Prosjekt: Project protocol for health consequences of female genital mutilation/cutting (FGM/C)

Mandate

The WHO Department of Reproductive Health and Research, and NORAD Department of Health and AIDS commissioned the NOKC to conduct a systematic review to support the organisations’ work concerning FGM/C among girls/women subjected to and at risk for the practice in all countries where FGM/C may occur. The overall aim of the systematic review is to support well-informed decisions in health promotion that inform work to reduce the practice of FGM/C and improve quality of services related to FGM/C.

 Purpose

This systematic review has two purposes: 1) to summarise empirical quantitative research describing the physical consequences of FGM/C on women ages 15 and older, and 2) to summarise empirical quantitative and qualitative research describing the physical and psychological consequences of FGM/C on girls ages 0-14.

Aims one and two will be pursued in separate reports because of their different scopes and datasets which will be included. Psychological consequences will not be examined for women because this issue was address in a previous project (Berg, 2010a). The age cut off of 15 years (at the time of study) was chosen in accordance with the Demographic and Health Surveys. These are nationally representative surveys, carried out every five years in several African countries, and for those where FGM/C occurs, a module of questions on FGM/C for women ages 15-49 is included in the survey. Further, FGM/C is usually carried out on girl under the age of 15, and age of marriage and age of consent in many African countries is around age 15.

 Background

Female Genital Mutilation/ Cutting (FGM/C) is a traditional practice that involves "all procedures involving partial or total removal of the external female genitalia or other injury to the female genital organs for non-medical reasons" (WHO, 2008), and is performed on pre-pubescent girls (WHO, 2006). A range of reasons, which vary across cultural groups, exist for FGM/C, but the practice is closely linked with ethnic identity, with FGM/C serving as an ethnic marker throughout the lifespan (UNICEF, 2005a). The practice is also rooted in tradition as well as religio-social beliefs such as the conviction that FGM/C is a religious requirement, that it is necessary to control women’s sexuality and preserve family honour (WHO, 1999), and that it is a prerequisite for marriage or an economic necessity in cases where women are largely dependent on men (UNFPA, 2007).

While FGM/C transcends geography, it is mainly found among diverse ethnic groups in about 28 countries in Africa as well as some countries in the Middle East and Asia (WHO, 2006; UNICEF, 2005b). National figures for African countries show a prevalence of FGM/C of more than 70% in Burkina Faso, Djibouti, Egypt, Eritrea, Ethiopia, Guinea, Mali, Mauritania, Northern Sudan, Sierra Leone, and Somalia. However, because of the magnitude of FGM/C among certain ethnic groups there is great variation in prevalence between and within countries (UNICEF, 2005b).

Given the practice is usually performed on young girls often without anaesthetics, it is reasonable to assume that it is traumatic and that these girls suffer psychological consequences as a result of FGM/C. A systematic review conducted by the NOKC summarized the results from four comparative studies that assessed risk for psychological consequences for women with FGM/C compared with women without FGM/C (in contrast with the proposed systematic review we included only comparative studies). On a study level, the results showed that significantly more women with FGM/C than without suffered from a psychiatric diagnosis, post-traumatic stress disorder, anxiety, phobias, and somatisation (Berg, 2010a). We also examined the sexual consequences of FGM/C, performing meta-analyses which showed that compared to women without FGM/C, women with FGM/C were 1.5 times more likely to experience pain during intercourse, twice as likely to report that they did not experience sexual desire, and  they experienced significantly less sexual satisfaction (Berg, 2010a).

On the physiological level, FGM/C causes permanent, irreparable changes in the external female genitalia and there are no known health benefits to FGM/C. Across the world, according to WHO estimates (2008), between 100-140 million girls/women live with the consequences of FGM/C. For example, a large study examining more than 28,000 women in birth clinics in African countries concluded that women with FGM/C were significantly more susceptible to problems related to childbirth (WHO Study Group, 2006). However, two smaller studies from Sweden could not confirm that there was a statistical correlation between FGM/C and childbirth-related problems (Essen, 2002; 2005). Also research on infertility as a consequence of FGM/C seems inconclusive. While a study that included women from the Central African Republic, Cote d’Ivoire, and Tanzania did not find a statistical association between FGM/C and infertility (Larsen, 2000), a case-control study from Sudan found a statistically as well as clinically significant association between FGM/C and infertility (Almroth, 2005). Part of these inconsistent results may be related to the fact that the more severe a woman’s cutting, the more likely it is that she will experience adverse consequences (WHO, 2008).

