PROTOCOL: Interventions to reduce the prevalence of female genital mutilation/cutting in African countries

1.1 Definition and classification of female genital mutilation/cutting1

Female genital mutilation/cutting (FGM/C) is a traditional practice that involves the partial or total removal or other injury to the female genital organs for cultural or other non-therapeutic reasons (WHO, 2008). The current classification describes four types of FGM/C: Type 1, clitoridectomy involves partial or total removal of the clitoris and/or the prepuce. Type 2, excision, involves partial or total removal of the clitoris and the labia minora, with or without excision of the labia majora. Type 3, 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. Infibulation is considered the most invasive type of FGM/C. Defibulation, opening of the covering seal, is often necessary prior to childbirth. Reinfibulation refers to the recreation of an infibulation after defibulation. Type 4, other, involves all other harmful procedures to the female genitalia for non-medical purposes, for example: pricking, piercing, incising, scraping and cauterizing (WHO, 1997).

1.2 Prevalence of FGM/C

FGM/C is practised in more than 28 countries in Africa, usually on girls under the age of 15 years, and in some countries in the Middle East and Asia. FGM/C is also practised by immigrant communities in a number of other countries, including Australia, Canada, France, New Zealand, Norway, Sweden, Switzerland, the United Kingdom, and the United States (HRP, 2006).

Recent figures for African countries show a prevalence of FGM/C of more than 70 percent in Burkina Faso, Djibouti, Egypt, Eritrea, Ethiopia, Guinea, Mali, Mauritania, Northern Sudan, and Somalia (Yoder & Kahn, 2008). However, there is great variation in prevalence between and within countries, reflecting ethnicity and tradition. Therefore, UNICEF (2005) has proposed that countries be categorized in three groups according to FGM/C prevalence rates: Group 1, 80 percent or higher prevalence, e.g. Ethiopia and Somalia, Group 2, 25-79 percent, e.g. Senegal and Kenya, and Group 3, 1-24 percent, e.g. Nigeria (Table 1).

1.3 Reasons for the practice of FGM/C

The practice of FGM/C is rooted in religious, personal and societal beliefs within a frame of psycho-sexual and social reasons such as control of women's sexuality and family honour, which is enforced by community mechanisms (WHO, 1999). While reasons for the practice vary across cultural groups, social reasons may include FGM/C as an initiation act for girls into womanhood, as an act of social integration and for the maintenance of social cohesion. Socio-economic reasons include beliefs that FGM/C is a prerequisite for marriage or an economic necessity in cases where women are largely dependent on men. Religious reasons rest on the belief that it is a religious requirement. Hygienic and aesthetic reasons for FGM/C include beliefs that the female genitalia are dirty and unsightly, and health reasons include beliefs that FGM/C enhances fertility and child survival. FGM/C may also be an important source of income for circumcisers (UNFPA, 2007).

1.4 Consequences of FGM/C

1.5 Interventions to reduce the prevalence of FGM/C

Efforts to abandon the practice of FGM/C in Africa have used several different approaches which, in turn, have had implication for interventions. These approaches include those based on human rights frameworks, legal mechanisms, health risks, alternative rites, positive deviance, training health workers as change agents, training and converting circumcisers, and the use of comprehensive social development processes. Interventions based on these approaches have targeted stakeholders at individual, interpersonal, community and national levels (Muteshi & Sass, 2005).

In 2007, the Population Reference Bureau (PRB) published their results of an extensive survey of current intervention projects taking place in African countries (Feldman-Jacobs & Ryniak, 2007). In total, PRB identified 92 projects, 27 of which were evaluated, mostly by observational designs. Only four of the 27 evaluated projects (15%) used a controlled before-and-after design, and about a dozen of the evaluations used cross-sectional or pre-post intervention questionnaires or interviews without a comparison group. While contributing great understanding about the range of interventions initiated to curb the prevalence of FGM/C, the overview did not reach any conclusions about the effectiveness of interventions.

