Abstract
Background
Previable prelabor rupture of membranes (PROM) in twin pregnancies is a rare but high‐risk condition associated with substantial neonatal mortality and maternal morbidity. Management options include expectant management and selective reduction, though guidance is limited and based primarily on singleton data. Emerging reports suggest selective reduction may improve outcomes for the unaffected co‐twin by prolonging latency and reducing maternal complications.
Objectives
To evaluate maternal, obstetric, and neonatal outcomes in twin pregnancies complicated by previable PROM before 24 weeks of gestation, and to compare outcomes following expectant management and selective reduction.
Search strategy
A comprehensive literature search was conducted in Medline, Embase, and Web of Science for studies published between January 1990 and September 2024.
Selection criteria
Eligible studies included case reports, case series, and cohort studies, reporting outcomes of twin pregnancies with previable prelabor rupture of membranes (PPROM) managed either expectantly or by selective reduction of the affected twin.
Data collection and analysis
Data on maternal, obstetric, and neonatal outcomes were extracted and pooled for analysis. Subgroup analyses were performed based on management strategy: selective reduction versus expectant management.
Main results
A total of 45 studies were included, comprising 286 twin pregnancies with previable PROM. Selective reduction was performed in 16.4% (n = 47) of the pregnancies. Across the entire cohort, no infant was discharged home alive in 32.0% of the pregnancies, one infant in 51.6%, and two infants in only 16.4%. Clinical chorioamnionitis occurred in 33.5%, maternal sepsis in 7.8%, septic shock in 1.6%, and hysterectomy in 0.8%. Compared to expectant management, selective reduction was associated with longer latency (15.0 ± 8.4 vs. 8.3 ± 5.7 weeks), higher gestational age at delivery (31.6 ± 7.7 vs. 24.4 ± 4.4 weeks), and higher birth weight (2663.6 ± 703.9 vs. 1251.3 ± 773.0 g) of the co‐twin, all P < 0.001. Following selective reduction compared to expectant management, the discharge home of one live infant was more common (66.0% vs. 33.8%, P < 0.001), yet the discharge of two infants was not possible (0.0% vs. 25.7%, P < 0.001). Following selective reduction, clinical chorioamnionitis and neonatal death were lower (19.1% vs. 40.3%, P = 0.008 and 3.6% vs. 17.1%, P = 0.033, respectively).
Conclusions
Twin pregnancies with PPROM are associated with high maternal and neonatal risks. Selective reduction may improve outcomes for the unaffected twin and reduce maternal morbidity. These findings underscore the need for individualized, multidisciplinary counseling and highlight the critical gaps in evidence guiding management in this rare clinical scenario.
Keywords: expectant management, neonatal outcomes, previable prelabor rupture of membranes, selective reduction, twin pregnancy
1. INTRODUCTION
Previability represents the earliest stage of fetal development at which there is a reasonable, though limited, chance of survival outside the womb. Currently, this threshold of neonatal viability is about 22–23 weeks' gestation in facilities equipped with neonatal intensive care units. Outcomes in previable prelabor rupture of membranes (PROM) are influenced by gestational age at rupture, the duration of membrane rupture, and the presence of complications like oligohydramnios, which increases the risk of pulmonary hypoplasia and other prematurity‐related conditions. Additionally, prolonged membrane rupture can lead to infections, such as chorioamnionitis, which further escalate the risks for both mother and fetus. 1 Although occurring in only 0.1%–0.37% of pregnancies, previable PROM is more frequently observed in twin than singleton pregnancies. 2 , 3 In twins, previable PROM affecting one fetus may increase the risks of prematurity or infection of the co‐twin. Consequently, prenatal counseling and management in such context require a delicate balance of risks and benefits for both fetuses. 1 , 4
The evidence is limited regarding maternal and neonatal outcomes of previable PROM in twin pregnancies, with most available data derived from studies on singleton gestations. 3 , 5 , 6 , 7 Outcomes in previable PROM are influenced by gestational age at rupture, the duration of membrane rupture, and the presence of complications like oligohydramnios, which increases the risk of pulmonary hypoplasia and other prematurity‐related conditions. Additionally, prolonged membrane rupture can lead to infections, such as chorioamnionitis, which further escalate the risks for both mother and fetus. 3 For twins, these risks are compounded as each fetus may be affected differently, and interventions carry unique implications for each twin. 5
Management options typically recommend termination of the entire pregnancy (TOP). Other options include expectant management to prolong gestation, and selective reduction to potentially improve outcomes for the remaining twin. However, evidence guiding these approaches is limited, and standardized guidelines are currently lacking. 8 This systematic review aimed to consolidate the available data on maternal and neonatal outcomes in twin pregnancies complicated by PROM. By examining the benefits and risks of current management strategies, we sought evidence to support informed clinical decision making for this high‐risk population.
