Author
Information
Thakur HS*,
Gupta AS**.
(* Assistant Professor, ** Professor. Department of Obstetrics
and Gynecology, Seth G.S. Medical
College & K.E.M. Hospital , Mumbai ,
India
Abstract
A patient
of twin to twin transfusion syndrome presented in early second trimester with
intrauterine fetal death of one of the twins. Pregnancy could be continued till
32 weeks with monitoring of the coagulation profile and ultrasound (USG). A
good perinatal outcome was achieved for the surviving twin. This case
highlights the fact that individualization of treatment during ANC period does
lead to good neonatal outcome in the surviving twin.
Introduction
Intrauterine
fetal death (IUFD) of one twin in a case of twin to twin transfusion syndrome
(TTTS) is relatively common and known complication of twin gestation. [1]
TTTS is a major cause of perinatal morbidity and mortality when complicated by
death of one twin.[2] Surviving twin is at risk of prematurity and
also neonatal death.[2, 3] Expectant management can be given in
cases of severe prematurity until fetal lung maturation occurs with strict
monitoring.
Case Report
A 23 yr
old primigravida presented in emergency with USG diagnosis of a monochorionic diamniotic
twin with twin to twin transfusion syndrome with 23 weeks of gestation age and
IUFD of one twin. She had an earlier USG scan of 18 weeks of gestation which
showed presence of cardiac activity in both twins, the biometry showed a lag of
3 weeks between the twins. USG Doppler done had diagnosed twin to twin
transfusion signals in a monochorionic diamniotic placentation. On presentation the couple expressed a desire
to salvage the surviving twin after all risks to the mother and the surviving
twin with continuing the pregnancy were explained. She was admitted, given bed
rest in the left lateral position and pregnancy was monitored with biochemical,
hematological and serological parameters. On obstetric USG polyhydramnios in
the surviving twin was detected, the amniotic fluid index (AFI) being 32 cm at
23 weeks of pregnancy. Maternal fasting and postprandial blood sugars were
normal. Obstetric USG showed no congenital malformation. Oral indomethacin was
given for 48 hours and was stopped as the AFI reduced to 27 cm. Fetal 2D ECHO
was done which was normal.
Serial monitoring of
CBC with platelets, coagulation profile, and obstetric USG and doppler were the
focus of her antenatal monitoring. The biometry on serial USG showed no growth
retardation in the live twin. Fetoplacental and uteroplacental pulsatility
index (PI) on Doppler were also normal. Middle cerebral artery Doppler studies
of the surviving twin was within range for the gestational age. She was given
prophylactic steroids as intramuscular dexamethasone 6 mg 12 hourly 4 doses to
achieve fetal lung maturity at 28 weeks of gestation as she was predisposed to
preterm labor which eventually did occur.
Investigation Chart
Day from
admission
|
HB
(gm%)
|
Platelets
(Lakhs /mm3)
|
PT
(test/control)
|
INR
|
Plasma
Fibrinogen
|
D1
|
8.4
|
2.8
|
8.7/9.7
|
0.91
|
501
|
D3
|
7.5
|
2.6
|
|||
D5
|
8.6
|
2.13
|
12.2/13.5
|
0.89
|
300
|
D8
|
8.6
|
2.64
|
12/13.5
|
0.87
|
220
|
D11
|
9.6
|
2.21
|
11.9/13.5
|
0.87
|
200
|
D15
|
9.5
|
2.20
|
12.8/13.5
|
0.94
|
220
|
D17
|
9.4
|
2.14
|
12.7/13.5
|
0.94
|
240
|
D19
|
9.6
|
2.28
|
12.1/13.5
|
0.88
|
400
|
D22
|
9.2
|
2.36
|
11.6/13.5
|
0.87
|
200
|
D29
|
9.4
|
1.5
|
12/13.5
|
0.88
|
210
|
D31
|
9.4
|
2.34
|
12.1/13.5
|
0.87
|
210
|
D33
|
9.8
|
2.59
|
11.8/13.5
|
0.86
|
270
|
D36
|
9.8
|
2.09
|
13.5/13.5
|
0.98
|
350
|
D43
|
10.3
|
2.16
|
14.6/13.5
|
1.09
|
360
|
D48
|
10
|
2.14
|
13.9/13.5
|
1.02
|
320
|
D50
|
11
|
2.47
|
12.8/13.5
|
0.94
|
220
|
D52
|
10.7
|
2.29
|
13.2/13.5
|
0.92
|
200
|
D53
|
9.9
|
2.06
|
12/14
|
0.98
|
220
|
D61
|
10.2
|
2.5
|
12/14
|
0.83
|
The patient
took discharge on request on day 53 of admission (32 weeks of gestation). She returned
within 8 days of discharge on day 61 at 33 weeks of gestation in advanced
preterm labor. She delivered the first
live born male twin baby weighing 1.55 kg and second twin, a macerated male
abortus of 365 gm. APGAR score live born was 9/10 at 1 minute and also at 5
minutes. Surprisingly, the amniotic fluid volume in the live born twin was normal
and the polyhydramnios was seen in the amniotic sac of the dead twin. Placenta
on gross inspection was monochorionic, diamniotic. The neonate stayed in NICU
for a month and was then discharged. The neonate showed no signs of disseminated
intravascular coagulopathy and cerebral impairment either clinically or on post
delivery investigation by the coagulation profile and USG of the brain
respectively.
