Full text of DOI:10.1007/BF02910983

INTRODUCTION
Majority of the swellings in the submandibular region are
either lymph node swellings or cases of chronic
sialadenitis involving the submandibular salivary gland. The
clinical picture and the history gives a clue as to the etiology
of the lesion. Hemangiomas involving the submandibular
salivary gland are a rare cause of swelling in this region.
We present a case of arteriovenous hemangioma involving
the submandibular gland.
CASE REPORT
A 20 year old girl presented with a swelling in the left
submandibular region since childhood, which had gradually
increased to its present size. There was no change reported
in the size of the swelling with meals or on bending or
straining. There was no history of sudden increase in the
size of the swelling.
Clinical examination revealed a solitary, firm swelling,
approximately 5x3 cm in size, mobile from side to side
with the overlying skin normal in colour, texture, and a
temperature similar to the surrounding skin.
Fine needle aspiration cytology done twice revealed only
blood. A sialogram revealed a normal duct system with
no evidence of any duct calculi (Fig.I). In view of the
inconclusive fine needle aspiration cytology, a doppler
ultrasound was done. The ultrasound showed multiple,
anechoic, poorly encapsulated masses almost totally
replacing the submandibular salivary gland parenchyma.
The anechoic masses showed scanty blood flow at rest
but filled up with blood on performing the valsalva
manoeuvre. No stone was detected on ultrasound. The
radiologic picture was suggestive of an arteriovenous
malformation (Fig.II).
.
At surgery an ill-defined, poorly encapsulated vascular
mass was found lying around and within the submandibular
gland. A hard stone like structure approximately 0.5 cm
'Professor & Head ,:Senior Resident, 'Senior Resident , Dept. of ENT , G.T.B Hospital and UCMS , Delhi.

and only 12% in the submandibular gland (Me Menamin
M et al 1997). A probable explanation of this difference is
the lack of a well-defined capsule and the presence of
neurovascular structures in the parotid gland.
Hemangiomas are found to occur more frequently in
females. It has been observed that the left side is more
frequently involved (Batsakis JG, 1986), a finding
supported by the case in study.
Arteriovenous hemangiomas can be divided into two
types-one occurring in deep locations and associated with
varying degrees of arteriovenous shunting, the other type
superficial, occurring in the dermis with no significant
shunting.
The deep form occurs mostly in young persons and is
regarded by some as an arteriovenous malformation
(Enzinger FM and Weiss SW 1995). It is thought to be,
due to partial persistence of fetal capillary bed, causing
abnormal connections between the arteries and veins.
These lesions are commonly seen in the head, neck and
the lower extremities. Lesions, which have large shunts,
may
the
in
e a bruit over the mass and raised temperature of
ng skin. In some lesions there may be an increase
if the extremity with enlarged veins.
in diameter was found lying within i e vascular lesion.
The mass was excised in toto and submitted for
histopathological examination. Microscopic examination
revealed medium and large sized arteries and veins lying
in close association with one another (Fig.III). However
no communication between the vessels was demonstrable
in any of the sections.There was a focus of calcification
in a vessel wall seen protruding into the lumen (Fig.IV).
The surrounding submandibular salivary gland parenchyma
showed marked dilatation of vessels and congestion. No
features suggestive of sialadenitis were seen. Based on
these features, a diagnosis of arteriovenous hemangioma
was arrived at.
H
iy, these lesions are difficult to diagnose. There
ies and veins seen lying in close association
tther. Serial sections may document presence
tween them. At times these lesions may
ipillary or cavernous hemangioma (Enzinger
SW, 1995).
ave been mostly seen in soft tissue cavernous
s and should be distinguished from salivary
i (Hopkins R, 1969). Plain radiographs may
multiple calcified phleboliths in upto 2% to
(Enzinger FM & Weiss SW, 1995). In the
diagnosis, ectopic oral calcification including
lymph nodes, myositis ossificans and
arasitic lesions must be included. A single
vas found in our case from the lesion. It was
plain radiographs as well as on ultrasound.
DISCUSSION
Hemangiomas are the most common lesions of the major
salivary glands reported during infancy and early childhood
(Batsakis JG, 1986). Salivary gland hemangiomas in the
adult have been rarely reported (Nussbaum et al 1976). In
a reference series from the Armed Forces Institute of
Pathology (AFIP), only 1.4% of all salivary gland tumors
were found to be benign mesenchymal tumors. Out of
these, 30% were hemangiomas. As many as 87.5% of
these mesenchymal tumors occurred in the parotid gland
pulebol
missed
Ultras* d shows hemangiomas as heterogeneous
hypoe
scanni
investi
ic lesions (Hopkins, R 1969). CT dynamic
ind magnetic resonance imaging are useful
'e techniques for hemangiomas.

Another differential diagnosis in such a case is an
intramuscular hemangioma, first reported in the anterior
belly of the digastric muscle (Slack et al, 1989).
2. Dempsey E. F & Murley R. S (1970) Vascular malformations
simulating salivary disease. Br J Plast Surg ; 23; 77-84
3. Enzinger FM & Weiss SW (1995). Benign Tumors and tumor
like lesions of blood vessels. In Soft Tissue Tumors. Third
edition (Enzinger FM & Weiss SW eds). Mosby pp 579-626.
Submandibular salivary gland hemangiomas are
exceedingly rare, with a total of 13 cases reported so far
(McMenamin M et al, 1997).
4. Hopkins R (1969). Submandibular sialolithiasis with a case of a
cavernous hemangioma presenting as a salivary calculus.Br J
Oral Surg 6(3); 215-21.
5. McMenamin M et al (1997). Cavernous hemangioma in the
submandibular gland masquerading as sialadenitis. Oral Surg Oral
Med Oral Pathol Oral Radiol Endod, 84; 146-8.
6. Nussbaum M, Tan S, and Som M L (1976). Hemangiomas of the
salivary glands.Laryngoscope 86: 1015-1019
7. Slack R.W.T. Milroy C & Parker A (1989). Rare submandibular
swelling (Capillary Hemangioma). Journal of Laryngology and
Otology 103; 632-633.
Most of these hemangiomas have been of the cavernous
type. The clinical features, as seen in our case, can lead
to an erroneous diagnosis of other benign neoplasms or
chronic sialadenitis involving the submandibular gland.
Hemangiomas of the salivary glands should be considered
in the differential diagnosis because there is a risk of
profuse bleeding per-operatively. The rarity of such a
lesion in this area prompted us to report this case.
Address for Correspondence:
Dr. P. P. Singh
A-5, Teachers' Flats, Hans Raj College,
University of Delhi, Delhi-11007
REFERENCES
I. Batsakis JG (1986). Pathology consultation. Vascular tumors
of the salivary glands. Annals Otol Rhinol Laryngol 95; 649-
650.