Per today, the best summary of the physical complications from FGM/C is a WHO literature review from 2000, "A systematic review of the health complications of female genital mutilation, including sequelae in childbirth." The review aimed to identify and summarize primary data on health complications following FGM/C, with particular emphasis on sequelae in childbirth, as well as psycho-sexual outcomes. The review included a range of study designs, identifying various types of complications, the most common of which were severe pain, bleeding, difficulty in passing urine and faeces, and infections. Around the same time, Obermeyer completed a related review of the health complications and sexual consequences following FGM/C, which concluded that "the powerful discourse that depicts these practises as inevitably causing death and serious ill health, and as unequivocally destroying sexual pleasure, is not sufficiently supported by the evidence" (Obermeyer, 1999, p79). The review was updated in 2005 (Obermeyer, 2005), with largely similar conclusions but also the acknowledgement that there were statistically higher risks documented for some but not all types of health conditions.

The above studies, and in particular the WHO report from 2000, are often cited documents to describe the health consequences of FGM/C. However, the literature searches for the reviews are out-dated, the reviews were not systematic, some outcomes were missing, and because no meta-analyses were performed, the risks of various health consequences were not quantified. The degree of health risk is important and can be specified, as has been shown in a systematic review on the sexual consequences of FGM/C conducted by researchers at NOKC (Berg, 2010a).

There is a need to update and synthesize the research on the health consequences following FGM/C. A systematic review on this issue will not only identify important new studies that have been conducted in recent years and identify the range of health consequences, but also quantify the risks and identify knowledge gaps. The results can assist in improving identification of and treatment of complications from FGM/C, informing guidelines on treatment, guiding future research, and supporting interventions towards the ultimate elimination of FGM/C.

 Methods

We will conduct a systematic review of the health consequences of FGM/C on girls/women in accordance with the NOKC handbook for summarizing evidence (Nasjonalt kunnskapssenter for helsetjenesten, 2011) and the guidelines in the Cochrane Handbook for Systematic Reviews of Interventions (Higgins and Green, 2009) with regards to defining the review question, searching for studies, selecting studies and collecting data, assessment of methodological quality, and, as far as possible, analysing data. Both organisations specialise in the synthesis of evidence concerning the efficacy and effectiveness of interventions - neither the NOKC handbook nor the Cochrane handbook specify the best approach for the synthesis of qualitative evidence. For this type of data, we will therefore rely on the examples and guidelines published by the Evidence for Policy and Practice Information and Co-ordinating Centre (EPPI-Centre) as well as the recommendations provided in courses attended by the project leader (Evidence synthesis of qualitative research in Europe (ESQUIRE); Methodological basis of HTA; Comprehensive systematic review training programme – JBI).  

  Search strategy:

We will conduct a comprehensive and systematic search for relevant literature in the following international databases:

• African Index Medicus
• Anthropology Plus
• British Nursing Index
• The Cochrane Library: Cochrane Database of Systematic Reviews (CDSR), Cochrane Central Register of Controlled Trials (CENTRAL), Database of Abstracts of Reviews of Effects (DARE), Health Technology Assessment Database (HTA)
• EMBASE
• MEDLINE
• MEDLINE In-Process & Other Non-Indexed Citations
• PILOTS
• POPLINE
• PsycINFO
• Social Services Abstracts
• Sociological Abstracts
• WHOLIS

 The search strategy will incorporate subject headings (e.g. MeSH terms in MEDLINE) and text words (in title and abstract) relating to FGM/C (e.g. genital cutting) and the four classifications thereof, including mutilation, circumcision, excision. We will not apply any methodology search filters in order to maximise the sensitivity of searches. We will neither restrict the searches to any specific languages nor publication dates. Search strategies will be prepared by information specialist Sari Ormstaad, in cooperation with the project group and commissioners.

We will supplement the electronic database searches with searches in sources for grey literature (OpenGrey, OpenSigle, OAIster), and browse websites of six international organizations that are engaged in projects regarding FGM/C:

• Population Council
• Population Reference Bureau (PRB)
• The Centre for Development and Population Activities (CEDPA)
• The United Nations Children’s Fund (UNICEF)
• The United Nations Population Fund (UNFPA)
• The World Health Organization (WHO)

 In addition to the searches mentioned above, we will scan reference lists of relevant reviews and included studies for further relevant references. We will also communicate with experts engaged in FGM/C related work and ask for studies about the consequences of FGM/C.

