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Nodular Fasciitis in the Buccal Mucosa: A Case Report
Oral November, Science International, 2004 November 2004, p.89―92 Nodular Fasciitis Copyright © 2004, Japanese Stomatology Society. All Rights Reserved.
89
Nodular Fasciitis in the Buccal Mucosa : A Case Report Tetsuro Ikebe, Yuichi Ogata, Yasuo Takamune, Kazutoshi Ota, Takehisa Obayashi and Masanori Shinohara Department of Oral and Maxillofacial Surgery, Sensory and Motor Organ Sciences, Faculty of Medical and Pharmaceutical Sciences, Kumamoto University （Chief : Professor Masanori Shinohara）
Abstract : A case of nodular fasciitis which arose from the buccal submucosal region is reported. One week after an incisional biopsy, the lesion enlarged alarmingly and protruded from the submucosa. Although a sarcoma was suspected because of rapid growth, the diagnosis of the biopsy was nodular fasciitis showing a haphazard arrangement of plump fibroblasts without atypical mitoses. After complete resection, no signs of recurrence were seen. Key words : nodular fasciitis, oral cavity, immunohistochemistry
Introduction
fibrosarcoma and malignant fibrous histiocytoma2.
Nodular fasciitis is a reactive, non-neoplastic lesion
We report a case of nodular fasciitis in the buccal
in which fibroblast-like cells proliferate rapidly to form
mucosa, which enlarged alarmingly after an incisional
a fibrous mass, and is thought to originate from
biopsy.
muscular fascia1. While the lesion is most common in the upper extremity with the forearm being predis-
Report of a case
posed, 13％ of all cases occur in the head and neck
A 53-year-old male was referred to our department
1
region . The incidence in the oral cavity is extremely
because of a dull pain in the right cheek during
rare.
mastication.
Since nodular fasciitis in the head and neck often
earlier. The patient had no history of oral injury.
develops adjacent to a bony prominence such as the
Clinical examination revealed a non-visible, but pal-
zygomatic arch and the angle of mandible, the reactive
pable well-demarcated mass under the right buccal
growth of fibrous tissue is thought to be initiated by
mucosa. The mass was firm and fixed to the underlying
trauma followed by inflammation. Because of its rapid
subjacent tissues. The overlying mucosa was movable
growth and haphazard proliferation of plump, spindle-
and showed almost normal appearance except for a
shaped fibroblasts, however, nodular fasciitis is often
small erosion facing the right lower second molar（Fig.
mistakenly diagnosed as a malignant tumor such as
1（A）） . There were no sings of odontogenic infection.
The symptom had appeared a month
The skin of the right cheek was intact. Received 2/16/04 ; revised 5/20/04 ; accepted 6/20/04. Grant support : Ministry of Education, Culture, Sports,
Magnetic resonance images（MRI）demonstrated a
Science and Technology, Japan ; No. 15390619
well-limited, homogeneous mass in the right buccal
Requests for reprints : Tetsuro Ikebe, Department of Oral
and Maxillofacial Surgery, Sensory and Motor Organ Sciences, Faculty of Medical and Pharmaceutical Sciences, Kumamoto University, 1―1―1 Honjyo, Kumamoto, Kumamoto 860―8556, Japan, Phone : 096―373―5288, Fax : 096―373―5286, E-mail : [email protected]
submucosal region with high signal intensity in the T1as well as T2-weighted images（Fig. 2） . The lesion seemed to be attached to the anterior border of the masseter muscle.
90
Oral Science International Vol. 1, No. 2
（A）
（B）
Fig. 1 Clinical appearance.（A）The right buccal mucosa before the first biopsy. （B）A mass protruding from the right buccal submucosa after the biopsy.
（A）
（B）
Fig. 2 MRI view of the mass on the right buccal mucosal region.（A）T1-weighted image.（B）T2-weighted image.
Because the clinical and image findings suggested a tumorous lesion, an incisional biopsy was performed. The histological diagnosis of the biopsy was granulation tissue or, alternatively, nodular fasciitis.