The objective of this study was to systematically review and describe maternal, obstetric, and neonatal outcomes in twin pregnancies complicated by previable PROM before 24 weeks of gestation that were managed with the intent to continue the pregnancy. We compared outcomes following expectant management and selective reduction.
2. METHODS
2.1. Adherence to reporting guidelines
The review was conducted in accordance with the Meta‐analysis of Observational Studies in Epidemiology (MOOSE) guidelines and reported following the Preferred Reporting Items for Systematic Reviews and Meta‐Analyses (PRISMA) checklist to ensure clarity and transparency (see Tables S1 and S2). 9 , 10 Prior to data extraction and analysis, the protocol was registered with the International Prospective Register of Systematic Reviews (PROSPERO; ID: CRD42024597637).
2.2. Eligibility criteria
We included case reports, case series, cohort studies, case–control studies, and observational studies. Reviews, commentaries, and guidelines were excluded. Studies were eligible if they reported outcomes of twin pregnancies complicated by previable PROM before 24 weeks of gestation, managed with the intent to continue the pregnancy, either expectantly or by selective reduction of the affected twin. This gestational‐age cutoff was chosen to capture a strictly previable cohort, considering the marked changes that occur in management and prognosis after 24 weeks, following the introduction of antenatal corticosteroids, magnesium sulfate, and active neonatal resuscitation. Accordingly, including 24–26‐week gestations would have introduced clinical heterogeneity and biased outcome estimates toward improved survival. Only studies published in English from 1990 through September 2024 were considered, to reflect more contemporary obstetric and neonatal practices. Exclusion criteria included studies reporting on singleton pregnancies, studies without separate data for twin pregnancies, and studies describing PROM occurring beyond 24 weeks of gestation. Pregnancies in which the entire pregnancy was electively terminated were also excluded.
2.3. Information sources and search strategy
A comprehensive literature search was performed across Embase, Medline, and Web of Science, covering studies from 1990 to September 2024. In collaboration with information specialists at Tel Aviv University Library, we designed a search strategy (Table S3). The population of interest was twin pregnancies with previable PROM. Our review focused on neonatal outcomes, including survival rates, and neonatal morbidities like pulmonary hypoplasia and prematurity‐related complications such as respiratory distress syndrome and intraventricular hemorrhage. The pregnancy outcomes evaluated included gestational age at delivery, and the proportions with chorioamnionitis, placental abruption, and cord prolapse. The maternal outcomes of interest included sepsis, admission to the intensive care unit, hysterectomy, and maternal mortality.
2.4. Selection process, data collection process, and data items
Two independent researchers (IS and LL) undertook the selection process according to the outlined PICO framework. After eliminating duplicate records, the reviewers assessed the titles and abstracts to determine initial eligibility, and then thoroughly evaluated the full texts of the remaining articles. To ensure a comprehensive search, reference lists of the included studies were examined for potentially relevant publications. Any discrepancies in study selection were resolved in discussion with a senior investigator (MO). The data collected included author names, study year, country, study details (e.g., year, location, duration, design, setting), the number of pregnancies, and the outcomes listed above. The data were systematically extracted into a preformatted Excel spreadsheet.
2.5. Assessment of methodological quality
To evaluate the methodological rigor of the case reports and case series included in this review, we applied a validated assessment tool that emphasizes key domains such as case selection, data ascertainment, and the clarity of reporting. 11 Three causality‐related questions were excluded, as they did not apply to this study. Each question allowed for three response options. Two independent reviewers assessed the quality of each study. Disagreements were resolved through discussion, ensuring reliable quality assessment.
2.6. Ethics
As this study is a systematic review and meta‐analysis of previously published data, neither ethical approval nor patient consent was required.
2.7. Statistical analysis
In the descriptive analyses, continuous variables are presented as medians with ranges, or as means with standard deviations, as appropriate. Categorical variables are presented as frequencies and percentages. Outcomes following selective reduction group and expectant management were compared using the independent t‐test or the Mann–Whitney U test for continuous variables, and the chi‐square test or Fisher exact test for categorical variables. A P value of less than 0.05 was considered statistically significant. All the analyses were performed using SPSS version 27.0 (IBM Corp., Armonk, NY).
