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Online ISSN (İngilizce)
1305-3124

Basılı ISSN (Türkçe)
1300-5251

Online ISSN (Türkçe)
1305-3132

Kuruluş
1993

Editör
Cihat Şen

Yardımcı Editörler
Murat Yayla, Oluş Api

A Case Achieved at 28th Gestational Age by Cervical Cerclage with Retained Plasenta After Expulsion of Nonviable Fetus Underwent Selective Termination in Multifetal Pregnancy

Pınar Özcan, Mehmet Serdar Kütük, Taha Takmaz, Havva Sevde İşlek

Künye

A Case Achieved at 28th Gestational Age by Cervical Cerclage with Retained Plasenta After Expulsion of Nonviable Fetus Underwent Selective Termination in Multifetal Pregnancy. Perinatoloji Dergisi 2019;27(0):- DOI: 10.2399/prn.19.0272005

Yazar Bilgileri

Pınar Özcan1,
Mehmet Serdar Kütük1,
Taha Takmaz2,
Havva Sevde İşlek3

  1. Bezmialem Vakıf Üniversitesi Kadın Hastalıkları ve Doğum Anabilim Dalı istanbul TR
  2. Bezmialem Vakıf Üniversitesi Kadın Hastalıkları ve Doğum Anabilim Dalı istanbul TR
  3. Bezmialem Vakıf Üniversitesi Kadın Hastalıkları ve Doğum Anabilim Dalı istanbul TR
Yazışma Adresi

Pınar Özcan, Bezmialem Vakıf Üniversitesi Kadın Hastalıkları ve Doğum Anabilim Dalı istanbul TR, drpinarozcan@hotmail.com

Yayın Geçmişi

Gönderilme Tarihi: 03 Haziran 2019

Kabul Edilme Tarihi: 03 Eylül 2019

Erken Baskı Tarihi: 03 Eylül 2019

Çıkar Çakışması

Çıkar çakışması bulunmadığı belirtilmiştir.

Amaç
We aimed to present a special case underwent the selective termination because of multiple structural anomaly and trisomy 13 in dichorionic and diamniotic pregnancy after stimulated intrauterin insemination. It achieved at 28th gestational age by cervical cerclage with retained plasenta after expulsion of nonviable fetus at 21th gestational age which underwent ST at 20th gestational age.
Olgu
22 years old patient (G1P0) underwent IUI for unexplained infertility. A dichorionic diamniotic twin pregnancy has been confirmed by the ultrasonographic examinationat 12 weeks of gestation. The selective termination for twin A was performed at 19 weeks of gestation because of trisomy 13 andat 21 week of gestation dead twin A with ventriculomegaly, vermis agenesis, micrognathia, micropthalmia, hypertelorism, polydactyly on both hands, atrioventricular channel defect in the heart, hyperechogenic and polycystic imaging on both kidneys was delivered normally. On the absence of infection parameters, Mcdonald-Schirokodkar cerclage were carried out after the delivery of the dead twin A under antibiotherapy and tocolysis.
Sonuç
Vaginal delivery was performed and 1150 gr male baby with 8/9 apgar score was born at 28 week of gestation. Cervical cerclage may take into consideration an alternative  to prolong the pregnancy, although the retention of nonviable fetal plasenta following expulsion of nonviable fetus after the selective reduction may increase maternal and fetal risk.
 
Anahtar Kelimeler

dichorionic diamniotic twin pregnancy, fetal reduction, trisomy 13, cervical cerclage, IUI, structural anomaly

