Original Article
Cesarean Section before the Onset of Labor and Subsequent Motor Function in Infants with Meningomyelocele Diagnosed Antenatally
N Engl J Med 1991; 324:662-666March 7, 1991
- Abstract
- Abstract
Background.
Meningomyelocele can now be detected before birth. Few data are available on its natural history, however, and optimal management at the time of delivery is controversial, although it has been suggested that labor and vaginal delivery may cause pressure on exposed nerve roots, resulting in additional loss of neural function.
Methods.
To assess the effect of labor and the type of delivery on the level of motor function in fetuses with uncomplicated meningomyelocele, we identified 200 cases of this disorder, accounting for 95 percent of the cases that occurred in the state of Washington during our 10-year study period. We compared the outcomes of 47 infants delivered by cesarean section before labor began, 35 delivered by cesarean section after a period of labor, and 78 who were delivered vaginally (another 40 were ineligible for the study). In cases of meningomyelocele detected prenatally, cesarean section was performed before the onset of labor if isolated meningomyelocele without severe hydrocephalus was present. The infants delivered in this manner were compared with those who were delivered either vaginally or by cesarean section after labor began.
Results.
At two years of age, the infants who had been exposed to labor were 2.2 times more likely to have severe paralysis than those delivered by cesarean section without labor (95 percent confidence interval, 1.7 to 2.8). Infants delivered by cesarean section before the beginning of labor had a mean (±SD) level of paralysis 3.3±3.0 segments below the anatomical level of the spinal lesion at two years of age, as compared with 1.1±2.3 for infants delivered vaginally and 0.9±4.1 for infants delivered by cesarean section after the beginning of labor (P<0.001 for both comparisons). Exposure to labor did not affect the frequency of neonatal complications or later intellectual performance.
Conclusions.
For the fetus with uncomplicated meningomyelocele, delivery by cesarean section before the onset of labor may result in better subsequent motor function than vaginal delivery or delivery by cesarean section after a period of labor. (N Engl J Med 1991; 324: 662–6.)
Media in This Article
Table 1
Gestational Age at Antenatal Diagnosis of Meningomyelocele and Outcome of Pregnancy.Table 2
Characteristics of Pregnancy and Level of Paralysis, According to Exposure to Labor and Type of Delivery.Article Activity
- Article
WHEN meningomyelocele is detected in a fetus, prenatal counseling and management may be problematic. Termination of the pregnancy may be chosen by the mother in many cases, whereas in others termination may not be possible because of personal or religious beliefs or because of the advanced stage of gestation at the time of diagnosis. Little information is available about the natural history of meningomyelocele detected in utero to provide guidance in such cases,1 2 3 4 and the benefits and risks of different modes of delivery are controversial.5 6 7 8 In 1970 Stark and Drummond8 suggested that labor and vaginal delivery could cause pressure on the neural plate and result in additional loss of neural function. In 1984, Chervenak et al.9 described a technique of cesarean delivery to avoid trauma to the exposed meninges. More recently, however, Bensen et al.6 found no benefits in terms of mortality, incidence of meningitis, or developmental outcome for cesarean section as compared with vaginal delivery for infants with meningomyelocele.
Since 1979 we have collected data on infants with meningomyelocele detected either before or at the time of delivery. For fetuses given an antenatal diagnosis of meningomyelocele who did not have severe hydrocephalus and who were carried to term, we have offered elective cesarean section before the onset of labor, together with immediate back closure, in an effort to preserve motor function.8 We compared the results in this group with those among infants with meningomyelocele detected at birth to assess the effects of labor and the type of delivery on the level of paralysis.
Methods
The records of all fetuses given an antenatal diagnosis of meningomyelocele at the Swedish Hospital Medical Center or University Hospital Medical Center in Seattle, between January 1, 1979, and December 31, 1988, were reviewed. We also reviewed the records of all infants in whom meningomyelocele was first detected at birth who were seen by the staff of the Division of Congenital Defects at Children's Hospital and Medical Center, Seattle, during the same period. The two groups comprised approximately 95 percent of the infants with meningomyelocele identified in the state of Washington during the 10-year study period.
