DISCUSSIONÂ
Salivary glands are frequently located in the parotid gland which is why
the majority of neoplastic processes and hemangiomas are studied at that
level(7,8). Hemangiomas are mesenquimalbenigns tumors and usually
presents as a unilateral hard mass (7). Within the parotid, it has been
described that 5% of benign tumors from this gland are hemangiomas
which is considered to be a rare tumor type (8). Diagnosis can be
challenging in this type of tumors, however it gets even difficult and
rare when it presents at the sublingual area, that usually compromise
the upper airway due to late diagnosis. (9)
Less than 20 submandibular benign tumor cases have been reported in
literature, between removable and biopsied lesions, it was found that
hemangiomas represented 1.7% of the cases (11). Clinical course varies,
and the majority presents only with pain or minor symptoms (11), but can
also present with recurrent upper respiratory infections (12) or severe
complications such as described in our case.
The diagnosis can be arduous in this type of benign tumors especially
due to their localization (10). Previous studies in parotid glands show
the efficacy of doppler ultrasound as first diagnostic step, followed by
MRI-imaging where further details can be seen (13). In our case,
ultrasound was highly beneficial to the diagnosis of hemangioma.
Some differential diagnosis can have similar clinical presentations such
as hemangioendotheliomas (14, 15), these are benign tumors with the same
origin as hemangiomas, but haschronic lesions and syndromes associated
(14). The presentation of this tumor is very rare and usually grows
within the submandibular triangle, where also pleomorphic adenomas can
be located (16). In our case, the submandibular hemangioma was found in
the submandibular triangle as well. Hemangiomas can grow extensively and
it can generate severe dysphagia (17), however this compromise may
become worse with acute respiratory distress and cardiac arrest due to
severe respiratory failure as happened to our patient.
Treatment has changed recently, according to a 40 years cohort study,
steroids and surgery have been well-tolerated by patients with excellent
response and very few adverse effects (18). Nowadays, majority of
hospitals follows a regimen based on oral propanolol which is usually
well tolerated as well (19).
In our case our patient followed this last regimen and up to this date
there has not been any issues so far.
As a conclusion; our patient had a hemangioma in a very rare location
with few previous reports. She had a severe complication managed in the
hospital with no recurrences or complications until today.