DOI: 10.1002/pd.4055
ORIGINAL ARTICLE
Parent’s attitude toward prenatal diagnosis and termination of pregnancy could be influenced by other factors rather than by the severity of the condition Ayman Alsulaiman1* and Khaled K. Abu-Amero2 1
Department of Genetics, Research Center King Faisal Hospital and RC, Riyadh, Saudi Arabia Ophthalmic Genetics Laboratory, Department of Ophthalmology, College of Medicine, King Saud University, Riyadh, Saudi Arabia *Correspondence to: Khaled K. Abu-Amero. E-mail:
[email protected]
2
ABSTRACT Objective The aim of this study is to investigate whether the severity of a particular condition alone influences parents’ attitudes toward prenatal diagnosis (PND) and termination of pregnancy (TOP) or are there other factors involved?
Methods A questionnaire that mainly focuses on parent’s attitude toward PND and TOP for 30 different hypothetical scenarios for a series of genetic, non-genetic, and non-medical conditions were completed by 400 Saudi parents. Results were compared and scored and parent comments were noted. Additionally, cross tabulation of thalassemia, considered the most severe and had the most favorable PND and TOP, against the 29 other conditions were carried out to find similarities and different views toward TOP and PND.
Results We found that parents’ attitudes toward PND and TOP for thalassemia are significantly associated with their attitudes in relation to all of the other conditions (p < 0.01).
Conclusion Saudi Parents’ attitudes toward TOP and PND are not always influenced by the severity of the condition. © 2013 John Wiley & Sons, Ltd.
Funding sources: King Faisal Specialist Hospital. Conflicts of interest: None declared
INTRODUCTION 1
Previous studies from Saudi Arabia showed similarities and differences between parents with and without an affected child in their attitudes toward prenatal diagnosis (PND) and termination of pregnancy (TOP) across a range of many diseases. We have previously shown that thalassemia was considered by most parents as a severe condition justifying TOP.2 Having said that, not all parents shared the same opinion as some thought that other genetic or medical conditions, apparently less severe than thalassemia, as the most severe condition. Some parents had different views on severity based on their circumstances. Individuals or parents, with low social and economical status, could favor TOP for conditions less severe than thalassemia, due to its impact on their life. Parents who perceive their affected children as having more serious problems may have a more negative perception of their own quality of life as a result. 3,4 Parents’ attitudes toward TOP and PND have been studied in various populations including Saudi Arabia. 1,5,6 Majority of these studies came to a main conclusion that parent’s attitude toward TOP and PND are largely influenced by the severity Prenatal Diagnosis 2013, 33, 257–261
of the condition. 7 Few studies had suggested that parent’s attitude could also be influenced by socioeconomic 8 and socio-cultural factors.9 Religious beliefs and traditional values also influence parent’s attitudes toward TOP and PND.10,11 These factors (socioeconomic, traditional values, and religious beliefs) had not been studied extensively especially in the settings of various medical conditions. Therefore, we are aiming to investigate how these factors influence parents’ attitudes toward PND and TOP in the settings of an extensive questionnaire detailing hypothetical scenarios for 30 different conditions. In this study, we compared thalassemia with other medical conditions and studied parent’s attitude toward PND and TOP for those conditions. We also took note of the parent’s additional comments and reported those here.
METHODS Study population This survey was conducted at the King Faisal Specialist Hospital and Research Center (KFSH&RC), Riyadh, Saudi Arabia. KFSH&RC is considered one of the biggest tertiary © 2013 John Wiley & Sons, Ltd.
