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Please cite this paper as: Ong S, Zamora J, Khan K, Kilby M. Prognosis for the co-twin following single-twin death: a systematic review. BJOG 2006;113:
992998.
Introduction
Intrauterine death of one fetus in a twin pregnancy is uncommon in the second or third trimester. However, the consequences to the surviving co-twin can be profound, especially in
monochorionic twins. These may include co-twin death, survival with cerebral impairment or preterm labour with its
sequelae.1 Recent changes in technology and prenatal ultrasound scan have meant that clinicians can diagnose this condition and have the option to intervene. Decision making
should be informed by existing prognostic evidence, but individual studies including data from case reports, follow up of
cohorts and twin registries tend to have imprecise results as the
event is uncommon. More precise information may be
992
Methods
The project was based on a prospective protocol developed
using widely recommended methods for systematic review of
observational studies.4,5
extracted to allow computation of rates, stratifying by chorionicity and other prognostic factors wherever possible.
Data synthesis
The extracted data were tabulated to allow qualitative inspection for clinical and methodological heterogeneity. Heterogeneity of rates was explored graphically using Forrest plot and
examined statistically using the chi-square test.7 Data from
individual studies were pooled within subgroups defined by
prognosis and outcome.8
Results
Identification of the literature
The electronic search yielded a total of 632 citations, of which
54 were considered potentially relevant. Examination of the
full manuscripts revealed that 26 did not meet the selection
criteria. This was mostly because the case series reported less
than five cases. Thus, a total of 28 primary studies were
selected for review1,2,934 (Figure 1, Table 1). One study was
derived from data obtained from three separate institutions.12
All other studies published data either from follow up of case
series (cohort studies) or from registries.
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Ong et al.
Citations excluded.
Did not meet selection criteria
n = 578
Studies excluded.
Did not meet selection criteria
n = 26
Outcome:
in utero death
Outcome:
neurological
abnormality
Outcome:
preterm delivery
n = 19
(904 pregnancies)
n = 17
(267 pregnancies)
n = 11
(100 pregnancies)
Figure 1. Flow diagram of study selection for systematic review. Outcome of surviving co-twin following single-twin death after 14 weeks of gestation.
Preterm delivery
994
Number of
cases
Data extractable
In utero Neurological Preterm
death abnormality delivery
Case series
Abdal-Khalig and
Sobande33
Aslan et al.11
Axt et al.10
Eglowstein and
DAlton30
Fusi and Gordon21
Gaucherand
et al.26
Ishimatsu et al.32
Jou et al.29
Kilby et al.15
Malinowski
et al.23
Malinowski
et al.9
Lin et al.24
Liu et al.34
Petersen and
Nyholm16
Prompeler et al.17
Saito et al.18
Santema et al.19
Sonneveld and
Correy31
van Heteren
et al.20
Wang et al.25
Woo et al.2
Zorlu et al.22
Birth register
Rydhstrom and
Ingemarsson
(1993)27
Rydhstrom (1994)28
Pharoah and Adi1
Glinianaia et al.13
Johnson and
Zhang14
Other
Bajoria et al.12
35
25
7
20
N
Y
Y
Y
Y
Y
Y
Y
Y
16
9
Y
Y
Y
Y
Y
Y
15
12
20
15
Y
Y
Y
N
Y
Y
Y
N
Y
Y
N
N
11
17
3
12
N
N
Y
Y
N
Y
Y
N
Y
43
30
29
25
N
N
Y
Y
N
Y
Y
N
N
Y
N
Y
11
9
7
9
N
Y
Y
Y
Y
N
Y
Y
N
206
326
597
164
3599
Y
Y
Y
Y
N
N
N
N
N
N
N
N
92
995
N, no; Y, yes.
Management strategy
Ong et al.
10
18
23
15
Congenital anomalies
13
Follow up
28
Objective outcome
Death
28
10
18
Neurological
20
40
60
80
100%
No/unclear/not reported
Yes
Figure 2. Quality assessment of studies in systematic review.
Preterm delivery
Monochorionic
Monochorionic
Abdal-khalig and Sobande
10
Axt et al.
12
Bajoria et al.
30
Eglowstein and DAlton
21
Fusi and Gordon
26
Gaucherand et al.
32
Ishimatsu et al.
29
Jou et al.
15
Kilby et al.
9
Malinowski et al.
16
Petersen and Nyholm
19
Santema et al.
31
Sonneveld and Correy
2
Woo et al.
33
10
Axt et al.
12
Bajoria et al.
30
Eglowstein and DAlton
21
Fusi and Gordon
26
Gaucherand et al.
32
Ishimatsu et al.
29
Jou et al.
24
Lin et al.
16
Petersen and Nyholm
18
Saito et al.
20
van Heteren
25
Wang et al.
2
Woo et al.
n = 19
n=4
n = 50
n=8
n=8
n=6
n = 12
n = 12
n=7
n=4
n=5
n = 13
n=7
n=5
Monochorionic
n=4
n = 21
n=8
n=6
n=5
n=9
n=8
n=7
n=4
n = 14
n=9
n=9
n=4
10
Axt et al.
