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Ethical Issues Surrounding Multifetal Pregnancy Reduction and Selective Termination
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ETHICAL ISSUES SURROUNDING MULTIFETAL PREGNANCY REDUCTION AND SELECTIVE TERMINATION Mark I. Evans, MD, Mark Paul Johnson, MD, Ruben A. Quintero, MD, and John C. Fletcher, PhD
Public fascination with multiple gestations has been timeless and extends in the media back 60 years to the Dionne quintuplets. Despite the national attention given recently to the Dildy sextuplets and other high-profile successes, the number of sad outcomes continues to rise geometrically. 28 Buried in the fascination about the wonders of multiple births has been an ignorance of the increased reproductive risks inherent in the event. In this article we address some of the ethical issues that arise in multiple gestations in which there is not only a potential conflict between the interests of mother and child but also among fetal siblings. The ethical issues involving multiple gestations can be divided into two clinical situations: (1) multiple gestations in which the number of fetuses in and of itself threatens the ability of the mother to carry them far enough in pregnancy to survive and (2) twin gestations, generally not a result of infertility treatment, in which one fetus is found to be abnormal. 16 After the first few publications, there has been a convention adopted in the literature, that is, the term multifetal pregnancy reduction is used for first-trimester procedures that are done because of fetal number per
From the Departments of Obstetrics and Gynecology (MIE, MJP, RAQ), Molecular Medicine and Genetics, and Pathology (MIE, MPJ), Wayne State University /Hutzel Hospital, Detroit, Michigan; and the Center for Biomedical Ethics, Health Sciences Center, University of Virginia, Charlottesville, Virginia (JCF)
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se. Selective termination is for procedures done predominantly in the second trimester because of a diagnosed fetal abnormality. 5
ETHICAL PRINCIPLES FOR THE PRACTICE OF MEDICINE AND CLINICAL RESEARCH
In our view, the selection of ethical guidance involves two interdependent claims. First, ethical guidance ought to be judged by the consequences of following it. Second, the consequences should be examined in terms of ethical principles widely respected across cultural, philosophical, and religious lines. A set of principles with wide acceptance in biomedical ethics is outlined below. 1. Respect for persons: The duty to respect the self-determination and choices of autonomous persons as well as to protect persons with diminished autonomy (e.g., young children, mentally retarded persons, and those with other mental impairments). 2. Beneficence: The obligation to secure the well-being of persons by acting positively on their behalf and, moreover, to maximize the benefits that can be attained. 3. Nonmaleficence: The obligation to minimize harm to persons and, whenever possible, to remove the causes of harm altogether. 4. Proportionality: The duty, when taking actions involving risks of harm, to balance risks and benefits so that actions have the greatest chance to result in the least harm and the most benefit to persons directly involved. 5. Justice: The obligation to distribute benefits and burdens fairly, to treat equals equally, and to give reasons for differential treatment based on widely accepted criteria for just ways to distribute benefits and burdens. The consequence of the four positions of ethical guidance outlined by us previously for cases of multiple pregnancies can be briefly examined in terms of these five principles:
Position 1: Proceeding to delivery in each case of multiple pregnancy because abortion, multifetal pregnancy reduction, and selective termination are ruled out because they (1) violate self-determination; (2) do not always result in the benefits of completely healthy children; (3) increase long-term harm and suffering to more persons; (4) effect a disproportionate relationship between the risks of premature delivery and the possible benefits of survival, because more persons will be harmed than benefited; and (5) are unfair to parents by burdening them with multiple children, some of whom may be severely impaired or suffer a lengthy, costly process of dying in neonatal intensive care. Position 2: Aborting each multiple pregnancy in hope of starting a new pregnancy (1) restricts choice of selective termination, if the
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procedure is available; (2) brings little benefit to infertile couples who may have great difficulty in a new pregnancy; (3) avoids the long-term harms and complications of premature delivery; (4) avoids many risks and harms but with few benefits; and (5) may be unfair to older couples with a long-term history of infertility. Position 3: Multifetal pregnancy reduction or selective termination in any multiple pregnancy, including twins, on request (1) maximizes self-determination; (2) if effective, benefits parents with at least one surviving child; (3) prevents abortion, reduces harms and complications of premature delivery, reduces or prevents stay in neonatal intensive care, increases incidence of selective termination to a larger number of cases (twins), increases incidence of fetal loss, and when ineffective will increase chances of spontaneous abortion, resulting in no child at all; (4) in light of fewer risks of twin pregnancies compared with multiples of three or more, it disproportionately results in more deaths of normal fetuses who would be less apt to suffer serious harm at delivery; and (5) distributes the benefits of selective termination fairly to all parents in all multiple pregnancies. Position 4: Multifetal pregnancy reduction or selective termination in pregnancies of three or more, but to no fewer than twins (1) restricts self-determination only of parents of normal twins; (2) if effective, benefits parents with two surviving children; (3) prevents abortion, provides protection against total loss of pregnancy in case of one fetal demise, reduces harms and complications of premature delivery; reduces length of stay in neonatal intensive care; prevents extension of selective termination to twins; when ineffective, runs risk of spontaneous abortion; (4) proportionately avoids the worst harms of the most dangerous multiple pregnancies with the most benefit to parents and survivors; and (5) distributes benefits and burdens of selective termination fairly to all parents in the most dangerous multiple pregnancies. ONE ABNORMAL TWIN
The Controversy Prior to the 1980s, the diagnosis of multiple gestations was limited to auscultation of fetal heart beats, crude outlines on radiographs, or limited ultrasound images. 7 With increased diagnostic abilities have come fundamental ethical problems. By far the most common situation involving one abnormal fetus in a multiple gestation is that of one twin who is shown to be either dysmorphic by ultrasound or aneuploid by invasive genetic studies. Since the earliest report by Aberg et aP in 1978, which detailed cardiac puncture for one fetus of twins with Hurler's disease, the subject has always drawn criticism. In 1981, a published report from New York detailed a midtrimester "selective feticide" of a
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Down's syndrome fetus when the other was normal. 27 The authors implied they were forced to attempt the procedure by the mother who threatened to abort both fetuses. Despite the disclaimers, they were criticized for intervening at risk to the normal twin. Sentiment against risking a normal twin continued in many forums through the 1980s. A set of guidelines for fetal therapy was first devised in 1982, which included avoidance of experimental procedures when there was a normal twin. 25 Such guidelines were later modified to permit procedures on an abnormal twin prior to the viability of the normal twin. 22' 27 This view is more consistent with a mother's right to abortion prior to fetal viability than is the restraint of her rights because of the presence of the normal twin. In the 1980s, despite the certainty of many diagnoses of one abnormal twin, very few publications discussed outcomes involved with selective intervention on one fetus. Several potential cases were not published by physicians engaged in such procedures primarily because of the "heat" that would be generated. Although toward the end of the decade, a few physicians risked the heat and began to publish their experiences. Through the mid-1990s, experience increased and included a 1994 collaborative report that proved the safety and efficacy of selective termination.9' is, 24 Loss rates increase with gestational age and vary with techniques. In the mid-1990s, the development of fiberoptic endoscopy has now allowed for selective termination to be performed on monozygotic twins. Umbilical cord ligation can be used for those cases in which use of potassium chloride would be contraindicated because of vascular communication between the twins that would have a high likelihood of damage to the survivor. 35, 35a The incidence of abnormalities is higher in twin pregnancies than in singleton pregnancies. 28 For dizygotic twins the risks are essentially the product of two singleton infants' risks. For example, the risk of a chromosome abnormality at age 35 is about 1 in 200, and the chance that one of a set of twins would be aneuploid is about 1 in 100. Overall, serious disorders are stated at 2% to 3% for singletons, and 4% to 7% for twins. For monozygotic pairs, aneuploidy risk is for "both" or "none," and is identical to the singleton rate. Disorders of laterality and dysmorphology are higher than for singletons or dyzygotic twins. Thus, the chance of facing one abnormal twin is not rare. 10 With increasing diagnostic capabilities, diagnoses of one abnormal twin have moved from the nursery to the second or even first trimester, and it is within the legal rights of the mother to consider termination of the pregnancy. Couples facing the diagnosis of one abnormal twin must confront an excruciating dilemma either to continue the pregnancy with the birth of an abnormal child or abort both fetuses, including the one that is normal. 9 , rn, 24 , 35, 35 a Such a choice transcends the traditional arguments about social abortion (which will not be reviewed here) and even
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those about abortion of a singleton abnormal fetus in an otherwise planned and wanted pregnancy. Ethical Justification for Selective Termination In assessing the ethical perspectives about twin selective termination, various arguments are possible. First, there are the traditional objections to aborting an abnormal pregnancy. The issues involved in selective termination, however, are far more complex because of the presence of the normal twin. In our thinking, the overriding principle is one of trying to preserve the most good and doing the least harm. As with all new techniques, there often is much uncertainty as to the true risks and benefits of such procedures. As with any termination procedure, there are risks of infection, ruptured membranes, and premature delivery, all of which can cause death to the normal twin and pose maternal risks. Furthermore, retention of a dead fetus carries known, although small, risks of coagulopathies, such as disseminated intravascular coagulation (DIC), which could be life threatening. 36 Our collaborative data have not shown a single case of DIC. We believe the risk of DIC is very low and more likely an issue related to much later gestational age spontaneous demises. 18 The approach to ethical reasoning about selective termination must be different from that for abortion per se and, therefore, requires a basic justification. By basic, we mean our reasoning ought not to be derived from precedents that appear to be morally similar but that on examination are not (i.e., arguments for social abortion). It is too simplistic to argue that because society permits abortion of normal fetuses on request in the first trimester, selective termination must fall well within the sphere of permissible actions created by abortion practices. Such reasoning derives an ethical imperative, an ought, from an existing cultural situation that is still ethically controversial. Any intentional action that could, or does, result in fetal harm or death requires a basic ethical justification. Otherwise, physicians would be liable to moral blame for destroying innocent human life without regard for the many ethical traditions that transmit respect for human life. For example, a basic ethical justification for fetal research, independent of arguments for and against abortion, was developed in the mid1970s,32 and even that process has evolved considerably. Both abortion and selective termination cause fetuses to die and are similar in this consequence. But selective termination may enable the surviving fetus to have a better chance for life. Moreover, morally they differ because the intent or aim is different. In abortion, the woman wants to end the whole pregnancy and enlists a physician's help. In selective termination cases, the woman wants to continue a pregnancy, and selective termination allows for the option of doing so without the burden of having an abnormal child as well.