Figure 1.
Macerated abortus.
Figure 2. Live
premature twin.
Discussion
The RCOG
study on multiple pregnancies with death of a co-twin recommends that the
management should depend on chorionicity, gestation and time since death. They
recommend policy of increased surveillance with delivery at 37 weeks in
dichorionic pregnancies.[4]
According
to ACOG guidelines it is recommended that if the death is the result of fetal
anomaly itself rather than maternal or uteroplacental pathology and the
pregnancy is remote from term then expectant management may be suitable.[5]
About 10-15% of monochorionic pregnancies are complicates by TTTS as the
placentas are more likely to have unidirectional artery-vein communication.[6]
This anastomosis results in hypovolemia in the donor twin and circulatory
overload in the recipient twin. Thus the donor twin is anemic whereas the recipient
is polycythemic. Polyuria due to circulatory overload in the recipient twin
leads to polyhydramnios and hypovolemia in the donor twin results in a growth
restricted fetus and oligohydramnios in their respective amniotic sacs.
After the
single fetal death in a monochorionic pregnancy the risk to the surviving twin
of death or neurological abnormality is of the order of 12% and 18%
respectively.[6] There were a few unique features in our case. We
were able to continue the pregnancy for 61 days after the co twin demise so
pregnancy progressed from 23 weeks to 33 weeks whereas in an international
study, median interval between the diagnosis of fetal death and the delivery
was only 11 days.[2] Our patient took home a healthy neonate whose Apgar
scores were 9/10 both at 1 minute and 5 minutes and birth weight was 1.55 kg. The
live twin birth was followed by the stillborn twin. The interesting feature was
the polyhydramnios in the stillborn’s amniotic sac that was contradictory to
the USG finding. As the fetus ceases to produce urine after death we postulate
that this twin was the recipient and circulatory overload was the cause of its
demise. However, the reduction of AFI with a short course of indomethacin is
unexplainable as indomethacin which is a prostaglandin inhibitor works by
decreasing the urine output. Risk of coagulopathies and thromboembolic
phenomenon are reported in the surviving co-twin in its intrauterine environment
due to vascular anastomosis. It is postulated that the thromoplastin from the dead
twin enters the surviving twin’s circulation and induces coagulopathy.[7]
Close monitoring of the surviving twin was done with the aim to detect onset of
any coagulopathy and to detect any dense echoes in the fetal brain as the
reported incidence of cerebral impairment in the surviving twin in a TTTs is
about 20%.[8]
Conclusion
Expectant
management can be given to case of TTTs with co-twin death with strict
antenatal, hematological, biochemical and USG monitoring until fetal lung
maturation occurs.
References
- Chelli D, Methni A, Boudaya F, Marzouki Y, Zouaoui B, Jabnoun S, et al. Twin pregnancy with single fetal death: etiology, management and outcome. J Gynecol Obstet Biol Reprod (Paris). 2009;38(7):580-7.
- Aslan H, Gul A, Cebeci A, Polat I, Ceylan Y. The outcome of twin pregnancies complicated by single fetal death after 20 weeks of gestation. Twin Res. 2004;7:1-4.
- Browne I, Brustman L, Santos A. Intrauterine Fetal Death. In: Datta S, ed. Anesthetic and Obstetric Management of High-Risk Pregnancy. 3rd ed. New York: Springer publications. 2003. pp 515-28.
- Kilby M, Baker P, Critchley H, Field D, editors. Multiple pregnancy. London: RCOG Press; 2006, 283-6.
- American College of Obstetricians and Gynecologists (ACOG). Multifetal gestation: complicated twin, triplet, and high-order multifetal pregnancy. Washington (DC): American College of Obstetricians and Gynecologists (ACOG); 2004 Oct. p. 14-16 (ACOG practice bulletin; no. 144).
- Royal College of Obstetricians and Gynecologists. Greentop Guidelines number 51. Management of monochorionic twin pregnancy. Edition 1. December 2008.
- Benirschke K. Intrauterine death of a twin: mechanisms, implications for surviving twin, and placental pathology. Semin Diagn Pathol. 1993 Aug; 10(3):222-31.
- Pharoah PO, Adi Y. Consequences of in-utero death in a twin pregnancy. Lancet 2000 May 6; 355(9215):1597-602.
- Suzuki S, Sawa R, Yoneyama Y, Otsubo Y, Araki T.Fetal middle cerebral artery Doppler waveforms in twin-twin transfusion syndrome. Gynecol Obstet Invest. 1999; 48(4):237-40.
- Sallam MAG. Twin-to-twin transfusion syndrome (TTTS). Ain Shams Journal of Anesthesiology. 2011 Jan; Vol 4-1: 99-104.
- Zach T. Twin-to-Twin Transfusion Syndrome updated Jun 14, 2013 Available from: http://emedicine.medscape.com/article/271752
Citation
Thakur HS,
Gupta AS.