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\begin{document}

\title{A case of peripheral odontogenic fibroma arising in the mandibular
premolar region of a teenager}



\author[1]{Kie Yamashiro}%
\author[2]{Katsuhisa Sekido}%
\author[1]{Yasushi Hariya}%
\author[1]{Michiko Okita}%
\author[1]{Masashi Harada}%
\author[3]{Masaharu Tatetsu}%
\affil[1]{Teine Keijinkai Hospital}%
\affil[2]{Toyama Red Cross Hospital}%
\affil[3]{Okinawa Prefectural Miyako Hospital}%


\vspace{-1em}



  
  \date{June 09, 2024}


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\selectlanguage{english}
\begin{abstract}
Peripheral odontogenic fibroma (POdF) is a rare, benign, and
ectomesenchymal tumor. We report a case of a 15-year-old girl who
developed POdF in the mandible. The lesion was resected, including the
periosteum. Histopathological findings revealed a small mass and
cord-like epithelium. No sign of post-operative recurrence appeared
after 16 months.%
\end{abstract}%



\sloppy


\textbf{1. Introduction}

Peripheral odontogenic fibroma (POdF) is a rare ectomesenchymal, benign
odontogenic tumor that most often occurs in adult women, frequently in
the mandible and anterior maxilla
regions.\textsuperscript{1,2}Histopathologically, the tumor consists of
fibrotic tissue with odontogenic epithelium and hard tissue showing
varying degrees of calcification, but its expression is
rare.\textsuperscript{3,4}

Along with a review of the literature, here we report a case of POdF
arising in the mandibular premolar region of a teenager.

\textbf{2. Case story / examination}

A 15-year-old girl with a chief complaint of swelling in the right
mandibular premolar region was referred to our department. She had a
history of surgery for a congenital ear fistula. The patient first
recognized a painless mass in her mandible at 6 years of age. On this
occasion, she was referred to a local dental clinic for the treatment of
dental caries and then to our hospital for the treatment of the
mandibular tumor. Intraoral examination revealed a well-defined,
painless, elastic hard mass, measuring 17\selectlanguage{ngerman}×17 mm in size, between the
right mandibular canine and first premolar (Figure 1). Panoramic
radiography showed a radiolucent area in the alveolar bone at the same
location (Figure 2). In addition, sandy calcification was observed
inside the lesion. Computed tomography (CT) revealed compressive bone
resorption of the alveolar bone between the mandibular right canine and
first premolar (Figure 3). Magnetic resonance imaging (MRI) revealed an
endophytic appearance of a well-defined enhancing mass on the right side
of the mandible on T2-weighted imaging (Figure 4).

A biopsy was performed under local anesthesia and a benign tumor with
fibroblast proliferation was diagnosed. Tumor resection was performed
under general anesthesia. The tumor was detached from the surrounding
bone surface and resected, including the periosteum. A bone depression
was observed around the tumor, and the bone surface was smooth. The
tumor was 17×17 mm in size and had a smooth surface. Histopathological
examination revealed spindle-shaped fibroblast-like cells that spread
subcutaneously. Scattered cord-like and lump-like epithelial components
were observed in the fibrous tissue near the base of the tumor (Figure
5A). No atypical cells or nuclear fission were observed. Cementum-or
bone-like calcifications were observed in the deep area of the tumor.
Epithelial components resembled Marasse's remaining epithelial or
Hertwig epithelial sheath and were positive for cytokeratin (CK) CK19
staining (Figure 5B, C). There were no signs of post-operative
recurrence after 16 months.

\textbf{3. Discussion}

Gardner described POdF in comparison with central odontogenic fibroma
and histopathologically separated it into peripheral ossifying
fibroma.\textsuperscript{5} POdF is a rare odontogenic tumor with
gradual growth consisting of a hard, elastic, smooth gingival
mass.\textsuperscript{1,3,4}

The onset age of POdF is 5 months to 84 years, and there is seemingly a
predominant peak in middle age, specifically, during patients'
forties.\textsuperscript{1,2,6,7} The most common sites to develop POdF
are known to be the anterior maxilla and mandible in adults, while some
reports show that POdF also occurs frequently in the mandibular canine
to premolar region.\textsuperscript{7,8} A total of 25 reports of POdF
occurring in patients younger than 19 have been published, of which, 12
reported POdF development in the mandible (Table
1).\textsuperscript{6,7,9--18} Among those, two cases, including the
present case, reported POdF occurring in the mandibular premolar region
of teenagers.\textsuperscript{10}

Histopathologically, POdF is characterized by odontogenic epithelium
scattered to various degrees in the fibrous
substrate.\textsuperscript{3,5,7} Moreover, bone-like, dentin-like, or
cementum-like hard tissue formation is observed, and it is classified
into epithelium-poor or epithelium-rich type according to the degree of
content of the dentin epithelium.\textsuperscript{19} Regarding the hard
tissue inside the lesion, bone-like and cementum-like hard tissue
formation have a reported frequency of 28.3\% and 15.2\%,
respectively.\textsuperscript{6} In the present case, a region was
described histopathologically as rich in cellular components as well as
a mixture of small mass and cord-like epithelial components similar to
Marasse's epithelial remnants and Hertwig epithelial sheath. Moreover,
bone-like calcification was observed in the deep areas of the tumor.