3. RESULTS
A total of 45 studies met the eligibility criteria for our systematic review, 4 , 5 , 12 , 13 , 14 , 15 , 16 , 17 , 18 , 19 , 20 , 21 , 22 , 23 , 24 , 25 , 26 , 27 , 28 , 29 , 30 , 31 , 32 , 33 , 34 , 35 , 36 , 37 , 38 , 39 , 40 , 41 , 42 , 43 , 44 , 45 , 46 , 47 , 48 , 49 , 50 , 51 , 52 , 53 , 54 yielding 286 pregnancies for analysis. Of these 47 pregnancies were managed by selective reduction and 239 by expectant management. The flow chart of study selection is presented in Figure 1, and the reasons for exclusion are detailed in Table S4. The characteristics of the included studies are summarized in Table 1. The quality assessment of the included studies is summarized in Table S5. Most studies demonstrated clear reporting and adequate ascertainment of exposure and outcomes, although in many, it was unclear whether all the eligible patients were reported. The mean gestational age at PROM was 19.5 ± 2.9 weeks (n = 145). Dichorionic diamniotic twins accounted for 95.9% (208/217), and monochorionic diamniotic twins for 4.1% (9/217). Selective reduction was performed in 16.4% of the pregnancies (47/286). Antibiotic administration was reported in 91.3% (221/242) of the pregnancies. Table 2 describes the characteristics of the study population.
FIGURE 1.
Flow chart.
TABLE 1.
Characteristics of studies included.
| Year | Location | Number of cases included | Type of twins | Twin A/B with PPROM | Antibiotic treatment | Selective reduction | Interval delivery | ||
|---|---|---|---|---|---|---|---|---|---|
| 1 | Borenstein et al 12 | 1990 | Israel | 1 | Not reported | B | + | − | − |
| 2 | Jensen et al 13 | 1991 | Denmark | 1 | Di‐Di | A | + | − | + |
| 3 | Long et al 14 | 1991 | UK | 1 | Di‐Di | A | − | − | + |
| 4 | Kruzel et al 15 | 1992 | USA | 1 | Not reported | A | + | − | + |
| 5 | Poeschmann et al 16 | 1992 | Netherlands | 1 | Di‐Di | A | − | − | + |
| 6 | Wittman et al 17 | 1992 | Canada | 1 | Di‐Di | A | + | − | + |
| 7 | Arias et al 18 | 1994 | USA | 8 | Di‐Di | A | + | − | + |
| 8 | de Jong et al 19 | 1995 | Netherlands | 1 | Di‐Di | A | + | − | + |
| 9 | Dorfman et al 20 | 1995 | USA | 1 | Di‐Di | A | − | + | − |
| 10 | Antsaklis et al 21 | 1996 | Greece | 2 | Di‐Di | A | + | − | + |
| 11 | Wu et al22 | 1996 | Taiwan | 1 | Di‐Di | A | + | − | − |
| 12 | Abboud et al 23 | 1997 | France | 2 | Di‐Di | A | One of 2 received ABX | − | + |
| 13 | Knight et al 24 | 1997 | Australia | 1 | Di‐Di | A | + | − | + |
| 14 | Bakos et al 25 | 1998 | Sweden | 1 | Di‐Di | A | + | − | + |
| 15 | Catte et al 26 | 1998 | Belgium | 11 | Di‐Di | A | Three received ABX | Performed for 3 | Occurred for 2 |
| 16 | Platt et al 27 | 1999 | USA | 1 | Di‐Di | A | + | − | + |
| 17 | Van Doorn et al 28 | 1999 | Netherlands | 1 | Not reported | A | + | − | + |
| 18 | Bollen et al 29 | 2000 | Belgium | 1 | Di‐Di | A | + | − | + |
| 19 | Farkouh et al 30 | 2000 | USA | 10 | Di‐Di | A | + | − | + |
| 20 | Clerici et al 31 | 2001 | Italy | 1 | Di‐Di | A | + | − | + |
| 21 | Ghulmiyyah et al 32 | 2004 | USA | 1 | Di‐Di | A | + | − | + |
| 22 | Livingston et al 33 | 2004 | USA | 7 | Di‐Di | Not reported | + | − | + |
| 23 | Cristinelli et al 34 | 2005 | France | 1 | Di‐Di | A | + | − | + |
| 24 | Wu et al 35 | 2010 | Taiwan | 1 | Di‐Di | B | + | − | − |
| 25 | Petousis et al 36 | 2012 | Greece | 2 | Di‐Di | A | + | − | + |
| 26 | Reinhard et al 37 | 2012 | Germany | 3 | Di‐Di | A | + | − | + |
| 27 | Qiu et al 38 | 2013 | China | 1 | Di‐Di | A | + | − | + |
