CASE REPORT
Angiomyxolipoma of Tongue
ABSTRACT
Manas Bajpai, Betina Chandolia and Manika Arora
Angiomyxolipoma is a rare histological variant of lipoma, characterized by proliferation of adipose tissue associated with
a variable amount of myxoid stroma with numerous thick- and thin-walled blood vessels. An exhaustive literature review
could reveal only 12 cases of angiomyxolipoma affecting subcutaneous tissue, subungual area and spermatic cord. In oral
cavity, only 2 cases of angiomyxolipoma have been reported to date, one case in the buccal mucosa and one case in the
floor of the mouth. Hence, this is only the third case of angiomyxolipoma affecting oral cavity and the first case of
angiomyxolipoma of the tongue. We report a case of a 51-year male presenting with a swelling on the right lateral border
of the tongue. The lesion was excised and microscopical examination confirmed the diagnosis of angiomyxolipoma.
Key Words: Angiomyxolipoma. Lipoma. Tongue.
INTRODUCTION
Angiomyxolipoma is a rare variant of lipoma, first
reported by Mai et al.1 Angiomyxolipoma shows a frank
predilection for males with only 2 cases reported in
females till date. It is characterized by proliferation of
adipose tissue with myxoid stroma and numerous
dilated vascular channels.2 Review of literature revealed
only 2 cases of intraoral angiomyxolipoma, one each in
buccal mucosa,3 and floor of the mouth.4 This is the first
case of angiomyxolipoma affecting the tongue. Surgical
excision is the preferred treatment modality; no
recurrence has been reported. This case report
addresses the importance of the definitive diagnosis of
angiomyxolipoma with an emphasis on histopathological
differential diagnoses and differentiating features.
proliferating blood vessels (Figures 2 and 3). Based on
histological examination, final diagnosis of angiomyxolipoma was rendered. Follow-up period of 2 years
was uneventful.
CASE REPORT
A 51-year male patient presented to the Department of
Oral and Maxillofacial Surgery, for the evaluation of a
painless swelling of one year duration on his right lateral
border of the tongue. Intraoral examination revealed
3 x 2 cm, well circumscribed swelling on the right lateral
border of the tongue, without any sign of ulceration.
Patient had slight difficulty in tongue movements
(Figure 1). The patient underwent complete excision of
the lesion under local anesthesia. The cut surface
showed yellowish mass with gelatinous material.
Histological examination of formalin-fixed, paraffin
embedded sections stained by Hematoxylin and Eosin
(H&E) stain revealed a network of benign mature
adipocytes with abundant myxoid areas and numerous
Figure 1: Clinical picture of the lesion.
Department of Oral and Maxillofacial Pathology, NIMS Dental
College, Jaipur, India.
Correspondence: Dr. Manas Bajpai, Assistant Professor,
Department of Oral and Maxillofacial Pathology, NIMS Dental
College, Jaipur, India.
E-mail: [email protected]
Received: June 01, 2016; Accepted: January 18, 2017.
252
Figure 2: Network of mature
adipocytes with proliferating
blood vessels and myxoid areas
(Hematoxylin and Eosin staining
X200).
Figure 3: Benign fat cells with numerous
proliferating capillaries and extensive
myxoid areas. (Hematoxylin and Eosin
staining X400).
Journal of the College of Physicians and Surgeons Pakistan 2017, Vol. 27 (4): 252-253
Angiomyxolipoma of tongue
Table I: Differential diagnoses and differentiating features of angiomyxolipoma.3,6
Diagnosis
Differentiating features
Myxolipoma
Myxolipoma consists of mature fat cells and areas of myxoid degeneration but lacks proliferating blood vessels.
Angiolipoma
Angiolipoma consists of proliferating blood vessels but lacks myxoid areas.
Spindle cell lipoma (SCL)
Angiomyolipoma
Angiofibroma
Lipoblastoma
Myxoid liposarcoma
SCL consists of spindle cells and pleomorphic cells, vascular channel may not be pronounced.
Angiomyolipoma consists of smooth muscle cells and proliferating blood vessels but lacks myxoid areas.
Angiofibroma lacks myxoid areas and mature fat cells.
Lipoblastoma characteristically consists of lipoblast and lacks proliferating blood vessels and myxoid areas.
Myxoid liposarcoma consists of lipoblast cells, show as cellular pleomorphism, increased mitosis and abnormal mitotic figures.
DISCUSSION
Lipomas are common mesenchymal tumors.5 In 1996,
Mai et al. reported a rare variant of lipoma of spermatic
cord in a 34-year male. Histologically, the tumor was
composed of network of mature fat cells with areas of
myxoid degeneration and proliferating blood vessels, as
it was designated as angiomyxolipoma.1 Since then,
only 12 cases of angiomyxolipomas have been reported
in English literature. In 2011, Martinez-Mata et al.
reported first case of angiomyxolipoma in the oral cavity,
occurring on the right buccal mucosa of a 12-year male.3
Since then, only one more case of oral angiomyxolipoma
has been reported in 2014 by Nair et al., occurring on
the floor of mouth of a 70-year male.4 The present case
report describes the third case of oral angiomyxolipoma
and the first case of angiomyxolipoma of the tongue.
Histological differential diagnosis of angiomyxolipoma
includes spindle cell lipoma, myxolipoma, angiolipoma,
angiomyolipoma, cellular angiofibroma, lipoblastoma
and myxoid liposarcoma (Table I). The lesion is usually
asymptomatic and presents as slowly growing, painless
well-demarcated, solitary, subcutaneous mass.1 Wide
local excision is the treatment of choice for angiomyxolipomas and they do not recur.
Angiomyxolipoma is a benign tumor of adipose tissue
characterized by proliferation of adipose tissue with
myxoid stroma and numerous dilated vascular channels.
This case report presents the first case of angiomyxolipoma of tongue. Angiomyxolipoma can show
histological features resembling some benign and
malignant tumors, i.e. myxoid liposarcoma. The
differentiation is important to prevent the aggressive
treatment modalities to be carried out for the treatment
of myxoid liposarcoma. This report also discusses the
microscopical differential diagnoses of angiomyxolipoma
with their differentiating features.
REFERENCES
1. Mai KT, Yazdi HM, Collins JP. Vascular myxolipoma (angiomyxolipoma) of the spermatic cord. Am J Surg Pathol 1996;
20:1145-8.
2. Za´mecnı´k M. Vascular myxolipoma (angiomyxolipoma) of
subcutaneous tissue. Histopathology 1999; 34:180-1.
3. Martı´nez-Mata G, Rocı´o MF, Juan LE, Paes AO, Adalberto
MT. Angiomyxolipoma (vascular myxolipoma) of the oral cavity.
Report of a case and review of the literature. Head Neck Pathol
2011; 5:184e187.
4. Nair S, Ramalingam WVBS, Bharadwaj R, Mohan S, Malik A.
Angiomyxolipoma: Unusual swelling of oral cavity. MJAFI
2013; 1-3.
5. Bajpai M, Kumar M, Agarwal D, Agrawal S, Gupta S, Kumar M.
Osteolipoma of the palate: An unusual presentation. Natl J
Maxillofac Surg 2014; 5:250-1.
6. Bajpai M, Pardhe N. Myxolipoma of oral cavity. Univ Res J
Dent 2016; 6:134-6.
Journal of the College of Physicians and Surgeons Pakistan 2017, Vol. 27 (4): 252-253
253