PEDIATRIC DENTAL JOURNAL 14(1): 141–146, 2004 141

Case Report Clinical and microbiological evaluations of gingival fibromatosis in children: Report of two cases

Kazuhiko Nakano, Naoko Nishiyama, Kiyoko Tamura, Hidekazu Sasaki and Takashi Ooshima

Department of Pediatric Dentistry, Osaka University Graduate School of Dentistry 1-8 Yamada-oka, Suita, Osaka 565-0871, JAPAN

Abstract Gingival fibromatosis is a rare clinical finding of non-inflammatory Key words gingival overgrowth. Here we present 2 cases of children with gingival Children, fibromatosis and provide a longitudinal analysis of the correlation between Gingival fibromatosis, the presence of periodontal and clinical condition. The first case, a Periodontal bacteria, boy treated from age 2 to 6, showed periodontitis in the lower anterior region, Polymerase chain reaction which recovered to a good periodontal condition by a professional therapeutic approach. The second case is a girl, treated for 2 years from age 10, who presented with extremely enlarged gingiva and severe , and a was carried out to eliminate the accumulation. In both cases, periodontal bacteria were assessed periodically using PCR (Polymerase Chain Reaction) method, and the results compared with the clinical conditions. In both cases, increased numbers of periodontal bacteria were detected during the bad gingival condition period. In addition, nigrescens and Prevotella intermedia were especially detected. Clinical condition seems to be influenced by the number of the periodontal bacteria present, which indicates that an effective monitoring of such bacteria would be beneficial for not only evaluating the current periodontal condition, but also for making an appropriate prognosis of a lesion.

Introduction of the maxillary tuberosities. Gingival fibromatosis causes esthetic and functional problems such as Gingival fibromatosis is an uncommon gingival malposition of teeth, prolonged retention of primary overgrowth associated with increased levels of teeth, and delayed eruption of permanent successors. mature collagen1). The enlarged gingivae associated In addition, hyperplastic lesions produce conditions with this condition are usually normal in color, firm favorable for the accumulation of dental plaque, in consistency, painless, and occasionally nodular causing the secondary inflammatory changes of with little inflammation. Some patients have a gingivitis, though alveolar bone is not usually hereditary predisposition, whereas others have no affected4). apparent genetic link2). usually In our previous study5), we reported the detection begins with the eruption of primary or permanent of 10 putative periodontopathic bacteria found in dentition. However, some cases have been reported subgingival plaque from 144 healthy children (2–13 to be present at birth while others have not been years old, 12 subjects for each year of age) using a identified until adult life3). Gingival enlargement polymerase chain reaction (PCR) method. Capno- may be generalized or localized to a specific site, cytophaga ochracea, sputigena, though it is frequently found at the palatal surface Campylobacter rectus, and Actinobacillus actino- mycetemcomitans were found in approximately 50% Received on June 20, 2003 of those plaque samples from all ages. Prevotella Accepted on November 14, 2003 nigrescens was also frequently detected as aging,

141 142 Nakano, K., Nishiyama, N., Tamura, K. et al. a) a)

b)

b) c)

Fig. 1 Oral photographs of case 1 Fig. 2 Periapical radiographs of case 1 a) 2 years 1 month b) 4 years 11 months c) 6 years 8 months a) 3 years 1 month b) 4 years 11 months

