BMC Public Health: WHO Systematic Review of Prevalence of Chronic Pelvic Pain: A Neglected Reproductive Health Morbidity

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Research article Open Access


WHO systematic review of prevalence of chronic pelvic pain: a
neglected reproductive health morbidity
Pallavi Latthe*1, Manish Latthe2, Lale Say3, Metin Gülmezoglu3 and
Khalid S Khan4

Address: 1Birmingham Women's Healthcare NHS Trust, Birmingham, UK, 2Tower Hill Medical Centre, Great Barr, Birmingham, 3UNDP/UNFPA/
WHO/World Bank Special Programme of Research, Development and Research Training in Human Reproduction, Department of Reproductive
Health and Research, World Health Organization, Geneva, Switzerland and 4Academic Department of Obstetrics & Gynaecology, University of
Birmingham, Birmingham, UK
Email: Pallavi Latthe* - pallavi.latthe@bwhct.nhs.uk; Manish Latthe - manish@latthe.freeserve.co.uk; Lale Say - sayl@who.int;
Metin Gülmezoglu - gulmezoglum@who.int; Khalid S Khan - k.s.khan@bham.ac.uk
* Corresponding author

Published: 06 July 2006 Received: 12 September 2005


Accepted: 06 July 2006
BMC Public Health 2006, 6:177 doi:10.1186/1471-2458-6-177
This article is available from: http://www.biomedcentral.com/1471-2458/6/177
© 2006 Latthe et al; licensee BioMed Central Ltd.
This is an Open Access article distributed under the terms of the Creative Commons Attribution License (http://creativecommons.org/licenses/by/2.0),
which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly cited.

Abstract
Background: Health care planning for chronic pelvic pain (CPP), an important cause of morbidity
amongst women is hampered due to lack of clear collated summaries of its basic epidemiological
data. We systematically reviewed worldwide literature on the prevalence of different types of CPP
to assess the geographical distribution of data, and to explore sources of variation in its estimates.
Methods: We identified data available from Medline (1966 to 2004), Embase (1980 to 2004),
PsycINFO (1887 to 2003), LILACS (1982 to 2004), Science Citation index, CINAHL (January 1980
to 2004) and hand searching of reference lists. Two reviewers extracted data independently, using
a piloted form, on participants' characteristics, study quality and rates of CPP. We considered a
study to be of high quality (valid) if had at least three of the following features: prospective design,
validated measurement tool, adequate sampling method, sample size estimation and response rate
>80%. We performed both univariate and multivariate meta-regression analysis to explore
heterogeneity of results across studies.
Results: There were 178 studies (459975 participants) in 148 articles. Of these, 106 studies were
(124259 participants) on dysmenorrhoea, 54 (35973 participants) on dyspareunia and 18 (301756
participants) on noncyclical pain. There were only 19/95 (20%) less developed and 1/45 (2.2%) least
developed countries with relevant data in contrast to 22/43 (51.2%) developed countries. Meta-
regression analysis showed that rates of pain varied according to study quality features. There were
40 (22.5%) high quality studies with representative samples. Amongst them, the rate of
dysmenorrhoea was 16.8 to 81%, that of dyspareunia was 8 to 21.8%, and that for noncyclical pain
was 2.1 to 24%.
Conclusion: There were few valid population based estimates of disease burden due to CPP from
less developed countries. The variation in rates of CPP worldwide was due to variable study quality.
Where valid data were available, a high disease burden of all types of pelvic pain was found.

