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Aggressive intrapartum management of lethal fetal anomalies: Beyond fetal beneficence
Aggressive Intrapartum Management of Lethal Fetal Anomalies: Beyond Fetal Beneficence JOSEPH A. SPINNATO, MD, VERNON D. COOK, MD, CURTIS R. COOK, MD, AND DEWARD H. VOSS, MD Objective: To evaluate management recommendations from the current literature for patients whose fetuses are certain to have lethal anomalies or absent (or virtually absent) cognitive function. These recommendations include termination of pregnancy or, for cases in the third trimester, nonaggressive intrapartum management, avoiding cesarean delivery for fetal indications. Methods: We report our experience with several patients who voiced opposition to nonaggressive intrapartum care and present a rationale for selectively aggressive, intraparturn management for some of these cases. Results: Four women whose fetuses had lethal anomalies requested aggressive intrapartum management. For three of the four, standard aggressive management of labor resulted in vaginal delivery of live-born infants who died shortly thereafter. The patients found comfort in the live births. The fourth patient accepted a recommendation to avoid fetal monitoring during labor, and the fetus was stillborn. This patient found the intrapartum experience to be very stressful. Conclusion: When a patient's desire to avoid an intrapartum stillbirth is strong enough that substantial psychological harm might result from one, the physician's beneficencebased obligation to her and respect for maternal autonomy justify selectively aggressive intrapartum therapy, even if no beneficence-based obligation to the fetus exists. (Obstet Gynecol 1995;85:89-92)
The ability to diagnose lethal fetal conditions accurately has expanded the available m a n a g e m e n t options for patients and practitioners. In addressing such cases, the recent literature 1-3 has focused primarily on the following choices: 1) termination of pregnancy, particularly w h e n early diagnosis is made, 2) nonaggressive management that withholds certain therapeutic options, such as cesarean delivery for fetal distress, and 3) aggressive m a n a g e m e n t that is offered when the prediction of death or absent cognitive function is uncerFrom the Department of Obstetrics and Gynecology, University of Louisville School of Medicine, Louisville, Kentucky.
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tain. In general, the justification for these m a n a g e m e n t options is made by the absence or virtual absence of a beneficence-based obligation to the affected fetus, a beneficence-based obligation to the mother, and respect for maternal autonomy. Stated simply, m a n y (if not most) patients and physicians choose either to terminate a pregnancy before the third trimester and avoid exposure of the patient to unnecessary risks, or in cases of continuing pregnancies, to not use therapeutic modalities that increase maternal risk (such as cesarean delivery for fetal indications) without a reasonable hope of fetal survival or cognitive function. In some institutions, prenatal detection of lethal anomalies has dramatically reduced inadvertent aggressive management of these fetuses. 4 We have encountered several patients whose fetuses have had lethal anomalies; despite thorough counseling and a seemingly clear understanding of their fetuses' prognoses, the patients were not only unwilling to terminate their pregnancies, but also adamantly opposed to nonaggressive intrapartum management that might result in stillbirth. This report describes four cases that illustrate the selective use of aggressive and incrementally aggressive therapies for such patients and presents a rationale for their use.
Case Reports The first patient was a 23-year-old primigravida, first seen for a second-opinion perinatal consultation. Marked oligohy~ dramnios was first noted 2 weeks earlier. Targeted ultrasonography performed elsewhere confirmed the findings and noted an enlarged fetal bladder and bilateral hydronephrosis. Percutaneous sampling of the fluid from the smaller kidney demonstrated values for osmolality, chloride, and sodium that were similar to those in plasma. The fetal karyotype was normal. The ultrasonographic findings were confirmed in our unit, and the fetal chest circumference was noted to be less than the tenth percentile for gestafional age, consistent with probable long-standing oligohydramnios. Nonaggressive
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management was recommended. The couple initially insisted on aggressive management including cesarean delivery for fetal benefit, if necessary. Follow-up evaluations over the next 5 weeks consistently demonstrated oligohydramnios and poor fetal chest growth. The patient presented at 35 weeks' gestation in active labor with a frank breech presentation and no fetal heart rate (FHR) abnormalities. Management options were discussed, and cesarean delivery was discouraged. We suggested that the parents spend time with the newborn in the delivery room rather than choose aggressive neonatal resuscitation. They agreed to avoid cesarean, but requested that intrapartum techniques discussed previously, such as amnioinfusion, be used to lessen the chance of intrapartum death and that neonatologists be present for the delivery. Neonatologists counseled the couple and agreed to provide initially aggressive, postnatal management until lethality