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Attitudes towards prenatal diagnosisand termination in women who have asibling with Down's syndromeL. Bryant a , J. D. Hewison a & J. M. Green ba Academic Unit of Psychiatry and Behavioural Sciences, School ofMedicine, University of Leeds, UKb Mother and Infant Research Unit, Department of HealthSciences, University of York, UKPublished online: 23 Jan 2007.

To cite this article: L. Bryant , J. D. Hewison & J. M. Green (2005): Attitudes towards prenataldiagnosis and termination in women who have a sibling with Down's syndrome, Journal ofReproductive and Infant Psychology, 23:2, 181-198

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Attitudes towards prenatal diagnosis and

termination in women who have a sibling with

Down’s syndrome

L. BRYANT & J. D. HEWISON

Academic Unit of Psychiatry and Behavioural Sciences, School of Medicine, University of Leeds,

UK

J. M. GREEN

Mother and Infant Research Unit, Department of Health Sciences, University of York, UK

Abstract This study surveyed the views of 78 women who had a sibling with Down’s

syndrome towards using prenatal diagnosis and termination. Other data were collected,

including participants’ perceptions of the difficulty of caring for a child with Down’s syndrome,

perceived familial approval of selective termination, and the quality of the sibling relationship.

Fifty-four per cent of respondents said they would use diagnostic tests for Down’s syndrome in a

future pregnancy, 37% would not, and 9% were unsure. In contrast, 33% would consider

terminating a pregnancy for Down’s syndrome, 53% would not consider termination, and 14%

were unsure. Logistic regression showed that the perceived difficulty of caring for a child with

Down’s syndrome and perceived parental approval of selective termination were the strongest

overall predictors of both attitudes. However, women who were younger than their affected

sibling were more likely to want to use prenatal diagnosis, and a generally favourable attitude

towards abortion predicted a favourable view towards terminating a pregnancy for Down’s

syndrome. In general, a positive picture emerged of having a sibling with Down’s syndrome

although around one-third of the women viewed the impact on themselves and their family as

negative and this was reflected in their attitudes towards prenatal testing and termination.

Introduction

When pregnant women make decisions about whether or not to undergo prenatal

diagnostic testing (PD) or termination for Down’s syndrome they may not have the

first-hand knowledge of the condition with which to inform this decision. One group

Address for correspondence: Dr Louise Bryant, Academic Unit of Psychiatry and Behavioural Sciences,

University of Leeds, 15 Hyde Terrace, Leeds LS2 9LT, UK. Tel: 0113 3431882; Fax: 0113 2433719; Email:

l.d.bryant@leeds.ac.uk

Received: 12 November 2003. Accepted: 1 June 2004

JOURNAL OF REPRODUCTIVE AND INFANT PSYCHOLOGY,VOL. 23, NO. 2, MAY 2005, pp. 181–198

ISSN 0264-6838/print/ISSN 1469-672X/online/05/020181-18# 2005 Society for Reproductive and Infant PsychologyDOI: 10.1080/02646830500129214

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who generally do have this knowledge are women who have a sibling with the condition.

Although the majority of women with a sibling with Down’s syndrome are likely to go

on to have children of their own, very little has been written about their views towards

using prenatal testing and termination. This is surprising considering the unique

position they hold. Many will have spent an extended period of time living with their

brother or sister with Down’s syndrome. They will know a great deal about the impact

of this on siblings and something about how parents and the extended family are

affected. They are likely to have experienced the attitudes of others to their sibling’s

condition, and may well be aware of some of the healthcare, education, and

employment issues associated with the person with learning difficulty in our society.

In addition, many will have thought about, and even dealt with, the situation where

their parents are no longer able to support their sibling as a primary carer (Fairbrother,

1988; Itzkowitz, 1995).

There is little literature on the attitudes that mothers of existing children with

Down’s syndrome hold towards personal use of prenatal testing and termination in

subsequent pregnancies (Boon, 1986; Bridle, 2000; Ekwo et al., 1985, 1987; Elkins

et al., 1986; Felker, 1994; Swerts, 1987). These studies report a wide range of views:

some mothers seeing testing as intrinsically offensive and an insult to themselves and

their child, while others welcome the tests as a means of ensuring they do not have

another child with Down’s syndrome. Most appear to be accepting of the existence of

testing even if they would not wish to terminate for Down’s syndrome themselves.

Sample sizes have generally been small however, and sampling methods diverse,

making it difficult to draw a consensus about the proportion of mothers who would and

wouldn’t test or terminate for Down’s syndrome in a subsequent pregnancy.

Studies that have considered the attitudes of the general public towards using

prenatal testing and termination for conditions including Down’s syndrome also report

a wide range of views (Drake et al., 1996; Evers-Kiebooms et al., 1993; Marteau et al.,

1995; Norup, 1997; Singer, 1993; Singer et al., 1999). However, from these studies it

can be concluded that most people hold more favourable attitudes towards the

availability of these technologies, but less favourable attitudes towards using them

themselves. Around two-thirds of the people studied indicated they would use prenatal

diagnosis for Down’s syndrome (Evers-Kiebooms et al., 1993; Singer, 1993; Singer

et al., 1999), whereas intentions of using termination for the condition were generally

closer to 40%. However, one study reported favourable attitudes towards having a

termination for Down’s syndrome in up to 60% of a ‘lay sample’ in the UK (Drake

et al., 1996).