One week
later, at the second visit, the lesion had enlarged more than expected and protruded from the buccal mucosa （Fig. 1（B））as if the biopsy had triggered the lesion to grow. The size of the mass was 32 × 22 mm. The rapid increase in size led us to suspect a sarcoma. A second biopsy was undertaken since we were concerned that the first biopsy failed to target the lesion correctly. The second biopsy was also diagnostic of nodular fasciitis. We resected the lesion with the surrounding tissues including a part of the buccinator, masseter and
Fig. 3 The nodular fasciitis mass removed from the right buccal mucosal region.
periosteum of mandible（Fig. 3） , even though simple local excision is the recommended treatment and the
that its origin was the fascia of masseter.
prognosis is excellent1，3. It was non-encapsulated and
The histology showed a haphazard arrangement of
fixed to the anterior border of the masseter, suggesting
spindle-shaped fibroblast-like cells in a myxoid matrix.
November, 2004
Nodular Fasciitis
（A）
91
（B）
Fig. 4 （A）Histological view of the surgical specimen showing a haphazard arrangement of the spindle-shaped cells（hematoxylin-eosin stain, original magnification ×40） .（B）Spindle-shaped cells showing occasional mitoses （hematoxylin-eosin stain, original magnification ×100） . The mitotic cells are indicated by arrows.
（B）
（A）
（C）
Fig. 5 Immunohistochemical staining of nodular fasciitis showing the expression of smooth muscle actin（A）and vimentin（B） （original magnification ×100） .（C）A number of small blood vessels in the lesion were also positive for smooth muscle actin（original magnification ×40） .
Mitotic figures without atypia were common and
（A） （B）） . There were numerous α-SMA-positive small
abundant vascular vessels and inflammatory cell
blood vessels in the lesion（Fig. 5（C）） .
infiltration were also observed（Fig. 4（A）（B）） .
After surgery, the mouth opening of the patient was
Immunohistochemical staining indicated that the
limited because of the buccal scar, but recovered to the
fibroblast-like cells in nodular fasciitis were positive for
normal range a month later. No evidence of recurrence
α-smooth muscle actin（SMA）and vimentin（Fig. 5
was seen at the one-year follow-up visit.
92
Oral Science International Vol. 1, No. 2
Discussion
of nodular fasciitis.
Nodular fasciitis is a benign lesion of uncertain
There are no specific histopathological markers of
etiology, and is also known as pseudosarcomatous
nodular fasciitis to aid the diagnosis.
fasciitis or pseudosarcomatous fibromatosis1. The most
immunohistochemical studies demonstrated that the
common localizations are the upper extremity（49％） ,
spindle fibroblastic cells in nodular fasciitis were
trunk（18％）, head and neck（13％）and lower extremity
positive for SMA and vimentin, but not desmin and
1
Several
（17％）. Nodular fasciitis has been reported to affect
cytokeratin, implying that their origin was myofibro-
males and females equally, commonly seen in young
blasts7，8. The immunostaining in the present study also
adults with 85％ younger than 50 years of age, and
showed that the fibroblast-like cells expressed SMA and
usually has an average diameter of around 2 cm4.
vimentin. In addition, the SMA-positive vessels were
On the basis of its anatomic location, nodular fasciitis
abundant in nodular fasciitis.
can be categorized into three types : subcutaneous,
number of blood vessels may feed the fibroblast-like cell
intramuscular, and intermuscular types1.
While the increasing
Histologi-
and enable to proliferate rapidly, the pathological
cally, it can also be classified into myxoid, cellular, and
relationship between spindle fibroblastic cells and
fibrous subtypes, and is thought to mature from myxoid
vascular smooth muscle cells remains unknown.
to fibrous subtype1.
The present case seems to be
categorized into submucosal（subcutaneous） , cellular subtype.
References 1． Batsakis J. G. : Tumors of the head and neck. Williams &
Although ultrasound, CT and MRI are commonly
Wilkins, Baltimore, 1979 pp. 259―261.
used to evaluate nodular fasciitis, there are no reports
2． Davies H. T., Bradley N., and Bowerman J. E. : Oral
describing specific image appearances for this lesion.
nodular fasciitis. Br J Oral Maxillofac Surg：27：147―151,
Among these imaging modalities, however, MRI may present more information about it. In the MRI, nodular fasciitis of the upper extremity and trunk appeared homogeneous, hyperintense to muscle on T1-weighted images and homogeneous with signal intensity greater than cutaneous fat on T2-weighted images5，6.
