Introduction
Research suggests approximately 14% of women are affected by extreme fear of childbirth (FOC), also known as tokophobia (Nilsson et al., Reference Nilsson, Hessman, Sjöblom, Dencker, Jangsten, Mollberg and Begley2018; O'Connell, Leahy-Warren, Khashan, Kenny, & O'Neill, Reference O'Connell, Leahy-Warren, Khashan, Kenny and O'Neill2017). The causes of FOC are thought to be linked to obstetric (Fairbrother, Thordarson, & Stoll, Reference Fairbrother, Thordarson and Stoll2018; Haines, Pallant, Karlström, & Hildingsson, Reference Haines, Pallant, Karlström and Hildingsson2011; Sydsjö et al., Reference Sydsjö, Angerbjörn, Palmquist, Bladh, Sydsjö and Josefsson2013; Sydsjö et al., Reference Sydsjö, Bladh, Lilliecreutz, Persson, Vyoni and Josefsson2014), psychological (Dencker et al., Reference Dencker, Nilsson, Begley, Jangsten, Mollberg, Patel and Sparud-Lundin2019; Hall, Stoll, Hutton, & Brown, Reference Hall, Stoll, Hutton and Brown2012; Jokić-Begić, Žigić, & Nakić Radoš, Reference Jokić-Begić, Žigić and Nakić Radoš2014; Lukasse, Vangen, Ãian, & Schei, Reference Lukasse, Vangen, Ãian and Schei2011) and socio-demographic factors (Haines et al., Reference Haines, Pallant, Karlström and Hildingsson2011; O'Connell, Leahy-Warren, Kenny, O'Neill, & Khashan, Reference O'Connell, Leahy-Warren, Kenny, O'Neill and Khashan2019; Ryding et al., Reference Ryding, Lukasse, Van Parys, Wangel, Karro, Kristjansdottir and Laanpere2015). FOC can impact on women's birth choices and outcomes. For example, those with FOC are more likely to opt for an elective caesarean section (CS) (Eide, Morken, & Bærøe, Reference Eide, Morken and Bærøe2019; Ryding et al., Reference Ryding, Lukasse, Van Parys, Wangel, Karro, Kristjansdottir and Laanpere2015; Sydsjö, Sydsjö, Gunnervik, Bladh, & Josefsson, Reference Sydsjö, Sydsjö, Gunnervik, Bladh and Josefsson2012), choose an epidural, have longer labours (Dencker et al., Reference Dencker, Nilsson, Begley, Jangsten, Mollberg, Patel and Sparud-Lundin2019; Logtenberg et al., Reference Logtenberg, Verhoeven, Rengerink, Sluijs, Freeman, Schellevis and Mol2018; Reck et al., Reference Reck, Zimmer, Dubber, Zipser, Schlehe and Gawlik2013) or have an emergency CS (Sydsjö et al., Reference Sydsjö, Sydsjö, Gunnervik, Bladh and Josefsson2012).
FOC has also been found to be related to postnatal psychological adjustment. For example, FOC is associated with postnatal post-traumatic stress disorder (Capik & Durmaz, Reference Capik and Durmaz2018; Söderquist, Wijma, & Wijma, Reference Söderquist, Wijma and Wijma2004; Wijma, Ryding, & Wijma, Reference Wijma, Ryding and Wijma2002; Wijma, Söderquist, & Wijma, Reference Wijma, Söderquist and Wijma1997), depression and anxiety (Räisänen et al., Reference Räisänen, Lehto, Nielsen, Gissler, Kramer and Heinonen2013; Rouhe, Salmela-Aro, Gissler, Halmesmäki, & Saisto, Reference Rouhe, Salmela-Aro, Gissler, Halmesmäki and Saisto2011) as well as antenatal depression and anxiety (Andersson, Sundström-Poromaa, Wulff, Åström, & Bixo, Reference Andersson, Sundström-Poromaa, Wulff, Åström and Bixo2004). The long-term impact of FOC on mother–infant outcomes has yet to be investigated; however, research has evaluated the impact of the psychological conditions associated with FOC such as anxiety and depression (Stein et al., Reference Stein, Pearson, Goodman, Rapa, Rahman, McCallum and Pariante2014). Antenatal mood disorders have been associated with altered patterns of foetal behaviour and heart rate responses (Kinsella & Monk, Reference Kinsella and Monk2009), more fearful or anxious behaviour in the infant, and increased risk of poor development and adverse child outcomes (Talge, Neal, & Glover, Reference Talge, Neal and Glover2007). Furthermore, antenatal depressive symptoms have been linked to poorer maternal responsiveness 12 months postnatally (Pearson et al., Reference Pearson, Melotti, Heron, Joinson, Stein, Ramchandani and Evans2012).
The development of FOC has still yet to be established in the literature; however, a systematic review of 89 studies found that a variety of factors may contribute to the development (Rondung, Thomtén, & Sundin, Reference Rondung, Thomtén and Sundin2016). In a theory of fear acquisition, Rachman (Reference Rachman1977) proposed that fears are developed through three pathways (conditioning, vicarious experiences and transmission of information), and the literature appears to support this model (Rondung et al., Reference Rondung, Thomtén and Sundin2016). For example, in terms of conditioning, research suggests that negative birth experiences cause future FOC (Lukasse et al., Reference Lukasse, Vangen, Ãian and Schei2011; Lukasse, Schei, & Ryding, Reference Lukasse, Schei and Ryding2014; Nilsson, Lundgren, Karlström, & Hildingsson, Reference Nilsson, Lundgren, Karlström and Hildingsson2012). Research also suggests that vicarious experiences such as viewing a live birth is associated with a reduction in fear (Stoll & Hall, Reference Stoll and Hall2013) and research also suggests that the transmission of information such as negative childbirth stories (Melender, Reference Melender2002; Tsui et al., Reference Tsui, Pang, Melender, Xu, Lau and Leung2007) and public discourses of birth (Fenwick, Hauck, Downie, & Butt, Reference Fenwick, Hauck, Downie and Butt2005; Melender, Reference Melender2002) also contribute to the development of FOC.