was confirmed. Fetal distress was not observed, and delivery was accomplished by the assisted breech extraction of a male infant with Apgar scores of 2 and 4 at I and 5 minutes, respectively. Umbilical vein acidemia was absent. Despite maximal ventilatory support, the infant died at 7 hours of age, with the clinical diagnosis of pulmonary hypoplasia. The couple later volunteered that they wished they had kept the baby in the delivery room with them, believing that the neonate suffered pain in the nursery. The second patient was a 22-year-old primigravida who was referred at 26 weeks' gestation when an office sonogram indicated a possible omphalocele. This finding was confirmed, and a ventricular septal defect was also noted. Amniocentesis was performed, and the karyotype indicated trisomy 18. Termination of the pregnancy and nonaggressive management were offered to the patient. She refused the former and, through most of the remainder of pregnancy, she requested aggressive intrapartum therapy, including cesarean delivery for fetal indications. Eventually, she agreed to aggressive intrapartum management that would stop short of cesarean delivery for fetal distress. Continuous fetal monitoring identified repetitive decelerations late in the active phase of labor. Standard measures, including repositioning and oxygen therapy, failed to ameliorate the tracing abnormalities. Forceps were used to shorten the second stage, and a 2195-g male infant was delivered, with Apgar scores of 7 and 8 at 1 and 5 minutes, respectively. The newborn's condition was initially stable in the nursery, and a decision was made to close the leaking omphalocele and insert a gastrostomy tube. The baby was discharged and died at home 2 weeks later from apparent cardiac failure. The mother's grief response was pronounced, including substance abuse and suicidal behavior that necessitated a brief psychiatric admission. Subsequently, she expressed particular gratitude for our intrapartum care. The third patient was a 19-year-old primigravida, referred at 29 weeks' gestation. Previous ultrasonographic examinations at 24 and 28 weeks' gestation demonstrated oligohydramnios, probable fetal growth retardation, and nonvisualization of the fetal kidneys and bladder. Evaluation in our unit confirmed these findings. After diagnostic amnioinfusion, rupture of membranes occurred. Options were discussed at length on several occasions. The couple initially desired fully
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aggressive management, including possible expectant management of the ruptured membranes. Over a 48-hour period, they came to accept and understand the poor prognosis of the fetus, agreeing to induction of labor with oxytocin and continuous monitoring. They also agreed to forego a cesarean for fetal indications. Variable decelerations recurred. Oxygen therapy, repositioning, and discontinuance of the oxytocin were followed by amnioinfusion, which reduced the severity and frequency of the decelerations. Assisted breech extraction of a 970-g male infant with Apgar scores of 3 and 5 at 1 and 5 minutes, respectively, was accomplished. Mild umbilical artery respiratory acidemia (pH 7.185, base excess -3.8) was noted. The baby was intubated and placed on a ventilator until a confirmatory postnatal sonogram was performed. Ventilatory support was removed, and the infant died within 3 hours of birth. The fourth patient was a 19-year-old primigravida, referred at 25 weeks' gestation with marked oligohydramnios, fetal thoracic circumference less than the fifth percentile, an enlarged heart with hypoplasia of the right ventricle, renal agenesis, and size less than the tenth percentile. Umbilical vein sampling revealed a normal male karyotype. Under the care of the referring physician, she entered spontaneous labor at 34 weeks' gestation. Variable FHR decelerations were present. The patient expressed a desire to deliver a live-born infant, but declined cesarean delivery for fetal distress. Amnioinfusion was not offered, and the fetus was monitored only intermittently. During labor, the patient told us that she "kept her fingers crossed" that the baby would be born alive. Thirty minutes before delivery, the fetal heart sounds were lost. The patient delivered a 1022-g stillborn male. She said it was very difficult to view her lifeless child. An autopsy confirmed the ultrasonographic findings.
Discussion Chervenak and McCullough 3 developed a "comprehensive m a n a g e m e n t s t r a t e g y " a n d d e s c r i b e d three categories of clinical situations b a s e d on the p r o b a b i l i t y of a n t e n a t a l d i a g n o s i s a n d the p r o b a b i l i t y of d e a t h or a b s e n t cognitive d e v e l o p m e n t a l capacity. T h e y stated that w h e n there are no b e n e f i c e n c e - b a s e d o b l i g a t i o n s to the fetus (category A, eg, a n e n c e p h a l y ) , o n l y t e r m i n a t i o n of p r e g n a n c y o r n o n a g g r e s s i v e m a n a g e m e n t s h o u l d be offered. W h e n there are m i n i m a l beneficenceb a s e d o b l i g a t i o n s to the fetus (category B, eg, t h a n a t o p h o r i c d y s p l a s i a ) , o n l y a g g r e s s i v e or n o n a g g r e s s i v e m a n a g e m e n t s h o u l d be offered. C a t e g o r y C a n o m a l i e s , those w h o s e d i a g n o s t i c accuracy or lethality or absence of cognitive function is uncertain, w e r e r e c o m m e n d e d for a g g r e s s i v e m a n a g e m e n t . W i t h i m p r o v e m e n t s in u l t r a s o n o g r a p h i c e q u i p m e n t a n d i n c r e a s i n g clinical experience, p r e n a t a l d i a g n o s i s has b e c o m e m o r e accurate in its p r e d i c t i o n of l e t h a l i t y or a b s e n t cognitive function. For m a n y institutions, the list of d i a g n o s e s that fulfill C h e r v e n a k a n d McCul-
