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Hyperplastic Dental Follicle: A Case Report and Literature Review

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DOI: 10.1155/2014/251892

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Case Reports in Dentistry
Volume 2014, Article ID 251892, 7 pages
http://dx.doi.org/10.1155/2014/251892

Case Report
Hyperplastic Dental Follicle: A Case Report and
Literature Review

Ligia Buloto Schmitd,1,2 Diego Maurício Bravo-Calderón,1

Cleverson Teixeira Soares,3 and Denise Tostes Oliveira1
1
Pathology Division, Department of Stomatology, Bauru School of Dentistry, University of São Paulo,
Alameda Octávio Pinheiro Brisolla 9-75, 17012-901 Bauru, SP, Brazil
2
ESFA Dental School, Rua Bernardino Monteiro 700, 29650-000 Santa Teresa, ES, Brazil
3
Lauro de Souza Lima Institute, Rodovia Comandante João Ribeiro de Barros, Km 225/226, 17034-971 Bauru, SP, Brazil

Correspondence should be addressed to Denise Tostes Oliveira; denisetostes@usp.br

Received 25 July 2014; Revised 12 September 2014; Accepted 15 September 2014; Published 8 October 2014

Academic Editor: Indraneel Bhattacharyya

Copyright © 2014 Ligia Buloto Schmitd et al. This is an open access article distributed under the Creative Commons Attribution
License, which permits unrestricted use, distribution, and reproduction in any medium, provided the original work is properly
cited.

Hyperplastic dental follicle is an odontogenic hamartomatous lesion associated with delayed or tooth eruption failure in young
patients. The occurrence of this pericoronal dental lesion may be single or multiple and it seems to be more frequent than literature
has reported. We present a literature review focusing on the etiopathogenesis and clinicopathological features of this hamartomatous
lesion in young patients. In addition, we reported a case of hyperplastic dental follicle causing delayed tooth eruption of 14-year-old
male patient. Microscopic analyses based on routine staining and immunohistochemistry were used to discuss the cells found in
pericoronal follicle. This paper reinforces the importance of association between clinical history and radiographic features with
microscopic pericoronal follicle examination for diagnosis of this hamartomatous lesion.

1. Introduction [5, 12, 13] and they suggested that this condition should be
considered a distinct pathology [12, 13].
Hyperplastic dental follicle has been described as odonto- The occurrence of hamartomas from odontogenic origin
genic hamartomatous lesion that occurs in pericoronal tis- seems to be more frequent than that which has been reported
sues of the unerupted tooth [1, 2]. Although its occurrence in the literature [14–16]. Furthermore, the diagnosis of hyper-
can be found in any age, most cases in the literature affected plastic dental follicle is important in order to distinguish this
young individuals [3–7], involving permanent first and sec- condition from other odontogenic tumors that present differ-
ond molars [3, 7–9]. ent pathogenesis and recurrence potential [8].
The radiographic appearance of hyperplastic dental We use this case of hyperplastic dental follicle as an
follicle is characterized by well-circumscribed radiolucent opportunity to review the literature focusing on the etio-
area with sclerotic borders surrounding the crown of an pathogenesis and clinicopathological features of this hamar-
unerupted tooth, frequently mimicking dentigerous cyst [4, tomatous lesion in young patient.
10]. Delayed or tooth eruption failure has been associated
with this hamartomatous lesion. Microscopically, the hyper-
plastic dental follicle consists of fibrous connective tissue con- 2. Case Presentation
taining odontogenic epithelium, multinucleated giant cells,
and calcification foci [3, 8, 11]. A 14-year-old black male presented to the Dental Clinic at
Recently, some authors described the occurrence of mul- Escola Superior São Francisco de Assis, Espı́rito Santo, Brazil,
tiple calcifying hyperplastic dental follicles associated with with the chief complaint of absence of a molar tooth in the
multiple unerupted teeth affecting young male patients inferior dental arch. His medical history was unremarkable.
2 Case Reports in Dentistry

Figure 1: Enlargement of gingival area associated with unerupted right mandibular second molar.