The systematic review also suggested that cognitive aspects may play an important role in FOC (Rondung et al., Reference Rondung, Thomtén and Sundin2016). For example, women with childbirth fear more commonly report having childbirth-related thoughts compared with women with no fear (Hildingsson, Thomas, Karlström, Olofsson, & Nystedt, Reference Hildingsson, Thomas, Karlström, Olofsson and Nystedt2010). Furthermore, FOC is negatively correlated with childbirth self-efficacy (Beebe, Lee, Carrieri-Kohlman, & Humphreys, Reference Beebe, Lee, Carrieri-Kohlman and Humphreys2007) and a woman's appraisal of her ability to cope with stressful situations (Ryding, Wijma, Wijma, & Rydhström, Reference Ryding, Wijma, Wijma and Rydhström1998; Söderquist et al., Reference Söderquist, Wijma and Wijma2004). The review found that behavioural aspects are also important (Rondung et al., Reference Rondung, Thomtén and Sundin2016) with FOC being associated with avoidance of pregnancy (Tsui et al., Reference Tsui, Pang, Melender, Xu, Lau and Leung2007), or avoidance of vaginal delivery (Dehghani, Sharpe, & Khatibi, Reference Dehghani, Sharpe and Khatibi2014; D'Cruz & Lee, Reference D'Cruz and Lee2014; Matinnia et al., Reference Matinnia, Faisal, Juni, Herjar, Moeini and Osman2015; Nieminen, Stephansson, & Ryding, Reference Nieminen, Stephansson and Ryding2009). Physiological aspects may also be associated with FOC, such as sleep disturbances, tachycardia, tenseness, restlessness and nervousness (Rondung et al., Reference Rondung, Thomtén and Sundin2016). This complex conceptualisation of the potential causes of FOC suggests that interventions for FOC need to focus on strategies to improve psychological, cognitive, behavioural and physiological aspects of the fear.
To prevent poorer outcomes related to FOC, early detection and evidence-based interventions are key, and should therefore be available to women. Reviews of the evidence suggest that interventions with an educational component may reduce FOC (Moghaddam Hosseini, Nazarzadeh, & Jahanfar, Reference Moghaddam Hosseini, Nazarzadeh and Jahanfar2018; Stoll, Swift, Fairbrother, Nethery, & Janssen, Reference Stoll, Swift, Fairbrother, Nethery and Janssen2018; Striebich, Mattern, & Ayerle, Reference Striebich, Mattern and Ayerle2018). However, previous reviews used narrow inclusion criteria such as requiring a minimum score on a FOC measure (Striebich et al., Reference Striebich, Mattern and Ayerle2018); requiring FOC to be measured twice (Stoll et al., Reference Stoll, Swift, Fairbrother, Nethery and Janssen2018); or only including clinical trials (Moghaddam Hosseini et al., Reference Moghaddam Hosseini, Nazarzadeh and Jahanfar2018). This means the reviews include very few studies (range = 7–15) and may exclude studies that could provide further insight into effective FOC treatments. Further, the previous reviews did not evaluate evidence quantitatively using meta-analysis. This may have led to a bias in conclusions where interventions that have been more widely examined are more likely to be evaluated as promising. Therefore, this study aimed to conduct a systematic review and meta-analysis to identify the effectiveness of FOC interventions for reducing FOC or rates of caesarean by request during the perinatal period.
Methods
Search strategies
Literature searches and study selection were conducted according to the Preferred Reporting Items for Systematic Reviews and Meta-analyses (PRISMA) guidelines (Moher et al., Reference Moher, Shamseer, Clarke, Ghersi, Liberati, Petticrew and Stewart2015) (online Supplementary Table S1). The protocol was registered with PROSPERO (CRD42018093095). Online databases were used to identify papers. Searches were carried out in Cochrane Library (1996–present), EMBASE (1947–present), Healthcare Management Information Consortium (HMIC; 1983–present), Medline (1946–present); PsychInfo (1967–present), PsychARTICLES (1806–present), PubMed (1996–present), SCOPUS (2004–present) and Web of Science (1997–present). Pre-planned searches were carried out using search terms combined with Boolean operators ‘OR’ and ‘AND’ (e.g. pregnancy OR perinatal OR postnat* AND fear of childbirth OR tokophobia AND Caesarean OR abdom* deliver* AND counselling OR intervention). Full search syntax can be found in the supporting information. Searches were conducted up to and including June 2019. Two searches were carried out: (1) desire for an elective CS and interventions; (2) FOC and interventions (see Appendix S1). Desire for an elective CS was used as a proxy measure for FOC. Reference lists of all identified papers were also searched.
Inclusion and exclusion criteria
The following inclusion criteria were applied: Participants – women in the perinatal period (the UK definition of perinatal period was used: pregnancy – 1 year after birth; NHS England, 2019) with FOC or tokophobia; Intervention – any intervention that was for women with FOC; Outcome – a measure of FOC or birth outcomes. Studies were excluded if they were conference abstracts, theses, non-English publications or non-empirical papers.
Study selection and data extraction
The results from both searches were imported into Eppi-Reviewer 4. All duplicate papers were removed and studies screened for eligibility based on their title and abstract by one reviewer (RW). A proportion (10% n = 284) were double screened by BRG. Reviewers agreed on inclusion/exclusion of studies 99% (n = 282/284) of the time. Studies that were not eligible were excluded. Full texts of studies that appeared to meet criteria or where it was unclear were then reviewed by one reviewer (RW) to determine whether they should be included. A proportion (10%, n = 9) were double screened by BRG. Reviewers agreed on inclusion/exclusion of studies 100% of the time. A decision to only double screen a proportion of titles and abstracts and papers was made based on the high level of agreement on screening suggesting that the inclusion and exclusion criteria were clear and that screening was accurate, the similar approach to double coding used in other reviews (Furuta, Sandall, & Bick, Reference Furuta, Sandall and Bick2012; Lucas, Olander, Ayers, & Salmon, Reference Lucas, Olander, Ayers and Salmon2019; Sambrook Smith, Lawrence, Sadler, & Easter, Reference Sambrook Smith, Lawrence, Sadler and Easter2019).
