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2ARC Centre of Excellence for Children and Families over the Life Course Centre, UQ, Australia
5Centre for Rural and Remote Health, James Cook University, Mount Isa, Australia
6Department of Health Sciences, Global Health unit, University Medical Center Groningen. The
Netherlands
9Department of Population Medicine. College of Medicine, QU Health, Qatar University. Doha, Qatar
This article has been accepted for publication and undergone full peer review but has not been
through the copyediting, typesetting, pagination and proofreading process, which may lead to
differences between this version and the Version of Record. Please cite this article as doi:
10.1111/1471-0528.16491
This article is protected by copyright. All rights reserved
Corresponding author:
Accepted Article
Tahmina Begum
80 Meiers Rd | Long Pocket Precinct | Indooroopilly | Queensland- 4068
Email: t.begum@uqconnect.edu.au
Running title: Caesarean deliveries on maternal request
Tweetable abstract:
Globally, the CD rate has doubled in the last 15 years to 21% and is increasing annually by 4%(12). Of
29·7 million globally performed CDs; around 6.2 million unrequired CDs were done in 2008 (9).
According to professional and press media, CD on maternal request (CDMR) is a significant
contributor on the unprecedented increase of unrequired CDs(13, 14). In some European countries,
CDMR constitutes 7-22% of CDs and is related to fear of childbirth (15). Citing obstetricians self-
reported data, a study from Australia has reported the CDMR rate as 17.3% (16). However, the
reliability of data is a concern due to methodological variation in defining CDMR (17)and in some
cases, proxy indications were used when it was not mentioned explicitly in the medical records (13, 14).
The recent systematic review findings on maternal preference on mode of birth raises a further
debate where globally only 15.6% of women reported CDMR as their first choice (18). However, to
rule out the widely criticqued statement on CDMR, an actual estimation of CDMR rate is required
instead of reporting subjective preference which has not been studied systematically yet(13, 17). To fill
this knowledge gap, we conducted a systematic review of the literature on global CDMR incidences
using a valid and reliable definition (13, 19). Meta-regression was used to explore the variation in
CDMR proportion by different subgroups and to identify significant predictors of CDMR. Knowing this
burden and its variation will be helpful for resource allocation and effective planning on the reduction
of CDMR rates.
Search Strategy
A comprehensive search strategy was developed using Boolean operators incorporating the key
search terms such as "caesarean delivery", "caesarean section", "caesarean birth", "maternal
request", "maternal demand", "patient demand", "patient request", "self-request", elective, planned,
"maternal decision". We used different combinations of free text and MeSH term for the key search
term. Five databases; PubMed, Embase, CINAHL, Medline and Google Scholar, were searched from
January 1985 to May 2019. Grey literature was searched from Grey Net International and ProQuest
online database. The lower limit was set to the year 1985 as the World health organisation (WHO)
brought the discussion on the adverse effect of un-necessary CDs for the first time during this year
(21). (Appendix S1).
The CDMR definition used in this review was approved by system experts during State of Science
Conference in 2006. A panel of 18 members representing different disciplines of medicine and public
health sat together to investigate the scientific evidence to recommend an easily applicable and valid
CDMR definition. The approved CDMR definition was "elective caesarean deliveries performed at
term for a singleton pregnancy without obstetric and or any medical reasons" (13). Accordingly, we
set our study inclusion and exclusion criteria under the format of population, intervention,
comparison, outcome and study design (PICOS)(22). We included observational studies that provide
complete information to compute CDMR proportion. However, articles reporting elective CDs from
pregnancy risk factors (2) and physician recommendation were excluded. The further exclusion
criteria were non- English articles, case notes, editorial review and conference abstract. We did not
include articles that talked about mother and physician's perception of CDMR instead of reporting
actual numbers of CDMR (Table S1, screening criteria).
We extracted CDMR data when it was mentioned explicitly in the caesarean section indication lists
and matched with our operational definition. However, four studies contradicted our CDMR case
definition, where the previous CD was mentioned as the cause of CDMR (25-28). We subtracted the
previous CD from total CDMR cases for those four articles.
