Oral Health Status of Russian Children with Unilateral Cleft Lip and Palate

Oral Health Status of Russian Children with Unilateral Cleft Lip and Palate Objective: To evaluate the oral and dental health of Russian children who underwent Frolova primary palatoplasty. Design: Eighty-nine children (62 boys and 27 girls; age range, 5 to 9 years) who had undergone repair of unilateral cleft lip and palate were included in this oral/dental evaluation. Factors such as stage of dentition; oral hygiene; carious, missing and restored teeth; and occlusion were recorded. Results: Twenty-six of the 89 patients presented with a palatal fistula. Oral hygiene of patients without a palatal fistula was significantly better than that of patients with a fistula (p.01). Forty-five percent of patients had eight or more decayed teeth. Significantly more patients with palatal fistulae had class II soft tissue facial profiles than those without palatal fistulae. Anterior crossbites were found in 48% of patients, unilateral posterior crossbites in 58%, and bilateral posterior crossbites in 12%. Conclusions: The high percentage of poor oral hygiene and dental caries found in this group of patients is likely due to the general unavailability of dental hygiene products and the high cost of these products when available. In addition, it seems there is limited understanding by parents of the importance of dental hygiene and appropriate diet in preventing dental disease. CLARA TURNER, D.M.D. ASIYA ZAGIROVA, D.M.D. LARISA FROLOVA, M.D. FRANK J. COURTS, D.D.S., PH.D. WILLIAM N. WILLIAMS, PH.D. KEY WORDS: caries, cleft lip and palate, dental health, occlusion, oral hygiene Review of the literature yields few reports on the oral hygiene of children with cleft lip and palate (CLP). Bokhout et al. (1996a, 1997) examined 151 2-year-old children (76 with oral cleft and 75 without oral cleft) for the presence or absence of plaque on the buccal and lingual surfaces of the teeth. Fiftyone percent had good oral hygiene with minimal or no dental plaque. Children with manifest caries (cavitation) had more gingival inflammation and poorer oral hygiene than children without manifest caries. Dahllöf et al. (1989) diagnosed gingival inflammation more frequently in children with clefts than in children without clefts, especially in the anterior maxillary area. They suggested that it may be more difficult for children with a cleft to achieve optimal cleaning in this region because of the anatomy of the cleft area, residual scar tissue, and immobility of the lip. Dr. Turner is Associate Professor and Dr. Courts is Associate Professor and Chairman, Department of Pediatric Dentistry, University of Florida College of Dentistry, Gainesville, Florida. Dr. Zagirova is Orthodontist and Dr. Frolova is Surgeon and Director, National Pediatric Center for Congenital Maxillofacial Pathology, Moscow, Russia. Dr. Williams is Associate Professor and Director, Craniofacial Center, University of Florida, Gainesville, Florida. Presented at the American Cleft Palate Craniofacial Association Annual Meeting, Toronto, Ontario, Canada, May 19, 1994. Submitted February 1997; Accepted November 1997. Reprint requests: Dr. Clara Turner, Associate Professor, Department of Pediatric Dentistry, University of Florida College of Dentistry, Gainesville, FL 32610. Similar to the limited literature base on oral hygiene of children with CLP, only a few studies have investigated the prevalence of caries in young children with CLP. Lauterstein and Mendelsohn (1964) compared 285 Swedish children from the county of Stockholm with CLP and 300 children without clefts from the same area (mean age, 8.5 years). These investigators found no significant difference between the two groups in diseased, missing, or filled teeth. They concluded that children with CLP from areas with fluoridated water had an average of 2.01 less decayed teeth than their peers with CLP from nonfluoridated areas. Johnson and Dixon s (1984) study of children in Cleveland, Ohio, (age range, 18 months to 4 years) reported more carious lesions in the incisors of children with CLP than in children with other craniofacial defects, such as cranial synostoses or hemifacial microsomia. Dahllöf et al. (1989) reported a higher prevalence and activity of caries in children with CLP compared to age- and sex-matched control subjects without clefts. Bokhout et al. (1996b) reported on the prevalence of Streptococcus mutans and lactobacilli in 18-month-old Dutch children with CLP, noting that these children have a high prevalence of colonization with these bacteria and that such early colonization presents a higher risk for caries in the primary dentition. Also of interest in this study was the association between children wearing acrylic preoperative orthopedic appliances and the presence of lactobacilli in their saliva. Bok- 489