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Background ing and the search was restricted to human and female.
Chronic pelvic pain (CPP) is a debilitating condition We also hand searched the bibliographies of all relevant
among women with a major impact on health-related reviews and primary studies to identify cited articles not
quality of life, work productivity and health care utilisa- captured by electronic searches. The search did not have
tion. It is the single most common indication for referral any language restrictions. We also attempted to contact
to women's health services accounting for 20% of all out- authors from indexed electronic abstracts on theses for
patient appointments in secondary care [1]. This leads to data.
a substantial burden on limited health care resources. For
example, $881.5 million are spent per year on its outpa- Study selection
tient management in the USA [2] and an estimated £158 Studies on CPP were selected using the following prede-
million are spent annually on the management of this fined criteria:
condition in the UK National Health Service [3].
Participants
Valid information about the true extent of CPP is an Non-pregnant women without cancer participating in sur-
essential consideration in resource allocation and health veys about rates of CPP. Women with or without known
care planning. In addition, these basic epidemiological endometriosis or irritable bowel syndrome were to be
data are necessary to monitor trends of the disease burden included
as well as to inform design of other research in this condi-
tion, like genetic and environmental epidemiology to Outcome
assess aetiology, qualitative studies to establish well being There is lack of consensus on the definition of CPP in the
and overall quality of life, and studies aimed at the devel- published literature [10]. We used a definition based on
opment of new treatment strategies [4]. The epidemiolog- duration and nature of pain (constant or intermittent,
ical features of CPP have been generously reported in the cyclical or noncyclical pain, that persisted for 3 months or
worldwide literature. The majority of the studies are lim- more [11]) and included three types: cyclical pain during
ited by small sample size and hence their estimates are menstruation (dysmenorrhoea), deep dyspareunia and
imprecise. The need to summarise these data have gener- noncyclical pelvic pain. Studies were included in the
ally received scant attention [5]. absence of information on duration of pain as long as it
was explicit that cases of acute pain were excluded.
A systematic literature review was performed to ascertain:
the prevalence rates of CPP according to the type of pain; There is a debate about the definition of CPP as recurrent
its geographical distribution; its variation within sub- pain such as that associated with isolated dysmenorrhoea
groups defined by age and development status of the and dyspareunia is often considered biologically distinct
country of origin; and the effect of study quality and rep- from chronic pain, however many women have overlap-
resentativeness on the rates. ping symptoms. For our review, CPP may be regarded as a
composite of chronic and recurrent pelvic pain.
Methods
Our systematic review followed a protocol developed Study design
using widely recommended methodology [6,7] and com- Cross sectional studies that reported the prevalence of
plied with the MOOSE statement [8]. CPP.

Data sources Data extraction and quality assessment


We searched general bibliographic databases: Medline Two reviewers (PML, ML) extracted data independently,
(1966–2004), Embase (1980–2004) and PSYCHINFO using a piloted form, on participants' characteristics, study
(1887–2004). We also searched specialist computer data- quality and rates of CPP. Data on studies not published in
bases: LILACS (Literatura Latinoamericana y del Caribe en English were extracted by people with a medical back-
Ciencias de la Salud 1982 to 2004), CINAHL (January ground with command of the relevant language. In some
1980 to 2004) and SCISEARCH (1974–2004). Our search studies the existence of multiple symptoms amongst indi-
term combination for electronic databases was as follows: viduals could not be evaluated separately due to the struc-
MeSH headings, text words and word variants for "pelvic ture of their questionnaires and their manner of reporting,
pain" or "dysmenorrhoea" or "dyspareunia" or "low so these were excluded. Two of the datasets in the system-
abdominal pain" were combined using Boolean operator atic review of dysmenorrhoea prevalence were extracted
and with terms like "prevalence" or "community survey" from the abstracts of theses were when the full copies
or "incidence". These were combined with terms repre- could not be obtained [12,13].
senting relevant study designs e.g. cross-section, survey
etc. according to recent recommendations [9] for search-

