school entry (Byrne et al, 1988; Cunningham et al, 1986; Sloper et al 1988). The aims of the present study were: 1) to provide comprehensive information on child and family functioning during the children's teenage years; 2) to define the factors related over time to the well-being of children, parents and siblings, and to the risk of poor child development and family distress; 3) to investigate parents' areas of concern, their knowledge of resources available, and their needs for help. The present study must be seen in the context of earlier work with the Cohort. Figure 1.1 shows the chronology of the Cohort studies. In particular, measures used in the Family Adaptation Study in (Sloper et al 1988) formed the basis of the present study, thus enabling the investigation of change over time, and the factors predictive of change. The data set and associated command file lodged with the ESRC data archive covers only the data collected in 1991, and do not therefore reflect the longitudinal aspects of the 2

3 study. The 1991 study collected scores on four outcome measures relating to the child. These were: self-sufficiency, academic attainments, social life and behaviour problems. With the exception of academic attainments scores, which were based on teachers' reports, scores were based on mothers' reports using a self-completion questionnaire. Two parental outcomes, measuring psychological distress and satisfaction with life, were obtained from data from questionnaires separately completed by mothers and fathers. Two outcome measures of sibling adaptation reflected feelings of self-worth, and reported behaviour problems. The former was based on sibling questionnaires, and the latter on mothers' questionnaires. A wide range of predictor variables was collected in order to investigate relationships with these outcomes. The study adopted the model of stress and coping developed by Folkman and Lazarus (1985) to inform the analysis of outcome measures. Data Preparation File "teendata.d91" contains a sequential data file relating to the 106 families. Data are recorded in fixed-field format, with 44 lines per case. Each set is made up of up to seven data sources (see Introduction to Codebook). Where any of these sources are missing (ie where no sibling or father is present, or in cases of nonresponse) a standardised sequence of defined missing values appears in the data set. The first 3 digits in each set identifies the 3

4 subject family. These subject numbers were established at the beginning of the project, and are not a complete sequence owing to deaths, moves from the area, or withdrawal from the Cohort. The annotated questionnaires and the sections A and B of the command file indicate the location of each variable, and values assigned to missing data. File "teencomd.d91" is a SPSS command file containing data definition, missing values, creation of derived variables, and recodes of other variables in order to facilitate bivariate and multivariate analysis. The content and sequence of the command file is described in a "comment" at the beginning of the file, and repeated at the end. Section C of the command file contains the commands creating outcome and predictor variables based on standardised scales, or on scales or indices developed during earlier studies with the Cohort. These measures and their computation are described below. The command file also includes some computations of variables obtained from the 1988 Family Adaptation Study. These computations are retained in the file for reference only, and appear as "comments" introduced by the line "comment 1988 variable re-codes follow:". They will not run on the data files covered in this paper. Other variables computed in section C represent sub-scales or alternative versions of measures which were not used or reported in 4

5 the final report. Where this is the case, the suffix * after the variable name denotes those referred to in the final report, and ** to main outcome measures. (These symbols only appear in comment entries, and are not part of the variable name). Methodology The remainder of this documentation is derived from the relevant chapter of the final report also deposited with the Archive. All the families in the Cohort were contacted by letter to inform them of the study and request their assistance. Following their agreement to participate, detailed self-report questionnaires were sent to mothers and fathers and an appointment was made to interview the mother at home. With the agreement of parents, a questionnaire was sent to the child's school to be completed by the child's teacher. Separate home visits were arranged, with a different interviewer, for study siblings to complete a questionnaire and semi-structured interview. Interviews were tape recorded with the participant's consent. The mean length of mother interviews was 1 hour 38 minutes (range 50 minutes to four hours); the mean length of sibling interviews was 1 hour 20 minutes, (range 45 minutes to 2 hours 20 minutes). Two interviewers were involved in the study. One conducted all interviews with mothers and the other all interviews with siblings. Both interviewers were trained in the use of the interview schedules, using pilot interviews at the start of the study, by the project director, who had been involved in earlier interviews with the Cohort 5