The
1989. 3． Martinez-Blanco M., Bagan J. V., Alba J. R., and Basterra J. : Maxillofacial nodular fasciitis : a report of 3 cases. J Oral Maxillofac Surg 60：1211―1214, 2002. 4． Bernstein K. E., and Lattes R. : Nodular（pseudosarcomatous）fasciitis, a nonrecurrent lesion : clinicopathologic study of 134 cases. Cancer 49：1668―1678, 1982.
present case was also hyperintense on T1-weighted as
5． Leung L. Y. J., Shu S. J., Chan A. C. L., Chan M. K., and
well as T2-weighted images, suggesting that the image
Chan C. H. S. : Nodular fasciitis : MRI appearance and
of nodular fasciitis in the oral cavity is similar to that in
literature review. Skeletal Radiol 31：9―13, 2002. 6． Wang X. L., De Schepper A. M. A., Vanhoenacker F., De
the upper extremity and trunk. As in our case, Davies et al. reported the nodular
Raeve H., Gielen J., Aparisi F., Rausin L., and Somville
fasciitis in the parasymphyseal region of the lower jaw
J. : Nodular fasciitis : correlation of MRI findings and
increased in size after an incisional biopsy2. Although surgical intervention may stimulate it to proliferate, preoperative biopsy seems to be inevitable to make a definitive diagnosis of nodular fasciitis. Because of its rarity in the oral cavity, oral surgeous may suspect the rapidly growing nodular fasciitis of a sarcoma.
histopathology. Skeletal Radiol 31：155―161, 2002. 7． Eversole L. R. Christensen R, Ficarra G., Pierleoni L., and Snapp J. P. : Nodular fasciitis and solitary fibrous tumor of the oral region. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 87：471―476, 1999. 8． Shlomi B., Mintz S., Jossiphov J., and Horovitz I. :
It is
Immunohistochemical analysis of a case of intraoral
important to carefully diagnose a mass that rapidly
nodular fasciitis. J Oral Maxillofac Surg 52：323―326,
grows after biopsy to avoid the unnecessary oversurgery
1994.
89
Nodular Fasciitis in the Buccal Mucosa : A Case Report Tetsuro Ikebe, Yuichi Ogata, Yasuo Takamune, Kazutoshi Ota, Takehisa Obayashi and Masanori Shinohara Department of Oral and Maxillofacial Surgery, Sensory and Motor Organ Sciences, Faculty of Medical and Pharmaceutical Sciences, Kumamoto University （Chief : Professor Masanori Shinohara）
Abstract : A case of nodular fasciitis which arose from the buccal submucosal region is reported. One week after an incisional biopsy, the lesion enlarged alarmingly and protruded from the submucosa. Although a sarcoma was suspected because of rapid growth, the diagnosis of the biopsy was nodular fasciitis showing a haphazard arrangement of plump fibroblasts without atypical mitoses. After complete resection, no signs of recurrence were seen. Key words : nodular fasciitis, oral cavity, immunohistochemistry
Introduction
fibrosarcoma and malignant fibrous histiocytoma2.
Nodular fasciitis is a reactive, non-neoplastic lesion
We report a case of nodular fasciitis in the buccal
in which fibroblast-like cells proliferate rapidly to form
mucosa, which enlarged alarmingly after an incisional
a fibrous mass, and is thought to originate from
biopsy.
muscular fascia1. While the lesion is most common in the upper extremity with the forearm being predis-
Report of a case
posed, 13％ of all cases occur in the head and neck
A 53-year-old male was referred to our department
1
region . The incidence in the oral cavity is extremely
because of a dull pain in the right cheek during
rare.
mastication.
Since nodular fasciitis in the head and neck often
earlier. The patient had no history of oral injury.
develops adjacent to a bony prominence such as the
Clinical examination revealed a non-visible, but pal-
zygomatic arch and the angle of mandible, the reactive
pable well-demarcated mass under the right buccal
growth of fibrous tissue is thought to be initiated by
mucosa. The mass was firm and fixed to the underlying
trauma followed by inflammation. Because of its rapid
subjacent tissues. The overlying mucosa was movable
growth and haphazard proliferation of plump, spindle-
and showed almost normal appearance except for a
shaped fibroblasts, however, nodular fasciitis is often
small erosion facing the right lower second molar（Fig.
mistakenly diagnosed as a malignant tumor such as
1（A）） . There were no sings of odontogenic infection.