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The methodological quality of all selected papers was due to the statistically significant heterogeneity in this
assessed to evaluate internal validity using the following subgroup of high quality representative studies. We how-
attributes [6]: (a) Study design to determine if CPP assess- ever, have reported the rate ranges for representative stud-
ment had been performed prospectively to minimise ies [18]. Publication and related biases were examined for
recall bias; (b) Adequacy of sampling by assessing by plotting log rates versus their corresponding variances
whether recruitment of participants was random or con- in a funnel plot. Funnel asymmetry was tested by Begg's
secutive or a convenience sample; (c) Sufficiently high test [19].
response rate (>80%); (d) Use of a validated measure-
ment tool to ascertain CPP [14] as this ensures that partic- Results
ipants' responses are a true representation of the The electronic search yielded a total of 1226 citations (fig-
underlying condition; (e) Sample size calculation so as to ure 1). On examination of titles and abstracts, 225 were
ascertain prevalence reliably. It was considered 'adequate' found to be potentially relevant and their full papers were
if the studies had mentioned that sample size was calcu- obtained. The reference lists of these revealed 32 further
lated and 'inadequate' if this was not done or not men- citations. After reviewing these, 109 papers were excluded
tioned explicitly. The studies were classified into high and (see additional file 3). The remaining 148 papers (see
low quality groups based on compliance with 3/5 quality additional file 2 for a complete list of included articles)
criteria or more. Representativeness of the sample for gen- met the inclusion criteria, which provided data on
eral population (source of sample) was considered sepa- 459972 participants. 30 studies overlapped and reported
rately to methodological quality as this relates to external more than one outcome or more than one group of pop-
validity. This distinction is important because internally ulation. There is very little data (1/148 papers) available
valid studies of women attending hospitals or for private from the least developed countries. 22/43 developed
health care checks may not be biased but they are less use- countries had published data on prevalence of CPP in
ful due to sampling of non-generalisable population contrast to 19/95 less developed and 1/46 least developed
groups.

Numerators and denominators were extracted or esti- Total citations identified from electronic searches 1226
mated from each study for computing rates and confi-
dence intervals (CI). In most studies, prevalence 1001 Citations excluded after screening abstract
measured how many women had CPP at a single point in
Papers retrieved for detailed evaluation: 225
time, i.e. point prevalence [14]. Period prevalence, based
on the number of women developing CPP during a
defined period of time, was reported only in a few studies. Searching of reference lists: 32

Data synthesis Papers excluded: 109


No/ Insufficient /unclear data 5
For each study, we computed prevalence rates and their
Not a primary data source 19
95% CI according to the three different types of CPP.
Not on prevalence of pelvic pain 50
Rates of the different CPP were mapped to depict the var-
Duplicate data 9
iation in prevalence by country of origin. Heterogeneity
Unobtainable 3
was explored in the log rates of CPP graphically using for-
Study performed in : pregnant/postnatal women 8
est plots of point estimates of rates and their 95% CI and : cancer 4
statistically using Cochrane Q and I2, a statistic that quan- : other specified disorders 7
tifies the degree of inconsistency across studies in a meta- : case-control study/case report 4
analysis on a scale ranging from 0–100% [15,16]. In
reviews with high degree of inconsistency meta-analysis is
not recommended. Meta-regression explored if inconsist-
Primary papers included in systematic review: 148
ency in results across individual studies could be
178 studies (some papers report more than one outcome/study):
explained by variations in countries' development status,
106 – dysmenorrhea
participants' age, representativeness of the sample and
54 - dyspareunia
methodological quality of the included studies [17]. For
18 -noncyclical CPP
development status we used the United Nations classifica-
tion (developed, less developed and least developed) for
countries. Study quality was assessed separately for indi- Figureselection
Study
chronic 1
pelvic pain
for systematic review on prevalence of
vidual items and scores. We performed both univariate Study selection for systematic review on prevalence of
and multivariate meta-regression analysis. We did not chronic pelvic pain.
pool results even from high quality representative studies

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countries (P < 0.0001) [the denominator is the number of


developed, less developed and least developed countries

Dysmenorrhea
in the world according to UN country classification].
Study quality assessment (figure 2) revealed deficiencies
in many areas of methodology: Two (1.2%) studies met
all five high quality criteria, 13 (7.1%) met 4/5 criteria
and 40 (22.5%) met three or more criteria.

The details of studies included in the systematic review on


prevalence of dysmenorrhoea, dyspareunia and noncycli-
cal pelvic pain are given in table 1, 2 and 3 respectively

Dyspareunia
(additional file 1). The data on prevalence of CPP in
included studies is summarised in figures 3, 4. Epimaps in
Figure 3 depict the available data by countries, on world-
wide prevalence of different types of chronic pelvic pain
by percentage.