Obstetrics & Gynecology
lough's category A criteria includes renal agenesis, acrania, exencephaly, severe forms of iniencephaly, allobar holoprosencephaly, trisomies 13 and 18 (particularly when major cranial or cardiac defects are present), parenchymal and obstructive renal disease with long-standing oligohydramnios, and possibly others. 5 Although some would probably object to the inclusion of these diagnoses in category A, few would disagree that counseling against aggressive management occurs commonly, even earnestly, when these diagnoses are made. Kirkinen et al 4 described cesarean delivery as inappropriate, useless, and associated with unnecessary maternal morbidity and extra cost when a fatal anomaly is present. Little guidance is available from the literature regarding patient requests for aggressive management, including cesarean delivery, when the fetal condition is lethal. Hassed et al 6 reported 130 pregnancies diagnosed with lethal conditions at or before 24 weeks' gestation. Termination of pregnancy was chosen by 67% (n = 87) and continuation of pregnancy was elected by 33% (n = 43). Postulated reasons for continuing the pregnancies included the alleviation of guilt and "to allow Mother Nature to take its course." The fetuses from these pregnancies were less likely to have an autopsy than the aborted fetuses. The authors speculated that these mothers had become more attached to their fetuses. Zeanah et al 7 evaluated 23 women 2 months after pregnancy termination for fetal anomalies and compared them with matched controls who had had a spontaneous loss. Seventeen percent of the patients in the termination group experienced major depression and 22% sought psychiatric help. The authors concluded that the grief response was similar to that of the women experiencing spontaneous loss. With regard to performing a medically unnecessary cesarean, Johnson et al s polled 112 obstetricians and asked them to consider 19 hypothetical clinical situations with varying degrees of justification for patientrequested cesarean delivery. As many as 38% of the respondents said they would agree to perform a cesarean at the request of a patient for various nonindications. The factors cited were patient autonomy and the maxim, "do no harm." Our experience with these four patients and an evaluation of pertinent ethical principles prompts us to propose that aggressive intrapartum therapy, which attempts to avoid fetal death, is appropriate for selected patients whose fetuses definitely have lethal anomalies. We do not view this as an issue of fetal beneficence and agree that no such obligations exist in these cases. Instead, our position stems from our beneficence-based
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obligation to the mother and from respect for maternal autonomy. The principle of nonmaleficence, understood in medicine as primum non nocere (first do no harm), is a central part of our societal morality. Although patient motives are heterogeneous, "do not kill" and "do not inflict harm" are driving forces in the opposition to terminating a pregnancy and are likely part of the desire to avoid intrapartum stillbirth. Indeed, for some, the argument centers around the distinction between killing and letting die. In medicine, active euthanasia (ie, killing) is generally rejected, but passive euthanasia (ie, letting die), defined as "to withhold or withdraw extraordinary means to prolong the life of the body when there is irrefutable evidence that biologic death is imminent," is seen as being morally justified. 9 In fact, this is the very argument that justifies nonaggressive management. However, protecting a fetus from dying in labor is not considered extraordinary means to some. Some patients (and possibly some physicians) understand ordinary means as "usual and customary" and extraordinary means as "unusual or a departure from custom. ''1° More correctly defined, "extraordinary means are all medicines, treatments and operations, which cannot be obtained or used without excessive expense, pain, or other inconvenience, or which, if used, would not offer a reasonable hope of benefit. ''11 Using this definition, our ordinary custom, to protect the intrapartum fetus from dying using intrapartum resuscitation and possibly cesarean delivery, becomes extraordinary care and is not morally required in a hopeless situation. The correctness of this ethical argument may not be accepted by some patients. The hope of patients for their unborn children is a powerful virtue that should not be set aside thoughtlessly. Their questioning of a terminal prognosis should always be understood. It should be noted that intrapartum resuscitation measures short of cesarean (such as oxygen therapy, fetal monitoring, amnioinfusion, elimination of hyperstimulation by tocolysis, or forceps delivery) involve minimal expense and little, if any, morbidity. They may not be so extraordinary to justify withholding them against maternal wishes. Their hidden expense occurs when intrapartum death is avoided or when the neonatologist is unwilling to accept prenatal evidence of lethality and postnatal expenses are incurred. These expenses may be minimized by careful antenatal involvement of neonatologists in the decision-making process. When rapid death is expected, as with renal agenesis, we have successfully encouraged couples to keep the infant in the birthing room, thus avoiding any added nursery costs or interventions. Our first three cases demonstrate how dialogue with
