Midwifery 29 (2013) e73–e77
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Commentary
Female genital mutilation and female genital schistosomiasis-bourouwel, the worm: Traditional belief or medical explanation for a cruel practice? ¨ Jurgen Wacker, MD, PhD (Professor)a,n, Adama Zida, PhD, MD (Professor)b, Christina Sitz, MD (Professor)a, Dagmar Schweinfurth (Medical Student)a, ¨ (Trainee in Gynecology)e, Janika Briegel (Medical Student)b, Anika Huser c Hermann Feldmeier, PhD, MD (Professor) , Joachim Richter, MD, PhD (Professor)d a
Department of Obstetrics and Gynecology Bruchsal, Teaching Hospital of the University of Heidelberg, Germany Service of Parasitology-Mycology of CHU-YO Ouagadougou, Burkina Faso Institute of Microbiology and Hygiene, Charite´ University Medicine, Berlin, Germany d Department of Hepatology, Infectiology and Tropical Medicine, University of D¨ usseldorf, Germany e Department of Obstetrics and Gynecology, Torgau, Germany b c
Introduction Female genital mutilation (FGM), defined as the partial or total removal of the external female genitalia for ritual or religious reasons, is routinely practised by ethnic groups in more than 20 countries across the North African savannah as well as in Egypt, the southern part of the Arab peninsula, Malaysia and Indonesia. The total number of women mutilated has been estimated 100– 140 million (WHO, 2008; cf. 85–115 million: Dehne et al., 1997). In Africa, three million young women are at risk to be circumcised annually (WHO, 2008). Two main types of mutilation have been described: Excision, i.e. partial or total removal of the clitoris and the labia minora, with our without excision of the labia maiora, and infibulation, i.e. narrowing of the vaginal opening, in addition to removal of the clitoris, partial or total removal of the labia minora and sections of the labia maiora, enabling the passage of menstrual blood and urine only through a small opening (WHO, 2008, 1994).
History and origins of FGM: Bourouwel Few Africanists and anthropologists have investigated the background of FGM (Assaad, 1980; El Dareer, 1982; Hosken, 1982; Dorkenoo and Elworthy, 1992; Dehne et al., 1997; Abdallah et al., 1996). Some scholars argue that FGM was first practiced in Ancient Egypt and spread from there to other areas in Africa and the Arab peninsula (e.g. Assaad, 1980), whereas others assume that it evolved independently as a puberty rite in different regions of Sub-Saharan Africa (Dorkenoo and Elworthy, 1992). So far, its provenance remains enigmatic. There is no straightforward connection between male circumcision and female excision, although both have been perceived as closely associated n
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[email protected] (J. Wacker).
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(e.g. King, 2001). In all societies where FGM is practised, male circumcision is also performed, but the male operation prevails in considerably more societies. FGM is a crime and no arguments whatsoever serve to justify the ordeal of its victims. However, in order to work towards preventing it, we consider it necessary to better understand the underlying motives of the tradition. Starting from the ethnological theory that a relation to aetiological concepts of illness can be established for most traditional remedies, our attention was drawn to explanations given by women with FGM in Burkina Faso. Explanations frequently quoted are tradition, religion, ease of birth, etc. (Wacker, 2011; Snow et al., 2001). Such explanations are in line with the concept of ‘rites de passage’. According to this theory, FGM was practised in indigenous communities of SubSaharan Africa as part of an initiation into adulthood (Dehne et al., 1997). In this context, excision would be thought to promote sexual health and fertility and to prevent disease. As the age at excision decreased, the focus would have shifted from STDs towards diseases common during childhood, such as diarrhoea (Dehne et al., 1997; Swanson, 1985). However, as very little is known about the prevalence and perception of STDs in Africa before and during the early stages of colonisation (Barton, 1991), this explanation remains unsatisfactory. Another explanation is quoted with remarkable uniformity by women from different areas and ethnic groups: The prevention of ‘bourouwel’ (‘worm’, Yagha) or similar diseases (guldjis, ‘worms’, Dori) was given as a reason for FGM by women from all ethnic groups in Burkina Faso (Fulani, Gourmantche´, Rimaibe´, Mossi: Barry, 1991; Dorkenoo and Elworthy, 1992; Dehne et al., 1997). Intriguingly, a similar aetiology is also found in Western Sudan. A midwife who also carried out FGM describes the procedure as curative of a worm disease of the external female genitalia. % According to her, the main characteristic of al-dudah, ‘the worm’, is discharge which causes the vulva to stick together. She describes a white worm jumping out when she held and cut the clitoris and vulva. Children afflicted by the worm disease would
