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 Table of Contents  
CASE REPORT
Year : 2021  |  Volume : 41  |  Issue : 4  |  Page : 188-193

Idiopathic gingival fibromatosis with periodontitis: A rare case report and literature


1 Department of Periodontics, Rural Dental College, Pravara Institute of Medical Sciences, Loni, Ahmednagar, Maharashtra, India
2 Department of OMFS, Rural Dental College, Pravara Institute of Medical Sciences, Loni, Ahmednagar, Maharashtra, India

Date of Submission02-Jun-2020
Date of Decision31-Jul-2020
Date of Acceptance03-Dec-2020
Date of Web Publication03-Feb-2021

Correspondence Address:
Dr. Shivani Sachdeva
Department of Periodontics, RDC, Pravara Institute of Medical Sciences, Loni, Rural Dental college Loni, Maharashtra, Ahmednagar
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jmedsci.jmedsci_165_20

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  Abstract 


Idiopathic gingival fibromatosis (IGF) is a hereditary condition with esthetic, functional, and periodontal impairment in patients. It is slowly progressive in nature. The enlargement might be nodular or localized and symmetrical in nature all along the dentition in both the arches and similar gender prediction uniformly. The present case report of IGF in a 21-year-old female patient covering three-fourths of crowns in posterior areas. The gingival enlargement was excised by external bevel gingivectomy along with the use of electrocautery. The gingival fibromatosis can be genetic, hereditary, or drug-influenced covering marginal, papillary or in severe, long-standing cases, it might cover crown of teeth, thus hampering mastication henceforth, functional impairment. The prognosis is uncertain, and the recurrence of risk is high.

Keywords: Gingival enlargement, idiopathic gingival enlargement, hereditary gingival fibromatosis, nonsyndromic gingival hyperplasia


How to cite this article:
Sachdeva S, Saluja H, Mani A. Idiopathic gingival fibromatosis with periodontitis: A rare case report and literature. J Med Sci 2021;41:188-93

How to cite this URL:
Sachdeva S, Saluja H, Mani A. Idiopathic gingival fibromatosis with periodontitis: A rare case report and literature. J Med Sci [serial online] 2021 [cited 2021 Oct 25];41:188-93. Available from: https://www.jmedscindmc.com/text.asp?2021/41/4/188/308681




  Introduction Top


Gingival fibromatosis (GF) is a rare entity that causes esthetic, functional, and periodontal impairement.[1],[2] It is characterized by the excessive pathological growth of gingival tissue, which might be localized or symmetrical. Localized growths are mostly nodular in nature around maxillary tuberosity and also on buccal and lingual aspects of the mandibular posterior.[3] Symmetrical or generalized gingival enlargements are more common, which occur in both the arches. These enlargements are also referred and described as “gingival enlargement,” which comprises hyperplasia, hypertrophy, or both.

GF is also referred as gingivomatosis, gingival enlargement, gingival hyperplasia, gingival overgrowth (GO), elephantiasis gingivae, familial elephantiasis, gigantism of the gingiva, and congenital macrogingivae.[4] GF can present as Hereditary GF, which may appear as an isolated entity, i.e., nonsyndromic and as a part of a genetic disease or syndrome, as drug-induced gingival overgrowth (DIGO) or as idiopathic gingival fibromatosis (IGF). GF is usually pale pink, firm, leathery in consistency, and presents a characteristic pebbled surface. The GO starts with the eruption of deciduous as well as permanent teeth.

IGF or hereditary GF is a rare form of hereditary condition that has no definite etiology. IGF has a prevalence of 1 in 750,000 individuals and can occur in both genders and in either of the jaws[5],[6] Investigations are in still in the process to establish the association in genetic linkage and heterogeneity.[7],[8] This condition may manifest as an autosomal dominant mode of inheritance predominantly or autosomal recessive mode.[9],[10],[11],[12] Autosomal-dominant forms of GF, which are usually nonsyndromic, have been genetically linked to the chromosome 2p21–p222 and 5q13–q22. At present, a mutation in the son of sevenless-1 (SOS-1) gene has been related as a potential etiological factor of nonsyndromic GF, but still, no precise linkage is ascertained.[13]


  Case Report Top


A female patient 21-year-old reported to out patient department of periodontics with facial disfigurement along the right side of face for the past 7 years. She also gave a history of prior excision of swelling at the age of 10 years and exfoliation of first molars. The swelling had increased as the teeth erupted in the oral cavity. She also complained of difficultly in mastication, and the oral hygiene index (OHI) was fair. The patient did not reveal any medical or systemic history, neither drug history nor family history.

