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ORIGINAL RESEARCH |
From the Departments of Obstetrics and Gynecology and Pediatrics, Tufts University School of Medicine, Boston, Massachusetts; and The Shriver Center for Mental Retardation, Inc., Waltham, Massachusetts.
Address reprint requests to: Steven J. Ralston, MD Department of Obstetrics and Gynecology Division of Maternal-Fetal Medicine Tufts University School of Medicine New England Medical Center 750 Washington Street Box #360 Boston, MA 02111 E-mail: sjralston{at}lifespan.org
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Abstract |
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Methods: A questionnaire was mailed to 51 mothers of children with aneuploidy. Women whose fetuses were diagnosed prenatally comprised the study group and those whose infants were diagnosed at birth were controls. Outcomes measured included an assessment of pregnancy management, neonatal outcome, subjective measures of depression and anxiety, and evaluation of women’s emotional and physical experience of the pregnancy. For outcomes measured by nonparametric survey questions, 20 women were needed in each arm to achieve a power of 80% to detect a 2-point difference on a 6-point scale; for our neonatal outcomes, 100 women were needed in each arm to achieve 80% power to detect a difference in length of stay (less than 1 week versus greater than 1 week) or need for surgery.
Results: Thirty-eight women (75%) responded. Most (86%) had children with trisomy 21. Seventeen women (45%) received their child’s diagnosis at birth; 21 (55%) had prenatal diagnoses. Demographic measures were similar except that women with prenatal diagnoses attended religious services more frequently (1–3 times per month versus once to several times per year, P = .04). Women with prenatal diagnosis had better perceptions of their physical experience of pregnancy (median score of 10 versus 6 on a 10-point visual analog scale, P = .005) and their emotional experience of the birth (median score of 7.5 versus 2, P = .001). Mental Health Inventory scores were similar between groups. Neonates without prenatal diagnoses were more likely to be transferred to tertiary centers after birth (70% versus 24%, P = .004); lengths of hospital stays and need for surgery were similar. Seventy-one percent (95% confidence interval [CI] 48, 89%) of women with prenatal diagnoses said they would have done nothing differently in the pregnancy compared with 29% (95% CI 10, 56%) of women with diagnoses at birth.
Conclusion: Early knowledge of fetal aneuploidy is beneficial to women who continue their pregnancies. These results might be useful when counseling women who do not intend to terminate abnormal pregnancies, but are considering prenatal diagnosis.
Women offer many reasons why they decline prenatal diagnosis or screening, whether it be invasive testing such as an amniocentesis or noninvasive testing such as serum marker screening.1 Among women who decline prenatal testing, up to two-thirds cite unwillingness to consider abortion an option and that they "would not do anything differently." Except for allowing couples the opportunity to make abortion decisions, there are few diseases which, when diagnosed antenatally, can be reliably and effectively treated in utero. There are exceptions to that rule: a diagnosis of spina bifida antenatally can change delivery route; the consequences of a bladder outlet obstruction may be ameliorated by a shunt; hydrops from fetal anemia can be cured with fetal transfusion; maternal steroid administration can improve outcomes for fetuses with congenital adrenal hyperplasia; and recent advances in minimally invasive fetal surgery have begun to show promise for select fetal malformations.
For congenital abnormalities that are not treatable in utero, there is conflicting information on the notion that prenatal diagnosis might change neonatal outcome,2–4 but for fetal aneuploidy, there is a paucity of such information. A growing body of literature in bioethics and disability studies is critical of the medical profession for promoting prenatal diagnosis of fetal aneuploidy as anything other than a means for selective pregnancy termination.5–7 It is true that rates of pregnancy termination for fetal aneuploidy vary from institution to institution and by diagnosis,8 and the view that prenatal diagnosis is a "search and destroy" mission is difficult to refute when there is little data about specific benefits of prenatal diagnosis of fetal aneuploidy. The purpose of this study was to assess whether antenatal diagnosis of fetal aneuploidy conferred any benefit to mothers or neonates when women elected to continue their pregnancies.