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In practice, selective midtrimester terminations in twins generally have been limited to centers that provide the technical expertise to carry out twin diagnoses, use methods of termination that carry minimal risk to the healthy twin, and manage the continuing pregnancy to avoid the potential dangers of a dead twin in the uterus. 38 Nevertheless, in our view, the option of selective termination meets the criteria of most good-least harm because it preserves the chance for couples to have a healthy child unburdened by abnormalities-the original intent of the pregnancy. An appropriate analogy would be an abortion of an abnormal child followed by a subsequent normal pregnancy. The intent is the same even if the methods are different, and the risk to the normal twin is higher. To be certain, the risks and benefits should be proportionate; however, nearly two decades of experience suggest that selective termination can be a relatively safe procedure. Despite our conclusion of the ethical probity of such procedures, we still hold concerns about the timing of the procedure and indications for it. We reaffirm our beliefs that such procedures only can be considered prior to viability and should not be performed for objectionable reasons, such as sex selection or simply a desire for only one child. The literature on the ethics of sex selection is expanding. 15 We believe that "social" sex selection is inconsistent with an open and equal society and inherently creates inequalities between the sexes. Despite disclaimers to the contrary, in our experience, the vast majority of sex selection requests are for boys. We do believe that sex selection can be appropriate, but, in general, only for sex-linked diseases for which diagnoses are not possible or completely reliable. 30 Until recently, Duchenne muscular dystrophy or hemophilia would have fit that category. 20 , 26 Ironically, in such cases, the selection is against males. The advent of increasingly sophisticated ultrasound has allowed diagnoses of abnormalities much earlier. Diagnosis of dysmorphic anomalies are becoming common in the first trimester. 6 We believe that political pressures against abortion may curtail the rights to abort late in the midtrimester, which will, in turn, further emphasize the need for earlier diagnosis and earlier intervention, making selective termination both technically easier, safer, and more ethically accepted. MULTIPLE PREGNANCIES
With a twin pregnancy, the obstetric outcome in uncomplicated cases is reasonably good. 36 In gestations of larger size, even perfectly normal fetuses may be at considerable risk from prematurity. RATIONALE FOR MULTIFETAL PREGNANCY REDUCTION
The first cases of multifetal pregnancy reduction (MFPR) were published in 1986 to 1988,3 , n, 17 and since then there have been several
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individual center and collaborative series that have allowed for the establishment of now relatively smooth risk curves that show increasing pregnancy loss rates and premature rates that correlate with both starting and finishing numbers. 12• 13, 14• 3 9, 42 When we first confronted the issues of MFPR in 1986, such cases were not reported in the literature, and ethical precedents for such procedures were lacking. We debated as to the best course of action under such circumstances, recognizing that the use of fertility drugs such as human menopausal gonadotropin (HMG, menotropins, and urofollitropin), assisted reproduction techniques such as in vitro fertilization, and gamete intrafallopian transfer in the treatment of infertility carried the risk of inducing multiple gestations. Despite reduced occurrences and better understanding of the mechanisms of fertility drugs over the last decade, the possibility for inducing multiple gestations still exists. 2 In fact, the occurrences of multifetal pregnancies continues to rise, not fall. Explanations for the rise are multifaceted but include a tendency for less caution in their use because of the knowledge that MFPR can be used to treat a multifetal pregnancy. 21 With Pergonal, and now more commonly Metrodin, twin pregnancies occur in about 10% to 30% of patients. Higher numbers of conceptuses are seen in about 1% to 30% of patients. In some centers, the numbers are actually much higher. 2• 21 The obstetric outcome of triplets (or more) is known to be significantly poorer than with singleton or even twin pregnancies. 36 The ability to carry four or more fetuses to viability is very problematic. Only two successful sextuplet pregnancies are known in the United States. A successful gestation of octuplets has never been substantiated. Thus, a couple faced with the prospect of an octuplet pregnancy has a very serious dilemma. With no intervention, they will lose all the fetuses. In 1985, a case of septuplet births in California was highlighted in the national media. 34 Ultimately, only three infants survived. All have mental and physical handicaps, and malpractice litigation was successfully brought. In the case of multifetal pregnancies, one management alternative is to abort the entire pregnancy and have the couple try to conceive again. Because another conception cannot be guaranteed, another more attractive possibility is to reduce the number of fetuses with the hope of increasing the probability of a good outcome for at least some of them. Selective termination in multiple pregnancy, usually of one abnormal twin, in the midtrimester had been reported in the literature on a few occasions in the 1980s,24• 27 • 38 but literature on the ethics of selective termination was essentially nonexistent when we first had to make hard choices. In our view, MFPR in these patients was the only alternative available to achieve a reasonable chance of successful pregnancy outcome. In all instances, the couples understood the potential risks of the procedure and its experimental nature. Informed consent was obtained after thorough counseling. We stressed that any attempt to reduce the number of fetuses would be experimental and could result in miscarriage of the
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pregnancy. Infection, bleeding, and other unknown risks were possible. If successful, the attempt could theoretically result in damage to the remaining fetuses. All felt strongly, however, that because of their infertility history their current pregnancy might be their only or last opportunity to have children. As of 1996 about 2000 cases have been reported. The rate of subsequent live twin births appears to be about 88%. The medical details have been elucidated in great detail elsewhere and will not be repeated here. 13 Ethical Justification for Multifetal Pregnancy Reduction
Although there still is some debate, more than a decade of clinical experiences with good outcomes has demonstrated the technical feasibility and safety of first-trimester MFPR, even in grand multiple pregnancies that has improved outcomes compared with nonreduced conceptions.29, 33, 41 In the mid-1980s, we recognized, however, that MFPR was an inherently controversial topic and would require a thorough, prior, ethical reflection. 17 First we recognized, as with selective termination for one abnormal twin, that a basic ethical justification was required. Second, we recognized that our reasoning should not be limited to cases of multiple pregnancies after iatrogenic effects of infertility treatment. We must be willing to reason similarly in cases in which the pregnant woman's health and the survival of any infants are endangered by a multifetal pregnancy, regardless of cause. MFPR in a multifetal pregnancy raises ethical issues different from those for one abnormal twin. In multifetal pregnancies, childbearing has been endangered by multiple fetuses, all of which may be normal. The patient may or may not regard this pregnancy as her final chance to have a child. She enlists a physician's help to facilitate the continuation of the pregnancy. Termination of one genetically abnormal twin is, in some respects, similar to cases of termination in multifetal pregnancies. In both instances the woman desires to complete the pregnancy. The harms and dangers of severe genetic disease and prematurity of multiple newborns can be similar in expected morbidity and mortality, the major difference being between a diagnosis of a congenital anomaly and a history of infertility. The chance of a subsequent normal pregnancy after abortion for genetic disorders is more likely than in cases that involve infertility. Despite generally good anecdotal outcomes in twin cases, we were reluctant to rely on midtrimester precedents because, at the time, the outcomes of midtrirnester terminations of an affected twin and selective birth of a healthy surviving twin had not been studied adequately. Without reliable knowledge of long-term beneficial consequences, however, the value of the precedent was dubious, even though ethical reasoning about the two problems might be similar.