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hospitals in the Middle East, which provides health services to all residents of Saudi Arabia. A questionnaire seeking attitudes toward prenatal testing and TOP for 30 different conditions were self-completed or interviewer assisted as required. These conditions were as follows: (1) not preferred gender, (2) cleft lip and palate, (3) mild LD/mental health (MH), (4) coronary at 50 years, (5) Alzheimer’s disease, (6) grossly overweight, (7) autism, (8) blindness, (9) deafness, (10) dwarfism, (11) moderate LD/MH, (12) cystic fibrosis, (13) fragile X syndrome, (14) Huntingdon’s disease, (15) cancer, (16) Turner’s syndrome, (17) absent limb, (18) high risk of alcoholism, (19) Klinefelter’s syndrome, (20) epilepsy, (21) phenylketonuria, (22) schizophrenia, (23) diabetes, (24) quadriplegia, (25) severe LD/MH, (26) proteus syndrome, (27) thalassemia, (28) Duchenne muscular dystrophy (DMD), (29) trisomy 13 or 18, and (30) anencephaly. The questionnaire contained scenario descriptions of 30 conditions (see previous text) and asked two questions for each condition: whether participants would consider a prenatal test and whether participants would consider a termination for an affected pregnancy. The response options for each of the two questions were ‘no’, ‘not sure’, or ‘yes’. Using this questionnaire, 422 Saudi parents attending different genetic clinics and outpatient clinics within 8-month period were interviewed by a trained research assistant and the author after written informed consent. There was an overwhelming response and 94.8% (400/422) of those approached agreed to participate. In our setting, half of the parents (n = 200) had an affected child and the other half without an affected child. The interview took from 20 to 25 min for each individual. The study had the approval of the Clinical Research and Ethics Committee of the (KFSH&RC). The questionnaire was constructed on the basis of a similar study in this area 1. It consisted of two sections. The first section included general questions on the demographic and personal characteristics. The second section were presented a list of 30 scenarios, parents were asked to answer one box for each of the two questions, the options of the answers were namely 1.No, 2.Yes, 3.Don‘t know. In addition, the parents were asked for a reason for their answer. The results of parents’ attitudes toward PND and TOP were presented elsewhere (Alsulaiman A and Hewison J, 2005). Statistical analysis was performed using the Statistical Package for the Social Sciences Version 11.0.
RESULTS An illustrative example used for cross tabulation of Thalassemia against severe learning difficulties (Table 1). The results
presented in Table 1 shows a strong association between the two conditions. Thalassemia was selected among the 30 conditions interrogated in the questionnaire, because Thalassemia is one of the commonest diseases in Saudi Arabia. It’s also one of the conditions for which parents in this study held the most favorable attitudes toward PND and TOP (see introduction). A Fisher’s exact test confirmed that there was a significant association between attitudes to the two conditions (p < 0.01). In each condition, nearly 90% of parents held favorable attitudes to PND, but the minority who rejected PND consisted of slightly different people on the two occasions. Then, we proceeded with cross tabulations between thalassemia and each of the 29 conditions listed in the questionnaire. The results for parent’s attitudes toward PND and TOP are presented in Tables 2 and 3, respectively. The results presented clearly indicate that parents’ attitudes toward PND and TOP for thalassemia are significantly associated with their attitudes in relation to all of the other conditions (Fisher’s exact p < 0.01 in all cases). However, for all conditions except non-preferred gender, parents were identified who held all combinations of attitudes, namely, favorable for both conditions, for neither condition, for thalassemia but not the other condition, and vice versa.
DISCUSSION Thalassemia is group of inherited blood disorders caused by reduced or absent synthesis of the alpha or beta chains of hemoglobin resulting in variable phenotypes ranging from severe anemia to clinically asymptomatic individuals. No one can argue about the severity of this condition and the devastating effect it had on the patients and their parents and sometime their close relatives who have to endure long-term care for their affected relative. Thalassemia is common in Saudi Arabia along the coastal strip of the red sea and the eastern province including Jubail, Qateef, Dammam, and Hofuf. It is not surprising to see from our survey that most parents judged thalassemia as the most severe condition justifying termination of pregnancy.2 Most of those parents thought that a child requiring a repeated blood transfusion is a burden and a problem to himself, his immediate relatives, and the society as a whole and they favor TOP in such a situation. Despite the fact that the severity of genetic condition plays an important role of parents’ attitudes toward PND and TOP, some parents might perceive the genetic testing for a specific condition differently. We hypothesize that parent’s attitudes toward TOP may be influenced also by their religious beliefs, cultural values, and the impact on their quality of life regardless of the severity of the condition. For example, even
Table 1 Attitudes to prenatal diagnosis for thalassemia cross tabulated with severe learning disability (% are of total sample) Total
Severe LD No Thalassemia
Total
Yes
No
29 (7.3%)
23 (5.8%)
Yes
19 (4.8%)
329 (82.3%)
348 (87.0%)
52 (13.0%)
48 (12%)
352 (88.0%)
400 (100%)
LD, learning difficulty.