There is little published research evidence on the views and prenatal testing

behaviour of siblings of people with Down’s syndrome or in fact any disability. In a

review chapter examining how parents of children with a disabling condition view

selective termination it was stated that ‘adult and teenage siblings of affected children

are usually quite accepting of prenatal diagnosis and often plan to have it themselves’

(Wertz, 1992, p. 181). However, this statement was supported by only one reference to

an American study on the reproductive choices of a group of haemophilic men and

their families, in which over a 6 year period none of the 10 carrier-sisters identified

‘carried an untested pregnancy to term’ (Miller et al., 1987). Some anecdotal evidence

does suggest that women who have a sibling with a learning difficulty hold a wide

variety of views on this subject, from a determination to terminate an affected

pregnancy to the complete opposite (Scotson, 1988). In relation to Down’s syndrome

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specifically, Carr (1995), reports on the responses of 27 non-affected siblings who were

approached about this issue as part of a longitudinal study of families with a child with

Down’s syndrome. At this point the sibling with Down’s syndrome was 21 years old.

Nearly two-thirds were reportedly not worried about having an affected child

themselves, four had been ‘sufficiently concerned’ to request, or intend to request,

prenatal diagnosis, and in the remainder of the families the issue had not been

discussed (Carr, 1995, pp. 126–127). No information about possible factors associated

with these actions or intentions was reported.

It is argued that women with a sibling with Down’s syndrome should be in a good

position to make testing choices that are consistent with their own values and attitudes

as well as being informed by knowledge, and as such, their views are worthy of further

investigation (Bekker et al., 1999; Marteau & Dormandy, 2001). Attitudes based on

direct experience are said to be stronger, more clearly and confidently held, more stable

and less easily changed and therefore better predictors of behaviour than those based on

indirect experience (Ajzen & Fishbein, 1980; Fazio et al., 1982; Fazio & Zanna, 1981;

Regan & Fazio, 1977). It may be expected that the more favourable the person’s

experience in relation to her sibling the less motivated she will be to avoid having a child

with Down’s syndrome herself, and that conversely, those women with an unfavourable

experience may be more motivated to avoid such an event.

The present study

The first aim of the study was to describe the attitudes of women with a sibling with

Down’s syndrome towards PD and termination of a pregnancy for Down’s syndrome.

Based on findings from the general population and studies of mothers of affected

children it was anticipated that the majority of siblings would hold favourable attitudes

towards the general availability of these procedures but that fewer would consider using

them personally. The second aim was to explore possible relationships between these

target attitudes and other variables as described below.

N The perceived burden of caring for a child with a disability has been one of the most

consistent predictors of attitudes towards the use of prenatal testing and termination

for disabling conditions (Davies & Doran, 1982; Denayer et al., 1992; Evers-

Kiebooms et al., 1993; Lawson, 2001; Marteau et al., 1992; Priest et al., 1998). It

was hypothesized that perceived burden would also be an important predictor of the

sibling’s views of PD and termination for Down’s syndrome.

N The perception of the severity of a prenatally diagnosed condition has also been

found to be a reliable predictor of the decision to terminate (Abramsky et al., 2001;

Drugan et al., 1990; Evans et al., 1996; Holmes-Siedle et al., 1987; Mansfield et al.,

1999; Verp et al., 1988). It was therefore considered likely that the participant’s

attitudes towards PD and termination would be related to the severity of the

problems experienced by their own sibling in terms of the level of learning difficulty

and health problems experienced.

N Although research is currently lacking in this area, it was hypothesized that the

nature of the sibling relationship would impact on target attitudes, for example, the

more positive the relationship between the siblings was felt to be, the less acceptable

the unaffected sibling would find PD and termination for Down’s syndrome.

N Finally, research with families of children with cystic fibrosis (Wertz et al., 1992;

Wertz et al., 1991) has shown that attitudes towards selective termination are

PRENATAL DIAGNOSIS AND TERMINATION 183

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associated with the respondent’s perceptions of the attitudes of their spouse, parents,

and siblings. It was therefore hypothesized that in this study, the respondent’s beliefs

about their family’s attitude towards termination of pregnancy for Down’s syndrome

would be a predictor of their own attitude.

Methods

Recruitment

The target population for this study were women resident in the UK aged 16 years or

over who had a sibling with Down’s syndrome. There is no central register of families of

people with Down’s syndrome or an obvious access route to this population. A

sampling strategy was therefore developed that accessed known groups of target

families first, and then used various supplementary methods to extend recruitment to a

wider range of women. Families of a child with Down’s syndrome who were members

of an existing research cohort were identified in Surrey (Carr, 1995) and in Manchester

(Cunningham, 1996). Study materials were sent to the lead researcher of each cohort

study who then passed them on to those parents who also had a non-affected daughter

aged 16 or over. Parents were requested to pass the study materials to their daughter.