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In our first cases, we began from the premise that a pregnancy that has been finally achieved, particularly in the face of previous infertility, is a basic human good worth preserving. To what lengths is it ethically acceptable to go to achieve and preserve a pregnancy? Some, such as Tiefel, 40 or the 1987 Vatican document,8 challenged the ethical acceptability of methods of fertility treatment (including in vitro fertilization). Tiefel viewed the anxiety that brought these women into fertility treatment as caused by "dehumanizing socialization" in which "having a child is the ultimate need and in which infertility is seen as personal failure and a sign of worthlessness." 40 Had they only come to terms with these (supposed) feelings, they would never have found themselves in this situation. We doubt whether "dehumanizing socialization" is the basic cause of the despair of infertile couples. Furthermore, we find it impossible to accept the Vatican's position that attempts to treat infertility involving anything but marital coitus are immoral. There are straightforward biologic causes of infertility that can be remedied by a number of methods, including in vitro fertilization. We do not accept the posture of passive resignation in the face of biologic problems of infertility. If something can be done about infertility, we believe there is a strong moral obligation to do it, because infertility is a significant form of human suffering. Nonetheless, we do agree with the Vatican report that one is not ethically permitted to do simply anything to achieve or preserve pregnancy. How should the human need for a child be evaluated on ethical grounds? Is there a moral duty to have a child of one's own? Is there a right to have a child, as some parents have argued? Tiefel40 makes a case, with which we agree, that there is no moral or legal right to have children in a positive or enabling sense. Society is not obligated to meet an entitlement of a child in the same sense that rights to life, freedom of speech, and protection from enemies are entitlements. On the other hand, the Vatican notwithstanding, there is a negative duty not to be restrained or prevented from childbearing. In our society we believe that right is guarded by the legal right to privacy. 40 Once having obtained help in becoming pregnant, women in this situation deserve help to achieve their aim, as long as the means used are proportionate to the ends sought. A proportionate relationship between means and ends entails two criteria. First, there must be no other way to achieve the end, in this instance, preservation of a viable and desired pregnancy. Second, in choosing among the means available the chosen one must be the one that results in least harm and results in the most good for all involved. 31 In our cases of multifetal pregnancies, there were only three real alternatives. We recognized that any of the three could lead to harm. First, the pregnancy could be electively aborted, causing death to all fetuses and possibly not succeeded by any future pregnancy. A second possibility was to do nothing and risk the birth of multiple premature infants, many of whom might die or be significantly impaired. 2 In such a choice, there was also a very high likelihood of a spontaneous loss of
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the entire pregnancy. The third alternative was MFPR. Negative outcomes included the deaths of multiple fetuses and potential harm to the remaining twins; however, in these circumstances, we concluded, and our patients agreed, that MFPR held out more potential benefits than the other two alternatives. MULTIFETAL PREGNANCY REDUCTION AND ABORTION RIGHTS
Views on the morality of abortion are obviously relevant to choices in multiple pregnancy because both raise the question of whether deliberate killing of a fetus is justified. We argue in the following discussion that abortion practices per se are not sufficient to justify MFPR in multifetal pregnancies; however, the structure of an ethical argument concluding that some abortions are morally justified is highly similar to the argument for MFPR. A typology of responses to MFPR can be constructed by inference from the literature on the morality of abortion. The actual views of particular authors, and the variation within religious and philosophical traditions, are far richer than the truncated typology presented here.
Position 1: Any human act deliberately taken to destroy human embryonic or fetal life ought to be morally condemned. 16 This position rules out MFPR in any case. Position 2: An exception to position 1 is permitted for abortion only to save the life of a pregnant woman, as in cases of uterine cancer or ectopic pregnancy. Whether this position would favor or reject MFPR in multifetal pregnancies turns mainly on the empiric questions of the degree of threat to the life of the mother of continuation of pregnancy with premature delivery. Position 3: Although abortion is a serious moral problem, some abortions are permissible for ethically valid reasons. 31 It follows that some cases of MFPR in multifetal pregnancies are permitted, especially to avoid predictable harms to the pregnancy woman, to remaining fetuses, or to survivors. Because this position attempts to draw lines between morally valid and invalid reasons for abortion, it follows that a compatible position on MFPR would search for some moral lines between permissible and impermissible cases. Position 4: A position for abortion on request for any pregnancy at any stage32 favors a stance on MFPR that permits termination of any number of fetuses, including twins, to any remainder. The main concern would be the health and well-being of the pregnant woman. Positions on the morality of abortion mainly rest on three sorts of premises: (1) the moral status of the human embryo and fetus; (2) the relative strengths of countervailing moral claims made in the interests
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of the pregnant woman, the family, or society; and (3) the determination of how much moral weight should be given to whether the pregnancy is wanted by the woman. All positions on abortion tend to accept the premise that each pregnancy should be wanted and planned with medical advice, if available. Position 1 accords moral status of "personhood" to embryos from fertilization and requires that society protect every fetus equally in every pregnancy. This position allows for no countervailing moral claims and accords little moral significance to the unwantedness of the pregnancy. Position 2 gives the same high moral status to the fetus throughout pregnancy and allows only one higher moral claim to override when continuation of pregnancy actually threatens the woman's life. An important variation of this position extends slightly the range of countervailing moral claims and bends to permit abortions in "tragic" circumstances resulting from rape or incest and for diagnosis of genetic disorders that are totally incompatible with physical or mental life. Position 3 involves a "graded" 37 approach to the moral status of the fetus that resists indifference at any stage but gradually raises society's protection of the nascent human being with stages of development. This view is more likely to recognize countervailing moral claims, especially in the first trimester of pregnancy. It gives an important, but not absolutely overriding, weight to the wantedness of pregnancy. For example, some unplanned pregnancies that occur after failure of contraception would not result in substantial harm to the woman, family, or society. Abortion under such circumstances, in this view, is morally objectionable because the harm done by abortion is worse than ·the supposed but empty harm that would be prevented by the abortion. This view is probably dominant in the sense of prevailing in practice and public policy in this society. "Dominance" is used here as a descriptive term, and not a moral judgment. Position 4 is skeptical of claims for significant moral status or the interests of the fetus until birth, and even then claims to personhood and equal protection might be overridable in certain cases. "Wantedness" of the pregnancy is treated as a matter of moral supremacy in any context. Do abortion practices justify MFPR in multiple pregnancy? Some might argue "if society permits abortion of a normal, unwanted fetus, what is so wrong about terminating eight, four, or three to two, or two to one in a wanted pregnancy?" 37 It is true that if society proscribed abortions, MFPR also would be illegal.37 What is legally permitted, however, is not always ethically sound. Using abortion practices as sole sources of justification of MFPR is inadequate given the premises of the third position, because some abortions are done for reasons that cannot be justified by ethical principles and that violate respect for fetal life (e.g., abortions for gender choice). To argue from the premise of what society permits in abortion would also encourage MFPR in cases of normal twins, a direction that would result in the loss of many normal fetuses. To reason from abortion practices and make ethical conclusions is dangerous because many other
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wrongs could be justified with this approach, such as exploitative experimentation with the embryo and fetus, especially with fetuses to be aborted. Also, arguments based on abortion practices per se could be extrapolated to justify infanticide or euthanasia. For example, if a mother in Africa has eight children and enough food for two and no help is available or on the way, is she justified in killing six of her children to save two? Two reasons are against it: each child has an equal right to protection, and food, even though scarce, will maintain life. If killing starving children is wrong, and if the moral status of the first-trimester fetus is the same as that of such children, then MFPR and selective termination are prima facie wrong. If the moral status of starving children, however, were held to be no higher than that of the first-trimester fetus and if contemporary abortion practices were a source of moral guidance, then mercy killing might be more justifiable. If the moral status of the first-trimester fetus is such that it is justifiable under some conditions to do an abortion (e.g., maternal health, rape, incest, genetic indications, or social and economic reasons), then moral lines can be more clearly drawn between acceptable and unacceptable cases of abortion, MFPR, selective termination, and euthanasia. MFPR and selective termination can be justified under some circumstances, whereas killing children in extreme danger from starvation cannot. Over the past several years, particularly as a result of the availability of donor eggs, the incidence of women in their late 40s or even 50s having children has increased substantially. 19 In such cases we are seeing a tendency to want to reduce to singleton pregnancies even those starting as twins. We believe that there is increased justification under these circumstances-both medically and socially-that mitigates our general prescription against two to one for "frivolous" reasons. We believe that not wanting to be 70 years old and still have two or more children in high school or college is a reasonable viewpoint, even if one wants to be pregnant at 50. An action by a physician that may or will result in fetal harm or death requires a justification in the light of basic ethical principles. Otherwise physicians are open to moral blame for indifference to many ethical traditions transmitting respect for human life. Along these lines, a basic ethical justification for fetal research, independent of arguments for and against abortion, was developed in the mid-1970s. 32 Also, society's members, including physicians, should help to reduce and prevent the underlying causes of unjustified abortions or casual taking of fetal life. ETHICAL PRINCIPLES AND MULTIPLE PREGNANCIES
Ethics involves the study and recommendation of bodies of guidance-rules, norms, and principles-that help to resolve difficult problems of moral choice such as were faced in these cases of selective termination.