(a) (b)

Figure 2: Radiographic aspect of the unerupted teeth. (a) Sectioned panoramic radiography: radiolucent well-defined area surrounding the
crown of tooth 47, extending to apical region in the anterior area. (b) Periapical radiography: delicate sclerotic border, normal enamel and
radicular formation, and absence of visible calcifications in pericoronal space.

Clinical examination showed enlargement of the gingi- fibers for vimentin (Figure 5). Based on clinical, radiological,
val region of unerupted right mandibular second molar and microscopic examination, the diagnosis established was
(Figure 1). hyperplastic dental follicle. The patient is under clinical
Radiographic examination revealed a radiolucent well- control and the tooth is in eruption process.
delimited area surrounding the crown of the unerupted tooth,
with one-third of root formed and calcified. In addition,
periapical radiography showed that dental follicle was limited 3. Review of the Literature
by thin sclerotic border with absence of visible calcifications.
We reviewed the English literature for hyperplastic dental
The radicular and enamel formation of the tooth were normal
follicle occurring in young patients (under 21 years old) and
(Figure 2). The clinical diagnosis was dentigerous cyst. No
the following inclusion criteria were used: (1) patients under
other radiologic abnormalities involving teeth were observed.
21 years old, (2) complete description of the teeth involved,
Surgical marsupialization of the lesion was performed under
(3) absence of systemic diseases, and (4) microscopic analysis
local anaesthesia exposing the dental crown and no fluid
of the dental follicle. Based on these criteria, we selected 13
accumulation was observed. The pericoronal follicle was
reports, including the present case, and the data are summa-
submitted to the Laboratory of Pathology of Bauru School of
rized in Table 1.
Dentistry, University of São Paulo, for histopathological
analysis (Figure 3).
Microscopic examination revealed noninflamed fibrous 4. Discussion
connective tissue with dense collagen, fusiform cells, and
giant multinuclear cells (Figures 4(a) and 4(b)). Odontogenic Over the past decades, some clinical reports had explored the
epithelial islands were scattered randomly and surrounded characteristics of the hyperplastic dental follicle associated
by calcification focus (Figure 4(c)). Some of these epithelial with delayed or tooth eruption failure, in an attempt to cat-
islands presented squamous metaplasia. Reduced enamel egorize it as a pathological distinct entity [8, 11]. Radiograph-
epithelium was not identified. The immunohistochemical ically, normal pericoronal follicle is considered to be in the
analysis showed that the multinucleated giant cells were neg- range of 2-3 mm [16]. Although the radiographic evidence of
ative for CD-68, HHF-35 and strongly positive for vimentin. a radiolucency around the crown of an unerupted tooth
The blood vessels were positive for HHF-35 and the collagen of no more than 5 mm in width is strongly suggestive of
Case Reports in Dentistry 3

(a) (b)

Figure 3: (a) Incisional biopsy of the lesion. (b) Fresh fragment of gingival and follicular tissues after removal.

(a) (b)

(c)

Figure 4: Histological aspect of the follicle: (a) pericoronal dental tissue with dense connective tissue; (b) multinucleated giant cells (arrows);
(c) islands of odontogenic epithelium (large arrow) and calcified focus (small arrow) (H&E; (a) = 50x, (b) = 400x, and (c) = 200x).