More recently, the authors of the proposed project specifically examined the effectiveness of interventions to reduce the prevalence of FGM/C in a systematic review (Denison et al, 2009). Through our literature search of February 2009 we identified a total of seven controlled studies, six of which could be obtained in full text. All six studies were controlled before-and-after studies carried out in African countries. In contrast to the PRB overview (Feldman-Jacobs & Ryniak, 2007), we included only controlled studies, i.e. studies with reference to a non-intervention comparison group, and we concluded that while the evidence base is insufficient to draw definite conclusions, there are possible advantageous developments as a result of interventions. Notably, the review highlighted the uncertainties regarding relevance of the interventions (e.g. regarding objectives, intervention targets, activities). That is, since it was not a focus of the systematic review, we were unable to provide any assessment of the degree to which the interventions were appropriate responses to the populations' needs with respect to FGM/C, including the degree to which factors that contribute to the perpetuation of the practice were taken into account in the interventions. It is apparent that the degree of relevance of the intervention exerts a considerable influence on an intervention's effectiveness in reaching its designated goals, and may to a large extent help explain variation in behavioral and other outcomes among members of groups.

In sum, two recent publications have examined aspects of interventions designed to reduce the prevalence of FGM/C. However, the effectiveness of interventions in the context of relevance has yet to be explored.

1.6 Contextual factors related to the continuance or discontinuance of FGM/C

FGM/C is a long-standing tradition that has become inseparable from ethnic and social identity among many groups (UNICEF, 2005). Disaggregation of data from the Demographic and Health Surveys (DHS)² shows that the practice of FGM/C varies by demographic variables such as age, urban-rural residence, and region or province, and also by variables such as education, ethnicity and religion (Yoder et al, 2004). Further analysis of DHS data by UNICEF suggests that educational attainment, a woman's own circumcision status and ethnicity have the greatest influence in explaining support or opposition to the practice (UNICEF, 2005). Thus, programmes designed to reduce the prevalence of FGM/C should be country specific and adapted to reflect regional, ethnic and socio-economic variances while also taking into account the diverse reasons why FGM/C is practised among a given ethnic or cultural group (UNICEF, 2005).

3.1 Criteria for inclusion and exclusion in the review

3.2 Search strategy for identification of relevant studies

The primary method of study identification will be electronic searches, as advised by the Cochrane Handbook (Higgins & Green, 2009)). For the recently completed systematic review of the effectiveness of interventions designed to prevent the prevalence of FGM/C we searched systematically for relevant literature up to February 2009 in 13 international databases: African Index Medicus, Anthropology Plus, British Nursing Index and Archive, The Cochrane Library (CENTRAL, Cochrane Database of systematic Reviews, Database of Abstracts of Reviews of Effects), EMBASE, EPOC, MEDLINE, PILOTS, POPLINE, PsychINFO, Social Services Abstracts, Sociological Abstracts, and WHOLIS. Under the guidance of one reviewer (ED) a research librarian performed the searches using a strategy incorporating subject headings (for example MeSH terms in MEDLINE, see http://www.nlm.nih.gov/mesh/) and text words (in title and abstract) relating to FGM/C and the four classifications thereof. No method filters were applied as we were more concerned about sensitivity than specificity and prepared to screen a large number of references. We did not restrict our searches by country or language. The MEDLINE search strategy served as the model for the other database searches using appropriate controlled vocabulary as applicable. Our search strategy for MEDLINE is shown:

Database: Ovid MEDLINE(R) In-Process & Other Non-Indexed Citations and Ovid MEDLINE(R) 1950 to Present 1.

Circumcision, Female/

We will include databases for ‘grey’ literature (defined here as “reports that are produced by all levels of government, academics, business and industry in print and electronic formats but that are not controlled by commercial publishers” [Higgins & Green 2009]): Demographic and Health Surveys, British Library for Development Studies, IDEAS, JOLIS, Google Scholar, and Google. Theses and dissertations will be included. In addition to the electronic database search, we will perform bibliographic back-referencing to identify new leads. We will also supplement the electronic search with searches in databases of six international organizations that are engaged in projects regarding FGM/C: Centre for Development and Population Activities (CEDPA), Population Council, Population Reference Bureau (PRB), The United Nations Children's Fund (UNICEF), The United Nations Population Fund, and The World Health Organization (WHO). Notably, Popline contains ministerial reports on FGM/C. We will also ask for suggestions for literature we may have missed from FGM/C experts, e. g. the research director of Population Council. Forward citation tracking will be performed through the ISI Web of Knowledge in order to identify further studies. Lastly, we will conduct a hand search of relevant journals (African Journal of Reproductive Health, Social Science & Medicine).