| 28 | Doger et al 39 | 2014 | Turkey | 2 | Di‐Di | A | + | − | + |
| 29 | Udealor et al 40 | 2015 | Nigeria | 1 | Di‐Di | A | + | − | + |
| 30 | Galvão et al 41 | 2016 | Portugal | 1 | Di‐Di | A | + | − | + |
| 31 | Myrick et al 42 | 2016 | USA | 30 | Di‐Di in 23, seven Mono‐bi | Twin A in 29, Twin B in one | 17 received ABX | − | Not reported |
| 32 | Yılanlıoğlu et al 43 | 2016 | Turkey | 6 | Di‐Di | Not reported | Not reported | + | − |
| 33 | Wagner et al 44 | 2017 | Germany | 27 | Di‐Di | Not reported | + | − | Not reported |
| 34 | Axelson et al 45 | 2018 | USA | 1 | Di‐Di | A | + | + | − |
| 35 | Lim et al 46 | 2018 | Canada | 27 | Di‐Di | A | 23 received ABX | Performed in 5 cases | Occurred for 6 |
| 36 | Abdelazim et al47 | 2019 | Kazakhstan | 1 | Di‐Di | A | + | − | + |
| 37 | Benito et al 48 | 2019 | Spain | 2 | Di‐Di | A | + | − | + |
| 38 | Alnoman et al 49 | 2020 | Canada | 1 | Di‐Di | A | + | − | − |
| 39 | Zajicek et al 50 | 2020 | Israel | 20 | Di‐Di | Twin A in seven, Twin B in 13 | + | Performed in 13 | Occurred in 3 |
| 40 | Zhou et al 51 | 2020 | China | 3 | Di‐Di | A | + | + | − |
| 41 | Xing et al 52 | 2021 | China | 2 | Mono‐Di | Not reported | + | − | − |
| 42 | Ponce et al 5 | 2023 | Spain | 36 | Cannot be determined | Not reported | + | − | Cannot be determined |
| 43 | Gadeer et al 53 | 2024 | Canada | 1 | Di‐Di | A | + | − | + |
| 44 | Goodfellow et al 4 | 2024 | UK | 30 | Cannot be determined | Not reported | Not reported | − | Not reported |
| 45 | Zhu et al 54 | 2024 | China | 28 | Di‐Di | Twin A in 15, Twin B in 13 | + | Performed in 16 | Not reported |
Note: +, Yes; −, No.
Abbreviations: ABX, antibiotics; Di‐Di, dichorionic diamniotic; Mono‐Di, monochorionic diamniotic; PPROM, previable prelabor rupture of membranes.
TABLE 2.
Pregnancy and management characteristics of the study population.
| Variable | N | |
|---|---|---|
| Maternal age, years, mean (±SD) | 101 | 32.1 ± 5.1 |
| Gravidity, mean (±SD) | 42 | 2.5 ± 1.6 |
| Parity, median (range) | 42 | 0 (0–3) |
| GA at PPROM, weeks, mean (±SD) | 145 | 19.5 ± 2.9 |
| Assisted reproduction (%) | 69/105 | 65.7 |
| PROM after amniocentesis/CVS | 11/73 | 15.1 |
| PROM after fetoscopy/selective termination (%) | 6/92 | 4.4 |
| Amniocentesis for IAI (%) | 27/130 | 20.8 |
| Selective reduction (%) | 47/286 | 16.4 |
| Rupture of lower sac (%) | 127/156 | 81.4 |
| MC‐DA twins (%) | 9/217 | 4.1 |
| DC‐DA twins (%) | 208/217 | 95.9 |
| Antibiotic administration (%) | 221/242 | 91.3 |
| Cerclage (%) | 25/195 | 12.8 |
Note: Denominators vary according to the number of cases for which data were reported in the original studies.
Abbreviations: CVS, chorionic villi sampling; DA, diamniotic; DC, dichorionic; GA, gestational age; IAI, intra‐amniotic infection; MC, monochorionic; PPROM, previable prelabor rupture of membranes; PROM, prelabor rupture of membranes; SD, standard deviation.
The mean gestational age at delivery was 22.8 ± 4.5 weeks for the affected twin and 24.9 ± 5.3 weeks for the co‐twin. The mean latency period was 5.5 ± 9.5 weeks for the affected twin and 9.7 ± 6.9 weeks for the co‐twin. The mean birth weight was 593 ± 442 g for the affected twin and 1692 ± 996 g for the co‐twin. Pulmonary hypoplasia was reported in 15.3% of the affected twins. Sepsis occurred in 21.4% of the affected twins and 9.8% of the co‐twins. Table 3 shows the perinatal outcomes of the affected twin versus the co‐twins following previable PROM.
