Literature DB >> 32154139

Unusual clinical and histologic findings in a child with mixed dentition with hereditary gingival fibromatosis: a case report.

Han Gao1,2, Jun Liang1, Xiaoxue Xia1, Zhaoming Deng1, Zhaoqiang Zhang3.   

Abstract

Hereditary gingival fibromatosis (HGF) is characterized by an accumulation of extracellular matrix resulting in a fibrotic enlargement of keratinized gingiva. The goal of this article is to describe one kindred affected with HGF and discuss the diagnosis, treatment, recurrence risk and histopathology features of the proband with mixed dentition. An 8-year-old boy reported with a chief complaint of gingival enlargement. A series of examinations were performed to make a diagnosis and family histories of the patient were recorded. Surgical therapy included gingivectomy and gingivoplasty technique were performed for the proband to remove excess gingiva. Collagen bundles in the permanent incisors area were prevalently significantly bulkier than that of the deciduous teeth area and the arrangement was much more regular. Microscopic evaluation revealed that epithelial hyperplasia was severe in tissue with intense inflammation, whereas tissue with mild inflammation showed only long and deep epithelial papillae. The patient was placed on a 3-month recall schedule and didn't demonstrate any recurrence of gingival overgrowth over a 1-year follow-up period. The study revealed that the eruption of permanent teeth played an important role in accelerating gingival hyperplasia. For the first time, it was confirmed from histological perspective that inflammation in gingival tissue was crucial to the gingival overgrowth of HGF. 2020 Translational Pediatrics. All rights reserved.

Entities:  

Keywords:  Genetic disease; case report; gingival fibromatosis; mixed dentition

Year:  2020        PMID: 32154139      PMCID: PMC7036643          DOI: 10.21037/tp.2019.12.01

Source DB:  PubMed          Journal:  Transl Pediatr        ISSN: 2224-4336


  22 in total

Review 1.  Hereditary gingival fibromatosis: characteristics and novel putative pathogenic mechanisms.

Authors:  L Häkkinen; A Csiszar
Journal:  J Dent Res       Date:  2007-01       Impact factor: 6.116

2.  Gingival fibromatosis and significant tooth eruption delay in an 11-year-old male: a 30-month follow-up.

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Journal:  Int J Paediatr Dent       Date:  2005-07       Impact factor: 3.455

Review 3.  Gingival overgrowth in children: epidemiology, pathogenesis, and complications. A literature review.

Authors:  Aikaterini Doufexi; Mina Mina; Effie Ioannidou
Journal:  J Periodontol       Date:  2005-01       Impact factor: 6.993

4.  Hereditary gingival fibromatosis: report of a five-generation family using cellular proliferation analysis.

Authors:  H Martelli-Junior; D P Lemos; C O Silva; E Graner; R D Coletta
Journal:  J Periodontol       Date:  2005-12       Impact factor: 6.993

5.  A mutation in the SOS1 gene causes hereditary gingival fibromatosis type 1.

Authors:  Thomas C Hart; Yingze Zhang; Michael C Gorry; P Suzanne Hart; Margaret Cooper; Mary L Marazita; Jared M Marks; Jose R Cortelli; Debora Pallos
Journal:  Am J Hum Genet       Date:  2002-02-26       Impact factor: 11.025

6.  REST Final-Exon-Truncating Mutations Cause Hereditary Gingival Fibromatosis.

Authors:  Yavuz Bayram; Janson J White; Nursel Elcioglu; Megan T Cho; Neda Zadeh; Asuman Gedikbasi; Sukru Palanduz; Sukru Ozturk; Kivanc Cefle; Ozgur Kasapcopur; Zeynep Coban Akdemir; Davut Pehlivan; Amber Begtrup; Claudia M B Carvalho; Ingrid Sophie Paine; Ali Mentes; Kivanc Bektas-Kayhan; Ender Karaca; Shalini N Jhangiani; Donna M Muzny; Richard A Gibbs; James R Lupski
Journal:  Am J Hum Genet       Date:  2017-07-06       Impact factor: 11.025

7.  In vitro testing the potential of a novel chimeric IgG variant for inhibiting collagen fibrils formation in recurrent hereditary gingival fibromatosis: chimeric antibody in a gingival model.

Authors:  K Gawron; K Lazarz-Bartyzel; M Lazarz; K Steplewska; K Pyrc; J Potempa; M Chomyszyn-Gajewska
Journal:  J Physiol Pharmacol       Date:  2014-08       Impact factor: 3.011

8.  Hereditary gingival fibromatosis associated with generalized aggressive periodontitis: a case report.

Authors:  Piero Casavecchia; M Ilhan Uzel; Alpdogan Kantarci; Hatice Hasturk; Serge Dibart; Thomas C Hart; Philip C Trackman; Thomas E Van Dyke
Journal:  J Periodontol       Date:  2004-05       Impact factor: 6.993

9.  Early onset of hereditary gingival fibromatosis in a 28-month-old.

Authors:  Gary H Breen; Rocco Addante; Candice C Black
Journal:  Pediatr Dent       Date:  2009 Jul-Aug       Impact factor: 1.874

Review 10.  Gingival fibromatosis: clinical, molecular and therapeutic issues.

Authors:  Katarzyna Gawron; Katarzyna Łazarz-Bartyzel; Jan Potempa; Maria Chomyszyn-Gajewska
Journal:  Orphanet J Rare Dis       Date:  2016-01-27       Impact factor: 4.123

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