Idiopathic Gingival Fibromatosis Associated with Generalized Aggressive Periodontitis: a Case Report

Clinical P RACTIC E Idiopathic Gingival Fibromatosis Associated with Generalized Aggressive Periodontitis: A Case Report Contact Author Rashi Chaturvedi, MDS, DNB Dr. Chaturvedi Email: rashichaturvedi@ yahoo.co.in ABSTRACT Idiopathic gingival fibromatosis, a benign, slow-growing proliferation of the gingival tissues, is genetically heterogeneous. This condition is usually part of a syndrome or, rarely, an isolated disorder. Aggressive periodontitis, another genetically transmitted disorder of the periodontium, typically results in severe, rapid destruction of the tooth- supporting apparatus. The increased susceptibility of the host population with aggressive periodontitis may be caused by the combined effects of multiple genes and their inter- action with various environmental factors. Functional abnormalities of neutrophils have also been implicated in the etiopathogenesis of aggressive periodontitis. We present a rare case of a nonsyndromic idiopathic gingival fibromatosis associated with generalized aggressive periodontitis. We established the patient’s diagnosis through clinical and radiologic assessment, histopathologic findings and immunologic analysis of neutrophil function with a nitro-blue-tetrazolium reduction test. We describe an interdisciplinary approach to the treatment of the patient. For citation purposes, the electronic version is the definitive version of this article: www.cda-adc.ca/jcda/vol-75/issue-4/291.html diopathic gingival fibromatosis is a rare syndromic, have been genetically linked to hereditary condition that has no specific the chromosome 2p21-p222 and 5q13-q22.4 Icause.1 Investigations are ongoing to es- Recently, a mutation in the son of sevenless-1 tablish the genetic linkage and heterogen- (SOS-1) gene has been suggested as a possible eity associated with it.2–5 This condition may cause of isolated (nonsyndromic) gingival fi- manifest as an autosomal-dominant6–9 or, less bromatosis, but no definite linkage has been commonly, an autosomal-recessive mode of established.5 inheritance,10–12 either as an isolated disorder Idiopathic gingival fibromatosis is a slowly or as part of a syndrome.6 Syndromic gin- progressive benign enlargement that affects gival fibromatosis has been associated with the marginal gingiva, attached gingiva and ancillary features such as hypertrichosis, interdental papilla. The fibromatosis may mental retardation, epilepsy, progressive sen- potentially cover the exposed tooth surfaces, sorineural hearing loss and abnormalities of causing esthetic and functional problems, the extremities, particularly of the fingers and and in extreme cases may distort the jaws. toes.6,13 Autosomal-dominant forms of gin- The gingival tissues are usually pink and gival fibromatosis, which are usually non- nonhemorrhagic, and have a firm, fibrotic JCDA • www.cda-adc.ca/jcda • May 2009, Vol. 75, No. 4 • 291 ––– Chaturvedi ––– Figure 1: Preoperative intraoral frontal view Figure 2: Preoperative intraoral right lat- Figure 3: Preoperative intraoral left of the maxillary and mandibular arches. eral view of the maxillary and mandibular lateral view of the maxillary and man- arches. dibular arches. consistency.1,13–17 Histopathologically, the bulbous in- Case History creased connective tissue is relatively avascular and has Diagnosis densely arranged collagen-fibre bundles, numerous fibro- A 23-year-old man reported to the outpatient depart- blasts and mild chronic inflammatory cells. The over- ment of our institute complaining of pain and swelling in lying epithelium is thickened and acanthotic, and has the gums and an inability to chew food. The patient was elongated rete ridges.1,13–18 Unusual findings include the also concerned about the progressively increased spacing presence of small calcified particles, amyloid deposits, between his upper front teeth and their movement away islands of odontogenic epithelium and osseous metaplasia from their original positions. in the connective tissue, and ulcerations of the overlying 19 The medical history revealed that the patient’s gums mucosa. had progressively enlarged over the last 6 to 7 years Aggressive periodontitis comprises another and that the patient had pain during mastication and group of genetically inherited diseases that represent increased tooth mobility, especially of all 4 first molars, a severe and rapidly progressive form of periodontitis. 