whereas forsythus and Prevotella inter- cleaning was performed with a shorter interval media were detected less frequently, and Porphyro- between sessions. An excisional biopsy was performed monas gingivalis and denticola were not under infiltration anesthesia at the age of 4 years 1 found. The purpose of the present study is to describe month, which was submitted to routine histopatho- two cases of gingival fibromatosis identified in a logical examination. The histopathological features 2-year-old boy and a 10-year-old girl, as well as demonstrated the existence of a large number of report on the longitudinal examination of clinical irregular collagen fibers under the extended rete peg, conditions and periodontopathic bacteria using a which was diagnosed as a gingival fibromatosis. At PCR method. 4 years 9 months, dental plaque was found moderately accumulated in the region of the lower anterior teeth, Case Reports and tooth brushing instruction was given again. Two months later, at 4 years 11 months, his Case 1 was improved and the swelling had changed into A 2-year-1-month-old boy, who had a cleft lip and a non-inflammatory hyperplastic lesion (Fig. 1b), palate, was referred to the Pedodontic Clinic of Osaka however, alveolar bone losses were still found around University Dental Hospital for oral prophylaxis. the primary lower lateral incisors and canines (Fig. There was no dental caries detected, but non- 2b). The degree of swelling reduced as the patient inflammatory gingival overgrowth was found at grew older. At 5 years 11 months, oral hygiene was the labial site of the lower anterior region (Fig. 1a). judged to be fair, and the gingiva still showed non- Professional tooth cleaning was performed and inflammatory swelling. When the patient was 6 proper tooth brushing instruction was given. At 2 years 7 months old, the lower primary incisors had years 4 months of age, the swelling had increased, exfoliated and soon thereafter permanent successor while slight gingival inflammation and mobility of teeth emerged into the oral cavity (Fig. 1c). the teeth in the region were identified. At 3 years 10 months, the gingival condition had worsened and Case 2 periapical radiographs showed alveolar bone loss A 10-year-2-month-old girl was referred to our clinic around the primary lower lateral incisors and canines with the chief complaint of swollen gingivae in the (Fig. 2a). As a result, routine professional tooth incisor regions that caused an esthetic problem. A GINGIVAL FIBROMATOSIS IN CHILDREN 143 a) plaque index6), and gingival index7). Pocket depth was measured to the nearest millimeter at 6 points around the circumference of each tooth (mesio-, mid-, and disto-buccal; and disto-, mid-, and mesio- lingual) from the to the deepest probeable point, using a round ended probe tip 0.4 mm in diameter. was scored immediate bleeding on probing, or (ם) ;as follows no bleeding. was scored as (מ) b) follows; (2) moderate mobility (1–2 mm) in a bucco- lingual direction and (1) slight mobility (0.2–1 mm) in a bucco-lingual direction, or (0) physiological mobility within 0.2 mm. Pus discharge was scored as no (מ) spontaneous pus discharge, or (ם) ;follows pus discharge.

Microbiological examinations Microbiological examinations were performed Fig. 3 Oral photographs of case 2 multiple times in both cases using a PCR assay. a) First visit at 10 years 2 months Subgingival plaque was collected from the lower b) Three months after gingivectomy at 11 years 2 months left primary canine in case 1 and the upper right permanent central incisor in case 2 with sterile Gracy curettes, and the samples were suspended clinical examination revealed severe generalized in 1 ml of sterile saline, after gently removing gingival overgrowth involving both the upper and supragingival plaque. Plaque samples in 1 ml of lower arches, which covered almost half of the saline were centrifuged at 15,000 rpm for 5 minutes tooth crown (Fig. 3a). She had no other medical to pellet the bacterial cells. Bacterial genomic DNA disorders and none of the members of her family had was then isolated from the pellet using a DNA gingivae problems. Dental plaque was found heavily isolation kit (Puregene, Gentra Systems, Minneapolis, accumulated in the region of the posterior teeth, MN, USA). which led us to initiate professional oral cleaning Expectorated whole saliva was collected from and tooth brushing instruction. At 10 years 5 months the patients and kept in sterile plastic tubes. The of age, her oral hygiene was still poor and at 10 years samples were mixed with Chelex 100 (Bio-Rad 8 months conventional gingivectomy was scheduled, Laboratories, Hercules, CA, USA) and incubated at as the gingiva had become inflamed. The procedure 56°C for 30 minutes, followed by boiling at 100°C was carried out under infiltration anesthesia at the for 10 minutes, as described by Mättö et al.8) Saliva age of 10 years 11 months and the histopathological samples were then centrifuged at 15,000 rpm for 20 diagnosis was gingival fibromatosis. When she was minutes and the supernatants were used as template 11 years 2 months old, the fibrous swelling had for PCR. completely disappeared and the periodontal condition Gram-negative anaerobic bacterial species, of the affected regions was shown to be almost P. gingivalis, T. denticola, C. ochracea, C. sputigena, normal (Fig. 3b). However, at 11 years 6 months of P. intermedia, P. nigrescens, C. rectus, A. actinomy- age, her oral hygiene was found to be poor with cetemcomitans, B. forsythus, and Eikenella corrodens the dental plaque accumulated in the region of the were selected, based on a list of putative peri- posterior teeth. Throughout the observation period, odontopathogens. The species-specific PCR primers alveolar bone loss was not identified by radiographic selected for use in the present study have been examination in any regions. described previously9). A ubiquitous primer set that matches almost all bacterial 16S rRNA genes was Clinical examinations used as a positive control. PCR amplification was Clinical parameters measured were probing depth, performed in a reaction mixture containing PCR bleeding on probing, pus discharge, tooth mobility, beads (Ready-To-Go; Amersham Pharmacia Biotech, 144 Nakano, K., Nishiyama, N., Tamura, K. et al.