A data extraction sheet was designed and used to extract relevant information from the full texts. This included: (1) measure used; (2) language of the measure; (3) country; (4) number of participants; (5) participant group; (6) participant demographics; (7) design of the study; (8) norming data for the measure; (9) cut-off scale of the measure; (10) how the measure was administered. The primary outcome measure was reduction in FOC. Other outcome measures were self-efficacy, obstetric outcomes and childbirth experience. Data extraction for all studies was completed by RW. BRG completed data extraction for a proportion of studies (10%, n = 7). Agreement was high (88%). RW and BRG extracted effect sizes and statistics for the meta-analysis in duplicate.
Quality assessment
The Cochrane Risk of Bias Tool (Higgins et al., Reference Higgins, Altman, Gøtzsche, Jüni, Moher, Oxman and Sterne2011) was used to assess quality. Due to the nature of psychological intervention studies, both participants and personnel were aware of the intervention; therefore, as all studies would score negatively on the performance bias items, this was not assessed. The tool was adapted so that each bias criterion could be answered as ‘yes’, ‘no’ or ‘not applicable’ (n/a). Items that were scored yes were assigned a score of 1, items that were scored no were assigned a score of 0. These were then averaged (excluding the answers scored n/a) and multiplied by 100. Studies that scored between 0 and 33 were labelled as having a high risk of bias, those scoring between 34 and 66 were labelled as having a medium risk of bias, studies that scored between 67 and 100 were labelled as having a low risk of bias.
Data analysis
Studies were synthesised narratively then meta-analysis used to determine whether FOC interventions were effective for two types of outcome: (1) FOC in late pregnancy; and (2) CS birth. Potential moderators of the effectiveness of FOC interventions were examined, i.e. risk of bias, country, study design, sample (only women with FOC v. all women; nulliparous v. mixed parity), intensity of the intervention and type of intervention. Studies were excluded from the meta-analysis if they did not report effect sizes for relevant outcomes (n = 31), were based on samples with men only (n = 1), did not have a control group (n = 5), or where the FOC intervention was provided intrapartum (n = 1). Effect sizes were calculated as Cohen's d using the difference between the pre- and post-test means to control for any pretest group differences, and the posttest standard deviations (Lipsey & Wilson, Reference Lipsey and Wilson2001). For Haapio, Kaunonen, Arffman, and Åstedt-Kurki (Reference Haapio, Kaunonen, Arffman and Åstedt-Kurki2017), Cohen's d was derived from the odds ratio (Borenstein, Hedges, Higgins, & Rothstein, Reference Borenstein, Hedges, Higgins and Rothstein2009). Meta-analysis was carried out using R where the computation of a Q statistic was made. To see if publication bias was of any concern in this analysis, a funnel plot and trim and fill analysis (Duval & Tweedie, Reference Duval and Tweedie2000) were carried out using R.
Results
Study characteristics
Searches identified a total of 4474 citations. Hand searches of reference lists of key papers identified a further 19 papers. After removing duplicates and screening through abstracts, titles and full texts, 66 papers from 48 studies remained for inclusion in the review (Fig. 1). Participants within the studies varied. Only three studies reported ethnicity, all recruited white women (range: 17–71%) and two studies reported recruiting Black women (range: 25–50%). University education was reported by 19 studies, 12 of these reported more than half of participants having completed university. Of the 18 studies that reported marital status, between 57% and 100% of the participants were married or cohabiting. Most papers (n = 27) used the Wijma Delivery Expectance Questionnaire (WDEQ-A) to measure FOC. Scores of ⩽37 are indicative of mild fear, scores of 38–65 moderate fear, scores of 66–84 severe fear, and scores of ⩾85 clinical fear (Wijma, Wijma, & Zar, Reference Wijma, Wijma and Zar1998). Average WDEQ-A scores ranged from 29.7 to 130.
Fig. 1. PRISMA Flow Diagram.
One paper described their outcome variable as ‘tokophobia’ (Pour-Edalati, Moghadam, Shahesmaeili, & Salehi-Nejad, Reference Pour-Edalati, Moghadam, Shahesmaeili and Salehi-Nejad2019) and one paper used the terms FOC and tokophobia interchangeably (Bulez, Ceber Turfan, & Sogukpinar, Reference Bulez, Ceber Turfan and Sogukpinar2019). The remaining studies used FOC as their outcome variable. The majority of papers examined FOC interventions with women (see online Supplementary Table S2). The exceptions were two studies with fathers (Bergström, Rudman, Waldenström, & Kieler, Reference Bergström, Rudman, Waldenström and Kieler2013; Ryding et al., Reference Ryding, Read, Rouhe, Halmesmäki, Salmela-Aro, Toivanen and Saisto2018) and one with couples (Ahmadi et al., Reference Ahmadi, Karami, Faghihzadeh, Jafari, Oskoei and Kharaghani2018). Studies were mostly conducted in non-English speaking countries: Iran (n = 8), Sweden (n = 9), Turkey (n = 9). Sample sizes ranged from 10 to 1887. Three studies had a high risk of bias, 30 had a medium risk of bias and 15 had a low risk of bias (online Supplementary Table S3).
Interventions identified
Interventions were grouped into six broad categories:
• Cognitive/cognitive behavioural (11 papers from six studies): these were interventions which used strategies to change cognitions (e.g. psychoeducation, thought restructuring, problem solving) and behaviours (e.g. exposure, relaxation).
• Other talking therapies (16 papers from 12 studies): interventions that used therapeutic conversation (e.g. counselling, haptotherapy, psychotherapy).
• Antenatal education (18 papers from 13 studies): these were interventions where the main focus was education about pregnancy and birth.
• Enhanced midwifery care (six papers from three studies) comprised continuity of carer or a midwife-led visit to delivery suite.
• Alternative interventions (13 papers from 12 studies): interventions involving specific therapies such as hypnobirthing, stand-alone relaxation, Pilates, art therapy.