Secondly, study details were sent to newsletters of organisations that would reach target

families. Thirdly, families of people with Down’s syndrome were contacted via the

manager of a residential care scheme for people with learning disabilities and invited to

participate. Finally, through women already recruited to the project, snowballing

provided potential participants who either contacted the researcher themselves or were

contacted by letter. The aim of this more purposive approach was to increase not only

the size but the diversity of the sample. In particular, it was aimed to recruit women

across as wide an age range as possible, and to access the views of women whose

siblings lived in residential care as well those who lived with their family. Recruitment

was carried out between September 1997 and January 1998 during which 125 potential

participants from across the UK were identified.

Design

The study employed a cross-sectional, self-report survey design, with questionnaires

being delivered and returned by post.

Measures

Background data on age, education, religion, and religiosity were collected along with

data on relevant aspects of the participant’s pregnancy history including experience of

diagnostic testing and termination. These socio-demographic variables have been

shown to significantly relate to attitudes towards prenatal testing and termination in

previous studies of the general population and women during pregnancy (Bell &

Stoneman, 2000; Drake et al., 1996; Furr and Seger, 1998; Green et al., 1993; Norup,

1997; Singer, 1993; Singer et al., 1999). Data relating directly to the sibling with

Down’s syndrome were collected including age, sex, birth order, perceptions of the

degree of learning difficulty experienced, and the existence of any physical and mental

health problems. In order to measure a general attitude towards abortion, participants

were asked whether termination would be acceptable for others and themselves,

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following rape, where the mother’s health was in danger, and for social reasons.

Participants could respond ‘Yes’, ‘No’, or ‘Not sure’ to these scenarios (‘Yes’53, ‘Not

sure’52, ‘No’51). A mean score was derived and used to indicate an attitude towards

abortion generally, with a higher score indicating a more favourable attitude.

Attitudes towards the availability and personal use of PD and termination for Down’s

syndrome

Participants were asked whether they thought PD for Down’s syndrome should be

freely available to all women and then what they themselves might do in a

(hypothetical) future pregnancy. These responses were measured on 7-point Likert

type scale (15strongly disagree/I would definitely not have prenatal diagnosis and

75strongly agree/I would definitely have prenatal diagnosis). An open ended item

invited respondents to explain their response if they wished. These were followed with

questions that asked whether the availability of termination for Down’s syndrome was

acceptable and secondly whether participants would consider it themselves. Three

response options were provided in each case, ‘Yes’, ‘No’, and ‘Not sure’.

Perception of burden

Perception of the burden of caring for a child with Down’s syndrome was measured

using the item ‘How do you think you could cope with a child with Down’s syndrome?’

Responses were captured on a 7-point Likert type scale (15‘I could handle this without

any problem’ and 75‘I couldn’t handle this at all’).

Women were also asked about perception of burden and views on PD and

termination for four other conditions; physical handicap, a condition where the child

would die within 2–6 months of birth, severe learning difficulties, and a long term

incapacitating illness. This paper deals only with the data relating to attitudes towards

Down’s syndrome.

The Quality of Relationships Inventory (QRI) (Pierce, 1994; Pierce et al., 1991,

1992, 1997) was used to measure the positive and negative aspects of the sibling

relationship. The QRI measures three relationship dimensions of depth, conflict, and

social support with independent sub-scales for each dimension. Only the depth and

conflict sub-scales were used in this study, as the items relating to social support were

considered inappropriate. The depth sub-scale assesses ‘the extent to which the

relationship is believed to exert a significant, positive impact on the person’s life’. The

conflict sub-scale assesses the extent to which the individual experiences angry or

ambivalent feelings towards the other person. Both sub-scales have been shown to have

high internal consistency (Cronbach’s alpha of 0.8 and 0.9), to be stable over a period

of 1 year, show moderate agreement between participants’ perceptions of the same

relationship, and to be sensitive to the range of conflict and depth experienced in family

relationships (Pierce et al., 1997). The items from these two sub-scales were integrated

to form 17 items with responses rated on a 4-point scale. For analysis purposes the

scores for the depth items were reversed, thus the lower the score the greater the level of

relationship depth this indicated.

The views of significant others about termination for Down’s syndrome were

collected using measures based on those employed in the study by Wertz et al. (1991),

participants were asked whether they thought their family members (parents, partner,

siblings (including sibling with Down’s syndrome)) would approve or disapprove of

PRENATAL DIAGNOSIS AND TERMINATION 185

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them having a termination for Down’s syndrome. Responses were measured on a 5-

point scale from 15‘Strongly Disapprove’ to 55‘Strongly Approve’.

Prior to distribution the questionnaire was piloted on a small sample of women who

met the study inclusion criteria and a number of changes were made to the wording of

some items. The questionnaire was prefaced with brief descriptions of prenatal testing

procedures to ensure that participants were aware of the tests before completing the

questionnaire.

Procedure

All potential participants were sent an information sheet, a consent form, ques-

tionnaire, and a prepaid envelope for return to the researcher. The research was carried

out within the guidelines of the BPS Ethical Principles for Conducting Research with

Human Participants (British Psychological Society, 1995). Ethical approval was sought

and obtained from the Psychology Ethics Committee at the University of Leeds before

data collection began.