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Table 2 Parents’ attitudes toward prenatal diagnosis in thalassemia and other conditions Condition
No to thalassemia and other condition(%)
Severe LD/MH Quadriplegia
No to thalassemia but yes to other condition(%)
Yes to thalassemia and other condition(%)
7.3
5.8
82.3
Yes to thalassemia but no to other condition(%) 4.8
8.5
4.5
81.8
5.3
10.8
2.3
83
4
Trisomy 13 or 18
9.3
3.8
81.5
5.5
Phenylketonuria
9.5
3.5
79.8
7.3
Diabetes
10.5
2.5
79.5
7.5
Proteus syndrome
10
3
79
8
DMDdystrophy
Anencephaly
10.3
2.8
79
8
Absent limb
10.5
2.5
77.8
9.3
Schizophrenia
10
3
77.3
9.8
Epilepsy
11
2
77.5
9.5
Cystic fibrosis
10.8
2.3
76.8
10.3
Cancer
10.8
2.3
75.3
11.8
Klinefelter’s syndrome
10.8
2.3
75
12
Fragile X
10.3
2.8
74.3
12.8
Deafness
10.8
2.3
74
13
Huntingdon’s disease
10.3
2.8
73
14
Moderate LD/MH
10
3
72.5
14.5
Blindness
10.8
2.3
72.3
14.8
Turner’s syndrome
10.8
2.3
70.3
16.8
Grossly overweight
11.3
1.8
69.8
17.3
Autism
10.3
2.8
67.8
19.3
9.8
3.3
66.8
20.3
High risk of alcoholism Mild LD/MH
11.3
1.8
64.5
22.5
Dwarfism
11.8
1.3
62.8
24.3
Cleft lip and palate
11.3
1.8
60.3
26.8
Coronary at 50 years
11.5
1.5
58.8
28.3
Alzheimer’s disease
12
1
55.5
31.5
Not preferred gender
11
2
43.8
43.3
The percentage of responses in each combination of attitudes. Conditions are listed in this table in rank order from most to least favored. DMD, Duchenne muscular dystrophy, LD, learning difficulty.
though cleft lip and palate had a very low ranking overall in term of severity, and thalassemia a much higher ranking, there were still some parents who rejected termination for thalassemia but accepted it for cleft lip and palate. Majority of the parents who favored TOP for cleft lip and palate and not thalassemia gave the following reason for their answer ‘ladies usually spend most of their time in front of the mirror for makeup purposes. I feel that if a lady is short, fat, deaf, blind, or missing a part of her body, she will suffer a lot socially. She will have difficult time finding a husband accepting her condition and she will not live a normal life socially, so termination of such an abnormality would be better.’ So clearly, the cultural values play a role in parent’s attitude to TOP. Another example where own personal views influences parents attitude toward TOP is when parents felt that their condition was ‘severe’ and should be terminated, but less Prenatal Diagnosis 2013, 33, 257–261
‘severe’ conditions according to their views, should not be terminated. Majority of those parents commented on this issue by saying ‘I feel we should terminate only the mentally handicapped, because they will be hopeless, but those who will be late affected in his or her age should not be terminated at all because he or she should at least live part of their lives without this condition and that we do not know when we will die anyway.’ Another example where religious believes influences parents attitude toward TOP is when parents rejected TOP for all conditions except alcoholism. Parents stated that ‘we should not terminate any kind of abnormality because it is all from God and we cannot do anything against God’s creation, but for the alcoholic condition, we should terminate the pregnancy because it is against Islamic Law.’ This statement stems from the Islamic religious belief, which prohibits alcoholism. © 2013 John Wiley & Sons, Ltd.