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be ailing or not gaining weight. They are believed to acquire it while crawling on the ground, whereas others were thought to have been born with it (Epelboin and Epelboin, 1976; El Dareer, 1982). Can these beliefs be traced back to the same origin? There are several arguments in favour of this theory. Even setting aside ethnogenetic theories, a strong connection exists between the Fulani of Burkina Faso and Sudan and Egypt, the mythological homeland ‘He´li et Yˆoyo au bord du Nil’ (Mone´nembo, 2004). On the other hand, as Hosken points out, Muslims all over Africa have taken part in the Haj in the past no less than today, which promoted cultural exchange including the import of Middle Eastern practices: For example, the tradition that barbers perform excisions among the Hausa of Nigeria may well reach back to Egypt as the only place where barbers have been known to carry out surgery (El Dareer, 1982; Hosken, 1982). Thus, we believe that the ‘bourouwel’ or worm disease explanation given by the women in Yagha and elsewhere should not only be taken seriously as an aetiological attribution, but also that it may point to the very origins of the practice. It should be kept in mind that the etiopathogenetic perspective of modern medicine does not apply without restrictions in cultures with traditional concepts of illness. ‘Worm disease’ may well be defined by clinical features (oedema, ulceration, systemic symptoms), as various types of maggots or ‘worms’ appear frequently in ulcerous tissue and infected wounds, while only occasionally actual parasites are seen emerging from swollen tissue (e.g. Dracunculus medinensis). Are there any diseases with characteristic lesions in the external female (and also male) genitalia which can be acquired at an early age? Different sets of evidence point to female genital schistosomiasis (FGS) as a likely candidate: The geographical distribution of FGM and Schistosoma haematobium in Africa, ancient sources from Egypt and the Graeco-Roman world, and clinical observations.
FGS: clinical characteristics A review article by Gabriele Poggensee and Hermann Feldmeier (2001) underlines the clinical importance of FGS. FGS is defined as the presence of ova and/or a characteristic pathology in the internal or external reproductive organs. Genital schistosomiasis as a clinical feature of S. haematobium infection was already described by Maden (1899). In 1949, Charlewood came to the conclusion: ‘that practically all gynaecologists who have been practising in Johannesburg or Durban for any length of time, have encountered gynaecological manifestations of Bilharzia’ (Charlewood et al., 1949). Later Gentilini stated that ‘in S. haematobium infection female genital schistosomiasis is clinically a frequent and anatomically a constant condition’ (Gentilini et al., 1986). It is therefore surprising that until recently the scientific literature on this disease offered more questions than answers. Thus, it seems appropriate at this point to give a brief summary of the effects of schistosomiasis on the external and internal female genitalia (Poggensee and Feldmeier, 2001): When after maturation in the liver, adult schistosomes leave the portal vein against the blood stream, the first obstacle to overcome is the anorectal plexus. Once this plexus is passed, worms gain easy access to the perivesical plexus, as well as to the plexus uterovaginalis, either by traversing the rectovaginal septum or through vascular links between the bladder and the reproductive organs. The abundant anastomoses between the different venous plexus of the small pelvis, the veins of which are almost without valves and allow blood to flow in either direction, offer a network of routes for the migration of worms to any genital organ.
There is circumstantial evidence that the deposition of eggs in the ovaries and the tubes leads to sterility, higher risk for EUP and enlargement of the ovaries. There is a variety of macro- and microscopic alterations induced by FGS in the lower genital tract. S. haematobium ova and adult worms have been found in endometrial tissue and in diagnostic or therapeutic curettage material. In a population based study in Madagascar, Leutscher et al. (1998) have shown that spontaneous abortions are more frequent in a S. haematobium endemic village compared to a non-endemic village. Symptoms associated with cervical schistosomiasis are dysmenorrhoea, menorrhagia, leucorrhoea, discharge, lower abdominal pain, post-coital bleeding, intermenstrual bleeding and dyspareunia. For the diagnosis of cervical FGS, a biopsy has to be taken. The clinical presentation of cervical lesions varies. Lesions may be fibroid-like, polypoid, ulcerative or have the aspect of coarse grained sandpaper (Berry, 1966). In tropical countries the macroscopic changes are of great importance for diagnosis. Rectovaginal and vesicovaginal fistulas have been found to be an extremely debilitating sequel of vaginal schistosomiasis (Diouf et al., 1973). The lesions of vulva and clitoris vary in size and presentation. Pruritus, progressive and relapsing swelling, painful or painless ulcerations, fibromata and a hypertrophic clitoris with eroded granular surface have been described (Crump et al., 2000; Poggensee and Feldmeier, 2001; Yirenya-Tawiah et al., 2011). In a few women, the development of lesions has been monitored. Genital schistosomiasis started with an irritation of the epithelium, followed by oedema and hyperaemia. Later, small nodules developed under the skin and papillomatous lesions appeared forming masses resembling condylomata (Desmond et al., 1994). Schistosomiasis induced vulvar growth has been observed in girls as young as three years, and vaginal lesions are known to occur before puberty (Al-Adnani and Saleh, 1982; Hegertun et al., in press). Furthermore, FGS interacts with other infectious diseases, notably sexually transmitted diseases (STDs). On the basis of clinical findings and available pathophysiological as well as immunological data it is conceivable that FGS of the cervix not only facilitates the infection with agents of STDs, but