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Materials and Methods |
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Outcomes measured included assessments of pregnancy and delivery management and neonatal medical outcomes. Measures of depression and anxiety levels in women were assessed using the Mental Health Inventory-5 scale.9 We evaluated women’s emotional and physical experience of pregnancy and birth through questions that asked them to grade their experiences on a 1–10 visual analog scale.10,11 Statistical analysis used 
2, Student t, and Mann-Whitney U tests (for non-normally distributed data) where appropriate using SPSS 10.0 (SPSS Inc., Chicago, IL). A statistical significance level of .05 was chosen. For outcomes measured by nonparametric survey questions, we calculated that 20 women were needed in each arm to achieve a power of 80% to detect a 2-point difference on a 6-point scale; for our neonatal outcomes, 100 women were needed in each arm to achieve an 80% power to detect a difference in length of stay (less than 1 week versus greater than 1 week) or need for surgery. The study was approved by the hospital Human Investigations Review Committee.
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Results |
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Table 1
shows the demographic distribution of the cohorts. Most women in both groups were well educated, married, white, and either Catholic or Protestant. There was a trend toward greater maternal age among those diagnosed before birth. The only demographic that was significantly different between the two groups was our measure of religiosity: women with prenatal diagnoses attended religious services more frequently.
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. Women who had prenatal diagnoses had better perception of their physical experience of pregnancy and their emotional experience of birth. There was no reported difference in how they graded their relationships with their children: almost all gave a response of 10 on a 1–10 visual analog scale (1 = poor, 10 = good). Mental Health Inventory scores (Table 3) were not significantly different between groups.
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. There were no differences in the lengths of hospital stays or need for surgery after birth. Infants diagnosed at birth were more likely to have been transferred to a tertiary center for evaluation or treatment.
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Women who did not receive diagnoses until birth were more likely to report that they would have asked for more tests such as serum screening or ultrasound examination. Only one woman said she would have had an abortion. That cohort of women was no more or less critical of the medical professionals they dealt with than the women who received antenatal diagnoses. Several women in each cohort commented on the poor delivery of the news at the time of diagnosis.
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Discussion |
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Neonatal outcomes were improved for women who received antenatal diagnoses of fetal aneuploidy only insofar as their children were less likely to require transfer to tertiary centers, with consequent separation of mother and child. Other neonatal outcomes were more difficult to assess with this study. Although designed to assess women’s emotional responses to prenatal diagnoses, a questionnaire is probably not the best instrument to measure short-term or long-term improvements in neonatal medical outcomes. In-depth psychosocial and physical assessments of children done prospectively would have helped address this issue, but were well beyond the scope of the current study. It was interesting that neonates with prenatal diagnoses were not more likely to require surgery: one would suspect that more fetuses with Down syndrome who had congenital heart disease (and thus require surgery) would be diagnosed antenatally, but perhaps those fetuses were also more likely to be aborted, as suggested by Drugan et al.12 In addition, one cannot discount the possibility of a type II error affecting these statistics. Our power calculation showed that for our negative findings in our neonatal outcomes, we needed 100 women in each arm to achieve a power of 80% to detect a difference in the length of stay (less than 1 week versus greater than 1 week) or need for surgery.
The most difficult data to collect and interpret involved emotional responses of women responding to the study. In designing the questionnaire, we chose carefully several measures of emotional well-being and satisfaction. Some of those, such as Mental Health Inventory-5, have been studied extensively and proven valid as questionnaire instruments.13 The visual scale questions used to assess respondents’ experiences of pregnancy and birth are similar to scales that have been used reliably to assess pain and other subjective data. The pages of the survey that allowed for respondents’ expository commentary proved to be replete with information on how they were affected by the process of receiving their child’s diagnosis, whether before or after the child’s birth.
There did not appear to be any long-term differences in the mood states between cohorts as assessed by the Mental Health Inventory-5, nor by their subjective assessments of their relationships with their children. That is not surprising because the benefits of antenatal diagnoses are likely to be temporally related to the acquisition of that knowledge, and are also likely to dissipate with time. The long-term benefit to mothers, children, or families raising these children would be even more difficult to measure, and as time passes from diagnosis, the number of confounding factors would increase, making it very difficult to conclude reliably that it was prenatal diagnosis that led to any change in outcome.