• Interventions during labour (two papers from two studies) used intrapartum music or emotional freedom technique (EFT)
Cognitive/cognitive behavioural
Six studies examined cognitive behavioural therapy (CBT) delivered online (n = 2; Baylis, Ekdahl, Haines, & Rubertsson, Reference Baylis, Ekdahl, Haines and Rubertsson2019; Hildingsson & Rubertsson, Reference Hildingsson and Rubertsson2019; Larsson et al., Reference Larsson, Karlström, Rubertsson, Ternström, Ekdahl, Segebladh and Hildingsson2017; Larsson, Hildingsson, Ternström, Rubertsson, & Karlström, Reference Larsson, Hildingsson, Ternström, Rubertsson and Karlström2019; Nieminen et al., Reference Nieminen, Malmquist, Wijma, Ryding, Andersson and Wijma2015; Nieminen, Andersson, Wijma, Ryding, & Wijma, Reference Nieminen, Andersson, Wijma, Ryding and Wijma2016; Rondung et al., Reference Rondung, Ternström, Hildingsson, Haines, Sundin, Ekdahl and Rubertsson2018) or face-to-face (n = 4; Kordi, Bakhshi, Masoudi, & Esmaily, Reference Kordi, Bakhshi, Masoudi and Esmaily2017; Saisto, Salmela-Aro, & Nurmi, Reference Saisto, Salmela-Aro and Nurmi2001; Sydsjö et al., Reference Sydsjö, Blomberg, Palmquist, Angerbjörn, Bladh and Josefsson2015; Uçar & Golbasi, Reference Uçar and Golbasi2019). Studies either had no control group (n = 1; Nieminen et al., Reference Nieminen, Malmquist, Wijma, Ryding, Andersson and Wijma2015, Reference Nieminen, Andersson, Wijma, Ryding and Wijma2016) or compared the intervention to other talking therapies (n = 2; Baylis et al., Reference Baylis, Ekdahl, Haines and Rubertsson2019; Hildingsson & Rubertsson, Reference Hildingsson and Rubertsson2019; Larsson et al., Reference Larsson, Karlström, Rubertsson, Ternström, Ekdahl, Segebladh and Hildingsson2017, Reference Larsson, Hildingsson, Ternström, Rubertsson and Karlström2019; Rondung et al., Reference Rondung, Ternström, Hildingsson, Haines, Sundin, Ekdahl and Rubertsson2018; Saisto et al., Reference Saisto, Salmela-Aro and Nurmi2001) or standard medical care (SMC; n = 3; Kordi et al., Reference Kordi, Bakhshi, Masoudi and Esmaily2017; Sydsjö et al., Reference Sydsjö, Blomberg, Palmquist, Angerbjörn, Bladh and Josefsson2015; Uçar & Golbasi, Reference Uçar and Golbasi2019). Two studies had a low risk of bias and the rest medium.
Three studies, including online CBT, found a reduction in FOC symptoms (Kordi et al., Reference Kordi, Bakhshi, Masoudi and Esmaily2017; Nieminen et al., Reference Nieminen, Andersson, Wijma, Ryding and Wijma2016; Uçar & Golbasi, Reference Uçar and Golbasi2019). However, there was no impact of CBT on birth preference, birth mode (Larsson et al., Reference Larsson, Karlström, Rubertsson, Ternström, Ekdahl, Segebladh and Hildingsson2017) or birth experience (Hildingsson & Rubertsson, Reference Hildingsson and Rubertsson2019), in fact one study found CBT was associated with an increase in negative birth outcomes (Sydsjö et al., Reference Sydsjö, Blomberg, Palmquist, Angerbjörn, Bladh and Josefsson2015).
Other talking therapies
Twelve studies examined other talking therapies (excluding CBT) delivered face-to-face (n = 11; Ahmadi et al., Reference Ahmadi, Karami, Faghihzadeh, Jafari, Oskoei and Kharaghani2018; Andaroon, Kordi, Kimiaei, & Esmaeily, Reference Andaroon, Kordi, Kimiaei and Esmaeily2017; Halvorsen, Nerum, Sørlie, & Øian, Reference Halvorsen, Nerum, Sørlie and Øian2010; Henriksen, Borgen, Risløkken, & Lukasse, Reference Henriksen, Borgen, Risløkken and Lukasse2018; Klabbers, Paarlberg, & Vingerhoets, Reference Klabbers, Paarlberg and Vingerhoets2018; Klabbers, Wijma, Paarlberg, Emons, & Vingerhoets, Reference Klabbers, Wijma, Paarlberg, Emons and Vingerhoets2019; Larsson, Karlström, Rubertsson, & Hildingsson, Reference Larsson, Karlström, Rubertsson and Hildingsson2015; Nerum, Halvorsen, Sørlie, & Øian, Reference Nerum, Halvorsen, Sørlie and Øian2006; Ryding, Persson, Onell, & Kvist, Reference Ryding, Persson, Onell and Kvist2003; Sjogren, Reference Sjogren1998; Soltani, Eskandari, Khodakarami, Parsa, & Roshanaei, Reference Soltani, Eskandari, Khodakarami, Parsa and Roshanaei2017; Sydsjö et al., Reference Sydsjö, Sydsjö, Gunnervik, Bladh and Josefsson2012) or over the telephone (n = 1; Fenwick et al., Reference Fenwick, Toohill, Gamble, Creedy, Buist, Turkstra and Ryding2015; Toohill et al., Reference Toohill, Fenwick, Gamble, Creedy, Buist, Turkstra and Ryding2014; Toohill, Callander, Gamble, Creedy, & Fenwick, Reference Toohill, Callander, Gamble, Creedy and Fenwick2017; Turkstra et al., Reference Turkstra, Mihala, Scuffham, Creedy, Gamble, Toohill and Fenwick2017). These approaches are a very diverse group, drawing on different theoretical frameworks including generic counselling, haptotherapy and psychotherapy. Studies either had no control group (n = 2) or compared other talking therapies to SMC (n = 7) or no intervention (n = 3). Two studies had a low risk of bias, one had high risk and the remaining medium risk.
