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BJOG: an International Journal of Obstetrics and Gynaecology January 2004, Vol. 111, pp.

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DOI: 1 0 . 1 0 4 6 / j . 1 4 7 1 - 0 5 2 8 . 2 0 0 3 . 0 0 0 0 9 . x

Monoamniotic twin pregnancies: antenatal management and perinatal results of 19 consecutive cases
Fabien Demaria, Francois Goffinet*, Gilles Kayem, Vassilis Tsatsaris, Madieh Hessabi, Dominique Cabrol
Objective To describe the obstetric management and perinatal outcome of antenatally diagnosed monoamniotic twin pregnancies (MATP) in a tertiary level maternity unit. Setting Port-Royal Maternity Hospital, Paris, France. Population MATP that progressed beyond 22 weeks seen from 1993 to 2001. Methods A retrospective chart review of all twin pregnancies. Diagnosis of MATP was made by ultrasonography and confirmed by placental pathology. Main outcome measure Perinatal mortality. Results Among the 1242 twins pregnancies delivered during the study period, 19 were monoamniotic. Four fetuses (10% of all births) had malformations. Perinatal mortality was high (n 12, 32%) because of fetal deaths (nine cases) and very preterm births (three neonatal deaths). No fetal deaths occurred after 29 weeks. Of the 15 women with at least one live fetus before labour, 6 gave birth by vaginal delivery (40%). No obstetric accidents occurred during vaginal deliveries. Conclusion Perinatal mortality of MATP is still very high, even with accurate, early antenatal diagnosis, intensified surveillance and delivery provided in a tertiary level hospital. The main causes of perinatal deaths are cord accidents in utero, congenital anomalies and very preterm births. INTRODUCTION Monoamniotic twin pregnancy (MATP) is a rare occurrence; its frequency is estimated at 1 per 25,000 pregnancies.1 4 Perinatal mortality in MATP is high, with reported rates ranging from 28% to 70% in the studies before 2001.5 The deaths are primarily related to prematurity and intrauterine fetal death due to cord accidents.5,6 The above figures come from the pooling of case series from several centres over very different periods, with variable types of recruitment and management, often diagnosed postnatally.1,2,7 9 Ultrasound now makes antenatal diagnosis possible in most cases, but data from single centres on their surveillance, delivery decisions and type of delivery are sparse. A recent study with antenatal diagnosis reported better perinatal outcome than previously observed.5 We report here the results and management of a consecutive series of 19 MATP that progressed beyond 22 weeks over the past nine years, all managed by the same team.

METHODS Charts of all twin pregnancies that progressed beyond 22 weeks seen at the Port-Royal Maternity Hospital (University of Rene Descartes-Paris V) from 1993 to 2001 were reviewed retrospectively. Our department, which handles 3500 deliveries per year, is a tertiary perinatal centre with onsite neonatal and maternal intensive care units. The diagnosis of chorionicity and amnionicity was always confirmed by histology examinations that were performed systematically in our centre for all twin pregnancies. All patients underwent a first trimester ultrasound examination, and chorionicity was diagnosed based on the following two criteria: 1. An early antenatal ultrasound was repeated by a different operator, and both observed10 a single placental mass, no intertwin membrane, same-sex twins, similar distribution of amniotic fluid around both fetuses and free movements for both. 2. The histological examination of the placenta systematically confirmed the chorionicity (monochorionic placenta) and amnionicity (absence of intertwin membrane).
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Department of Obstetrics and Gynecology, Maternity PortRoyal, Cochin-Saint Vincent-de-Paul hospital, University Paris V, 123 Bd de Port-Royal, 75014 Paris, France
* Correspondence: Dr F. Goffinet, Department of Obstetrics and Gynecology, Maternity Port-Royal, Cochin-Saint Vincent-de-Paul hospital, University Paris V, 123 Bd de Port-Royal, 75014 Paris, France. D RCOG 2004 BJOG: an International Journal of Obstetrics and Gynaecology