Giriş
Multi-fetal gestation is a well-known, adverse outcome of stimulated IUI treatment. Multi-fetal pregnancies compared to singletons are associated with a higher risk of maternal and obstetrical complications such as miscarriage, preterm birth, low-birth weight, preeclampsia, gestational diabetes and perinatal mortality. [1] The multiple gestation rate can be reduced with single embryo transfer in IVF treatment, however, in OS-IUI treatments, control of the multiple gestation rate is very limited.
The effect of assisted reproduction on the risk of major congenital malformations remains controversial. According to the result of studies focused on the risk of major congenital malformations associated with ART and IUI treatment, the use of ART and IUI was related to an increased risk of major birth defects (adjusted OR, 3.11; 95% CI, 1.33–7.27 for the risk of major urogenital malformations in ART; adjusted OR, 2.02; 95% CI, 1.10–3.71 for risk of major musculoskeletal malformations in IUI; adjusted OR, 1.66; 95% CI, 1.00–2.79 for an increased risk of any MCM in ART and adjusted OR, 7.18; 95% CI, 1.59–32.53 for urogenital malformations in ART). [2, 3] Even the adjustment, a contribution of the underlying subfertility problems cannot completely ruled out given the differences in the severity of subfertility. There are a few studies which evaluated the risk of major congenital malformations associated with the use of stimulated IUI. Furthermore, these studies had some limitations including suboptimal sample size, lack of appropriate comparison groups and lack of the establishment of the risk of different types of congenital malformations. The incidence of fetal abnormalities has been reported to be higher in multiple pregnancy than in singletons. [4, 5, 6]
             Women with multifetal pregnancies in which one fetus is abnormal and the other is normal have three options: expectant management (do nothing), terminate the pregnancy (both normal and abnormal fetuses), or selective termination (ST) of the abnormal fetus or fetuses. A specific fetus is terminated because of a structural anomaly or a chromosomal abnormality by using selective termination. [7] There are several approaches which have been described for embryo reduction. [8, 9] The most commonly applied methods is ultrasound-guided transabdominal injection of potassium chloride into the fetal heart. [10, 11] The technique of selective termination because of a structural anomaly or a chromosomal abnormality is not different from those in singleton pregnancies. But there are several additional considerations resulting from a multiple pregnancy include the procedure-related risk of pregnancy loss, the possibility of maternal coagulopathy secondary to the prolonged retention of a nonviable fetus and the appropriate technique. The complete pregnancy loss rate of selective termination (ST) is at 2,5-7% and it is proportional to the starting and ending number of fetuses. [12,13,14]
We aimed to present a special case underwent ST because of multiple structural anomaly and trisomy 13 in dichorionic and diamniotic pregnancy after stimulated IUI. It achieved at 28th gestational age by cervical cerclage with retained plasenta after expulsion of nonviable fetus at 21th gestational age which underwent ST at 20th gestational age.
Olgu
Twenty-two-year-old patient with gravida 1, parity 0 which is not able to concieve despite a year of unprotected sexual intercourse has applied to IVF unit of Faculty of Medicine of Bezmialem University. During investigation of infertility, her hormone profile was normal, a hysterosalpingography revealed that uterin cavity was normal and the fallopian tubes were bilaterally patent. Her partner’s spermiogram was consistent with normal values. The patient underwent ovulation induction and intrauterin insemination for unexplained infertility. She has got pregnant with her second ovulation induction and IUI. A dichorionic diamniotic twin pregnancyhas been confirmed by the ultrasonographic examinationat 12 weeks of gestation and twins had a normal translucence measurement of 1.4 mm and 1.6 mm at 12 weeks of gestation. She was referred to our perinatology clinic for the routine second trimester detailed obstetric ultrasonography at 20 weeks of gestation.An anomaly scan was performed at gestational age of 20 weeks of pregnancy. In the ultrasonographic examination, Twin A had multiple fetal abnormalities, which strongly suggest trisomy 13, including ventriculomegaly, vermian agenesis, micrognathia, micropthalmia, hypertelorism, polydactyly on both hands, atrioventricular channel defect in the heart, hyperechogenic and polycystic imaging on both kidneys (Figure 1). Twin B showed normal fetal anatomy without detected major and/or minor abnormality. The family was informed about the risk of pregnancy and genetic consultancy was provided about genetic abnormality and major congenital malformations. Counselling contained detailed discussion of the risk of mid-term  ST procedure which includes 3-7,5 % risk of total pregnancy loss. [15] The family was also informed that Trisomy 13 is universally lethal condition and continuation of pregnancy as twin might be an alternative approach albeit it has its attendant risks specific for multiple pregnancies and the potential need for prolonged intensive care for live-born with multiple anomalies. The parents opted for selective reduction of twin A due to their concern for prolonged suffering for the affected fetus A. ST for twin A with the presumptive diagnosis of trisomy 13 was performed by transabdominal guided intrathorasic injection of potasium chloride at 20th weeks. Amniocentesis performed during the termination procedure confirmed trisomy 13.
After ST, the patient readmitted to the hospital with vaginal bleeding at 21 week of gestation. She had powerfull uterine contractionand the ultrasonographic examination revealed that cervical length was 20 mm (Figure 2). Antibiotherapy (ampiciline+sulbactam IV 2 g/4id per day for 48 h followed by PO 375 mg/2id for 5 days plus azithromycin PO 500mg per day for 7days) and tocolysis (indomethacin rectal 100mg once, followed by PO 25mg/4id for 2 days) were started. Despite antibiotherapy and tocolysis, after fourth day of therapy, amniotic leakage occured and the dead twin A was delivered normally with full dilated cervix but the placenta of dead twin A was not delivered. On the absence of enfection parameters, Mcdonald-Schirokodkar cerclage were carried out after the delivery of the dead twin A under antibiotherapy and tocolysis. The patient was daily followed up with blood and clinic enfection parameters. The patient was discharged and weekly followed up at the outpatient clinic. 15 days after cerclage, the patient readmitted with abdominal pain and vaginal bleeding. She had powerfull uterine contraction and cervical length was 23 mm at 25 week of gestation. Antibiotherapy and tocolysis were started again as described above. Antenatal corticosteroids to enhance fetal lung maturity (betamethasone IM 12mg/d for48h) were done. Magnesium sulfate for neuroprotection with a loading dose of 4g IV in 20 min followed by a maintenance dose of 2g/h for 24 h were also given. There was no response of uterine contraction to antibiotherapy and tocolysis and the presence of enfection, vaginal cephalic delivery was performed and  1150 gr male baby with 8/9 apgar score was born at 28 week of gestation. During admission to NICU, the baby was not underwent intubation and it was treated with only CPAP for one week and it was stayed in neonatal unit for two months and it was discharged as 3000 gr two monhs later. There was no complication related to prematurity in neonatal unit.
Tartışma
We reported a special case to achieve at 28th gestational age by cervical cerclage with retained plasenta after expulsion of nonviable fetus with a structural anomaly and trisomy 13 underwent ST in dichorionic-diamniotic pregnancy after stimulated IUI.
ST of a specific fetus in multiple pregnany because of a structural anomaly or a chromosomal abnormality is a widely accepted procedure. Hovewer, ST of a fetus in multiple pregnancy is not similar to a singleton pregnancy, this procedure has additional factors that need to be considered in the context of a multiple pregnancy. The most unintended complication related to ST is the loss of total pregnancy before 24th gestational weeks. The rate of unintended pregnancy loss is generally associated with the gestational age at ST, the number of fetuses and having more than one fetus selectively terminated. The patients who had severe anomalies visible by ultrasound and invasive diagnostic procedures before ST is most likely related to higher risk of pregnancy loss. The other important risk related to ST is the possibility of pre-term birth of a normal fetus and long-term sequelae resulted from prematurity. Preterm labor is one of the most contributors of perinatal morbidity/mortality. The other important risk related to ST is the possibility of maternal risks from retantion of nonviable fetal tissue following the selective reduction. After the expulsion of fetus, the retantion of plasenta is associated with chorioamnionitis, hemorrhage and preterm delivery. The optimal timing for performing SF has been reported in the literature to be as early as particularly before 18–20 weeks. [16,17,18] Likewise, there are several technically problem related to ST of a specific fetus in multiple pregnany when compare to fetal reduction for elective indications in multiple pregnany. Basically, fetal reduction for elective indications could be accompanied with fewer complications when compare to ST of a specific fetus in multiple pregnany. [19] Because, during fetal reduction for elective indications, the technically most easily accessible fetus can be selected to reduce as well as the fetus furthest away from the internal cervical os can be reduced to minimize the risk of premature rupture of membranes followed by premature delivery. [20] On the other hand, selective fetal reduction is associated with excellent perinatal outcomes in dichorionic twins while selective reduction in monochorionic twins has an increased procedure-related complications and preterm delivery rate. A large single-centre series of selective fetal reduction in monochorionic and dichorionic twins for structural, chromosomal and genetic abnormalities demonstrated that selective termination in both monochorionic and dichorionic pregnancies is safe and effective with relatively low loss and prematurity rates. [21]. Cervical cerclage has been used widely in the management of pregnancies considered at high risk of preterm birth. Cerclage in twin pregnancies with dilated cervix ≥ 1cm before 24 weeks significantly increase latency period from diagnosis to delivery (6,7 weeks). Because of longer latency period from diagnosis to delivery, it decreases incidence of spontaneous preterm birth at any given gestational age and improves perinatal outcome when compared to expectant management. [22]
 