TABLE 3.
Perinatal outcomes of the affected twin versus the co‐twin following previable PROM.
| Affected twin | Co‐twin | |||
|---|---|---|---|---|
| N | N | |||
| GA at delivery, weeks, mean (±SD) | 132 | 22.8 ± 4.5 | 173 | 24.9 ± 5.3 |
| Latency period, weeks, mean (±SD) | 104 | 5.5 ± 9.5 | 86 | 9.7 ± 6.9 |
| Birth weight (g), weeks, mean (±SD) | 44 | 593 ± 442 | 77 | 1692 ± 996 |
| IVH (%) | 4/46 | 8.7 | Not reported | |
| BPD (%) | 6/43 | 14.0 | 2/43 | 4.7 |
| NEC (%) | 7/46 | 15.2 | Not reported | |
| RDS (%) | 4/11 | 36.4 | 14/56 | 25.0 |
| Anemia (%) | 1/12 | 8.3 | Not reported | |
| Orthopedic problems (%) | 3/13 | 23.1 | Not reported | |
| PDA (%) | 2/12 | 16.7 | 4/50 | 8.0 |
| ROP (%) | 3/46 | 6.5 | 3/50 | 6.0 |
| Sepsis (%) | 3/14 | 21.4 | 5/51 | 9.8 |
| Pulmonary hypoplasia (%) | 9/59 | 15.3 | Not reported | |
Note: Denominators vary according to the number of cases for which data were reported in the original studies.
Abbreviations: BPD, bronchopulmonary dysplasia; GA, gestational age; IVH, intraventricular hemorrhage; NEC, necrotizing enterocolitis; PDA, patent ductus arteriosus; PROM, prelabor rupture of membranes; RDS, respiratory distress syndrome; ROP, retinopathy of prematurity; SD, standard deviation.
Table 4 shows the maternal outcomes in the pregnancies complicated by previable PROM of one twin. Interval delivery occurred in 52.6% of the pregnancies (71/135). Clinical chorioamnionitis was reported in 33.5% (69/206), and chorioamnionitis on pathology was found in 46.9% of the examined placentas (15/32). Maternal sepsis occurred in 7.8% (9/115) of the pregnancies. Placental abruption was reported in 5.9% (10/169).
TABLE 4.
Maternal outcomes in pregnancies complicated by previable PROM of one twin.
| N | (%) | |
|---|---|---|
| Interval delivery | 71/135 | 52.6 |
| Cesarean delivery | 34/67 | 50.7 |
| Clinical chorioamnionitis | 69/206 | 33.5 |
| Chorioamnionitis on pathology | 15/32 | 46.9 |
| Placental infarcts | 2/32 | 6.3 |
| Placental abruption | 10/169 | 5.9 |
| Cord prolapse | 7/147 | 4.8 |
| Maternal sepsis | 9/115 | 7.8 |
| Septic shock | 2/128 | 1.6 |
| Hysterectomy | 1/125 | 0.8 |
| Intensive care unit admission | 1/115 | 0.9 |
| Maternal death | 0/125 | 0 |
Note: Denominators vary according to the number of cases for which data were reported in the original studies.
Abbreviation: PROM, prelabor rupture of membranes.
Livebirths of one twin occurred in 42.6% (69/162), and livebirths of both twins in 68.9% (111/161). Neonatal death of one twin was reported in 15.0% (28/186). One infant was discharged home alive in 51.6% (66/128), and two infants were discharged home alive in 16.4% (21/128). Table 5 shows neonatal survival and discharge outcomes following previable PROM of one twin.
TABLE 5.
Neonatal survival and discharge outcomes following previable PROM of one twin.
| N | (%) | |
|---|---|---|
| Livebirth – one twin | 69/162 | 42.6 |
| Livebirth – both twins | 111/161 | 68.9 |
| Neonatal death – one twin | 28/186 | 15.0 |
| Neonatal death – both twins | 2/161 | 2.7 |
| No infant discharged home alive | 41/128 | 32.0 |
| One infant discharged home alive | 66/128 | 51.6 |
| Two infants discharged home alive | 21/128 | 16.4 |
Note: Denominators vary according to the number of cases for which data were reported in the original studies.
Abbreviation: PROM, prelabor rupture of membranes.