Three studies, including other talking therapies carried out over the phone, found a reduction in FOC (Andaroon et al., Reference Andaroon, Kordi, Kimiaei and Esmaeily2017; Soltani et al., Reference Soltani, Eskandari, Khodakarami, Parsa and Roshanaei2017; Toohill et al., Reference Toohill, Fenwick, Gamble, Creedy, Buist, Turkstra and Ryding2014). However, two studies found no change in women's FOC after receiving other talking therapy (Larsson et al., Reference Larsson, Karlström, Rubertsson and Hildingsson2015; Ryding et al., Reference Ryding, Persson, Onell and Kvist2003). All three studies that looked at the impact of other talking therapies on birth preference found a reduction in desire for a CS (Fenwick et al., Reference Fenwick, Toohill, Gamble, Creedy, Buist, Turkstra and Ryding2015; Halvorsen et al., Reference Halvorsen, Nerum, Sørlie and Øian2010; Nerum et al., Reference Nerum, Halvorsen, Sørlie and Øian2006). Three studies found other talking therapy was associated with a lower risk of CS (Ahmadi et al., Reference Ahmadi, Karami, Faghihzadeh, Jafari, Oskoei and Kharaghani2018; Fenwick et al., Reference Fenwick, Toohill, Gamble, Creedy, Buist, Turkstra and Ryding2015; Toohill et al., Reference Toohill, Callander, Gamble, Creedy and Fenwick2017); however, two studies found an increased risk of CS (Henriksen et al., Reference Henriksen, Borgen, Risløkken and Lukasse2018; Sydsjö et al., Reference Sydsjö, Sydsjö, Gunnervik, Bladh and Josefsson2012).
Antenatal education
Thirteen studies examined antenatal education classes. All but two (Khedr & Eldeen, Reference Khedr and Eldeen2017; Kulkarni, Wright, & Kingdom, Reference Kulkarni, Wright and Kingdom2014) interventions were delivered face-to-face. Studies either had no control group (n = 2; Khedr & Eldeen, Reference Khedr and Eldeen2017; Kulkarni et al., Reference Kulkarni, Wright and Kingdom2014) or compared antenatal education to SMC (n = 10) or no intervention (n = 1; Taheri, Mazaheri, Khorsandi, Hassanzadeh, & Amiri, Reference Taheri, Mazaheri, Khorsandi, Hassanzadeh and Amiri2014). Five studies were RCTs with low risk of bias (Bergström et al., Reference Bergström, Rudman, Waldenström and Kieler2013; Haapio et al., Reference Haapio, Kaunonen, Arffman and Åstedt-Kurki2017; Masoumi et al., Reference Masoumi, Kazemi, Oshvandi, Jalali, Esmaeili-Vardanjani and Rafiei2016; Ozdemir, Cilingir, Ilhan, Yildiz, & Ohanoglu, Reference Ozdemir, Cilingir, Ilhan, Yildiz and Ohanoglu2018; Rouhe et al., Reference Rouhe, Salmela-Aro, Toivanen, Tokola, Halmesmäki, Ryding and Saisto2015).
All but one study (Masoumi et al., Reference Masoumi, Kazemi, Oshvandi, Jalali, Esmaeili-Vardanjani and Rafiei2016) found that antenatal education was associated with a reduction of FOC in women (El-Malky, El-Homosy, Ashour, & Shehada, Reference El-Malky, El-Homosy, Ashour and Shehada2018; Gökçe İsbir, İnci, Önal, & Dikmen-Yıldız, Reference Gökçe İsbir, İnci, Önal and Dikmen-Yıldız2016; Haapio et al., Reference Haapio, Kaunonen, Arffman and Åstedt-Kurki2017; Karabulut, Coşkuner Potur, Doğan Merih, Cebeci Mutlu, & Demirci, Reference Karabulut, Coşkuner Potur, Doğan Merih, Cebeci Mutlu and Demirci2016; Kizilirmak & Başer, Reference Kizilirmak and Başer2016; Serçekuş & Başkale, Reference Serçekuş and Başkale2016; Taheri et al., Reference Taheri, Mazaheri, Khorsandi, Hassanzadeh and Amiri2014) and men (Bergström et al., Reference Bergström, Rudman, Waldenström and Kieler2013). Antenatal education was also associated with a change in birth preferences (Ozdemir et al., Reference Ozdemir, Cilingir, Ilhan, Yildiz and Ohanoglu2018), an increased likelihood of having a spontaneous vaginal birth (Rouhe et al., Reference Rouhe, Salmela-Aro, Toivanen, Tokola, Halmesmäki, Ryding and Saisto2015) and a more positive birth experience (Rouhe et al., Reference Rouhe, Salmela-Aro, Toivanen, Tokola, Halmesmäki and Saisto2013).
Enhanced midwifery care
Three studies examined enhanced midwifery care, including a continuity of care model (n = 2; Hildingsson, Rubertsson, Karlström, & Haines, Reference Hildingsson, Rubertsson, Karlström and Haines2018; Hildingsson, Karlström, Rubertsson, & Haines, Reference Hildingsson, Karlström, Rubertsson and Haines2019; Hildingsson et al., Reference Hildingsson, Rubertsson, Karlström and Haines2019; Lyberg & Severinsson, Reference Lyberg and Severinsson2010a, Reference Lyberg and Severinsson2010b) or a midwife-led visit to a delivery suite (Sydsjö et al., Reference Sydsjö, Bladh, Lilliecreutz, Persson, Vyoni and Josefsson2014). One study had a high risk of bias, and the remaining had a medium risk of bias. The results showed that continuity of care was evaluated positively by women (Lyberg & Severinsson, Reference Lyberg and Severinsson2010a, Reference Lyberg and Severinsson2010b) and was associated with reduced FOC, increased satisfaction with care (Hildingsson et al., Reference Hildingsson, Rubertsson, Karlström and Haines2018; Hildingsson et al., Reference Hildingsson, Karlström, Rubertsson and Haines2019a) and an improved birth experience (Hildingsson, Rubertsson, Karlström, & Haines, Reference Hildingsson, Rubertsson, Karlström and Haines2019b). The impact of visiting the delivery suite with a midwife prior to labour was associated with a shorter duration of labour for multiparous women. However, rate of emergency CS was higher in the intervention group (Sydsjö et al., Reference Sydsjö, Bladh, Lilliecreutz, Persson, Vyoni and Josefsson2014).
