Oral Fibrolipoma-A Rare Histological Entity: Report of 3 Cases and Review of Literature

Abstract

Lipomas are rare benign soft tissue mesenchymal neoplasms in the oral cavity, representing 1% of all benign oral tumors. Fibrolipoma (FL), an uncommon, histological variant of the classic lipoma, mostly affects the buccal mucosa. Very few cases of FL have been reported in the English literature. To the best of our knowledge a review of the English literature showed 33 cases of FL affecting the oral cavity.
The diagnosis and differentiation of FL with clinically similar lesions such as fibroma, mucocele and pleomorphic adenoma are very essential for a correct treatment plan and complete follow-up. Due to the rarity of oral cavity fibrolipoma reports, three cases and a review of literature is presented here.

Full text

2010; Vol. 7, No. 4
Case Report
Oral Fibrolipoma-A Rare Histological Entity: Report of 3
Cases and Review of Literature
B.S. Manjunatha
1

, G.S. Deepak Pateel
2
, V. Shah
3
1
Associate Professor, Deptment of Oral Pathology and Microbiology, Sumandeep Vidyapeeth, K M Shah Dental College and Hos-
pital, Pipariya,Vadodara, Gujarat State, India.
2
Senior lecturer
, Department of Oral Pathology and Microbiology, Sumandeep Vidyapeeth, K M Shah Dental College and Hospi-
tal, Pipariya,Vadodara, Gujarat State, India.
3
Professor,
Department of Oral Pathology and Microbiology, Sumandeep Vidyapeeth, K M Shah Dental College and Hospital,
Pipariya,Vadodara, Gujarat State, India.



Corresponding author:
B.S. Manjunatha, Associate
Professor, Dept of Oral Pathol-
ogy, KM Shah Dental College
Sumandeep Vidyapeeth Pi-
pariya--391760 Waghodia
Vadodara, Gujarat, India.
drmanju26@hotmail.com
Received:15 April 2010
Accepted: 20 October 2010
Abstract:
Lipomas are rare benign soft tissue mesenchymal neoplasms in the oral cavity, represent-
ing 1% of all benign oral tumors. Fibrolipoma (FL), an uncommon, histological variant of
the classic lipoma, mostly affects the buccal mucosa. Very few cases of FL have been re-
ported in the English literature. To the best of our knowledge a review of the English lit-
erature showed 33 cases of FL affecting the oral cavity.
The diagnosis and differentiation of FL with clinically similar lesions such as fibroma,
mucocele and pleomorphic adenoma are very essential for a correct treatment plan and
complete follow-
up. Due to the rarity of oral cavity fibrolipoma reports, three cases and a
review of literature is presented here.
Key Words: Lipoma; Buccal Mucosa; Adipose Tissue Neoplasms; Adipocytes
Journal of Dentistry, Tehran University of Medical Sciences, Tehran, Iran (2010; Vol.7, No.4)
INTRODUCTION
Lipoma is a benign mesenchymal soft tissue
neoplasm of mature adipocytes of which of
about 20% occurs in the head and neck region.
However, oral lipomas comprise of only 1% to
4% of cases and these usually present as
painless, well-circumscribed, slow-growing
submucosal mass or superficial lesions, mainly
in the buccal mucosa.
These are usually composed of mature adipo-
cytes, surrounded by a thin fibrous capsule [2].
Oral lipoma is usually slow growing and rarely
recurs after surgical treatment. Hence, the
prognosis of these benign tumors is considered
good [3, 4]
Histologically, lipomas are classified as simple
lipoma or variants such as fibrolipoma, spindle
cell lipoma, intramuscular or infiltrating li-
poma, angiolipoma, salivary gland lipoma
(sialolipoma), pleomorphic lipoma, myxoid
and atypical lipomas [5,6].
FL of the oral cavity has been infrequently re-
ported. To the best of our knowledge, the re-
view of the English literature till early 2009
revealed a total of 33 cases of intraoral FL
(Table 1).
As oral lipomas are relatively rare, few large
case series have been published in the English
literature [1,7-9].
A review by Fregnani et al. [1] revealed the
buccal mucosa as the most common site of oc-
currence and the tongue as the second most
common site.