6 families. Response Rates One hundred and twenty four families were asked to participate in the study. Six families refused; five had moved out of the area; one was ineligible as the child was no longer living at home; and another seven later declined to take part or could not be contacted to arrange interviews. Thus, of the 119 eligible families (ie still in the area, with the child living at home), 13 did not take part, whilst 106 provided at least a partial response. This represents a response rate of 89 per cent. The sample included 15 single parent mothers and one single parent father. Of the 105 mothers, 99 per cent (104) were interviewed and 96 per cent (101) returned questionnaires. Of the 91 fathers 78 per cent (70) returned questionnaires. Teacher questionnaires were returned for 99 of the 105 eligible children (94 per cent; one child was educated at home). The criteria for inclusion of siblings in the study were age (between 10 and 18 years old), residence at home and, if more than one sibling in a family fell into these categories, the sibling nearest in age to the child with Down's syndrome. All 63 siblings in the 106 participating families who were identified as eligible for the study completed interviews and questionnaires. The lower response rate from fathers could have resulted in bias in the fathers' data. This question was examined by comparing families 6

7 of fathers who did not respond with those who did respond on demographic and outcome variables. There were no significant differences and therefore no evidence of bias in the fathers' sample. Questionnaires and Interviews: Choice of Methods The choice of methods was based on the previous study of family adaptation in the Cohort (Sloper et al, 1988). The use of the questionnaires devised for the previous study allowed collection of comparable data for longitudinal investigation. Questionnaire measures provide an economical means of data collection but do not allow individuals to express views in their own way and highlight issues which are of particular importance to them. In the more descriptive and exploratory areas of the study, centring around current needs and concerns, more qualitative data were required. The semi-structured interview was chosen as a method for this part of the study. Similarly, for the study of siblings' responses and adaptation, it was deemed important to obtain the siblings' own views, allowing them to provide their own emphasis on important issues in an interview situation, and to obtain quantitative data which would allow investigation of factors related to variance in adaptation. For this latter purpose, established instruments, in questionnaire form, were used. As previous studies with the Cohort had not included siblings, the questionnaires and interviews were piloted with 12 siblings of children with learning disabilities, who were not part of the Cohort. 7

8 The age range of the pilot group was 10 to 18 years. Both mothers and fathers completed questionnaires. Data from the earlier study showed that measures of stress, adaptation and coping could be obtained from both parents through questionnaires and interview data from fathers added little to the questionnaire data in this area. The earlier study and other studies in the area of needs and service contacts (eg Glendinning, 1986) have shown that mothers, in their role as the main carer, have considerably more contacts with services than fathers. Therefore, only mothers were interviewed (apart from one single parent father) as their responses were felt to provide the most complete picture of current needs and help from services. Likewise, sections of the questionnaire which related to the child- health status and history, behaviour problems, social life, self-sufficiency; to the study sibling, and to contacts with services, were only included in the mothers' schedule. Missing Data Missing data in the study were of two types: i) the non-completion of questionnaires detailed above and ii) occasional response omissions on individual questions or scales for otherwise satisfactory interviews and questionnaires. Attempts were made to minimise ii) by interviewers scanning completed questionnaires during the visit to the family and checking on any missing data, eg where two pages had been turned over together. 8

9 Missing data cause two main problems: a) bias in the sample if the data obtained are not representative of the population; and b) the cumulative effect of occasional omissions on calculation of indices and on multivariable analyses. With respect to a), as noted earlier there was no evidence of substantial bias due to non-responding of some fathers. In the case of b), when one or more of the components of a composite measure, or one or more of the factors in a multivariable analysis are missing, then all the data are deemed to be missing for that subject. In indices and analyses involving many measures the overall effect of this could be to severely reduce the sample size. In order to minimise the possible bias and the reduction in sample size from missing data due to response omissions, a number of guidelines were adhered to (Sloper et al., 1988): (i) Questions with more than 10% of data missing due to occasional response omissions were considered to be unreliable and/or biased and would be excluded from the analysis. No questions were excluded on this basis. (ii) Subjects who failed to respond to more than 10% of items required for a composite measure or index would be excluded from the calculation of that measure. This occurred for between one and eight subjects on individual indices. (iii) For the remaining response omissions on items included in indices, missing data were assigned a mean score. Thus, the respondent's mean score was obtained for valid items, and this was 9