Analysis

All data were analysed using SPSS version 10. The non-normal distribution of many of

the variables led to non-parametric statistics being used in most tests. Categorical data

were analysed using the x2 test.

Results

Response rate

Table 1 sets out the various methods of recruitment and their associated response rates.

Overall 82 questionnaires were returned, of which 78 were useable (63%). The response

rate from the existing cohorts were the lowest, as might be expected as families received the

study materials unsolicited. However, the very low response rate from the Surrey cohort

suggests that the materials did not always reach their target, either because parents did not

pass them on or because contact details held by the lead researcher were out of date. In

addition, the children with Down’s syndrome in the Surrey cohort had all been born in

1963/1964 (compared with 1973 for the Manchester cohort) thus some parents would be

elderly and many female siblings no longer of reproductive age.

Table 1. Participant recruitment methods and response rates.

Recruitment source

Number of

questionnaires

posted

Number of

questionnaires

returned

Percentage

response

rate

Manchester cohort 38 25 66

Surrey cohort 31 10 32

Snowballing from existing contacts 25 19 76

Advertising in newsletters of Down’s syndrome

associations, Mencap and others.

27 21 78

Recruitment via residential housing scheme 4 3 75

Totals 125 78 63

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Sample characteristics

On average, participants had lived with their sibling with Down’s syndrome for 14 years

(range 0 to 25 years), and 21 (27%) were currently living together in the parental home.

Two women reported that they had never lived with their sibling because he or she had

been placed ‘in care’ soon after birth. Two participants had a child with Down’s

syndrome as well as a sibling. Participant socio-demographic information is provided in

Table 2. Although there were clusters of participants from Surrey, Manchester, and

West Yorkshire (where the study was carried out) participants came from a wide

geographical area including Scotland and Wales. It is not known to what degree this

sample were representative of the population of women with a sibling with Down’s

syndrome and ethnicity was not recorded.

Characteristics of the siblings with Down’s syndrome

The socio-demographic information on the siblings with Down’s syndrome provided in

Table 3 suggests that this group represented a fairly typical range of characteristics and

health issues. Health problems were reported in 40% of siblings, mainly congenital

heart defect and hearing difficulties. Two siblings were no longer living having died in

early adulthood. Current or previous mental health problems were reported for 27% of

Table 2. Socio-demographic characteristics of the women in the study.

Age Mean (SD): 28.1 (8.6)

Range: 16 to 47 years

Marital status Married or living as married: 41 (53%)

Parity With children: 31 (40%)

Age at leaving formal educationa Sixteen or younger: 19 (25%)

Seventeen/eighteen: 20 (26%)

After eighteen: 38 (49%)

Religious affiliation Protestant: 41 (53%)

Roman Catholic: 14 (18%)

No religious affiliation: 23 (29%)

Influence of religion over important life decisions No influence: 39 (50%)

A little influence: 19 (24%)

Quite a lot of influence: 20 (26%)

an577 (missing data).

Table 3. Characteristics of the sibling with Down’s syndrome.

Ageb Mean (SD): 24.1 years, Range: 7 to 41 years

Birth order of sibling with Down’s syndromea Older than participant: 25 (33%)

Younger than participant: 51 (66%)

Twin: 1 (,1%)

Sex Male: 58%

Perceived level of learning difficulty Mean (SD): 3.73 (0.99), Range: 1 (very low) to 7

(very severe)

Current living arrangementsb Parental home: 56 (74%)

Residential care: 19 (25%)

Living independently: 1 (1%)

an577 (missing data), bn576 (two siblings no longer living)

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siblings, most noticeably depression. Seven siblings were said to have behavioural

difficulties. A sub-group of 11 siblings (14%) who had a higher than average mean age

of 31.7 years were said to have both physical and mental health problems. In summary,

the affected siblings varied greatly in the nature and perceived severity of their

difficulties, a variation known to be in itself a feature of Down’s syndrome.

Attitudes towards the availability and personal use of PD and termination of pregnancy for

Down’s syndrome

The responses to the questions measuring attitude towards PD were grouped into three

categories; responses of 5 or above on the scale were considered to be favourable attitudes,

responses of 3 and below were considered to be unfavourable attitudes, and a response of 4

was considered to indicate attitude uncertainty, ambivalence, or neutrality. Figure 1 gives

the relative frequencies with which the different attitudes were held. In line with other

studied populations the sample were more likely to support the availability of diagnosis and

termination for Down’s syndrome than they were to consider using them themselves.

Of the 42 participants who indicated that they would probably or definitely undergo

PD in a future pregnancy, 56% said that this was because they would want to terminate

a pregnancy if the baby had Down’s syndrome. They explained that this was due to the

negative impact they felt their sibling’s disability had had on themselves and their

family, for example, one woman wrote:

Figure 1. Attitudes towards the availability and personal use of prenatal diagnosis and termination of pregnancy for

Down’s syndrome.