Alternative interventions
Twelve studies examined alternative interventions, including relaxation (e.g. guided relaxation, meditation/mindfulness, hypnobirthing; Baleghi, Akerdi, & Pasha, Reference Baleghi, Akerdi and Pasha2016; Bulez et al., Reference Bulez, Ceber Turfan and Sogukpinar2019; Byrne, Hauck, Fisher, Bayes, & Schutze, Reference Byrne, Hauck, Fisher, Bayes and Schutze2014; Fisher et al., Reference Fisher, Hauck, Bayes and Byrne2012; Hunter et al., Reference Hunter, Bormann, Belding, Sobo, Axman, Reseter and Miranda Anderson2011; Pour-Edalati et al., Reference Pour-Edalati, Moghadam, Shahesmaeili and Salehi-Nejad2019), exercise (n = 2; Guder, Yalvac, & Vural, Reference Guder, Yalvac and Vural2018; Guszkowska, Reference Guszkowska2014), art therapy (n = 2; Sezen & Ünsalver, Reference Sezen and Ünsalver2019; Wahlbeck, Kvist, & Landgren, Reference Wahlbeck, Kvist and Landgren2018) or group psychodynamic (Saisto, Toivanen, Salmela-Aro, & Halmesmäki, Reference Saisto, Toivanen, Salmela-Aro and Halmesmäki2006), role play (Navaee & Abedian, Reference Navaee and Abedian2015) and heart rate monitoring (Narita, Shinohara, & Kodama, Reference Narita, Shinohara and Kodama2018). Ten studies had control groups; no studies were RCTs.
Relaxation-style interventions and art therapy were associated with a decrease in FOC (Baleghi et al., Reference Baleghi, Akerdi and Pasha2016; Bulez et al., Reference Bulez, Ceber Turfan and Sogukpinar2019; Byrne et al., Reference Byrne, Hauck, Fisher, Bayes and Schutze2014; Fisher et al., Reference Fisher, Hauck, Bayes and Byrne2012; Pour-Edalati et al., Reference Pour-Edalati, Moghadam, Shahesmaeili and Salehi-Nejad2019; Sezen & Ünsalver, Reference Sezen and Ünsalver2019; Wahlbeck et al., Reference Wahlbeck, Kvist and Landgren2018); however, all but one of these studies had a medium or high risk of bias. Results for the remaining interventions were less clear. Role play reduced FOC; however, a reduction in FOC was also found in the control group (Navaee & Abedian, Reference Navaee and Abedian2015). One exercise intervention including yoga, Pilates and body ball found no impact on FOC (Guszkowska, Reference Guszkowska2014) whereas another Pilates course was found to reduce FOC (Guder et al., Reference Guder, Yalvac and Vural2018). Lastly, heart rate variability biofeedback was associated with lower FOC over time (from 32–34 to 36–67 weeks) (Narita et al., Reference Narita, Shinohara and Kodama2018).
Interventions during labour
Two interventions were carried out during labour, one had low risk of bias and the other had medium risk. One used an RCT to evaluate the effect of EFT [exposure therapy and somatic stimulation using acupressure points (i.e. tapping) compared to breathing awareness or SMC and found a reduction of FOC in the intervention group (Irmak Vural & Aslan, Reference Irmak Vural and Aslan2019)]. Another RCT using marching songs and cheerful music during labour found no impact on FOC, sense of power and self-control (Phumdoung, Youngvanichsate, & Wongmuneeworn, Reference Phumdoung, Youngvanichsate and Wongmuneeworn2011).
Quantitative meta-analysis
Characteristics of studies
Twenty-eight papers from 22 studies were included in the meta-analysis (see online Supplementary Table S4). The majority of papers (79%) were carried out in Scandinavian (Sweden or Finland) or Middle-Eastern (Iran, Turkey or Egypt) countries. A variety of types of intervention were employed, the most frequent being antenatal education (n = 10; 36%). The intensity of interventions varied from six or more sessions (high intensity) to two or fewer sessions. Control groups were mostly routine care, although in six papers the control was some form of counselling. Design of the studies included randomised controlled trials (n = 12), quasi-experimental (n = 14) and a pilot study (n = 2). Papers were recent with a median publication year of 2017.
Publication bias
The trim and fill analysis and funnel plot showed that there were 0 estimated studies missing indicating that there is no significant funnel plot asymmetry (model estimate = −1.3673; s.e. = 0.3006, p < 0.0001; τ 2 = 1.837; s.e. = 0.600). As can be seen from Fig. 2, studies are evenly distributed around the central effect size and there is no evidence of studies that have non-significant or opposite results being omitted.
Fig. 2. Publication bias funnel plot.
Types of outcome
For the majority of papers (n = 23), the outcome was a measure of FOC. The remaining papers (n = 12) recorded the mode of childbirth (whether or not a CS). There was overlap between papers (see online Supplementary Table S4).
Fear of childbirth
Most papers (n = 16) used the Wijma Delivery Expectancy/Experience Questionnaire to measure FOC (Wijma et al., Reference Wijma, Wijma and Zar1998) (W-DEQ).