In a subanalysis of pregnancies managed expectedly, for those with previable PROM occurring before 20 versus 20–23+6 weeks of gestation, the latency period for the first twin was similar (6.38 ± 6.74 weeks and 3.96 ± 6.78 weeks, respectively, P = 0.119). However, the latency period for the second twin was significantly longer in pregnancies with earlier than later previable PROM (10.48 ± 6.13 vs. 5.50 ± 3.70 weeks). For pregnancies with a rupture before 20 weeks compared to 20–23+6 weeks, one infant was more often discharged home (54.1% vs. 31.0%, P = 0.038), and two infants less often discharged (13.5% vs. 38.1%, P = 0.014). The proportion of pregnancies in which no infant was discharged home alive was similar between the groups (32.4% vs. 31.0%, P = 0.888).
Another subanalysis focused on outcomes of selective reduction (N = 47) versus expectant management (N = 239). Monochorionic diamniotic twin pregnancies were observed in 0/47 (0.0%) of the selective reduction group and in 9/170 (5.3%) of the expectant management group (P = 0.107). Antibiotic treatment was administered in 39/41 (95.1%) women following selective reduction and in 182/201 (90.5%) of those managed expectantly (P = 0.343). Cerclage was performed in 1/31 (3.2%) of the selective reduction group and 24/164 (14.6%) of the expectant management group (P = 0.081). PROM in sac A occurred in 26/41 (63.4%) of the selective reduction group and in 101/115 (87.8%) of the expectant management group (P = 0.001). The gestational age at PROM was lower in the selective reduction than the expectant management group (18.1 ± 2.5 weeks versus 19.6 ± 2.9 weeks, P = 0.005). Table 6 compares pregnancy and neonatal outcomes between the selective reduction and expectant management groups following previable PROM. In the selective reduction group, interval delivery was less frequent (16.1% vs. 63.5%, P < 0.001) and latency to delivery of the co‐twin was longer (15.0 ± 8.4 weeks vs. 8.3 ± 5.7 weeks, P = 0.004). For the selective reduction group, the mean gestational age at delivery of the co‐twin was higher (31.6 ± 7.7 weeks vs. 24.4 ± 4.4 weeks, P < 0.001) and the mean birth weight was greater (2663.6 ± 703.9 vs. 1251.3 ± 773.0 g, P < 0.001). Following selective reduction, one infant was more frequently discharged home alive (66.0% vs. 33.8%, P < 0.001), the discharge of two infants was not possible (0.0% vs. 25.7%, P < 0.001). In the selective reduction group, neonatal death and clinical chorioamnionitis occurred less frequently (3.6% vs. 17.1%, P = 0.033 and 19.1% vs. 40.3%, P = 0.008). These findings are summarized in the forest plots demonstrating mean differences in latency and gestational age at delivery, as well as odds ratios for neonatal survival and infectious complications (Figure 2a,b).
TABLE 6.
Pregnancy and neonatal outcomes following selective reduction versus expectant management for previable PROM.
| Variable | Selective reduction | Expectant management | P value |
|---|---|---|---|
| Interval delivery | 5/31 (16.1) | 66/104 (63.5) | <0.001 |
| Latency, the affected twin (weeks) | 7.7 ± 16.5 | 4.9 ± 7.2 | 0.226 |
| Latency, the co‐twin (weeks) | 15.0 ± 8.4 | 8.3 ± 5.7 | 0.004 |
| GA delivery, the affected twin (weeks) | 28.1 ± 8.8 | 22.8 ± 4.2 | 0.009 |
| GA delivery, the co‐twin (weeks) | 31.6 ± 7.7 | 24.4 ± 4.4 | <0.001 |
| Birth weight, the affected twin (g) | – | 593.0 ± 442.0 | – |
| Birth weight, the co‐twin (g) | 2663.6 ± 703.9 | 1251.3 ± 773.0 | <0.001 |
| No infant discharged home alive | 16/47 (34.0) | 42/109 (38.5) | 0.594 |
| One infant discharged home alive | 31/47 (66.0) | 47/139 (33.8) | <0.001 |
| Two infants discharged home alive | 0/47 (0.0) | 45/175 (25.7) | <0.001 |
| Neonatal death | 1/28 (3.6) | 27/158 (17.1) | 0.033 |
| Clinical chorioamnionitis | 9/47 (19.1) | 60/149 (40.3) | 0.008 |
| Chorioamnionitis on pathology | 0/7 (0.0) | 15/25 (60.0) | 0.002 |
| Placental abruption | 1/30 (3.3) | 9/139 (6.5) | 0.508 |
| Cord prolapse | 0/30 (0.0) | 7/117 (6.0) | 0.17 |
| Maternal sepsis | 0/30 (0.0) | 9/85 (10.6) | 0.063/0.059 # |
| Hysterectomy | 0/30 (0.0) | 1/95 (1.1) | 0.573 |
| ICU admission | 0/30 (0.0) | 1/85 (1.2) | 0.551 |
| Maternal death | 0/30 (0.0) | 0/95 (0) | NA |
| Septic shock | 0/30 (0.0) | 2/98 (2.0) | 0.43 |
Note: Denominators vary according to the number of cases for which data were reported in the original studies. The data are presented as mean ± SD for continuous variables, and as number (percentage) for categorical variables.
