Anti-fibrotic potential of a chimeric IgG variant in cell culture established from hereditary gingival fibromatosis with a high propensity for recurrence and immunoreactive component within the lesion (original) (raw)
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Journal of physiology and pharmacology : an official journal of the Polish Physiological Society, 2014
Gingival fibromatosis is a progressive enlargement of the gingiva. It may hinder oral cavity hygiene and result in underlying bone loss. The long-term benefits of surgery cannot be predicted. On the other hand, alternative, efficient and non-invasive methods are not available at present. The aim of this study was to test the inhibitory effects of a chimeric IgG variant on collagen fibril formation in the cell culture of gingival fibroblasts taken from a patient with hereditary gingival fibromatosis with a high propensity for recurrence. Gingival biopsies were collected from the mandibular gingiva and used for histological evaluation as well as to establish a fibroblast culture. A histological evaluation was made in haematoxylin-eosin and Heidenhain's trichrome stained tissue sections. The inhibitory effect of a chimeric antibody on collagen fibril formation was determined in fibroblast cultures by using a collagen-specific Western blot and immunofluorescent staining. A histologi...
Gingival fibromatosis: clinical, molecular and therapeutic issues
Orphanet Journal of Rare Diseases, 2016
Gingival fibromatosis is a rare and heterogeneous group of disorders that develop as slowly progressive, local or diffuse enlargements within marginal and attached gingiva or interdental papilla. In severe cases, the excess tissue may cover the crowns of the teeth, thus causing functional, esthetic, and periodontal problems, such as bone loss and bleeding, due to the presence of pseudopockets and plaque accumulation. It affects both genders equally. Hereditary, drug-induced, and idiopathic gingival overgrowth have been reported. Hereditary gingival fibromatosis can occur as an isolated condition or as part of a genetic syndrome. The pathologic manifestation of gingival fibromatosis comprises excessive accumulation of extracellular matrix proteins, of which collagen type I is the most prominent example. Mutation in the Son-of-Sevenless-1 gene has been suggested as one possible etiological cause of isolated (non-syndromic) hereditary gingival fibromatosis, but mutations in other genes are also likely to be involved, given the heterogeneity of this condition. The most attractive concept of mechanism for drug-induced gingival overgrowth is epithelial-to-mesenchymal transition, a process in which interactions between gingival cells and the extracellular matrix are weakened as epithelial cells transdifferentiate into fibrogenic fibroblast-like cells. The diagnosis is mainly made on the basis of the patient's history and clinical features, and on histopathological evaluation of affected gingiva. Early diagnosis is important, mostly to exclude oral malignancy. Differential diagnosis comprises all pathologies in the mouth with excessive gingival overgrowth. Hereditary gingival fibromatosis may present as an autosomal-dominant or less commonly autosomal-recessive mode of inheritance. If a systemic disease or syndrome is suspected, the patient is directed to a geneticist for additional clinical examination and specialized diagnostic tests. Treatments vary according to the type of overgrowth and the extent of disease progression, thus, scaling of teeth is sufficient in mild cases, while in severe cases surgical intervention is required. Prognosis is precarious and the risk of recurrence exists.
Study About Hereditary Gingival Fibromatosis and Its Impact on Dental Treatment
2019
Introduction: Gingival Fibromatosis orGingival Hyperplasia is a rare disorderwith slow and progressive growth andis characterized by being a neoplasm inthe gingival tissue, where it can be foundin a localized manner. This study aimsto elucidate the characteristics and possiblemanifestations, aiming to facilitateits identifi cation, treatment and control.Methodology: This is a literature review.Forty-two articles and theses with languagesin English, Spanish, French, and Portuguesewere researched, whose sourcesare Scielo and Pubmed, and 25 articleswere included for their relevance to thetopic. Literature review: Gingival fi bromatosisis a clinical manifestation presentin several genetic syndromes that canlead to a craniofacial involvement that ischaracterized by the expansion and accumulationof connective tissue with increasednumber of cells and collagen fi bers.Fibromatosis has a proliferation of well-differentiated fi broblasts with varying amountsof collagen between proliferating c...
