Infant oral mutilation (IOM) is a practice that differs from the more commonly recognized practices of culturally based dental modification in several important aspects: it is performed for perceived medical benefit, it is performed on infants who are incapable of consenting to this practice, and it can have disastrous health consequences. In 1969, Pindborg⁹ first documented the practice of removing the mandibular primary canine tooth follicles in infants in the Acholi district of Uganda, which was being carried out under the belief that these follicles were responsible for febrile illnesses. In his article, Pindborg⁹ acknowledged two previous reports. One, from 1882,¹⁰ described a Kenyan tribe that extracted the primary mandibular incisors and then inserted the tip of a hot knife into the extraction socket to destroy the follicle of the permanent tooth. A second, from 1932,¹¹ described a Sudanese tribe that practiced removal of the primary canine tooth buds within the first month of life followed by extraction of the permanent mandibular teeth after their eruption. However, although these reports preceded his, Pindborg⁹ was the first to link this practice to the belief that it would cure or prevent infant febrile illnesses. In his study, he noted evidence of primary canine tooth extraction in close to 50 percent of the children aged 4 years and younger in the Ugandan tribe he studied.

In practice, IOM is a crude form of dental ablation or "germectomy" in which the healer removes the developing primary canine tooth follicles from below the alveolar process in infants aged 1 month to 1 year. Traditional healers or other village elders such as grandmothers typically use unsterilized tools to excise these tooth follicles without providing local anesthetic. In addition to knives and fingernails, the healers’ "tools" may include bicycle spokes, rusty nails or wires.¹²

The terms "false teeth" or "nylon teeth," as used by rural villagers, describe the developing protuberances on the alveolar process and the underlying tooth follicles, thought to resemble worms, that traditional folklore suggests are the cause of high temperature, vomiting, loss of appetite and diarrhea in young children. These tooth follicles are believed to represent "false" teeth because they are unmineralized soft-tissue masses occurring in place of the "real" teeth. In areas in which the practice of IOM is endemic, severe diarrhea is one of the main causes of the high infant mortality rate,¹³ so prevention or treatment of these illnesses takes on great significance.

Forty-five¹⁴ to 85 percent¹⁵ of mothers in some rural areas of Africa believe that removing a child’s tooth buds is an effective treatment for severe diarrhea. This belief is reinforced by the fact that parents first notice these developing tooth buds when infants are being weaned from breast-feeding, a period during which infants are most likely to have their first bouts of enteritis. In addition, dehydration caused by diarrhea may desiccate the oral mucosa, potentially making the alveolar protuberance associated with the developing canine tooth follicles appear even more prominent. Paradoxically, the fact that many children become seriously ill and even die after undergoing this procedure may reinforce further how "dangerous" these tooth buds are.

This practice is believed to be of relatively recent onset. Graham and colleagues¹⁶ suggested that this practice may be an adaptation by traditional village healers of a procedure practiced by colonial dentists in the early 1900s in which they incised the alveolar mucosa over erupting teeth to decrease the pain associated with tooth eruption. IOM is believed to have originated in northern Uganda, although the practice has spread to neighboring countries during the last 30 to 40 years.¹⁷ The political unrest and war during Idi Amin’s term as ruler of Uganda in the 1970s may have spearheaded the spread of this practice because many of the fighters involved in these conflicts came from areas of northern Uganda, where the practice of IOM was well-entrenched.¹⁸

The diverse terms used to describe the practice of IOM mirror the numerous geographical and tribal variations. "Ebinyo" (or "ebino") translates as "false teeth." Variations include "killer teeth" (based on the belief that the child will die if they are not removed) and "vinyl teeth."¹⁹ Less common forms of IOM include incising over the unerupted primary canine tooth protuberance to elicit bleeding ("haifat"²⁰) and uvulectomy, involving the crude amputation of the uvula ostensibly to treat vomiting or upper respiratory symptoms.²¹

Although IOM is believed to be extremely rare in developed countries, investigators have documented the sporadic continuation of this practice among recent émigrés in Western countries. In 2000, Rodd and Davidson²² suggested that the practice of canine tooth enucleation was occurring in children born in England of Somali parents, possibly being performed during visits to Somalia. Similarly, Holan and Mamber²³ reported that parents of Ethiopian children who had emigrated to Israel in the early 1990s suggested that the practice was occurring in their community.

There have been no documented reports of IOM’s being performed in the United States and only one published report documenting the dental sequelae from IOM among children residing in the United States. Graham and colleagues¹⁶ described three Somali children in Washington state with dental malformations secondary to IOM. Researchers have documented similar cases among Ugandan children living in the United Kingdom²⁴ and an Ethiopian girl living in Sweden.²⁵

	CASE REPORT

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Our report involves a family of five well-nourished and healthy Sudanese siblings, ranging in age from 2.5 to 10 years, who had emigrated recently from a refugee camp. They came to the pediatric dental clinic at Creighton University (Omaha, Neb.) with multiple intraoral effects attributed to prior IOM. All of the children had been cooperative during dental treatment.