Abbreviations: GA, gestational age; ICU, intensive care unit; PROM, prelabor rupture of membranes; SD, standard deviation.
P value one‐sided.
FIGURE 2.
(a) Forest plot showing mean differences (95% confidence interval [CI]) for latency and gestational age (GA) at delivery in affected twins and co‐twins. (b) Forest plot showing odds ratios (95% CI) for neonatal survival, chorioamnionitis, and maternal infectious complications.
When comparing selective reduction of twin A versus twin B, an interval delivery occurred in 5/16 (31.3%) of twin A cases compared to 0/9 (0.0%) of twin B (P = 0.123). Livebirth of one twin was reported in 13/16 (81.3%) of twin A and 9/9 (100.0%) of twin B (P = 0.280), and one infant discharged home alive occurred in 13/16 (81.3%) versus 9/9 (100.0%) (P = 0.280). No neonatal deaths were reported in either group. There were no cases of neonatal complications—including respiratory distress syndrome (RDS), sepsis, patent ductus arteriosus (PDA), or bronchopulmonary dysplasia (BPD)—in either group (data available for twin A: 11; twin B: 9). Chorioamnionitis occurred in 1/16 (6.3%) of twin A versus 0/9 (0.0%) of twin B (P = 1.000), and placental abruption occurred in 1/15 (6.7%) versus 0/9 (0.0%) (P = 1.000).
4. DISCUSSION
This systematic review of 286 twin pregnancies highlights the high maternal and neonatal risks associated with previable PROM before 24 weeks. Neonatal morbidity and mortality were substantial, with no infant discharged alive following about one‐third of the pregnancies, and frequent maternal complications, particularly chorioamnionitis and sepsis. Overall, selective reduction improve outcomes for the co‐twin by prolonging latency and reducing maternal morbidity, though it eliminated the chance of dual survival.
TOP is widely accepted as a reasonable option in the setting of previable PROM, given the high risk of adverse maternal and neonatal outcomes. Yet, major guidelines from the Society for Maternal‐Fetal Medicine, and Royal College of Obstetricians and Gynecologists do not differentiate between singleton and twin pregnancies, and management recommendations are largely based on data from singletons. 8 , 55 This creates a critical gap in evidence‐based guidance for twin gestations. 56 Our findings support the inclusion of TOP in counseling discussions for these pregnancies. 55 , 56 Neonatal morbidity and mortality were high across the cohort, with no infant discharged home alive following about one‐third of the pregnancies. Maternal complications were also substantial, including a 33.5% rate of clinical chorioamnionitis, nearly 8% developing sepsis, and one woman requiring hysterectomy. Among those managed expectantly, nearly one‐third of the women in both gestational age groups (before 20 weeks and 20–23+6 weeks) did not take a live infant home. Moreover, chorioamnionitis rates were high in both groups, 31.4% and 55.8%, respectively (P = 0.040). These findings emphasize the need for comprehensive, honest counseling that includes not only the possibility of neonatal death and dual loss, but also the considerable maternal risks that can accompany prolonged expectant management. Importantly, our sub‐analysis revealed a longer latency period for the second twin in pregnancies with PPROM occurring before 20 weeks versus 20–23+6 weeks of gestation. However, it remains unclear as to whether pregnancies with rupture at 22–23+6 weeks, lying within the gray zone of periviability, are more likely to result in better outcomes. There is limited evidence to guide management in this range. Furthermore, as our review focused on pregnancies up to 23+6 weeks, we lack sufficient data regarding outcomes in the 24–26‐week gestational window. In this periviable period, clinical decisions remain especially complex and underexplored.
