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AOGS S Y S T E M A TI C R E VI E W

Worldwide prevalence of tocophobia in pregnant women:


systematic review and meta-analysis
MAEVE A. O’CONNELL1 , PATRICIA LEAHY-WARREN2, ALI S. KHASHAN1,3, LOUISE C. KENNY1 &

SINEAD M. O’NEILL1
1
Irish Centre for Fetal and Neonatal Translational Research (INFANT Center), Department of Obstetrics & Gynecology,
Cork University Maternity Hospital, Wilton, Cork, 2School of Nursing & Midwifery, Brookfield Health Sciences Complex,
University College Cork, Cork, and 3Department of Epidemiology & Public Health, Western Gateway Building, University
College Cork, Cork, Ireland

Key words Abstract


Epidemiology, fear of childbirth, pregnancy,
systematic review, tocophobia, W-DEQ A Introduction. Tocophobia is defined as a severe fear of pregnancy and childbirth.
There is increasing evidence that tocophobia may have short-term and long-term
Correspondence adverse effects on mother and baby. We performed a systematic review and meta-
Maeve A. O’Connell, INFANT Research analysis to determine the global prevalence of tocophobia in pregnancy. Material
Center, Cork University Maternity Hospital,
and methods. Relevant articles were identified through searching six relevant
Wilton, Cork T12 YE02, Ireland.
databases: MEDLINE, CINAHL, Pubmed, PsycINFO, Maternity & Infant Care
E-mail: maeveanneoconnell@gmail.com
and Scopus between 1946 and April 2016. We used search terms for tocophobia
Conflicts of interest prevalence in pregnant women that we agreed on with a medical librarian. There
The authors have stated explicitly that there were no language restrictions. Two review authors independently assessed data
are no conflicts of interest in connection with for inclusion, extracted data and assessed quality using a standardized appraisal
this article. tool. Meta-analysis was performed to determine the overall pooled-prevalence of
tocophobia. Several subgroup and sensitivity analyses were conducted.
Please cite this article as: O’Connell MA,
Results. Thirty-three studies were included in the systematic review from 18
Leahy-Warren P, Khashan AS, Kenny LC,
O’Neill SM. Worldwide prevalence of countries of which data from 29 studies were used in the meta-analysis of
tocophobia in pregnant women: systematic 853 988 pregnant women. Definition of tocophobia varied, whereas prevalence
review and meta-analysis. Acta Obstet rates ranged between 3.7 and 43%. The overall pooled prevalence of tocophobia,
Gynecol Scand 2017; 96:907–920. using a random-effects model, was 14% (95% CI 0.12–0.16). Significant
heterogeneity was observed (I2 = 99.25%, p = 0.00), which was not explained in
Received: 22 September 2016 subgroup analyses including tocophobia definition used, screening trimester and
Accepted: 19 March 2017
parity. Conclusion. The prevalence of tocophobia is estimated at 14% and
DOI: 10.1111/aogs.13138
appears to have increased in recent years (2000 onwards). Considerable
heterogeneity (99.25%) was noted that may be attributed to lack of consensus on
the definition of tocophobia, so our results should be interpreted with caution.

Abbreviations:CAQ, Childbirth Attitudes Questionnaire; FOBS, Fear of Birth


Scale; FOC, fear of childbirth; W-DEQ A, Wijma Delivery Experience
Questionnaire Part A.

Key Message
Introduction
Definitions of tocophobia vary widely. This meta-ana-
Over the last 30 years, there has been increasing interest lysis estimated a global pooled-prevalence of 14%;
in tocophobia (severe fear of childbirth) both in empirical however, this should be interpreted with caution
research and clinical practice (1–5). Tocophobia has been because of significant heterogeneity. This is the first
defined as “an unreasoning dread of childbirth,” and fur- systematic review of the prevalence of tocophobia,
ther classified into primary (affecting nulliparous women) which affects a significant minority of women.
and secondary (affecting parous women usually after a

ª 2017 Nordic Federation of Societies of Obstetrics and Gynecology, Acta Obstetricia et Gynecologica Scandinavica 96 (2017) 907–920 907
Worldwide prevalence of tocophobia M.A. O’Connell et al.