Table 1 Longitudinal changes in periodontal bacteria in subgingival plaque and saliva, and periodontal conditions in the area of the lesion near the lower left primary canine 3Y 4Y 5Y 6Y 4M 9M 11M 6M Gram-negative anaerobic bacteria (מ)מ (מ)מ (מ)ממ P. gingivalis (מ)מ (מ)מ (מ)ממ T. denticola (מ)מ (ם)ם (ם)מם C. ochracea (ם)ם (ם)ם (ם)םם C. sputigena (מ)מ (מ)מ (מ)ממ P. intermedia (מ)מ (מ)מ (מ)מם P. nigrescens (ם)ם (מ)מ (ם)םם A. actinomycetemcomitans (מ)מ (מ)מ (מ)ממ B. forsythus (מ)מ (מ)מ (ם)םם C. rectus (מ)ם (מ)ם (ם)םם E. corrodens Periodontal condition Pocket depth (mm) 3322 ממממ Bleeding on probing Gingival Index 1111 Plaque Index 1210 Mobility 1001 ממממ Pus exudate .of periodontal bacteria in saliva (ם) or presence (מ) Symbols in parenthesis note the absence Saliva could not be collected from the patient at the age of 3 years 4 months.

Piscataway, NJ, USA), as described previously9). C. sputigena and E. corrodens. Table 2 summarizes the longitudinal changes of Results periodontal conditions and bacteria in case 2. At 10 years 2 months of age, C. ochracea, P. nigrescens, Table 1 shows the longitudinal changes of the C. rectus, and B. forsythus were detected in the periodontal conditions and bacteria in case 1. At the plaque sample, and C. ochracea, C. sputigena, A. age of 3 years 4 months, when his gingivae exhibited actinomycetemcomitans, and B. forsythus in saliva. the inflamed condition, C. ochracea, C. sputigena, At 10 years 5 months, when periodontal pocket P. nigrescens, C. rectus, A. actinomycetemcomitans, depths reached 7 mm, 8 of the species, except and E. corrodens were detected in subgingival for P. gingivalis and T. denticola, were detected in plaque from the lower primary left canine. At the age plaque sample. At 10 years 8 months old, when of 4 years 9 months, when oral hygiene was poor, gingivae were inflamed, C. ochracea, C. sputigena, C. sputigena, C. rectus, A. actinomycetemcomitans, A. actinomycetemcomitans, and E. corrodens were and E. corrodens were each detected in plaque detected in plaque samples, as well as the former 3 sample, while C. ochracea was detected in saliva bacteria in saliva. At 11 years 2 months, 4 months samples in addition to the 4 species found in after the gingivectomy, periodontal bacteria were the plaque samples. At 5 years 11 months, when not detected from either plaque or saliva samples. the gingiva showed non-inflammatory hyperplastic At 11 years 6 months old, when her oral hygiene was lesions, C. ochracea, C. sputigena, and E. corrodens poor, only P. intermedia was detected in the saliva were found in plaque, and C. ochracea and sample. C. sputigena were found in saliva. At 6 years 6 months, when the primary canine showed slight Discussion mobility due to root resorption by adjacent lateral incisor, A. actinomycetemcomitans was detected again We considered that case 1 should be regarded as a from both plaque and saliva samples, along with rare case of gingival fibromatosis, because of the GINGIVAL FIBROMATOSIS IN CHILDREN 145