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I have understood from a personal level how a child with this handicap not only

affects the lives of the parents but the whole family, and adds additional burdens

and stress on others in the relationship.

In contrast, 27% of those who indicated that they would use PD (n529) said they

would not consider termination but wanted to be prepared for the birth of a child with

Down’s syndrome. The remaining 17% of this group said they wanted PD so that in

the event of a positive result they could make a termination decision based on their

current circumstances and partner’s wishes.

Of those women who would definitely not or probably not, undergo PD in a future

pregnancy, virtually all stated that this was because they would not consider

termination of a child with Down’s syndrome. This group talked about the favourable

impact of their sibling on themselves and their family. For example, a woman who also

had a child with Down’s syndrome wrote:

My sister has been the greatest positive influence of my life—you don’t vote

against that!

This group of women also cited miscarriage risks and the fact that no test is 100%

accurate as reasons for not wanting to use PD.

The remaining 9% who had indicated they were unsure whether they would want to

use PD in a future pregnancy (n57) reported experiencing ambivalence about the pros

and cons of the tests, for example whether knowledge gained was worth the associated

risk of miscarriage. A number reported their difficulty in responding to hypothetical

scenarios; an acknowledged problem with this type of attitude research (Green, 1995).

Of the women who had been pregnant, 65% (n520) gave details of their experiences

of being offered PD; three had accepted testing with results proving negative and 17

women had refused tests. Of the two women who also had a child with Down’s

syndrome, one had refused diagnostic tests and the other had not been offered a test;

both held an unfavourable attitude towards PD in future pregnancies.

Relationships between target attitudes and other measured variables

Table 4 shows that religious influence on important life decisions was significantly

associated with both attitudes towards PD and termination. Religious affiliation was

associated only with PD: the majority of women who were Catholic indicated that they

would not use PD in a future pregnancy. The participants’ age was associated with

Table 4. Results: relationships between participant background variables and attitude towards PD and termination

for Down’s syndrome.

Variable Attitude to PD Attitude to termination

Participant’s age Kruskal-Wallis x2(2)50.09 Kruskal-Wallis x2(2)59.18*

Education levelc x2(2)52.18 x2(2)50.83

Parityc x2(1)51.30 x2(1)52.38

Religionc x2(2)56.93* x254.10

Religious influencec x2(2)513.84** x2(2)55.97*

*p,0.05, **p,0.01, c those in ‘not sure’ categories excluded due to small numbers.

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attitude towards termination for Down’s syndrome but not towards using PD. Women

who would consider having a termination were, on average, 6.8 years older than those

who would not consider it. The lack of a significant relationship between age and

attitude towards using PD probably reflects the number of younger women who

indicated they would want testing for knowledge purposes only. No relationship was

identified between the level of education (as reported by the participant) and either of

the target attitudes. Whether the participant already had children or not, did not relate

to attitudes towards testing and termination.

As identified in previous studies, a significant relationship was found between the

scores for general attitude to abortion and attitude towards termination for Down’s

syndrome (Kruskal-Wallis x2(2)533.49, p,0.005). Those women who found abortion

generally more acceptable were also more likely to say they would consider termination

for Down’s syndrome.

Sixty percent of participants (n547) scored three or below on the perceived burden

of care scale, indicating that they believed they could cope with a child with Down’s

syndrome. Of these, 17 women felt they could cope with an affected child without any

problems, including the two women who actually had a child with the condition. Of the

26% (n520) of participants who scored over 4 in this scenario indicating that coping

with a child with Down’s syndrome would be difficult for them, eight women felt they

would not be able to cope at all. The perceived burden of care associated with having a

child with Down’s syndrome was significantly related to attitudes towards using PD

(Kruskal Wallis x2(2)513.83, p,0.01) and having a termination for the condition

(Kruskal Wallis x2(2)520.81, p,0.01). Burden scores were not related to perceived

level of learning difficulty of the sibling or to their physical health problems; however,

higher levels of perceived burden were associated with the existence of mental health

problems (Mann Whitney U5399.5, p,0.05) and with the sibling’s (increasing) age

(Rho +0.23, p,0.05). The current living arrangement of the affected sibling was also

associated with perceived burden. Participants whose sibling lived in the parental home

perceived care of a child with Down’s syndrome as significantly less burdensome than

those whose sibling lived in residential care (Mann Whitney U5296, p,0.01).

Sibling characteristics in relation to target attitudes

Table 5 shows the results of analyses of attitudes towards PD and termination for

Down’s syndrome by the affected sibling characteristics. A significantly more

Table 5. Results: relationships between characteristics of the sibling with Down’s syndrome and attitudes towards

PD and termination.

Dependent variable Attitude to prenatal diagnosis Attitude to termination

Age of sibling Kruskal-Wallis x2(2)54.63 Kruskal-Wallis x2(2)514.17**

Sex of siblingc x2(1)50.73 x2(1)51.94

Birth orderc x2(1)58.29** x2(1)50.25

Perceived degree of learning

difficulty

Kruskal-Wallis x2(2)50.36 Kruskal-Wallis x2(2)50.47

Physical health problemsc x2(1)50.97 x2(1)50.42

Mental health problemsc x2(1)51.86 x2(1)510.18**

*p,0.05, ** p,0.01, c those in ‘not sure’ categories excluded due to small numbers.