490 Cleft Palate Craniofacial Journal, November 1998, Vol. 35 No. 6 hout et al. (1996a, 1997) also reported finding an increased prevalence of caries in 2.5-year-old children with CLP, as well as an increased incidence of dental caries in the primary dentition of children up to age 4 with CLP. The highest incidence of dental caries in the CLP group was found in teeth adjacent to the cleft and in the primary molars. The influence of the original defect of CLP and subsequent treatments, such as presurgical orthopedics and surgical procedures, as well as timing of the surgery, have been widely studied in relation to the development of occlusion. Schwartz et al. (1984), in an investigation designed to establish a method for classifying infants with CLP according to their risk for developing arch collapse and crossbites, examined 35 cases of UCLP and reported 18 (52%) with unacceptable (at least two teeth in crossbite) occlusion in the primary dentition; 18 (55%) of 33 cases had unacceptable occlusion in the early mixed dentition. Friede et al. (1993) studied 32 children with cleft palate who underwent repair by the Wardill-Kilner technique and reported maxillary dimensions to be significantly reduced, both before and after surgery, compared to children without clefts. More recently, Kramer et al. (1996) reported that soft and hard palatal closure in one stage had a significant impeding influence on posterior sagittal palatal growth compared to closure of the soft palate only and that transverse palatal growth and development in children with CLP is more dependent on the type and severity of the cleft than on the type of palatal surgery. Mars and Houston (1990) reported that Sri Lankan males with unrepaired clefts had potential for normal maxillary growth, whereas subjects who had lip repair in early infancy had relatively normal maxillary growth; however, maxillary hypoplasia was common when the palate had also been repaired early. In contrast, Filho et al. (1996) reported that differences in dentofacial morphologic characteristics in Brazilian males with UCLP seem to be influenced principally by the surgically repaired lip and not by palatal repair. In February 1993, several members of the University of Florida Craniofacial Center (UFCFC) were invited to visit the National Pediatric Center for Congenital Maxillofacial Pathology (NPC) in Moscow, Russia. The primary objective of this visit was to evaluate velopharyngeal competency and speech proficiency in a group of Russian children who had their cleft palates repaired with the Frolova primary palatoplasty technique. The technique is described by Williams et al. (1998). The NPC staff also requested UFCFC s partnership in evaluating the oral health and occlusal conditions of these children. The results of that evaluation are presented in this report. METHODS The limited materials and equipment at NPC permitted only visual assessment of the oral and dental condition of 89 children with repaired CLP (62 boys and 27 girls, age range, 5 to 9 years). Examinations performed by UFCFC and NPC plastic surgeons determined that 26 of the 89 patients had a palatal fistula. All fistulae were in the hard palate and were of various sizes and shapes. All of the children s clefts had been surgically repaired at least 2 years before the oral/dental evaluation. The oral examinations were conducted using a dental operatory light, mirror, and tongue blades. Radiographs and explorers were not available. Information was recorded about the stage of dentition; level of oral hygiene; frequency of carious, missing, and restored teeth; occlusion; and facial profile. All dental evaluations were