3.3 Details of study coding categories

3.4 Statistical procedures and conventions

For the synthesis of effectiveness evidence (Research question 1; Synthesis 1, Figure 1, Appendix 6), the studies will be grouped by intervention, and key intervention features, outcomes and effect estimates will be described in tables. Sensitivity analyses will be used to examine the stability of the effect estimates in relation to quality of studies.

With respect to statistical analyses, we will present dichotomous data for the outcomes listed in the inclusion criteria in results tables when pre- and post scores for both intervention and comparison groups are reported by study authors, allowing comparison. We will estimate effects of interventions in two ways. First, we estimate effect by the adjusted absolute risk difference (ARD) in which the pre-post change score (in percentage points) in the comparison group will be subtracted from the pre-post change score (in percentage points) in the intervention group. Whether ARD is deemed to be large is a judgment whereby we also take baseline difference into account. Second, we will estimate effect by the relative risk (RR) and 95 percent confidence interval (95%CI) based on post-intervention data. The benefit of using this approach is that effect can be estimated even though prognostic similarity at baseline cannot be assumed due to non-randomization. Because none of the effect studies we have identified so far is randomized we will need to use this approach. Should randomized studies be identified in the update search and included in the review we will synthesize these separately. An obvious drawback is that some element of judgment of baseline similarity will be present when conclusions are drawn from the results. We will present continuous data with mean difference and 95%CI.

Forest plots of effect sizes for each grouping of outcomes will be presented. Subgroup analyses or moderator analyses will be used to explore sources of heterogeneity. If sufficient numbers of studies are identified (a minimum of ten studies with one dichotomous moderating variable) meta-regression will be used to perform moderator analyses. Potential moderating variables are intervention content and underlying theory of action (e.g. educational only, training health workers as change agents, comprehensive social development), stakeholder group/s involved, and setting.

3.5 Treatment of quantitative and qualitative research

The contribution of quantitative and qualitative data describing contextual factors will pertain primarily to the understanding of heterogeneous results from the intervention studies, and to help define interventions more precisely in relation to their contexts (Research question 2).

We will utilize an integrative evidence approach, whereby quantitative evidence has methodological dominance. Data extraction and analyses of quantitative and qualitative evidence will largely be completed in separate streams (Figure 1, Appendix 6). Data from cross-sectional survey studies (quantitative data) will be combined with data from studies which examined various stakeholders' perspective of factors influencing the continuance and discontinuance of FGM/C (qualitative ‘views’ studies). Our integrative evidence approach is largely based on published examples and guidelines from the Evidence for Policy and Practice Information and Co-ordinating Centre (EPPI) (e.g., Harden et al., 2004; Shepherd et al., 2006; Thomas & Harden, 2008). The synthesis will be aggregative (Dixon-Woods et al., 2006) and focus on summarizing data by pooling conceptually similar data from the quantitative studies and the qualitative studies. First, we will analyze the two sets of evidence separately (each step is delineated below). Results from the quantitative data set will be used as organizing principles for the qualitative data analysis. Throughout the analysis we will place most weight on the quantitative results, such that the qualitative results are subsumed under the quantitative results and the qualitative results extend the results from the quantitative analysis.

With respect to the quantitative studies (Synthesis 2, Figure 1, Appendix 6), we will use a generic inverse variance approach (Higgins, 2009) to synthesize the extracted quantitative data across studies. Further, for each study and each group of stakeholders, we will review the units extracted and categorize them according to our predefined categories, such as expressed reasons for continuing FGM/C. We will then determine the frequencies of these categories in order to create a ranked list of factors, ending up with one list for each stakeholder group in each country/local region. Thus, we will be able to both synthesize data across studies (settings) and describe local variation.