previous birth experience) tocophobia (6,7). There is, “tocophobia,” “fear of childbirth,” “fear of labor,” “fear of
however, no one agreed definition of tocophobia and birth,” “childbirth related fear,” “childbirth related anxiety,”
much of the published literature to date refers to toco- “fear in pregnancy,” “antenatal” and “childbirth.”
phobia as a severe “Fear of Childbirth (FOC)” rather than
“an unreasoning dread of childbirth” (8,9). Factors
Study selection
including anxious personality types, previous sexual
abuse, past traumatic birth or any traumatic experience Published observational studies including pregnant
in health care, previous miscarriages, long duration of women of any age and origin and reporting the preva-
infertility, smoking, low social supports and poor partner lence of tocophobia (or sufficient data in order for us
relationships have been associated with primary and sec- to compute this estimate) were eligible for inclusion.
ondary tocophobia (9–13). Two researchers (MOC and SMON) independently
While there are no standard criteria for defining toco- reviewed study titles and abstracts applying the inclu-
phobia, the Wijma Delivery Expectancy Questionnaire sion/exclusion criteria. Full-text studies were obtained
Part A (W-DEQ A) is the most commonly used tool for where required and where consensus was not reached,
assessment and diagnosis (14,15). Other tools include the a third reviewer (PLW) ensured agreement. Reference
Fear of Birth Scale (FOBS) and the Childbirth Attitudes lists of eligible studies were hand searched for further
Questionnaire (14,16,17). The FOBS is a Visual Analogue potentially eligible studies. The following data were
Scale consisting of two questions, developed to encourage abstracted from each study using a standardized form
compliance in completion of the questionnaire due to the by two reviewers (MOC, SMON): author, year, study
length of the W-DEQ A (consisting of 33 questions). The location (country), study design, scale used, sample size
prevalence of tocophobia has also been reported by analy- and prevalence. If it was considered that a study had
sis of the International Classification of Diseases 10th collected data on the prevalence of tocophobia but had
Revision codes, assigned to women who attended toco- not reported it, the authors were contacted for this
phobia clinics in countries where care pathways are well information.
established (9,18).
It is reported that 6–10% of pregnant women suffer
Quality assessment
with FOC that affects everyday life (1,18–20). However,
lack of consistency in defining tocophobia has led to vari- Quality assessment of each study was independently
ation in prevalence reports (21,22). Therefore, the aims of evaluated by two reviewers (MOC and SMON) using a
this systematic review were to: assess how “tocophobia” is standardized tool including eight questions to assess bias
defined in the literature, and provide the first quantitative (25) (see Supplementary material, Appendix S2) pertain-
pooled estimate of the prevalence of tocophobia in preg- ing to the following criteria: target population, sampling
nant women by synthesizing the data from eligible studies ascertainment methods, response rate, information on
(where feasible) in a meta-analysis. non-responders, if the sample was representative, data
collection methods, use of a validated tool for tocopho-
bia, and prevalence with 95% CIs. Each study received a
Material and methods
score of between 0 and 8 points, based on meeting the
The review adheres to the Preferred Reporting Items for prescribed criteria as agreed by the reviewers. High-qual-
Systematic Reviews and Meta-Analysis (PRISMA) guideli- ity studies were defined as those receiving a score of ≥5
nes (23) and has been registered on the International out of 8.
prospective register of systematic reviews (PROSPERO
ID: CRD42015017443) (24).
Statistical analyses
Search results were compiled in ENDNOTE REFERENCE MAN-
Sources
AGER version X7 (Clarivate Analytics, New York, NY,
Six electronic databases (PubMed, CINAHL, PsycINFO, USA). Characteristics of the included studies were sum-
Maternity & Infant Care, Scopus, and MEDLINE) were marized and presented in Table 1. For the meta-analysis,
searched for all published literature up until 11 April an overall pooled prevalence was calculated using the
2016 using a detailed search strategy and without date or sample size and the proportion of women with tocopho-
language restrictions (see Supplementary material, bia and the fixed or random-effects model as appropriate.
Appendix S1). Medical subject headings or keyword terms Using the metaprop command, we generated pooled pro-
for tocophobia during pregnancy were combined accord- portions and an overall pooled estimate with inverse vari-
ing to the principles of Boolean logic including: ance weights derived from a random-effects model (26)

908 ª 2017 Nordic Federation of Societies of Obstetrics and Gynecology, Acta Obstetricia et Gynecologica Scandinavica 96 (2017) 907–920
Table 1. Characteristics and quality assessment of studies included in the systematic review.

Trimester Tocophobia Quality


Study (year) Country Study design (sample size) Scale screened Parity prevalence (out of 8)

Adams et ala (2012) Norway Prospective cohort (2206) W-DEQ A ≥85 3rd All 7.5% 5
Aksoy et al. (2015) Turkey Cross-sectional (817) W-DEQ A ≥85 3rd Multi-parous 15.7% 5
M.A. O’Connell et al.

Areskog et al. (1981) Sweden Qualitative (139) Interview 2nd All 6.5% 5
Christiaens et al. (2011) Belgium/The Prospective cross-cultural comparison CAQ 3rd All Not reported 4
Netherlands study (799)
Fenwick et al. (2009) Australia Prospective cohort (401) W-DEQ A ≥71 3rd All 26.2% 7
Gao et al. (2015) China Cross-sectional (353) Chinese CAQ 3rd All No tocophobia reported, 5
moderate fear only
Geissbuehler et al. (2002) Switzerland Prospective cohort (8528) 50 Qs 2nd All 5.3% 6
Haines et al. (2011) Australia/Sweden Prospective cross-cultural (123/386-509 FOBS VAS tool (cut-off 5) 2nd All 31.1% Australia 5
combined) 29.5% Sweden
Hall et al. (2009) Canada Cross-sectional (650) W-DEQ A ≥66 3rd All 24.9% 7
Heimstad et al. (2006) Norway Prospective cohort (1452) W-DEQ A ≥95 2nd All 7.3% 4
Jaju et al. (2015) South India Cross-sectional (368) Interview based on ICD- 3rd Nulli-parous 17.7% 4
10 Code
Kjaergaard et al. (2008) Sweden/Denmark Prospective cohort (110/55–165 W-DEQ A ≥85 3rd Nulli-parous 10.9% 5
combined)
Two separate studies carried out over
2 time periods 1996/2004–2005
Laursen et al. (2008) Denmark Population cohort (Danish Birth Register) Phone interviews 2nd & 3rd Nulli-parous 11.74% 5
(30 380)
Lowe (2000) USA Cross sectional (280) CAQ 3rd Nulli-parous 19.29% 4
Lukasse et al. (2014) BIDENS (Belgium Cross sectional (6870) W-DEQ A ≥85 All All 11.2% 6
Iceland, Denmark,
Estonia, Norway
Sweden)
Matinnia et al. (2015) Iran Cross sectional (342) Fear related to Pregnancy 2nd Nulli-parous 42.9% 3
& Childbirth
Questionnaire
Niemenen et al. (2009) Sweden Cross sectional (1635) W-DEQ A ≥85 All All 15.5% 6
Nordeng et al.a (2012) Norway Birth cohort study (1984) W-DEQ A ≥85 3rd All 7.8% 8
Pazzagli et al. (2015) Italy Prospective correlational design (158) W-DEQ A in Italian 3rd All 9.5% 7