10 multiplied by the total number of items in the scale to produce an estimate of the respondent's likely total score. This was deemed to be valid where only the total scores, not individual item scores, were used. Selection of Measures The investigation required the assessment of a wide range of variables. The selection of descriptor and outcome variables was informed by the theoretical model of family adaptation as utilised in the earlier Cohort study. The measures included both established scales with demonstrated reliability and validity and measures specifically devised for the earlier study. The properties of all measures were subject to detailed investigation in that study. The results of this investigation confirmed that the measures provided reliable and valid data (see Sloper et al, 1988: Appendix B for details). Outcome measures: Parent functioning Two outcome measures were used: (i) Parental distress. The Malaise Inventory (Rutter, Tizard and Whitmore, 1970), a 24 item checklist of psychosomatic symptoms was used to measure distress. This measure has been used in a number of studies of families with a disabled child (eg Bradshaw and Lawton, 1978; Quine and Pahl, 1991). (ii) Satisfaction with life. An index of parents' perceived satisfaction with current life circumstances was developed in the 10

11 earlier study. This was completed from a range of measures, chosen to give a broad perspective on satisfaction with life within the family, including general satisfaction, satisfaction with social support, amount of child care, housework, time for self, going out with partner and with family, financial circumstances and parenting, and parents' ratings on a ladder scale of their perceptions of their present position between their own ideas of the worst and ideal life, based on methods devised by Cantril (1965). The computation of the index was based on the weighted scores established in the previous study (Sloper et al, 1991). In the present study, the components of the index were reduced from eight to six, as some items were felt to be repetitious. (See Appendix 2). Outcome measures: Child functioning These comprised: (i) Self-Sufficiency Index (Turner et al, 1991a). A 272 item scale measuring two areas of child functioning: personal selfsufficiency and community self-sufficiency. The scale was developed from the Pathways to Independence Scale (Jeffree and Cheseldine, 1982). Additional items were included to increase sensitivity for the Cohort sample, and scoring was on a three-point scale (can do, can do with help, cannot do), instead of yes/no, in order to overcome problems of completion noted by Jeffree and Cheseldine for the original scale. The scale was completed by mothers. (ii) Academic Attainments (Sloper et al, 1990a). A 58 item scale, completed by teachers, relating to reading and use of written 11

12 information, number skills and writing skills. The checklist was based on those devised by Lorenz (1985) in a study of children with Down's syndrome. Items were scored on a three-point scale (can do, can do with help, cannot do). (iii) Social Life Index (Sloper et al, 1990b). A measure of the frequency of the children's social contacts with other children was obtained through a questionnaire completed by mothers concerning the child's attendance at organised groups and activities and informal play contacts. (iv) Behaviour Problems Index (Byrne et al, 1988). This was a questionnaire completed by mothers and comprised the Behavioural Screening Questionnaire (Richman, Stevenson and Graham, 1982) and a number of questions covering the wider range of problems considered more appropriate to a population of children with Down's syndrome from the questionnaires of Gath (1978) and Wing and Gould (1978). Descriptor variables These measures were selected for the earlier study to reflect a broad spectrum of child and family characteristics which the literature suggests may be associated with child and family functioning. Child Variables These were gender; chronological age; position in family; health problems - the Child Health Index (Turner et al, 1990); mental age and IQ based on assessments in 1986 on the McCarthy Scales of Children's Abilities (McCarthy, 1972); distractibility and excitability based on the ADIECAS scale completed by teachers (Evans 12

13 and Hogg, 1984; Turner et al, 1991b); and the amount of supervision and caretaking needed using the measure of Richman et al (1982). Intensity of early intervention in infancy and type of school attended were also included as descriptor variables. It should be noted that assessments of cognitive development were not carried out in the 1991 study. However, the use of the 1986 measures this context was deemed reliable as a number of earlier investigations of the large quantity of developmental data obtained on the Cohort children has shown very high consistency over time in relative scores (eg Cunningham, 1983; Sloper et al, 1988). Thus, whilst children would have progressed in developmental level between 1986 and 1991, their position relative to each other would have remained largely the same. Demographic variables were social class; unemployment; parental educational level; adequacy of housing; car ownership; marital status; age of parent; number of children in family; and financial problems. These measures were based on parents' answers to questions in the self-report questionnaire. Parental and family resources A number of published scales were used to assess the personal resources of parents and the resources within the family system. These were contained within the self-report questionnaires completed by mothers and fathers. 13