21
Fusi and Gordon
26
Gaucherand et al.
32
Ishimatsu et al.
29
Jou et al.
16
Petersen and Nyholm
18
Saito et al.
31
Sonneveld and Correy
20
van Heteren
25
Wang et al.
2
Woo et al.
Dichorionic
10
Axt et al.
12
Bajoria et al.
30
Eglowstein and DAlton
21
Fusi and Gordon
26
Gaucherand et al.
32
Ishimatsu et al.
24
Lin et al.
16
Petersen and Nyholm
18
Saito et al.
2
Woo et al.
Dichorionic
Abdal-khalig and Sobande
10
Axt et al.
12
Bajoria et al.
30
Eglowstein and DAlton
21
Fusi and Gordon
26
Gaucherand et al.
32
Ishimatsu et al.
15
Kilby et al.
9
Malinowski et al.
16
Petersen and Nyholm
1
Pharoah and Adi
28
Rydhstrom
19
Santema et al.
31
Sonneveld and Correy
2
Woo et al.
33
n = 16
n=3
n = 42
n = 11
n=8
n=2
n=3
n = 13
n=7
n=7
n = 181
n = 47
n = 16
n=9
n=1
n=3
n=7
n=5
n=9
n=7
n=5
n = 17
n=4
n = 11
n=7
n=2
Dichorionic
10
n=3
n = 33
n = 11
n=6
n=2
n=3
n=3
n=6
n = 10
n=1
Axt et al.
21
Fusi and Gordon
32
Ishimatsu et al.
16
Petersen and Nyholm
18
Saito et al.
31
Sonneveld and Correy
n=2
n=6
n=2
n=6
n=6
n=1
Undefined
Zorlu et al.
0.0
0.2
0.4
0.6
0.8
1.0
22
n=9
0.0
0.2
0.4
0.6
0.8
1.0
0.0
0.2
0.4
0.6
0.8
1.0
Figure 3. Forrest plots of outcomes for the surviving co-twin following single-twin death according to chorionicity.
996
Study
Monochorionic
n/ N
Dichorionic
n/ N
OR (fixed)
95% CI
OR (fixed)
95% CI
Co-twin death
Abdal-khalig and Sobande
10
Axt et al.
30
Eglowstein and DAlton
26
Gaucherand et al.
32
Ishimatsu et al.
15
Kilby et al.
16
Petersen and Nyholm
31
Sonneveld and Correy
9
Malinowski et al.
19
Santema et al.
2
Woo et al.
12
Bajoria et al.
21
Fusi and Gordon
33
0/19
0/4
0/8
0/6
0/12
0/7
0/5
0/7
1/4
2/13
1/5
13/50
1/8
0/16
0/3
0/11
0/2
0/3
0/13
0/7
0/9
0/7
0/16
0/1
1/42
1/8
Not estimable
Not estimable
Not estimable
Not estimable
Not estimable
Not estimable
Not estimable
Not estimable
6.43 (0.21201.07)
7.17 (0.31163.82)
1.00 (0.0240.28)
14.41 (1.80115.53)
1.00 (0.0519.36)
148
138
6.04 (1.8419.87)
0/4
0/4
2/21
1/8
1/4
4/9
2/14
3/6
1/5
1/7
0/6
0/1
0/33
0/11
0/3
0/3
0/10
1/6
0/2
0/3
Not estimable
Not estimable
8.59 (0.39188.26)
4.60 (0.16128.54)
3.00 (0.09102.05)
5.73 (0.23142.55)
4.20 (0.1897.55)
5.00 (0.3472.77)
1.67 (0.0558.28)
1.62 (0.0551.11)
82
78
4.07 (1.3212.51)
5/5
6/9
13/17
2/3
2/4
4/7
3/6
1/2
2/6
2/2
1/1
4/6
45
23
Neurological abnormality
16
Preterm delivery
16
Ishimatsu et al.
Saito et al.
Axt et al.
18
10
31
11.00
2.00
6.50
0.33
0.33
0.67
(0.43284.30)
(0.0944.35)
(0.8549.69)
(0.0112.82)
(0.0112.82)
(0.076.41)
1.91 (0.705.21)
0. 0 0 1 0. 0 1
0. 1
m o n oc h o r i o n i c b e t t e r
10
100
1000
d i c h o r i o n i c b et t e r
Figure 4. Meta-analysis of studies providing information of co-twin death, neurological abnormality and preterm delivery following single-twin
death after 14 weeks of gestation. A comparison of odds between monochorionic and dichorionic pregnancies.
1 Pharoah PO, Adi Y. Consequences of in-utero death in a twin pregnancy. Lancet 2000;355:1597602.
2 Woo HH, Sin SY, Tang LC. Single foetal death in twin pregnancies:
review of the maternal and neonatal outcomes and management.
Hong Kong Med J 2000;6:293300.
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References
Ong et al.
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