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In these cases, a conflict exists between two duties. If the pregnancy is wanted, there is a duty to benefit the pregnant woman by preserving her pregnancy unless unacceptable physical or mental harm to her would ensue. There is a second moral duty, however, not to destroy human life, including fetal and embryonic life, without ethically justifiable reasons. Are there ethically justifiable reasons for MFPR? What are the basic criteria of ethical reasoning? Having the facts, decision makers must then make choices about the best approach to the problem. Facts per se are not the source of ethics. Human beings choose among various forms of practical guidance to resolve ethical problems. Many physicians claim to approach ethics on a case-by-case basis, as if ethical guidance must be created for each problem. This claim likely rests on unexamined ideas and professional ideology. Systematic studies of what physicians actually do in ethical problems in clinical research and surgery 6 find that they rely on constant sets of ethical beliefs and practices rather than variable case-by-case choices.
SUMMARY
MFPR and selective terminations satisfy the criteria of enabling pregnancies to continue with the least harm and most benefits to all involved. The surviving infants can be saved from certain death (abortion) or higher risks of severe harm and death and of an extended stay in neonatal intensive care (premature delivery). In the hands of trained operators, MFPR and selective termination is, in our opinion, the best means to protect the mother's health and well-being, given it is available and approved by the parents. MFPR and selective termination avoid the trauma of abortion of a wanted pregnancy, enable the parents to achieve the goal of having their own child, and avoid the dangers of delivery of multiple premature infants. There is no doubt that any procedure that involves the death of a fetus will be hotly argued despite the potential for greater good. 15 We acknowledge that it will be impossible to convince those who cannot morally accept the taking of any life regardless of the circumstances. We hope, however, that we have shown there is a place for MFPR and selective termination in a very limited number of circumstances and the ethical probity of MFPR and selective termination as an option in such cases.
References 1. Aberg A, Mitelman F, Cantz M: Cardiac puncture of a fetus with Hurler's disease avoiding abortions of unaffected co-twin. Lancet 2:990, 1978 2. American Fertility Society: Assisted reproductive technology in the United States and Canada: 1993 results generated from the Society for Assisted Reproductive Technology from the American Fertility Society registry. Fertil Steril 64:13-21, 1995
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3. Berkowitz RL, Lynch L, Chitkara U, et al: Selective reduction of multifetal pregnancies in the first trimester. N Engl J Med 318:1043, 1988 4. Berkowitz RL, Lynch L, Lapinski R, et al: First trimester transabdominal multifetal pregnancy reduction: A report of two hundred completed cases. Am J Obstet Gynecol 169:17-21, 1993 5. Berkowitz RL, Lynch L: Selective reduction: An unfortunate misnomer. Obstet Gynecol 75:873-874, 1986 6. Bronshtein M, Timor-Tritsch IE, Rottem S: Early detection of fetal anomalies. In Timor-Tritsch IE, Rottem S (eds): Transvaginal Sonography. New York, Elsevier Science, 1991, pp 327-371 7. Bronsteen RA, Evans MI: Multiple gestation. In Evans MI, Fletcher JC, Dixler AO, et al (eds): Fetal Diagnosis and Therapy: Science, Ethics, and the Law. Philadelphia, JB Lippincott, 1989, pp 242-266 8. Cahill LS, McCormick RA: The Vatican document on bioethics. America 156:246248, 1987 9. Chitkara U, Berkowitz RL, Wilkins IA, et al: Selective second-trimester termination of the anomalous fetus in twin pregnancies. Obstet Gynecol 73:690-694, 1989 10. Drugan A, Johnson MP, Evans MI: Principles of inheritance. In Evans MI (ed): Reproductive Risks and Prenatal Diagnosis. Norwalk, Appleton & Lange, 1992, pp 3-23 11. Dumez Y, Oury JF: Method for first trimester selective abortion in multiple pregnancy. Contrib Gynecol Obstet 15:50, 1986 12. Evans MI, Dommergues M, Timor-Tritsch I, et al: Transabdominal vs. transcervical/ vaginal multifetal pregnancy reduction: International collaborative experience of more than 1000 cases. Am J Obstet Gynecol 170:902-909, 1994 13. Evans MI, Dommergues M, Wapner RI, et al: International collaborative experience of 1789 patients having multifetal pregnancy reduction: A plateauing of risks and outcomes. J Soc Gyn Invest 3:23-26, 1996 14. Evans MI, Dommergues M, Wapner RJ, et al: Efficacy of transabdominal multifetal pregnancy reduction: Collaborative experience among the world's largest centers. Obset Gynecol 82:61-67, 1993 15. Evans MI, Drugan A, Bottoms SF, et al: Attitudes on the ethics of abortion, sex selection, and selective pregnancy termination among health care professionals, ethicists, and clergy likely to encounter such situations. Am J Obstet Gynecol 164:10921099, 1991 16. Evans MI, Fletcher JC, Rodeck C: Ethical problems in multiple gestations: Selective termination. In Evans MI, Fletcher JC, Dixler AO, et al (eds): Fetal Diagnosis and Therapy: Science, Ethics, and the Law. Philadelphia, JB Lippincott, 1989, pp 266-276 17. Evans MI, Fletcher JC, Zador IE, et al: Selective first trimester termination in octuplet and quadruplet pregnancies: Clinical and ethical issues. Obstet Gynecol 71:289-296, 1988 18. Evans MI, Goldberg JD, Dommergues M, et al: Efficacy of second trimester selective termination for fetal abnormalities: International collaborative experience among the world's largest centers. Am J Obstet Gynecol 171:90-94, 1994 19. Evans MI, Hume RF, Polak S, et al: The geriatric gravida: Multifetal pregnancy reduction (MFPR), donor eggs, and aggressive infertility treatments. The Central Association of Obsetricians and Gynecologists, 1996 meeting, in press 20. Evans MI, Johnson MP, Hoffman EP, et al: Fetal tissue biopsies. In James DK, Steer PJ, Weiner CP, et al (eds): High Risk Pregnancy: Management Options. Philadelphia, WB Saunders, 1994, pp 1015-1022 21. Evans MI, Littman L, St Louis L, et al: Evolving patterns of iatrogenic multifetal pregnancy generation: Implications for aggressiveness of infertility treatments. Am J Obstet Gynecol 172:1750-1753, 1995 22. Fletcher JC, Evans MI: Ethics in reproductive genetics. In Chervenak FA, McCullough LB (eds): Ethical Dilemmas in Obstetrics, Clinical Obstetrics and Gynecology, in press 23. Fletcher JC: Ethics in experimental fetal therapy: Is there an early consensus? In Evans MI, Fletcher JC, Dixler AO, et al (eds): Fetal Diagnosis and Therapy: Science, Ethics, and the Law. Philadelphia, JB Lippincott, 1989, pp 438-446
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24. Golbus MS, Cunningham N, Goldberg JD, et al: Selective termination of multiple gestations. Am J Med Genet 31:339-348, 1988 25. Harrison MR, Filly RA, Golbus MS, et al: Fetal treatment 1982. N Engl J Med 307:1651-1652, 1982 26. Hoffman EP, Fishbeck KH, Brown RH, et al: Dystrophin characterization in muscle biopsies from Duchenne and Becker muscular dystrophy patients. N Engl J Med 318:1363-1368, 1988 27. Kerenyi T, Chitkara U: Selective birth in twin pregnancy with discordancy for Down's syndrome. N Engl J Med 304:1525-1527, 1981 28. Luke B, Keith LG: The contribution of singletons, twins, and triplets to low birthweight, infant mortality, and handicap in the United States. J Reprod Med 37:661-666, 1992 29. Macones GA, Schemmer G, Pritts E, et al: Multifetal reduction of triplets to twins improves perinatal outcome. Am J Obstet Gynecol 169:982-986, 1993 30. Martin LS, Ben-Yoseph Y, Ebrahim SAD, et al: Laboratory teclmiques for prenatal diagnosis. In Hillier SG, Kitchener H, Neilson JP (eds): Scientific Essentials of Reproductive Medicine, Philadelphia, WB Saunders, in press 31. McCormick RA: How Brave a New World? Washington DC, Georgetown University Press, 1981 32. National Commission for the Protection of Human Subjects for Biomedical and Behavioral Research: Report and Reocmmendations. Research on the Fetus. Washington DC, US Government Printing Office, DHEW Publication No (05) 76-127, 1975 33. Newman RB, Hamer C, Miller C: Outpatient triplet management: A contemporary revue. Am J Obstet Gynecol 161:547-555, 1989 34. People Magazine, May 26, 1986 35. Quintero RA, Reich H, Puder KS, et al: Brief report: Umbilical cord ligation of an acardiac twin by fetoscopy at 19 weeks of gestation. N Engl J Med 330:469--471, 1994 35a. Quintero RA, Romero R, Reich H, et al: In utero percutaneous umbilical cord ligation in the management of complicated monochorionic multiple gestations. Journal of Ultrasound in Obstetrics and Gynecology, July 1996, in press 36. Reece EA, Hobbins JL, Mahoney MH, et al: Medicine of the Fetus and Mother. Philadelphia, JB Lippincott, 1992, pp 877-878 37. Robertson JA: The right to procreate and in utero fetal therapy. J Leg Med 3:333336, 1982 38. Rodeck CH, Mibashan RS, Abramowicz T, et al: Selective feticide of the affected twin by fetosopic air embolism. Prenat Diag 2:189, 1982 39. Tabsh KM: Transabdominal multifetal pregnancy reduction: Report of 40 cases. Obstet Gynecol 75:739, 1990 40. Tiefel HO: Human in vitro fertilization: A conservative view. JAMA 247:3235, 1982 41. Versmold H: Fetal reduction-not needed. FIGO, 1991, Singapore 42. Wapner RJ, Davis GH, Johnson A: Selective reduction of multifetal pregnancies. Lancet 335:90-93, 1990
Address reprint requests to Mark I. Evans, MD Department of Obstetrics and Gynecology Hutzel Hospital 4707 St. Antoine Detroit, MI 48201
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ETHICAL ISSUES SURROUNDING MULTIFETAL PREGNANCY REDUCTION AND SELECTIVE TERMINATION Mark I. Evans, MD, Mark Paul Johnson, MD, Ruben A. Quintero, MD, and John C. Fletcher, PhD
Public fascination with multiple gestations has been timeless and extends in the media back 60 years to the Dionne quintuplets. Despite the national attention given recently to the Dildy sextuplets and other high-profile successes, the number of sad outcomes continues to rise geometrically. 28 Buried in the fascination about the wonders of multiple births has been an ignorance of the increased reproductive risks inherent in the event. In this article we address some of the ethical issues that arise in multiple gestations in which there is not only a potential conflict between the interests of mother and child but also among fetal siblings. The ethical issues involving multiple gestations can be divided into two clinical situations: (1) multiple gestations in which the number of fetuses in and of itself threatens the ability of the mother to carry them far enough in pregnancy to survive and (2) twin gestations, generally not a result of infertility treatment, in which one fetus is found to be abnormal. 16 After the first few publications, there has been a convention adopted in the literature, that is, the term multifetal pregnancy reduction is used for first-trimester procedures that are done because of fetal number per
