Taiwanese Journal of & Gynecology 55 (2016) 109e111

Contents lists available at ScienceDirect

Taiwanese Journal of Obstetrics & Gynecology

journal homepage: www.tjog-online.com

Case Report Successful ablation of acardiac twin with Histoacryl

* Vladimír Ferianec, Mikulas Redecha , Peter Papcun, Marian Krizko Jr., Ivan Hollý, Karol Holoman

Department of and Obstetrics, Comenius University, Bratislava, Slovakia article info abstract

Article history: Objective: Acardiac twin occurs in 1:35,000 pregnancies. Several techniques have been described to treat Accepted 7 April 2014 this condition. Some techniques have been suggested as golden standard; however, new are still being tried. Keywords: Case report: This is a case of a 32-year-old patient who had successful ablation of the acardiac twin with acardiac twin Histoacryl. The diagnosis of the acardiac twin was made in the 11 weeks þ 3 weeks of pregnancy. Due to fetal ablation the development of myocardial hypertrophy and pericardial transudate of the pumping fetus, we had occlusion material (Hyalobarrier) performed ablation of the acardiac twin with Histoacryl in the 21 weeks' þ5 weeks' gestation. The procedure was uneventful, and the healthy fetus had no signs of distress. In the 33 weeks' þ5 weeks' gestation, she had Cesarean section due to distress of the healthy fetus. The female baby was healthy, weighing 2380 g, Apgar score 9/10. The mummified mass of acardiac fetus weighted 300 g. Nine months later, the child is doing well. Conclusion: Histoacryl is suitable for the ablation of an acardiac twin. Further studies are needed to prove the efficacy of this technique. Copyright © 2016, Taiwan Association of Obstetrics & Gynecology. Published by Elsevier Taiwan LLC. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/ 4.0/).

Introduction The mortality rate of the pumping twin is more than 50% [2]. Today, we have several therapeutic possibilities. The treatment of choice is An acardiac-twin pregnancy occurs very rarely, only in 1:35,000 selected according to the week of the pregnancy and the level of deliveries and in one in 100 monozygotic twins [1]. The twins are development of each of the mentioned pathomechanism. At hand, always monochorionic. The pathomechanism of the acardiac-twin we have the possibility of pregnancy termination, expectation formation lies in the preexistence of arterio-arterial anastomosis, management, intrafetal ablation, or interventional anastomosis either on the surface of the placenta or in the placental arterial sclerotization (alcohol ablation, thermal ablation, laser ablation, system, or more rarely as a direct connection of the fetal umbilical bipolar coagulation). Several new techniques, like radiofrequency cords [2]. Thus, the acardiac twin does not have its own placenta, ablation, are being investigated, but more studies are needed to and the arterial pressure of one twin overpowers that of the other prove their safety and efficacy [3]. Based on these reports, we have twin. It is rare, but for the healthy fetus, a very serious complication. tried to treat the condition of acardiac-twin pregnancy by, in other The acardiac twin does not have its own heart pump, and its medical fields, a well-tried method. We have used TissueSeal's function is overtaken by the heart of the healthy sibling with all the Histoacryl (B-Braun, Bratislava, Slovakia) in order to occlude the circulation consequences on it. The blood flow of the acardiac twin circulation of the acardiac twin and interrupt the twin-reversed- is then reversed. The whole mechanism creates a so-called twin- arterial-perfusion sequence. Histoacryl is used as a tissue seal for reversed-arterial-perfusion sequence. In most cases, the head and wound closure or for vessel closure by interventional radiologists. It upper body of the acardiac twin do not develop. The healthy fetus consists of monomeric n-butyl-2-cyanoacrylate, which polymer- develops arterial-steal phenomenon with the development of car- izes quickly in connection with tissue fluid. diac insufficiency, polyhydramnion, and consequently heart failure. Case Report

