Skip to main content
Top
Published in: Clinical Oral Investigations 6/2021

01-06-2021 | Fibromatosis | Original Article

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

Authors: Eirini Boutiou, Ioannis A. Ziogas, Dimitrios Giannis, Aikaterini-Elisavet Doufexi

Published in: Clinical Oral Investigations | Issue 6/2021

Login to get access

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.
Literature
3.
go back to reference Goddard WH, SDG (1856) Case of hypertrophy of the gums. Dent Regist West 9(9):276–282 Goddard WH, SDG (1856) Case of hypertrophy of the gums. Dent Regist West 9(9):276–282
7.
go back to reference Bansal A, Narang S, Sowmya K, Sehgal N (2011) Treatment and two-year follow-up of a patient with hereditary gingival fibromatosis. J Indian Soc Periodontol 15:406–409CrossRef Bansal A, Narang S, Sowmya K, Sehgal N (2011) Treatment and two-year follow-up of a patient with hereditary gingival fibromatosis. J Indian Soc Periodontol 15:406–409CrossRef
10.
go back to reference DeAngelo S, Murphy J, Claman L et al (2007) Hereditary gingival fibromatosis--a review. Compend Contin Educ Dent 28:138–143 quiz 144, 152PubMed DeAngelo S, Murphy J, Claman L et al (2007) Hereditary gingival fibromatosis--a review. Compend Contin Educ Dent 28:138–143 quiz 144, 152PubMed
13.
go back to reference Aboujaoude S, Cassia A, Moukarzel C (2016) Diode laser versus scalpel in the treatment of hereditary gingival fibromatosis in a 6-year old boy. Clin Pract 6:895CrossRef Aboujaoude S, Cassia A, Moukarzel C (2016) Diode laser versus scalpel in the treatment of hereditary gingival fibromatosis in a 6-year old boy. Clin Pract 6:895CrossRef
15.
go back to reference Rasila D, Asi K, Mahajan A (2016) A rare case report of hereditary gingival fibromatosis with atrial septal defect. IP Int J Periodontol Implantol 1:105–107 Rasila D, Asi K, Mahajan A (2016) A rare case report of hereditary gingival fibromatosis with atrial septal defect. IP Int J Periodontol Implantol 1:105–107
16.
go back to reference Karthikeyan BV, Khanna D, Prabhuji MLV (2016) The autosomal dominant inheritance of hereditary gingival fibromatosis: a case report. N Y State Dent J 82:43–46PubMed Karthikeyan BV, Khanna D, Prabhuji MLV (2016) The autosomal dominant inheritance of hereditary gingival fibromatosis: a case report. N Y State Dent J 82:43–46PubMed
17.
go back to reference Parkarwar P, Patil K, Anjum A, Choudhari R, Doshi Y, Birangane R (2015) Idiopathic gingival fibromatosis – a rare case report. Int JAppl Dent Sci 1:156–160 Parkarwar P, Patil K, Anjum A, Choudhari R, Doshi Y, Birangane R (2015) Idiopathic gingival fibromatosis – a rare case report. Int JAppl Dent Sci 1:156–160
19.
go back to reference Ahmed S, Ali Z (2015) Rare case of idiopathic gingival fibromatosis affecting primary dentition. J Ayub Med Coll Abbottabad 27:933–935PubMed Ahmed S, Ali Z (2015) Rare case of idiopathic gingival fibromatosis affecting primary dentition. J Ayub Med Coll Abbottabad 27:933–935PubMed
20.
go back to reference Ercoli C, Bartolino M, Montesani L, Docimo R (2015) Gingival fibromatosis: a case report. Eur J Paediatr Dent 16:233–235CrossRef Ercoli C, Bartolino M, Montesani L, Docimo R (2015) Gingival fibromatosis: a case report. Eur J Paediatr Dent 16:233–235CrossRef
21.
go back to reference Camilotti RS, Ferreira TB, Antonini F et al (2015) Resection of gingival fibromatosis with high-power laser. J Dent Child 82:47–52 Camilotti RS, Ferreira TB, Antonini F et al (2015) Resection of gingival fibromatosis with high-power laser. J Dent Child 82:47–52
