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Table of Contents   
SHORT COMMUNICATION  
Year : 2011  |  Volume : 22  |  Issue : 3  |  Page : 489-492
Gingival fibromatosis with hemi-osseous hyperplasia of jaws, focal maxillary viral papillomatosis of gingiva, fissured tongue and congenitally missing anterior teeth: A case report and surgical management of a new syndrome


1 Department of Periodontics, Vishnu Dental College, Vishnupure, Bhimivaram, WG DT, Andhra Pradesh, India
2 Department of Oral and Maxillofacial Pathology, Vishnu Dental College, Vishnupure, Bhimivaram, WG DT, Andhra Pradesh, India

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Date of Submission08-Feb-2010
Date of Decision23-Sep-2010
Date of Acceptance20-Nov-2010
Date of Web Publication3-Nov-2011
 

   Abstract 

Gingival fibromatosis is characterized by fibrotic enlargement of the gingiva that can occur as inherited or sporadic form. Inherited form can be an isolated trait or as a component of a syndrome. This article reports a 35 year old male patient affected by gingival fibromatosis associated with hemiosseous hyperplasia of mandible, maxilla, and zygoma on the right side, viral papillomatosis of maxillary anterior gingiva, fissured tongue and congenitally missing anterior teeth. The patient was subjected to phase I and phase II periodontal therapy. There was no evidence of recurrence of the enlargement after one year but the papillomatosis recurred. Gingival fibromatosis has been reported to be associated with various other abnormalities but not with those described in our case. This observation raises the possibility that the coexistence of these entities in our case may represent a new syndrome.

Keywords: Gingival fibromatosis, hereditary gingival fibromatosis, hemi osseous hyperplasia, viral papillomatosis (HPV), syndrome

How to cite this article:
Reddy M S, Manyam R, Babu M N, Saraswathi T R. Gingival fibromatosis with hemi-osseous hyperplasia of jaws, focal maxillary viral papillomatosis of gingiva, fissured tongue and congenitally missing anterior teeth: A case report and surgical management of a new syndrome. Indian J Dent Res 2011;22:489-92

How to cite this URL:
Reddy M S, Manyam R, Babu M N, Saraswathi T R. Gingival fibromatosis with hemi-osseous hyperplasia of jaws, focal maxillary viral papillomatosis of gingiva, fissured tongue and congenitally missing anterior teeth: A case report and surgical management of a new syndrome. Indian J Dent Res [serial online] 2011 [cited 2019 Dec 10];22:489-92. Available from: http://www.ijdr.in/text.asp?2011/22/3/489/87078
Gingival fibromatosis (GF) is the fibrous enlargement of the gingiva. It may be hereditary or drug induced as side effect of systemic drugs such as anticonvulsants, immunosuppresents and calcium channel blockers. [1] It is considered idiopathic, in the absence of any known cause. As an inherited disorder it may be part of a genetic syndrome [2],[3],[4] and more recently associated with generalized aggressive periodontitis [5] or it may be isolated and known as hereditary gingival fibromatosis (HGF). [2] HGF is traditionally considered to have an autosomal-dominant pattern of inheritance whereas syndromic forms with a dominant or recessive Mendelian transmission pattern has been reported. [2],[3] GF may also associate with dental abnormalities that includes generalized thin hypoplastic amelogenesis imperfecta, intrapulpal calcifications, delay of tooth eruption, periapical radiolucencies in unerupted teeth, dental agenesis, root dilacerations and in one case association of mental retardation is recently reported. [4] In this paper we present a case of GF associated with hemi-osseous hyperplasia of mandible, maxilla and zygoma and focal maxillary viral papillomatosis of gingiva, fissured tongue and congenitally missing anterior teeth and the surgical management of a new syndrome.


   Case Report Top


A 35-year-old male reported with the complaint of recurrent gingival overgrowth. He had developed generalized gingival enlargement during childhood which had been surgically excised. The enlargement had recurred after 8 years and slowly reached the present size. As the enlargement has assumed a huge size causing esthetic and functional impairment, the patient reported for treatment.