Extraoral examination

Facial disfigurement and incompetent and protruding lips. Lymph nodes were nonpalpable.

Intraoral examination

The enlargement was on all facial and lingual/palatal aspects with pale pink color. It was generalized or symmetric enlargement [Figure 1]. The gingiva was firm and fibrosed in consistency. There were pockets with both true and false components, mean clinical attachment loss was 5–7 mm. Mobility was Grade III with all the third molar and with right maxillary second molar as well as left mandibular second molar. While the maxillary anteriors and mandibular anterior teeth were grade II mobile. According to Bokenkamp et al. 1994, we classify gingival enlargement as Grade 3 as it covered three fourth of crown, in particular in the maxillary and mandibular posteriors arches.[14] All the first molars were absent with flaring of anterior teeth with increased intermaxillary rest position. The OHI scores depicted fair oral hygiene, but attachment loss was severe. Since, we cannot use the terminology aggressive periodontitis (AP) hence, according to the latest classification by American Academy of Periodontology (AAP 2017) the present case will be regarded as nonplaque induced periodontitis with Stage IV and Grade C.[15] There was a rapid rate of destruction which exceeded the expectations of given biofilm deposits. The clinical patterns were suggestive of periods of rapid progression and early onset.
Figure 1: Preoperative view. (a) Extraoral facial view (b) Enlargement in mandibular left side (c) Right maxillary view (d) Left maxillary view

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Investigations

All blood investigations were found to be in the normal range, no mental retardation, no fatigue, and weight loss. Thyroid indices were all normal. There was no possible association with syndromes associated with GF. A list of possible syndromes are depicted in [Table 1].
Table 1: List of syndromes and diseases associated with gingival fibromatosis

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On panoramic view, there was severe bone loss, and molars were floating. Provisional diagnosis of immunoglobulin (IG) E with isolated GF nonsyndromic along with Stage IV and Grade C periodontitis was confirmed.

Treatment protocol

The treatment protocol was planned full-mouth disinfection by mechanical and chemical plaque control therapy to remove the periodontal pathogens followed by surgical treatment to eradicate the unesthetic overgrowth. As the overgrowth might become the nidus for food debris and harbor millions of periodontal pathogens later so, gingivectomy was planned along with nutritional supplementation with Vitamin c for good gingival health.

In the etiotrophic phase, full-mouth disinfection was carried out in single sitting with initial chlorhexidine mouthwash (0.2%). Scaling and root planning were performed under local anesthesia. After 3 weeks, external bevel gingivectomy was done with radical excision of gingival enlargement in the maxillary right posterior area and mandibular left posterior region along with the removal of floating third molars. The enlargements were excised with scalpel and bipolar electrocautery was used for excision in posterior regions of the mandibular left side as well as maxillary right side to avoid excessive bleeding [Figure 2].
Figure 2: Surgical view (a) Panaromic radiograph; (b) Excision of mandibular posterior lesion with electrocautery; (c) Excised lesion of manibular posterior lesion and maxillary right posterior area along with extracted teeth

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The tissue was sent for histopathological examination. Histologically, it was described as a severe hyperplasia of the epithelium with hyperkeratosis along with elongation of rete pegs. The increase in the tissue mass was primarily the result of an increase in thickening of the collagen bundles in the connective tissue stroma. The patient was recalled after 15 days with no extraoral swelling and uneventful healing of edentulous maxillary and mandibular ridges after surgery [Figure 3]. The patient was referred to the prosthodontic department for further treatment.
Figure 3: Postoperative view extraorally and intraorally (a) Extraoral view; (b) Healed maxillary arch; (c) Healed mandibular arch

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  Discussion Top


Gingival enlargements could be classified on the basis of etiology and pathologic conditions in [Table 2]. In most of the cases, we can hit upon the etiology and eradicate the underlying pathology. Similarly, in cases of inflammatory enlargement the triggers, which are periodontal pathogens, can be eradicated by mechanical and chemical plaque control.
Table 2: Classification of gingival enlargement according to etiology and pathologic changes

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Drugs influenced gingival enlargements (DIGO) are primarily caused due to genetic predisposition, and the presence of local factors plaque and calculus deteriorate the condition more critically. The anticonvulsants, immunosuppressants, and calcium channel blockers come under this category. DIGO can be treated by either medicinal or surgical modalities. In the medicinal therapy, we give the drug substitutes, followed by mechanical plaque control. In most cases, the enlargements resolve by means of medicinal approach, and gums become healthy, but still, there are situations where surgical approach by gingivectomy has to be carried out for removal of overgrowth keeping both functional and esthetic demands in mind.