Table 2 Longitudinal changes in periodontal bacteria in subgingival plaque and saliva, and periodontal conditions in the area of the lesion near the upper right central incisor 10Y 10Y 10Y 11Y 11Y 2M 5M 8M 2M 6M Gram-negative anaerobic bacteria (מ)מ (מ)מ (מ)מ (מ)מ (מ)מ P. gingivalis (מ)מ (מ)מ (מ)מ (מ)מ (מ)מ T. denticola (מ)מ (מ)מ (ם)ם (ם)ם (ם)ם C. ochracea (מ)מ (מ)מ (ם)ם (ם)ם (ם)מ C. sputigena (ם)מ (מ)מ (מ)מ (מ)ם (מ)מ P. intermedia (מ)מ (מ)מ (מ)מ (מ)ם (מ)ם P. nigrescens (מ)מ (מ)מ (ם)ם (ם)ם (ם)מ A. actinomycetemcomitans (מ)מ (מ)מ (מ)מ (מ)ם (ם)ם B. forsythus (מ)מ (מ)מ (מ)מ (מ)ם (מ)ם C. rectus (מ)מ (מ)מ (מ)ם (מ)ם (מ)מ E. corrodens Periodontal condition Pocket depth (mm) 67635 םמםממ Bleeding on probing Gingival Index 22201 Plaque Index 11002 Mobility 00111 מממממ Pus exudate .of periodontal bacteria in saliva (ם) or presence (מ) Symbols in parenthesis note the absence

occurrence of alveolar bone loss in the primary months (Table 1), since it had been detected at a low dentition. Kharbonda et al.10) reported 6 cases of frequency from the age of 2 to 5 and its detection gingival fibromatosis among 3 generations of a rate increased with age. family. Of them, a 17-year-old boy showed extensive Dibart et al.15) reported a case of prepubertal enlargement of the gingiva, which induced inability periodontitis, in which putative periodontopathogens to close the lips, and the dental radiograph showed a such as P. intermedia and A. actinomycetemcomitans generalized alveolar bone loss in the molar region. were identified. A. actinomycetemcomitans has been His 6-year-old cousin with primary dentition showed reported to be limited to and generalized enlargement of the gingiva, however, is frequently isolated from child plaque samples16). no alveolar bone loss was noted. There are some In the present case 1, A. actinomycetemcomitans other reports of gingival fibromatosis with primary was also frequently isolated from the lesion, while dentition, but none describe alveolar bone loss4,11,12). P. intermedia was never detected. Furthermore, it In prepubertal periodontitis, alveolar bone loss is very unusual that 6 species of periodontopathic usually progresses rapidly, while acute destruction bacteria were detected at the age of 3 years 4 months is intermittent followed by prolonged periods of (Table 1), as the mean number of bacterial species remission13,14). In the present case 1, alveolar bone detected in healthy 3-year-old children was reported loss was found in the region of the primary lateral to be 2.755). In addition, the number of bacteria incisor and canine. The degree of bone loss detected in case 1 decreased as the patient grew up. remained unchanged for 3 years and the gingival Since the gingival overgrowth condition had been pockets were only 2–3 mm. A bacteriological already found at the age of 2 years 1 month and examination showed the presence of C. ochracea, slight mobility was recorded at 3 years 4 months, the C. sputigena, A. actinomycetemcomitans, C. rectus, patient may have had active destruction at an earlier and E. corrodens in the area of the lesion, which are age and then gone into remission for a long period. frequently detected in healthy children5). It may be One of the common clinical approaches for unusual that P. nigrescens was detected at 3 years 4 gingival fibromatosis is a gingivectomy followed by 146 Nakano, K., Nishiyama, N., Tamura, K. et al. plaque control in the affected area17). In the present 135, 1964. case 2, a gingivectomy was successful in eliminating 7) Löe, H. and Silness, J.: in pregnancy. (I) Prevalence and severity. Acta Odont the bad gingival condition, while removal of the Scand 21: 533–551, 1963. hyperplastic gingiva resulted in a drastic reduction of 8) Mättö, J., Sasrela, M., Alaluusua, S., Oja, V., periodontopathic bacteria. Several species including Jousimies-Somer, H. and Asikainen, S.: Detection P. intermedia, which is rarely isolated from healthy of Porphylomonas gingivalis from saliva by PCR by children5), were detected prior to the procedure. Three using a simple sample-processing method. J Clin months after the gingivectomy, none of the bacteria Microbiol 36: 157–160, 1998. 9) Amano, A., Kishima, T., Kimura, S., Takiguchi, were detected, however, P. intermedia was detected M., Ooshima, T., Hamada, S. and Morisaki, I.: 7 months after the gingivectomy. P. intermedia is Periodontopathic bacteria in children with Down regarded as one of the major virulent species for syndrome. 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