23.
go back to reference Inagaki K, Kamei H, Mitani A, Noguchi T (2014) Management of traumatic dental injury after periodontal surgery in patient with hereditary gingival fibromatosis: case report. Oral Health Dent Manag 13:260–265PubMed Inagaki K, Kamei H, Mitani A, Noguchi T (2014) Management of traumatic dental injury after periodontal surgery in patient with hereditary gingival fibromatosis: case report. Oral Health Dent Manag 13:260–265PubMed
24.
go back to reference Gohil M, Padhiyar N, Gohil A (2013) Idiopathic gingival fibromatosis. J Res Med Den Sci 1:23–26CrossRef Gohil M, Padhiyar N, Gohil A (2013) Idiopathic gingival fibromatosis. J Res Med Den Sci 1:23–26CrossRef
30.
go back to reference Dhadse PV, Yeltiwar RK, Pandilwar PK, Gosavi SR (2012) Hereditary gingival fibromatosis. J Indian Soc Periodontol 16:606–609CrossRef Dhadse PV, Yeltiwar RK, Pandilwar PK, Gosavi SR (2012) Hereditary gingival fibromatosis. J Indian Soc Periodontol 16:606–609CrossRef
32.
go back to reference Butchi Babu K, Pavankumar K, Anuradha BR, Arora N (2011) Hereditary gingival fibromatosis - a case report and management using a novel surgical technique. RSBO 8:453–458 Butchi Babu K, Pavankumar K, Anuradha BR, Arora N (2011) Hereditary gingival fibromatosis - a case report and management using a novel surgical technique. RSBO 8:453–458
34.
go back to reference Anand Nayak P, Nayak UA, Khandelwal V, Ninave N (2011) Idiopathic gingival fibromatosis. Int J Clin Pediatr Dent 4:77–81CrossRef Anand Nayak P, Nayak UA, Khandelwal V, Ninave N (2011) Idiopathic gingival fibromatosis. Int J Clin Pediatr Dent 4:77–81CrossRef
35.
go back to reference Ramnarayan BK, Sowmya K, Rema J (2011) Management of idiopathic gingival fibromatosis: report of a case and literature review. Pediatr Dent 33:431–436PubMed Ramnarayan BK, Sowmya K, Rema J (2011) Management of idiopathic gingival fibromatosis: report of a case and literature review. Pediatr Dent 33:431–436PubMed
38.
go back to reference Duddu MK, Muppa R, Reddy GSP, Reddy PVN (2012) Non syndromic gingival fibromatosis in a mild mental retardation child. Contemp Clin Dent 3:S206–S209CrossRef Duddu MK, Muppa R, Reddy GSP, Reddy PVN (2012) Non syndromic gingival fibromatosis in a mild mental retardation child. Contemp Clin Dent 3:S206–S209CrossRef
39.
go back to reference Pol DG, Lobo TM, Pol SD (2016) Idiopathic gingival fibromatosis with asymmetrical presentation and electrosurgical management. J Indian Soc Periodontol 20:98–102CrossRef Pol DG, Lobo TM, Pol SD (2016) Idiopathic gingival fibromatosis with asymmetrical presentation and electrosurgical management. J Indian Soc Periodontol 20:98–102CrossRef
40.
go back to reference Shetty AK, Shah HJ, Patil MA, Jhota KN (2010) Idiopathic gingival enlargement and its management. J Indian Soc Periodontol 14:263–265CrossRef Shetty AK, Shah HJ, Patil MA, Jhota KN (2010) Idiopathic gingival enlargement and its management. J Indian Soc Periodontol 14:263–265CrossRef
42.
go back to reference Breen GH, Addante R, Black CC (2009) Early onset of hereditary gingival fibromatosis in a 28-month-old. Pediatr Dent 31:286–288PubMed Breen GH, Addante R, Black CC (2009) Early onset of hereditary gingival fibromatosis in a 28-month-old. Pediatr Dent 31:286–288PubMed
45.
go back to reference Anegundi RT, Sudha P, Nayak UAPJ (2006) Idiopathic gingival fibromatosis: a case report. Hong Kong Dent J 3:53–57 Anegundi RT, Sudha P, Nayak UAPJ (2006) Idiopathic gingival fibromatosis: a case report. Hong Kong Dent J 3:53–57
