Localized juvenile spongiotic gingival hyperplasia: A report of 27 cases.

BACKGROUND: Localized juvenile spongiotic gingival hyperplasia (LJSGH) is a poorly understood but distinctive inflammatory hyperplasia occurring in children and young adults. Fewer than 100 cases have been reported since its initial description. METHODS: During the period of 2015 to 2018, cases of LJSGH were identified, retrieved and their clinical and histopathological data reviewed. RESULTS: There were 27 cases, with a median age of 13 years (range 7-72 years). Twenty-four of 27 patients were less than 20 years old, and in three cases the patients were over 60 years of age. The most commonly affected site was the anterior maxillary gingiva presenting as a solitary, red, and papillated lesion. Typical microscopic findings included elevated areas of variably acanthotic, spongiotic nonkeratinized epithelium with elongated rete ridges, accompanied by a neutrophilic-rich infiltrate. An abrupt transition between epithelium affected by LJSGH and normal mucosa was characteristic. LJSGH typically exhibited full-thickness epithelial expression of CK19 without expression of estrogen and progesterone receptors. CONCLUSIONS: The clinical and histopathologic characteristics of LJSGH are unique and consistent. Despite the name, the condition is not limited to juveniles and can occur in adults. LJSGH in adults and juveniles shares the same spectrum of histopathologic and immunohistochemical findings.

Nifedipine induced gingival enlargement in an edentulous patient: a case report with one year follow up.

BACKGROUND: Gingival enlargement due to calcium channel blockers is a common complaint reported by patients. It can be localized or generalized and can range from mild to severe, affecting patients appearance and function. Nifedipine induced gingival enlargement is noticed only in 10 % of patients and very few cases of Nifedipine induced gingival enlargement in an edentulous patient have been documented in the literature. CASE PRESENTATION: Here in, we report a case of gingival enlargement in a 70 year old hypertensive edentulous patient who was on low dose Nifedipine therapy. Patient wanted complete dentures. We planned to excise the overgrowth and followed up for 1 year. CONCLUSION: Nifedipine induced gingival enlargement noticed only in 10 % of patients. Hence, there is a need for physicians and dentist to make a coordinated treatment plan and practice care while prescribing these drugs which are associated with gingival overgrowth.

Distribution of biopsied non plaque-induced gingival lesions in a Chilean population according to the classification of periodontal diseases.

BACKGROUND: Many gingival lesions are not induced by plaque. The aim of this study was to analyze the frequency of biopsied non-plaque-induced gingival lesions (NPIGL) in a Chilean population. METHODS: One thousand twelve cases of biopsied gingival lesions with confirmed anatomopathologic diagnosis were included, from the records of the Oral Pathology Referral Institute (OPRI), Faculty of Dentistry, University of Chile, between years 1990 and 2009. RESULTS: The most frequent non plaque-induced gingival lesions categories from biopsied cases included hyperplastic lesions, malignancies and benign neoplasms. The most frequent diagnoses in each category were fibrous hyperplasia (35.47%), squamous cell carcinoma (3.85%) and giant cell fibroma (2.08%), respectively. From all lesions, only 8.3% fitted in the specified categories of the current classification of periodontal diseases. CONCLUSIONS: The most frequent biopsied NPIGL were hyperplastic lesions and neoplasms. These categories represent relevant lesions to be included in a future periodontal classification system to improve the care needs of the patients, as well as early diagnosis and treatment.

Localized Juvenile Spongiotic Gingival Hyperplasia: Report of Two Cases.

OBJECTIVE: Localized juvenile spongiotic gingival hyperplasia (LJSGH) is a painless gingival swelling that histologically exhibits hyperplasia of the non-keratinized stratified squamous epithelium, intercellular edema and spongiosis of the spinus layer, and exocytosis of inflammatory cells. LJSGH pathogenesis remains to be elucidated, while a possible origin from the gingival sulcus epithelium is nowadays proposed. STUDY DESIGN: We report two cases of LJSGH with immunohistochemical evaluation of cytokeratins (CKs) 18 and 19. RESULTS: Both cases concerned 12-year-old boys, who presented with a well-circumscribed bright red pedunculated papillary swelling on the marginal gingiva of the left maxillary lateral incisor. With the provisional diagnosis of LJSGH, the lesions were excised under local anesthesia and histological examination supported the final diagnosis of LJSGH. In both cases, the lesional epithelium showed intense and mild positivity for CK19 and CK18, respectively, while the adjacent normal gingival epithelium expressed CK19, but not CK18, only in the basal cell layer. The postoperative course was uneventful in both patients and no recurrence has been reported. CONCLUSION: LJSGH is a recently introduced entity that is worth attention in the clinical pediatric dentistry. Clinical and histological examination is required for the final diagnosis, while immunohistochemistry has shed light to LJSGH pathogenesis.