The overall mean effect size was −1.27 (z = −4.53, p < 0.0001) indicating that women in the intervention group experienced much less FOC compared to those in the control group. There was, however, considerable heterogeneity in the size of the effect reported (Q = 431.43, p < 0.0001; I 2 = 97.7). All papers reported a lower FOC following intervention except Rondung et al. (Reference Rondung, Ternström, Hildingsson, Haines, Sundin, Ekdahl and Rubertsson2018) (Fig. 3 and Table 1). In three studies (Ahmadi et al., Reference Ahmadi, Karami, Faghihzadeh, Jafari, Oskoei and Kharaghani2018; El-Malky et al., Reference El-Malky, El-Homosy, Ashour and Shehada2018; Taheri et al., Reference Taheri, Mazaheri, Khorsandi, Hassanzadeh and Amiri2014), the effect of the intervention was greater than four standard deviations, and an analysis of studentised residual indicated that these were significant outliers. When the analysis was re-run without these outliers, the overall effect was smaller but large and remained significant (d = −0.80, z = −6.50, p < 0.0001). Significant heterogeneity of effect sizes remained (Q = 138.80, p < 0.0001; I 2 = 87.3).
Fig. 3. Fear of childbirth forest plot.
Table 1. Effect sizes for fear of childbirth interventions on FOC reduction
a Results from the same study.
Examination of moderators found country was the only moderator of effectiveness (Table 2). Studies in Middle-Eastern countries reported much larger effect sizes (d = −1.54, z = −2.23, p = 0.026, n = 14) than those from Scandinavian countries (d = −0.20, z = −0.34, p = 0.731, n = 5). Whether the intervention was antenatal education or other talking therapies did not significantly moderate the effect. It was not possible to examine other intervention types because there were too few studies.
Table 2. Moderators of effect of intervention on fear of childbirth (k = 23 unless otherwise stated)
*p < 0.05; **p < 0.01.
Mode of delivery
Twelve studies recorded whether birth was by CS (Fig. 4 and Table 3). Overall, the odds of a CS were lower in the intervention group compared to the control group, but this difference was not statistically significant (OR = 0.80, z = −1.85, p = 0.065). The test for homogeneity was also not significant [Q (df = 11) = 15.23, p = 0.172]. Examination of studentised residuals indicated there were no significant outliers.
Fig. 4. Caearean section by choice forest plot.
Table 3. Effect sizes for fear of childbirth interventions on CS rate reduction
a Results from the same study.
Two significant modifiers were found: country and type of intervention (Table 4). In Scandinavian countries, the odds of a CS were significantly lower in the intervention group (OR 0.66, 95% CI 0.49–0.90), whereas in Middle-Eastern countries, women in the intervention group were more likely to have a CS (OR 1.07, 95% CI 0.76–1.52). Those who undertook talking therapies were less likely to have a CS (OR 0.48, 95% CI 0.48–0.90) compared to those who received antenatal education (OR 1.01, 95% CI 0.78–1.31).
Table 4. Moderators of effect of intervention on fear of childbirth (k = 12 unless otherwise stated)
*p < 0.05; **p < 0.01.
Discussion
Main findings
This review aimed to identify interventions that reduced FOC and CS rates. The review identified 66 papers from 48 studies that investigated six types of intervention. Most studies were carried out in non-English speaking countries and only two recruited fathers-to-be. The quality of the studies varied with 30 out of 48 having a medium risk of bias.
Results from the meta-analysis suggest that most interventions, regardless of the approach, reduce FOC. This is in line with results from the qualitative synthesis which found that within each intervention approach, the majority of studies found a reduction in FOC. The studies that did not find a reduction of FOC were counselling run by midwives (Ryding et al., Reference Ryding, Persson, Onell and Kvist2003), self-reported counselling (Larsson et al., Reference Larsson, Karlström, Rubertsson and Hildingsson2015), antenatal education classes run by midwives with a focus on physical wellbeing (Masoumi et al., Reference Masoumi, Kazemi, Oshvandi, Jalali, Esmaeili-Vardanjani and Rafiei2016), an exercise intervention including yoga, pilates and body ball (Guszkowska, Reference Guszkowska2014) and listening to marching songs during labour (Phumdoung et al., Reference Phumdoung, Youngvanichsate and Wongmuneeworn2011).
The effect of interventions on CS rates was varied and insignificant overall, with studies of CBT (Larsson et al., Reference Larsson, Hildingsson, Ternström, Rubertsson and Karlström2019) finding no reduction in CS rates, and enhanced midwifery care finding increased likelihood of CS (Sydsjö et al., Reference Sydsjö, Bladh, Lilliecreutz, Persson, Vyoni and Josefsson2014). Talking therapies were more consistently associated with reduced CS rates whether delivered by telephone (Fenwick et al., Reference Fenwick, Toohill, Gamble, Creedy, Buist, Turkstra and Ryding2015; Toohill et al., Reference Toohill, Callander, Gamble, Creedy and Fenwick2017) or face-to-face (Ahmadi et al., Reference Ahmadi, Karami, Faghihzadeh, Jafari, Oskoei and Kharaghani2018). Results from the moderator analysis support this, in that women who undertook talking therapies were less likely to have a CS compared to those who received antenatal education. This suggests talking therapies may be potentially effective for reducing CS rates in women with FOC. However, the majority of these studies had a medium risk of bias and the results were not completely consistent with two studies finding that other talking therapies were associated with an increased risk of CS (Henriksen et al., Reference Henriksen, Borgen, Risløkken and Lukasse2018; Sydsjö et al., Reference Sydsjö, Sydsjö, Gunnervik, Bladh and Josefsson2012). More research is therefore needed.
Interpretation
The results from the qualitative and quantitative analysis of this review suggest that most intervention approaches investigated reduce FOC. Only five studies across all intervention groups found null results in terms of FOC reduction, compared to 25 studies that found a reduction in FOC. It may therefore be the delivery of an intervention, rather than the theoretical underpinning that makes it effective at reducing FOC. This is supported by a recent meta-synthesis of women's experiences of interventions for FOC (O'Connell, Khashan, & Leahy-Warren, Reference O'Connell, Khashan and Leahy-Warren2020) that found interventions with a woman-centred ethos, where women felt listened to and where a trusting relationship was able to develop were crucial for women to move from fear to ownership of childbirth. Further, most women felt empowered when they attended FOC interventions, and this was facilitated by supportive alliances, education and birth choices. It could therefore be suggested that providing women with a supportive space to explore their fear is more important than providing a specific approach in terms of reducing FOC. However, more research is needed to understand whether this is the case.