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91
the p a r e n t s a n d their a g r e e m e n t w i t h a g g r e s s i v e intrap a r t u m m a n a g e m e n t s h o r t of cesarean d e l i v e r y a l l o w e d t h e m to a v o i d a b a n d o n i n g their child d u r i n g labor. W e s p e c u l a t e that as a c o n s e q u e n c e of o u r counseling, the p a t i e n t s c a m e to u n d e r s t a n d that cesarean is i n d e e d a n e x t r a o r d i n a r y m e a n s w h e n the fetus is e x p e c t e d to die. The f o u r t h case d e m o n s t r a t e s h o w different a p a t i e n t ' s experience is w h e n c o n t i n u o u s m o n i t o r i n g a n d amn i o i n f u s i o n are w i t h h e l d . As p o i n t e d o u t b y Johnson et al, 8 obstetricians h a v e at times e x p r e s s e d a n d d e m o n s t r a t e d a w i l l i n g n e s s to p e r f o r m an obstetrically u n n e c e s s a r y cesarean d e l i v e r y for the p s y c h o l o g i c a l benefit of a patient. A n obstetrician's fear of litigation m a y also be a c o m p e l l i n g reason. W e p e r c e i v e such decisions as d r i v e n b y m a t e r n a l beneficence. R e a s o n a b l e a t t e m p t s (that is, efforts that r e s p e c t m a t e r n a l a u t o n o m y a n d d o not use i n t i m i d a tion) to d i s s u a d e the p a t i e n t f r o m cesarean d e l i v e r y b e c a u s e of the i n c r e a s e d risks are a p p r o p r i a t e . H o w ever, if there is r e a s o n a b l e e v i d e n c e or s u s p i c i o n that s u b s t a n t i a l p s y c h o l o g i c a l h a r m m i g h t be d o n e to the p a t i e n t w h o s e fetus dies d u r i n g labor, the e x p e n s e a n d risks of a g g r e s s i v e i n t r a p a r t u m m a n a g e m e n t can be seen as m a t e r n a l l y beneficent, w i t h o u t violating the p r i n c i p l e of non-maleficence. W e share w i t h m o s t obstetricians b o t h a desire to a v o i d i n c r e a s i n g m a t e r n a l m o r b i d i t y a n d m o r t a l i t y a n d the cost of cesarean delive r y w h e n the fetus is e x p e c t e d to die. H o w e v e r , for those p a t i e n t s w h o s e risk of p s y c h o l o g i c a l t r a u m a ~ J u l d m o s t likely exceed the risks of a cesarean, the latter m a y be the beneficent choice to m a k e on the rare occasion w h e n a g g r e s s i v e i n t r a p a r t u m m e a s u r e s fail. A l t h o u g h w e g e n e r a l l y agree w i t h the r e c o m m e n d a tions of C h e r v e n a k a n d M c C u l l o u g h 3 r e g a r d i n g termin a t i o n or n o n a g g r e s s i v e t h e r a p y for c a t e g o r y A a n o m alies a n d a g g r e s s i v e v e r s u s n o n a g g r e s s i v e m a n a g e m e n t for c a t e g o r y B a n o m a l i e s , c o u n s e l i n g r e g a r d i n g the p o s s i b l e use of i n c r e m e n t a l l y a g g r e s s i v e i n t r a p a r t u m m a n a g e m e n t techniques s e e m s a p p r o p r i a t e for patients w h o voice o p p o s i t i o n to n o n a g g r e s s i v e m a n a g e m e n t . This c o u n s e l i n g m a y a l l o w the p a t i e n t w h o m i g h t o t h e r w i s e reject n o n a g g r e s s i v e m a n a g e m e n t to accept selectively a g g r e s s i v e t h e r a p y a n d a v o i d cesarean delivery.
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References 1. Chervenak FA, Farley MA, Walters L, Hobbins JC, Mahoney MJ. When is termination of pregnancy during the third trimester morally justifiable? N Engl J Med 1984;310:501-4. 2. Chervenak FA, McCullough LB. Nonaggressive obstetric management. An option for some fetal anomalies during the third trimester. JAMA 1989;261:3439-40. 3. Chervenak FA, McCullough LB. An ethically justified, clinically comprehensive management strategy for third-trimester pregnancies complicated by fetal anomalies. Obstet Gynecol 1990;75:311-6. 4. Kirkinen P, Jouppila P, Herva R. Fatal fetal abnormalities. Route of delivery and effect of the development of antepartum diagnostics in the last 13 years. J Reprod Med 1992;37:645-8. 5. Sanders RC. Prenatal ultrasonic detection of anomalies with a lethal or disastrous outcome. Radiol Clin North Am 1990;28:16377. 6. Hassed SJ, Miller CH, Pope SK, Murphy P, Quirk JG, Cunnif C. Perinatal lethal conditions: The effect of diagnosis on decision making. Obstet Gynecol 1993;82:37-42. 7. Zeanah CH, Dailey JV, Rosenblatt MJ, Devereux NS. Do women grieve after terminating pregnancies because of fetal anomalies? A controlled investigation. Obstet Gynecol 1993;82:270-5. 8. Johnson SR, Elkins TE, Strong C, Phelan JP. Obstetric decisionmaking: Responses to patients who request cesarean delivery. Obstet Gynecol 1986;67:847-50. 9. AMA House of Delegates Statement, 1973. In: Rachels R. The end of life and euthanasia. Oxford: Oxford University Press, 1986:88, 192-3. 10. Beauchamp TL, Childress JF. Principles of biomedical ethics. 3rd ed. New York: Oxford University Press, 1989. 11. Kelly G. The duty to preserve life. Theological Studies 1951;12:550.
