Consequences of in-utero death in a twin pregnancy

doi:10.1016/S0140-6736(00)02215-7

The Lancet

Volume 355, Issue 9215, 6 May 2000, Pages 1597-1602

https://doi.org/10.1016/S0140-6736(00)02215-7 Get rights and content

Summary

Background

Twins have a higher mortality and morbidity than singletons and, among twins, the surviving co-twin of a fetus that dies in utero is particularly at risk. We did a cohort study to quantify mortality and serious morbidity in co-twin survivors of fetuses that died in utero.

Methods

We collected data of all registered twin births in England and Wales between 1993 and 1995 in which one twin was registered as having died in utero. Copies of all death certificates of these fetuses and death certificates of live-born co-twins of fetuses that died in utero were obtained from the Office for National Statistics. A questionnaire was sent to the general practitioners of all surviving co-twins to find out if the child had any disability.

Findings

There were 434 fetal death/live-birth same-sex twin pairs. Among the live births, there were 59 neonatal deaths, seven postneonatal deaths (first 28 days), and five infant deaths (< 1 year). In three of the five deaths, the cause of death was cerebral palsy. Ten of the children who survived infancy were lost to follow up. Responses were received from general practitioners for 241 of 353 survivors (68% response). Of the 241 respondents, 23 had cerebral palsy and 28 had other cerebral impairment. Among the children who survived to infancy, the prevalence of cerebral palsy was 106 (95% Cl 70–150) per 1000 and prevalence of other cerebral impairment was 114 (80–160) per 1000. There were 163 fetal death/live-birth different-sex twin pairs. Of the live births, 13 died in the neonatal period and four were lost to follow up. Of the 146 survivors, responses were received from the general practitioners for 102 (70% response). Three of the 102 had cerebral palsy and 12 had other cerebral impairment. The prevalence of cerebral palsy was 29 (95% Cl 6–83) per 1000 and of other cerebral impairment 118 (62–196) per 1000 infant survivors.

Interpretation

The live-birth co-twin of a fetus that died in utero is at increased risk of cerebral impairment, the overall risk is 20% (95% Cl 16–25). The gestational-age-specific prevalence of cerebral palsy after fetal death of the co-twin is much higher than that reported for the general twin population.

Introduction

Twins have a greater risk of fetal and infant death than singleton births.1, 2, 3 At particular risk among twins are monozygotic twins who have poorer survival rates than dizygotic twins.4, 5 Not only is fetal and neonatal mortality higher in monozygotic compared with dizygotic twins, but in a twin pregnancy, if one fetus dies in utero, reports from case series have shown that the surviving co-twin is at increased risk of major morbidity such as cerebral palsy, learning disabilities, and gut atresias.6, 7, 8, 9, 10, 11, 12 The increased risk for cerebral impairment in the surviving co-twin has also been shown in population-based registers of cerebral palsy.13, 14 In the case reports, the risk seems to be confined to monozygotic twins, specifically monochorionic ones.

We aim to quantify the risk of serious morbidity in the survivor of a twin pregnancy when the co-twin died in utero (a fetal death).

Section snippets

Patients and procedure

The study cohort comprised all registered twin births in England and Wales between 1993 and 1995. The routinely published data on registrations of fetal deaths, live births, and infant deaths in twins according to sex were used.

The Office for National Statistics (ONS) provided copies of the Fetal Death Registration certificates of all fetal deaths from twin pregnancies. The ONS also provided the Death Registration certificates of those twins who were live born but who subsequently died and

Results

There were 25 506 twin pairs in England and Wales in 1993-95. Of these, 153 (0·6%) pairs died in utero, 613 (2·4%) pairs had one co-twin who died (fetal death/livebirth pairs), and 24 739 (97·0%) pairs were both live births.

Table 1 shows these data according to whether the twins were of the same sex. When comparing twins of the same sex with twins of different sex, the risk ratio for both twins dying in utero was 6·17 (95% CI 3·34–11·38; p < 0·0001) and one death in utero of one of the twins

Discussion

There is only one published study¹⁷ that provides specific cerebral palsy prevalence rates for singleton and twin gestational age based on a population of 102 059 singletons and 2367 twins (table 4). It is evident that, when one twin dies in utero, for all gestational age-groups, the risk of cerebral-palsy among the same-sex twins is higher than in different-sex twins.