  Selection of literature:

Three reviewers (Berg+Denison, Berg+Underland) will independently read all titles/and or abstracts resulting from the literature searches. We will compare our judgements and obtain full text copies of the studies deemed relevant. The same pairs of reviewers, acting independently, will classify the studies read in full text as relevant (meets all inclusion criteria) and therefore to be included, or not relevant and therefore to be excluded. We will then compare our judgements and include studies that meet all inclusion criteria. For each of the two screening levels, pre-designed inclusion forms will be used. The inclusion forms will contain questions regarding types of studies, types of participants, type of FGM/C, and outcomes measured.

When considered necessary, we will contact the authors of studies to aid the decision process. Differences in opinion in the screening process will be resolved through re-examination of the record and subsequent discussion. If consensus cannot be reached, a third reviewer will be consulted. Studies considered in full text but excluded will be listed in an appendix and reasons for exclusion provided.

 Inclusion/exclusion criteria:

Study designs

• systematic reviews
• cohort studies
• case-control studies
• case series
  • cross-sectional studies
  • case reports.

 We will use study design features (as defined in the Cochrane glossary) not study design labels to designate the studies. For aim one, we will exclude qualitative studies and all studies without a quantitative measure of a physical consequence of FGM/C. For aim two (physical and psychological consequences of FGM/C on girls ages 0-14), we will in addition to the study designs listed above  also include qualitative studies in which girls portray how they experienced the procedure on the psychological level. Consistent with similar systematic reviews, we define qualitative evidence as "papers had to report results of qualitative (i.e. text-based and interpretive) analysis based on qualitative methods of data collection" (Smith, 2005 p826).

Population

Girls and women who have been subjected to any type of FGM/C, as classified by the WHO (2008). We will enforce no limitations on age, race/ethnicity, nationality or other participant characteristics. We will pursue aims one and two in separate reports and median age of the participants will be used to decide the age cut off if there is a mix of ages below and over age 15 (mean will be used if median age is not available). We will exclude studies about FGM/C where genital cutting was performed for medically indicated or purely cosmetic reasons.

We will not examine consequences of a woman’s FGM/C on other individuals, such as her sexual partners or babies during birth.

Interest/Event

FGM/C classified as type I to type IV according to the WHO modified typology. Type I, clitoridectomy, involves partial or total removal of the clitoris and/or the prepuce. Type II, excision, involves partial or total removal of the clitoris and the labia minora, with or without excision of the labia majora. Type III, infibulation, involves narrowing of the vaginal orifice with creation of a covering seal by cutting and appositioning the labia minora and/or the labia majora, with or without excision of the clitoris. Type IV, other, involves all other harmful procedures to the female genitalia for non-medical purposes, for example: pricking, piercing, incising, scraping and cauterization (WHO, 2008).

Comparison

Both studies with and without a comparison group are eligible for inclusion. The reasons for this is that FGM/C is a non-medically indicated procedure, which means that all complications can be considered unintended adverse effects and will rarely have been studied with rigorous study design. Indeed, experimental studies for this issue would not be ethically justifiable. Based on our previous systematic reviews regarding FGM/C (Denision, 2009; Berg, 2010a; Berg; 2010b; Berg, forthcoming) we are aware that studies which compare women with and without FGM/C on various outcomes exist, but our impression is that these studies are in short supply. When the study reports a comparison group, the study must compare either 1) any type of FGM/C vs no FGM/C, or 2) one type of FGM/C vs another type, e.g. type I vs type III, as defined by the WHO (2008).

Outcomes

We will include all types of physical consequences / complications following FGM/C, both short- and long term consequences experienced by girls/women. These include but are not limited to:

Primary outcomes

• Death
• Shock
• Infections (e.g. tetanus, hepB,  STIs, vaginal tract infections, chronic pelvic infections)
• Infertility
• Fistula (urethra-vaginal)
• Pain (e.g. headaches , backache, chronic pelvic pain)
• Urinary complications (e.g. retention, incontinence, painful urination, time of urination)

 Secondary outcomes

• Bleeding/haemorrahage (e.g. post-coital bleeding )
• Menstrual complications (e.g. painful menses, hematocolpos, dysmenorrhoea)
• Vaginal calculus formation
• Cysts
• Tissue injury (to bladder, etc.), fractured/displaced bones
• Urethral meatal stenosis/ urethral stricture
• Abscesses
• Keloid and other scarring
• Obstetric complications (e.g. episiotomy, caesarean section)

The outcomes must be self-reported by the girls/women having experienced these or documented by health personnel and study investigators. When physical outcomes pertain to children, we will accept reports by the girl’s parents. We will exclude secondary reports, i.e. of the type where participants or others report consequences experienced by neighbours, friends, acquaintances, etc.  