From the Departments of Obstetrics and Gynecology (MIE, MJP, RAQ), Molecular Medicine and Genetics, and Pathology (MIE, MPJ), Wayne State University /Hutzel Hospital, Detroit, Michigan; and the Center for Biomedical Ethics, Health Sciences Center, University of Virginia, Charlottesville, Virginia (JCF)
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se. Selective termination is for procedures done predominantly in the second trimester because of a diagnosed fetal abnormality. 5
ETHICAL PRINCIPLES FOR THE PRACTICE OF MEDICINE AND CLINICAL RESEARCH
In our view, the selection of ethical guidance involves two interdependent claims. First, ethical guidance ought to be judged by the consequences of following it. Second, the consequences should be examined in terms of ethical principles widely respected across cultural, philosophical, and religious lines. A set of principles with wide acceptance in biomedical ethics is outlined below. 1. Respect for persons: The duty to respect the self-determination and choices of autonomous persons as well as to protect persons with diminished autonomy (e.g., young children, mentally retarded persons, and those with other mental impairments). 2. Beneficence: The obligation to secure the well-being of persons by acting positively on their behalf and, moreover, to maximize the benefits that can be attained. 3. Nonmaleficence: The obligation to minimize harm to persons and, whenever possible, to remove the causes of harm altogether. 4. Proportionality: The duty, when taking actions involving risks of harm, to balance risks and benefits so that actions have the greatest chance to result in the least harm and the most benefit to persons directly involved. 5. Justice: The obligation to distribute benefits and burdens fairly, to treat equals equally, and to give reasons for differential treatment based on widely accepted criteria for just ways to distribute benefits and burdens. The consequence of the four positions of ethical guidance outlined by us previously for cases of multiple pregnancies can be briefly examined in terms of these five principles:
Position 1: Proceeding to delivery in each case of multiple pregnancy because abortion, multifetal pregnancy reduction, and selective termination are ruled out because they (1) violate self-determination; (2) do not always result in the benefits of completely healthy children; (3) increase long-term harm and suffering to more persons; (4) effect a disproportionate relationship between the risks of premature delivery and the possible benefits of survival, because more persons will be harmed than benefited; and (5) are unfair to parents by burdening them with multiple children, some of whom may be severely impaired or suffer a lengthy, costly process of dying in neonatal intensive care. Position 2: Aborting each multiple pregnancy in hope of starting a new pregnancy (1) restricts choice of selective termination, if the
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procedure is available; (2) brings little benefit to infertile couples who may have great difficulty in a new pregnancy; (3) avoids the long-term harms and complications of premature delivery; (4) avoids many risks and harms but with few benefits; and (5) may be unfair to older couples with a long-term history of infertility. Position 3: Multifetal pregnancy reduction or selective termination in any multiple pregnancy, including twins, on request (1) maximizes self-determination; (2) if effective, benefits parents with at least one surviving child; (3) prevents abortion, reduces harms and complications of premature delivery, reduces or prevents stay in neonatal intensive care, increases incidence of selective termination to a larger number of cases (twins), increases incidence of fetal loss, and when ineffective will increase chances of spontaneous abortion, resulting in no child at all; (4) in light of fewer risks of twin pregnancies compared with multiples of three or more, it disproportionately results in more deaths of normal fetuses who would be less apt to suffer serious harm at delivery; and (5) distributes the benefits of selective termination fairly to all parents in all multiple pregnancies. Position 4: Multifetal pregnancy reduction or selective termination in pregnancies of three or more, but to no fewer than twins (1) restricts self-determination only of parents of normal twins; (2) if effective, benefits parents with two surviving children; (3) prevents abortion, provides protection against total loss of pregnancy in case of one fetal demise, reduces harms and complications of premature delivery; reduces length of stay in neonatal intensive care; prevents extension of selective termination to twins; when ineffective, runs risk of spontaneous abortion; (4) proportionately avoids the worst harms of the most dangerous multiple pregnancies with the most benefit to parents and survivors; and (5) distributes benefits and burdens of selective termination fairly to all parents in the most dangerous multiple pregnancies. ONE ABNORMAL TWIN
The Controversy Prior to the 1980s, the diagnosis of multiple gestations was limited to auscultation of fetal heart beats, crude outlines on radiographs, or limited ultrasound images. 7 With increased diagnostic abilities have come fundamental ethical problems. By far the most common situation involving one abnormal fetus in a multiple gestation is that of one twin who is shown to be either dysmorphic by ultrasound or aneuploid by invasive genetic studies. Since the earliest report by Aberg et aP in 1978, which detailed cardiac puncture for one fetus of twins with Hurler's disease, the subject has always drawn criticism. In 1981, a published report from New York detailed a midtrimester "selective feticide" of a
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Down's syndrome fetus when the other was normal. 27 The authors implied they were forced to attempt the procedure by the mother who threatened to abort both fetuses. Despite the disclaimers, they were criticized for intervening at risk to the normal twin. Sentiment against risking a normal twin continued in many forums through the 1980s. A set of guidelines for fetal therapy was first devised in 1982, which included avoidance of experimental procedures when there was a normal twin. 25 Such guidelines were later modified to permit procedures on an abnormal twin prior to the viability of the normal twin. 22' 27 This view is more consistent with a mother's right to abortion prior to fetal viability than is the restraint of her rights because of the presence of the normal twin. In the 1980s, despite the certainty of many diagnoses of one abnormal twin, very few publications discussed outcomes involved with selective intervention on one fetus. Several potential cases were not published by physicians engaged in such procedures primarily because of the "heat" that would be generated. Although toward the end of the decade, a few physicians risked the heat and began to publish their experiences. Through the mid-1990s, experience increased and included a 1994 collaborative report that proved the safety and efficacy of selective termination.9' is, 24 Loss rates increase with gestational age and vary with techniques. In the mid-1990s, the development of fiberoptic endoscopy has now allowed for selective termination to be performed on monozygotic twins. Umbilical cord ligation can be used for those cases in which use of potassium chloride would be contraindicated because of vascular communication between the twins that would have a high likelihood of damage to the survivor. 35, 35a The incidence of abnormalities is higher in twin pregnancies than in singleton pregnancies. 28 For dizygotic twins the risks are essentially the product of two singleton infants' risks. For example, the risk of a chromosome abnormality at age 35 is about 1 in 200, and the chance that one of a set of twins would be aneuploid is about 1 in 100. Overall, serious disorders are stated at 2% to 3% for singletons, and 4% to 7% for twins. For monozygotic pairs, aneuploidy risk is for "both" or "none," and is identical to the singleton rate. Disorders of laterality and dysmorphology are higher than for singletons or dyzygotic twins. Thus, the chance of facing one abnormal twin is not rare. 10 With increasing diagnostic capabilities, diagnoses of one abnormal twin have moved from the nursery to the second or even first trimester, and it is within the legal rights of the mother to consider termination of the pregnancy. Couples facing the diagnosis of one abnormal twin must confront an excruciating dilemma either to continue the pregnancy with the birth of an abnormal child or abort both fetuses, including the one that is normal. 9 , rn, 24 , 35, 35 a Such a choice transcends the traditional arguments about social abortion (which will not be reviewed here) and even
