|Year : 2021 | Volume
| Issue : 4 | Page : 188-193
Idiopathic gingival fibromatosis with periodontitis: A rare case report and literature
Shivani Sachdeva1, Harish Saluja2, Amit Mani1
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 Submission||02-Jun-2020|
|Date of Decision||31-Jul-2020|
|Date of Acceptance||03-Dec-2020|
|Date of Web Publication||03-Feb-2021|
Dr. Shivani Sachdeva
Department of Periodontics, RDC, Pravara Institute of Medical Sciences, Loni, Rural Dental college Loni, Maharashtra, Ahmednagar
Source of Support: None, Conflict of Interest: None
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
| Introduction|| |
Gingival fibromatosis (GF) is a rare entity that causes esthetic, functional, and periodontal impairement., 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. 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. 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, Investigations are in still in the process to establish the association in genetic linkage and heterogeneity., This condition may manifest as an autosomal dominant mode of inheritance predominantly or autosomal recessive mode.,,, 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.
| Case Report|| |
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.
Facial disfigurement and incompetent and protruding lips. Lymph nodes were nonpalpable.
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. 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. 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|
Click here to view
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|
Click here to view
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.
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|
Click here to view
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|
Click here to view
| Discussion|| |
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|
Click here to view
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.
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. 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|| |
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.
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
Conflicts of interest
There are no conflicts of interest.
| References|| |
Gawron K, Łazarz-Bartyzel K, Potempa J, Chomyszyn-Gajewska M. Gingival fibromatosis: Clinical, molecular and therapeutic issues. Orphanet J Rare Dis 2016;11:9.
Chaturvedi R. Idiopathic gingival fibromatosis associated with generalized aggressive periodontitis: A case report. J Can Dent Assoc 2009;75:291-5.
Bozzo L, Almedia CP, Seully C, Akfred MJ. Hereditary gingival fibromatosis, report of an extensive 4 generation pedigree. Oral Surg Oral Med Oral Pathol 1998;86:304-7.
Pappachan B, Narayan JV, Nayak A. Idiopathic gingival fibromatosis: A neglected case. Indian J Radiol Imaging 2002;12:335-38. [Full text]
Bittencourt LP, Campos V, Moliterno LF, Ribeiro DP, Sampaio RK. Hereditary gingival fibromatosis: Review of the literature and a case report. Quintessence Int 2000;31:415-8.
Tiwana PS, De Kok IJ, Stoker DS, Cooper LF. Facial distortion secondary to idiopathic gingival hyperplasia: Surgical management and oral reconstruction with endosseous implants. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2005;100:153-7.
Hart TC, Pallos D, Bowden DW, Bolyard J, Pettenati MJ, Cortelli JR. Genetic linkage of hereditary gingival fibromatosis to chromosome 2p21. Am J Hum Genet 1998;62:876-83.
Hart TC, Pallos D, Bozzo L, Almeida OP, Marazita ML, O'Connell JR, et al.
Evidence of genetic heterogeneity for hereditary gingival fibromatosis. J Dent Res 2000;79:1758-64.
Bozzo L, de Almedia OP, Scully C, Aldred MJ. Hereditary gingival fibromatosis. Report of an extensive four-generation pedigree. Oral Surg Oral Med Oral Pathol 1994;78:452-4.
Bozzo L, Machado MA, de Almeida OP, Lopes MA, Coletta RD. Hereditary gingival fibromatosis: Report of three cases. J Clin Pediatr Dent 2000;25:41-6.
Martelli H Jr., Lemos DP, Silva CO, Graner E, Coletta RD. Hereditary gingival fibromatosis: Report of a five-generation family using cellular proliferation analysis. J Periodontol 2005;76:2299-305.
Singer SL, Goldblatt J, Hallam LA, Winters JC. Hereditary gingival fibromatosis with a recessive mode of inheritance. Case reports. Aust Dent J 1993;38:427-32.
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.
Bokenkamp A, Bohnhorst B, Beier C, Albers N, Offner G, Brodehl J. Nifedipine aggravates cyclosporine a-Inuced hyperplasia. Pediatr Nephrol 1994;8:181.
Caton JG, Armitage G, Berglund T, Chapple IL, Jepsen S, Kornman KS, et al
. A new classification scheme for periodontal and periimplant diseases and conditions – Introduction and keys changes from the 1999 classification. J Clin Perioontol 2017;45 Suppl 20:S1-8.
Ramachandra SS, Hegde M, Prasad UC. Gingival enlargement and mesiodens associated with generalized aggressive periodontitis: A case report. Dent Update 2012;39:364-6, 369.
[Figure 1], [Figure 2], [Figure 3]
[Table 1], [Table 2]