3 of which had to be extracted about 2 years before. Characteristically, these diseases tend to exhibit fam- Nothing in the patient’s medical and family history ilial aggregation and there are no factors in the medical seemed related to the condition, and no associated endo- 20 history that seem associated with the condition. crinological abnormality could be detected. The patient Aggressive forms of periodontitis are currently con- was not receiving any anti-epileptic, antihypertensive or sidered to be multifactorial diseases that develop as a immunosuppressive medications that could contribute result of complex interactions between specific host to the gingival enlargement. However, the patient had genes and the environment. Various factors have been smoked up to 20 cigarettes a day for the past 4 to 5 years identified that increase the risk of developing aggressive and drank alcohol about 2 or 3 times a week. periodontitis, including familial aggregation, single nu- Intraoral examination revealed moderate-to-severe cleotide polymorphisms, neutrophil functional defects, gingival overgrowth of a firm, dense and fibrotic consist- antibodies to specific bacteria, herpes virus infection, ency that involved both the maxillary and mandibular smoking and stress.21 arches (Figs. 1 to 3). The patient had generalized tooth Generalized aggressive periodontitis usually affects mobility with a severe pathologic migration, especially people under 30 years of age. They have a poor serum- of the upper anterior teeth. Full-mouth periodontal antibody response to infecting agents and pronounced charting, including assessment of probing depth and clin- episodic destruction of the attachment and alveolar bone. ical attachment level, revealed deep pockets throughout The loss of attachment affects at least 3 permanent teeth the mouth, and scanty plaque and calculus deposits. other than first molars and incisors.22 The radiographic findings, which corroborated those We report here an unusual case of a nonsyndromic, of the clinical examination, revealed severe generalized idiopathic gingival fibromatosis associated with gen- alveolar bone loss (Fig. 4). The peripheral blood results eralized aggressive periodontitis. Only one other case were normal and correlated with an absence of any with such an association has been reported to date.18 history of systemic disease. We discuss our patient’s clinical findings, histopath- Based on all these findings, a provisional diagnosis of ologic evaluation, analysis of neutrophil function and idiopathic gingival enlargement with generalized aggres- treatment plan. sive periodontitis was made. 292 JCDA • www.cda-adc.ca/jcda • May 2009, Vol. 75, No. 4 • ––– Idiopathic Gingival Fibromatosis ––– Figure 4: Preoperative panoramic radiograph of the maxillary and Figure 5: Gingival tissues excised during mandibular arches showing severe generalized alveolar bone loss. periodontal surgery were sent for histopathologic assessment. Figure 6: Photomicrograph of the Figure 7: Photomicrograph of the con- Figure 8: Photomicrograph illustrating the specimen illustrating the presence nective tissue illustrating the presence of presence of dense collagen bundles in the of a thickened acanthotic epithelium densely arranged collagen bundles with connective tissue with multiple areas of with elongated rete ridges and a numerous fibroblasts and few mixed inflam- neovascularization (original magnification densely fibrous connective tissue with matory cells, mainly around blood vessels 100×). numerous fibroblasts (original magni- (original magnification 400×). fication 100×). Histopathologic Findings tetrazolium (NBT) reduction test. During this test, The attached gingiva excised from the buccal and polymorphonuclear leucocytes (PMNLs) were stimulated interdental areas during surgery (Fig. 5) was immedi- with heat-killed spores of Candida albicans to evaluate ately fixed in 10% buffered formaldehyde solution and the oxidative pathway. Normal granulocytes metabolic- sent for histopathologic examination. The specimen was ally activated by phagocytosis reduce NBT to blue for- processed and then embedded in paraffin wax. Multiple mazan. Eighty percent of the patient’s cells contained 5-mm serial sections were prepared, stained with hema- reduced blue formazan, which was the percentage of toxylin and eosin, and viewed under a trinocular light PMNLs producing a respiratory burst, thereby indicating microscope with flat-field objective lenses at 100× and the normal functioning of the oxygen-dependent leuko- 400× magnifications. cytic bactericidal system. The microscopic evaluation of these sections revealed Treatment parakeratinized stratified squamous acanthotic epithelia with thin long rete ridges extending into the connective The patient initially underwent phase 1 periodontal tissue. The underlying connective tissue showed dense therapy that comprised scaling and root planing, oral wavy bundles of collagen fibres containing numerous fi- hygiene instructions and adjunctive administration of systemic doxycycline (100 mg twice a day, on the first day, brocytes and fibroblasts. Some sections in the connective

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