Localized juvenile spongiotic gingival hyperplasia possibly originates from the junctional gingival epithelium-an immunohistochemical study.

AIMS: To immunohistochemically evaluate the cytokeratin (CK) pattern of expression in localized juvenile spongiotic gingival hyperplasia (LJSGH) as compared with the gingival epithelium (GE). METHODS AND RESULTS: Ten cases of LJSGH were semiquantitatively evaluated for the immunohistochemical pattern of CK1/10, CK4, CK8/18, and CK19. GE controls were taken from 10 cases of reactive gingival fibroepithelial hyperplasia. GEs showed mean positivity rates of 80% for both CK1/10 and CK4, and 5% for both CK8/18 and CK19. LJSGHs showed mean positivity rates of 65% for CK19, 60% for CK8/18, 30% for CK4, and 5% for CK1/10. The differences between LJSGHs and GEs were statistically significant (P < 0.01). CONCLUSIONS: The LJSGH pattern of CK expression is reminiscent of the profile described in the literature for the junctional epithelium (JE). Possibly, JE exteriorized from the gingival sulcus would be more prone to irritation from a variety of sources, resulting in inflammation and hyperplasia, with the subsequent development of LJSGH.

Localized juvenile spongiotic gingival hyperplasia featuring unusual p16INK4A labeling and negative human papillomavirus status by polymerase chain reaction.

BACKGROUND: Localized juvenile spongiotic gingival hyperplasia (LJSGH) is a distinct type of gingival hyperplastic lesion with specific clinicopathologic features. Evaluation of the morphological characteristics of LJSGH indicates the potential role of human papillomavirus (HPV) infection as an underlying etiopathogenetic mechanism. METHODS: All cases of LJSGH from 2008 to present were retrieved. Clinical and demographic data were collected. HPV status was investigated by p16INK4A immunohistochemistry and HPV-Polymerase chain reaction (PCR). RESULTS: Twenty-one cases of LJSGH were identified, 14 (66.7%) affecting males and seven (33.3%) females (M:F = 2:1, age range: 8-36, mean: 13 years). All lesions were well-demarcated, exophytic, erythematous, and hemorrhagic with granular or slightly papillary surface. Preponderance for the maxillary gingiva (19, 90.5%) was observed. Two (9.5%) patients presented with recurrence 20 and 21 months after excision (mean follow-up: 18.7 months). Histopathologically, all LJSGH lesions featured epithelial hyperplasia with intense neutrophilic exocytosis and spongiosis. All cases demonstrated positivity for p16INK4A with the majority of specimens (47.6%) intensely decorated in >50% of the overlying epithelium with focal immunostaining observed in 47.6% and diffuse in 52.4%. Thirteen cases (61.9%) were negative for HPV DNA by PCR, while two (9.5%) were suspicious for the presence of low levels of HPV DNA but definitive genotyping was not possible. One case (4.8%) displayed positivity for HPV-31. The remaining five cases failed the PCR reaction. CONCLUSIONS: Human papillomavirus does not participate in the pathogenesis of LJSGH. P16INK4A expression in the absence of detectable HPV DNA can likely be attributed to the intense inflammation associated with LJSGH.

Whole-Exome sequencing identifies FAM20A mutations as a cause of amelogenesis imperfecta and gingival hyperplasia syndrome.