47.
go back to reference Tavargeri AK, Kulkarni SS, Sudha P (2004) Idiopathic gingival fibromatosis - a case report. J Indian Soc Pedod Prev Dent 22:180–182PubMed Tavargeri AK, Kulkarni SS, Sudha P (2004) Idiopathic gingival fibromatosis - a case report. J Indian Soc Pedod Prev Dent 22:180–182PubMed
50.
go back to reference Katz J, Guelmann M, Barak S (2002) Hereditary gingival fibromatosis with distinct dental, skeletal and developmental abnormalities. Pediatr Dent 24:253–256PubMed Katz J, Guelmann M, Barak S (2002) Hereditary gingival fibromatosis with distinct dental, skeletal and developmental abnormalities. Pediatr Dent 24:253–256PubMed
51.
go back to reference Khan U, Mustafa S, Saleem Z, Azam A, Khan ZA (2012) Hereditary gingival fibromatosis diagnosis and treatment. Pakistan Oral Dent J 32:226–231 Khan U, Mustafa S, Saleem Z, Azam A, Khan ZA (2012) Hereditary gingival fibromatosis diagnosis and treatment. Pakistan Oral Dent J 32:226–231
52.
go back to reference Kelekis-Cholakis A, Wiltshire WA, Birek C (2002) Treatment and long-term follow-up of a patient with hereditary gingival fibromatosis: a case report. J Can Dent Assoc 68:290–294PubMed Kelekis-Cholakis A, Wiltshire WA, Birek C (2002) Treatment and long-term follow-up of a patient with hereditary gingival fibromatosis: a case report. J Can Dent Assoc 68:290–294PubMed
53.
go back to reference Brennan MT, O’Connell BC, Rams TE, O’Connell AC (1999) Management of gingival overgrowth associated with generalized enamel defects in a child. J Clin Pediatr Dent 23:97–101PubMed Brennan MT, O’Connell BC, Rams TE, O’Connell AC (1999) Management of gingival overgrowth associated with generalized enamel defects in a child. J Clin Pediatr Dent 23:97–101PubMed
57.
go back to reference Hallett KB, Bankier A, Chow CW et al (1995) Gingival fibromatosis and Klippel-Trenaunay-Weber syndrome. Case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 79:578–582CrossRef Hallett KB, Bankier A, Chow CW et al (1995) Gingival fibromatosis and Klippel-Trenaunay-Weber syndrome. Case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 79:578–582CrossRef
61.
go back to reference Johnson BD, el-Guindy M, Ammons WF et al (1986) A defect in fibroblasts from an unidentified syndrome with gingival hyperplasia as the predominant feature. J Periodontal Res 21:403–413CrossRef Johnson BD, el-Guindy M, Ammons WF et al (1986) A defect in fibroblasts from an unidentified syndrome with gingival hyperplasia as the predominant feature. J Periodontal Res 21:403–413CrossRef
63.
go back to reference Emerson TG (1965) Hereditary gingival hyperplasia. A family pedigree of four generations. Oral Surg Oral Med Oral Pathol 19:1–9CrossRef Emerson TG (1965) Hereditary gingival hyperplasia. A family pedigree of four generations. Oral Surg Oral Med Oral Pathol 19:1–9CrossRef
66.
go back to reference Byars LTSB (1944) Congenital macrogingivae (fibromatosis gingivae) and hypertrichosis. Surg J 15:964–970 Byars LTSB (1944) Congenital macrogingivae (fibromatosis gingivae) and hypertrichosis. Surg J 15:964–970
67.
go back to reference Manoj M, Garg R, Babu K (2017) Unusual presentation of familial gingival fibromatosis among male siblings. J Indian Soc Periodontol 21:152–155CrossRef Manoj M, Garg R, Babu K (2017) Unusual presentation of familial gingival fibromatosis among male siblings. J Indian Soc Periodontol 21:152–155CrossRef
71.
