Hereditary gingival fibromatosis in children: a systematic review of the literature
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ORIGINAL ARTICLE
Hereditary gingival fibromatosis in children: a systematic review of the literature Eirini Boutiou 1
&
Ioannis A. Ziogas 2 & Dimitrios Giannis 3 & Aikaterini-Elisavet Doufexi 4
Received: 25 August 2020 / Accepted: 4 November 2020 # Springer-Verlag GmbH Germany, part of Springer Nature 2020
Abstract Objectives Hereditary gingival fibromatosis (HGF) is an uncommon, inherited condition with slow and progressive fibrous hyperplasia of the gingiva. Due to its association with mastication, speech, and occlusion problems, early diagnosis is important. We sought to summarize the available data regarding the epidemiology, clinical characteristics, and outcomes of children with HGF (< 18 years). Methods A systematic literature review of the MEDLINE and Cochrane Library databases was conducted with respect to the Preferred Reporting Items for Systematic Reviews and Meta-Analyses statement (end-of-search date: March 1, 2019). Results A total of 99 articles reporting on 146 patients were included. The mean age was 10.82 ± 3.93 years, and generalized gingival enlargement was seen in 97.16% (95% CI 92.69 to 99.14). Jaw, gingival, and teeth abnormalities; poor oral hygiene; eating; or speech difficulties were typical HGF-induced, while 60.90% had extraoral manifestations (95% CI 52.41 to 68.78). The disease was most commonly inherited in an autosomal dominant manner (88.41%, 95% CI 78.5 to 94.26), and about one-third of the patients had syndromic HGF (33.85%, 95% CI 23.50 to 46.00). Gingivectomy was performed in the majority of cases (91.15%, 95% CI 84.31 to 95.29), and recurrence was seen in 33.85% (95% CI 23.50 to 46.00). Conclusion HGF should be suspected in children with nodularity and gingival fibrosis, teeth abnormalities, or jaw distortion. Family history can help to establish the diagnosis. Clinical relevance More cases should focus on longer-term follow-up after gingivectomy as disease recurrence is not uncommon. Keywords Hereditary gingival fibromatosis . Gingival hypertrophy . Gingival overgrowth . Gingivectomy
Introduction Hereditary gingival fibromatosis (HGF) is a rare, hereditary, benign disease characterized by slow and progressive fibrous hyperplasia of the gingiva [1, 2] and was initially reported by Goddard and Gross in 1856 [3]. It has a prevalence of 1:175,000 according to phenotype and 1:350,000 according * Eirini Boutiou [email protected] 1
Dental School, Aristotle University of Thessaloniki, 54124 Thessaloniki, Greece
2
Medical School, Aristotle University of Thessaloniki, Thessaloniki, Greece
3
Institute of Health Innovations and Outcomes Research, The Feinstein Institute for Medical Research, Manhasset, NY, USA
4
Private Practice Limited to Periodontics and Implant Dentistry, Thessaloniki, Greece
to genotype, and it can affect both mandibular and maxillary gingiva [4]. The mode of inheritance is still controversial; while it is generally considered to be an autosomal dominant disease, there are a few studies demonstrating that it may also follow an autosomal recessive
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