Antenatal diagnosis was accomplished by measurement of maternal serum alpha-fetoprotein, amniotic fluid alpha-fetoprotein, and amniotic fluid acetylcholinesterase and by ultrasonography, which was also used to identify the anatomical level of the spinal lesion, the presence and degree of hydrocephalus, and any associated anomalies. For the purpose of this study, we defined severe hydrocephalus as an in utero head circumference at least 4 SD above the mean for gestational age and a cerebral cortex of less than 1 cm as measured by ultrasonography through the anterior horns of the lateral cerebral ventricles.10 , 11 Analysis of fetal chromosomes was performed in all cases detected antenatally. Termination of pregnancy was offered to the mother if the diagnosis was made up to the 24th week of gestation and after the 24th week if there were also multiple-system anomalies or chromosomal anomalies thought to be incompatible with life.
The mothers who elected to continue the pregnancy were followed up and delivered at either Swedish Hospital Medical Center or University Hospital Medical Center. Ultrasound examinations were performed at least monthly to assess fetal growth, amniotic fluid volume, fetal movement, and degree of hydrocephalus and to screen for other anomalies. The mothers also received counseling from perinatologists, neonatologists, and pediatricians from the Birth Defects Clinic at Children's Hospital and Medical Center. In the absence of severe hydrocephalus, all mothers carrying a fetus given an antenatal diagnosis of meningomyelocele were offered the option of delivering by cesarean section before the onset of labor (prelabor cesarean section) as a means of avoiding the potentially adverse effects of labor or vaginal delivery on the exposed nerve roots of the fetus. Because of the grim prognosis for fetuses with meningomyelocele and severe hydrocephalus in utero, prelabor cesarean section was not offered to improve fetal outcome when both conditions were present. In some of these cases, cesarean section was performed to avoid trauma to the mother from the large fetal head. In most of these cases, vaginal delivery took place after cranial decompression.
All the mothers who were offered the option chose to undergo cesarean section before the onset of labor. Serial amniocenteses to assess fetal lung maturity were begun at 36 weeks, and cesarean section was performed as soon as fetal pulmonary maturation was documented by a lecithin:sphingomyelin ratio of at least 2.0 in amniotic fluid,5 , 7 , 9 10 11 12 unless spontaneous labor began before the cesarean section could be performed. In this situation, cesarean section was performed as soon as possible after the onset of labor. Neonatologists were present at delivery. Spinal lesions were wrapped in sterile bandages, and the infants were placed in sterile isolettes and handled with sterile technique until back-closure surgery, which was scheduled to follow delivery as soon as feasible.13 14 15 At the time of surgery, the length and width of the spinal lesions were measured, at the skin surface, from the last area of normal skin and subcutaneous tissue.
For infants first given a diagnosis of meningomyelocele at the time of delivery, we reviewed the medical records for information on the presence or absence of labor (defined as regular uterine contractions occurring over a period of at least two hours and cervical dilatation of more than 2 cm), the type of delivery, and the indications for cesarean section, if any. Data on neonatal complications were collected for both the group diagnosed antenatally and the group diagnosed at the time of delivery.
The anatomical level of the spinal lesion was defined as the most caudal posterior vertebral arch that appeared intact on x-ray films. All films were obtained as part of urologic care and were reviewed by a single examiner. The age at which x-ray films of the infants were obtained ranged from two to four years, since they were obtained only when indicated clinically. However, the levels of lesions as indicated by radiography do not change with age.