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those about abortion of a singleton abnormal fetus in an otherwise planned and wanted pregnancy. Ethical Justification for Selective Termination In assessing the ethical perspectives about twin selective termination, various arguments are possible. First, there are the traditional objections to aborting an abnormal pregnancy. The issues involved in selective termination, however, are far more complex because of the presence of the normal twin. In our thinking, the overriding principle is one of trying to preserve the most good and doing the least harm. As with all new techniques, there often is much uncertainty as to the true risks and benefits of such procedures. As with any termination procedure, there are risks of infection, ruptured membranes, and premature delivery, all of which can cause death to the normal twin and pose maternal risks. Furthermore, retention of a dead fetus carries known, although small, risks of coagulopathies, such as disseminated intravascular coagulation (DIC), which could be life threatening. 36 Our collaborative data have not shown a single case of DIC. We believe the risk of DIC is very low and more likely an issue related to much later gestational age spontaneous demises. 18 The approach to ethical reasoning about selective termination must be different from that for abortion per se and, therefore, requires a basic justification. By basic, we mean our reasoning ought not to be derived from precedents that appear to be morally similar but that on examination are not (i.e., arguments for social abortion). It is too simplistic to argue that because society permits abortion of normal fetuses on request in the first trimester, selective termination must fall well within the sphere of permissible actions created by abortion practices. Such reasoning derives an ethical imperative, an ought, from an existing cultural situation that is still ethically controversial. Any intentional action that could, or does, result in fetal harm or death requires a basic ethical justification. Otherwise, physicians would be liable to moral blame for destroying innocent human life without regard for the many ethical traditions that transmit respect for human life. For example, a basic ethical justification for fetal research, independent of arguments for and against abortion, was developed in the mid1970s,32 and even that process has evolved considerably. Both abortion and selective termination cause fetuses to die and are similar in this consequence. But selective termination may enable the surviving fetus to have a better chance for life. Moreover, morally they differ because the intent or aim is different. In abortion, the woman wants to end the whole pregnancy and enlists a physician's help. In selective termination cases, the woman wants to continue a pregnancy, and selective termination allows for the option of doing so without the burden of having an abnormal child as well.
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In practice, selective midtrimester terminations in twins generally have been limited to centers that provide the technical expertise to carry out twin diagnoses, use methods of termination that carry minimal risk to the healthy twin, and manage the continuing pregnancy to avoid the potential dangers of a dead twin in the uterus. 38 Nevertheless, in our view, the option of selective termination meets the criteria of most good-least harm because it preserves the chance for couples to have a healthy child unburdened by abnormalities-the original intent of the pregnancy. An appropriate analogy would be an abortion of an abnormal child followed by a subsequent normal pregnancy. The intent is the same even if the methods are different, and the risk to the normal twin is higher. To be certain, the risks and benefits should be proportionate; however, nearly two decades of experience suggest that selective termination can be a relatively safe procedure. Despite our conclusion of the ethical probity of such procedures, we still hold concerns about the timing of the procedure and indications for it. We reaffirm our beliefs that such procedures only can be considered prior to viability and should not be performed for objectionable reasons, such as sex selection or simply a desire for only one child. The literature on the ethics of sex selection is expanding. 15 We believe that "social" sex selection is inconsistent with an open and equal society and inherently creates inequalities between the sexes. Despite disclaimers to the contrary, in our experience, the vast majority of sex selection requests are for boys. We do believe that sex selection can be appropriate, but, in general, only for sex-linked diseases for which diagnoses are not possible or completely reliable. 30 Until recently, Duchenne muscular dystrophy or hemophilia would have fit that category. 20 , 26 Ironically, in such cases, the selection is against males. The advent of increasingly sophisticated ultrasound has allowed diagnoses of abnormalities much earlier. Diagnosis of dysmorphic anomalies are becoming common in the first trimester. 6 We believe that political pressures against abortion may curtail the rights to abort late in the midtrimester, which will, in turn, further emphasize the need for earlier diagnosis and earlier intervention, making selective termination both technically easier, safer, and more ethically accepted. MULTIPLE PREGNANCIES
With a twin pregnancy, the obstetric outcome in uncomplicated cases is reasonably good. 36 In gestations of larger size, even perfectly normal fetuses may be at considerable risk from prematurity. RATIONALE FOR MULTIFETAL PREGNANCY REDUCTION
The first cases of multifetal pregnancy reduction (MFPR) were published in 1986 to 1988,3 , n, 17 and since then there have been several
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individual center and collaborative series that have allowed for the establishment of now relatively smooth risk curves that show increasing pregnancy loss rates and premature rates that correlate with both starting and finishing numbers. 12• 13, 14• 3 9, 42 When we first confronted the issues of MFPR in 1986, such cases were not reported in the literature, and ethical precedents for such procedures were lacking. We debated as to the best course of action under such circumstances, recognizing that the use of fertility drugs such as human menopausal gonadotropin (HMG, menotropins, and urofollitropin), assisted reproduction techniques such as in vitro fertilization, and gamete intrafallopian transfer in the treatment of infertility carried the risk of inducing multiple gestations. Despite reduced occurrences and better understanding of the mechanisms of fertility drugs over the last decade, the possibility for inducing multiple gestations still exists. 2 In fact, the occurrences of multifetal pregnancies continues to rise, not fall. Explanations for the rise are multifaceted but include a tendency for less caution in their use because of the knowledge that MFPR can be used to treat a multifetal pregnancy. 21 With Pergonal, and now more commonly Metrodin, twin pregnancies occur in about 10% to 30% of patients. Higher numbers of conceptuses are seen in about 1% to 30% of patients. In some centers, the numbers are actually much higher. 2• 21 The obstetric outcome of triplets (or more) is known to be significantly poorer than with singleton or even twin pregnancies. 36 The ability to carry four or more fetuses to viability is very problematic. Only two successful sextuplet pregnancies are known in the United States. A successful gestation of octuplets has never been substantiated. Thus, a couple faced with the prospect of an octuplet pregnancy has a very serious dilemma. With no intervention, they will lose all the fetuses. In 1985, a case of septuplet births in California was highlighted in the national media. 34 Ultimately, only three infants survived. All have mental and physical handicaps, and malpractice litigation was successfully brought. In the case of multifetal pregnancies, one management alternative is to abort the entire pregnancy and have the couple try to conceive again. Because another conception cannot be guaranteed, another more attractive possibility is to reduce the number of fetuses with the hope of increasing the probability of a good outcome for at least some of them. Selective termination in multiple pregnancy, usually of one abnormal twin, in the midtrimester had been reported in the literature on a few occasions in the 1980s,24• 27 • 38 but literature on the ethics of selective termination was essentially nonexistent when we first had to make hard choices. In our view, MFPR in these patients was the only alternative available to achieve a reasonable chance of successful pregnancy outcome. In all instances, the couples understood the potential risks of the procedure and its experimental nature. Informed consent was obtained after thorough counseling. We stressed that any attempt to reduce the number of fetuses would be experimental and could result in miscarriage of the
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pregnancy. Infection, bleeding, and other unknown risks were possible. If successful, the attempt could theoretically result in damage to the remaining fetuses. All felt strongly, however, that because of their infertility history their current pregnancy might be their only or last opportunity to have children. As of 1996 about 2000 cases have been reported. The rate of subsequent live twin births appears to be about 88%. The medical details have been elucidated in great detail elsewhere and will not be repeated here. 13 Ethical Justification for Multifetal Pregnancy Reduction
Although there still is some debate, more than a decade of clinical experiences with good outcomes has demonstrated the technical feasibility and safety of first-trimester MFPR, even in grand multiple pregnancies that has improved outcomes compared with nonreduced conceptions.29, 33, 41 In the mid-1980s, we recognized, however, that MFPR was an inherently controversial topic and would require a thorough, prior, ethical reflection. 17 First we recognized, as with selective termination for one abnormal twin, that a basic ethical justification was required. Second, we recognized that our reasoning should not be limited to cases of multiple pregnancies after iatrogenic effects of infertility treatment. We must be willing to reason similarly in cases in which the pregnant woman's health and the survival of any infants are endangered by a multifetal pregnancy, regardless of cause. MFPR in a multifetal pregnancy raises ethical issues different from those for one abnormal twin. In multifetal pregnancies, childbearing has been endangered by multiple fetuses, all of which may be normal. The patient may or may not regard this pregnancy as her final chance to have a child. She enlists a physician's help to facilitate the continuation of the pregnancy. Termination of one genetically abnormal twin is, in some respects, similar to cases of termination in multifetal pregnancies. In both instances the woman desires to complete the pregnancy. The harms and dangers of severe genetic disease and prematurity of multiple newborns can be similar in expected morbidity and mortality, the major difference being between a diagnosis of a congenital anomaly and a history of infertility. The chance of a subsequent normal pregnancy after abortion for genetic disorders is more likely than in cases that involve infertility. Despite generally good anecdotal outcomes in twin cases, we were reluctant to rely on midtrimester precedents because, at the time, the outcomes of midtrirnester terminations of an affected twin and selective birth of a healthy surviving twin had not been studied adequately. Without reliable knowledge of long-term beneficial consequences, however, the value of the precedent was dubious, even though ethical reasoning about the two problems might be similar.