Our results show that selective reduction was associated with improved outcomes for the unaffected co‐twin, including a significantly lower rate of neonatal death and a higher chance of discharging one infant home alive. However, this benefit was at the expense of eliminating the possibility of taking both infants home, which obviously was only possible in the expectant management group. This trade‐off aligns with the central goal of selective reduction in this setting: to stabilize the intrauterine environment and favor the survival of the unaffected twin, particularly when the prognosis for the affected twin is poor. 50 Prolonged latency and higher gestational age at delivery have been reported following selective feticide. 50 , 54 Zhu et al., for instance, found that gestational age at delivery was higher and delivery beyond 32 weeks more common following selective reduction. 54 However, they also noted that selective feticide was only beneficial when performed for rupture of the upper sac. In contrast, our findings suggest a broader applicability. While about one‐third of the selective reductions involved rupture of the lower sac, improved outcomes were observed, including longer latency and lower maternal infection rates. Although the data is limited, we did not observe a difference in livebirth rates between reductions performed in twin A versus twin B. From a maternal perspective, selective reduction was associated with fewer complications, particularly clinical chorioamnionitis. This supports the idea that removal of the affected twin may reduce ongoing inflammation and the risk of ascending infection. 5 , 54 Importantly, for both the selective reduction and expectant management groups, the rates of antibiotic treatment were comparable, suggesting that these outcome differences were not driven by disparities in medical management.
Our findings underscore the need for individualized, family‐centered decision making in the management of previable twin pregnancies complicated by PPROM. Choices regarding expectant management or termination, including selective reduction, are often ethically complex and deeply personal. Prognoses can rapidly evolve, shifting from the possibility of dual survival to escalating maternal risk with limited fetal benefit. As emphasized by Ponce et al., 5 a multidisciplinary approach is essential, incorporating input from maternal‐fetal medicine specialists, neonatologists, and, when appropriate, palliative care teams. Counseling should be realistic, empathetic, and grounded in individualized risk assessments, while considering local thresholds for neonatal viability and aligning with the family's values and goals.
Strengths of this systematic review study include strict eligibility criteria, and a comprehensive search strategy across major databases. By focusing on a clearly defined clinical question, the study offers important insights to guide counseling and management decisions for a rare and high‐risk obstetrical condition. Comparisons between selective reduction and expectant management enabled direct assessment of maternal, pregnancy, and neonatal outcomes between these two approaches. However, several limitations must be considered. Our review was limited to pregnancies with rupture before 24+0 weeks, although the 24–26‐week window may still be considered by some as part of the previable or early periviable period. We acknowledge that excluding those gestations narrowed the dataset and may have omitted outcomes relevant to this gray zone. However, because management practices and neonatal interventions typically change after 24 weeks, inclusion of such gestations would have introduced clinical heterogeneity and biased estimates toward better outcomes. The included studies were case reports and small case series, thus increasing the risk of selection, information, and reporting biases. Heterogeneity in management protocols, outcome definitions, and reporting quality limited the ability to standardize analyses and may have introduced confounding. Additionally, some PROM events occurred after invasive procedures, which may be associated with better outcomes and could bias results. Subgroup analyses including only such cases were not performed due to the small number of reports. It remains unclear whether selective reduction is more beneficial when the affected sac is A or B, and this uncertainty limits the ability to refine clinical recommendations. Importantly, long‐term neonatal outcomes, especially regarding neurologic development, were not consistently reported, and thus remain a significant gap in the current evidence. Moreover, the possibility of publication bias cannot be fully ruled out. As most of the included studies were descriptive case reports or small series, the findings should be viewed as hypothesis‐generating rather than definitive, and as reflecting current evidence gaps rather than guiding clinical practice.
In conclusion, this systematic review highlighted the complex clinical landscape of twin pregnancies complicated by previable PROM before 24 weeks, revealing high rates of neonatal morbidity, maternal complications, and difficult trade‐offs between survival and risk. Selective reduction may offer improved outcomes for the co‐twin and reduce maternal morbidity, yet at the expense of dual survival. Shared decision making, guided by multidisciplinary counseling and individualized risk assessment, remains essential to support families navigating these challenging scenarios.
AUTHOR CONTRIBUTIONS
M.O.: manuscript drafting and interpretation of data for the article, M.F.W.: contributed to the concept and design of the article and data acquisition. F.K.: contributed to data acquisition and data analysis. R.S.K.: contributed to the concept and design of the article and data acquisition. A.A.: contributed to the concept and design of the article and interpretation of data for the article. L.L.: contributed to the concept and design of the article and interpretation of data. I.S.: contributed to the concept and design of the article; data acquisition, data analysis, and interpretation of data for the article. All listed authors should have contributed to the manuscript substantially and have agreed to the final submitted version.
FUNDING INFORMATION
None.
COMPETING INTERESTS
The authors have no relevant financial or non‐financial interests to disclose.
Supporting information
Table S1.
Table S2.
Table S3.
Table S4.
Table S5.
DATA AVAILABILITY STATEMENT
Research data are not shared.
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Associated Data
This section collects any data citations, data availability statements, or supplementary materials included in this article.
Supplementary Materials
Table S1.
Table S2.
Table S3.
Table S4.
Table S5.
Data Availability Statement
Research data are not shared.