ª 2017 Nordic Federation of Societies of Obstetrics and Gynecology, Acta Obstetricia et Gynecologica Scandinavica 96 (2017) 907–920
Raisanen et al. (2014) Finland Retrospective cohort (788 317) ICD Code O99.80 All All 3.7% 8
Rouhe et al. (2009) Finland Prospective cohort(1276) W-DEQ A ≥100 All All 7.5% 7
Ryding et al. (1998) Sweden Prospective cohort (1981) W-DEQ A ≥84 3rd All 10.0% 7
Salomonssen et al. (2013) Sweden Cross-sectional (1000) W-DEQ A ≥85 2nd Nulli-parous 20.8% 6
Sluijs et al. (2012) The Netherlands Prospective cohort (105) W-DEQ A ≥85 3rd All 12.4% 6
Spice et al. (2009) Canada Cross-sectional (110) W-DEQ A ≥85 3rd All 9.1% 4
Storksen et al.a (2012) Norway Cross-sectional (1642) W-DEQ A ≥85 3rd All 8% 6
Worldwide prevalence of tocophobia

909
Worldwide prevalence of tocophobia M.A. O’Connell et al.

(out of 8)
in STATA software Version 13.1 (StataCorp, College Sta-

screened for tocophobia; 1st trimester, weeks 1–12; 2nd trimester, weeks 13–20; 3rd trimester, 21 onwards; All, refers to both nulliparous and multiparous women; ICD-10, International Classifi-
Scale; ICD-10, International Classification of Diseases; J-WDEQ A, Japanese Wijma Delivery Experience Questionnaire Part A; Trimester screened, refers to the gestation at which women were
CAQ, Childbirth Attitudes Questionnaire; W-DEQ A, Wijma Delivery Experience Questionnaire Part A; Qs, Questions; CI, Confidence Intervals; FOBS, Fear of Birth Scale; VAS, Visual Analogue
Quality tion, TX, USA).

7
7
6

5
5
8
J-WDEQ A cut-off not
Subgroup and sensitivity analyses
prevalence estimate A priori sensitivity analyses included studies with a W-
DEQ A ≥85 for tocophobia, by parity (nulliparous
confirmed – no

women only, multiparous women only), by screening tri-


Tocophobia
prevalence

mester (first trimester, second trimester, third trimester).


22.10%

10.9%

18.5%
25.9%
11.1%
4.9%

A priori subgroup analyses included: by study quality


(high vs. low), by region (Scandinavia vs. Rest of Europe
Nulli-parous vs. Australia vs. America vs. Asia), and by time period
(1980s vs. 1990s vs. 2000–2009 vs. 2010–2016).
Parity

All

All
All
All

All
All

Heterogeneity assessment
Trimester
screened

Heterogeneity between studies was assessed by examining


the study characteristics presented in Table 1. In addition,
2nd
2nd
2nd
3rd

3rd
3rd
3rd

the I2 statistic was used to determine statistical hetero-


geneity according to the Cochrane Handbook for System-
Aurora Clinic? Or feeling
Two questions –attending

atic Reviews threshold recommendations (27). For this


very negative about

meta-analysis, where heterogeneity was >50% the ran-


W-DEQ A ≥85.8

dom-effects model was used (28).


W-DEQ A ≥85

W-DEQ A ≥66
W-DEQ A ≥85
J-WDEQ A

FOBS >60

birth?

Results
Scale

Results of the systematic search are presented in Figure 1,


which yields 33 studies eligible for inclusion in the sys-
tematic review (29). Twenty-four high-quality studies and
five low-quality studies were included in the meta-analy-
Prospective cohort study (2662)

sis. Where there was more than one publication on a


Study design (sample size)

cohort of patients (i.e. the same population), data on the


Prospective cohort (496)
Cross-sectional (1410)

prevalence of tocophobia were taken from those that


Cross-sectional (240)

Cross-sectional (606)

Cross-sectional (196)
Cross-sectional (506)

described the total population rather than a subset.

Study characteristics
Study characteristics are presented in Table 1. One study
was published in 1981 (30), one study in the late 1990s
(31), 14 studies between 2000 and 2009 (15,18,19,21,32–
41) and 17 studies between 2010 and April 2016
(1,8,9,16,17,20,42–52). Study settings included the follow-
ing: USA (32), Canada (37), Australia (36,46,49), Sweden
Australia
Country

Sweden

Sweden

Sweden
Sweden
Sweden

(15,16,19,29–31,33,38,40,41,47,49), Norway (20,35,43,45),


Japan

Finland (9,21), Switzerland (34), Denmark (18,19), Italy


Data from same population.

(1), Turkey (8), Iran (51), China (48), Japan (52), South
Waldenstrom et al. (2006)

India (50), and the Netherlands (44). One study was con-
Ternstrom et al. (2015)
Takegata et al. (2014)

Wiklund et al. (2007)

ducted across six countries: Belgium, Iceland, Denmark,


Table 1. Continued

Toohill et al. (2014)

cation of Diseases.