dentigerous cyst or odontogenic tumors, the hyperplastic Of the 18 patients with hyperplastic dental follicles found
dental follicle should be considered in the clinical diagnosis in the literature and that matched our inclusion criteria
[1, 17, 18]. (Table 1), most of them involved multiple teeth (14 cases
The etiopathogeny of hyperplastic dental follicle is reported). Only 4 patients under 21 years presented with sin-
unclear and in some reported cases the teeth affected pre- gle affected tooth, including our case reported. Regarding the
sented defective enamel formation such as amelogenesis multiple cases, the number of involved teeth ranged from 4 to
imperfecta [5, 7, 10] or enamel dysplasia [9]. 16 different teeth. Two authors reported patients with hyper-
According to our English review, the patients’ age range plastic dental follicles affecting two distinct teeth in the same
varied from 5 years to 19 years old (Table 1) and the rela- individual (first and second mandibular molars) and it was
tionship between female and male was 1 : 1.4, showing higher not considered multiple occurrence [3, 8].
occurrence of hyperplastic dental follicle in young male than When the characteristics of enamel mineralization were
in female gender. analyzed we verified that the teeth crowns were normal in
4 Case Reports in Dentistry

(a) (b)

(c)

Figure 5: Microscopic immunohistochemical features: (a) the giant multinucleated cells showed negative reactivity for antibody anti-CD-68
(arrow) indicating no monocyte origin; (b) the blood vessels were positive for anti-HHF-35 but the giant multinucleated cells were negative for
this antibody (arrow); (c) strong vimentin immunopositivity of the giant multinucleated cells was observed indicating mesenchymal origin
(arrow) ((a) = 400x, (b) = 400x, and (c) = 400x).

14 patients with hyperplastic dental follicle and 4 patients immunohistochemistry is not necessary to establish the final
presented with amelogenesis imperfecta or enamel dysplasia diagnosis, in the present case, the immunoprofile of the
(Table 1). Furthermore, it is important to reinforce that giant cells was investigated. The hyperplastic dental follicle
there is no relationship between multiple hyperplastic dental showed typical characteristics such as positive reactivity of
follicle and genetic syndromes involving multiple unerupted the collagen for vimentin and of the blood vessels for HHF-35.
teeth such as Gardner syndrome and cleidocranial dysplasia Furthermore, the multinucleated giant cells were negative for
[19]. CD-68, HHF-35 and strongly positive for vimentin, confirm-
Microscopic findings in hyperplastic dental follicle ing the mesenchymal origin of these cells. According to pre-
include the presence of fibrous connective tissue, wavy col- vious study [11], the multinucleated giant cells in hyperplastic
lagen fibers, strands and islands of odontogenic epithelium, dental follicle are fibroblasts and seem to be associated with
multinucleated giant cells, and varying sizes of basophilic the production of myxoid matrix. It is important to reinforce
mineralized areas presenting round cementum-like or psam- that the presence of stellate and giant fibroblasts is commonly
momatous calcifications [12, 19]. In Table 1, we described all detected in gingival tissue of young individuals [2].
cases of hyperplastic dental follicle with calcification foci Since 1980, when Gardner [1] first described the hyper-
in patients under 21 years. These calcified areas varied plastic dental follicle, there are difficulties regarding the
in size, amount, and microscopic appearance, some of pathological differentiation of this hamartomatous lesion
them resembling woven bone, osteodentin, and cementum with other odontogenic tumors, particularly with central
while others presenting psammomatous calcification and odontogenic fibroma. Both lesions present similar clinical
Liesegang ring-like structures [5, 9, 12, 13, 17]. The presence of and histopathological characteristics and the distinction may
morphologically distinct calcifications associated with nests be challenging [1, 17, 21, 22]. It has been considered that
of odontogenic epithelium is frequently observed in normal hamartomatous lesion such as hyperplastic dental follicle
pericoronal dental tissues or in odontogenic cyst and tumors presents a less aggressive behavior than tumors as odon-
[20, 21]. Moreover, according to our review (Table 1), calci- togenic fibroma and that the clinical outcome may be an
fication areas seem to be a common finding in hyperplastic important tool for diagnosis [8, 21].
dental follicle of the young individuals. The mechanism by which some follicles become hyper-
The histopathological diagnosis of hyperplastic dental fol- plastic and cause retarded eruption or impaction of teeth is
licle is based on hematoxylin eosin routine staining. Although not completely known. However, it has been suggested that
Case Reports in Dentistry 5

Table 1: Review of the clinical and microscopic characteristics found in hyperplastic dental follicles of young patients.