With respect to the qualitative evidence (Synthesis 3, Figure 1, Appendix 6), our analysis is thematic and 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 summarizing the findings of the different studies under thematic headings (Dixon-Woods, 2005). The data for synthesis of qualitative studies are in text form. In order to synthesize these, we will copy all findings verbatim into a word processing program. These data extracts include both extracts from research participants and extracts of the interpretations made by the researchers. We will organize the findings from each study according to whether the factors are continuance or discontinuance factors, for each stakeholder group separately. Next, we will examine the findings of each study in turn and assign descriptive codes to describe the findings. Codes will be created without prejudging the meaning of the data and inductively to capture meaning and content of each sentence or phrase. For example, we will code the finding “People perform FGC to reduce a girl's sexual desire” under continuance factors as ‘It curbs women's sexuality’. During the coding process, the reviewers will look for similarities and differences between codes in order to start grouping them. Next, we will group findings into thematic categories. This will be based on commonality of meaning as well as frequency and strength of various stakeholders' cognitions about FGM/C, separately for each stakeholder group. Consistent with thematic analysis, it 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 explained by Dixon-Woods (2005), thematic analysis can be data driven, i.e. driven by the themes identified in the studies that are included, or theory driven, i.e. driven by themes identified through assessment of the literature. For this systematic review, given quantitative evidence will be given methodological dominance, we will work by using both a priori codes from the included quantitative studies to seek out evidence from the qualitative findings, as well as allowing themes to emerge from the qualitative findings. Text units appearing to deal with related content will be identified and sorted into categories, to which we assign thematic headings, such as ‘reduced sexual interest’ and ‘affirming community membership’. We will do this separately for each stakeholder group, thus developing broader concepts that captured similar themes from different papers. The organization of findings into related areas will first be conducted individually by two reviewers, who then discuss and agree on a set of categories. We will select a set of quotations for each category that represents views that appear frequently, thereby describing the stakeholders' understandings of the factors related to the continuance and discontinuance of FGM/C.

In the last qualitative analysis step, having created categories that represent 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. The two main reviewers will work together and examine the themes and their corresponding codes in light of the review question, inferring continuance and discontinuance factors from the views stakeholders were expressing about FGM/C. Through discussion and reflection, the main authors will come to a consensus on overall understanding and develop a comprehensive set of analytic themes, across stakeholder groups, such as ‘sexual restraint’ and ‘community allegiance’. For each analytic theme, we will select a set of quotations that capture the essence of each theme.

Once both the quantitative and qualitative sets of data are analyzed, they will be integrated (Synthesis 4, Figure 1, Appendix 6). The integration will involve creating a matrix (table) in which we juxtapose the list of quantitative factors and thematic categories for each stakeholder group. We will work from the quantitative results and seek out evidence from the qualitative results, thus working “down” from pre-existing quantitative understandings. We will generate a set of statements that represents the aggregation of the underlying forces of the continuation and discontinuation of FGM/C from different papers.

The procedure described above was used by Berg and Denison in a systematic review of factors promoting and hindering the practice of female genital mutilation/cutting (FGM/C) from the viewpoints of stakeholders residing in Western countries (Berg 2010b) In the final step of the analysis (Synthesis 5, Figure 1, Appendix 6) the results from Synthesis 1 (effectiveness of interventions) will be integrated with the results from Synthesis 4 (factors related to the continuance and discontinuance of FGM/C) in a realist synthesis model that aims to explain the extent to which intervention programs have heeded and built upon factors related to the continuance and discontinuance of FGM/C, the extent to which interventions have been provided to the most appropriate stakeholder groups, and which forces have been overlooked as critical program elements.

Realist synthesis is concerned with review of complex social interventions (Pawson, 2006). It follows the same “standard” sequence as systematic reviews, i.e. identifying the research question, searching for primary studies, quality appraisal, extracting the data, synthesizing the data, and dissemination of the results. The basic components of realist explanation are outcome patterns, generative mechanisms, and contextual conditions. The result of a realist synthesis would be described in an abstract model explaining how efficacy (outcome patterns) of an intervention varies depending on the particular configuration of its constituent mechanisms and contexts (Pawson, 2006). In our proposed synthesis (Synthesis 5, Figure 1, Appendix 6) the effect estimates extracted from the effectiveness studies will provide the outcome patterns. The data describing the interventions will be used to identify, or generate hypotheses about, the change theories underlying the interventions. The results from Synthesis 4 will provide data concerning contextual conditions (e.g. factors related to the continuance and discontinuance of FGM/C in different stakeholder groups). Recent examples of realist syntheses are Greenhalg et al. (2007) and Kane et al. (2010).