One criticism that could be made of this project is that the groups studied were not truly comparable, that women who sought prenatal diagnoses and then continued pregnancies were dissimilar to women who received diagnoses after birth of their children. Our measure of religiosity—the number of times these women attended religious services—was different between groups. This makes intuitive sense because women who choose not to abort after an antenatal diagnosis might be more religious. Yet it seems unlikely that this difference alone would account for the consistent declaration of these women that the antenatal diagnosis was useful to them. Not one of these women mentioned her dependence on religion or God in helping her through the pregnancy; almost all commented on the benefit of meeting specialists, learning about Down syndrome, and the time to prepare for the birth of a special needs child. Demographics are different between women who accept and those who decline prenatal diagnosis,14 so it might be impossible to find ideally comparable groups of women for this type of study. A randomized trial that withholds prenatal diagnosis from one cohort would not be ethical, so this type of cohort data is the best means available to study this question.
As with any questionnaire survey with less than a 100% response rate, a selection or nonresponse bias is an inherent risk. This questionnaire was anonymous, so we were unable to make conclusions about the demographics of women who chose not to respond. Anonymous surveys do not necessarily improve response rates,15 but anonymity was believed to be necessary because of the emotionally charged nature of the study. Given the kinds of questions asked, we believe our response rate of 75% was excellent.
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Footnotes |
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Received September 14, 2000. Received in revised form December 11, 2000. Accepted January 12, 2001.
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References |
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2. Kumar RK, Newburger JW, Gauvreau K, Kamenir SA, Hornberger LK. Comparison of outcome when hypoplastic left heart syndrome and transposition of the great arteries are diagnosed prenatally versus when diagnosis of these two conditions is made only postnatally. Am J Cardiol 1999;83:1649–53.[Medline]
3. Bonnet D, Coltri A, Butera G, Fermont L, Le Bidois J, Kachaner J, et al. Detection of transposition of the great arteries in fetuses reduces neonatal morbidity and mortality. Circulation 1999;99: 916–8.
4. Simpson LL, Harvey-Wilkes K, D’Alton ME. Congenital heart disease: The impact of delivery in a tertiary care center on SNAP scores (scores for neonatal acute physiology). Am J Obstet Gynecol 2000;182:184–91.[Medline]
5. Saxton M. Disability rights and selective abortion. In: Solinger R, ed. Abortion wars: A half century of struggle, 1950–2000. Berkeley, CA: University of California Press, 1997:374–95.
6. Asch A. Reproductive technology and disability. In: Cohen S, Taub N, eds. Reproductive laws for the 1990s. Clifton, NJ: Humana Press, 1989:69–124.
7. Parens E, Asch A. The disability rights critique of prenatal genetic testing. Reflections and recommendations. Hastings Cent Rep 1999;29:S1–22.[Medline]
8. Mansfield C, Hopfer S, Marteau TM. Termination rates after prenatal diagnosis of Down syndrome, spina bifida, anencephaly, and Turner and Klinefelter syndromes: A systemic literature review. Prenat Diagn 1999;19:808–12.[Medline]
9. Stewart AL, Hays RD, Ware JE Jr. The MOS short-form general health survey. Reliability and validity in a patient population. Medical Care 1988;26:724–35.[Medline]
10. McDowell I, Newell C. Measuring health: A guide to rating scales and questionnaires. 2nd ed. New York: Oxford University Press, 1996.
11. Luria RE. The validity and reliability of the visual analogue mood scale. J Psychiat Res 1975;12:51–7.[Medline]
12. Drugan A, Greb A, Johnson MP, Krivchenia EL, Uhlmann WR, Moghissi KS, et al. Determinants of parental decisions to abort for chromosomal abnormalities. Prenat Diagn 1990;10:483–90.[Medline]
13. Berwick DM, Murphy JM, Goldman PA, Ware JE Jr, Barsky AJ, Weinstein MC. Performance of a five-item mental health screening test. Medical Care 1991;29:169–76.[Medline]
14. Press N, Browner CH. Characteristics of women who refuse an offer of prenatal diagnosis: Data from the California maternal serum alpha fetoprotein blood test experience. Am J Med Genet 1998;78:433–45.[Medline]
15. Campbell MJ, Waters WE. Does anonymity increase response rate in postal questionnaire surveys about sensitive subjects? A randomised trial. J Epidemiol Community Health 1990;44:75–6.
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