Our study aimed, primarily, to quantify the risk of serious morbidity in the surviving twin when the co-twin died in utero. We

References (35)

EL Potter
Twin zygosity and placental form in relation to the outcome of the pregnancy
Am J Obstet Gynecol
(1963)
M Melnick
Brain damage in a survivor after in utero death of monozygous co-twin
Lancet
(1977)
JA Enbom
Twin pregnancy with intrauterine death of one twin
Am J Obstet Gynecol
(1985)
AAGL Schinzel et al.
Monozygotic twinning and structural defects
J Pediatr
(1979)
R Bejar et al.
Antenatal origin or neurologic damage in newborn infants: II multiple gestations
Am J Obstet Gynecol
(1990)
SG Weig et al.
Patterns of cerebral injury and clinical presentation in the vascular disruptive syndrome of monozygotic twins
Pediat Neurol
(1995)
CM Moore et al.
Intrauterine disseminated intravascular coagulation: a syndrome of multiple pregnancy with a dead twin fetus
J Pediatr
(1969)
RF Ellis et al.
The Northwestern University multihospital twin study II: mortality of first versus second twin
Acta Genet Med Gemellol
(1979)
PPS Nylander
Perinatal and infant mortality in twins
Acta Genet Med Gemellol
(1979)
BJ Botting et al.
Recent trends in the incidence of multiple births and associated mortality
Arch Dis Child
(1987)

MC Myrianthopoulos

An epidemiologic survey of twins in a large prospectively studied population

Am J Hum Genet

(1970)

K Yoshida et al.

Outcome of the surviving cotwin of a fetus papyraceus or of a dead fetus

Acta Genet Med Gemellol

(1986)

ZJ Hagay et al.

Management and outcome of multiple pregnancies complicated by the antenatal death of one fetus

J Reprod Med

(1986)

W Szymonowicz et al.

The surviving monozygotic twin

Arch Dis Child

(1986)

L Fusi et al.

Twin pregnancy complicated by single intrauterine death: problems and outcome with conservative management

Br J Obstet Gynaecol

(1990)

RL Anderson et al.

Central system damage and other anomalies in surviving fetus following second trimester antenatal death of a cotwin: report of four cases and literature review

Prenatal Diagnosis

(1990)

JK Grether et al.

Twinning and cerebral palsy: experience in four Northern Californian counties, births 1983 through 1985

Pediatrics

(1993)