14 Parental personality was assessed using the Eysenck Personality Inventory (Eysenck and Eysenck, 1964); locus of control was measured on the Brief Locus of Control Scale (Lumpkin, 1983); marital relationships were assessed using the Measure of Marital Satisfaction (Kelso et al, 1984); social support was assessed using the Inventory of Parent Experiences (Crnic and Greenberg, 1983) and the characteristics of the family system were measured on the Family Environment Scale (Moos and Moos, 1981) and a subscale of this measure the Family Relations Index (Holahan and Moos, 1983). Measures of service contact and perceived helpfulness of services were also included in questionnaires. Mother-child, mother-sibling and sibling-child relationships were rated on responses to a number of interview questions using ratings based on those of Richman et al, (1982). Life events Occurrence of life events in the last year was measured by a 42-item checklist (Cheang and Cooper, 1984). This also measured the individual's perception of the strain of the events on a 10 point Likert type scale. Coping strategies The Ways of Coping Questionnaire (Revised) (Folkman and Lazarus, 1985), as adapted to measure the ways in which parents coped with problems concerning their children with Down's syndrome (Knussen et al, 1992), was used. In the previous study, factor analysis of responses to the questionnaire produced a five 14

15 factor structure: (1) practical coping; (2) wishful thinking; (3) stoicism; (4) seeking emotional social support; and (5) passive acceptance (Knussen et al, 1992). The same factors were used in the present study. Proportional scores were used in all analyses as they take into account individual differences in the relative use of ways of coping (Vitaliano et al, 1987). In addition to these measures the short version of the Marlowe-Crowne Social Desirability Scale (Strahan and Gerbasi, 1972) was used to measure social desirability. Sibling measures As the sibling measures have not been included in previous studies, further details of the measures and their reliability and validity are given in Appendix 1. Three outcome measures were used to assess sibling adaptation: (i) Behaviour problems were assessed using the Rutter Scale A (Rutter et al, 1970) completed by mothers. This is a 31 item scale with each item scored 0 to 2, suitable for children age 6 to 16 years. A cut off score of 13 has been designated as indicating risk of significant behavioural disturbance. Reliability and validity of the scale has been demonstrated in a number of studies. (ii) Self esteem was measured using six subscales of the Self- Perception Profile for Adolescents (Harter, 1988): scholastic competence, social acceptance, physical appearance, behavioural 15

16 conduct, close friendship and global self-worth. This measure was completed by siblings. (iii) Anxiety was measured using the Revised Children's Manifest Anxiety Scale (Reynolds and Richmond, 1978). The descriptor variables used for the analysis of sibling outcome comprised child and parent characteristics, demographic measures and a number of variables related to the roles and resources of the siblings themselves. The Parental Support subscale of the Social Support Scale for Children (Harter, 1985) was used to assess siblings' perceptions of the support and regard which parents manifest towards them. Six subscales of the Sibling Problems Questionnaire (McHale et al, 1986) were used to assess the perceptions of siblings regarding their brother or sister with Down's syndrome. The subscales were feelings of rejection towards sibling; perceived favouritism towards sibling; positive reaction by peers; positive reaction by parents; ability to cope with the sibling's disability; and feelings of being burdened. Interviews with siblings also provided measures of roles and responsibilities, perceived parental expectations and relationships with parents and the child with Down's syndrome. Mothers' questionnaires included questions on their perceptions of the effect on siblings of the child with Down's syndrome. Sibling status factors were age, gender and position in family relative to the child with Down's syndrome. 16

17 Parent interviews Seven main topics were covered in the interviews with mothers. These were parent-child relationships; current concerns; the effects of the child with Down's syndrome on the family; satisfaction with the child's school; school-leaving and transition to adulthood; helpseeking; and experience of link worker and respite care services. The analysis explored the relationships between descriptor and outcome variables. Different variables were designated as descriptor or outcome, according to the specific analysis. Thus, in keeping with the transactional model of family functioning, the same variable can be an outcome variable in one analysis and a descriptor variable in another analysis. For example, scores on the Behaviour Problems Index were designated as a descriptor variable in relation to parental adaptation and an outcome variable in relation to child functioning. Table 1 summarises the main outcome and descriptor variables defined in the 'teencomd.d91' command file. Table 1 Outcome and descriptor variables in relation to child, parent and sibling functioning KEY M = obtained from mothers' responses F = obtained from fathers' responses T = obtained from teachers' responses S = obtained from siblings' responses A = assessment by researchers 1. Outcome Variables - Child Functioning 17