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favourable attitude towards using PD was expressed by those whose affected sibling

was older than them, but birth order was not found to relate to attitude towards

termination.

Quality of the sibling relationship

The mean scores were 2.04 for depth (SD 0.57, range 1.0 to 3.25) and 1.67 for conflict

(SD 0.48, range 1.0 to 3.4). Conflict scores did not relate significantly to either of the

attitude variable scores and the relationship between attitude towards using PD and

sibling relationship depth was not significant. However, the relationship between

attitude towards undergoing a termination for Down’s syndrome and sibling relation-

ship depth was significant (F (2,74)58.28, p,0.01). A lower level of relationship depth

was associated with a favourable attitude towards undergoing a termination for Down’s

syndrome.

Perceived approval of family members towards termination for Down’s syndrome

Around one-half of participants perceived that their family would disapprove of them

terminating a pregnancy for Down’s syndrome (48% of mothers, 46% of fathers, and

44% of non-affected siblings). While most participants were not sure how their sibling

with Down’s syndrome would feel about them terminating an affected pregnancy, 35%

of those who answered this question (n563) felt they would disapprove or said they

would be distressed. Two participants felt their sibling would approve. One woman

wrote:

I just know that I could never tell her if I terminated - it would make her question

the value of her own life.

Overall, 26% of participants felt their partners would disapprove of them having a

termination for Down’s syndrome. Ten percent of participants reported definite

differences between their own and their partner’s attitude towards termination in that

they would not consider termination, but thought that their partner would approve of

this action. There was least discrepancy between participant’s views and the perceived

view of their mother towards termination than between the views of any other family

member.

With the exception of the perceived view of the sibling with Down’s syndrome, the

perceived views of family members towards termination for the condition were strongly

inter-correlated (Rho . +0.6, p,0.001 in all cases). Chi-square analyses were

conducted to investigate the relationship between participants’ attitudes towards

termination for Down’s syndrome and their families’ perceived approval of this action.

The responses ‘strongly’ and ‘probably approve’ were collapsed into an ‘approve’

category, and responses ‘strongly’ and ‘probably disapprove’ were collapsed into a

‘disapprove’ category. Cases where the family member’s view had been given as ‘don’t

know’ were excluded. Due to the quantity of missing data, an analysis of the perceived

approval of the sibling with Down’s syndrome was not conducted. In all tests, the

perceived family approval of termination for Down’s syndrome was significantly related

to the participant’s attitude towards using this procedure. Chi-square statistics for these

relationships were: partner, x2 (2, n541)530.1, mother, x2 (2, n564)533.9, father, x2

(2, n559)527.6, and other siblings, x2 (2, n549)525.2; all relationships significant at

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p,0.001. Values given are likelihood ratio chi-square statistics as these tests are less

sensitive to small sample sizes.

Logistic regression analyses were carried out to establish the relative predictive

importance of those variables found previously in the bi-variate analyses to relate

significantly with the two target attitude variables. The variable ‘attitude towards PD’

was dichotomised by categorising responses of 4 or less on the scale as an ‘unfavourable

attitude’ (46%) and responses of 5 and above as a ‘favourable attitude’ (54%). The

attitude to termination variable was dichotomised by collapsing the ‘not sure’ and ‘yes’

categories, on the basis that participants who had given a ‘not sure’ response had not

completely ruled out termination. After this split, 53% of participants remained in the

‘no to termination’ category, and 47% in a ‘would consider’ category. As perceived

family views towards termination were significantly correlated, a single summed

variable of the mother’s and father’s perceived approval was calculated and named

‘family approval’, with partner and sibling views being excluded due to the level of

missing data.

Forward stepwise regression equations were used, with the psychological variables

entered in the first step and the demographic variables added in the second step

(participant’s age, sibling’s age and religious influence, and birth order in the PD

analysis). The results of these analyses are given in Table 6. In the model to predict

attitudes towards personal use of PD, the final model (family approval of termination,

perceived burden, and birth order variables) accurately predicted 79% of those who did

not intend to use testing and 77% of those who did (78% overall). The Nagelkerke R2

value indicated that 60% of the variability in these attitudes was explained by the

variables in the model. The model to predict attitudes towards personal use of

termination for Down’s syndrome (family approval of termination, perceived burden,

and general attitude to abortion variables) accurately predicted 90% of those who

would not consider termination, and 81% of those who would (86% overall). The

Nagelkerke R2 value indicated that 74% of the variability in these attitudes was

explained by the variables included in the model.

In summary, results supported the hypotheses that perceived family views towards

termination for Down’s syndrome and perception of burden would predict

participants’ attitudes to PD and termination for Down’s syndrome. However, the

Table 6. Forward stepwise logistic regression analyses to predict attitudes towards personal use of PD and

termination for Down’s syndrome: variables retained by the final model.