Dysmenorrhoea
The prevalence rates ranged from 1.7% [2] to 97% [20] in
106 studies including 125249 women. Prevalence rates
for cyclical pelvic pain in the UK reported were between
45% (12% reporting severe dysmenorrhoea) [21] to 97% Noncyclical pelvic pain
[20] (14% severe) for any dysmenorrhoea in community
based studies and between 41–62% in hospital based
studies [22-24]. In other European countries it was similar
[25,26]. The lowest prevalence was reported in Bulgaria
(8.8%) in women hospitalised with adnexitis between the
ages of 19–41 years and the highest was in Finland (94%)
in girls aged 10–20 years [27]. In 20 high quality studies
with representative samples, the rate of dysmenorrhoea Figure 3of worldwide prevalence of chronic pelvic pain
Epimaps
was reported between 16.8 [28] to 81% [29]. There was Epimaps of worldwide prevalence of chronic pelvic pain.
substantial heterogeneity in forest plots and I2 statistic was
98% (figure 4). The funnel plot for dysmenorrhoea was
asymmetrical (Begg's test P = 0.02; figure 5a) but not for representativeness, age < 25 years or development status
representative studies (P = 0.333). Metaregression (Table of the country (developed versus less developed versus
4) showed validated measurement tool to be a significant least developed).
factor to explain heterogeneity but not study quality score,

Figure
Quality
lence of of
2chronic
studiespelvic
included
pain in systematic review on preva-
Quality of studies included in systematic review on
prevalence of chronic pelvic pain. (Data presented as
100% stacked bars; figures in the stacks represent number of Figure
Prevalence
4 of different types of chronic pelvic pain
studies). Prevalence of different types of chronic pelvic pain.

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ity that was statistically significant in meta-regression


Begg's funnel plot with pseudo 95% confidence limits
analysis (table 4) but age under 60 was not (P = 0.15).

0
Non-cyclical pelvic pain
The prevalence rates ranged from 4.0% [35] to 43.4% [36]
-1 in 18 studies including 299740 women. Two recent stud-
ies stated a 3 month prevalence of 15% in women aged
18–50 years in the USA [2] and 24% in ages between 12–
logr

-2
70 in the UK [37]. In less developed countries in South
Dysmenorrhoea
- 5a East Asia the prevalence rates varied from 5.2% in India,
-3
8.8% in Pakistan to 43.2% in Thailand [36]. Amongst the
3 high quality studies with representative samples, the
-4
rate range of noncyclical pain was 2.1 [37] to 24 [29]% as
2 0 .2 .4
s.e. of:
logr reported from the primary studies. There was heterogene-
ity across studies (I2 = 99%). Metaregression (table 4)
0 revealed that prospective design, adequate sampling strat-
egy and sample size estimation tended to describe lower
prevalence of noncyclical pelvic pain though none of
logr

-2
these were significant (table 4). The funnel plot for non-
cyclical pelvic pain was asymmetrical (Begg's test P =
-4 0.048; figure 5c) but not for representative studies (Begg's
Dyspareunia
- 5b
test P = 0.88).
-6
0 .5 1 Discussion
s.e. of:
logr
This is the first systematic review of the worldwide preva-
2
lence of CPP. The variation in rates of CPP worldwide was
explained by variable study quality. The number of popu-
0 lation-based studies yielding estimates of burden of CPP
from less developed countries was low. High quality liter-
ature comprising representative samples revealed a high
logr

-2
burden of disease for all types of pelvic pain, however,
there remained heterogeneity in these subgroup of stud-
-4
Noncyclical pelvic
- pain 5c ies.