Oral Surgery Oral Medicine Oral Pathology Oral Radiology and Endodontology, 2008
Objective. To examine the histomorphologic and histomorphometric features of tissue from 3 unrelated families with hereditary gingival fibromatosis (HGF). Study design. Twelve affected individuals from 3 HGF families and 3 control subjects were evaluated. Gingival samples were fixed in formalin and embedded in paraffin for hematoxylin and eosin stain to count the number of fibroblast and inflammatory cells. Sirius red staining was performed to quantitate the amount of collagen present. Results. Histomorphologic analysis of HGF showed extension of epithelial rete ridges into the underlying lamina propria and the presence of collagen bundles in the connective tissue. Analysis of the mean area fraction of collagen showed that there were significant increases in the collagen fraction for all HGF types compared with control subjects (P Ͻ .05). There were significant increases in the number of fibroblasts for HGFa and HGFb compared with control subjects (P Ͻ .05). The number of fibroblasts for HGFc were similar to that for control subjects. Conclusions. The collagen fraction was significantly greater in all HGF types compared with controls. The number of fibroblasts was significantly increased in 2 of the 3 HGF types compared with controls. These data indicate that different mechanisms may be responsible for tissue enlargement in different forms
Study About Hereditary Gengival Fibromatosis and Its Impact on Dental Treatment
2019
Introduction: Gingival Fibromatosis or Gingival Hyperplasia is a rare disorder with slow and progressive growth and is characterized by being a neoplasm in the gingival tissue, where it can be found in a localized manner. This study aims to elucidate the characteristics and possible manifestations, aiming to facilitate its identifi cation, treatment and control. Methodology: This is a literature review. Forty-two articles and theses with languages in English, Spanish, French, and Portuguese were researched, whose sources are Scielo and Pubmed, and 25 articles were included for their relevance to the topic. Literature review: Gingival fi bromatosis is a clinical manifestation present in several genetic syndromes that can lead to a craniofacial involvement that is characterized by the expansion and accumulation of connective tissue with increased number of cells and collagen fi bers. Fibromatosis has a proliferation of well-diff erentiated fi broblasts with varying amounts of collagen between proliferating cells and can be classifi ed as: drug-induced and hereditary. Through histopathology, the stratifi ed squamous epithelium is obese with prominent and elongated ridges, dense connective tissue and disorganized cells with slow but progressive growth. It is also a rare disorder of non-neoplastic gingival tissue (1 case/750,000 people) that presents normal coloration, fi rm consistency and absence of infl ammatory signs. Conclusion: The expression of Gingival Fibromatosis has several origins, making it necessary to know the causative factor in the patient to prepare the treatment plan according to their needs, so the conversation with the patient through the anamnesis is so important at the beginning of treatment. Oral manifestations are important clinical fi ndings to assist in the diagnosis of genetic syndromes, allowing the dentist to guide their patients and clarify doubts about dental impairment.
American Journal of Case Reports, 2016
Objective: Rare disease Background: Hereditary gingival fibromatosis is characterized by slowly progressive enlargement of the gingiva that can present as an isolated condition or a part of various syndromes. Case Report: An 11-year-old female reported with a gingival lesion that caused masticatory problems and poor oral hygiene. Periodontal examination revealed a dense tissue covering 30% of her teeth crowns within both jaws. Panoramic x-ray showed a normal bone height and teeth positioning. The patient did not use any medications, but a similar condition was also present in other family members. The patient was diagnosed with hereditary gingival fibromatosis. Surgery was carried out to remove excess of gingival tissue. Post-surgical healing was uneventful, but four weeks after the first surgery, the condition recurred amounting to 45% of the initial tissue volume presenting in the mandible, and 25% in the maxilla. Two months later, no significant growth was noted in the mandible, while in the maxilla, growth increased to 40% of the pre-operative state. Analysis by polarized microscope showed a significant increase of thin fibrotic fibrils that contributed 80% of the total pool of collagen fibrils in the patient's gingiva, but only 25% in healthy gingiva. The patient was receiving outpatient care for follow-up every three months and surgical intervention had not been planned as long as her periodontal health would not be compromised. Conclusions: It is currently not clear whether the extent of the fibrosis had a mechanistic association with the ratio of gingival tissue re-growth in our case study. Further studies are needed to explain this association and improve the management of this condition.
Journal of Periodontology, 2005
Background: Hereditary gingival fibromatosis (HGF) is an uncommon condition characterized by an accumulation of extracellular matrix resulting in a fibrotic enlargement of the gingiva. The goal of this article is to describe one kindred affected with HGF and discuss the diagnosis, treatment, and control of the disease. The pattern of inheritance, histopathologic characteristics, and proliferative potential of epithelial and mesenchymal cells of HGF are also emphasized. Methods: To characterize the pattern of inheritance and the clinical appearance of gingival overgrowth, 117 family members were examined. The recurrence risk was estimated by the use of a genetic analysis program. Immunohistochemistry against the proliferating cell nuclear antigen (PCNA) and pKi-67 was performed to assess cellular proliferation of normal gingiva (NG) and HGF cells. Results: Examination of the family pedigree demonstrated an autosomal dominant trait of inheritance, and a sibling recurrence risk of 0.085 and an offspring recurrence risk of 0.078, indicating that HGF was a consequence of genetic alteration with low penetrance. Unaffected and affected members transmitted the disease to their offspring. The affected patients showed a generalized but mild gingival overgrowth. Surgical treatment consisted of a combination of gingivectomy and gingivoplasty. Histologic examination showed that the gingival lesions of all patients were quite similar, with increased amounts of collagen fiber bundles in the connective tissue. Immunohistochemistry revealed that the proliferative potential of epithelial cells was significantly higher in the HGF group compared to the NG group, whereas mesenchymal cells from both groups were negative for the proliferative markers. Conclusion: Our data demonstrated that, in the studied family, HGF is transmitted by an autosomal dominant pattern with incomplete disease penetrance, and although the gingival enlargement resulted from an excessive accumulation of collagen fibers, HGF is characterized by an increase in the proliferation rate of epithelial cells.
The Autosomal Recessive Inheritance of Hereditary Gingival Fibromatosis
Case Reports in Dentistry, 2013
Hereditary gingival fibromatosis (HGF) is a rare condition which is marked by enlargement of gingival tissue that covers teeth to various extents leading to aesthetic disfigurement. This study presents a case of a 28-year-old female patient and 18-year-old male who belong to the same family suffering from HGF with chief complaint of overgrowing swelling gingiva. The presence of enlarged gingiva with the same eruption was found in their other family members with no concomitant drug or medical history, and the occurrence of HGF has been found in one generation of this family which may indicate the autosomal recessive inheritance pattern of HGF. Hereditary gingival fibromatosis is an idiopathic condition as its etiology is unknown and it was found to recur in some cases even after surgical treatment. Both patients underwent thorough oral prophylaxis and later surgical therapy to correct the deformity.