The level of paralysis (motor level), in contrast to the anatomical level of the lesion, was defined as the lowest level of purposeful movement, on either the right or the left side, exclusive of spastic or reflex activity. In children with meningomyelocele, this level is as low as or lower than the anatomical level. The motor levels were identified by physical therapists who recorded sequential segmental motor-level determinations with use of a standard muscle-examination procedure, as described by Sharrard,16 at 6, 12, 18, and 24 months and yearly thereafter. The motor levels determined at the 24-month examination were used in the analyses described in this report. Both motor and anatomical levels were converted to numerical values as follows: cervical, 1 through 8; thoracic, 9 through 20; lumbar, 21 through 25; sacral, 26 through 29; and no loss of motor function, 30. To determine the difference between the anatomical level and the motor level, we subtracted the numerical value of the last intact vertebral arch from that of the lowest motor level.
To evaluate long-term outcome and identify any developmental disabilities, we assessed the infants at 6, 12, 18, and 24 months and yearly thereafter. At these times the Bayley Scales of Infant Development17 were administered by occupational therapists or trained psychometricians. The Bayley Scales consist of the Mental Development Index and the Psychomotor Development Index. The mean (±SD) standardized value for the Mental Development Index is 100±16. Since the Psychomotor Development Index is affected by motor performance and therefore may not be reliable in children with meningomyelocele,13 14 15 only the Mental Development Index is reported here. The scores obtained on the Mental Development Index at two years of age were used in the analyses reported here.
The level of motor function, the score on the Bayley Mental Development Index, and the incidence of neonatal complications in the infants delivered by cesarean section before the onset of labor were compared with those in the infants delivered after labor, either by cesarean section or vaginally. The results were stored, retrieved, and analyzed with a computerized data-management system described elsewhere.1 , 13 14 15 Analysis of variance, t-tests, chi-square tests, and Fisher's exact tests were used to examine the mean difference between motor and anatomical levels for infants delivered by cesarean section without labor, cesarean section after the onset of labor, and vaginal delivery to determine whether the degree of motor paralysis was related to the time of diagnosis of meningomyelocele and to evaluate the significance of any association between the level of motor paralysis and the type of delivery. Maximum-likelihood estimates of relative risk and 95 percent confidence intervals were calculated to determine the risk of motor paralysis associated with exposure to labor and with the type of delivery.
Results
A total of 200 cases of meningomyelocele were identified during the 10-year study period: 81 before delivery and 119 at the time of delivery. The 81 cases in which diagnosis was made antenatally and their outcomes are summarized in Table 1Table 1
Gestational Age at Antenatal Diagnosis of Meningomyelocele and Outcome of Pregnancy.. In 119 cases the diagnosis of meningomyelocele was made at the time of delivery. Eleven cesarean sections were performed before the onset of labor because of abnormal fetal position or previous cesarean section. In 108 cases, delivery occurred after the onset of labor — by cesarean section in 30 and vaginally in 78.Regardless of the route of delivery, we excluded from further analysis all cases in which pregnancy was terminated (n = 25), which included all cases of chromosomal anomalies (n = 7), multiple-system anomalies (n = 10), and isolated spina bifida (n = 8). We also excluded all cases in which pregnancy was terminated because of intrauterine or neonatal death (n = 17) or severe hydrocephalus (n = 12).11 , 18 , 19
A total of 160 infants with meningomyelocele survived the neonatal period and were followed up. Forty-seven infants were delivered by cesarean section before labor began, and 113 were delivered after the onset of labor. Among the latter, 35 infants were delivered by cesarean section, and 78 were delivered vaginally. There was no difference between the infants delivered by cesarean section before labor began and those delivered after the onset of labor in the size of the spinal lesions (calculated as length times width). The mean (±SD) size of the lesion was 26.8±22.1 cm2 in the prelabor-cesarean-section group and 26.6±19.6 cm2 in the two groups exposed to labor (P = 0.95). Within the group delivered by cesarean section before labor, no significant differences were noted in the distribution of motor levels according to gestational age at the time of diagnosis.
