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      Prenatal management and perinatal outcome in giant placental chorioangioma complicated with hydrops fetalis, fetal anemia and maternal mirror syndrome


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          Giant placental chorioangiomas have been associated with a number of severe fetal complications and high perinatal mortality.

          Case presentation

          We report a case of giant chorioangioma with fetal hydrops, additionally complicated by severe anemia, mild cardiomegaly with hyperdinamic heart circulation and maternal mirror syndrome. Intrauterine blood transfusion and amniodrainage was performed at 29 weeks. Worsening of the fetal and maternal condition prompted us to proceed with delivery at 29 + 5 weeks. The newborn died 3 hours later due to pulmonary hypoplasia and hemodynamic failure. Maternal course was favourable, mirror syndrome resolved in the second day and the patient was discharged four days following delivery.


          In the case described here, fetal condition got worse despite of the anemia correction and amniodrainage. Our outcome raises the issue whether additional intrauterine clinical intervention, as intersticial laser, should have been performed to stop further deterioration of the fetal condition when progressive severe hydrops develops.

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          Most cited references20

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          Mirror syndrome: a systematic review of fetal associated conditions, maternal presentation and perinatal outcome.

          Mirror syndrome, also referred to as Ballantyne's syndrome, is normally defined as the development of maternal edema in association with fetal hydrops. The incidence of mirror syndrome is low and few cases have been published. We describe a case report in association with fetal Ebstein anomaly and provide a systematic review on the fetal associated conditions, maternal presentation and perinatal outcome reported for mirror syndrome. A PubMed database search was done until December 2008 (English, French or German) without any restriction of publication date or journal, using the following key words: Ballantyne syndrome, Mirror syndrome, Triple edema, Pseudotoxemia, Maternal hydrops syndrome, Pregnancy toxemia, Acute second trimester gestosis, and Early onset preeclampsia. Reported cases were considered eligible when fetal associated conditions, maternal symptoms and fetal outcome were clearly described. Among 151 publications a total of 56 reported cases satisfying all inclusion criteria were identified. Mirror syndrome was associated with rhesus isoimmunization (29%), twin-twin transfusion syndrome (18%), viral infection (16%) and fetal malformations, fetal or placental tumors (37.5%). Gestational age at diagnosis ranged from 22.5 to 27.8 weeks of gestation. Maternal key signs were edema (80-100%), hypertension (57-78%) and proteinuria (20-56%). The overall rate of intrauterine death was 56%. Severe maternal complications including pulmonary edema occurred in 21.4%. Maternal symptoms disappeared 4.8-13.5 days after delivery. Mirror syndrome is associated with a substantial increase in fetal mortality and maternal morbidity. Copyright 2010 S. Karger AG, Basel.
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            Giant placental chorioangioma: natural history and pregnancy outcome.

            Giant placental chorioangiomas are associated with a high prevalence of pregnancy complications and a poor perinatal outcome. The aim of the study was to evaluate the natural history, intrauterine treatment and outcome of pregnancies complicated by giant placental chorioangioma. This was a retrospective study of 19 cases of giant placental chorioangioma, in which the natural history, intrauterine treatment and outcome of pregnancy were evaluated. Eighteen of the 19 cases were associated with a wide variety of fetal complications, including polyhydramnios, growth restriction, hyperdynamic circulation, cardiomegaly, anemia and non-immune hydrops. Two-thirds of the cases developed complications that required either elective delivery for fetal growth restriction (n = 6) or intervention for cardiovascular effects (n = 7). Fetal therapy, when required, was tailored to the particular complication and resulted in a successful pregnancy in all cases. The presence of large placental chorioangioma is associated with the development of fetal growth restriction and hyperdynamic state in a significant number of cases. This series elucidates some of the putative underlying mechanisms for these complications, and reports on a safe and effective treatment modality, percutaneous ultrasound-guided interstitial laser therapy. (c) 2009 ISUOG. Published by John Wiley & Sons, Ltd.
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              Perinatal outcome after prenatal diagnosis of placental chorioangioma.

              To review the prenatal complications, management, and perinatal outcome in pregnancies complicated by placental chorioangioma. Cases of placental chorioangioma diagnosed prenatally as part of a prospective, multicentric program for fetal diagnosis and therapy were identified. All cases were evaluated with color flow imaging. In the latter part of the study, three-dimensional power Doppler angiography was also used to study the vascular pattern of the tumor. Information on maternal demographics, prenatal sonographic findings, pregnancy complications, antenatal intervention, and perinatal outcome was obtained by reviewing the medical records or contacting the referring obstetricians. In the 5-year period from January 1997 to December 2001, 11 cases of placental chorioangioma were diagnosed prenatally. Nine cases were diagnosed in singleton and two in twin pregnancies. Among the nine cases occurring in singletons, five (56%) were associated with pregnancy complications, including polyhydramnios (n = 3), oligohydramnios (n = 2), fetal growth restriction (n = 2), and nonimmune hydrops (n = 1). Amniodrainage was required in one of these cases, allowing prolongation of pregnancy until term. Four (44%) singletons delivered before 35 weeks. Overall, two fetuses died, including one twin due to complications of twin-twin transfusion syndrome and another with hydrops after alcohol injection into the chorioangioma. In four pregnancies, no prenatal complications were detected in spite of continuous growth and vascularity of the placental mass in three of them. Placental chorioangioma is associated with an increased risk of pregnancy complications, the most common being polyhydramnios and preterm delivery. In selected cases, amniodrainage allows continuation of the pregnancy with improving perinatal outcome. Fetuses who develop hydrops are at the highest risk for perinatal death, with limited therapeutic options being available. Close follow-up is advised, even in those cases with no associated findings at the time of the diagnosis.

                Author and article information

                BMC Pregnancy Childbirth
                BMC Pregnancy Childbirth
                BMC Pregnancy and Childbirth
                BioMed Central
                28 July 2012
                : 12
                : 72
                [1 ]Unidad de Gestión Clínica de Genética, Reproducción y Medicina Fetal. Instituto de Biomedicina de Sevilla (IBIS), Hospital Universitario Virgen del Rocío/CSIC/Universidad de Sevilla, Sevilla, Spain
                [2 ]Servicio de Obstetricia y Ginecologia, Hospital Juan Ramón Jiménez, Huelva, Spain
                [3 ]Centro de Investigación Biomédica en Red de Enfermedades Raras (CIBERER), Sevilla, Spain
                [4 ]Director de la Unidad de Gestión Clínica de Genética, Reproducción y Medicina Fetal, Hospital de la Mujer, Hospital Universitario Virgen del Rocío, Avda. Manuel Siurot s/n, 41013, Sevilla, Spain
                Copyright ©2012 García-Díaz et al.; licensee BioMed Central Ltd.

                This is an Open Access article distributed under the terms of the Creative Commons Attribution License ( http://creativecommons.org/licenses/by/2.0), which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

                : 23 November 2011
                : 20 July 2012
                Case Report

                Obstetrics & Gynecology
                fetal chorioangioma,fetal anemia,mirror syndrome,fetal therapy,hydrops fetalis


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