performed by a UFCFC pediatric dentist and an NPC orthodontist. Each dental evaluation was completed by the pediatric dentist and the orthodontist together, and consensus was reached about the data recorded for each patient. Oral Hygiene The oral hygiene of each patient was categorized as either poor, fair, good, or excellent. A condition of poor was chosen if the entire buccal surface of the most posterior mandibular molar was covered with plaque, fair if two-thirds of the surface showed plaque, good if only the gingival one-third of the tooth was covered with plaque, and excellent if no plaque was evident. Because disclosing solution was not available, it is very likely that the amount of plaque was higher than reported. Carious Teeth The carious condition of each patient was also judged on a four point scale: patients with no carious teeth were classified as caries free; those with one to three carious teeth, as having a low caries rate; four to seven carious teeth, moderate caries rate; and eight or more carious teeth, high caries rate. A carious lesion was recorded only if frank cavitation had occurred. If a tooth had a carious lesion and a restoration, it was recorded as a filled tooth. Because the examiners did not have an explorer and/or radiographs, many carious lesions may have been missed. Data were collected for carious, filled, and missing teeth; however, because examiners were unable to determine whether teeth were missing because of caries, exfoliation, or the CLP, only dft and DFT are reported. Occlusion Occlusion was evaluated in terms of the sagittal relationship by noting anterior crossbites and the occlusal relationship of the molars. For patients without first permanent molars, the second primary molars were used to assess the angle classification. The vertical relationship was evaluated by assessing the depth of the overbite, and the transverse relationship was judged by the presence or absence of unilateral or bilateral posterior crossbites. A crossbite was recorded if one or more teeth were in crossbite. Facial Profile The soft tissue facial profile was visually estimated for each patient. The profile was classified according to the method de-

Turner et al., ORAL HEALTH STATUS OF RUSSIAN CHILDREN 491 FIGURE 1 Effect of fistula on oral hygiene. scribed by Proffit and Ackerman (1986). Superficial facial features were viewed from the patients right side while the patient was sitting in a comfortable upright position. The patient was positioned to look straight ahead and to have the Frankfurt horizontal plane parallel to the floor. According to Proffit and Ackerman, a convex profile indicates a skeletal class II jaw relationship, whereas a concave profile indicates a skeletal class III relationship but does not indicate which jaw is at fault. Convexity or concavity of the facial profile was established by viewing the relationship between two lines, one line dropped from the bridge of the nose to the base of the upper lip and the second extending from the base of the upper lip to the chin. If these line segments formed a straight line, a class I classification was recorded. An angle between the lines indicating either profile convexity (upper jaw prominent relative to chin) was a class II classification, and profile concavity (upper jaw behind chin) was a class III classification. Oral Hygiene RESULTS Of the 89 patients examined, only 6% (n 5) were rated as having excellent oral hygiene, 28% (n 25) had good, 44% (n 39) had fair, and 22% (n 20) had poor. Chisquare analysis revealed that the oral hygiene of patients without a palatal fistula was significantly better (p.01) than that of patients with a fistula (Fig. 1). Dental Caries Six percent (n 5) of the 89 patients were judged to be caries free, 12% (n 11) had a low caries rate, 37% (n 33) had a moderate caries rate, and 45% (n 40) had a high caries rate (Fig. 2). The mean dft was 7.31 (SD, 1.70), and the mean DFT was 1.45 (SD, 3.69). These values are significantly higher than those reported in the United States and other parts of the world (Ripa, 1988; Kaste et al., 1996). There was no statistical difference in caries rate between patients with a fistula and those without a fistula. Occlusal Evaluation An anterior crossbite was