Enamel Single or multiple

Authors/year Age/sex Teeth affected∗ Calcification
alteration occurrence
van Heerden et al., 1990 [10] 14-year-old Present Amelogenesis
13 different teeth Multiple
female imperfecta
18, 17, 14, 13, 12,
Gomez et al., 1998 [17] 15-year-old male 23, 24, 27, 33, 43, Present Unremarkable Multiple
and 48
5-year-10-
month-old 36 and 46 Present Unremarkable Not specified
Onishi et al., 2003 [3] female
6-year-old Present
36 Unremarkable Single
female
Walker et al., 2004 [4] 6-year-old Present
85 Unremarkable Single
female
Feller et al., 2006 [9] 17, 27, 37, 35, 33, Present Enamel
12 years old Multiple
45, and 47 displasia
Nikitakis et al., 2006 [8] 14-year-old male 37 and 47 Present Unremarkable Not specified
17, 16, 15, 14, 24,
Roquebert et al., 2008 [5] 25, 26, 27, 37, 36, Present Amelogenesis
10-year-old male Multiple
35, 34, 44, 45, imperfecta
46, and 47
8-year-9-
Quintella et al., 2008 [6] month-old 36 Present Unremarkable Single
female
15-year-old male 13, 23, 33, and 43 Absent Unremarkable Multiple
Sun et al., 2010 [24] 12-year-old
13, 23, 33, and 43 Absent Unremarkable Multiple
female
17, 13, 23, 27, 37, Present
11-year-old male Unremarkable Multiple
36, and 47
17, 13, 23, 27, 37, Present
14-year-old male Unremarkable Multiple
Cho et al., 2011 [12] and 47
11-year-old male 17, 27, 37, and 47 Present Unremarkable Multiple
17, 15, 25, 27, 37, Present
15-year-old male Unremarkable Multiple
35, and 47
18, 17, 27, 28, 38,
Jamshidi et al., 2013 [13] 19-year-old male 37, 33, 43, 47, Present Unremarkable Multiple
and 48
O’Connell et al., 2014 [7] 7-year-old 16, 26, 36, and Present Amelogenesis
Multiple
female 46 imperfecta
Present case 14-year-old male 47 Present Unremarkable Single
∗
The ISO system by the World Health Organization was used for dental notation.

the presence of hamartomatous pericoronal areas as observed causing delayed eruption should be removed in order to
in our case reported may induce active tissue remodeling and release the teeth from impaction and the routine microscopic
result in fibrosis [2, 11]. On the other hand, Kim et al. [23] examination should be performed in order to differentiate the
confirmed that the turnover of extracellular matrix is neg- hamartomatous follicle from other neoplastic odontogenic
atively affected by downregulation of metalloproteinases in tumors [14].
hyperplastic dental follicle when compared to normal peri- Based on present case and in our review, the diagnosis
coronal follicle, suggesting this factor as the responsible for of hyperplastic dental follicle in young patients should be
abnormal tooth eruption. established when a clinical history of delayed tooth eruption
Our case reported and other clinical cases reviewed rein- exhibits radiographic image of enlarged pericoronal space in
force the need to perform a careful radiograph examination association with distinct microscopic features. So, the clini-
in young patients with single hyperplastic dental follicle in cians should be aware of the existence of this pathology in
order to investigate the possible multiple occurrence of this young individuals and systematically send removed pericoro-
lesion affecting unerupted teeth. Whenever possible, follicles nal follicles to microscopic analysis.
6 Case Reports in Dentistry

In conclusion, we presented a literature review and a [6] C. Quintella, J. H. Damante, G. Janson, E. Guerra, M. R. de
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tial histogenetic source of intraosseous epithelial odontogenic
tumors,” Journal of Oral Pathology and Medicine, vol. 36, no. 4,
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