In this sense, there are so far only four reports of POdF with hard
tissue inside the lesion among teenagers.\textsuperscript{17} Although
few reports have been published on hard tissue formation in POdF, it has
been suggested that the odontogenic epithelium inside the lesion may
induce hard-tissue formation, which requires a lengthy time to develop
in teenagers.\textsuperscript{17} Thus, it has been speculated that the
frequency of calcification in POdF might be low.

Some reports of CK-positive cells in the odontogenic epithelium of POdF
exist.\textsuperscript{20} In the present case, CK19-positive epithelial
components resembled Marasse's epithelial remnants and Hertwig
epithelial sheath.

POdF was hypothesized to originate from the periodontal ligament, tooth
sac, and dental papilla.\textsuperscript{17} Central odontogenic fibroma
originates from the periodontal ligament or tooth sac according to the
2005 World Health Organization classification, while other authors have
reported the possibility of periodontal ligament origin as
well.\textsuperscript{19} We considered periodontal ligament or tooth
sac as the origin of the tumor described here for the following reasons:
the tumor was observed during the tooth-replacement period, tumor growth
was observed from the neck of the adjacent tooth, fibroblast growth
around the tumor did not resemble that of dental papilla, and Marasse's
epithelial remnants and Hertwig epithelial sheath-like cells were
observed.

The rate of POdF recurrence after surgery is reportedly
low.\textsuperscript{17,21} However, some studies have reported an early
recurrence rate of 50\% (29/58 cases).\textsuperscript{2,22,23}

\textbf{4. Conclusions}

We resected the tumor along with the periosteum and preserved the
adjacent tooth; there were no signs of recurrence at 14 months after the
operation. For POdF that develop in young people, long-term follow-up is
necessary in consideration of the risk of recurrence.

\textbf{Authorship list:}

Author Contributions:

Kie Yamashiro , Katsuhisa Sekido, Yasushi Hariya contributed to the
manuscript preparation. Michiko Okita, Masashi Harada, Yasushi Hariya,
and Masaharu Tatetsu contributed to the patient management. All authors
read and approved the final manuscript.

\textbf{Acknowledgments:} The authors would like to thank Dr. Eiji
Nakayama, Professor at Health Sciences University of Hokkaido, for
assistance with diagnostic imaging, and Dr. Tomoyuki Oouchi, Chief of
the Department of Pathology at Keiyukai Sapporo Hospital, for assistance
with the pathological investigation.

\textbf{Conflict of interest:}

The authors have no conflicts of interest to declare.

\textbf{Abbreviations:} CK, cytokeratin; CT, computed tomography; MRI,
magnetic resonance imaging; POdF, peripheral odontogenic fibroma

\textbf{Consent}

Confirmation that the author has obtained written informed consent from
patient(s).

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\textbf{Table 1.} Reports of peripheral odontogenic fibroma occurring in
patients under 19 years of age.

\textbf{Figure legends}

\textbf{Figure 1. Intraoral view. (A, B)} Intraoral view revealed a
well-defined, painless, elastic hard mass, measuring 17×17 mm in size
between the right mandibular canine and first premolar.

\textbf{Figure 2. Panoramic X-ray.} Panoramic radiography showed a
radiolucent area in the alveolar bone between the right mandibular
canine and first premolar.

\textbf{Figure 3. Computed tomography findings} . (\textbf{A, B} )
Compressive bone resorption of the alveolar bone between the right
canine and first premolar of the mandible.

\textbf{Figure 4. Magnetic resonance imaging findings.} Endophytic
appearance of a well-defined enhancing mass on the right side of the
mandible on T2-weighted images.

\textbf{Figure 5. Histopathological findings.} (\textbf{A} ) Scattered
cord-like and lump-like epithelial components were observed in the
fibrous tissue near the base of the tumor (H-E staining, ×40).

(\textbf{B} ) Cementum- or bone-like calcifications were observed.
Epithelial components resembled Marasse's remaining epithelial or
Hertwig epithelial sheath and were positive CK19 (H-E staining, ×400;
CK19 staining, ×40).\selectlanguage{english}
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