Address reprint requests to: Joseph A. Spinnato, M D Division of Maternal-Fetal Medicine Department of Obstetrics and Gynecology University of Louisville Louisville, KY 40292
Received March 9, 1994. Received in revisedform August 1, 1994. Accepted August 15, 1994.
Copyright © 1995 by The American College of Obstetricians and Gynecologists.
Obstetrics & Gynecology
The ability to diagnose lethal fetal conditions accurately has expanded the available m a n a g e m e n t options for patients and practitioners. In addressing such cases, the recent literature 1-3 has focused primarily on the following choices: 1) termination of pregnancy, particularly w h e n early diagnosis is made, 2) nonaggressive management that withholds certain therapeutic options, such as cesarean delivery for fetal distress, and 3) aggressive m a n a g e m e n t that is offered when the prediction of death or absent cognitive function is uncerFrom the Department of Obstetrics and Gynecology, University of Louisville School of Medicine, Louisville, Kentucky.
VOL. 85, NO. 1, JANUARY 1995
tain. In general, the justification for these m a n a g e m e n t options is made by the absence or virtual absence of a beneficence-based obligation to the affected fetus, a beneficence-based obligation to the mother, and respect for maternal autonomy. Stated simply, m a n y (if not most) patients and physicians choose either to terminate a pregnancy before the third trimester and avoid exposure of the patient to unnecessary risks, or in cases of continuing pregnancies, to not use therapeutic modalities that increase maternal risk (such as cesarean delivery for fetal indications) without a reasonable hope of fetal survival or cognitive function. In some institutions, prenatal detection of lethal anomalies has dramatically reduced inadvertent aggressive management of these fetuses. 4 We have encountered several patients whose fetuses have had lethal anomalies; despite thorough counseling and a seemingly clear understanding of their fetuses' prognoses, the patients were not only unwilling to terminate their pregnancies, but also adamantly opposed to nonaggressive intrapartum management that might result in stillbirth. This report describes four cases that illustrate the selective use of aggressive and incrementally aggressive therapies for such patients and presents a rationale for their use.
Case Reports The first patient was a 23-year-old primigravida, first seen for a second-opinion perinatal consultation. Marked oligohy~ dramnios was first noted 2 weeks earlier. Targeted ultrasonography performed elsewhere confirmed the findings and noted an enlarged fetal bladder and bilateral hydronephrosis. Percutaneous sampling of the fluid from the smaller kidney demonstrated values for osmolality, chloride, and sodium that were similar to those in plasma. The fetal karyotype was normal. The ultrasonographic findings were confirmed in our unit, and the fetal chest circumference was noted to be less than the tenth percentile for gestafional age, consistent with probable long-standing oligohydramnios. Nonaggressive
0029-7844/95/$9.50 89 0029-7844(94)00310-A
management was recommended. The couple initially insisted on aggressive management including cesarean delivery for fetal benefit, if necessary. Follow-up evaluations over the next 5 weeks consistently demonstrated oligohydramnios and poor fetal chest growth. The patient presented at 35 weeks' gestation in active labor with a frank breech presentation and no fetal heart rate (FHR) abnormalities. Management options were discussed, and cesarean delivery was discouraged. We suggested that the parents spend time with the newborn in the delivery room rather than choose aggressive neonatal resuscitation. They agreed to avoid cesarean, but requested that intrapartum techniques discussed previously, such as amnioinfusion, be used to lessen the chance of intrapartum death and that neonatologists be present for the delivery. Neonatologists counseled the couple and agreed to provide initially aggressive, postnatal management until lethality was confirmed. Fetal distress was not observed, and delivery was accomplished by the assisted breech extraction of a male infant with Apgar scores of 2 and 4 at I and 5 minutes, respectively. Umbilical vein acidemia was absent. Despite maximal ventilatory support, the infant died at 7 hours of age, with the clinical diagnosis of pulmonary hypoplasia. The couple later volunteered that they wished they had kept the baby in the delivery room with them, believing that the neonate suffered pain in the nursery. The second patient was a 22-year-old primigravida who was referred at 26 weeks' gestation when an office sonogram indicated a possible omphalocele. This finding was confirmed, and a ventricular septal defect was also noted. Amniocentesis was performed, and the karyotype indicated trisomy 18. Termination of the pregnancy and nonaggressive management were offered to the patient. She refused the former and, through most of the remainder of pregnancy, she requested aggressive intrapartum therapy, including cesarean delivery for fetal indications. Eventually, she agreed to aggressive intrapartum management that would stop short of cesarean delivery