18 Self-Sufficiency Index (M) Academic Attainments Index (T) Social Life Index (M) Behaviour Problems Index (M) 2. Outcome Variables - Parent Functioning Satisfaction with Life Index (M,F) Malaise Inventory (M,F) 3. Outcome Variables - Sibling Functioning Behaviour Problems (M) Self-Esteem (S) Anxiety (S) 4. Descriptor Variables (a) Child Gender Age Position in family Mental age (A, 1988 study) IQ (A, 1988 study) Excitability (T) Distractibility (T) Supervision needs (M) Caretaking needs (M) Child Health Index (M) Intensity of early intervention Type of school (b) Demographic Social class (M,F) Parental education (M,F) Father's employment/unemployment (F) Mother's employment/unemployment (M) Housing adequacy (M,F) Single parent family (M,F) Age of parent (M,F) Number of children in family (M) Car ownership (M,F) Financial problems (M,F) Table 1 Outcome and descriptor variables in relation to child, parent and sibling functioning (continued) 18

19 (c) Family Eysenck Neuroticism Score (M,F) Eysenck Extraversion Score (M,F) Locus of Control (M,F) Marital relationship (M,F) Amount of social support (M,F) Family Environment Scale/Family Relations Index (M,F) Contacts with services (M) Mother-child relationship (M) Mother-sibling relationship (M,S) Sibling-child relationship (M,S) Numbers of life events (M,F) Life events strain (M,F) Coping strategies (M,F) 1. Practical coping 2. Wishful thinking 3. Stoicism 4. Seeking emotional social support 5. Passive acceptance Marlowe-Crowne Social Desirability Score (M,F) (d) Sibling Age Gender Older/younger than Down's syndrome child Perceived parental support (S) Roles and responsibilities (S) Parental expectations (S) Perceived problems related to Down's syndrome child (S) The Issue of Social Desirability The effects of social desirability on responses on self report measures has long been debated (eg Block, 1965; Nevid, 1983; Strosahl, Linehan and Chiles, 1984), particularly in relation to the significant correlations often found between social desirability scores and other self report measures. The debate centres on whether such correlations are theoretically inconsistent with constructs being measured, and thus bring into question the validity of the measures, or whether the covariation is theoretically predictable, 19

20 and thus may strengthen the case for the construct validity of the measures. If the latter case is argued, the measures must still be discriminable from each other and not be confounded. In order to allow investigation of these issues in the present study the Marlowe-Crowne Social Desirability Scale was included in the questionnaires and the univariate relationships between outcome measures which involved parents' reports, and their Marlowe-Crowne scores (MARCTOTM, MARTOTF) were examined. The results are reported in Chapter 2 of the final report. Results were similar to those found in the previous study, suggesting some evidence of a social desirability 'response set' operating on some of the self report measures, for some parents. However, the magnitude of the correlations does not suggest confounding. The entry of Marlowe- Crowne scores as the first factor in all regression analyses which involved outcome measures based on parent reports was designed to control for a response set effect when determining which descriptor variables were significantly related to outcome. In all cases, Marlowe-Crowne scores were not significantly related to outcome in the final equations. This suggests that the solutions provided a valid measure of relationships which were not contaminated by social desirability response sets. Data Management and Statistical Analysis The data were analysed in five phases: 1. The establishment of a data base: the questionnaires and 20

21 interviews were coded and entered onto computer files. Interview responses were subject to qualitative analysis (Miles and Huberman, 1984). Designation of an initial coding frame for interview responses was carried out after completion of the first twenty interviews. Responses to each question were examined and coding categories devised to reflect commonalities in responses. The process was carried out jointly by two researchers. The use of a coding manual allowed the addition of categories, where appropriate, during coding of subsequent interview material. All interviews were coded by the interviewer concerned and reliability checks on coding were carried out by a second researcher coding a subset of 30 interviews from interview transcripts. Reliabilities ranged from 70 to 90 per cent with a mean reliability of 82.2 per cent. 2. Indices were created for composite measures. 3. Descriptive data were obtained from frequency counts on appropriate variables. 4. Replication of the 1988 multivariable analysis of outcome measures. In order to investigate the similarities and differences between the patterns of variables associated with outcome in the two studies and address the question of whether a different set of descriptor variables would be associated with outcome in the children's teenage years, the procedures used for the analysis of the 1988 data were replicated. 21

