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      Monoamniotic Twins with One Fetal Anencephaly and Cord Entanglement Diagnosed with Three Dimensional Ultrasound at 14 Weeks of Gestation

      case-report

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          Abstract

          A 29-year-old pregnant woman with parity 0-0-0-0 was diagnosed with monoamniotic twin pregnancy discordant for anencephaly at 14 weeks gestation. Umbilical cord entanglement, which is an important cause of fetal death in monoamniotic twins, was confirmed by three-dimensional ultrasound. Cesarean section was performed at 34 weeks of gestation, and the normal newborn infant was discharged without any complications. We report a case of monoamniotic twin pregnancy discordant for anencephaly and diagnosed with cord entanglement by three-dimensional ultrasound at 14 weeks of gestation, and now report it along with a literature review.

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          Cord entanglement and perinatal outcome in monoamniotic twin pregnancies.

          Edward Dornan, B Thilaganathan, S Bhide … (2010)
          To assess the prevalence of cord entanglement and perinatal outcome in a large series of monoamniotic twin pregnancies and to review the recent literature on similar published large series. Prospective observational study of all prenatally detected cases of monoamniotic twin pregnancies during an 8-year period in a tertiary fetal medicine unit. A Medline database review for publications since 2000 containing five or more cases of monoamniotic pregnancies that showed data on cord entanglement and perinatal outcome was also undertaken. A total of 32 monoamniotic pregnancies were diagnosed during the study period, including three conjoined twins, seven pregnancies with twin reversed arterial perfusion (TRAP) syndrome, three surgical pregnancy interruptions for discordant fetal abnormality and one miscarriage before 16 weeks' gestation. The remaining 18 monoamniotic pregnancies were included in the study analysis. All monoamniotic pregnancies were complicated with antenatal cord entanglement diagnosed by B-mode and color Doppler ultrasound. There were 34 live births and a double intrauterine death diagnosed at 19 + 2 weeks' gestation. There were two late neonatal deaths, one from congenital complete heart block and the other after surgery for transposition of the great arteries. The overall perinatal loss rate was 11.1% after 16 weeks and 5.9% after 20 weeks' gestation. The cumulative rates of cord entanglement and perinatal mortality in the reviewed literature were 74% and 21%, respectively. Umbilical cord entanglement is present in all monoamniotic twins when it is systematically evaluated by ultrasound and color Doppler. Perinatal mortality in monoamniotic twins is mainly a consequence of conjoined twins, TRAP, discordant anomaly and spontaneous miscarriage before 20 weeks' gestation. Expectantly managed monoamniotic twins after 20 weeks have a very good prognosis despite the finding of cord entanglement. The practice of elective very preterm delivery or other interventions to prevent cord accidents in monoamniotic twins should be re-evaluated.
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            Perinatal outcomes in monoamniotic gestations.

            D. Lockwood, E Funai, H Roque … (2003)
            A comprehensive review of monoamniotic twin gestations reported between 1990 and 2002 was performed to estimate current perinatal mortality and morbidity rates, as well as the predictive value of an antenatal diagnosis of cord entanglement for poor obstetric outcomes. A Medline literature review using the search term 'monoamniotic' and limited to articles published in the English language between 1990 and 2002 was performed. A total of 133 continuing, non-conjoined twin monoamniotic pregnancies with delivery information were identified. Perinatal loss per 2-week interval was relatively constant at 2-4% from 15 to 32 weeks. However, of the 131 fetuses reaching 33 weeks, the percentage loss significantly increased to 11.0% at 33-35 weeks and 21.9% at 36-38 weeks compared to that at 30-32 weeks. Overall perinatal mortality was 23.3%. Of all losses, 61.2% involved both twins and 38.8% involved only one fetus. Cord entanglements were documented antenatally in 22.6% of reports. There was a statistically significant decrease in the average number of neonatal intensive care unit days for non-anomalous neonates (10.6 +/- 7.7 vs. 32.6 +/- 32.0), average gestational age at the time of delivery (30.4 +/- 7.6 vs. 32.6 +/- 4.1), as well as a decrease in the prevalence of total (8.3% vs. 27.7%) and non-anomalous (7.0% vs. 21.6%) perinatal mortality in pregnancies with an antenatal diagnosis of cord entanglement compared to those without the antenatal diagnosis of cord entanglement. The presence of fetal anomalies was associated with a 42.9% perinatal mortality rate. Contrary to previous reports, there is a significant increase in the incidence of perinatal loss beyond 32 weeks among monoamniotic twins, suggesting that delivery after corticosteroid therapy should be strongly considered at this gestational age.
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              High perinatal survival in monoamniotic twins managed by prophylactic sulindac, intensive ultrasound surveillance, and Cesarean delivery at 32 weeks' gestation.