One case of FL has been reported in the lower
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Journal of Dentistry, Tehran University of Medical Sciences Manjaunatha et al.
2010; Vol. 7, No. 4
lip area. A male predilection was noted from
the data available in the literature (Table 1).
However, in contrast some have also shown a
female predilection [9].
The time of development of the tumor to diag-
nosis is often unknown but has been reported
to vary from two months to 21 years [10].
The mean age of occurrence of FL is 34 years
with a range from 3 to 56 years. In this present
case report we analyze three cases of fibroli-
poma and discuss the clinico-pathological fea-
tures.
CASE REPORT
Two male patients (cases 1 & 2) came to KM
Shah Dental College and Hospital, Vadodara
from September 2008 to March 2009 and pre-
sented with chief complain of growth in the
right cheek of at least 6 months duration. One
male patient in May 2009 reported to the out
patient department for a set of complete den-
ture for his upper edentulous jaw and was un-
aware of such a lesion on the palate.
The detailed clinical features of all three cases
of oral fibrolipoma comprising age and gender
of the patient, size, appearance and duration of
the lesion are summarized in Table 2. All three
patients were male, with the mean age of 66.6
years (range, 55 to 75 years).
The chief complaint was a solitary painless
nodule in all three cases of which two were
seen on the buccal mucosa (case 1 and 2) and
one was on the palate (case 3) (Fig 1). The size
of the tumor varied from 1 cm to 3.0 cm with a
mean dimension of 1.8 cm. Clinically, all
cases presented as painless, well-
circumscribed, sub-mucosal nodules, soft to
semi-firm in consistency and pale pinkish in
color similar to the adjacent normal mucosa.
The lesion was sessile in two cases (Fig 2) and
pedunculated in one case.
The duration of this lesion ranged from 6 years
to 10 years. It is interesting to note that clini-
cally all cases were diagnosed as ‘fibroma’.
Histopathologically, all cases revealed admix-
ture of round to oval, variably sized typical
adipocytes interspersed with dense collagen
fibers in a connective tissue stroma (Figs 3 &
4). Stratified squamous epithelium covering
the surface was also seen in these cases.
The microscopic diagnosis of ‘fibrolipoma’
was given for all three cases.
A follow-up of 6 months in all three cases
found no evidence of disease.
DISCUSSION
Lipomas are benign soft tissue neoplasms of
adipose tissue origin and are relatively un-
common in the oral cavity, representing about
1% to 5% of all benign oral lesions [1]. The
first description of oral lipoma was provided in
1848 by Roux in a review of alveolar masses
which he referred it as a ‘yellow epulis’ [11].
Lipomas can occur in various anatomic sites
including the major salivary glands and vari-
ous parts of the mouth.
The English literature review showed a vari-
able distribution of these intraoral lipomas but
approximately half were related to the cheek
and the remaining were found in the tongue,
floor of the mouth, lips, palate and gingiva
[12-16] Generally, oral lipomas have been re-
ported to occur in all ages but are frequently
seen after 40 years of age [13].
Hatziotis, in a review of the literature, reported
that 80% of the patients were over 40 years of
age, 64% were over 50 years and 40% over 60
years with an age range of 2-87 years [17].
Fibrolipoma is a benign soft tissue tumor that
rarely occurs in the oral and maxillofacial re-
gions, and is classified as a variant of conven-
tional lipoma by the WHO [18,19].
FL is a histological variant of simple lipo-
maand differs from theclassicvariant,posed of
mature adiposetissue interspersed by bands of
connective tissue.
FL of the oral cavity has been infrequently re-
ported. To the best of our knowledge, the re-
view of the English Fibrolipoma usually litera-
ture till early 2009 revealed a total of33 cases
227