Amelogenesis imperfecta (AI) describes a clinically and genetically heterogeneous group of disorders of biomineralization resulting from failure of normal enamel formation. AI is found as an isolated entity or as part of a syndrome, and an autosomal-recessive syndrome associating AI and gingival hyperplasia was recently reported. Using whole-exome sequencing, we identified a homozygous nonsense mutation in exon 2 of FAM20A that was not present in the Single Nucleotide Polymorphism database (dbSNP), the 1000 Genomes database, or the Centre d'Etude du Polymorphisme Humain (CEPH) Diversity Panel. Expression analyses indicated that Fam20a is expressed in ameloblasts and gingivae, providing biological plausibility for mutations in FAM20A underlying the pathogenesis of this syndrome.

Inflammatory environment induces gingival tissue-specific mesenchymal stem cells to differentiate towards a pro-fibrotic phenotype.

BACKGROUND INFORMATION: Human gingival tissues are prone to hyperplasia under inflammatory stimuli. We have identified gingival tissue-specific mesenchymal stem cells (GMSCs) and found their functional change being correlated with drug-induced gingival hyperplasia. However, whether these cells exhibit characteristics of pro-fibrotic phenotype under inflammatory condition remains unknown. RESULTS: GMSCs isolated from human normal gingival tissues (N-GMSC) and inflammatory gingival tissues (I-GMSC) were cultured in vitro, representative cytokines were added to simulate the in vivo inflammatory environment. Under the influence of the inflammatory cytokines, GMSCs exhibited higher rate of proliferation than those under normal condition, while their potential for osteogenic and adipogenic differentiation was suppressed. The expression of matrix metalloproteinases (MMP)-1, MMP-2, IL-1, IL-6, TNF-α and type 1 collagen was significantly higher in I-GMSCs than in N-GMSCs. Furthermore, compared with dental pulp stem cells, GMSCs showed different pattern of gene expression and extracellular matrix formation in inflammatory environment. CONCLUSIONS: Inflammatory microenvironment induces GMSCs to differentiate towards a pro-fibrotic phenotype, which could underlie the hyperplastic appearance of inflammatory gingiva.

A quantitative analysis of mast cells in inflammatory periapical and gingival lesions.

AIM: The aim of the study was to quantify the presence of mast cells in various inflammatory lesions like periapical granuloma, periapical cyst, inflammatory gingival hyperplasia and pyogenic granuloma. Mast cell degranulation and association with lymphocytes were also recorded in an attempt to understand the role of mast cells in the pathogenesis of these inflammatory lesions. MATERIALS AND METHODS: The quantification of mast cells was done on toluidine blue stained sections of all the four groups of lesions, using the image analyzer software, Image-Pro-Express (Media Cybernetics, USA). RESULTS: An increased number of mast cells in various inflammatory lesions with a significant difference between the four groups were noted. Mast cell number tended to be greater in the lesions present in the anterior region of the mouth than in the posterior region of the oral cavity. The mean mast cell number decreased with the increasing age which was directly correlated with the age of the patients. Mast cell site, distribution, degranulation and its association with fibroblasts, lymphocytes and blood vessels were noted. CONCLUSION: The location of mast cells in different areas, their association with lymphocytes, fibroblasts, endothelial cells, and the phenomenon of degranulation helps to appreciate the release of various mediators and multiple interactions among these cells, leading to increased vascular permeability, angiogenic response, collagen synthesis, regulation of inflammation, bone resorption, and extracellular matrix destruction, thus contributing to the pathogenesis of these inflammatory lesions.

Evaluation of ABCB5 immunostained epithelial stem cells in oral squamous cell carcinoma, inflammatory gingival hyperplasia and normal mucosa.

Oral cancer is the most prevalent head and neck cancer. Although tumor markers have been investigated for detecting the progression and prognosis of oral cancer, no reliable marker has been identified. We investigated the expression of ATP binding cassette subfamily B member 5 (ABCB5) positive stem cells in oral squamous cell carcinoma (OSCC) and in inflammatory gingival hyperplasia. We used tissue samples from normal subjects, patients with inflammatory gingival hyperplasia, and patients with OSCC. Samples were investigated using anti-ABCB5 monoclonal antibody immunohistochemistry to detect epithelial stem cells. Staining density, intensity, and immunoreactive scores of ABCB5 were analyzed for the three study groups. We found ABCB5 immunostaining in all three study groups, but different distributions of ABCB5 expression in different layers of the epithelium. We found no significant difference in staining intensity between inflammatory hyperplasia and normal mucosa, but we found significantly stronger expression in OSCC compared to normal and inflammatory hyperplasia individually. Elevated expression of ABCB5 in OSCC suggests an increased subpopulation of tumor cells with an undifferentiated stem cell phenotype, which facilitates cancer initiation and progression.