go back to reference Straka M, Danisovic L, Bzduch V et al (2016) The significance of electron microscopic examination of gingiva in cases of Hunter syndrome and hereditary gingival fibromatosis. Neuro Endocrinol Lett 37:353–360PubMed Straka M, Danisovic L, Bzduch V et al (2016) The significance of electron microscopic examination of gingiva in cases of Hunter syndrome and hereditary gingival fibromatosis. Neuro Endocrinol Lett 37:353–360PubMed
77.
go back to reference Wagaiyu EG, Ng’ang’a RN, Kemoli AM (2009) Hereditary gingival fibromatosis: report of family case series. East Afr Med J 86:491–493PubMed Wagaiyu EG, Ng’ang’a RN, Kemoli AM (2009) Hereditary gingival fibromatosis: report of family case series. East Afr Med J 86:491–493PubMed
84.
go back to reference Bozzo L, Machado MA, de Almeida OP et al (2000) Hereditary gingival fibromatosis: report of three cases. J Clin Pediatr Dent 25:41–46CrossRef Bozzo L, Machado MA, de Almeida OP et al (2000) Hereditary gingival fibromatosis: report of three cases. J Clin Pediatr Dent 25:41–46CrossRef
89.
go back to reference Skrinjaric I, Bacic M (1989) Hereditary gingival fibromatosis: report on three families and dermatoglyphic analysis. J Periodontal Res 24:303–309CrossRef Skrinjaric I, Bacic M (1989) Hereditary gingival fibromatosis: report on three families and dermatoglyphic analysis. J Periodontal Res 24:303–309CrossRef
93.
go back to reference Raeste AM, Collan Y, Kilpinen E (1978) Hereditary fibrous hyperplasia of the gingiva with varying penetrance and expressivity. Scand J Dent Res 86:357–365PubMed Raeste AM, Collan Y, Kilpinen E (1978) Hereditary fibrous hyperplasia of the gingiva with varying penetrance and expressivity. Scand J Dent Res 86:357–365PubMed
102.
go back to reference McIndoe A, Smith O (1958) Congenital familial fibromatosis of the gums with the teeth as a probable etiological factor: report of an affected family. Br J Plast Surg 11:62–71CrossRef McIndoe A, Smith O (1958) Congenital familial fibromatosis of the gums with the teeth as a probable etiological factor: report of an affected family. Br J Plast Surg 11:62–71CrossRef
103.
go back to reference Büchner HJ (1957) Diffuse fibroma of the gums. Report of two cases. J Am Dent Assoc Dent Cosm 24:1937 Büchner HJ (1957) Diffuse fibroma of the gums. Report of two cases. J Am Dent Assoc Dent Cosm 24:1937
104.
go back to reference Savara SB, Suher T, Everett FG, Burns AG (1954) Hereditary gingival fibrosis. Study of a family. J Periodontol 25:12–21CrossRef Savara SB, Suher T, Everett FG, Burns AG (1954) Hereditary gingival fibrosis. Study of a family. J Periodontol 25:12–21CrossRef
105.
go back to reference Ruggles S (1925) Primary hypertrophy of the gums. J AMA 84:20–24 Ruggles S (1925) Primary hypertrophy of the gums. J AMA 84:20–24
107.
go back to reference Skrinjaric I, Bacic M, Poje Z (1983) Hereditary gingival fibromatosis with hemophilia B. Acta Stomatol Croat 17:89–94PubMed Skrinjaric I, Bacic M, Poje Z (1983) Hereditary gingival fibromatosis with hemophilia B. Acta Stomatol Croat 17:89–94PubMed
Metadata
Title
Hereditary gingival fibromatosis in children: a systematic review of the literature
Authors
Eirini Boutiou
Ioannis A. Ziogas
Dimitrios Giannis
Aikaterini-Elisavet Doufexi
Publication date
01-06-2021
Publisher
Springer Berlin Heidelberg
Keyword
Fibromatosis
Published in
Clinical Oral Investigations / Issue 6/2021
Print ISSN: 1432-6981
Electronic ISSN: 1436-3771
DOI
https://doi.org/10.1007/s00784-020-03682-x

Other articles of this Issue 6/2021

Clinical Oral Investigations 6/2021 Go to the issue