for fetal distress. Continuous fetal monitoring identified repetitive decelerations late in the active phase of labor. Standard measures, including repositioning and oxygen therapy, failed to ameliorate the tracing abnormalities. Forceps were used to shorten the second stage, and a 2195-g male infant was delivered, with Apgar scores of 7 and 8 at 1 and 5 minutes, respectively. The newborn's condition was initially stable in the nursery, and a decision was made to close the leaking omphalocele and insert a gastrostomy tube. The baby was discharged and died at home 2 weeks later from apparent cardiac failure. The mother's grief response was pronounced, including substance abuse and suicidal behavior that necessitated a brief psychiatric admission. Subsequently, she expressed particular gratitude for our intrapartum care. The third patient was a 19-year-old primigravida, referred at 29 weeks' gestation. Previous ultrasonographic examinations at 24 and 28 weeks' gestation demonstrated oligohydramnios, probable fetal growth retardation, and nonvisualization of the fetal kidneys and bladder. Evaluation in our unit confirmed these findings. After diagnostic amnioinfusion, rupture of membranes occurred. Options were discussed at length on several occasions. The couple initially desired fully
90 Spinnato et al
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aggressive management, including possible expectant management of the ruptured membranes. Over a 48-hour period, they came to accept and understand the poor prognosis of the fetus, agreeing to induction of labor with oxytocin and continuous monitoring. They also agreed to forego a cesarean for fetal indications. Variable decelerations recurred. Oxygen therapy, repositioning, and discontinuance of the oxytocin were followed by amnioinfusion, which reduced the severity and frequency of the decelerations. Assisted breech extraction of a 970-g male infant with Apgar scores of 3 and 5 at 1 and 5 minutes, respectively, was accomplished. Mild umbilical artery respiratory acidemia (pH 7.185, base excess -3.8) was noted. The baby was intubated and placed on a ventilator until a confirmatory postnatal sonogram was performed. Ventilatory support was removed, and the infant died within 3 hours of birth. The fourth patient was a 19-year-old primigravida, referred at 25 weeks' gestation with marked oligohydramnios, fetal thoracic circumference less than the fifth percentile, an enlarged heart with hypoplasia of the right ventricle, renal agenesis, and size less than the tenth percentile. Umbilical vein sampling revealed a normal male karyotype. Under the care of the referring physician, she entered spontaneous labor at 34 weeks' gestation. Variable FHR decelerations were present. The patient expressed a desire to deliver a live-born infant, but declined cesarean delivery for fetal distress. Amnioinfusion was not offered, and the fetus was monitored only intermittently. During labor, the patient told us that she "kept her fingers crossed" that the baby would be born alive. Thirty minutes before delivery, the fetal heart sounds were lost. The patient delivered a 1022-g stillborn male. She said it was very difficult to view her lifeless child. An autopsy confirmed the ultrasonographic findings.
Discussion Chervenak and McCullough 3 developed a "comprehensive m a n a g e m e n t s t r a t e g y " a n d d e s c r i b e d three categories of clinical situations b a s e d on the p r o b a b i l i t y of a n t e n a t a l d i a g n o s i s a n d the p r o b a b i l i t y of d e a t h or a b s e n t cognitive d e v e l o p m e n t a l capacity. T h e y stated that w h e n there are no b e n e f i c e n c e - b a s e d o b l i g a t i o n s to the fetus (category A, eg, a n e n c e p h a l y ) , o n l y t e r m i n a t i o n of p r e g n a n c y o r n o n a g g r e s s i v e m a n a g e m e n t s h o u l d be offered. W h e n there are m i n i m a l beneficenceb a s e d o b l i g a t i o n s to the fetus (category B, eg, t h a n a t o p h o r i c d y s p l a s i a ) , o n l y a g g r e s s i v e or n o n a g g r e s s i v e m a n a g e m e n t s h o u l d be offered. C a t e g o r y C a n o m a l i e s , those w h o s e d i a g n o s t i c accuracy or lethality or absence of cognitive function is uncertain, w e r e r e c o m m e n d e d for a g g r e s s i v e m a n a g e m e n t . W i t h i m p r o v e m e n t s in u l t r a s o n o g r a p h i c e q u i p m e n t a n d i n c r e a s i n g clinical experience, p r e n a t a l d i a g n o s i s has b e c o m e m o r e accurate in its p r e d i c t i o n of l e t h a l i t y or a b s e n t cognitive function. For m a n y institutions, the list of d i a g n o s e s that fulfill C h e r v e n a k a n d McCul-
Obstetrics & Gynecology