For aim two, we will include the range of psychological consequences on girls age 0-14 following FGM/C, such as anxiety, depression, hostility, loss of trust towards caretakers. Qualitative studies will be included because this type of data is uniquely suited to describe the lived experiences and reflections surrounding the procedure.

Other:

Unpublished reports, abstracts, brief and preliminary reports will be considered for inclusion on the same basis as published reports. We will include all publication years and languages. When considered likely to meet the inclusion criteria, studies will be translated to English using Google translator, unless professional translation is considered necessary.

In sum, studies eligible for the systematic review are those that quantitatively report the occurrence of a physical complication on girls/women having been subjected to FGM/C, and for aim two also qualitative studies will be considered.

 

Assessment of methodological quality:

With respect to assessment of methodological quality of included studies, two reviewers (Berg+Denison or Berg+Underland) will independently assessed the quality of studies, using appropriate checklists for each included study design (see below). The two reviewers will discuss and agree upon a final decision of strong, moderate or weak methodological quality for each study. Where differences in judgments occur, this will be resolved by re-examination of the publication and subsequent discussion. If consensus cannot be reached, a third reviewer will be consulted.

For cohort studies, case-control studies, and case series we will use the respective NOKC checklists. For case reports we are searching for an appropriate checklist and consider using questions developed by Oliveira and colleagues (2006). Given our focus on consequences of exposure to FGM/C, the NOKC assessment tool for cross sectional studies will be used but modified by the addition of five questions from the NOKC quality assessment tool for cohort studies in order to capture whether 1) the compared groups (women with FGM/C and women without FGM/C) were selected from the same population; 2) the groups were comparable with respect to important backgrounds factors; 3) exposure and outcome were measured in the same way in the two groups; 4) the person who assessed the outcome was blind to whether participants were exposed or not; and 5) known, potentially important confounders had been considered in the study design and/or analyses, resulting in an adapted checklist with 12 questions (this modified checklist was successfully used by us previously, in Berg, 2010). For aim two, we will use the qualitative studies assessment tool designed by the Critical Appraisal Skills Programme (CASP) to assess the methodological quality of qualitative studies (the checklist was successfully used by us previously, in Berg, 2010b; Berg, forthcoming). The paired reviewers’ assessments of each checklist question of each study will be listed in an appendix.

Extraction of data and analysis:

Two authors (Berg+Denison or Berg+Underland) will independently extract data from the included sources using a pre-designed data recording form. They will then compare their results and if differences in data extracted occur this will be resolved by re-examination of the publication and subsequent discussion. The following data will be extracted from included studies and presented in text and tables:

• Title, authors, and other publication details
• Study design
• Methodological study quality
• Sample characteristic including residency, current age, etc.
• FGM/C characteristics such as type of cutting, age of cutting, type of practitioner
• Health consequences

We will extract and group dichotomous and continuous data for all outcomes (health consequence/complication) meeting the inclusion criteria. We will group the data according to outcomes across the studies, and present the results of these in text and tables. We will prioritise presenting results from those studies with highest internal validity and when a large number of studies document an outcome, results from those studies with lowest internal validity will be presented in an appendix. When possible we will present data for women with FGM/C and without FGM/C separately in the results tables to allow for comparison, and similarly, we will present data for women with different types of FGM/C separately.

For aim two, if qualitative studies are included, two reviewers (Berg+Denison) will read the texts independently of each other and extract all text data related to psychological consequences from FGM/C. This data extraction will be inclusive, as recommended by Briggs and colleagues (2007). Thus, these data extracts will include both extracts from research participants and extracts of the interpretations made by the researchers (Sandelowski, 2003; Thomas, 2008). We will copy all findings in the form of sentences, phrases or text units appearing to deal with consequences following FGM/C verbatim onto a pre-designed data extraction form.

With respect to quantitative data analyses, when possible, we will estimate effect on dichotomous variables by the relative risk (RR) and 95% confidence interval (95%CI) and effect on continuous variables by mean difference (MD, or standardized mean difference when possible) and 95%CI. For case-control studies where dichotomous variables are presented we will estimate effect by the odds ratio (OR) and 95%CI, because a case-control design involves the selection of research subjects on the basis of the outcome measurement rather than on the basis of the exposure. With respect to the quantitative data with only one group of girls/women, we will use the generic inverse variance approach (Higgins and Green, 2009) to synthesize the extracted consequences across studies. Further, if studies are sufficiently similar, we will pool those that can be grouped together and use the statistical technique of meta-analysis to estimate risk, with RevMan v5.1. (Cochrane Collaboration meta-analysis software). To be pooled, the same outcome/consequence must be assessed in a similar manner in similar populations across similar studies. Standard analysis procedures will be used; i.e. Mantel-Haenszel random effects meta-analyses will be conducted for dichotomous outcomes and inverse-variance random effects meta-analyses for continuous outcomes. We will also examine between-study heterogeneity, with the Chi-square (Chi²) and I-square (I²) tests. A high value shows that most of the variability across studies is due to heterogeneity rather than to chance.