Another potential reason for this finding is that there is a wide range of different fears involved in FOC as well as individualised responses (Wigert et al., Reference Wigert, Nilsson, Dencker, Begley, Jangsten, Sparud-Lundin and Patel2020). Further, studies do not always differentiate between primary tokophobia (women who have not given birth before) and secondary (women who have given birth before) tokophobia. This is an important distinction to make, because secondary tokophobia usually develops after a previous traumatic birth (Bhatia & Jhanjee, Reference Bhatia and Jhanjee2012) and, although not specific to childbirth, there is an evidence base about how trauma and PTSD can be treated [National Institute for Health and Care Excellence (NICE), 2018].
The findings from previous systematic reviews that identified interventions with an educational component as being promising at reducing FOC (Moghaddam Hosseini et al., Reference Moghaddam Hosseini, Nazarzadeh and Jahanfar2018; Stoll et al., Reference Stoll, Swift, Fairbrother, Nethery and Janssen2018; Striebich et al., Reference Striebich, Mattern and Ayerle2018) warrants further exploration. This review found that interventions with an educational component were the most studied (n = 19 studies), but not the most effective. It is therefore possible that the findings from previous systematic reviews can be explained by the fact that interventions with an educational component appeared to be most promising because they had been studied more. However, it is not clear if this is the only explanation, therefore more high-quality RCTs are needed to examine the effectiveness of different types and components of interventions on FOC.
The varied and insignificant effect of FOC interventions on CS rates is unsurprising given the many physiological, psychosocial, contextual, organisational and cultural factors that influence whether a woman wants, requires or has a CS. This is illustrated by the finding that country of study moderated the effect of FOC interventions on CS rates, suggesting cultural and organisational context are important. For example, FOC interventions were found to reduce CS rates amongst women in Scandinavian countries and increase rates in Middle Eastern countries (Iran, Egypt and Turkey). This is perhaps reflective of maternity care in these countries. Iran and Egypt both provide a medicalised model of maternity care, with high mortality and very little antenatal education (Aghlmand et al., Reference Aghlmand, Akbari, Lameei, Mohammad, Small and Arab2008; Choices and Challenges in Changing Childbirth Research Network, 2005; El-Kurdy, Hassan, Hassan, & El-Nemer, Reference El-Kurdy, Hassan, Hassan and El-Nemer2017; TorkZahrani, Reference TorkZahrani2008). Further, the rates of CS are higher in these countries at 48–52% (Azami-Aghdash, Ghojazadeh, Dehdilani, Mohammadi, & Asl Amin Abad, Reference Azami-Aghdash, Ghojazadeh, Dehdilani, Mohammadi and Asl Amin Abad2014; Ministry of Health and Population et al., 2015; Santas & Santhas, Reference Santas and Santhas2018) than in Scandinavian countries (15–20%) (Pyykönen et al., Reference Pyykönen, Gissler, Løkkegaard, Bergholt, Rasmussen, Smárason and Tapper2017). Additionally, it is important to note that in some cases, a planned CS can improve women's birth experience, such as where there is not sufficient time for FOC intervention, or where FOC has not improved following intervention. A planned CS may be the next most appropriate clinical step and can allow for women to feel a sense of control of their birth experience. This suggests individual, cultural and organisational norms may influence whether FOC interventions affect CS rates, and the wider care environment should be taken into consideration when selecting a FOC intervention.
Strengths and limitations
A strength of this review is the broad inclusion criteria, meaning all identified studies on FOC interventions were included. Another strength is the meta-analysis which provides a novel quantitative understanding of the effectiveness of FOC interventions in more robustly designed studies. Limitations of the review include the decision to only double screen 10% of abstracts and full texts. This may have meant some papers were missed; however, the high concordance of the double screening makes this seem unlikely. Further limitations are the low number of studies with different intervention types that could be included in the meta-analysis. There was great heterogeneity of the effect sizes for the FOC outcome across the studies (e.g. Q = 431.43; Q = 138.80). This makes interpretation of the results more difficult. Classification of interventions was challenging because they were complex interventions with multiple components. Some components could overlap between intervention categories. Components of interventions were also minimally described in many papers. Similarly, the methodological quality of studies was varied, with 38 out of the 48 studies had medium to high risk of bias. The way studies were carried out was also variable. For example, different outcome measures were used making it difficult to compare the baseline levels of FOC in all studies. Furthermore, those that used the WDEQ-A and were comparable recruited women with a range of WDEQ-A scores making results difficult to interpret (M = 29.7–130). Additionally, where participant demographics were reported, women were often highly educated, married and white, meaning these results may not be generalisable to those from more marginalised populations. Due to these limitations, the results from these studies should be interpreted cautiously.
Conclusion
Overall, this review suggests that interventions for FOC are effective in reducing FOC but have variable effects on CS rates. FOC interventions do not affect CS rates overall, but this is influenced by cultural and organisational context. High-quality RCTs of different FOC interventions that not only evaluate type of intervention but also determine which components of interventions are most effective for particular presentations are needed in order to design optimal FOC interventions.
Supplementary material
The supplementary material for this article can be found at https://doi.org/10.1017/S0033291721002324
Acknowledgements
We gratefully acknowledge Rachel Mycroft, Anne Ward and Lucy Stephenson who approached us with the idea for this review in order to inform the development of a Pan-London Perinatal Mental Health Networks Fear of Childbirth (Tokophobia) and Traumatic Experience of Childbirth Best Practice Toolkit.
Author contributions
RW carried out the searches and the qualitative analysis, extracted data for the meta-analysis, drafted and finalised the manuscript. RB carried out the meta-analysis and provided feedback on the manuscript. BRG extracted and prepared data for meta-analysis, finalised figures and tables and provided feedback on the manuscript. RM conceptualised the review and provided feedback on the manuscript. SA oversaw the entire process, providing detailed feedback on methodology, qualitative synthesis and manuscript.
Financial support
Partly funded through City, University of London, Higher Education Innovation Fund. PROSPERO registration 2018 CRD42018093095.
Conflict of interest
None.
Ethical standards
Not applicable.
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