Quality assessment
Two reviewers independently did the quality appraisal by using "risk of bias assessment" tool
proposed by Hoy et al. (29). The validity of the tool was reported most precise among all available
quality assessment checklists used for evaluation of prevalence studies (29). Accordingly, a ten-point
scale was used to assess both the external and internal validity of the study. External validity was
evaluated through the first four items consisting of representativeness, sampling strategy and non-
response bias. Internal validity was appraised subsequently by measurement and analytic bias
highlighted under items five to nine and item10 respectively (Table S2A). The individual articles
were assessed against all ten potential safeguards and assigned one summary quality count which
had a possible maximum of ten points. Of course, a study with a maximum count will not necessarily
be devoid of bias, and this count was used to rank studies between one and zero with the best study
having rank one (30).
Outcome variable:
We defined two CDMR proportion: 1) Absolute proportion of CDMR by the number of CDMR over
total deliveries (CDMR-TD) and 2) Relative proportion of CDMR; the number of CDMR over total
CDs (CDMR-CD). We also report CDMR proportion out of total elective CDs depending on data
Moderator variables
Geographical region, economic region, study site, study design, data source, study years, study
population and mean maternal age. World- Bank classification of 2015 was used to classify studies
by economic status and geographical region (32). By economic category, each study could fall under
any of the categories: high income (HI), upper middle income (UMI), lower middle income (LMI) or
low income (LI) group. The geographical region was specified as; Europe and Central Asia, North
America, East Asia, Middle East, Sub Saharan Africa. Whereas, to describe the study site as an
urban or rural setting, we utilised the study reported data. The variable "data source" re-categorised
into two categories, namely, national where a representative number of delivering women from a
country were surveyed and health facility, which did the survey among selected health facilities. The
study year was grouped into two; studies conducted before and after 2010. The study design was
assessed by data collection nature, either prospective or retrospective. Since mean parity was not
available from all studies, we categorised study by their study population; nulliparous and parous.
Whereas, mean maternal age was retrieved directly from the individual study.
Statistical analysis
Initially, an estimate of the logit transformed CDMR proportions were generated using MetaXL
software version 5.3 (33). The software also computes the standard error of the logit transformed
proportions as well as a quality effects weight for each study under the quality effects model. The
latter makes use of the quality rank generated for each study from the bias assessment (34). Since the
CDMR proportion from an individual study was expected to be small, the logit transformation helps to
stabilise the variance and avoid pooled estimates outside the 0 – 1 range. The advantage of using
the quality effects model is that the between-study variability is adjusted based on observed study
quality instead of the random variable assigned by the random-effects model. Another benefit of the
quality effects model is that it avoids overdispersion and spurious statistical significance (35, 36).
Subsequently, a quality effect weighted meta-regression was done to investigate the possible
sources of variability in CDMR proportions observed between studies. The transformed pooled
estimate of CDMR along with all other moderator variables was imported in Stata software (version
16.0) to run the meta-regression analyses. Since all moderators were categorical, the meta-
Results
Search outcome
In total, 615 articles were retrieved from the initial search, and among them, 158 were discarded as
the duplicates. From the initial screening of title and abstract, 53 articles added for full-text review.
The full text was not available for 16 studies and 13 more articles excluded from eligibility screening.
One study from low-income countries did not meet eligibility as CDMR was grouped under CD from
non- medical causes (3). Additionally, seven more articles added from reference tracking that made
the final count of 31 articles. (Figure 1)
The highest number of articles were from the continent "East Asia" (n=12) followed by "Europe and
Central Asia" (n=10). In the economic subgroup, 15 articles were from HI, 14 from UMI and two from
LMI countries; none was from the LIC group. At the country level, one-third of our included articles
were from China (n=11). The data collection period for individual article ranged from 2 months (38) to
18 years(39). The majority of the studies used data from health facilities, whereas nine of them used
population-level data (25, 27, 39-45). The studies done in the urban context were higher in number
(n=12). Few population-based studies used specific code to report CDMR (25, 27, 41). In Taiwan, the
code 0373B was assigned by the national insurance company and consistently being used in
Around five million total births from 14 countries constituted the study sample. The absolute
proportion of CDMR ranged from 0.2% to 42%, and the majority of them (20 articles) had a rate
below 5%. The relative proportion ranged from 0.9% to 60%. The study with the lowest CDMR
percentage was from Ireland (46) while China had the highest CDMR percentage (42). Most
importantly, more than 50% of CDs were reported to be from the maternal request in three Chinese
studies (42, 47, 48). Elective CD proportion was available for 16 articles and ranged in between 2% to
64%. Summary findings are presented in Table S3.