Variables in model

Attitude towards PD Attitude towards termination

B SE Wald B SE Wald

General attitude to

termination

23.20 1.06 9.19**

Perceived burden

of care

0.64 0.22 8.42** 1.10 0.32 12.16**

Family approval of

termination

0.51 0.15 12.21** 0.72 0.23 9.94**

Birth order 22.10 0.81 6.68*

* p,0.05 ** p,0.01

(Carpenter & Carpenter, 1997; Carr, 1995; Wertz, 1992)

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perceived level of the sibling’s disability did not appear to relate to the target attitudes

with the exception of the existence of mental ill-health, and this did not retain

significant predictive power once input to logistic regression. The quality of the sibling

relationship as measured by the QRI also contributed in a less significant way than

anticipated. This suggests either that the measure used was not a sufficiently sensitive

one for this sample or that quality of the sibling relationships was not an important

factor in relation to the target attitudes. No norms for sibling relationships obtained

using the QRI are available, so the relationship depth and conflict scores obtained in

this study could not be compared against ‘typical’ sibling pairings. In general

demographic variables did not add any significant predictive power to the regression

models over and above that provided by the psychological variables, with the exception

of the effect of birth order on the attitude towards PD. The possible reasons for these

and the other relationships identified will now be discussed.

Discussion

This study has demonstrated that like other sections of the population, women with a

sibling with Down’s syndrome can hold a variety of views about the availability and

personal usage of prenatal diagnosis and termination for the condition; there is no

consensus resulting from a ‘shared’ experience. The polarisation seen in the

distribution of attitude scores with relatively few participants expressing uncertain

views in regards to this topic, supports the theory that attitudes based on direct

experience are strongly held. The increasing ages of both participant and sibling were

related to favourable attitudes towards termination for Down’s syndrome and, in the

case of the affected sibling’s age, to an increased co-morbidity of physical and mental

health problems in the sibling, and a higher perception of the burden associated with

the condition. Cognitive and physical abilities are seen to decline around middle age in

some individuals with Down’s syndrome, although the relative contributions of

genetics and environment to this decline continues to be debated (Wishart, 1998). It

might be that some participants with older siblings have experienced these problems

and so perceive care to be more burdensome than those whose siblings were still young.

One participant (42 years) wrote of how she had changed her opinions about prenatal

testing as her sister with Down’s syndrome had grown older:

She seems to find life harder and is much less happy and settled than she was. It

seems that life’s experiences have confused rather than enriched her sense of

wellbeing and purpose. This is hard to watch and support and I’ve seen parenting

get harder for my mother rather then the reverse. This has made me question my

original thinking.

However, neither the perceived level of learning difficulty of the sibling, nor existence of

physical health problems significantly related to either of the target attitudes or to

perceived burden of care suggesting that factors other than actual degree of disability

may also be involved.

The ages of the participant and sibling with Down’s syndrome were significantly and

positively correlated and so it is difficult to determine whether these relationships had

meaning independent of each other. For example, a number of other studies have

found that women become more favourable in their attitudes towards termination for

abnormality as they get older (Green et al., 1993; Kramer et al., 1998; Singer et al.,

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1999). Having a child with a disability may be more threatening to an established

lifestyle and women might also become less idealistic about raising a child with a

disability as they get older. In addition, a cohort effect may have been at work in that

older people with Down’s syndrome may have had fewer educational and social

opportunities than their younger counterparts, thus increasing their physical and

emotional dependence on their family. A combination of these reasons is likely to

account for the relationship between participant and sibling age and attitudes towards

termination for Down’s syndrome.

The reasons for the predictive relationship between birth order and attitude towards

using PD but not termination for Down’s syndrome are unknown. However, of the 11

women who said they would want to use PD for preparation purposes only, seven had

affected siblings who were older than them. Some of these participants had commented

on the shock that their parents had experienced on the birth of their affected child and

said they wanted to avoid this. In seven out of eight of these cases the older sibling was

the first-born child of the family. In addition, the mothers of the ‘older sibling’ group

had been significantly younger at the time of the birth of the affected child than the

mothers of the ‘younger sibling’ group (mean ages 28.7 years versus 36.5 years). These

factors might increase awareness that a woman can have a child with Down’s syndrome

at any age with the resulting preference for more information and preparation time than

their own mother had.

Although attitudes to testing and abortion for Down syndrome were related to degree

of religious influence, and attitude towards testing was related to religious affiliation,

neither variable contributed significant predictive power in the logistic regression

analyses. Over one-third of women in this study with favourable attitudes to PD and

termination reported some degree of religious influence in their lives. Over one-third of

women who indicated that they would not consider termination said that religion did

not influence their decisions at all. None of the participants cited religion, or ‘pro life’

morality as the reason for not wanting to use PD or have a termination. Instead,

participants wrote of the value of people with Down’s syndrome as family members,

and of knowing that they could cope practically with an affected child. The influence of

religious belief in the testing and termination literature tends to be viewed in a rather

simplistic fashion, as if the existence of such influence was self-explanatory. This might

serve to support a belief that families with a disabled child are somehow more self-

sacrificing than others ignoring the wider psychological aspects of religious belief. It is

possible that having personal experience with a family member with a disability

overrides religious belief in terms of influencing attitudes towards PD and termination.

