For the present pregnancy, she followed the national guide of prenatal care during the first trimester of pregnancy. No maternal infections were diagnosed, and the pregnancy had a low risk of genetic disorders at standard first-trimester screening. A prophylactic cervical cerclage was decided to be performed, considering the maternal history, even though the cervix was 32 mm long at the vaginal ultrasound measurement. The cervical cerclage was performed without any maternal complications at 14 weeks of gestations. The patient was also prescribed intravaginal progesterone.

The pregnancy evolution was in normal ranges until 20 weeks of gestation, when funneling was observed for 2.5 cm of the cervix, with opened internal cervical os and closed external cervical os (Figure 1). As an extreme measure, it was decided to be added an Arabin vaginal pessary, which was placed uneventful surrounding the cervical cerclage. For one week, the pregnancy had no problem, until the current emergency presentation for vaginal bleeding.
 

The present clinical examination revealed the amniotic pouch, easily observed inside the cervical pessary at speculum evaluation, and reduced vaginal bleeding. The abdominal ultrasound identified a 21-week fetus with normal cardiac rhythm, fetal movements, 14 cm amniotic fluid index, and homogenous placentae (Figure 2). The patient was admitted in the obstetric department. In the following 12 hours, the patient received tocolytic therapy, the vaginal bleeding was reduced, and a detailed counseling was performed regarding the therapeutic options.
 

The removal of the pessary was performed, and there was an impressive clinical identification. The amniotic pouch was visible through an impressive cervical rupture. It was observed a 3-cm rupture on the right lateral cervical side cranial of the cervical cerclage which was in place (Figure 3). We decided to try to perform another vaginal cerclage under anesthesia above the present one. During the procedure, under the anesthetic muscular relaxation, it was observed that the cervical rupture was about 6 cm in diameter, the amniotic pouch could not be reduced, and another cervical cerclage could not be performed. The present cervical cerclage was removed. The cervix was closed. The patient did not accept the idea of abortion, and she wanted an expectative management. The following 12 hours were without any complications, but later an important vaginal bleeding occurred. The patient accepted that the pregnancy was compromised, and she consented for the termination of pregnancy.
 

The peculiarity is that the fetus was evacuated trough the cervical rupture and, also, the uterine curettage following the abortion was made using the same pathway. The cervix was 2 cm opened. The cervical rupture was repaired after curettage using multiple “X” resorbable sutures (Figure 4). The patient was discharged four days after the procedure, and the follow-up was favorable.
 

Discussion

The specialty literature does not contain many cases similar to the one presented, with most studies focusing on the role of cervical cerclage and/or cervical pessary in preventing preterm birth.

In a study from 2020 that included 81 patients with singleton pregnancies between 2005 and 2017, the effectiveness of the McDonald cerclage versus the combination of cerclage and Arabin pessary in preventing birth before 34 weeks was evaluated. The results did not show statistically significant differences between the two groups regarding birth before 34 weeks. However, the duration of neonatal intensive care unit (NICU) admission and birth weight were lower and higher, respectively, in the combined group, especially in patients with cervical canal length below the third percentile⁽²⁾.

A study conducted at our center in 2021, involving 75 patients with cervical insufficiency, who were divided into three study arms (cerclage group, pessary group, and the combined group), revealed that the average gestational age at birth was higher in the combined group (38.33 weeks) compared to the cerclage group (37.82 weeks) and the pessary group (35.73 weeks). Additionally, the separate groups exhibited higher rates of spontaneous abortion compared to the combined group, which did not experience any spontaneous abortion or preterm birth events.

Furthermore, premature rupture of membranes in patients with a shorter cervical canal length was more frequent in the pessary group. These results shed light on the potential benefits of combined interventions in the management of cervical insufficiency, suggesting that they may lead to improved pregnancy outcomes compared to cerclage or pessary alone⁽³⁾.

A randomized clinical feasibility study, conducted in an open-label manner in 2021, which included 18 patients with singleton pregnancies and risk factors for preterm birth (such as a history of spontaneous preterm birth, preterm premature rupture of membranes before 34 weeks, or cervical surgeries), who agreed to participate, aimed to compare the efficacy of cerclage versus pessary, as well as intravaginal progesterone at a dose of 200 mg. The results indicate that such a study is feasible, but more eligible participants are necessary to draw conclusive findings⁽⁴⁾.

A recent study from Germany demonstrates that early interventions (such as cerclage, pessary, progesterone, and combinations thereof) lead to a longer interval between intervention and birth, particularly concerning very low birth weight infants born before 32 weeks. The study included 575 newborns from 424 mothers who underwent interventions between 20 and 25 weeks to prevent preterm birth. The combined intervention group had a higher average gestational age (27 weeks) and a higher average birth weight (980 g) compared to single interventions⁽⁵⁾.