Not all but some studies mentioned the reason for CDMR. The study from Ghana stated that
unavailability of the assisted vaginal delivery and proper labour monitoring facilities made the mother
less confident in choosing normal birth (49). Whereas, physician incompetency to handle complicated
labour was suspected in one study from China (42) Study from Nigeria documented that when women
prefer CD obstetricians were less likely to oppose this(50).
Only eight of 31 articles (25, 27, 38-40, 44, 47, 48) explained the determinants of CDMR. In general, the
commonly cited determinants were increasing maternal age(40, 44), primi mother(27, 38, 44, 47), residing in
urban area(38), excess pregnancy weight gain(38, 44), fear of childbirth(25, 26) and delivering in a well-
equipped health facility (38).
Study quality
Out of ten available safeguards, the lowest achieved count was six (50-52), and the highest was ten(40,
41, 44, 53). Only few of the studies mentioned their representativeness to national population(25, 27, 40, 41,
43, 44, 53, 54) and some did not discuss about representativeness against target population(26, 28, 45, 46, 48-52,
55-60), that jeopardized external validity. Outcome definition of CDMR did not match in four articles,
and the previous CD was included as a cause of CDMR there (25-28). Two other studies used maternal
self-reported data that suggest possible reporting bias (42, 43) Quality appraisal findings are depicted
in (Table S2B)
We explored the reason for heterogeneity by doing meta-regression based subgroups analysis
across eight subgroups (Table 1). The absolute proportion of CDMR was 11 folds higher in upper-
middle-income countries (11.71, 95% CI: 5.34-23.52) than in high-income countries. Across five
geographical regions, the Middle East had the highest CDMR estimates, followed by East Asia. The
articles that reported data beyond 2010 had higher CDMR proportion than those published before
that. Studies reporting CDMR proportion from nulliparous women had higher rates than the studies
with parous study populations. Similarly, CDMR was higher among women with mean age below 28
years. As suggested by R2 value, economic region individually explained 81% variation in CDMR-TD
and 74% in CDMR-CD proportion.
[Table 1:Here]
Table 2 on multivariable metaregression shows that studies from high-income countries reporting
CDMR beyond 2010 had a lower proportion of CDMR-TD ( 0.39%, 95% CI: 0.12-1.26) than their
counterpart (1.53%; 95% CI:1.12-2.09). A similar pattern was observed for the rest of the economic
regions. Economic region and study year together explained 84% of the variation in the CDMR-TD
and 76% of the variation in CDMR-CD. Comparison between Random and Quality Effect output from
univariable and multivariable models are presented in Table S4 & Table S5.
[Table 2: Here]
Discussion
Main findings
To our knowledge, this is the first systematic review and meta-regression that reports the proportion
and major factors associated with CDMR using evidence across the globe. The absolute CDMR
proportion varied significantly (0.2% to 42%) across the countries. The economic region and study
year contributed around 84% of the variability in the CDMR rates. The upper-middle-income
This study followed a standard protocol and methodology of systematic review and meta-regression
analysis (20, 61, 62). The methodological challenges and validity of data around CDMR were addressed
by using a valid and reliable definition of CDMR. We checked the comparability of our work with
other related reviews. Two systematic reviews on CDMR can be mentioned, one by Viswanathan,
2006(63), and another by Schantz et al., 2019 (17). The first review only focused on developed
countries’ CDMR rates, whereas the 2nd review examined the methodological variation of CDMR
data collection. As a secondary outcome, Schantz et al. reported the range of global CDMR rates
though their search strategy was not comprehensive. Their review was based on a single search
engine and done by a single reviewer (17). We addressed the limitations of these two reviews in our
study . Additionally a main strength of this review is the use of the quality effects meta-regression to
explain the observed variability across studies.
One of the limitations of our review is that non-English articles were not included. Ability to include
articles in Spanish or Portuguese language could have captured more data from Brazil and Latin
America, where the epidemic rise of CD has been noticed (12, 64). The other limitation of our review is
that it relies on the study reported data that can vary according to the medical record-keeping system
of that country. Since the majority of the articles explicitly mentioned the maternal request as the
cause of CD, there is less chance of reporting bias. However, to cite this review as global CDMR
incidence needs cautious interpretation as data from low-income countries are missing, and a wide
range of CDMR incidence is noted across the studies.