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Table 3 Parents’ attitudes toward termination of pregnancy in thalassemia and other conditions. The percentage of responses in each combination of attitudes Condition
No to thalassemia and other condition(%)
No to thalassemia but yes to other condition(%)
Yes to thalassemia and other condition(%)
Yes to thalassemia but no to other condition(%)
Anencephaly
36.8
23.3
34.5
5.5
Trisomy 13 or 18
41
19
34
6
Severe LD/MH
39.8
20.3
31.8
8.3
Quadriplegia
43
17
27.8
12.3
DMD
48.3
11.8
31.5
8.5
Diabetes
50.3
9.8
24.3
15.8
Proteus syndrome
50.8
9.3
24.5
15.5
Schizophrenia
52.8
7.3
23.8
16.3
Phenylketonuria
54
6
22
18
High risk of alcoholism
49.5
10.5
17
23
Klinefelter’s syndrome
54.8
5.3
22
18
Epilepsy
54
6
20.5
19.5
Absent limb
54.8
5.3
20.3
19.8
Cystic fibrosis
55.5
4.5
19.8
20.3
Turner’s syndrome
54.8
5.3
17
23
Huntingdon’s disease
55.3
4.8
17.3
22.8
Fragile X syndrome
55.5
4.5
16.5
23.5
Cancer
55
5
15.5
24.5
Autism
57.3
2.8
13.5
26.5
Blindness
57.5
2.5
13.3
26.8
Moderate LD/MH
57.5
2.5
12.8
27.3
Grossly overweight
57.5
2.5
11.5
28.5
Dwarfism
57
2.8
10
30
Deafness
57.5
2.5
9.8
30
Alzheimer’s disease
57.3
2.8
7
33
Mild LD/MH
58.5
1.5
6.8
33.3
Coronary at 50y
59
1
5.8
34.3
Cleft lip and palate
58.8
1.3
4.3
35.8
Not preferred gender
60
0
0
40
Conditions are listed in this table in rank order from most to least favored. DMD, Duchenne muscular dystrophy; LD, learning difficulty.
Whenever the parents were willing to terminate a particular condition in the scenarios, they had their own justification, even if the condition was not ‘severe’ according to other parents’ opinion. Hence, parents had similar and different views toward termination of the pregnancy according to their individual experience. The examples of parent’s responses are not restricted to the Saudi society as other studies came to the same conclusion.12–14 Additionally, we found in this study how parents perceive the condition depends on their individual experience. Hence, parents’ views are specific to them and specific to that condition. This is consistent with the view of Koch (2001)15 that the severity of the condition that leads to impairment or disability is defined by social prejudice, not by physical or medical facts. Because humans see themselves in the framework of the reactions of others,16 parents’ concerns are often related to perceptions Prenatal Diagnosis 2013, 33, 257–261
of self-image rather than physical disability or impairment. However, it is disability and impairment which are reported by physicians, in terms of biological features and the kind of treatment that the disabled child needs. In addition, the informed choice of the parents is very important, because there is a gap between description of disabilities and potential disabilities and consequence for family life as the child develops. It must also be remembered that even if parent’s attitudes reported were valid, attitudes should not be confused with the behavior; what people say does not always indicate what they will do. For instance, previous studies showed that a majority of those at risk of Huntington’s disease said they would have a genetic test when it became available, yet only a small proportion underwent the test when it did become available.17 Similar findings have been obtained for predictive testing for cancers in both Europe and USA.18,19 Other studies © 2013 John Wiley & Sons, Ltd.
Attitudes toward PND and TOP is not always influenced by severity of condition
have shown that after finding out that their child is affected with Down’s syndrome, a much higher proportion of parents are willing to terminate the pregnancy than attitude studies would suggest 20. This might indicate that when the parents face the real implications in their life, they might change their minds and go for termination. Hence, genetic counselors should explore the implications that parents might face with their affected child according to their culture. However, in a country such as Saudi Arabia, where termination is not allowed in all genetic conditions, there is no point in the genetic counselor exploring the implications with the family, in the case of conditions for which termination is not allowed. Therefore, in line with the notions of informed choice and freedom but within the boundaries permitted by law, it is important to explore the broader social and cultural context in which decisions are taken and allow parents to explain their concerns according to their individuals’ experience,
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which might influence their attitudes toward prenatal diagnosis and termination of pregnancy.
WHAT’S ALREADY KNOWN ABOUT THIS TOPIC? • In the Saudi society, attitudes toward prenatal diagnosis and termination of pregnancy is influenced by the severity of the condition.
WHAT DOES THIS STUDY ADD? • Saudi parents’ attitudes toward prenatal diagnosis and termination of pregnancy for various medical and genetic conditions varied on the basis of individual circumstances. Some parents were clearly influenced by their religious beliefs, others by cultural values, and some by the impact on their quality of life regardless of the severity of the condition.
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