PERINATAL MORTALITY REMAINS VERY HIGH IN MONOAMNIOTIC TWIN PREGNANCIES

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Our centre is part of a perinatal network covering approximately 30,000 births a year. Numerous twin pregnancies are referred to us during the first trimester or transferred during the second or third from various maternity clinics in the network. Our centre manages approximately 150 twin pregnancies each year and thus has one of the largest twin populations in France. Specific management for twin pregnancies was implemented many years ago.11 Management of MATP is based upon this program, but surveillance is intensified at 24 weeks and delivery planned for around 36 weeks. This management has changed very little during the nine years of this study and includes the following points. The woman sees her obstetrician every three weeks from 15 weeks for a standard antenatal visit. The visit covers a checklist of questions for the mother, blood pressure measurement, weighing, urinary glucose and protein analysis, measurement of fundal height and a pelvic examination. Iron, folate and vitamin D supplements are prescribed systematically. Hospitalisation during pregnancy, corticosteroid administration and cerclage are not routine, each takes place on a case-by-case basis, as deemed necessary. Maternity leave is prescribed at 15 weeks, with rest at home. Weekly home visits by a midwife begin at 22 weeks: thisroutine medical examinations include blood pressure measurement, urinary glucose and protein analysis, measurement of fundal height and when appropriate clinical assessment of cervical state. The role of the midwife also includes questioning and advising the woman as well as

routine inquiries about need to consult one of our teams psychologists or when appropriate the psychiatrist for the maternity ward. Similarly, social workers provide necessary services during pregnancy and after the delivery. The following ultrasound examinations are performed: one during the first trimester, another around 16 18 weeks, one at 22 weeks and then every 2 weeks. Starting at 24 weeks, the following items are examined: morphologic assessment, biometry to verify the growth of two fetuses; a biophysical examination is performed and reported qualitatively (we do not use a score): fluid quantity, trunk, limb and thoracic movements. An umbilical artery Doppler is systematic, while the cerebral artery Doppler is performed only in cases of intrauterine growth retardation or umbilical Doppler abnormalities. Cord anomalies (knots and tangles) are not sought systematically; cervical ultrasound is not routine. There are no pre-established decision criteria; in the case of abnormalities, the woman is admitted and her case discussed at a staff meeting. After 28 weeks, additional consultations (similar to those with the obstetrician) take place twice weekly with a midwife at the maternity and include fetal heart rate recording. Delivery is planned around 36 weeks. Standard perinatal outcomes were analysed. A summary variable called neonatal distress was defined by the presence of at least one of the following criteria: neonatal death, 5 minute Apgar score <7, pH <7.15, transfer to neonatal intensive unit care (NICU). Third-stage haemorrhage was defined as a bleeding > 500 mL. We compared categorical variables with the m2 or Fishers exact test, as appropriate.

Table 1. Management of the 19 monoamniotic twin pregnancies. Case no. 1 2 3 4 5 6 7 8 9 10 11 12 13 14 15 16 17 18 19 Age (years) 38 42 26 25 30 39 33 31 33 33 28 27 23 28 23 28 32 30 32 Parity (IvF) P2 P3 P0 P1 P2 P2 P0 P0 P0, Ivf P1 P0 P0, Ivf P0 P0 P1 P0 P0, Ivf P0 P0 GA at diagnosis 12 11 11 12 10 12 12 12 13 13 13 11 10 13 20 9 13 12 15 GA at the in utero transfer 25 23 no no no 28 27 no 29 29 no no 28 32 36 18 24 26 no Hospital visit 5 4 16 5 0 5 12 12 14 2 4 Home midwife visits 20 9 5 2 23 12 2 18 10 16 2 Antenatal stay in hospital (days)* cause 4 PTL 24/28 polyhydramnios/IUFD T2 29/5 PTL/cholestasis 15 IUGR T2 5 IUGR T2 43/2/2 PTL/PTL/PTL 2 PTL 7/30 PTL/PTL 1 IUGR T2 3/3 pre-eclampsia/pre-eclampsia 13 surveillance 4 polyhydramnios Course of steroids 1 2 5 0 2 2 2 0 2 1 0 0 0 1 0 0 0 0 0 In utero fetal death no T2 IUFD no no no no no no no no no no no no no IUFD both twins IUFD both twins IUFD both twins IUFD both twins

T1 twin 1; T2 twin 2; GA gestational age; P parity; Ivf in vitro fertilisation; spont spontaneous pregnancy; IUGR intrauterine growth retardation; IUFD intrauterine fetal death; PTL preterm labour. * For some women, there were multiple admissions with sometimes different causes.