The mean gestational age at delivery was significantly lower for the prelabor-cesarean-section group than for either the group delivered by cesarean section after labor began or the vaginal-delivery group (F = 3.05, P = 0.05). The mean gestational ages in two groups exposed to labor did not differ significantly from one another (Table 2Table 2
Characteristics of Pregnancy and Level of Paralysis, According to Exposure to Labor and Type of Delivery.). The mean duration of labor was 10.8 hours in the labor-plus-cesarean-section group, as compared with 7.3 hours in the vaginal-delivery group (P<0.05).The mean anatomical levels and mean motor levels for the three groups at two years of age are also shown in Table 2. As determined by radiography, there was no significant difference in the mean anatomical level of spinal lesions between the children delivered by cesarean section without labor and those delivered by cesarean section after labor began; the anatomical level among the children delivered vaginally was significantly lower than that for either cesarean-section group (F = 4.89, P<0.01). The mean motor level was not significantly different in the prelabor-cesarean-section group and the vaginal-delivery group, but each was significantly lower than that in the labor-plus-cesarean-section group (F = 5.03, P<0.01).
When the anatomical levels were subtracted from the motor levels, the mean (±SD) difference was greater for the prelabor-cesarean-section group (3.3±3.0) than for either the vaginal-delivery group (1.1±2.3) or the labor-plus-cesarean-section group (0.9±4.1, F = 8.67, P<0.001) (Table 2). There was no significant difference between the two groups exposed to labor in the difference between the motor level and the anatomical level, but each of these groups differed significantly from the prelabor-cesarean-section group.
Significant differences in the severity of paralysis were found between the infants delivered by cesarean section without labor and those who were exposed to labor (Table 3Table 3
Level of Paralysis (Motor Level) at Two Years of Age, According to Exposure to Labor and Type of Delivery.*). Forty-five percent of the prelabor-cesarean-section group had sacral levels of paralysis or no loss, as compared with 16 percent of the groups exposed to labor, whether delivery was by cesarean section or vaginal X2 = 2.6, P = 0.005). The chance of severe paralysis (at the level of vertebra T-12 to L-3) was 2.2 times greater in the groups exposed to labor than in the prelabor-cesarean-section group (95 percent confidence interval, 1.7 to 2.8). No significant differences were found between the vaginal-delivery group and the labor-plus-cesarean-section group with respect to either severe levels of paralysis (T-12 to L-3) or minimal levels (sacral levels or no loss), but the likelihood of paralysis in each of these groups was significantly greater than in the prelabor-cesarean-section group. These differences were not influenced by maternal age, the infant's sex, the anatomical level of the spinal lesion, or the distance from the family's home to the tertiary care center.No significant differences were found among the prelabor-cesarean-section group, the labor-plus-cesarean-section group, and the vaginal-delivery group in the frequency of placement of a central nervous system shunt, either overall or for any given motor level. No serious wound dehiscence occurred in any group; central nervous system infections were rare, and their frequency did not differ among the study groups.
No differences were found in the means (±SD) for the highest score achieved up to and including 24 months of age on the Bayley Mental Development Index between the prelabor-cesarean-section group and the groups exposed to labor (prelabor-cesarean-section group, 89±20; groups exposed to labor, 89±22) or in the mean scores at 24 months on the Bayley Mental Development Index (prelabor-cesarean-section group, 94±15; groups exposed to labor, 85±22; P<0.8). The likelihood that this finding was due to chance does not exceed 5 percent.
Discussion
As measurements of maternal serum alpha-fetoprotein and ultrasonography are used more widely, more fetuses with meningomyelocele will be identified before birth. Since the termination of pregnancy may not always be an option, the optimal perinatal management of these cases is extremely important. In over 60 percent of the cases (13 of 21) in our series in which the diagnosis of isolated meningomyelocele was made up to 24 weeks of gestation (Table 1), the mothers elected to continue the pregnancy.