present in 48% (n 43) of the 89 patients. There was no significant difference (p.05) in the frequency of anterior crossbite when the group with palatal fistula was compared to the group without palatal fistulae. The molar relationship for the entire group was judged as class I in 76% (n 68) of patients, class II in 7% (n 6), and class III in 17% (n 15). There was no significant difference in molar relationship (p.05) between the groups of patients with and without a palatal fistula. There were no significant difference (p.05) in the vertical relationship, as measured by incisal overbite, between the groups with and without a fistula (Table 1). Unilateral crossbites were present in 58% (n 52) of patients, and bilateral posterior crossbites were present in 12% (n 11). No signif-

492 Cleft Palate Craniofacial Journal, November 1998, Vol. 35 No. 6 FIGURE 2 Percentage of carious teeth. icant difference in posterior crossbite was found between the two groups. Facial Profile Evaluation Soft tissue facial profile for the group of 89 patients was judged as class I for 69% (n 61) of patients, class II for 11% (n 10), and class III for 20% (n 18). For patients without a palatal fistula, 76% (n 48) had a class I profile, 6% (n 4) had class II, and 18% (n 11) had class III. For the 26 patients with a palatal fistula, 50% (n 13) had a class I profile, 23% (n 6) had class II, and 27% (n 7) had class III. The group with a palatal fistula had significantly more class II profiles than the group without palatal fistulae (Fig. 3). TABLE 1 Vertical Relationship Overbite Without Fistula With Fistula Total 0% 25% 50% 75% 100% 48% (n 30) 30% (n 19) 8% (n 5) 9% (n 6) 5% (n 3) DISCUSSION The intent of this study was to determine the oral/dental condition of 89 Russian children with UCLP repaired by the Frolova primary palatoplasty technique. Sixty-six percent (n 59) were judged to have either fair or poor oral hygiene. Although communication with the Russian parents was difficult, it is known from parent interviews at UFCFC that the 58% (n 15) 34% (n 9) 4% (n 1) 4% (n 1) 0% (n 0) 51% (n 45) 31% (n 28) 7% (n 6) 8% (n 7) 3% (n 3) priority of dental care for children with CLP may be low because of the parents focus on the numerous medical procedures required to correct the birth defect during the first decade of life. In addition, parents of the Russian children reported limited availability of basic dental care products such as toothbrushes and toothpaste. The parents noted that even when available, the cost of these products was prohibitive. In addition to limited products and high costs, many families have to travel long distances to reach a dental clinic. The significantly higher incidence of poor oral hygiene observed in patients with a fistula may have been exacerbated by the drainage of nasal flora through the fistula into the oral cavity. The tenacious nature of nasal fluids may increase adherence of plaque to the teeth. There are no other known reports in the literature against which these findings can be compared or to know whether these findings were spurious or predictable. In this study, 45% of the children (n 40) had at least eight cavities. This high rate of carious lesions may also be related to diet and a lack of water fluoridation. Even though data about the childrens diets were not collected, the Russian dental and medical staff at NPC noted that Russian children typically have a high-carbohydrate diet. None of the 89 families reported having fluoridated water or using a fluoride supplement. It seems reasonable to conclude that with the poor oral hygiene findings, along with the allegedly high-carbohydrate diet and a lack of fluoridated water, a high rate of carious lesions would be expected. Of the children examined in this study, 48% had anterior crossbites, 58% had unilateral posterior crossbites, and 12% had bilateral posterior crossbites. The cause of crossbite in patients with CLP is an area of debate, with some researchers