The first challenge related to our case is that we had to undergo reduction of the twin closest to the cervical os for ST because the structurally or chromosomally abnormal fetus located on closest to the internal cervical os. It may be increase the risk of premature rupture of membranes after reduction. The second challenge is that the risk results from the prolonged retention of plasenta of nonviable fetus after expulsion of the structurally or chromosomally abnormal fetus. We would not like to remove the plasenta of abnormal fetus after expulsion of abnormal fetus because its plasenta located between uterine wall and the non-affected co-twin. If we tried to remove this plasenta before cervical cerclage, it would result in premature rupture of membranes of the non-affected co-twin and thus total pregnancy loss. Therefore, we performed cervical cerclage after expulsion of abnormal fetus to prolong the pregnancy and to achieve viability of the non-affected co-twin. On the other hand, it may be considered as good prognostic factor to be dichorionic twins.
Sonuç
ST is a safe procedure with a low rate of pregnancy loss when performed by experienced operators. However, many of the risks related to multifetal pregnancies including  preterm labor and delivery may reduce with ST so that ST would be a benefit to the remaining fetus(es) by terminating a fetus with a lethal anomaly.  But unintended pregnancy loss before 24th gestational weeks is the most important complication of ST. Cervical cerclage may take into consideration an alternative  to prolong the pregnancy in those cases, although the retantion of nonviable fetal plasenta following expulsion of nonviable fetus after ST may increase maternal and fetal risk. Prophylactic antibiotherapy and close follow-up after cervical cerclage seems to be a reasonable choice to prevent the loss of non-affected co-twin and achieve viability of non-affected co-twin.
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Dosya / Açıklama
Figure 2
20 mm cervical length
Figure 1
a)vermis agenesis b)micrognathia c)micropthalmia and hypertelorism d)postaxial polydactyly e)hyperechogenic kidney