The symptom had appeared a month
The skin of the right cheek was intact. Received 2/16/04 ; revised 5/20/04 ; accepted 6/20/04. Grant support : Ministry of Education, Culture, Sports,
Magnetic resonance images（MRI）demonstrated a
Science and Technology, Japan ; No. 15390619
well-limited, homogeneous mass in the right buccal
Requests for reprints : Tetsuro Ikebe, Department of Oral
and Maxillofacial Surgery, Sensory and Motor Organ Sciences, Faculty of Medical and Pharmaceutical Sciences, Kumamoto University, 1―1―1 Honjyo, Kumamoto, Kumamoto 860―8556, Japan, Phone : 096―373―5288, Fax : 096―373―5286, E-mail : [email protected]
submucosal region with high signal intensity in the T1as well as T2-weighted images（Fig. 2） . The lesion seemed to be attached to the anterior border of the masseter muscle.
90
Oral Science International Vol. 1, No. 2
（A）
（B）
Fig. 1 Clinical appearance.（A）The right buccal mucosa before the first biopsy. （B）A mass protruding from the right buccal submucosa after the biopsy.
（A）
（B）
Fig. 2 MRI view of the mass on the right buccal mucosal region.（A）T1-weighted image.（B）T2-weighted image.
Because the clinical and image findings suggested a tumorous lesion, an incisional biopsy was performed. The histological diagnosis of the biopsy was granulation tissue or, alternatively, nodular fasciitis.
One week
later, at the second visit, the lesion had enlarged more than expected and protruded from the buccal mucosa （Fig. 1（B））as if the biopsy had triggered the lesion to grow. The size of the mass was 32 × 22 mm. The rapid increase in size led us to suspect a sarcoma. A second biopsy was undertaken since we were concerned that the first biopsy failed to target the lesion correctly. The second biopsy was also diagnostic of nodular fasciitis. We resected the lesion with the surrounding tissues including a part of the buccinator, masseter and
Fig. 3 The nodular fasciitis mass removed from the right buccal mucosal region.
periosteum of mandible（Fig. 3） , even though simple local excision is the recommended treatment and the
that its origin was the fascia of masseter.
prognosis is excellent1，3. It was non-encapsulated and
The histology showed a haphazard arrangement of
fixed to the anterior border of the masseter, suggesting
spindle-shaped fibroblast-like cells in a myxoid matrix.
November, 2004
Nodular Fasciitis
（A）
91
（B）
Fig. 4 （A）Histological view of the surgical specimen showing a haphazard arrangement of the spindle-shaped cells（hematoxylin-eosin stain, original magnification ×40） .（B）Spindle-shaped cells showing occasional mitoses （hematoxylin-eosin stain, original magnification ×100） . The mitotic cells are indicated by arrows.
（B）
（A）
（C）
Fig. 5 Immunohistochemical staining of nodular fasciitis showing the expression of smooth muscle actin（A）and vimentin（B） （original magnification ×100） .（C）A number of small blood vessels in the lesion were also positive for smooth muscle actin（original magnification ×40） .
Mitotic figures without atypia were common and
（A） （B）） . There were numerous α-SMA-positive small
abundant vascular vessels and inflammatory cell
blood vessels in the lesion（Fig. 5（C）） .
infiltration were also observed（Fig. 4（A）（B）） .
After surgery, the mouth opening of the patient was
Immunohistochemical staining indicated that the
limited because of the buccal scar, but recovered to the
fibroblast-like cells in nodular fasciitis were positive for
normal range a month later. No evidence of recurrence
α-smooth muscle actin（SMA）and vimentin（Fig. 5
was seen at the one-year follow-up visit.
92
Oral Science International Vol. 1, No. 2
Discussion
of nodular fasciitis.