In conditioned enlargements like in pregnancy, puberty and Vitamin c deficiency enlargements, effective plaque control and supplementation by Vitamin c in case of Vitamin c deficiency enlargement can take care for healthy gingiva. The prognosis for inflammatory, DIGO, conditional enlargements is good to fair. However, there are also cases where family, medical, prenatal, drug histories were noncontributory and with no definite etiology. This category of GF is known as idiopathic and incidence is sporadic.

GF is rare (one in 750,000 individuals) and benign progressive condition which may be related to hereditary factors. IGF or hereditary GF can occur as an isolated condition or as part of a genetic syndrome. Mutation in the SOS-1 gene has been suggested as one doable etiological cause of isolated (nonsyndromic) hereditary GF, but mutations in other genes are also likely to be involved, given the heterogeneity of this condition.[13]

The present case was diagnosed as IGF with severe periodontitis with Stage IV and Grade C with rapid and early onset of periodontal destruction. Earlier, these conditions were regarded as AP, but now the newer classification (AAP 2017) classification has eradicated the terminology of AP. However still, the linkage can be suspected in cases of IGF or hepatocyte growth factor (HGF) and their association with advanced periodontitis. Though in literature, sporadic cases have been reported with both IGE or HGF and associated with AP.

A similar case of GF was recently reported by Ramachandra et al.[27] reported a case of gingival enlargement associated with generalized AP and the presence of mesiodens. The common features of GF and localized AP are their onset around puberty, female predilection, hereditary back-ground, and progression in the presence of minimal local factors, although secondary involvement can aggravate the preexisting condition.

Histopathologically, it involves hyperplasia of the epithelium with elongated rete ridges extending into the underlying connective tissue. The connective tissue consists of excess collagen but has relatively few fibroblasts and blood vessels. The treatment varies depending upon the etiology. The supporting periodontal therapy includes removal of etiological factors, nonsurgical periodontal therapy and surgical excision of lesion or gingivectomy using either scalpel, electrocautery, or lasers.

Though the prognosis of such cases is awful sometimes and chances of recurrence are high. In the present case also, there was a history of recurrence, but results after surgical therapy were good and the prognosis was fair, with no recurrence for a period of 6 months and the patient will be kept under follow-up. Appropriate time for removal of gingival enlargement is at the age of 3, 6, and 12 years along with effectual plaque control. Tooth eruption many a times is associated with gingival inflammation and pain, so chances of lack of oral hygiene and GO in patients with genetic predisposition is quite common and should be corrected timely to avoid the unnecessary deterioration of the condition. The best time for treatment is 3 years because by this time, all the deciduous teeth erupt, by 6 years as all the incisors and first molars erupt and by 12 years as second molars and premolars erupt. Well said a stitch in time saves nine.


  Conclusion Top


Utmost importance in GF case is the differential diagnosis. Sufficient effort and time should be spent on the case history and proper evaluation of the patient to associate all the possible vulnerable link between the cause of GF in the particular patient so that the chain can be broken down by removal of the etiological factor. The recurrence rate cannot be predicted so well; the psychological, esthetic, and functional benefits far overshadow the risk of recurrence. Oral hygiene and the superimposition of plaque accumulation have a crucial effect on the prognosis of GF.

Consent

Patient has given her consent for her images and other clinical information to be reported in the journal. The patient understands that her name and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.

Declaration of patient consent

The authors certify that they have obtained all appropriate patient consent forms. In the form, the patient has given her consent for her images and other clinical information to be reported in the journal. The patient understand that name and initials will not be published and due efforts will be made to conceal identity, but anonymity cannot be guaranteed.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.



 
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Ma Y, Sun Z, Hu Y, Liu Y, Jin L, Zhang F. Non-syndromic hereditary gingival fibromatosis in three Chinese families is not due to SOS1 gene mutations. Cell Biochem Biophys 2014;70:1869-73.  Back to cited text no. 13
    
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    Figures

  [Figure 1], [Figure 2], [Figure 3]
 
 
    Tables

  [Table 1], [Table 2]



 

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