Estonia, Norway, and Sweden (42). Study population


Zar et al. (2001)
Zar et al. (2002)

sizes ranged from 105 to 788 317 (9,44). One study


Study (year)

included multiparous women (8), seven studies included


nulliparous women and 25 studies were not restricted by
parity.
a

910 ª 2017 Nordic Federation of Societies of Obstetrics and Gynecology, Acta Obstetricia et Gynecologica Scandinavica 96 (2017) 907–920
M.A. O’Connell et al. Worldwide prevalence of tocophobia

Idenficaon
Records idenfied through Addional records idenfied
database searching through other sources
(n = 533) (n = 5)

Records aer duplicates removed


(n = 468)
Screening

Records excluded
Records screened (n = 439)
(n = 468)

Full-text arcles
Full-text arcles assessed excluded, ineligible
for eligibility (n = 1)
Eligibility

(n = 34)

Studies included in
qualitave synthesis
(n = 33)
Included

Studies included in
quantave synthesis
(meta-analysis)
(n = 29)

Figure 1. Flow chart of systematic search. [Color figure can be viewed at wileyonlinelibrary.com].

interviews using standardized questions or self-reported


Definition of tocophobia
questionnaires completed in the clinic or returned via
Tocophobia was defined using a variety of measures and post (18,30,40,49,51). Sampling was done in different lan-
cut-offs. Most [21/33 studies (1,2,8,15,19–21,31,33,35– guages, and in the case of standardized instruments (W-
39,42,44,45,50,52,53)] used the W-DEQ Part A to assess DEQ A, FOBS, CAQ) the studies ensured correct transla-
tocophobia [of which three studies (20,43,45) used the tion of the questionnaires in the following ways, which
same cohort], meaning that 19 different cohorts in this varied according to study: the questionnaire was trans-
review used the W-DEQ Part A as a tool to assess toco- lated into the most commonly spoken languages of the
phobia. Whereas the majority of included studies used study area (forward translation); the various language
W-DEQ Part A, only a minority of the total study popu- versions of the questionnaire were translated by both lay
lation (21 619/853 988) were assessed with this tool. and professional translators (expert back translation);
Other methods used to define tocophobia included the draft versions of the translated questionnaire were
FOBS [three studies (16,21,49)], Childbirth Attitudes assessed for accuracy and validated by professionals who
Questionnaire (CAQ) [three studies (17,32,48)] and were fluent in one or more of the languages (pre-testing)
International Statistical Classification of Diseases and (16,17,44,49). One study (52) was the first to use the W-
Health Related problems 10th Revision [1 study (9)] DEQ A in the Japanese language and so needs to be vali-
(Table 1). A Finnish study comprised the largest study dated in further studies.
population (n = 788 317), which reported the prevalence Of the 21 studies that used the W-DEQ Part A, two
of tocophobia based on an International Statistical Classi- used ≥100 as a cut-off for tocophobia (21,35), one used
fication of Diseases and Health Related problems 10th ≥95 (35), one used ≥85.8 (41), 12 used ≥85
Revision Code allocated to all women who attended toco- (8,19,20,33,37,41,42,46,47,53), one used ≥84 (31), one
phobia clinics during the period of the study (9). In addi- used ≥71 (36) and two used ≥66 (15,37). Studies that
tion, tocophobia was measured using phone interviews used the FOBS estimated a much higher prevalence esti-
with pre-defined standardized questions, face-to-face mate (double the other prevalence estimates) than the

ª 2017 Nordic Federation of Societies of Obstetrics and Gynecology, Acta Obstetricia et Gynecologica Scandinavica 96 (2017) 907–920 911
Worldwide prevalence of tocophobia M.A. O’Connell et al.

other studies included in the review. Regarding screening sample selection was unclear or not reported or did not use
trimester, four studies questioned women in all trimesters validated tools for tocophobia.
(9,30,42,47), 12 studies recruited women in the second
trimester (12–27 weeks) (2,16,18,30,34,35,37,40,42,
Prevalence of tocophobia – meta-analysis
49,51,53) and 17 studies recruited women in the third tri-
mester (28–41 weeks) (1,8,15,18–20,30–33,36,37,39, Of the 33 studies included in the systematic review, data from
41,42,44,50). Of these studies, one recruited in both the 28 studies were included in the meta-analysis. One study (49)
second and third trimesters (18). Data on the prevalence included two cohorts from Australia and Sweden which we
of tocophobia were available for two population-based split into two studies for the purpose of the meta-analysis
(9,18) and 31 hospital-based cohorts of pregnant women. (Haines et al.), resulting in 29 studies in total. A fixed-effects
model yielded a 4% (95% CI 0.04–0.04) prevalence of toco-
phobia in pregnant women. Due to significant heterogeneity
Quality assessment
(I2 = 99.5%, P < 0.0001), a random-effects model was used
Study quality was assessed independently by two reviewers and a pooled prevalence of 14% (95% CI 0.12–0.16) for toco-
(MOC, SMON). While there was variation in the quality of phobia, with considerable heterogeneity (I2 = 99.25%) (Fig-
the studies, overall quality was considered high (26/33 ure 2) was obtained.
studies with a score of 5 or more out of 8) (Table 1). Seven
studies were considered low quality (a score of at least 4
Sensitivity analyses
out of 8) due to the following: the target population was
not clearly defined, the response rate was not reported, W-DEQ A ≥85. The twelve studies that used a W-
information on non-responders was not provided or the DEQ A score of ≥85 as the definition of tocophobia

Proportion meta-analysis plot [random effects]


Study ES (95% CI)

Adams et al* (2012) 0.07 (0.06, 0.09)