Medical history was not significant. Extra-oral examination revealed unilateral facial asymmetry on the right side and incompetence of lips. Intra-oral examination revealed massive diffuse gingival enlargement without significant gingival inflammation [Figure 1]. Bone sounding ruled out any underlying bone involvement. The anterior part of the palatal gingiva showed focal papillomatosis [Figure 2]. Permanent teeth from the canine-canine region were missing in the maxillary and mandibular anterior region and patient did not give history of extraction. The remaining teeth were totally embedded within the gingival overgrowth and were displaced. The oral hygiene index (OHI) of the individual was fair. The tongue showed midline fissure, was atrophied on the right side and was deviated towards the right side on protrusion.
Figure 1: Diffuse gingival enlargement

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Figure 2: Palatal gingiva showing focal papillomatosis

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Routine hematological investigations and biochemical investigations including serum calcium, phosphate, acid and alkaline phosphates were within normal limits. Abdominal ultrasound was normal. HIV infection was ruled out by Tridot. Polymerase chain reaction (PCR) was done for viral typing of HPV 16 and 18 for gingival papillamatosis and showed positive results for HPV16.

Orthopantomogram showed hyperplasia of the right maxilla, zygoma and mandible involving the body, ramus and condyle. Bony trabeculae showed coarse thick granularity on the right side of the mandible, maxilla and zygoma. Generalized horizontal bone loss of the alveolar bone was evident [Figure 3]. Computed tomography (CT) scan confirmed hemi-osseous hyperplasia, focal granular trabeculae in the mandibular and maxillary molar areas as seen in the orthopantomogram and showed increased osseodensity with increased width of alveolar bone on the right side [Figure 4].
Figure 3: Orthopantomograph showing hyperplasia of the right maxilla, zygoma and mandible involving the body, ramus and condyle

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Figure 4: CT scan confirms hemi–osseous hyperplasia

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Treatment included extraction of the mandibular right second molar as it was grossly decayed, followed by Phase I periodontal therapy (oral hygiene instructions, scaling and root planning). In Phase II therapy, quadrant-wise periodontal surgical treatment was planned one week later as clinical signs of inflammation were minimal after initial therapy.

Surgical therapy included external bevel gingivectomy in association with gingivoplasty on the left maxillary and mandibular areas as well as maxillary right palatal surface and mandibular right lingual surface as it involved the soft tissue alone which was confirmed by bone sounding. On the right buccal maxillary and mandibular areas, internal bevel gingivectomy combined with open-flap debridement followed by ostectomy and osteoplasty were done in order to remove the bone which was confirmed by bone sounding. Later, the flap was sutured and periodontal dressing was given for one week followed by 0.2% chlorhexidine mouth rinse for two weeks. The excised tissue was sent for histopathological evaluation.

Histological examination of the papillomatous lesion showed cytopathic changes involving a wider area of the epithelium. Cells of the stratum intermedium exhibited vacuolar degeneration and inclusion bodies confirming HPV 16 positivity. In the other regions, it showed hyperplastic stratified squamous epithelium with irregular, elongated rete ridges. The connective tissue showed dense bundles of collagen fibers and absence of inflammatory component [Figure 5]. Bone tissue showed deformed thinned cortical bone, presenting the "Mountain-Valley" appearance. Even though the pericortical bone showed osteoid, peripheral osteoblast rimming was absent. The connective tissue showed areas of collagenolysis [Figure 6].
Figure 5: Connective tissue showing dense bundles of collagen fibers

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Figure 6: Bone tissue showing thinned cortical bone which was deformed presenting a "Mountain–Valley" appearance

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After the last periodontal surgery and post-surgical follow-up visits, the patient returned periodically for observation. The papillomatosis on the maxillary palatal surface has reoccurred after six months.


   Discussion Top


Gingival fibromatosis (GF) is the fibrous enlargement of the gingiva. It may be hereditary or drug-induced as a side-effect of systemic drugs like anticonvulsants, immunosuppressants and calcium channel blockers. [1] It is considered idiopathic, in the absence of any known cause. As an inherited disorder, it may be part of a genetic syndrome [2],[3],[4] and more recently generalized aggressive periodontitis [5] or it may be isolated and known as hereditary GF (HGF). [2] HGF is traditionally considered to have an autosomal-dominant pattern of inheritance whereas syndromic forms have been reported with a dominant or recessive Mendelian transmission pattern. [2],[3] GF may also be associated with dental abnormalities that include generalized thin hypoplastic amelogenesis imperfecta, intrapulpal calcifications, delayed eruption, periapical radiolucencies in unerupted teeth, dental agenesis, root dilacerations and in one case, association with mental retardation has been reported. [4]

Gingival fibromatosis constitutes a component of many syndromes and the clinical findings are reported extensively in the literature. [6] Clinical findings such as hemi-osseous hyperplasia of the mandible, maxilla and zygoma; viral papillomatosis of the maxillary anterior gingiva; fissured tongue; congenitally missing anterior teeth associated with abnormal trabecular pattern of cancellous bone and increased bone density, associated with massive diffuse GF as observed in our case are not documented in the literature available.