When possible (there is a sufficient number of similar studies) we will perform sensitivity analyses for 1) performer (health care provider and traditional provider), 2) age (at which FGM/C was done, at onset of complications, or time between procedure and onset), and 3) type of FGM/C (according to WHO modified typology, 2008).

We will apply the instrument Grading of Recommendations Assessment, Development and Evaluation (GRADE) with GRADE-Profiler version 3.6 to assess the extent to which we can have confidence in the effect estimates (Guyatt et al., 2011). For more details about the GRADE system we refer to publications by the GRADE Working Group (gradeworkinggroup.org). We will use the standard definitions in grading the quality of the evidence (Balshem et al., 2011):

Quality	Definition of quality
High	We are very confident that the true effect lies close to that of the estimate of effect
Moderate	We are moderately confident in the effect estimate: The true effect is likely to be close to the estimate of the effect, but there is a possibility that it is substantially different
Low	Our confidence in the effect estimate is limited: The true effect may be substantially different from the estimate of the effect
Very low	We have very little confidence in the effect estimate: The true effect is likely to be substantially different from the estimate of effect

In order to synthesize any qualitative data we may have extracted for aim two, we will examine all findings in the form of text, and use a thematic analysis which draws on published EPPI studies (e.g., Harden et al., 2004; Shepherd et al., 2006). Thematic analysis involves identifying prominent or recurring themes in the literature and subsequently summarizing the findings of the different studies under thematic headings (Dixon-Woods, 2005). For this systematic review, we will organize the findings from each study according to type of psychological consequence. We will then examine the findings of each study in turn and group them into thematic categories. This will be based on commonality of meaning, which involves taking concepts from one study and recognizing the same concept in another study, though they may not have been expressed using identical words. As delineated by Dixon-Woods (2005), we will apply a thematic analysis that is data driven, i.e. driven by the themes identified in the studies that are included, allowing themes to emerge from the qualitative findings. The organization of findings into thematic categories will first be conducted individually by two reviewers (Berg+Denison), who then will discuss and agree on a set of categories. We will select a few quotations for each category that represent views that appear frequently. If possible (enough data exist), in the last qualitative analysis step, having created categories that represented descriptive themes of the findings, we will then combine categories to create synthesized themes. This will involve reflecting on the thematic categories as a whole and looking for similarities and differences among the categories. Two reviewers will work together and examine the themes in light of the consequences they seem to represent. Through discussion and reflection, we will arrive at a consensus on overall understanding and we will develop a set of analytic themes, each of which will be represented by a few quotations that capture the essence of the theme.

 Activities and timetable

• Project protocol: The commissioners (WHO and NORAD contact persons) will be asked to comment and confirm the project protocol before it is reviewed by two internal (NOKC) individuals with methodological expertise and by two external persons with expertise in the area of FGM/C. This is expected to take place in December 2011. The NOKC leader group will have final approval of the project protocol. We aim to obtain approval in January-February 2012.
• Search for literature: In collaboration with the commissioners, the project coordinator will work with a NOKC search specialist to develop a search strategy (Sari Ormstaad). This is expected to occur in January 2012. The sensitive search strategy used for three previous NOKC systematic reviews about FGM/C will serve as start point.
• Selection of literature: Three reviewers (Berg+Denison and Berg+Underland) will select literature in January-April 2012. The same three reviewers will evaluate the methodological quality of selected studies in April-June 2012.
• Extraction of data and analysis: Three reviewers (Berg+Denison and Berg+Underland) will extract data from selected studies. Berg will be responsible for creating tables, performing GRADE, write the reports and coordinate the collaboration among commissioners, reviewers and the working group. We expect this work to be completed by the end of October 2012.
• Review and publication: The reports will be reviewed by two internal (NOKC) individuals with methodological expertise and by two external persons with expertise in the area of FGM/C. This is expected to take place in October-November 2012. The NOKC leader group will have final approval of the reports, which is expected towards the end of December 2012. The report for aim two will likely be completed in 2013 as completion of the report for aim one will be prioritized.