Turner et al., ORAL HEALTH STATUS OF RUSSIAN CHILDREN 493 FIGURE 3 Effect of fistula on facial profile. supporting the theory of growth center disturbance associated with the original defect (Friede et al., 1993; Kramer et al., 1996) and others arguing that surgical treatment and/or timing inhibit growth (Mars and Houston, 1990; Filho et al., 1996). Most investigators, however, agree that a combination of the cleft along with the surgical treatment are the factors that primarily influence growth that may lead to crossbites. Sixty-nine percent of the group of 89 children was judged as having a class I facial profile, with a greater number of patients with a palatal fistula having a class II profile. That more of the children with a palatal fistula had a class II profile may be related to less tension in the palatal tissue due to relaxation of the tissue after the fistula is opened. There are no studies comparing growth outcome (soft or hard tissue) after Frolova palatoplasty with outcome after other surgical techniques. CONCLUSION This study assessed the oral and dental health of a group of 89 Russian children who were born with UCLP and had undergone palatal repair using a surgical technique developed at NPC in Moscow. Nearly one-third of the children had a palatal fistula, and these children had significantly poorer oral hygiene than those without a fistula. The results of this study also provide some insight into dental health and dental care in Russia as practiced before the collapse of the former Soviet Union. It would be interesting to investigate whether this same level of dental disease or dental care exists today, both for the general population and for individuals with CLP. REFERENCES Bokhout B, Hofman FXWM, van Limbeek J, Kramer GJC, Prahl-Andersen B. Incidence of dental caries in the primary dentition in children with a cleft lip and/or palate. Caries Res 1997;31:8 12. Bokhout B, Hofman FXWM, van Limbeek J, Kramer GJC, Prahl-Andersen B. Increased caries prevalence in 2.5-year-old children with cleft lip and/or palate. Eur J Oral Sci 1996a;104:518 522. Bokhout B, van Loveren C, Xavier WM, Buijs JF, van Limbeek J, Prahl-Andersen B. Prevalence of Streptococcus mutans and lactobacilli in 18-monthold children with cleft lip and/or palate. Cleft Palate Craniofac J 1996b;33: 424 428. Dahllöf G, Ussisoo-Joandi R, Ideberg M, Modeer T. Caries, gingivitis, and dental abnormalities in preschool children with cleft lip and/or palate. Cleft Palate J 1989;26:233 237. Filho LC, Normando ADC, DaSilva Filho OG. Isolated influences of lip and palate surgery on facial growth: comparison of operated and unoperated male adults with UCLP. Cleft Palate Craniofac J 1996;33:51 56. Friede H, Persson EC, Lilja J, Elander A, Lohmander-Agerskov A, Söderpalm E. Maxillary dental arch and occlusion in patients with repaired clefts of the secondary palate. Scand J Plast Reconstr Surg Hand Surg 1993;27:297 305. Johnson DC, Dixon M. Dental caries of primary incisors in children with cleft lip and palate. Cleft Palate J 1984;21:104 109. Kaste LM, Selwitz RH, Oldakowski RJ, Brunelle JA, Winn DM, Brown LJ. Coronal caries in the primary and permanent dentition of children and adolescents 1 17 years of age: United States, 1988 1991. J Dent Res 1996; 75:631 641. Kramer GJC, Hoeksma JA, Prahl-Andersen B. Early palatal changes after initial palatal surgery in children with cleft lip and palate. Cleft Palate Craniofac J 1996;33:104 111. Lauterstein A, Mendelsohn M. An analysis of the caries experience of 285 cleft palate children. Cleft Palate J 1964;1:314 319. Mars M, Houston WJB. A preliminary study of facial growth and morphology in unoperated male unilateral cleft lip and palate subjects over 13 years of age. Cleft Palate J 1990;227:7 10.

494 Cleft Palate Craniofacial Journal, November 1998, Vol. 35 No. 6 Proffit WR, Ackerman JL. Orthodontic diagnosis: the development of a problem list. In: Proffit WR, Fields HW, Ackerman JL, Thomas PM, Tullock JFC, eds. Contemporary Orthodontics. St. Louis: Mosby; 1986:123 167. Ripa LW. Nursing caris: a comprehensive review. Pediatr Dent 1988;10:268 282. Schwartz BH, Long RE, Smith RJ, Gipe DP. Early prediction of posterior crossbite in the complete unilateral cleft lip and palate. Cleft Palate J 1984;21: 76 81. Williams WN, Bzoch KR, Dixon-Wood V, Seagle MB, Nackashi JA, Marks RG, Frolova LY, Serova Y, Gonchakov GV, Scheslavsky S, Shmelkova T, Zagirova A. Velopharyngeal function for speech after the Frolova primary palatoplasty technique. Cleft Palate Craniofac J 1998;35:482 488.