Detailed family history and clinical examination of the patient's parents and siblings ruled out inheritance as a causative factor for GF in our case.

The radiological appearance of cancellous bone as granular instead of thin linear trabeculae was developmental and it was seen only on the right side of the mandible, maxilla and zygoma. The gingival growth was also massive on the right maxilla and mandible when compared to the left side. Therefore, the impact of pressure effect over the adjacent tissues was more on the right side than on the left. This explains the pressure atrophy of the right side of the tongue. Gingival enlargement begins at the time of eruption of permanent dentition but can develop with the eruption of the primary dentition also. It is rarely present at birth. [7] Associations of GF with anodontia or oligodontia have been reported before. [8]

In some cases, the gingiva becomes so firm and dense as to feel like bone on palpation. This finding was observed in our case and prompted us to do bone sounding before periodontal surgery. Bone sounding revealed underlying bone enlargement, later confirmed by CT scan (hemi-osseous hyperplasia). Although in few cases gingival hyperplasia occurs, the alveolar bone is not affected. [7]

Suggested treatment varies according to the degree of severity. When the enlargement is minimal, good scaling and oral hygiene instructions may be sufficient to maintain good oral health. Massive gingival enlargements however demand surgical correction, considering the impairment of function and esthetics. Various techniques used for excision of gingival overgrowth include external or internal bevel gingivectomy followed by gingivoplasty, apically positioned flap, electrocautery and carbon dioxide laser. In the present case, internal bevel gingivectomy followed by ostectomy and osteoplasty were performed on the right maxillary and mandibular buccal surface. In other areas, gingivectomy followed by gingivoplasty was performed. Recurrence of GF after treatment has been reported over a period of 2-14 years. This case is a rare entity of massive GF associated with papillamatosis, dento-osseous developmental anomalies not reported earlier in the literature and we consider it as a new syndrome–- "Sesha-–Ravi Syndrome".

 
   References Top

1.Sakamoto R, Nitta T, Kamikawa Y, Kono S, Kamikawa Y, Sugihara K, et al. Histochemical, immunohistochemical and ultrastructural studies of gingival fibromatosis: A case report. Med Electron Microsc 2002;35:248-54.  Back to cited text no. 1
    
2.Kavvadia K, Pepelassi E, Alexandridis C, Arkadopoulou A, Polyzois G, Tossios K. Gingival fibromatosis and significant tooth eruption delay in an 11 year old male. A 30 month follows up. Int J Pediatr Dent 2005;15:294-302.  Back to cited text no. 2
    
3.Hakkinen L, Csiszar A. Hereditary gingival fibromatosis: Characteristics and novel putative pathogenic mechanism. J Dent Res 2007;86:25-34.  Back to cited text no. 3
    
4.Martelli-Júnior H, Bonan PR, Dos Santos LA, Santos SM, Cavalcanti MG, Coletta RD. Case report of a New Syndrome associating gingival fibromatosis and dental abnormalities in a consanguineous family. J Periodontol 2008;79:1287-96.  Back to cited text no. 4
    
5.Casavecchia P, Uzel MI, Kantarci A, Hasturk H, Dibart S, Hart TC, et al. Hereditary gingival fibromatosis associated with generalized aggressive periodontitis. A case report. J Periodontol 2004;75:770-8.  Back to cited text no. 5
    
6.Dongari-Bagtzoglou A. Drug's associated gingival enlargement. J Periodontol 2004;75:1424-31.  Back to cited text no. 6
    
7.Bittencourt LP, Campos V, Moliterno LF, Ribeiro DP, Sampaio RK. Hereditary gingival fibromatosis: Review of literature and case report. Quintessence Int 2000;31:415-8.  Back to cited text no. 7
    
8.Gorlin RJ, Cohen MM Jr, Hennekam RC. Syndromes with Gingival/Periodontal components. In: Gorlin RJ, Cohen MM Jr, Henneken RC (editors). Syndromes of the Head and Neck. 4 th ed. New York, NY: Oxford University Press; 2001. p. 1093.  Back to cited text no. 8
    

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Correspondence Address:
M Sesha Reddy
Department of Periodontics, Vishnu Dental College, Vishnupure, Bhimivaram, WG DT, Andhra Pradesh
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0970-9290.87078

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    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6]



 

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