-6 We believe that the findings of our study are valid as our


0 .5 1
s.e. of:
logr review methodology was rigorous. A prospective review
protocol was used and a concerted effort made to identify
ies
Funnel
Figure plots
5 of the three types of pelvic pain prevalence stud- all the available evidence without language restriction. We
Funnel plots of the three types of pelvic pain preva- made concerted efforts to report this systematic review as
lence studies. [a – dysmenorrhoea; b – dyspareunia; c – suggested by the MOOSE consensus statement [8]. Both
noncyclical pelvic pain] the methodology and the rates of CPP varied among the
included primary studies and we explored the reasons for
variations comprehensively. This review represents the
best available evidence on the estimates of the prevalence
Dyspareunia of CPP at the time of writing and rate ranges from high
The prevalence rates ranged from 1.3% [30] to 45.7% [31] quality studies of representative samples provide the best
in 54 studies including 35973 women. The rates of dys- information available for targeting services at women suf-
pareunia varied from 1.1% in Sweden [32] to 45% [31] in fering from pelvic pain.
US studies. In 18 high quality studies with representative
samples, the rate range of dyspareunia was reported to be The variation in geographical distribution may be related
8 [33] to 21.1 [34]% Studies were markedly heterogene- to study characteristics, study quality, age groups included
ous (I2 = 97.9%) and the funnel plot for dyspareunia was and definitions used rather than intrinsic differences
asymmetrical (Begg's test P = 0.001; figure 5b) but not in between the prevalence of CPP between the different pop-
representative studies (P = 0.227). The representativeness ulations. Other plausible explanations might be differ-
of sample provided the main explanation for heterogene- ences in the prevalence of sexually transmitted infections,

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availability of medical and other resources or cultural dif- Competing interests


ferences. Although we have included studies from 1924 The author(s) declare that they have no competing inter-
onwards, majority of the studies are from 1980 onwards. ests.
The population demographics are unlikely to have under-
gone major changes over this period, making the studies Authors' contributions
relevant to current populations' Nevertheless, one has to Dr. P Latthe: Conception, design, search, study selection,
bear in mind the changes in patterns of smoking, sexually data extraction, data synthesis, writing the manuscript
transmitted infections and contraception during this time
and their consequences for pelvic pain [38]. Substantial Dr. M Latthe: Data extraction, tables
differences in and even complete absence of definitions,
together with differences in age ranges of the populations Dr L Say, Dr AM Gülmezoglu: Revising the manuscript,
studied also complicate the interpretation of our findings. figures

One study noted the overlaps among different pain disor- Prof. KS Khan: Conception, design, data synthesis, writing
ders (irritable bowel syndrome, interstitial cystitis etc) and revising the manuscript
[39], A better understanding of the epidemiology of these
disorders would facilitate our understanding of somato- Additional material
sensory disorders in general. We explored for heterogene-
ity using sophisticated technique of metaregression. We
found that validated measurement tool, representative-
Additional file 2
List of studies included in the systematic review of prevalence of chronic
ness and high quality were the variables that were statisti- pelvic pain
cally significant (P < 0.05) for dysmenorrhoea, Click here for file
dyspareunia and noncyclical pelvic pain respectively. In [http://www.biomedcentral.com/content/supplementary/1471-
the meta-regression for countries classification, there are 2458-6-177-S2.doc]
so few least developed countries that the power is very
low. Also, it is worth pointing out that there is a potential Additional file 3
risk of aggregation bias when age is used as a variable in Appendix of excluded studies
Click here for file
meta-regression. [http://www.biomedcentral.com/content/supplementary/1471-
2458-6-177-S3.doc]
The information on the rates of dysmenorrhoea, dyspare-
unia have implication for provision of services to policy- Additional file 1
makers in terms of provision of improved access for these Tables 1, 2 and 3 of tables of characterstics of included studies on dysmen-
women to health care resources as well as the develop- orrhoea, dyspareunia and noncyclical pelvic pain respectively
ment of appropriate treatment protocols. Future epidemi- Click here for file
[http://www.biomedcentral.com/content/supplementary/1471-
ological studies should ideally be prospective, with
2458-6-177-S1.doc]
explicit definitions of the outcome and representative of
the general population. Close attention must be paid to
study design and to the use the validated measurement
tools for validity and comparability of the results across
Acknowledgements
studies and regions. Such efforts will need funding agen- Mary Publicover, Librarian, Birmingham Women's HealthCare Trust for
cies to be willing to support broader and more systems her help with the searches; Luciano Mignini, Sam Pretlove, H. Kuntz,
oriented approach [40]. Tomoo Shaktari and Stefka Ritchie for translation of foreign language man-
uscripts; Daniel Wojdyal for producing the Epimaps
Conclusion
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