Child 1, the youngest boy at 2.5 years of age, was missing all four primary canine teeth (Figure 1) at his first dental examination. Scar tissue was evident on the mucosa overlying the alveolar ridges where the primary canine tooth follicles presumably had been removed. This scar tissue was most prominent in the area of the left maxillary canine tooth. In the mandibular right canine tooth area, a slight protuberance of the labial alveolar process was evident. Although we ordered radiographic studies to determine the fate of the missing primary canine teeth, we were unable to obtain enough cooperation from this patient to obtain diagnostic radiographs.

View larger version (108K): [in this window] [in a new window]   Figure 1. Clinical photo of child 1, the youngest of five siblings who had immigrated to the United States from a rural village in Sudan. This 2.5-year-old boy was missing all four primary canine teeth. Scar tissue, most prominent in the area of the left premaxilla, was evident in the mucosa overlying the canine alveolar ridges from where the primary canine tooth follicles had been extracted.

After we concluded that child 1 had undergone IOM, we retrospectively examined the dental records of this patient’s four siblings, who had been treated over a six- to 12-month period before child 1’s first dental examination. Although we had not considered the possibility of IOM when the four siblings came for initial dental treatment, on retrospective examination we noted that all had dental side effects consistent with IOM.

Child 2, a 4.5-year-old girl, was missing all four primary canine teeth (Figures 2A and 2B). There was obvious loss of alveolar bone height at the sites of the missing primary canine teeth, presumably secondary to a combination of surgical trauma and failure of alveolar bone development because of the missing primary canine teeth. We noted normally developing permanent canine teeth on the panoramic radiograph (Figure 2C).

View larger version (80K): [in this window] [in a new window]   Figure 2. Clinical and radiographic presentations of child 2, a 4.5-year-old girl who was missing all four primary canine teeth. A. Right lateral view. B. Left lateral view. C. Panoramic radiograph depicting normally developing permanent canine teeth.

View larger version (67K): [in this window] [in a new window]   Figure 3. Radiographs of child 3, a 6-year-old girl. The treatment plan called for restoring the carious lesions (A, B) and extracting the malformed left maxillary primary canine tooth. C. Initial radiographs also showed that the mandibular primary canine teeth were missing (D), the mandibular permanent lateral incisors were erupting distal to the retained primary lateral incisors (D) and the right and left maxillary primary canine teeth were present but abnormally small (C). A panoramic radiograph taken at a follow-up appointment confirmed that all four permanent canine teeth were developing normally (E). The right maxillary primary canine tooth was present.

View larger version (37K): [in this window] [in a new window]   Figure 4. Panoramic radiograph of child 4, a 9-year-old boy, who was missing the maxillary right primary canine tooth. He also had three malformed primary canine teeth, presumably the result of damage to the developing dental follicle.

View larger version (93K): [in this window] [in a new window]   Figure 5. A. Panoramic radiograph of child 5, a 10-year-old boy, who had a rotated, severely deformed right mandibular primary canine tooth with a well-defined periapical radiolucency at the apex. The authors subsequently extracted the tooth. B. The permanent right mandibular lateral incisor also was missing. C and D. The extracted malformed right mandibular primary canine tooth shown in lingual (C) and lateral (D) views.

	DISCUSSION

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The problem of intentional dental mutilation is compounded in many developing African countries by a lack of belief in Western medical practices and inadequate access to trained dentists. For example, the World Health Organization²⁷ estimates that in Ethiopia, only 93 dentists serve a population of more than 77 million people. As a result, many parents receive their counseling on dental and medical care from tribal elders or traditional healers.

Although the true prevalence of this practice is not known, in contrast to Pindborg’s⁹ belief that "since the native way of life is changing rapidly, it will not be possible much longer to observe dental alterations of this sort," it appears that this practice actually may have spread across a much wider area of Africa. In one study involving 398 Sudanese children aged 4 to 8 years living in Khartoum, those in the lowest socioeconomic strata were three times more likely to have undergone IOM than were those in the highest socioeconomic group.²⁰ In another cross-sectional study at a hospital in northern Uganda, 30 percent of children aged 0 to 4 years had undergone IOM.²⁸ These numbers are comparable with those in other studies, which have shown prevalence ranging from 15²⁹ to 80 percent or more,³⁰ depending on geographic location. Although most reports indicate that the mandibular primary canine tooth follicles are removed preferentially, researchers also have documented geographic and tribal preferences in which the maxillary canine tooth follicles are removed.³¹