Aksoy et al (2015) 0.16 (0.13, 0.18)
Areskog et al (1981) 0.06 (0.03, 0.12)
Fenwick et al (2009) 0.26 (0.22, 0.31)
Geissbuehler et al (2002) 0.05 (0.05, 0.06)
Haines et al A (2011) 0.30 (0.22, 0.38)
Haines et al B (2011) 0.31 (0.26, 0.36)
Hall et al (2009) 0.06 (0.04, 0.08)
Heimstad et al (2006) 0.07 (0.06, 0.09)
Jaju et al (2015) 0.18 (0.14, 0.22)
Kjaergaard et al (2008) 0.11 (0.07, 0.17)
Laursen et al (2008) 0.11 (0.11, 0.12)
Lowe (2000) 0.19 (0.15, 0.24)
Lukasse et al (2014) 0.11 (0.10, 0.12)
Matinnia et al (2015) 0.43 (0.38, 0.48)
Niemenen et al (2009) 0.16 (0.14, 0.17)
Pazzagli et al (2015) 0.09 (0.05, 0.15)
Raisanen et al (2014) 0.04 (0.04, 0.04)
Rouhe et al (2009) 0.08 (0.06, 0.09)
Ryding et al (1998) 0.10 (0.09, 0.11)
Salomonssen et al (2013) 0.21 (0.17, 0.25)
Sluijs et al (2012) 0.12 (0.07, 0.20)
Spice et al (2009) 0.09 (0.04, 0.16)
Ternstrom et al (2015) 0.22 (0.19, 0.26)
Toohill et al (2014) 0.05 (0.04, 0.06)
Waldenstrom et al (2006) 0.11 (0.10, 0.12)
Wiklund et al (2007) 0.19 (0.15, 0.22)
Zar et al A (2001) 0.26 (0.19, 0.33)
Zar et al B (2002) 0.11 (0.08, 0.14)
Overall (l 2 = 99.25%, p = 0.00) 0.14 (0.12, 0.16)

.25 .5 .75 1
Proportion (95% confidence interval)

Figure 2. Forest plot of the pooled prevalence of tocophobia for all studies included in the meta-analysis. [Color figure can be viewed at
wileyonlinelibrary.com].

912 ª 2017 Nordic Federation of Societies of Obstetrics and Gynecology, Acta Obstetricia et Gynecologica Scandinavica 96 (2017) 907–920
M.A. O’Connell et al. Worldwide prevalence of tocophobia

detected a pooled prevalence of 12% (95% CI 0.09–0.14) By region. The prevalence of tocophobia found in Scan-
and significant heterogeneity (I2 = 95.41%, p = 0.00) dinavia was 12% (95% CI 0.09–0.15) (I2 = 99.51%,
using the random-effects model (Figure 3). p = 0.00) (see Supplementary material, Fig. S3). In the
Rest of Europe the prevalence was 8% (95% CI 0.04–
Parity. Studies including nulliparous women (Figure 4), 0.13) (I2 = 99.51%, p = 0.00), in Australian studies the
yielded a pooled prevalence of 16% (95% CI; 0.14–0.19) prevalence was 23% (95% CI 0.07–0.39) (I2 = 98.63%,
with significant heterogeneity (I2 = 99.42%, p = 0.00). p = 0.00), in American studies the prevalence was 11%
Studies including multiparous women (Figure 5), resulted (95% CI 0.03–0.20) (I2 = 92.97%, p = 0.00) and in Asian
in a pooled prevalence of 12% (95% CI 0.10–0.14) and studies the prevalence was 25% (95% CI 0.11–0.40)
significant heterogeneity (I2 = 97.81%, p = 0.00). (I2 = 97.69%, p = 0.00).

Screening trimester. In one study, women were By time period. One study looked at the prevalence of
screened in the first trimester of pregnancy and this was tocophobia in the 1980s, which was 6% (95% CI 0.03–
not included in a sensitivity analysis (42). Studies that 0.12) (see Supplementary material, Fig. S4). Prevalence of
screened women in the second trimester (Figure 6), tocophobia was reported by one study in the 1990s at
yielded a pooled prevalence of 14% (95% CI 0.12–0.16) 10% (95% CI 0.09–0.11). Fourteen studies between 2000
and significant heterogeneity remained (I2 = 98.1%, and 2009 examined the prevalence of tocophobia, which
p = 0.00). Studies that screened in the third trimester was 12% (95% CI 0.10–0.15) (I2 = 98.18%, p = 0.00),
(see Supplementary material, Fig. S1) yielded a pooled and 13 studies conducted between 2010 and 2016 resulted
prevalence of 12% (95% CI 0.10–0.14), with significant in a pooled prevalence of 17% (95% CI 0.13–0.21)
heterogeneity (I2 = 97.78%, p = 0.00). (I2 = 98.98%, p = 0.00). Overall heterogeneity was highly
significant (I2 = 99.26%, p = 0.00).
Subgroup analyses
Studies not eligible for inclusion in the meta-
Study quality. The prevalence of tocophobia in the high- analysis. Three studies (17,48,52) did not include data
quality studies was 13% (95% CI 0.11–0.15) (I2 = 99.3%, that could be included in the meta-analysis and two stud-
p = 0.00) compared with 19% (95% CI 0.08–0.30) ies (43,45) included the same population as a third study
(I2 = 97.96%, p = 0.00) in the low-quality studies (see (20). A brief summary of the studies not included in the
Supplementary material, Fig. S2). meta-analysis is presented in Table 2.