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F. DEMARIA ET AL.

RESULTS During the study period, 1242 twin pregnancies were seen at our centre: 954 were dichorionic diamniotic, 269 monochorionic diamniotic and 19 monoamniotic. Characteristics and management of the 19 MATP are summarised in Table 1. Perinatal outcome for live births is summarised in Table 2 and causes of fetal deaths are reported in Table 3. The mean gestational age at delivery was 31 [5] weeks and the vaginal delivery rate was 53% (fetal deaths included). Three of the 19 pregnancies involved serious congenital malformations: one omphalopagus conjoined twins (case 15) and two pregnancies in which one of the twins had anencephaly (case 16) and acardia (case 18). Four pregnancies were both twins died in utero ended by vaginal delivery, with no mechanical complications. One of these occurred at 24 weeks (case 17), with cord entanglement and fetal hydrops. Two others occurred at 26 weeks (cases 16 and 18), both with probable twin twin transfusion syndromes. Tocolysis at 24 weeks failed for another woman (case 19) and both twins died during

labour (we do not perform caesarean sections before 25 weeks). In a fifth pregnancy (case 2), second twin was found to be dead in utero at 29 weeks. The mother was hospitalised for surveillance and underwent a caesarean four weeks later for fetal heart rate anomalies in the remaining twin. At delivery, the cords were found to be knotted and tangled. In total, there were nine fetal deaths (24%) from five pregnancies, all between 24 and 29 weeks. After exclusion of the pregnancies where both twins died in utero, the rate of vaginal delivery was 40% (6/15). The neonatal results in this subgroup were similar in the vaginal delivery and caesarean groups, in particular for the number of NICU transfers (6/12 vs 12/17, P 0.26), neonatal distress (6/12 vs 14/17, P 0.14) and neonatal deaths (2/12 and 1/17, P 0.35). Three children died in the NICU because of their extreme prematurity and type IV intraventricular haemorrhages. In all, both twins survived the perinatal period in 12 of 19 pregnancies (63%), and one of the pair survived in two more pregnancies. Overall, there were 26 survivors of 38 twins (68%).

Table 2. Perinatal outcome of live births. Case Sex Weight (g) GA at no. delivery 1 2 3 4 5 6 7 8 F F M M M F F F T1 T2 T1 T2 T1 T2 T1 T2 T1 T2 T1 T2 T1 T2 T1 T2 T1 T2 T1 T2 T1 T2 T1 T2 T1 T2 T1 T2 T1 T2 800 840 1990 740 1740 1590 2780 2300 1130 790 1140 950 1580 1800 2310 1700 2410 1680 890 1050 2850 2630 2400 2650 1200 1100 1310 1180 2800 2600 25 33 32 35 28 28 35 34 Onset of labour and mode of delivery Days in NICU T1 4 T2 9 NN outcome

spontaneous onset, VD abnormal FHR for T1 at 33, elective CS spontaneous onset, VD spontaneous onset, VD abnormal FHR T2 (IUGR), elective CS abnormal FHR T2 (IUGR), elective CS induced labour for moderate IUGR T1, VD induced labour for abnormal FHR T1 (twin twin syndrome), CS for dystocia spontaneous onset, VD abnormal FHR T1, elective CS induced labour at term, CS for dystocia spontaneous onset, CS for abnormal FHR T2, spontaneous onset, CS for chorioamnionitis and dystocia spontaneous onset, VD conjoined twins, elective CS

T1 neonatal death at day 4 with IVH 4 T2 neonatal death at day 9 with IVH 4 normal for T1/ T2 fetal death at 29 weeks normal for both twins normal for both twins

T1 T2 T1 T2

8 8 48 48

normal for both twins normal for both twins normal for both twins

T1 7 T2 T1 T2 T1 T2 7 5 9 69 23

normal for both twins

9 10 11 12 13 14 15

F F F M F F F

34 29 37 37 28 32 38

normal for both twins T1 IVH 3: normal outcome T2 neonatal death at day 23 with IVH 4 normal for both twins normal for both twins

T1 T2 T1 T2 T1 T2

56 56 5 5 3 3

normal for both twins normal for both twins alive and well after surgery

GA gestational age; VD vaginal delivery; CS caesarean section; FHR fetal heart rate; IVH intracerebral ventricular haemorrhage; NICU neonatal intensive unit care.