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In our first cases, we began from the premise that a pregnancy that has been finally achieved, particularly in the face of previous infertility, is a basic human good worth preserving. To what lengths is it ethically acceptable to go to achieve and preserve a pregnancy? Some, such as Tiefel, 40 or the 1987 Vatican document,8 challenged the ethical acceptability of methods of fertility treatment (including in vitro fertilization). Tiefel viewed the anxiety that brought these women into fertility treatment as caused by "dehumanizing socialization" in which "having a child is the ultimate need and in which infertility is seen as personal failure and a sign of worthlessness." 40 Had they only come to terms with these (supposed) feelings, they would never have found themselves in this situation. We doubt whether "dehumanizing socialization" is the basic cause of the despair of infertile couples. Furthermore, we find it impossible to accept the Vatican's position that attempts to treat infertility involving anything but marital coitus are immoral. There are straightforward biologic causes of infertility that can be remedied by a number of methods, including in vitro fertilization. We do not accept the posture of passive resignation in the face of biologic problems of infertility. If something can be done about infertility, we believe there is a strong moral obligation to do it, because infertility is a significant form of human suffering. Nonetheless, we do agree with the Vatican report that one is not ethically permitted to do simply anything to achieve or preserve pregnancy. How should the human need for a child be evaluated on ethical grounds? Is there a moral duty to have a child of one's own? Is there a right to have a child, as some parents have argued? Tiefel40 makes a case, with which we agree, that there is no moral or legal right to have children in a positive or enabling sense. Society is not obligated to meet an entitlement of a child in the same sense that rights to life, freedom of speech, and protection from enemies are entitlements. On the other hand, the Vatican notwithstanding, there is a negative duty not to be restrained or prevented from childbearing. In our society we believe that right is guarded by the legal right to privacy. 40 Once having obtained help in becoming pregnant, women in this situation deserve help to achieve their aim, as long as the means used are proportionate to the ends sought. A proportionate relationship between means and ends entails two criteria. First, there must be no other way to achieve the end, in this instance, preservation of a viable and desired pregnancy. Second, in choosing among the means available the chosen one must be the one that results in least harm and results in the most good for all involved. 31 In our cases of multifetal pregnancies, there were only three real alternatives. We recognized that any of the three could lead to harm. First, the pregnancy could be electively aborted, causing death to all fetuses and possibly not succeeded by any future pregnancy. A second possibility was to do nothing and risk the birth of multiple premature infants, many of whom might die or be significantly impaired. 2 In such a choice, there was also a very high likelihood of a spontaneous loss of
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the entire pregnancy. The third alternative was MFPR. Negative outcomes included the deaths of multiple fetuses and potential harm to the remaining twins; however, in these circumstances, we concluded, and our patients agreed, that MFPR held out more potential benefits than the other two alternatives. MULTIFETAL PREGNANCY REDUCTION AND ABORTION RIGHTS
Views on the morality of abortion are obviously relevant to choices in multiple pregnancy because both raise the question of whether deliberate killing of a fetus is justified. We argue in the following discussion that abortion practices per se are not sufficient to justify MFPR in multifetal pregnancies; however, the structure of an ethical argument concluding that some abortions are morally justified is highly similar to the argument for MFPR. A typology of responses to MFPR can be constructed by inference from the literature on the morality of abortion. The actual views of particular authors, and the variation within religious and philosophical traditions, are far richer than the truncated typology presented here.
Position 1: Any human act deliberately taken to destroy human embryonic or fetal life ought to be morally condemned. 16 This position rules out MFPR in any case. Position 2: An exception to position 1 is permitted for abortion only to save the life of a pregnant woman, as in cases of uterine cancer or ectopic pregnancy. Whether this position would favor or reject MFPR in multifetal pregnancies turns mainly on the empiric questions of the degree of threat to the life of the mother of continuation of pregnancy with premature delivery. Position 3: Although abortion is a serious moral problem, some abortions are permissible for ethically valid reasons. 31 It follows that some cases of MFPR in multifetal pregnancies are permitted, especially to avoid predictable harms to the pregnancy woman, to remaining fetuses, or to survivors. Because this position attempts to draw lines between morally valid and invalid reasons for abortion, it follows that a compatible position on MFPR would search for some moral lines between permissible and impermissible cases. Position 4: A position for abortion on request for any pregnancy at any stage32 favors a stance on MFPR that permits termination of any number of fetuses, including twins, to any remainder. The main concern would be the health and well-being of the pregnant woman. Positions on the morality of abortion mainly rest on three sorts of premises: (1) the moral status of the human embryo and fetus; (2) the relative strengths of countervailing moral claims made in the interests
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of the pregnant woman, the family, or society; and (3) the determination of how much moral weight should be given to whether the pregnancy is wanted by the woman. All positions on abortion tend to accept the premise that each pregnancy should be wanted and planned with medical advice, if available. Position 1 accords moral status of "personhood" to embryos from fertilization and requires that society protect every fetus equally in every pregnancy. This position allows for no countervailing moral claims and accords little moral significance to the unwantedness of the pregnancy. Position 2 gives the same high moral status to the fetus throughout pregnancy and allows only one higher moral claim to override when continuation of pregnancy actually threatens the woman's life. An important variation of this position extends slightly the range of countervailing moral claims and bends to permit abortions in "tragic" circumstances resulting from rape or incest and for diagnosis of genetic disorders that are totally incompatible with physical or mental life. Position 3 involves a "graded" 37 approach to the moral status of the fetus that resists indifference at any stage but gradually raises society's protection of the nascent human being with stages of development. This view is more likely to recognize countervailing moral claims, especially in the first trimester of pregnancy. It gives an important, but not absolutely overriding, weight to the wantedness of pregnancy. For example, some unplanned pregnancies that occur after failure of contraception would not result in substantial harm to the woman, family, or society. Abortion under such circumstances, in this view, is morally objectionable because the harm done by abortion is worse than ·the supposed but empty harm that would be prevented by the abortion. This view is probably dominant in the sense of prevailing in practice and public policy in this society. "Dominance" is used here as a descriptive term, and not a moral judgment. Position 4 is skeptical of claims for significant moral status or the interests of the fetus until birth, and even then claims to personhood and equal protection might be overridable in certain cases. "Wantedness" of the pregnancy is treated as a matter of moral supremacy in any context. Do abortion practices justify MFPR in multiple pregnancy? Some might argue "if society permits abortion of a normal, unwanted fetus, what is so wrong about terminating eight, four, or three to two, or two to one in a wanted pregnancy?" 37 It is true that if society proscribed abortions, MFPR also would be illegal.37 What is legally permitted, however, is not always ethically sound. Using abortion practices as sole sources of justification of MFPR is inadequate given the premises of the third position, because some abortions are done for reasons that cannot be justified by ethical principles and that violate respect for fetal life (e.g., abortions for gender choice). To argue from the premise of what society permits in abortion would also encourage MFPR in cases of normal twins, a direction that would result in the loss of many normal fetuses. To reason from abortion practices and make ethical conclusions is dangerous because many other
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wrongs could be justified with this approach, such as exploitative experimentation with the embryo and fetus, especially with fetuses to be aborted. Also, arguments based on abortion practices per se could be extrapolated to justify infanticide or euthanasia. For example, if a mother in Africa has eight children and enough food for two and no help is available or on the way, is she justified in killing six of her children to save two? Two reasons are against it: each child has an equal right to protection, and food, even though scarce, will maintain life. If killing starving children is wrong, and if the moral status of the first-trimester fetus is the same as that of such children, then MFPR and selective termination are prima facie wrong. If the moral status of starving children, however, were held to be no higher than that of the first-trimester fetus and if contemporary abortion practices were a source of moral guidance, then mercy killing might be more justifiable. If the moral status of the first-trimester fetus is such that it is justifiable under some conditions to do an abortion (e.g., maternal health, rape, incest, genetic indications, or social and economic reasons), then moral lines can be more clearly drawn between acceptable and unacceptable cases of abortion, MFPR, selective termination, and euthanasia. MFPR and selective termination can be justified under some circumstances, whereas killing children in extreme danger from starvation cannot. Over the past several years, particularly as a result of the availability of donor eggs, the incidence of women in their late 40s or even 50s having children has increased substantially. 19 In such cases we are seeing a tendency to want to reduce to singleton pregnancies even those starting as twins. We believe that there is increased justification under these circumstances-both medically and socially-that mitigates our general prescription against two to one for "frivolous" reasons. We believe that not wanting to be 70 years old and still have two or more children in high school or college is a reasonable viewpoint, even if one wants to be pregnant at 50. An action by a physician that may or will result in fetal harm or death requires a justification in the light of basic ethical principles. Otherwise physicians are open to moral blame for indifference to many ethical traditions transmitting respect for human life. Along these lines, a basic ethical justification for fetal research, independent of arguments for and against abortion, was developed in the mid-1970s. 32 Also, society's members, including physicians, should help to reduce and prevent the underlying causes of unjustified abortions or casual taking of fetal life. ETHICAL PRINCIPLES AND MULTIPLE PREGNANCIES
Ethics involves the study and recommendation of bodies of guidance-rules, norms, and principles-that help to resolve difficult problems of moral choice such as were faced in these cases of selective termination.