Manjuanatha et al.ORAL FIBROLIPOMA - Report of 3 cases and Review
2010; Vol. 7, No.3
manifests as long-lasting sessile of intraoral
FL. A very recent study showed that 27% of
41 cases of oral lipomas were FL [9].
Fibrolipomas have been reported in the
esophagus, pharynx, colon, trachea, larynx and
other locations [20,21].
round to ovoid sub-mucosal nodules affecting
the buccal mucosa, tongue, labial mucosa, pa-
rotid region and palate, as shown by our litera-
ture review (Table 1).
In this case report, we present three such
rare cases occurring in the oral cavity and a
review of previous published reports (Ta-
ble 1).The present report showed an age
range from 55 to 75 years (mean age, 66.6
years).FL of our series and the literature
review also suggests a male predominance.
Author
Age /Sex
*
Site
Duration
No. of
Cases
Recurrence
Saitoh et al 1995
16 3/F Parotid NA 1 NED 3 years
Dattilo et al 1996
14 45/M Tongue 10 years
1 NA
Epivatianos et al 2000
13 NA Tongue NA 2 NA
Fregnani et al 2003
1 NA Buccal Mu-
cosa NA 18 NED 26.5
months
Furlong MA et al 2004
7 NA Parotid Buccal
mucosa NA 2 NA
Bandéca MC 2007
23
42/M Lower lip NA 1 NED 60 months
Capodiferro S et al 2008
15 43/M Labial mucosa
8 months
1 NED 10 months
Freitas et al 2009
9
56/F Buccal mucosa NA 7 NA
*Agein years, M Male, FFemale, NED--no evidence of disease; NA--not available
Fig 1.
Photograph showing lesion in the
palate in case-3.
Fig 2.
Macroscopic appearance of the
lesion in case-2.
Fig 3.
Microphotograph showing round
to oval shaped adipocytes admixed with
fibrous connective tissue stroma (H & E
Stain, 100X magnification).
Table 1.
Summary of previous reported cases of oral fibrolipoma
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Journal of Dentistry, Tehran University of Medical Sciences Manjaunatha et al.
2010; Vol. 7, No. 4
According to the literature, it is difficult to
value the real incidence of this neoplasm be-
cause it appears as painless and slow-growing
clinical appearance. In reality, the patient re-
fers to the clinician only when it becomes
symptomatic and for aesthetic or functional
problems. In a large series of studies on li-
poma by Fregnani et al and de Feitas et al,
39% and 27% were reported microscopically
as fibrolipoma, respectively [1,9]. Whereas
other similar case series have reported a very
low incidence of fibrolipoma cases [2,13,17].
These differences could be explained by real
racial and geographic characteristics or simply
by different diagnostic criteria.
Table 2.
Clinical details of our cases
The size of the tumor depends on the location
of the lesion, but rarely exceeds 25 mm in di-
ameter [22].
Lipomas are freely mobile in relation to the
surrounding tissues and may clinically have a
semi-lucent yellow color because of the thin
overlying epithelium.
In some cases it is possible to observe the su-
perficial blood vessels as well.
The consistency of this lesion varies from soft
to firm, depending on the quantity and distri-
bution of fibrous tissue and the depthof the
tumor [23].
In some cases, a sensation of fluctuation may
be recognized in these tumors [24].
The clinical differential diagnosis of intra-oral
FL may include many benign mesenchymal
tumors like fibromas, simple lipomas, and mi-
Case
Age/ Gender
*
Site
*
Size Appearance Symptoms Duration Consistency
Case 1 75 /M Right BM
3 cm
Pedunculated None 10 years Soft
Case 2 55 /M Right BM
1 cm
Sessile None 6 months Firm
Case 3 70 /M Soft Palate
1.5 cm
Sessile None 6months Soft
*Agein years, M Male, FFemale, BM Buccal mucosa
Fig 4. Microphotograph showing round to oval shaped adipocytes admixed with dense collagenous fibers and fibroblasts (H
& E Stain, 400X magnification).
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Manjuanatha et al.ORAL FIBROLIPOMA - Report of 3 cases and Review
2010; Vol. 7, No.3
nor salivary gland tumors either benign or ma-
lignant.We diagnosed fibrolipoma based on
the presence of mature adipose tissue inter-
spersed by bands of broad or fascicles of dense
connective tissue fibers with the capsule.
Lipoma and fibrolipoma both are usually well
circumscribed and have a thin capsule. Fibrol-
ipoma differs from the classic variant because
the mature adipose tissue is interspersed by
bands of connective tissue.
The proliferative activity of FL revealed a
greater proliferative rate than other simple
variants which indicates the need for accurate
diagnosis of such variants with high prolifera-
tive activity and further encourages similar
studies [1].
Furthermore liposarcoma of the oral cavity is
exceedingly rare, but this entity cannot be dis-
tinguished from its benign counterpart at clini-
cal examination [15] .Therefore, accurate his-
tological examination is mandatory, and the
differential diagnosis is based on the detection
of a lack of lobular architecture, areas of
prominent fibrosis and, most importantly, on
the presence of multivacuolated adipose cells
with indented nuclei (lipoblasts), which are
typically present in liposarcoma in variable
proportions. The lesion in all three patient was
surgically excise without any complications.
Post- operative follow-up of six months
showed no recurrence in any of the cases. The
data from the literature review showed no evi-
dence of recurrence in any reports and the du-
ration of follow-up ranged from minimum 10
months to maximum 5 years. The average fol-
low-up period in the literature review was ap-
proximately 3 years.
However, two cases of giant FL [25,26] have
not been considered in the present report.
These contradictory findings authenticate the
need for a meticulous analysis of intra-oral FL
cases.
CONCLUSION
Microscopic/histological findings provided by
Oral Pathologists should be combined with
clinical features for accurate diagnosis. FL
represents a distinct clinico-pathologic and
biologic entity with an increased growth po-
tential compared with the classic lipoma and
not associated with any syndrome and with a
low recurrence rate.
The case presented here adds to the existing
few cases of FL reported in English literature.
Lesions appearing alike clinically often pre-
sent diverse and overlapping histopathological
features and in such cases, they pose a diag-
nostic dilemma for general dentists. So, the
authors recommend that histopathological ex-
amination of excised tissue is a gold standard
investigative procedure along with consulta-
tion with an Oral Pathologist for correct diag-
nosis has a very significant role in providing
successful treatment and preventing any ma-
lignant transformation, as recommended ear-
lier. It is very much essential for new cases to
be documented and followed carefully so that
more accurate, better treatment modalities
could be drawn to prevent further damages.
ACKNOWLEDGMENTS
The authors wish to thank Chancellor and Pro-
Chancellor, Sumandeep Vidyapeeth, Pipariya,
Vadodara, India, for all the encouragement and
support given during the study.
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