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PERINATAL MORTALITY REMAINS VERY HIGH IN MONOAMNIOTIC TWIN PREGNANCIES Table 3. Cause of intrauterine fetal death. Case Sex no. 2 16 17 F F F Weight (g) T2 T1 T2 T1 T2 T1 T2 T1 T2 740 940 900 1220 420 900 720 620 520 GA at death 29 26 24 Cause of fetal deaths

25

18 19

M F

26 24

entanglement and knots in the cords T2 anencephaly and hydrops fetalis twin twin syndrome fetal deaths both twins at 24 weeks after premature rupture of membranes; T1, hydrops fetalis, cord entanglement T2, acardia twin twin syndrome both twins died during labour

GA gestational age; IUGR intrauterine growth retardation.

DISCUSSION Perinatal mortality in monoamniotic twins remains very high even with intensified monitoring in a tertiary level centre. The principal reason for this high level of perinatal mortality is the number of fetal deaths occurring before 30 weeks and very preterm births. These poor results do not appear to be associated with the type of delivery. Most data concerning monoamniotic twins are rather old; they often involve only postnatal diagnosis (with numerous home deliveries) and sometimes lack histologic (placental) confirmation of the diagnosis.1,2 Moreover, the largest series are very heterogeneous: they include cases from several centres and it is not clear that all cases from all centres were considered or from what gestation. Only the recent study by Allen et al.5 is comparable to ours: it evaluates all consecutive cases diagnosed antenatally in a single centre. The substantial differences in monitoring and perinatal results found by reviews compared with publications reporting several cases and individual case reports explain the difficulty in interpreting the results about pregnancy management, type of delivery and perinatal mortality.5,12 Congenital anomalies are reported to be frequent in monoamniotic twins between 10% and 15% according to series, composed principally of terata and conjoined twins (11% in our series).2,3,5,12 14 The other dominant complication are cord entanglements and knots found at a rate of 58% in Quigleys series and 48% (9 for 19 pregnancies) in ours.14 These may cause cord compression followed by asphyxia, thus leading to fetal death. Sutter15 reported that 75% of deaths were related to cord complications (compared with three cases among nine fetal deaths in our series). These complications occur, mainly during the first two trimesters, because of the free movement of the two fetuses in a single amniotic cavity. Thus, the latest of the fetal deaths among the 24 monoamniotic twin pregnancies reported by Carr et al.9 occurred at 30 weeks. At 32 weeks among the 20 cases reported by Tessen and Zlatnik3 and at 29 weeks among the 25 cases
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reported by Allen et al.5 We found no double fetal deaths after 26 weeks, although a single death occurred at 29 weeks. Perinatal mortality is very high in our series, 12 cases in 38 births (or 32%), much higher than the 0.7% in the general pregnant population in France.16 This perinatal mortality rate is similar to those reported by Lumme and Saarikoski6 in their review (28 47%). However, it is much higher than that found in the other recent study of cases followed in a single centre (12%).5 This difference is explained in part by the presence of only a single fetal death for cord causes in Allens study, compared with three in ours. In all the other deaths, at least one of the twins had a malformation or fetal hydrops. In addition, there were two pregnancies with twin twin transfusion syndromes, which have previously been described in monoamniotic twins.12 This fetal mortality rate of 24% is twice that found in Allens series and systematic review (10% and 13% of fetal deaths, respectively).5 Antenatal surveillance probably does not explain this difference since eight of the nine fetal deaths in our series occurred between 24 and 26 weeks, and our surveillance during that period seems comparable to that in the study by Allen et al. All mothers of twins born before 34 weeks had received prophylactic corticosteroid administration except for one woman (case 13) who underwent a caesarean delivery within four hours of in utero transfer for preterm labour. These differences may have arisen by chance due the small number of cases. Accordingly, the conclusions by Allen et al. that regular fetal surveillance and appropriate steroid administration result in good perinatal outcome and that the risk of fetal death is lower than previously reported seem too optimistic. One reason that vaginal delivery is considered to be contraindicated is the description by some authors of obstetric accidents of collision and impaction of the twins in the maternal pelvis.1,8,17 None of our vaginal deliveries involved this type of problem. Possible cord complications at the moment of delivery are also mentioned as a reason for caesarean sections. In our series, one case required us to cut cord of the second twin because its cord was wrapped tightly around the neck of the other twin with no consequences to either baby (case 4). The number of vaginal deliveries with live infants (6 of 15 pregnancies) in our series is too small to allow us to assert that these accidents are rare. Nonetheless, we found that trial of vaginal delivery (10 of the 15 pregnancies without double fetal deaths) caused no related perinatal complication. The essential element in management of monoamniotic twins is the prevention of fetal deaths, but it is difficult to reach a consensus about specific protocols. Some recommend systematic pulmonary maturation with corticosteroids, repeated weekly, and delivery by caesarean at 32 weeks.8 Other teams, like ours, have not found excess mortality after 30 32 weeks and continue the pregnancy beyond 35 weeks if possible.3,5,9,12,13 Most authors do not