also alters the natural history of such infections. Two infectious agents are of particular concern: the Human Immunodeficiency Virus (HIV) and the oncogenic Human Papilloma Viruses (HPV). Besides Herpes simplex virus (HSV-2), HPV is the most prevalent sexually transmitted infectious agent in women and without any doubt, HPV plays a central role in the aetiology of cervical neoplasia (zur Hausen et al., 1981; Bosch et al., 1995). Therefore any evidence pointing to an association between FGS and HPV warrants careful consideration. Several reasons give credit to the hypothesis that cervical schistosomiasis may play a pivotal role in HPV induced cervical cancer. Firstly, there is convincing evidence that FGS develops during puberty (Poggensee and Feldmeier, 2001). Hence, egg-induced lesions, particularly at the cervix, may be present at the very beginning of sexual activity. Thinning, erosions and ulceration of the cervix epithelium due to egg associated pathology, should ease the penetration and implantation of HPV. In this way, FGS could facilitate the infection with HPV at very young age, which in turn would favour the development of cervical malignancy at a younger age than normal. In fact, in Sub-Saharan Africa, women with cervical cancer are surprisingly young. In a study in Tanzania Moubayed et al. found that 18% of women with FGS and cervical cancer were younger than 25 years. By contrast, in women with cervical cancer and absence of genital schistosomiasis only 5% were in this age group (Moubayed et al., 1955).
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The observation that in HIV-infected patients, schistosomal coinfection accelerates HIV disease progression and facilitates viral transmission to sexual partners suggests that urogenital schistosomiasis is a public health issue that has so far not received due attention (Mbabazi et al., 2011).
Distribution of FGM and Schistosoma haematobium As shown on the maps below, the geographical distribution of FGM and S. haematobium, which primarily causes urogenital schistosomiasis, is strikingly similar. The difference in distribution in the Eastern part of South Africa may be due to the migration of Bantus from the Kalahari Desert, where neither FGM was practised nor FGS existed, to South Africa. The observation that FGM is not performed in Madagascar, where FGS is documented, may be explained by the fact that its first settlers originally immigrated from Asia (Figs. 1 and 2).
Ancient sources on FGM and FGS In Egypt, high prevalence of S. haematobium infections is also documented for ancient times. Ova have been found in mummies from the 20th dynasty (1250–1000 BCE: Duke, 2008). Probably, the prevalence increased as irrigation agriculture along the Nile was established. There is evidence that suggests that schistosomiasis was perceived as a worm disease with urogenital manifestations. It has been connected to the so-called ‘aaa disease’ mentioned abundantly in medical papyri, a worm disease affecting the stomach (e.g. Papyrus Ebers (16th century BCE) 62, 19.11–19.19). With the word ‘aaa’, a discharging phallus is associated as a determinative. At first glance, this suggests haematuria as a symptom of urogenital schistosomiasis. While this connection can no longer be regarded as simple and straightforward, it still remains probable that schistosomiasis was indeed the ‘aaa disease’, as the main argument against this is based on the assertion that haematuria may not have been regarded as pathologic (Duke, 2008). This seems questionable given the debilitating complications of urogenital schistosomiasis which accompany haematuria. The earliest source locating the origins of FGM in Ancient Egypt is Strabo, a Greek geographer (about 64 BCE to 24 CE). The indirect nature of the evidence from Ancient Egypt itself (e.g. an ‘uncircumcised girl’ is mentioned in an inscription, 19th to 18th century BCE) may be due to a traditionally more secretive character of FGM, possibly as a religious rite in the context of temple life (King, 2001). By comparison, male circumcision is documented from the Old Kingdom onwards (27th to 22nd century BCE). Examination of mummies mostly relies on inspection only and has therefore been inconclusive so far, as certain mummification techniques involved fixation of the labia maiora in a way that resembled infibulation, so that no conclusions can be drawn as to the state of the female external genitalia before mummification (King, 2001). The ascription to Ancient Egypt continued in the Graeco-Roman and Arab medical tradition. Several important medical textbooks from the fifth to ninth century CE which probably draw on lost writings by Soranus (first to second century CE) offer detailed accounts of FGM. They contain instructions on how to perform clitoridectomy in women with hypertrophy of the clitoris, a condition that was associated with discomfort and shame for the patients. The procedure is recommended to take place before marriage. By only removing part of the clitoris, fistulas were thought to be prevented. These sources quote medical reasons for FGM, thus firmly locating the procedure in rational Graeco-Roman medicine which influenced clinical practice in Byzantine and Arab cultures. What remains
Fig. 1. Geographic distribution of FGM in Africa (WHO, 2008). Prevalence of FGM. Red: 75.1% or more, burgundy: 50.1–75%; orange: 25.1–50%; mauve: 10.1–25%; pink: o 10%, grey: missing data, FGM not widely practiced. (For interpretation of the references to colour in this figure legend, the reader is referred to the web version of this article.)