22 These procedures comprised three stages: (i) Univariate analysis The relationships between outcome and descriptor variables were examined using one way analysis of variance for categorical descriptor variables and correlations or regression plots for continuous variables. These analyses informed the selection of variables for multivariable analyses and led to the following stages. The linearity of all relationships was examined to determine whether variables could be entered in multiple regression analyses. Categorical variables were recoded to form a dichotomy or dummy variable were created. All variables significantly related to outcome at the univariate level proved suitable for entry into multiple regression analyses. For each outcome a subset of descriptor variables was identified for inclusion in multivariable regression analysis. Descriptors were included in the subsets if they were related to the outcome at the five per cent level of significance. (ii) Bivariate analysis In order to reduce problems arising from multicollinearity of the independent variables, correlations between descriptor variables were computed and where variables were confounded (intercorrelations exceeded 0.59), bivariate analyses were used to determine which of the variables should be entered in the multivariable analysis. (iii) Multivariable analysis The principal method of analysis used was multiple regression. This was preferred over multiple 22

23 analysis of variance because of the continuous or ordinal level of the majority of variables. The major merits of multiple regression are: (i) It provides a precis of the associations between descriptor and outcome variables by eliminating those descriptor variables whose covariance with the outcome measure is shared by other variables with more powerful associations. (ii) The regression equation indicates the maximum level of explained variance that is possible with the available information, and therefore the extent to which unknown or unmeasured factors may be important as indicators of the outcome variable. (iii) The equation identifies conditions and circumstances likely to predict the outcome measured, although causality cannot be presumed from cross-sectional data. (iv) The data may suggest possible causal models with at least some of the variables in the final equation. This can inform models for further longitudinal research. The format of the multivariable analyses is described below. Variables from the subsets of descriptors were entered into regression analyses in five stages, determined by the theoretical model of the stress and coping process. For parental outcome measures, the first analysis comprised the possible stressors of child characteristics and life events. The second analysis included 23

24 family characteristics and resources and the third comprised coping strategies. For child outcome measures, the first analysis comprised child characteristics, the second comprised demographic measures, and the third parental resources and coping strategies. For siblings, a further set of variables comprised siblings' own characteristics. At each stage, the overall F for the set of descriptors was computed. If this proved to be significant, the significance of t for each independent variable was examined and only variables showing significance at the five per cent level were selected for inclusion in the final analysis. This method is recommended by Cohen and Cohen (1983) to control for and balance Type I and Type II errors in regression analyses and reduce the problems of multivariable analyses which include a high number of independent variables. In order to determine the most powerful combination of variables related to the outcome measures, the significant variables from each stage were entered together in the final stepwise regression analyses. Social desirability scores were entered first into all multivariable analyses involving parent report outcome measures to control for social desirability response effects. 5. Path analysis of outcome variables Path analysis was used to investigate the relationships between 1988 predictor and outcome variables, 1991 descriptor variables and 1991 outcome variables. Procedures followed are reported in full in Chapter 2 of the final report, and are not repeated here as the deposited data set does not include longitudinal material. 24

25 REFERENCES Block S (1965) The Challenge of Response Sets: Unconfounding, Meaning, Acquiescence and Social Disability in the MMPI. New York: Appleton-Century-Crofts Bradshaw J and Lawton D (1978) Tracing the causes of stress in families with handicapped children. British Journal of Social Work, 8, Byrne E A, Cunningham C C and Sloper P (1988) Families and Their Children with Down's syndrome: One Feature in Common. London: Routledge Cantril H (1965) The Pattern of Human Concerns. New Brunswick, NJ: Cheang A and Cooper C L (1984) Psychosocial factors in breast cancer. Stress Medicine, 1, Cohen J and Cohen P (1983) Applied Multiple Regression Correlation Analysis for the Behavioural Sciences. Hillsdale, New Jersey: Lawrence Erlbaum Crnic K A and Greenberg M T (1983) Inventory of Parent Experiences: Manual. Seattle, WA: University of Washington Cunningham C C (1983) Early Development and its Facilitation in Infants with Down's Syndrome. University of Manchester: Hester Adrian Research Centre. Report to DHSS Cunningham C C, Sloper P, Rangecroft A and Knussen C (1986) The Effects of Early Intervention on the Occurrence and Nature of Behaviour Problems in Children with Down's Syndrome. University of Manchester: Hester Adrian Research Centre. Report to DHSS 25