Proportion meta-analysis plot [random effects]


Study ES (95% CI)

Adams et al (2012) 0.07 (0.06, 0.09)


Aksoy et al ( 2015) 0.16 (0.13, 0.18)
Hall et al (2009) 0.06 (0.04, 0.08)
Kjaergaard (2008) 0.11 (0.07, 0.17)
Lukasse et al (2014) 0.11 (0.10, 0.12)

Niemenen et al (2009) 0.16 (0.14, 0.17)


Pazzagli et al (2015) 0.09 (0.05, 0.15)
Salomonssen et al (2013) 0.21 (0.17, 0.25)

Spice et al (2009) 0.09 (0.04, 0.16)


Toohill et al (2014) 0.05 (0.04, 0.06)

Wiklund et al (2007) 0.19 (0.15, 0.22)


Zar et al (2002) 0.11 (0.08, 0.14)
2
Overall (l = 95.41%, p = 0.00) 0.12 (0.09, 0.14)

.25 .5 .75 1
Proportion (95% confidence interval)

Figure 3. Sensitivity analysis: Forest plot of the pooled prevalence of tocophobia including studies which used W-DEQ A ≥85 as the definition
for tocophobia. [Color figure can be viewed at wileyonlinelibrary.com].

ª 2017 Nordic Federation of Societies of Obstetrics and Gynecology, Acta Obstetricia et Gynecologica Scandinavica 96 (2017) 907–920 913
Worldwide prevalence of tocophobia M.A. O’Connell et al.

Proportion meta-analysis plot [random effects]


Study ES (95% CI)

Adams et al (2012) 0.09 (0.07, 0.11)


Areskog et al (1981) 0.05 (0.01, 0.14)
Fenwick et al (2009) 0.33 (0.26, 0.40)
Geissbuehler et al (2002) 0.05 (0.05, 0.06)
Haines et al A (2012) 0.33 (0.26, 0.41)
Haines et al B (2012) 0.37 (0.23, 0.52)
Hall et al (2009) 0.55 (0.38, 0.71)
Heimstad et al (2006) 0.05 (0.03, 0.07)
Jaju et al (2015) 0.18 (0.14, 0.22)
Kaergaard (2008) 0.11 (0.07, 0.17)
Laursen et al (2008) 0.12 (0.11, 0.12)
Lukasse et al (2014) 0.11 (0.10, 0.12)
Lowe (2000) 0.19 (0.15, 0.24)
Matinnia et al (2015) 0.43 (0.38, 0.48)
Niemenen et al (2009) 0.15 (0.12, 0.17)
Pazzagli et al (2015) 0.12 (0.06, 0.21)
Raisanen et al (2014) 0.02 (0.02, 0.03)
Rouhe et al (2009) 0.07 (0.05, 0.10)
Salomonssen et al (2013) 0.21 (0.17, 0.25)
Ternstrom et al (2015) 0.21 (0.17, 0.27)
Toohill et al (2014) 0.06 (0.05, 0.09)
Wiklund et al (2007) 0.19 (0.15, 0.22)
Zar et al A (2001) 0.26 (0.17, 0.37)
Overall (l 2 = 99.42%, p = 0.00) 0.16 (0.14, 0.19)

.25 .5 .75 1
Proportion (95% confidence interval)

Figure 4. Sensitivity analysis: Forest plot of the prevalence of tocophobia for studies that included nulliparous women only. [Color figure can be
viewed at wileyonlinelibrary.com].

women (42,54). We carried out a subgroup analysis that


Discussion showed tocophobia to be more prevalent in nulliparous
To our knowledge, this is the first systematic review and women (who have never experienced childbirth before),
meta-analysis of the prevalence of tocophobia in pregnant this is similar to the findings of nine previous studies
women. Overall, the pooled prevalence of tocophobia was (1,2,15,16,20,36,37,42,49).
14%. Subgroup analyses according to region showed a The results of our study are clinically relevant in the
significant difference in the prevalence of tocophobia. For following respects. First, we identified a lack of a clear
example, in Scandinavia the prevalence was 12% com- operational definition for tocophobia. Although tocopho-
pared with 8% in the rest of Europe and 23% in Aus- bia has become a term commonly used to describe severe
tralia. Furthermore when we looked at the prevalence of FOC, a clear, consistent operational definition is lacking
tocophobia by time period, it was lower in the earlier (9,55). This was reflected in the literature where several
years (1980s, 1990s) but increased in more recent years tools were used to assess FOC and tocophobia (Table 1).
(2000 onwards). However, our findings need to be inter- The W-DEQ A questionnaire was employed in 19 studies,
preted with caution since significant heterogeneity was and although there is a recommended cut-off point for
found (I2 = 99.25%, p = 0.00). Extensive pre-specified the definition of tocophobia (≥85), some studies used dif-
subgroup and sensitivity analyses did not explain the sig- ferent cut-off points (1,15,31,56). Terms used included
nificant heterogeneity in the meta-analysis. Differences in “high childbirth-related fear,” “intense fear,” “high child-
the way studies were conducted and information was col- birth fear,” “severe childbirth fear” or “severe FOC”
lected and recorded as well as variations in the social and (8,16,30,34,44,48,51). It is important to recognize that it
cultural characteristics of women included in these studies may be normal for pregnant women to have worries
may explain the heterogeneity (28). (17,34,36,57) (recurrent but unspecific thoughts) as birth
There has been conflicting evidence as to the preva- is unpredictable; however, fears can be strong, specific
lence of tocophobia in nulliparous and multiparous and continuous (57). It has been suggested that when a

914 ª 2017 Nordic Federation of Societies of Obstetrics and Gynecology, Acta Obstetricia et Gynecologica Scandinavica 96 (2017) 907–920
M.A. O’Connell et al. Worldwide prevalence of tocophobia

Proportion meta-analysis plot [random effects]


Study ES (95% CI)

Adams et al* (2012) 0.06 (0.05, 0.08)