Nodular fasciitis is a benign lesion of uncertain
There are no specific histopathological markers of
etiology, and is also known as pseudosarcomatous
nodular fasciitis to aid the diagnosis.
fasciitis or pseudosarcomatous fibromatosis1. The most
immunohistochemical studies demonstrated that the
common localizations are the upper extremity（49％） ,
spindle fibroblastic cells in nodular fasciitis were
trunk（18％）, head and neck（13％）and lower extremity
positive for SMA and vimentin, but not desmin and
1
Several
（17％）. Nodular fasciitis has been reported to affect
cytokeratin, implying that their origin was myofibro-
males and females equally, commonly seen in young
blasts7，8. The immunostaining in the present study also
adults with 85％ younger than 50 years of age, and
showed that the fibroblast-like cells expressed SMA and
usually has an average diameter of around 2 cm4.
vimentin. In addition, the SMA-positive vessels were
On the basis of its anatomic location, nodular fasciitis
abundant in nodular fasciitis.
can be categorized into three types : subcutaneous,
number of blood vessels may feed the fibroblast-like cell
intramuscular, and intermuscular types1.
While the increasing
Histologi-
and enable to proliferate rapidly, the pathological
cally, it can also be classified into myxoid, cellular, and
relationship between spindle fibroblastic cells and
fibrous subtypes, and is thought to mature from myxoid
vascular smooth muscle cells remains unknown.
to fibrous subtype1.
The present case seems to be
categorized into submucosal（subcutaneous） , cellular subtype.
References 1． Batsakis J. G. : Tumors of the head and neck. Williams &
Although ultrasound, CT and MRI are commonly
Wilkins, Baltimore, 1979 pp. 259―261.
used to evaluate nodular fasciitis, there are no reports
2． Davies H. T., Bradley N., and Bowerman J. E. : Oral
describing specific image appearances for this lesion.
nodular fasciitis. Br J Oral Maxillofac Surg：27：147―151,
Among these imaging modalities, however, MRI may present more information about it. In the MRI, nodular fasciitis of the upper extremity and trunk appeared homogeneous, hyperintense to muscle on T1-weighted images and homogeneous with signal intensity greater than cutaneous fat on T2-weighted images5，6.
The
1989. 3． Martinez-Blanco M., Bagan J. V., Alba J. R., and Basterra J. : Maxillofacial nodular fasciitis : a report of 3 cases. J Oral Maxillofac Surg 60：1211―1214, 2002. 4． Bernstein K. E., and Lattes R. : Nodular（pseudosarcomatous）fasciitis, a nonrecurrent lesion : clinicopathologic study of 134 cases. Cancer 49：1668―1678, 1982.
present case was also hyperintense on T1-weighted as
5． Leung L. Y. J., Shu S. J., Chan A. C. L., Chan M. K., and
well as T2-weighted images, suggesting that the image
Chan C. H. S. : Nodular fasciitis : MRI appearance and
of nodular fasciitis in the oral cavity is similar to that in
literature review. Skeletal Radiol 31：9―13, 2002. 6． Wang X. L., De Schepper A. M. A., Vanhoenacker F., De
the upper extremity and trunk. As in our case, Davies et al. reported the nodular
Raeve H., Gielen J., Aparisi F., Rausin L., and Somville
fasciitis in the parasymphyseal region of the lower jaw
J. : Nodular fasciitis : correlation of MRI findings and
increased in size after an incisional biopsy2. Although surgical intervention may stimulate it to proliferate, preoperative biopsy seems to be inevitable to make a definitive diagnosis of nodular fasciitis. Because of its rarity in the oral cavity, oral surgeous may suspect the rapidly growing nodular fasciitis of a sarcoma.
histopathology. Skeletal Radiol 31：155―161, 2002. 7． Eversole L. R. Christensen R, Ficarra G., Pierleoni L., and Snapp J. P. : Nodular fasciitis and solitary fibrous tumor of the oral region. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 87：471―476, 1999. 8． Shlomi B., Mintz S., Jossiphov J., and Horovitz I. :
It is
Immunohistochemical analysis of a case of intraoral
important to carefully diagnose a mass that rapidly
nodular fasciitis. J Oral Maxillofac Surg 52：323―326,
grows after biopsy to avoid the unnecessary oversurgery
1994.