26 Evans P L C and Hogg J (1984) A classroom scale for use with mentally retarded children. British Journal of Clinical Psychology, 23, Eysenck H J and Eysenck S B G (1964) Manual of the Eysenck Personality Inventory. London: University of London Press Folkman S and Lazarus R S (1985) If it changes it must be a process: A study of emotion and coping during three stages of a college examination. Journal of Personality and Social Psychology, 48, Gath A (1978) Down's syndrome and the Family: The Early Years. London: Academic Press Glendinning C (1986) A Single Door: Social Work with the Families of Disabled Children. London: Allen and Unwin Harter S (1985) Manual for the Social Support Scale for Children. University of Denver Harter S (1988) Manual for the Self Perception Profile for Adolescents. University of Denver Holahan C J and Moos R H (1983) The quality of social support: measures of family and work relationships. British Journal of Clinical Psychology, 22, Holahan C J and Moos R H (1986) Personality, coping and family resources in stress resistance: A longitudinal analysis. Journal of Personality and Social Psychology, 51, Jeffree D M and Cheseldine S E (1982) Pathways to Independence. Sevenoaks: Hodder and Stoughton Educational Kelso J, Stewart M A, Bullers L and Eginton R (1984) The Measure of 26

27 Marital Satisfaction: A questionnaire to screen parents for marital problems. Child Psychiatry and Human Development, 15, Knussen C, Sloper P, Cunningham C C and Turner S (1992) The use of the Ways of Coping (Revised) questionnaire with parents of children with Down's syndrome. Psychological Medicine, 22, Lorenz S (1985) Long Term Effects of Early Intervention in Infants with Down's Syndrome. University of Manchester: Unpublished PhD thesis Lumpkin J R (1983) Validity of a brief locus of control scale for survey research. Psychological Reports, 57, McCarthy D (1972) Manual for the McCarthy Scales of Children's Abilities. New York: Psychological Corporation McHale S M, Sloan J and Simeonsson R J (1986) Sibling relationships with autistic, mentally retarded and non-handicapped brothers and sisters. Journal of Autism and Developmental Disorders, 16, Miles M B and Huberman A M (1984) Qualitative Data Analysis: A Sourcebook of New Methods. London: Sage Moos R H and Moos B E (1981) Family Environment Scale: Manual. Palo Alto, CA: Consulting Psychologist Press Nevid J S (1983) Hopelessness, social desirability and construct validity. Journal of Consulting and Clinical Psychology, 51, Quine L and Pahl J (1991) Stress and coping in mothers caring for a 27

28 child with severe learning difficulties: A test of Lazarus' transactional model of coping. Journal of Community and Applied Social Psychology, 1, Reynolds C R and Richmond B O (1978) What I think and feel: A revised measure of children's manifest anxiety. Journal of Abnormal Child Psychology, 6, Richman N, Stephenson J and Graham P (1982) Pre-school to School - A Behavioural Study. London: Academic Press Rutter M, Tizard J and Whitmore K (1970) Education, Health and Behaviour. London: Longman Sloper P, Cunningham C C, Knussen C and Turner S (1988) A Study of the Process of Adaptation in a Cohort of Children with Down's Syndrome and Their Families. University of Manchester: Hester Adrian Research Centre. Report to DHSS Sloper P, Cunningham C C, Turner S and Knussen C (1990a) Factors related to the academic attainments of children with Down's syndrome. British Journal of Educational Psychology, 60, Sloper P, Knussen C, Turner S and Cunningham C C (1991) Factors related to stress and satisfaction with life in families of children with Down's syndrome. Journal of Child Psychology and Psychiatry, 32, Sloper P, Turner S, Knussen C and Cunningham C C (1990b) Social life of school children with Down's syndrome. Child: Care, Health and Development, 16,

29 Strahan R and Gerbasi K C (1972) Short homogeneous versions of the Marlowe-Crowne Social Desirability Scale. Journal of Clinical Psychology, 28, Turner S, Sloper P, Cunningham C C and Knussen C (1990) Health problems in children with Down's syndrome. Child: Care, Health and Development, 16, Turner S, Sloper P, Knussen C and Cunningham C C (1991a) Factors relating to self-sufficiency in children with Down's syndrome. Journal of Mental Deficiency Research, 35, Turner S, Sloper P, Knussen C and Cunningham C C (1991b) The validity and applicability of the ADIECAS classroom rating scale in a sample of children with Down's syndrome. Journal of Mental Deficiency Research, 35, Vitaliano P P, Maiuro R D, Russo J and Becker J (1987) relative scores in the assessment of coping strategies. Raw versus Journal of Behavioural Medicine, 10, 1-8 Wing L and Gould J (1978) Systematic recording of behaviours and skills of psychotic children. Journal of Autism and Childhood Schizophrenia, 8,