Can chlorhexidine mouthwash twice daily ameliorate cyclosporine-induced gingival overgrowth?

BACKGROUND/PURPOSE: Gingival overgrowth can be induced in patients treated with cyclosporine-A (CsA), an immunosuppressant often used following organ transplantation. A pre-existing rat model designed to mimic CsA-induced gingival overgrowth in humans was used to test the effectiveness of frequent application of a chlorhexidine antiplaque solution in reducing the overgrowth. METHODS: Four groups of rats were fed CsA. One group received chlorhexidine mouthwash twice a day, the second group received chlorhexidine mouthwash once a day, the third group received chlorhexidine mouthwash every other day, and the fourth group did not receive chlorhexidine mouthwash all. A fifth negative control group received only mineral oil. Overgrowth was determined by measuring the changes in the gingival probing depth and the keratinized gingival width on molars. A gingival histological examination was performed. RESULTS: Rats treated with mouthwash twice daily exhibited decreased probing depths and gingival widths without statistical significance. Histological examination revealed that CsA treatment caused gingival enlargement, whereas chlorhexidine treatment twice a day diminished the enlargement. CONCLUSION: These findings suggest that chlorhexidine mouthwash used twice daily may reduce the severity of CsA-induced gingival overgrowth. Further research is warranted to determine the optimal dose and treatment regimen.

Identification of 2 novel ANTXR2 mutations in patients with hyaline fibromatosis syndrome and proposal of a modified grading system.

Juvenile hyaline fibromatosis (JHF) and infantile systemic hyalinosis (ISH) are rare, autosomal recessive disorders of the connective tissue caused by mutations in the gene encoding the anthrax toxin receptor 2 protein (ANTXR2) located on chromosome 4q21. Characteristically, these conditions present with overlapping clinical features, such as nodules and/or pearly papules, gingival hyperplasia, flexion contractures of the joints, and osteolytic bone defects. The present report describes a pair of sibs and three other JHF/ISH patients whose diagnoses were based on typical clinical manifestations and confirmed by histopathologic analyses and/or molecular analysis. A comparison of ISH and JHF, additional thoughts about new terminology (hyaline fibromatosis syndrome) and a modified grading system are also included.

Giant gingival pseudoepitheliomatous hyperplasia in lung squamous cell carcinoma.

We here describe a case of giant primary gingival pseudoepitheliomatous hyperplasia in a 53-year-old Chinese male patient with lung squamous cell carcinoma (SCC). The pathogenesis of the lesion and the deferential diagnosis from invasive SCC are also discussed. To our knowledge, such a hugeous primary pseudoepitheliomatous hyperplasia of the gingiva accompanied with lung SCC is unusual.

Can gingival hyperplasia caused by cyclosporin A be prevented by removing dental plaque? / Megelozheto-e a ciklosporin A okozta fogíny-hyperplasia a dentalis plakk eltávolításával?

In Hungary, there are around 3500 people living with a transplanted organ, there are around 400 total donations, including between 250 and 300 kidney transplants per year. Due to the development of immunosuppression and surgical techniques, the survival rate of patients has improved, and therefore dentists are increasingly confronted with gingival hyperplasia due to immunosuppressive therapy in addition to oro-dental lesions resulting from renal failure. The prevalence of gingival hyperplasia with cyclosporin A can be as high as 90%. The aim of our study is to raise awareness of the importance of dental plaque in the development of cyclosporin A induced gingival hyperplasia and to emphasize that gingival hyperplasia and the risk of organ rejection can be prevented or greatly reduced by the development of appropriate individual oral hygiene and successful periodontal outpatient therapy. Particular mention should be paid to the importance of interdisciplinary cooperation and regular patient care. Our examinations and treatments were carried out at the Division of Periodontology at the Department of Dentistry and Oral Surgery of the University of Pecs, Hungary.