This might also be true in relation to the other socio-demographic variables measured,

in that a high degree of direct experience of the condition very much dilutes the effects

of education and age that are seen to be influential on the attitudes of women in general

population studies.

The significance of the mental health problems of the sibling with Down’s syndrome

in relation to the target attitudes and perception of burden is also not easy to interpret.

The causes of psychological distress are frequently complex and the state of a person’s

family relationships may be both a cause and effect of mental ill-health. Research has

shown that some parents have a lifelong problem adjusting to their child’s disability and

a number of authors have written about a phenomenon called ‘chronic sorrow’ (Bruce

et al., 1996; Hobdell & Deatrick, 1996; Teel, 1991). The son or daughter with Down’s

syndrome is cared for, loved, and valued, but covert sadness accompanies interactions

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with the ‘unexpected child, to whom they could never totally adjust’ (Carpenter and

Carpenter, 1997, p. 11). Echoes of chronic sorrow could be heard in some open-ended

responses by those participants who indicated they would consider termination for

Down’s syndrome.

My mum always says that if anything happened to her and my dad she would not

want my sister and myself burdened by my brother. It is such a shame that he may

never lead a totally normal life.

The strength of the relationships between the perceived approval or disapproval of

family members of termination for Down’s syndrome and the views of the participant

supports the view that attitudes are ‘embedded in family relationships’ (Eagly &

Chaiken, 1993). Wertz and colleagues reported similar strong predictive relationships

in their study of families of children with cystic fibrosis (Wertz et al., 1991, 1992). The

authors commented that, ‘for persons living in a family context, it is difficult to make an

abortion decision that contradicts the values of their spouses and extended families’

(Wertz et al., 1991, p. 994). This might be true more generally, and Gilligan has

previously argued that women make decisions about abortions within a complex

network of relationships and responsibilities (Gilligan, 1982). However, to date,

familial influence on prenatal testing and termination decisions in the general

population has not been researched in any depth.

Cunningham (1996) has suggested that brothers and sisters ‘take the view of the sibling

with Down syndrome from their parents’ (p. 91), and Levine (2002) that ‘children mirror

the emotions and concerns of their parent (in relation to their affected sibling)’ (p. 170).

Difficulties adjusting to a child’s disability may be passed on to the next generation,

manifesting in the unaffected siblings as an inability to accept a ‘potential child’ with a

disability. One sister who had used PD during her pregnancy wrote:

I wanted to make sure (as far as possible) that my child did not have a mental

handicap. We discovered that my son was ‘genetically normal’. From then on I

could accept him and enjoy the pregnancy. It was an emotional discovery.

Siblings of affected individuals may only have their own family’s experience on which to

base their attitudes towards Down’s syndrome. Whereas mothers frequently have

contact through special nurseries or support groups, a sibling may have never met

another woman in a comparable situation to herself with whom to discuss her views and

feelings. The knowledge that many others share the attitudes she herself holds may be

of value in a pre-test counselling situation. Some siblings may welcome the chance to

discuss their concerns about having an affected child or value the availability of prenatal

diagnosis without the accompanying shadow of termination. However, an assumption

on behalf of health professionals that women with a sibling with Down’s syndrome

would want testing was considered to be deeply offensive by some participants.

It is proposed that the findings contribute significantly to a gap in the knowledge in

this area and provide a number of hypotheses testable by future research. Although the

data was collected in 1997–1998 they are still considered to be of relevance. Attitudes

towards learning disability have been shown to change relatively slowly (Rees et al.,

1991) and attitudes towards prenatal testing and termination have also shown little

material alteration over the past 15 years or so. However, the findings do have to be

considered within the context of the sample used, as the degree to which the study

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group represents the wider population of siblings in the UK is not known. The

difficulties of obtaining a representative sample from this population mean that the

sample recruited to this study may well be biased in some way, however, as there is no

known sampling frame for this population it is difficult to say in what way the bias

occurs. The percentage of people leaving education at age 18 or older was higher than

might have been expected, and as the recruitment materials were in English, the sample

is unlikely to reflect a great deal of ethnic diversity. The method of recruitment for the

cohorts meant that filtering via parents probably affected the response rates negatively,

however, there is no reason why this filtering would have occurred in a way as to build

in a systematic bias to the sample. Therefore, while it is accepted that the method of

sample selection was not ideal in a methodological sense, in the real world it was a

pragmatic approach to accessing a hard-to-reach population.

It is also acknowledged that in this study no consideration was taken of male sibling

views, however, it may be predicted that brothers of individuals with Down syndrome

will also have views that may influence a couple’s prenatal testing decisions. In general,

the role of the male partner’s views in informing women’s testing and termination

decisions has been neglected by research, however, there is no evidence to suggest that

men hold more or less favourable attitudes on this topic than do women. In conclusion,

a number of limitations of the study mean that generalisation to a wider population of

siblings of people with Down’s syndrome should not be made. However, the findings

begin to address an important gap in the knowledge in this area, which could be

usefully developed by further research.

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14:

46 1

5 Ju

ne 2

013