Aksoy et al (2014) 0.16 (0.13, 0.18)
Areskog et al (1981) 0.08 (0.03, 0.16)
Fenwick et al (2009) 0.20 (0.15, 0.26)
Geissbuehler et al (2002) 0.05 (0.05, 0.06)
Haines et al A (2011) 0.31 (0.25, 0.37)
Haines et al B (2011) 0.26 (0.17, 0.38)
Hall et al (2009) 0.06 (0.04, 0.10)
Heimstad et al (2006) 0.07 (0.05, 0.09)
Lukasse et al (2014) 0.11 (0.10, 0.12)
Niemenen et al (2009) 0.35 (0.32, 0.39)
Pazzagli et al (2015) 0.07 (0.02, 0.15)
Raisanen et al (2014) 0.05 (0.04, 0.05)
Rouhe et al (2009) 0.08 (0.06, 0.10)
Ternstrom et al (2015) 0.13 (0.09, 0.17)
Toohill et al (2014) 0.04 (0.02, 0.05)
Zar et al (2001) 0.26 (0.17, 0.37)
Overall (l 2 = 97.81%, 0.12 (0.10, 0.14)
p = 0.00)

.25 .5 .75 1
Proportion (95% confidence interval)

Figure 5. Sensitivity analysis: Forest plot of the pooled prevalence of tocophobia for studies which included multiparous women only. [Color
figure can be viewed at wileyonlinelibrary.com].

Proportion meta-analysis plot [random effects]


Study ES (95% CI)

Geissbuehler et al (2002) 0.05 (0.05, 0.06)


Haines et al A (2011) 0.31 (0.26, 0.36)
Haines et al B (2011) 0.30 (0.22, 0.38)
Hall et al (2009) 0.06 (0.04, 0.08)
Heimstad et al (2006) 0.05 (0.04, 0.06)
Laursen et al A (2008) 0.08 (0.07, 0.08)
Lukasse et al (2009) 0.08 (0.07, 0.08)
Matinnia et al (2015) 0.43 (0.38, 0.48)
Salomonssen et al (2013) 0.21 (0.17, 0.25)
Ternstrom et al (2015) 0.22 (0.19, 0.26)
Toohill et al (2014) 0.05 (0.04, 0.06)
Waldenstrom et al (2006) 0.11 (0.10, 0.12)
Overall (l 2 = 98.10%, 0.14 (0.12, 0.16)
p = 0.00)

.25 .5 .75 1
Proportion (95% confidence interval)

Figure 6. Sensitivity analysis: Forest plot of the pooled prevalence of tocophobia using studies that screened women in the second trimester (13–
27 weeks of gestation) only. [Color figure can be viewed at wileyonlinelibrary.com].

woman expresses FOC during pregnancy and requests burden for public health. Moreover, our study revealed
support, this could be in itself a definition (55). Second, an apparent increase in the prevalence of tocophobia over
this is the first time a pooled-prevalence has been calcu- the last 30 years. Therefore, our results highlight the need
lated for tocophobia giving an indication of the overall for clinicians and the healthcare service to be aware of

ª 2017 Nordic Federation of Societies of Obstetrics and Gynecology, Acta Obstetricia et Gynecologica Scandinavica 96 (2017) 907–920 915
Worldwide prevalence of tocophobia M.A. O’Connell et al.

Table 2. Studies not included in the meta-analysis. The FOBS (a two-question visual analogue scale) is
Gao et al., 2015 (48) Moderate levels of fear reported. No deemed a feasible tool used to prompt referral in clinical
tocophobia reported. practice (22,57) and has been validated in samples of
Takegata et al., 2014 JW-DEQ A Mean Score= 50.0 (n=240). Swedish and Australian populations (sensitivity 89% and
(52) Sense of coherence is a direct cause of specificity 79%) (22,49). It is argued that there is likely to
fear of childbirth be high compliance because it is easily understood (22).
High sense of coherence works as a
Screening for FOC is suggested in order to offer appro-
resiliency factor to cope with birth and
reduce fear of birth
priate referral as there is evidence that women may bene-
Nordeng et al., 2012 7.8% of the study population had fit if offered timely antenatal support (2,3,22,62,63).
(43)a tocophobia (W-DEQ A≥85) (n = 1984) However, similarly to the introduction of other screening
Tocophobia was significantly associated assessments to the antenatal booking appointment, it may
with use of psychotropic drugs, but not be envisaged that time constraints in the clinic and lack
the use of analgesics or medications in of clear referral pathways may be barriers to the effective-
general
ness of this tool (64–66).
Storksen et al., 2012 8% of the study population had tocophobia
(45)a (W-DEQ A≥85) (n = 1642)
There is considerable evidence endorsing the need for
While presence of anxiety or depression improved perinatal psychological support in maternity
increased prevalence of tocophobia, the services (3–5,54,67,68). Reasons for tocophobia may be
majority of women with tocophobia had complex (3,33,69) and include lack of trust in or wor-
neither anxiety nor depression ries about unfriendly staff (36), being left alone in
JW-DEQ, A Japanese Version Wijma Delivery Experience Question-
labor, appearing silly and lack of involvement in deci-
naire, Part A; W-DEQ A, Wijma Delivery Experience Questionnaire Part sion-making (13,32,40) as well as trauma and previous
A. sexual abuse. In addition, FOC often coincides with
a
Nordeng et al. and Storksen et al. include the same study population depressive and compulsive personalities predisposing
as Adams et al. (2012) included in the meta-analysis. women to postnatal depression and post-traumatic
stress disorder (3,35,67,70). Hence, various strategies
and encourage women to express FOC as identifying have been proposed to help women cope with FOC,
women with tocophobia early in pregnancy may provide i.e. psycho-education, birth preparation (2,57). There is
an opportunity to support maternal mental health evidence that continuity of care, developing meaningful,
(3,22,49). This is important as there is growing evidence trusting relationships, involving women fully in deci-
linking tocophobia with increased maternal cortisol levels sion-making and working in partnership to provide
as well as the exacerbation of other mental health issues, woman-centered care can improve outcomes (63,71–73)
which may lead to serious and long-term consequences but there is no standardized care pathway for women
for mother and baby (54). with tocophobia in pregnancy (68). Future researchers
Tocophobia is difficult to quantify. Currently, the W- could strive to develop appropriate interventions aimed
DEQ A is used as the reference standard for assessment at identifying pregnant women at risk of tocophobia,
and ‘on the spot’ diagnosis (1,14). As mentioned, we such as decision aids, which are increasingly being used
found a variation in the cut-off point used for the W- in healthcare settings (74).
DEQ A. A criticism of this tool has been that it may This comprehensive systematic review was based on a
exclude some women who could benefit from support, detailed search carried out on six relevant databases with
therefore some studies used a slightly lower cut-off point no language or date restrictions and is based on a proto-
(66 or 71 rather than 85) (15,36), resulting in more refer- col that is registered on the International prospective reg-
rals for intervention. Moreover, an in-depth psychometric ister of systematic reviews database (24). This protocol
analysis of the W-DEQ A advised that calculating a total was available on the National Institute for Health
score and using a cut-off to define tocophobia may not Research website and subsequently, the systematic review
be appropriate as this is based on the premise that the followed standardized reporting guidelines (24,75). The
W-DEQ A is uni-dimensional (36,58–61). The use of strength of our review lies in the large number of studies,
subscales has been advocated to determine specific rea- which allowed extensive sensitivity and subgroup analyses
sons behind the woman’s fear and identify risk factors to be conducted.
that might make a woman more vulnerable, such as lack The main limitation in the study was the very high
of social support (58). In addition to the issues outlined statistical heterogeneity evident from the I2 estimates in
above, the W-DEQ A is lengthy and impractical for clini- the meta-analyses. It was not possible to carry out a
cal use, so researchers are striving to establish more prac- subgroup analysis on maternal age, social supports and
tical tools (14,22). existing mental health due to lack of such data in the