lough's category A criteria includes renal agenesis, acrania, exencephaly, severe forms of iniencephaly, allobar holoprosencephaly, trisomies 13 and 18 (particularly when major cranial or cardiac defects are present), parenchymal and obstructive renal disease with long-standing oligohydramnios, and possibly others. 5 Although some would probably object to the inclusion of these diagnoses in category A, few would disagree that counseling against aggressive management occurs commonly, even earnestly, when these diagnoses are made. Kirkinen et al 4 described cesarean delivery as inappropriate, useless, and associated with unnecessary maternal morbidity and extra cost when a fatal anomaly is present. Little guidance is available from the literature regarding patient requests for aggressive management, including cesarean delivery, when the fetal condition is lethal. Hassed et al 6 reported 130 pregnancies diagnosed with lethal conditions at or before 24 weeks' gestation. Termination of pregnancy was chosen by 67% (n = 87) and continuation of pregnancy was elected by 33% (n = 43). Postulated reasons for continuing the pregnancies included the alleviation of guilt and "to allow Mother Nature to take its course." The fetuses from these pregnancies were less likely to have an autopsy than the aborted fetuses. The authors speculated that these mothers had become more attached to their fetuses. Zeanah et al 7 evaluated 23 women 2 months after pregnancy termination for fetal anomalies and compared them with matched controls who had had a spontaneous loss. Seventeen percent of the patients in the termination group experienced major depression and 22% sought psychiatric help. The authors concluded that the grief response was similar to that of the women experiencing spontaneous loss. With regard to performing a medically unnecessary cesarean, Johnson et al s polled 112 obstetricians and asked them to consider 19 hypothetical clinical situations with varying degrees of justification for patientrequested cesarean delivery. As many as 38% of the respondents said they would agree to perform a cesarean at the request of a patient for various nonindications. The factors cited were patient autonomy and the maxim, "do no harm." Our experience with these four patients and an evaluation of pertinent ethical principles prompts us to propose that aggressive intrapartum therapy, which attempts to avoid fetal death, is appropriate for selected patients whose fetuses definitely have lethal anomalies. We do not view this as an issue of fetal beneficence and agree that no such obligations exist in these cases. Instead, our position stems from our beneficence-based
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obligation to the mother and from respect for maternal autonomy. The principle of nonmaleficence, understood in medicine as primum non nocere (first do no harm), is a central part of our societal morality. Although patient motives are heterogeneous, "do not kill" and "do not inflict harm" are driving forces in the opposition to terminating a pregnancy and are likely part of the desire to avoid intrapartum stillbirth. Indeed, for some, the argument centers around the distinction between killing and letting die. In medicine, active euthanasia (ie, killing) is generally rejected, but passive euthanasia (ie, letting die), defined as "to withhold or withdraw extraordinary means to prolong the life of the body when there is irrefutable evidence that biologic death is imminent," is seen as being morally justified. 9 In fact, this is the very argument that justifies nonaggressive management. However, protecting a fetus from dying in labor is not considered extraordinary means to some. Some patients (and possibly some physicians) understand ordinary means as "usual and customary" and extraordinary means as "unusual or a departure from custom. ''1° More correctly defined, "extraordinary means are all medicines, treatments and operations, which cannot be obtained or used without excessive expense, pain, or other inconvenience, or which, if used, would not offer a reasonable hope of benefit. ''11 Using this definition, our ordinary custom, to protect the intrapartum fetus from dying using intrapartum resuscitation and possibly cesarean delivery, becomes extraordinary care and is not morally required in a hopeless situation. The correctness of this ethical argument may not be accepted by some patients. The hope of patients for their unborn children is a powerful virtue that should not be set aside thoughtlessly. Their questioning of a terminal prognosis should always be understood. It should be noted that intrapartum resuscitation measures short of cesarean (such as oxygen therapy, fetal monitoring, amnioinfusion, elimination of hyperstimulation by tocolysis, or forceps delivery) involve minimal expense and little, if any, morbidity. They may not be so extraordinary to justify withholding them against maternal wishes. Their hidden expense occurs when intrapartum death is avoided or when the neonatologist is unwilling to accept prenatal evidence of lethality and postnatal expenses are incurred. These expenses may be minimized by careful antenatal involvement of neonatologists in the decision-making process. When rapid death is expected, as with renal agenesis, we have successfully encouraged couples to keep the infant in the birthing room, thus avoiding any added nursery costs or interventions. Our first three cases demonstrate how dialogue with