We can divide the adverse effects from these practices into immediate, short-term complications occurring soon after the procedure and long-term psychological and dental side effects. In the period immediately after the procedure, the most common risks include excessive bleeding, infection, osteomyelitis of the jaws, noma, tetanus, meningitis, aspiration bronchopneumonia, transmission of infectious diseases (including HIV and hepatitis) and death.³² The significant morbidity and mortality associated with these practices is well-documented and likely is made worse by the fact that healers perform the procedure on infants who already are acutely ill from severe diarrheal illness. Mosha³³ documented more than 120 patients treated for complications and 10 deaths resulting from IOM at a hospital in Tanzania. In another study conducted in northern Uganda, researchers found that approximately 2 percent of all pediatric admissions between 1992 and 1998 were related to complications arising from IOM, representing the 10th leading cause of admission.³⁴ The most common complications in this study were septicemia, anemia, meningitis, osteomyelitis and tetanus. More than 20 percent of these patients had fatal outcomes. The average age of the admitted patients was 5 months.

Assessing the psychological effects of the practice of IOM can be difficult; however, more tangible long-term physical adverse effects include reduced weight gain during the first year of life³⁵ and dentoalveolar sequelae ranging from hypoplasia of the crown to complete tooth agenesis because of damage to the dental lamina of the underlying succedaneous tooth, damage to or ablation of neighboring teeth and dentofacial malocclusions.²³ Pindborg⁹ described effects on the permanent mandibular canine teeth ranging from peg- or shovel-shaped malformations of the crown to partially split teeth and severe crown malformations. Pindborg⁹ also documented transposition of the lateral incisor and canine teeth, early eruption of the permanent canine teeth and the presence of unerupted, impacted or missing permanent canine teeth. In at least one case, investigators documented the development of odontomalike tooth structures after IOM.¹⁶ Although rare, this possibility is not unexpected, since researchers have associated trauma to the primary tooth with the development of odontomalike structures in clinical case reports.³⁶ Levy³⁷ demonstrated that inserting a needle into the developing molar follicle of a rat can induce alterations to the developing tooth that are histologically similar to that of a compound odontoma. However, the histologic presentation of these malformed teeth is more a reflection of disordered tooth development than representative of the formation of a true odontoma, since the total tooth count is unchanged in these cases.

Although the adverse effects associated with the practice of IOM are well-recognized by health care providers in Africa, knowledge about this practice is not widespread among dental professionals in North America. In many cases, dental practitioners treating refugees from Africa with missing or malformed permanent anterior teeth may assume that these teeth were extracted or damaged as the result of a tribal custom performed on the permanent teeth, and they may overlook the possibility that the observed effects are the result of incidental damage to the succedaneous tooth follicles following extirpation of the primary canine tooth follicle. Because dental practitioners may encounter patients who have been subjected to these practices, it is important that they be aware of the social factors behind this practice, as well as be able to recognize its dental and psychological sequelae.

Variations of the practice of IOM include rubbing herbs over the alveolar protuberance associated with the developing canine tooth follicle³⁸ and making an incision in the mucosa over the alveolar process adjacent to the unerupted primary canine tooth protuberance. Researchers have associated this latter practice with the development of enamel defects in the primary canine teeth.²⁰ These defects, characterized by enamel hypoplasia, hypomineralization or both, typically are located on the middle one-third of the buccal surface. These enamel defects were 3.5 times more prevalent on the buccal surface of primary canine teeth in patients who had undergone this form of mutilation.²⁰ Overall, 28 percent of children who had undergone this form of IOM showed evidence of isolated enamel defects of the primary canine teeth, compared with 8 percent of children who had no apparent history of IOM. However, attributing enamel defects in the primary teeth to IOM-like practices is complicated by the belief that most children who underwent this practice did so as a result of systemic illness such as fever, which alone can cause enamel defects.

Representatives of health organizations are attempting to reduce the incidence of IOM through a multipronged approach directed at rural villagers and traditional healers. They are aiming to explain the adverse effects of this practice, provide counseling regarding the use of intensive rehydration in the management of childhood diarrheal illnesses and reduce the incidence of waterborne illnesses by providing clean water. However, until the scourge of childhood illness can be reduced in rural areas of Africa, it is unlikely that these practices will be eradicated.

	CONCLUSIONS

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Cultural practices around the world continue to challenge the conventional standards of health. Many of these practices act as barriers to reducing childhood mortality and disease. Although in many cases concerted attempts to diminish these practices through education are successful, some evidence suggests that practices such as IOM actually may be increasing. Although there is no reason to believe that these practices are occurring in North America, it is reasonable to expect that as more people from developing countries immigrate to the United States and Canada, dentists and allied dental personnel may observe the long-term effects of these practices. Findings that should alert the clinician to a possible history of IOM include the absence of primary and succedaneous canine teeth or lateral incisors and malformed permanent canine teeth. Enamel defects on the permanent canine teeth, although a potential consequence of IOM, are not sufficiently uncommon in the general population to be considered strong evidence of IOM.