916 ª 2017 Nordic Federation of Societies of Obstetrics and Gynecology, Acta Obstetricia et Gynecologica Scandinavica 96 (2017) 907–920
M.A. O’Connell et al. Worldwide prevalence of tocophobia

included studies. These factors are reported to be asso-


ciated with tocophobia (9,18,40,54). When we con- Acknowledgments
ducted a subgroup analysis including only studies that We would like to acknowledge Prof. John Browne,
used the W-DEQ A to define tocophobia, significant Department of Epidemiology & Public Health, at Univer-
heterogeneity remained, suggesting that this issue is sity College Cork for his postgraduate module on System-
more complex than simply being explained by variation atic Reviews as well as Maura Flynn the librarian at
in the definition used. The authors acknowledge that Brookfield School of Nursing & Midwifery at University
the prevalence of tocophobia depends on several factors College Cork.
including various personality characteristics, previous
birth experiences and cultural determinants including
local obstetric norms, personal and religious beliefs Funding
(17,55,69). Furthermore, many of the studies included This work was carried out as part of doctoral studies at
in the systematic review were of a cross-sectional design The Irish Center for Fetal and Neonatal Translational
that only capture FOC at one point in time during Research (INFANT Center), which is supported by
pregnancy (see Table 1). Science Foundation Ireland (grant no. 12/RC/2272).
It is possible that questionnaires may not be applicable
in different countries and in other cultural contexts (even
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ª 2017 Nordic Federation of Societies of Obstetrics and Gynecology, Acta Obstetricia et Gynecologica Scandinavica 96 (2017) 907–920 919
Worldwide prevalence of tocophobia M.A. O’Connell et al.

for childbearing women. Cochrane Database Syst Rev. Figure S1. Sensitivity analysis: Forest plot of the pooled
2016;4:CD004667. prevalence of tocophobia using studies that screened
73. Nieuwenhuijze M, Korstjens I, de Jonge A, de Vries R, women in the third trimester (27–42 weeks of gestation)
Lagro-Janssen A. On speaking terms: a Delphi study on only.
shared decision-making in maternity care. BMC Pregnancy Figure S2. Subgroup analysis: Forest plot of the pooled
Childbirth. 2014;14:223. prevalence of tocophobia in high- and low-quality studies
74. Agoritsas T, Heen AF, Brandt L, Alonso-Coello P, as determined by the quality assessment score. High-qual-
Kristiansen A, Akl EA, et al. Decision aids that really ity studies were studies that scored 5 or more out of a
promote shared decision making: the pace quickens. BMJ.
maximum of 8.
2015;350:g7624.
Figure S3. Subgroup analysis: Forest plot of the pooled
75. MOOSE Guidelines for Meta-Analyses and Systematic
prevalence of tocophobia by study region.
Reviews of Observational Studies*. *Modified from Stroup
Figure S4. Subgroup analysis: Forest plot of the pooled
DF, Berlin JA, Morton SC, Olkin I, Williamson GD,
prevalence of tocophobia according to the time period in
Rennie D, et al. Meta-analysis of observational studies in
epidemiology: a proposal for reporting Meta-analysis Of
which the studies were conducted.
Observational Studies in Epidemiology (MOOSE) group. Appendix S1. Search strategy.
JAMA; 2000. Appendix S2. Quality assessment tool.

Supporting information
Additional Supporting Information may be found in the
online version of this article:

920 ª 2017 Nordic Federation of Societies of Obstetrics and Gynecology, Acta Obstetricia et Gynecologica Scandinavica 96 (2017) 907–920

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