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In these cases, a conflict exists between two duties. If the pregnancy is wanted, there is a duty to benefit the pregnant woman by preserving her pregnancy unless unacceptable physical or mental harm to her would ensue. There is a second moral duty, however, not to destroy human life, including fetal and embryonic life, without ethically justifiable reasons. Are there ethically justifiable reasons for MFPR? What are the basic criteria of ethical reasoning? Having the facts, decision makers must then make choices about the best approach to the problem. Facts per se are not the source of ethics. Human beings choose among various forms of practical guidance to resolve ethical problems. Many physicians claim to approach ethics on a case-by-case basis, as if ethical guidance must be created for each problem. This claim likely rests on unexamined ideas and professional ideology. Systematic studies of what physicians actually do in ethical problems in clinical research and surgery 6 find that they rely on constant sets of ethical beliefs and practices rather than variable case-by-case choices.
SUMMARY
MFPR and selective terminations satisfy the criteria of enabling pregnancies to continue with the least harm and most benefits to all involved. The surviving infants can be saved from certain death (abortion) or higher risks of severe harm and death and of an extended stay in neonatal intensive care (premature delivery). In the hands of trained operators, MFPR and selective termination is, in our opinion, the best means to protect the mother's health and well-being, given it is available and approved by the parents. MFPR and selective termination avoid the trauma of abortion of a wanted pregnancy, enable the parents to achieve the goal of having their own child, and avoid the dangers of delivery of multiple premature infants. There is no doubt that any procedure that involves the death of a fetus will be hotly argued despite the potential for greater good. 15 We acknowledge that it will be impossible to convince those who cannot morally accept the taking of any life regardless of the circumstances. We hope, however, that we have shown there is a place for MFPR and selective termination in a very limited number of circumstances and the ethical probity of MFPR and selective termination as an option in such cases.
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3. Berkowitz RL, Lynch L, Chitkara U, et al: Selective reduction of multifetal pregnancies in the first trimester. N Engl J Med 318:1043, 1988 4. Berkowitz RL, Lynch L, Lapinski R, et al: First trimester transabdominal multifetal pregnancy reduction: A report of two hundred completed cases. Am J Obstet Gynecol 169:17-21, 1993 5. Berkowitz RL, Lynch L: Selective reduction: An unfortunate misnomer. Obstet Gynecol 75:873-874, 1986 6. Bronshtein M, Timor-Tritsch IE, Rottem S: Early detection of fetal anomalies. In Timor-Tritsch IE, Rottem S (eds): Transvaginal Sonography. New York, Elsevier Science, 1991, pp 327-371 7. Bronsteen RA, Evans MI: Multiple gestation. In Evans MI, Fletcher JC, Dixler AO, et al (eds): Fetal Diagnosis and Therapy: Science, Ethics, and the Law. Philadelphia, JB Lippincott, 1989, pp 242-266 8. Cahill LS, McCormick RA: The Vatican document on bioethics. America 156:246248, 1987 9. Chitkara U, Berkowitz RL, Wilkins IA, et al: Selective second-trimester termination of the anomalous fetus in twin pregnancies. Obstet Gynecol 73:690-694, 1989 10. Drugan A, Johnson MP, Evans MI: Principles of inheritance. In Evans MI (ed): Reproductive Risks and Prenatal Diagnosis. Norwalk, Appleton & Lange, 1992, pp 3-23 11. Dumez Y, Oury JF: Method for first trimester selective abortion in multiple pregnancy. Contrib Gynecol Obstet 15:50, 1986 12. Evans MI, Dommergues M, Timor-Tritsch I, et al: Transabdominal vs. transcervical/ vaginal multifetal pregnancy reduction: International collaborative experience of more than 1000 cases. Am J Obstet Gynecol 170:902-909, 1994 13. Evans MI, Dommergues M, Wapner RI, et al: International collaborative experience of 1789 patients having multifetal pregnancy reduction: A plateauing of risks and outcomes. J Soc Gyn Invest 3:23-26, 1996 14. Evans MI, Dommergues M, Wapner RJ, et al: Efficacy of transabdominal multifetal pregnancy reduction: Collaborative experience among the world's largest centers. Obset Gynecol 82:61-67, 1993 15. Evans MI, Drugan A, Bottoms SF, et al: Attitudes on the ethics of abortion, sex selection, and selective pregnancy termination among health care professionals, ethicists, and clergy likely to encounter such situations. Am J Obstet Gynecol 164:10921099, 1991 16. Evans MI, Fletcher JC, Rodeck C: Ethical problems in multiple gestations: Selective termination. In Evans MI, Fletcher JC, Dixler AO, et al (eds): Fetal Diagnosis and Therapy: Science, Ethics, and the Law. Philadelphia, JB Lippincott, 1989, pp 266-276 17. Evans MI, Fletcher JC, Zador IE, et al: Selective first trimester termination in octuplet and quadruplet pregnancies: Clinical and ethical issues. Obstet Gynecol 71:289-296, 1988 18. Evans MI, Goldberg JD, Dommergues M, et al: Efficacy of second trimester selective termination for fetal abnormalities: International collaborative experience among the world's largest centers. Am J Obstet Gynecol 171:90-94, 1994 19. Evans MI, Hume RF, Polak S, et al: The geriatric gravida: Multifetal pregnancy reduction (MFPR), donor eggs, and aggressive infertility treatments. The Central Association of Obsetricians and Gynecologists, 1996 meeting, in press 20. Evans MI, Johnson MP, Hoffman EP, et al: Fetal tissue biopsies. In James DK, Steer PJ, Weiner CP, et al (eds): High Risk Pregnancy: Management Options. Philadelphia, WB Saunders, 1994, pp 1015-1022 21. Evans MI, Littman L, St Louis L, et al: Evolving patterns of iatrogenic multifetal pregnancy generation: Implications for aggressiveness of infertility treatments. Am J Obstet Gynecol 172:1750-1753, 1995 22. Fletcher JC, Evans MI: Ethics in reproductive genetics. In Chervenak FA, McCullough LB (eds): Ethical Dilemmas in Obstetrics, Clinical Obstetrics and Gynecology, in press 23. Fletcher JC: Ethics in experimental fetal therapy: Is there an early consensus? In Evans MI, Fletcher JC, Dixler AO, et al (eds): Fetal Diagnosis and Therapy: Science, Ethics, and the Law. Philadelphia, JB Lippincott, 1989, pp 438-446
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Address reprint requests to Mark I. Evans, MD Department of Obstetrics and Gynecology Hutzel Hospital 4707 St. Antoine Detroit, MI 48201