The results of this study suggest that cesarean delivery before the onset of labor may result in improved motor function, as compared with delivery after the onset of labor, either by cesarean section or vaginal, without affecting intellectual performance. Cesarean section after the onset of labor resulted in levels of paralysis no different from those in children delivered vaginally, but larger studies are needed to ascertain whether delivery by cesarean section after labor has begun has any benefit in terms of motor performance. The better motor performance in the infants delivered by cesarean section without labor could be due to either, or both, of two factors. First, scheduled cesarean section before the onset of labor allows for coordinated delivery with appropriate specialists present and thus permits rapid back closure in a timely and predictable manner. Second, cesarean section before the onset of labor precludes trauma to nerves from the forces of labor.
The infants in the prelabor-cesarean-section group had a much greater chance of having low lumbar or sacral paralysis, or none, than the infants born after labor began. Low lumbar and sacral levels of paralysis allow the hips and knees to be flexed and extended. The preservation of muscle function at these levels enables the patient to walk with minimal mechanical assistance, or none, as an adult, whereas patients with higher-level lesions generally require wheelchairs. These are important socioeconomic and psychosocial differences.
Since we found that labor affected motor function but not intellectual performance, our findings are consistent with those of Bensen et al.,6 who examined intellectual performance and mortality but not motor performance. We identified no differences in mortality, morbidity, or intellectual performance between infants with meningomyelocele who were delivered before the onset of labor and those born after the onset of labor.
These results were collected prospectively. Since patients were not randomly assigned to treatment groups, however, some selection bias could be present in the prelabor-cesarean-section group. In addition, antenatal diagnosis could introduce bias by selecting more severely affected fetuses for termination of pregnancy and allowing less severely affected fetuses to reach delivery. However, there were no differences between the groups in the size of the spinal lesions. The anatomical levels of the lesions (determined by x-ray films) were similar in the prelabor-cesarean-section and labor-plus-cesarean-section groups, but significantly lower in the vaginal-delivery group. Despite the lower mean anatomical levels in the vaginal-delivery group, there was a twofold increased risk of severe paralysis as compared with that in the prelabor-cesarean-section group. Furthermore, these differences were not influenced by maternal age, the infant's sex, the anatomical level of the lesion, or the distance from the family's home to the perinatal center. Since infants with severe hydrocephalus, chromosomal anomalies, and other anomalies were excluded from all three groups, no important selection bias appears to have been at work. A prospective, randomized trial would be extremely difficult, if not impossible, to perform, given the small numbers of cases and the difficulties of randomly assigning patients to treatment groups in multicenter studies of new forms of technology.20
Chromosomal abnormalities were found in 10 percent of the fetuses given a diagnosis before delivery, and multiple-system anomalies with normal karyotypes were found in an additional 15 percent. Another 15 percent had severe hydrocephalus. These results are consistent with previous reports,21 , 22 and we feel strongly that fetal karyotyping and serial ultrasound examinations should be performed for all such fetuses whose mothers contemplate the continuation of the pregnancy.
Only 2 of the 12 infants with meningomyelocele and severe hydrocephalus survived; these two have midthoracic lesions and are profoundly retarded. We do not recommend prelabor cesarean section to improve fetal outcome when severe hydrocephalus is present; in some cases, however, cesarean section may be necessary to avoid severe maternal trauma.
In conclusion, for the fetus with meningomyelocele without severe hydrocephalus and with a normal karyotype, scheduled delivery by cesarean section before the onset of labor may result in improved motor function.
Supported in part by the March of Dimes Birth Defects Foundation and by gifts from Mr. Steve Largent to the Birth Defects Clinic at Children's Hospital and Medical Center, Seattle.
Source Information
From the Divisions of Perinatal Medicine (D.A.L., K.A.H., D.E.H.) and Ultrasound (D.A.N.), Swedish Hospital Medical Center, and the Departments of Pediatrics (T.W., D.B.S.) and Obstetrics and Gynecology (T.J.B.). University Hospital Medical Center, both in Seattle. Address reprint requests to Dr. Luthy at the Department of Obstetrics and Gynecology and the Division of Perinatal Medicine, Swedish Hospital Medical Center, 747 Summit Ave., Seattle, WA 98104.
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