We have diagnosed an acardiac twin in the 12th week of * Corresponding author. Department of Gynaecology and Obstetrics, School of , Comenius University, Ruzinovska 6, 821 01, Bratislava, Slovakia. pregnancy in a 35-year-old patient, gravida 2, para 1, with biam- E-mail address: [email protected] (M. Redecha). nionic monochorial twin pregnancy. The patient, after being fully http://dx.doi.org/10.1016/j.tjog.2014.04.033 1028-4559/Copyright © 2016, Taiwan Association of Obstetrics & Gynecology. Published by Elsevier Taiwan LLC. This is an open access article under the CC BY-NC-ND license (http://creativecommons.org/licenses/by-nc-nd/4.0/). 110 V. Ferianec et al. / Taiwanese Journal of Obstetrics & Gynecology 55 (2016) 109e111

not show signs of increased distress and had normal heart rate. The whole procedure took 12 minutes. Further observations of the pumping twin showed normal heart rate and Doppler flowmetry. The patient was released from the hospital after 2 days of follow- up with planned outpatient visits. In the following weeks, the heart function of the healthy fetus restored, including the peri- cardial transudate. The fetus grew in physiological margins. Meanwhile, the acardiac twin showed signs of mummification and slow shrinkage, with no influence on the healthy fetus. The amniotic-fluid volume of the acardiac twin reduced continuously (Figure 1). In the 33rd week of pregnancy, 11 weeks after the intervention, the fetus showed signs of distress, and pathological levels of umbilical-blood-flow indexes with signs of fetal blood- flow centralization. Therefore, after the corticoid lung prepara- tion of the fetus, we have performed delivery by Cesarean section in the 33 weeks þ 5 weeks of pregnancy. The female baby was healthy, weighing 2380 g, with Apgar score 9/10, umbilical blood pH 7.22, with base excess 4.6 mmol/L. The acardiac female fetus resembled a mummified mass weighing 300 g (Figure 2A). The X- ray examination of the acardiac fetus with placenta revealed foci Figure 1. Magnetic-resonance-imaging scan of the healthy and acardiac fetus after ablation. of Histoacryl in the umbilical cord of the acardiac twin (Figure 2B). Nine months after the delivery, the surviving child is doing well with no signs of related embolic phenomenon. consulted and informed, decided to continue the pregnancy. Further ultrasound examinations revealed that the acardiac twin Discussion did not develop head, skull, and upper extremities. The stomach was absent and the trunk absently ended with multiseptal Several attempts have been made to occlude the umbilical ar- hygroma. Until the 17th week, the healthy fetus did not show signs tery of the acardiac twin, including fetal ablation or interventional of fetal distress. We have performed prenatal echocardiography of anastomosis closure (e.g., laser ablation, alcohol ablation, ter- the “pumping fetus” in the 20th week of the pregnancy. It has moablation) [4]. This case has shown a different possibility of developed myocardial hypertrophy and pericardial transudate. invasive intrauterine treatment approach to acardiac twins with a Because of the presumed development of chronic fetal hypoxia tissue-sealing material. We have tried this technique because of its and consequently congenital heart failure in the 21 weeks þ 5 presumed safety, simplicity, and experience in treating adult- weeks of pregnancy, after fully informing the mother about the patient conditions by interventional radiologists. Since our insti- status of the fetus, we have performed intrauterine intervention tution is a tertiary medical university center, under our laws, we by intra-arterial ablation of the acardiac twin. As the ablation did not need an agreement from the regional ethics committee. material, we have used 0.3 mL of TissueSeal's Histoacryl, diluted However, the patient had to sign a detailed informed consent, and by 6:1 ratio with a diluting liquid (Lipiodol Ultra Fluid, GUERBET, was fully acknowledged before the intervention. Even though the France). Under ultrasound guidance, we have inserted a 22-gauge golden standard in these cases is considered laser ablation, it needle into the acardiac-twin main feeding umbilical-cord vessel. cannot be used in every patient [5]. In each case, an individual We have then applied the ablating material in 5% glucose during approach and management are necessary. In order to prevent 30 seconds. Glucose was used, because in contact with saline or preterm premature rupture of membranes after fetal ablation, other material, the tissue glue agglutinates. Consequently, after mini-invasive techniques have to be selected. For laser techniques, the application of the material, the blood flow of the acardiac twin a relatively big insertion into the intra-amnionic space is needed, has stopped immediately. At the same time, the healthy twin did which is associated with a higher risk of preterm premature

