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Hereditary gingival fibromatosis in children: a systematic review of the literature

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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.

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Authors and Affiliations

  1. Dental School, Aristotle University of Thessaloniki, 54124, Thessaloniki, Greece

    Eirini Boutiou

  2. Medical School, Aristotle University of Thessaloniki, Thessaloniki, Greece

    Ioannis A. Ziogas

  3. Institute of Health Innovations and Outcomes Research, The Feinstein Institute for Medical Research, Manhasset, NY, USA

    Dimitrios Giannis

  4. Private Practice Limited to Periodontics and Implant Dentistry, Thessaloniki, Greece

    Aikaterini-Elisavet Doufexi

Authors
  1. Eirini Boutiou
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  2. Ioannis A. Ziogas
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  3. Dimitrios Giannis
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  4. Aikaterini-Elisavet Doufexi
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Contributions

All authors contributed to the study conception and design. Material preparation, data collection, and analysis were performed by Eirini Boutiou, Ioannis A. Ziogas, Dimitrios Giannis, and Aikaterini-Elisavet Doufexi. The first draft of the manuscript was written by Eirini Boutiou, and all authors commented on previous versions of the manuscript. The final manuscript was read and approved by all authors.

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Correspondence to Eirini Boutiou.

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Boutiou, E., Ziogas, I.A., Giannis, D. et al. Hereditary gingival fibromatosis in children: a systematic review of the literature. Clin Oral Invest 25, 3599–3607 (2021). https://doi.org/10.1007/s00784-020-03682-x

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