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F. DEMARIA ET AL. sonographic diagnosis, detection of cord entanglement, and obstetric management. Obstet Gynecol 1995;86:218 222. Beasley E, Megerian G, Gerson A, Roberts NS. Monoamniotic twins: case series and proposal for antenatal management. Obstet Gynecol 1999;93:130 134. Carr SR, Aronson MP, Coustan DR. Survival rates of monoamniotic twins do not decrease after 30 weeks gestation. Am J Obstet Gynecol 1990;163:719 722. Rodis JF, Vintzileos AM, Campbell WA, Deaton JL, Fumia F, Nochimson DJ. Antenatal diagnosis and management of monoamniotic twins. Am J Obstet Gynecol 1987;157:1255 1257. Papiernik E, Keith L, Oleszczuk JJ, Cervantes A. What interventions are useful in reducing the rate of preterm delivery in twins? Clin Obstet Gynecol 1998;41:12 23. Su LL. Monoamniotic twins: diagnosis and management. Acta Obstet Gynecol Scand 2002;81:995 1000. Dubecq F, Dufour P, Vinatier D, et al. Monoamniotic twin pregnancies. Review of the literature, and a case report with vaginal delivery. Eur J Obstet Gynecol Reprod Biol 1996;66:183 186. Quigley J, Rochester N. Monoamniotic twin pregnancy. A case record with review of the literature. Am J Obstet Gynecol 1935:354 362. Sutter J, Arab H, Manning FA. Monoamniotic twins: antenatal diagnosis and management. Am J Obstet Gynecol 1986;155:836 837. Beaumel C, Doisneau L, Vatan M. La situation demographique en 1999. INSEE Resultats. Paris: Demographie-Societe, 2001. McLeod FN, McCoy DR. Monoamniotic twins with an unusual cord complication. Case report. Br J Obstet Gynaecol 1981;88:774 775.

perform caesarean routinely, but vaginal delivery is rarely recommended or accepted. Our view is that vaginal delivery is acceptable if the twins are of similar size, the first twin is in a cephalic presentation and the fetal Doppler is normal. At 36 weeks, we advocate either induction of labour or caesarean delivery with paediatricians present at delivery.

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9.

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References
1. Raphael S. A review of the literature and a report of 5 new cases. Am J Obstet Gynecol 1961:323 330. 2. Simonsen M. Monoamniotic twins. Acta Obstet Gynecol Scand 1966;45:43 52. 3. Tessen JA, Zlatnik FJ. Monoamniotic twins: a retrospective controlled study. Obstet Gynecol 1991;77:832 834. 4. Colburn DW, Pasquale SA. Monoamniotic twin pregnancy. J Reprod Med 1982;27:165 168. 5. Allen VM, Windrim R, Barrett J, Ohlsson A. Management of monoamniotic twin pregnancies: a case series and systematic review of the literature. Br J Obstet Gynaecol 2001;108:931 936. 6. Lumme RH, Saarikoski SV. Perinatal deaths in twin pregnancy: a 22year review. Acta Genet Med Gemellol (Roma) 1988;37:47 54. 7. Aisenbrey GA, Catanzarite VA, Hurley TJ, Spiegel JH, Schrimmer DB, Mendoza A. Monoamniotic and pseudomonoamniotic twins:

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12. 13.

14. 15. 16. 17.

Accepted 4 September 2003

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