              Neena L. Chappell, L Pasquini, Louise Fisk … (2006)
              Increased perinatal mortality in monoamniotic twin pregnancies is attributed to cord accidents in utero and at delivery. We evaluated the following parameters in monoamniotic pregnancies: (1) the incidence of cord entanglement; (2) the effect of sulindac on amniotic fluid volume and stability of fetal lie; and (3) the perinatal outcome with our current management paradigm. This is a retrospective review of monoamniotic pregnancies of >or=20 weeks' gestation managed with serial ultrasound surveillance, medical amnioreduction and elective Cesarean delivery at 32 weeks' gestation. Mean amniotic fluid index (AFI) and change in AFI in monoamniotic pregnancies managed with oral sulindac was compared with 40 gestation-matched monochorionic-diamniotic controls. Among 44 monoamniotic pregnancies, 20 with two live structurally normal twins at 20 weeks' gestation satisfied the inclusion criteria. All fetuses survived to 28 days postnatally despite early prenatal cord entanglement in all but one case. Whereas AFI remained stable throughout gestation in the controls, the AFI fell in those patients on sulindac from a mean value of 21.0 cm (95% CI, 18.5-23.6 cm) at 20 weeks to a mean of 12.4 cm (95% CI, 10.1-14.6 cm) at 32 weeks (ANOVA P across gestation = 0.001) but mainly remained within normal limits. Fetal lie was stabilized in 11/20 cases in the monoamniotic group compared with 13/40 in the control group (P < 0.0001). Cord entanglement appears unpreventable, as it typically occurs in early pregnancy. Sulindac therapy reduces AFI, leads to more stable fetal lie, and may prevent intrauterine death by diminishing the risk of constricting cords that are already entangled. Perinatal survival in monoamniotic pregnancies managed by a regime of sulindac from 20 weeks' gestation, close ultrasound surveillance and elective abdominal delivery at 32 weeks' gestation seems empirically higher than that in the literature. Copyright 2006 ISUOG. Published by John Wiley & Sons, Ltd.
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                Author and article information

                Journal
                Journal ID (nlm-ta): Int J Med Sci
                Journal ID (publisher-id): ijms
                Title: International Journal of Medical Sciences
                Publisher: Ivyspring International Publisher (Sydney )
                ISSN (Electronic): 1449-1907
                Publication date Collection: 2011
                Publication date (Electronic): 29 September 2011
                Volume: 8
                Issue: 7
                Pages: 573-576
                Affiliations
                Department of Obstetrics and Gynecology, St. Vincent's Hospital, Catholic University of Korea, Seoul Korea
                Author notes
                ✉ Corresponding author: Guisera Lee, MD., Department of Obstetrics, St. Vincent hospital, The Catholic University of Korea, 93-6 Ji-dong Paldal-gu Suwon-si , Gyeonggi-do , 442-723, Republic of Korea. E-mail: leegsr@ 123456catholic.ac.kr ; Tel: 82-31-249-7114; Fax: 82-31-254-7481

                Conflict of Interest: The authors have declared that no conflict of interest exists.

                Article
                Publisher ID: ijmsv08p0573
                PMC ID: 3198252
                PubMed ID: 22022209
                SO-VID: c2300535-65ac-4141-a040-0f9d803afa1d
                Copyright © © Ivyspring International Publisher. This is an open-access article distributed under the terms of the Creative Commons License (http://creativecommons.org/licenses/by-nc-nd/3.0/). Reproduction is permitted for personal, noncommercial use, provided that the article is in whole, unmodified, and properly cited.
                History
                Date received : 26 June 2011
                Date accepted : 19 September 2011
                Categories
                Subject: Case Report

                ScienceOpen disciplines: Medicine
                Keywords: anencephaly,cord entanglement,3d,monoamniotc twin
                Data availability:
                ScienceOpen disciplines: Medicine
                Keywords: anencephaly, cord entanglement, 3d, monoamniotc twin

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