Interpretation
The Lancet global series on CD rate reported that 106 out of 169 countries in the world had their CD
above the threshold level(12). Our review on the subgroups of CD has identified that data on CDMR is
rarely reported, only 14 countries across the world reported CDMR proportions. Thus claiming
maternal request as a major driver of increasing CD rate needs more evidence. Our review identified
a higher CDMR proportion after 2010 compared to 2010. However, when we used interaction terms
in between economic region and study year, the proportion of CDMR was always lower beyond 2010
We found fewer studies that report population-level data (25, 26, 41). The challenge of reporting CDMR
using population-level data was mentioned as a unavailability of specific ICD code for CDMR under
national vital statistics (61). In the absence of specific code, CDMR data extracted from medical
records followed the exclusion method, by excluding CDs done from clinical indications(40). The data
collected by this method carry more chance of bias when maternal requests are omitted; instead,
relative clinical conditions are recoded as CD indication (31). The opposite example also persists in
the context of high CD rate, medical practitioners, when obliged to justify CD indications, tend to
swing CD indications as maternal request (57). Both these forms of misclassification provide incorrect
CDMR estimates (57).
The evidence around risk vs benefit of CDMR is also not comprehensive that hinders better maternal
engagement in decision making process (66). With an attempt to generate robust evidence, the
recent Cochrane systematic review could not produce a single trial on CDMR vs planned vaginal
birth(67). However, sporadic data reported higher maternal and neonatal health risk from CDMR
To overcome the current research gap, future research should focus on the appropriate reporting of
CDMR using a consistent definition and prospectively designed research (72, 73). A stable reduction of
CDMR rate should be supported by the improved maternal and neonatal health indicators.
Additionally, women who opted for CD on their own should be interviewed to learn about the level of
engagement of physician in the decision-making process(46, 47, 74). But, even more important, women’s
preference for the mode of birth should be explored during early week of pregnancies to identify the
prospective risk group of women who may need more counselling and support around childbirth(43).
Conclusion
Caesarean delivery on maternal request seems commonly discussed but less reported as
highlighted by our systematic review. Unfortunately, the bulk of current evidence is derived from
sources using an unreliable definition of CDMR that makes it difficult to differentiate the actual
contribution of maternal request on the medically no indicated caesarean sections. That, in turn, will
have implications for adequate planning and dissemination of interventions to reduce CDMR
incidence. Therefore, appropriate reporting of CDMR should be a key priority in maternal health
policies and practices. Moreover, better maternal engagement and supports are required to reduce
CDMR incidence and related health and financial burden.
Contribution to authorship
TB1, KMSUR and AAM wrote the protocol. TB1 and KMSUR independently did the article screening
and quality check. TB1 extracted data. Differences in opinion were resolved by discussion between
TB1, KMSUR, AAM and SD. Data analysis was done by TB1, YF, AAM and SD Data interpretation
and manuscript writing by TB1, AAM, SD, SUR, JS, YF, SA and TB 2.
Disclosure of interests
None
Funding
None
Acknowledgement
Authors admit the contribution of Marcos Riva, librarian of the University of Queensland for his help
during the literature search.
Reference
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Caption list:
Figure 1: PRISMA flow diagram on the article screening process and output on the global incidence
of caesarean deliveries on maternal request
Table 2: Multivariable meta-regression§ analysis of CDMR by the interaction of economic region and
study year
Table S1: Study inclusion criteria for systematic review on the global incidence of CDMR using
PICOS format
Table S2A: Risk of Bias assessment tool to do quality appraisal of the observation studies on on
caesarean deliveries for maternal request
Table S3: Summary of the observational studies reporting the incidence of caesarean deliveries on
maternal request
Table S4: Model comparison on univariable analysis findings on caesarean deliveries for maternal
request by subgroups
Table S5: Model comparison on multivariable analysis findings on caesarean deliveries for maternal
request by subgroups
Appendix S1: Search Strategy for the global incidence of the caesarean deliveries on maternal
request
*
Subgroups CDMRa-TDb **
CDMR-CDc
d d
% (95% CI ) % (95% CI )
High income & study year<2010 1.53(1.12-2.09) 5.98(4.87-7.31)
High Income & Study year >=2010 0.39(0.12-1.26) 1.82(0.52-6.2)