Figure 2. (A) Acardiac fetus with placenta after delivery. (B) X-ray picture with hyperechogenic places of Histoacryl. V. Ferianec et al. / Taiwanese Journal of Obstetrics & Gynecology 55 (2016) 109e111 111 rupture of membranes [5]. Therefore, we have chosen the 22- Conflicts of interest gauge needle and the application of TissueSeal's Histoacryl as the least invasive type of procedure. Furthermore, the acardiac twin The authors have no conflicts of interest relevant to this article. can be developed into several different types, and according to this, the appropriate technique and approach have to be selected [3,6]. We think that, in case of intervention, the decision has to be References made as early as possible and the intervention performed in the earlier weeks of the pregnancy. This way, we can avoid some [1] Yu HY, Lau T, Sahota DS, Ting Y, Leung T. Perinatal outcome of monochorionic specific complications associated with acardiac-twin feticide [7,8]. twin pregnancies. Zhongua Fu Chan Ke Za Zhi 2013;48:405e10. Some authors describe umbilical-cord transection to be effective [2] Moore TR, Gale S, Benirschke K. Perinatal outcome of forty-nine pregnancies complicated by acardiac twinning. Am J Obstet Gynecol 1990;163:907e12. in these cases [9]. However, we think that these approaches mean [3] Lee H, Bebbington M, Crombeholme TM. The North American too big interventional and represent a high risk for the course of Network Registry data on outcomes of radiofrequency ablation for twin- the pregnancy. Ville [5] describes a high percentage of failure reversed arterial perfusion sequence. Fetal Diagn Ther 2013;33:224e9. [4] Tan T, Sepulveda W. Acardiac twin: a systematic review of minimally invasive when using this laser ablation, even though it is considered to be treatment modalities. Ultrasound Obstet Gynecol 2003;22:409e19. the standard care of therapy. In order to minimize the risks, we [5] Ville Y. Selective feticide in monochorionic pregnancies: toys for the boys or have used a “tissue-sealing” material, Histoacryl. The material has standard care? Ultrasound Obstet Gynecol 2003;22:448e50. [6] Yamaguchi K, Honma K. An acardiac twin with advanced development of the appropriate qualities for vascular closure and even for amnion- brain: a histologic and volumetric study. Clin Neuropathol 2014;33:84e90. defect closure [10]. The extremely fast complete closure of the [7] Patra S, Gupta JK, Patra R. A rare anomaly in twin pregnancydacardiac twin. acardiac-fetus vascular system even prevented blood-steal phe- J Indian Med Assoc 2012;110:931e2. nomenon from the healthy fetus. There are no data about using [8] Novak CM, Patel SV, Baschat AA, Hickey KW, Petersen SM. Maternal coagul- opathy after umbilical cord occlusion for twin reversed arterial perfusion Histoacryl in the treatment during pregnancy. However, it is a sequence. Obstet Gynecol 2013;122:498e500. widely used material by interventional radiologists for ablation of [9] Bermúdez C, Tejada P, Gonzalez F, Perez-Wulff J, Araujo L, Azpúrua H, et al. tumors and in as wound closure [11]. Its price is V75, and Umbilical cord transection in twin-reverse arterial perfusion syndrome with the use of a coaxial bipolar electrode (Versapoint). J Matern Fetal Neonatal it is fully covered by health insurance. Med 2003;14:277e8. According to our experience, it seems that Histoacryl is also a [10] Bilic G, Brubaker C, Messersmith PB, Mallik AS, Quinn TM, Haller C, et al. suitable material for the ablation of an acardiac twin and for the Injectable candidate sealants for fetal membrane repair: bonding and toxicity in vitro. Am J Obstet Gynecol 2010;202:85. safety of the mother. However, further experience is needed in [11] Schneider G, Otto K. In vitro and in vivo studies on the use of Histoacryl as soft order to prove its efficacy. tissue glue. Eur Arch Otorhinolaryngol 2012;269:1783e9.