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the p a r e n t s a n d their a g r e e m e n t w i t h a g g r e s s i v e intrap a r t u m m a n a g e m e n t s h o r t of cesarean d e l i v e r y a l l o w e d t h e m to a v o i d a b a n d o n i n g their child d u r i n g labor. W e s p e c u l a t e that as a c o n s e q u e n c e of o u r counseling, the p a t i e n t s c a m e to u n d e r s t a n d that cesarean is i n d e e d a n e x t r a o r d i n a r y m e a n s w h e n the fetus is e x p e c t e d to die. The f o u r t h case d e m o n s t r a t e s h o w different a p a t i e n t ' s experience is w h e n c o n t i n u o u s m o n i t o r i n g a n d amn i o i n f u s i o n are w i t h h e l d . As p o i n t e d o u t b y Johnson et al, 8 obstetricians h a v e at times e x p r e s s e d a n d d e m o n s t r a t e d a w i l l i n g n e s s to p e r f o r m an obstetrically u n n e c e s s a r y cesarean d e l i v e r y for the p s y c h o l o g i c a l benefit of a patient. A n obstetrician's fear of litigation m a y also be a c o m p e l l i n g reason. W e p e r c e i v e such decisions as d r i v e n b y m a t e r n a l beneficence. R e a s o n a b l e a t t e m p t s (that is, efforts that r e s p e c t m a t e r n a l a u t o n o m y a n d d o not use i n t i m i d a tion) to d i s s u a d e the p a t i e n t f r o m cesarean d e l i v e r y b e c a u s e of the i n c r e a s e d risks are a p p r o p r i a t e . H o w ever, if there is r e a s o n a b l e e v i d e n c e or s u s p i c i o n that s u b s t a n t i a l p s y c h o l o g i c a l h a r m m i g h t be d o n e to the p a t i e n t w h o s e fetus dies d u r i n g labor, the e x p e n s e a n d risks of a g g r e s s i v e i n t r a p a r t u m m a n a g e m e n t can be seen as m a t e r n a l l y beneficent, w i t h o u t violating the p r i n c i p l e of non-maleficence. W e share w i t h m o s t obstetricians b o t h a desire to a v o i d i n c r e a s i n g m a t e r n a l m o r b i d i t y a n d m o r t a l i t y a n d the cost of cesarean delive r y w h e n the fetus is e x p e c t e d to die. H o w e v e r , for those p a t i e n t s w h o s e risk of p s y c h o l o g i c a l t r a u m a ~ J u l d m o s t likely exceed the risks of a cesarean, the latter m a y be the beneficent choice to m a k e on the rare occasion w h e n a g g r e s s i v e i n t r a p a r t u m m e a s u r e s fail. A l t h o u g h w e g e n e r a l l y agree w i t h the r e c o m m e n d a tions of C h e r v e n a k a n d M c C u l l o u g h 3 r e g a r d i n g termin a t i o n or n o n a g g r e s s i v e t h e r a p y for c a t e g o r y A a n o m alies a n d a g g r e s s i v e v e r s u s n o n a g g r e s s i v e m a n a g e m e n t for c a t e g o r y B a n o m a l i e s , c o u n s e l i n g r e g a r d i n g the p o s s i b l e use of i n c r e m e n t a l l y a g g r e s s i v e i n t r a p a r t u m m a n a g e m e n t techniques s e e m s a p p r o p r i a t e for patients w h o voice o p p o s i t i o n to n o n a g g r e s s i v e m a n a g e m e n t . This c o u n s e l i n g m a y a l l o w the p a t i e n t w h o m i g h t o t h e r w i s e reject n o n a g g r e s s i v e m a n a g e m e n t to accept selectively a g g r e s s i v e t h e r a p y a n d a v o i d cesarean delivery.
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References 1. Chervenak FA, Farley MA, Walters L, Hobbins JC, Mahoney MJ. When is termination of pregnancy during the third trimester morally justifiable? N Engl J Med 1984;310:501-4. 2. Chervenak FA, McCullough LB. Nonaggressive obstetric management. An option for some fetal anomalies during the third trimester. JAMA 1989;261:3439-40. 3. Chervenak FA, McCullough LB. An ethically justified, clinically comprehensive management strategy for third-trimester pregnancies complicated by fetal anomalies. Obstet Gynecol 1990;75:311-6. 4. Kirkinen P, Jouppila P, Herva R. Fatal fetal abnormalities. Route of delivery and effect of the development of antepartum diagnostics in the last 13 years. J Reprod Med 1992;37:645-8. 5. Sanders RC. Prenatal ultrasonic detection of anomalies with a lethal or disastrous outcome. Radiol Clin North Am 1990;28:16377. 6. Hassed SJ, Miller CH, Pope SK, Murphy P, Quirk JG, Cunnif C. Perinatal lethal conditions: The effect of diagnosis on decision making. Obstet Gynecol 1993;82:37-42. 7. Zeanah CH, Dailey JV, Rosenblatt MJ, Devereux NS. Do women grieve after terminating pregnancies because of fetal anomalies? A controlled investigation. Obstet Gynecol 1993;82:270-5. 8. Johnson SR, Elkins TE, Strong C, Phelan JP. Obstetric decisionmaking: Responses to patients who request cesarean delivery. Obstet Gynecol 1986;67:847-50. 9. AMA House of Delegates Statement, 1973. In: Rachels R. The end of life and euthanasia. Oxford: Oxford University Press, 1986:88, 192-3. 10. Beauchamp TL, Childress JF. Principles of biomedical ethics. 3rd ed. New York: Oxford University Press, 1989. 11. Kelly G. The duty to preserve life. Theological Studies 1951;12:550.
Address reprint requests to: Joseph A. Spinnato, M D Division of Maternal-Fetal Medicine Department of Obstetrics and Gynecology University of Louisville Louisville, KY 40292
Received March 9, 1994. Received in revisedform August 1, 1994. Accepted August 15, 1994.
Copyright © 1995 by The American College of Obstetricians and Gynecologists.
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