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Twins have a significantly higher risk of stillbirth, preterm delivery, perinatal morbidity and mortality. Single intrauterine fetal death is a relatively common and devastating occurrence for families and clinicians alike. Monochorionic twins are up to 13 times more likely to suffer an intrauterine death when compared to singletons. Additionally, longer term neurological sequelae affect monochorionic twins disproportionately. The timing of the death of the co-twin and the fetal order at the time of death have an impact on the outcome for the survivor. The risk in dichorionic pregnancies is lower with respect to neurological injury; however, the risk of prematurity remains high, particularly where the leading twin dies in utero. Recent published studies report lower rates of neurological injury in monochorionic twin pregnancies that incur an intrauterine fetal death after foetoscopic laser ablation for twin-to-twin transfusion syndrome – an important finding for patient counselling.
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This study aimed to describe fetal magnetic resonance neuroimaging findings in pregnancies complicated by single fetal demise after laser photocoagulation for twin-twin transfusion syndrome.
This was a single-center retrospective analysis of a cohort of prospectively collected patients in a monochorionic twin registry who had fetoscopic laser photocoagulation for twin-twin transfusion syndrome with single fetal demise at follow-up. Magnetic resonance neuroimaging was offered 3 to 4 weeks after the demise to assess for potential neurologic sequelae. Magnetic resonance images were interpreted by 2 board-certified neuroradiologists and classified as normal, mildly abnormal, or severely abnormal. The groups were compared on the basis of recipient vs donor demise using the Fisher exact test and Mann-Whitney U test. Multivariate logistic regression was performed to determine risk factors for abnormal magnetic resonance neuroimaging.
In 378 laser photocoagulation procedures, 64 cases (16.9%) of single demise were identified (36 in the donor group and 28 in the recipient group). Of note, 6 patients had rupture of membranes with nonviable delivery (3 from each group). Moreover, 40 patients (69%) underwent magnetic resonance imaging. Of those patients, 12 (30%) had abnormal findings: 10 (83%) were associated with mild changes, and 2 (17%) were associated with severe findings. Abnormal magnetic resonance neuroimaging was seen in 3 of 22 patients (14%) after donor demise and 9 of 18 patients (50%) after recipient demise (P=.02). Logistic regression revealed that recipient vs donor demise was an independent risk factor for abnormal magnetic resonance imaging. In addition, 2 pregnancies with severe magnetic resonance imaging findings had complicated courses.
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Citation Excerpt :
Twin pregnancies are associated with increased stillbirth rates, a thirteenfold increase in monochorionic, and a fivefold increase in dichorionic twins compared with singleton pregnancies [1,2]. Single intrauterine fetal death (IUD) occurs in approximately 6% of all twin pregnancies [3]. The risk of death for the co-twin after IUD is 15% and 3% for monochorionic (MC) and dichorionic (DC) twin pregnancies, respectively.
Thanks to shared circulation in monochorionic twins, single intrauterine fetal death (IUD) may lead to acute feto-fetal transfusion (aFFTR). The objective of the study was to describe our model of aFFTR simulation after IUD in monochorionic (MC) twins.
Prospective study analyzed 99 fresh MC placentas with the physiological course. A specially designed protocol was used for the preparation and analysis of the placentas. A pair of infusion sets fixed together using a mechanical mercury sphygmomanometer cuff was connected to the cannulated umbilical arteries. The tonometer was pressurized up to 30 and 40 mmHg. A positive finding of aFFTR was determined as the amount exceeding 1 ml of dye flowed out of the umbilical cord simulating a dead fetus. The number and types of anastomoses, types, and distances between cords insertions, and the size of the placental areas for each fetus were also statistically analyzed. The placental angioarchitecture with and without proven aFFTR was statistically compared, odds ratio (OR) and multivariable logistic analysis were performed.
A total of 49/99 (49.5%) cases of aFFTR was proven, and the average transfusion time of 1 ml was 30 s (19–46 s). aFFTR was present in 49/78 (62.8%) of placentas with arterio-arterial (AA) anastomosis. The median diameter of AA anastomoses with the present, and absent aFFTRF was 2.0 mm and 1.0 mm, respectively. The proven interfetal transfusion was 8%, 31%, and 61% in AA anastomoses with a diameter below 0,5 mm, 0,5–1,5 mm, and above 1,5 mm, respectively (p < 0,001). AA anastomoses diameter >1.5 mm had OR of 44.2 (95% CI 5.54–352.39). In the case of coexistence of AA anastomosis and umbilical cord distance ≤5th percentile, the aFFTRF occurred in 90.9%.
The potential risk of aFFTR in monochorionic twins is mainly due to the presence and nature of AA anastomoses. The diameter and length of the vessels play a crucial role, which is clinically related to the distance of the umbilical cords insertions.
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ArticlesConsequences of in-utero death in a twin pregnancy

Summary

Background

Methods

Findings

Interpretation

Introduction

Section snippets

Patients and procedure

Results

Discussion

Am J Obstet Gynecol

Lancet

Am J Obstet Gynecol

J Pediatr

Am J Obstet Gynecol

Pediat Neurol

J Pediatr

The Northwestern University multihospital twin study II: mortality of first versus second twin

Acta Genet Med Gemellol

Perinatal and infant mortality in twins

Acta Genet Med Gemellol

Recent trends in the incidence of multiple births and associated mortality

Arch Dis Child

An epidemiologic survey of twins in a large prospectively studied population

Am J Hum Genet

Outcome of the surviving cotwin of a fetus papyraceus or of a dead fetus

Acta Genet Med Gemellol

Management and outcome of multiple pregnancies complicated by the antenatal death of one fetus

J Reprod Med

The surviving monozygotic twin

Arch Dis Child

Twin pregnancy complicated by single intrauterine death: problems and outcome with conservative management

Br J Obstet Gynaecol

Central system damage and other anomalies in surviving fetus following second trimester antenatal death of a cotwin: report of four cases and literature review

Prenatal Diagnosis

Twinning and cerebral palsy: experience in four Northern Californian counties, births 1983 through 1985

Pediatrics

Articles
Consequences of in-utero death in a twin pregnancy