A case report from 2021 illustrates the potential complications associated with inserting a pessary in the context of cervical insufficiency and bulging membranes in a twin pregnancy. Despite the availability of various management options, such as vaginal progesterone, cervical cerclage and cervical pessary, determining the most effective treatment strategy remains uncertain, particularly in cases of dilated cervix and exposed membranes in twin pregnancies. In this specific instance, a twin pregnancy at 22+6/7 weeks of gestation was admitted to the hospital due to cervical insufficiency with bulging membranes. Following an unsuccessful attempt at cervical cerclage, an Arabin pessary was inserted without administering antibiotics, given the absence of signs of infection.

Subsequently, caesarean delivery was performed at 24 weeks of gestation due to spontaneous preterm labor and rupture of membranes, with the first baby presenting in a transverse lie position. Tragically, the first twin was stillborn, with the Arabin pessary found adherent to the fetal head, contributing to funisitis/sepsis and mechanical insult. The second baby was born alive. The subsequent investigations suggested that the adherence of the pessary to the fetal head led to the demise of the first twin.

This case underscores the importance of carefully evaluating the antibiotic treatment in cases of bulging membranes and dilated cervix, even in the absence of signs or symptoms of infection. Furthermore, it suggests caution in pessary insertion, especially during the second trimester, due to the risk of dislocation within the uterus during contractions and the potential harm to extremely preterm fetuses. Such insights contribute to the ongoing discussion surrounding the management of cervical insufficiency in twin pregnancies, emphasizing the need for individualized approaches and careful consideration of potential risks and benefits⁽⁶⁾.

Another case report from 2021 highlights a severe ischemic gangrene of the scalp in an extremely preterm infant born at 24+5/7 weeks of gestation and admitted to the neonatal intensive care unit following a caesarean section due to maternal chorioamnionitis. Notably, an Arabin pessary had been inserted in addition to a previous cervical cerclage due to cervical insufficiency at 21+5/7 weeks of pregnancy. Subsequently, at 23+5/7 weeks of pregnancy, preterm rupture of membranes occurred, and both devices were retained to support fetal viability.

At 24+4/7 weeks of pregnancy, the mother developed maternal chorioamnionitis, prompting an urgent caesarean section. Transvaginal manual manipulation was necessary during the procedure. Upon admission to the NICU, the infant presented severe shock, requiring high-dose vasopressors and blood products. Following the surgical repair, a bilateral grade IV intracranial hemorrhage was observed. Subsequently, the decision was made to withdraw the life support.

It is speculated that maternal chorioamnionitis and local infection may have acted as predisposing factors, with the presence of the pessary potentially contributing to uterine contractions and its manipulation during the procedure acting as a precipitating factor. This case underscores the importance of carefully considering the risks and benefits of interventions such as pessary insertion, particularly in the context of preterm labor and maternal complications like chorioamnionitis⁽⁷⁾.

In a retrospective population-based cohort study conducted in Ohio from 2006 to 2015, involving 1,428,655 singleton and twin live births, the researchers evaluated the association between cerclage use during pregnancy and severe maternal morbidity. The study revealed that more than one in ten women who underwent cerclage experienced adverse maternal outcomes. Even after adjusting for factors such as gestational age at delivery, the risk of serious adverse events remained more than twofold higher for pregnancies with cerclage compared to those without. Among the individual adverse outcomes observed in the cerclage group, chorioamnionitis, unplanned operation after delivery, and maternal hospital transfer were the most common, affecting 4.4%, 4.2% and 3.4% of pregnancies, respectively. Less common adverse events included unplanned hysterectomy, uterine rupture, blood transfusion, and maternal ICU admission, with lower frequencies observed in both groups⁽⁸⁾.

Conclusions

Although a recommended and proven method for preventing preterm birth, the use of cerclage with or without the Arabin pessary can lead to unfavorable consequences, both for pregnancy viability and maternal obstetric future. The present case is rare but worth mentioning, as not many articles in the specialty literature address the negative aspects associated with the use of cerclage and cervical pessary. A particularly notable aspect in this case is the patient’s obstetric history, which places her at an increased risk for spontaneous abortion or preterm birth, prompting the cerclage to be performed under conditions of a long cervical canal at 14 weeks of pregnancy. Additionally, medical and psychological counseling for patients facing such a diagnosis is crucial, as severe maternal complications, such as uterine rupture and emergency hysterectomy, can threaten not only the woman’s obstetric future, but also her life.

Autori pentru corespondenţă: Romina-Marina Sima E-mail: romina.sima@umfcd.ro

CONFLICT OF INTEREST: none declared.

FINANCIAL SUPPORT: none declared.

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