puzzling is the relative abundance of the sources on a condition that is mostly seen in a rare disease today (congenital adrenal hyperplasia). Varying perceptions of the normal size of the clitoris are a possible explanation, but another one may be that hypertrophy of the clitoris as well as pseudohypertrophy (i.e. Oedema and tissue hypertrophy of the praeputium clitoridis and labia minora) do occur in FGS and may thus have been more common. To sum up, the origins of FGM as a medical procedure are connected to Ancient Egypt, where the prevalence of FGS was as high or even higher than today, and schistosomiasis may have been perceived as a ‘worm disease’ with a urogenital aspect, which is evocative of the ‘bourouwel’ aetiology. It may be speculated that FGM was practiced to treat symptoms of FGS. Thus, the ancient evidence may argue in favour of a connection between FGS and the origins of FGM.
FGM and FGS today Is there a connection between FGS and FGM? The first author (JW), together with colleagues in countries with endemic FGS infections, has made the observation that clinical symptoms of this disease rarely occur in countries where FGM is performed regularly (Wacker et al. 2005, 2008; Wacker, 2007). The lack of evidence of a clinical manifestation of FGS in the introitus, the perineum and the external female genitalia in general could perhaps be explained by the scar remaining from the excision. These scars quite likely prevent schistosoma eggs from penetrating the outer genitalia. Another observation pertaining to this issue was made by the last author (JR) in Malawi. Here, FGM is not widespread. However, women with FGS call upon traditional healers to surgically
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¨ Fig. 2. Distribution of Schistosoma haematobium, S. japonicum and S. mekongi (from Tropenmedizin in Klinik und Praxis, Thomas Loscher und Gerd – Dieter Burchard; ¨ Thieme – Verlag Stuttgart, 2010), Loscher and Burchard (2010).
remove the papulous lesions of the outer genitalia. This practice may be seen as a residue of early cutting practice. In short, can it be argued that the tradition of FGM originated from the observation that women with scarring of the external genitalia were less likely to develop the visible lesions characteristic of FGS? This theory may be supported by the ‘bourouwel’ or worm disease concept frequently quoted as an explanation for FGM. Features of FGS such as tissue hypertrophy, lesions and discharge may have been interpreted as arising from worm infestation. If this theory is supported by further evidence, it may be possible to bridge the gap between traditional beliefs and modern medicine by addressing a common aetiology. While we are well aware of the speculative nature of these suggestions, we should like to argue that our findings merit further consideration. In an issue as controversial as FGM, any chance should be taken to further our understanding of traditional practice. If nothing else, the coexistence of FGM and FGS constitutes a significant bias in research which deserves closer attention. For example, a significant percentage of schistosomiasis-free girls and women with FGM (infibulation type) show haematuria and leucocyturia which has led to systematic errors in schistosomiasis research (Feldmeier et al., 1993, 1995). Descriptions of typical lesions of FGS are also biased by the fact that the external genitalia of many female patients in endemic areas have been altered (Laven et al., 1998). We would therefore like to ask you about your experience as midwives and doctors: 1. Have you ever seen any visible signs of FGS, such as ulcerations, granuloma, tumours or vesicorectal fistulas not connected to birth complications in women with FGM? 2. In your experience, is there an increasing number of women with visible FGS in areas where FGM is now performed less frequently? 3. Would you be willing to take part in a corresponding investigation? Note It is not the aim of this paper to trivialise or excuse the cruelty of the ritual of female genital cutting. Mutilation of young women and girls must be eliminated all over the world. The authors have published abundantly on this subject (see bibliography). In the novel ‘Sisters of Isaac’ (Isaaks Schwestern, 2011), I have described
the ordeal of women affected by FGM. However, the connection between FGS and FGM may help to illuminate the beginnings of FGM from the point of view of medical history, taking into account its probable roots in Ancient Egypt. It may emerge that the wise midwives of Ancient Egypt meant well when they initiated a cruel practice.
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