|Year : 2014 | Volume
| Issue : 2 | Page : 52-54
Submandibular vascular hamartoma with phleboliths mimicking sialolithiasis
Sharad Ramdas1, Anita Ramdas2, Moses M Ambroise2, Renu G'Boy Varghese2
1 Department of Plastic and Reconstructive Surgery, Pondicherry Institute of Medical Sciences, Pondicherry, India
2 Department of Pathology, Pondicherry Institute of Medical Sciences, Pondicherry, India
|Date of Web Publication||8-Dec-2014|
Department of Pathology, Pondicherry Institute of Medical Sciences, Kalapet - 605 014, Pondicherry
Source of Support: None, Conflict of Interest: None
The differential diagnoses of radiopacities in the orofacial region include sialoliths, calcified lymph nodes, tuberculous nodes, and uncommon phleboliths. We report a case of a 37-year-old lady with intermittent unilateral left-sided submandibular swelling caused by a vascular hamartoma with phleboliths, which was initially diagnosed as submandibular sialolithiasis.
Keywords: Hamartoma, phleboliths, sialolithiasis, submandibular
|How to cite this article:|
Ramdas S, Ramdas A, Ambroise MM, Varghese RG. Submandibular vascular hamartoma with phleboliths mimicking sialolithiasis. J Clin Sci 2014;11:52-4
| Introduction|| |
Hemangiomas and vascular malformations are common lesions in the head and neck region, particularly in the young and have been reported frequently in the parotid gland. Submandibular vascular malformations are comparatively uncommon.  Vascular malformations may have phleboliths which are probably the result of changes in blood flow dynamics. These can be mistaken clinically as sialoliths as both present with unilateral intermittent facial swellings. Some phleboliths are poorly calcified making radiographic detection difficult. However, imaging has progressed to involve ultrasound, computerized tomography (CT), and magnetic resonance imaging (MRI) (with or without sialography) to distinguish between these abnormalities, which with enhancement depict these lesions fairly well. ,,,
We present a case of intermittent unilateral left-sided submandibular swelling in a female patient caused by a vascular hamartoma with phleboliths, where the phleboliths were initially diagnosed clinically and radiologically as submandibular sialolithiasis.
| Case report|| |
A 37-year-old lady presented to our plastic surgery clinic with a history of swelling in the left submandibular region for the past 10 years. She complained of intermittent pain and an increase in size of the swelling over the last 5 years.
On examination, the left submandibular swelling was 7 × 4 cm [Figure 1] with normal overlying skin. The patient experienced mild tenderness on palpation. The temperature of the overlying skin was normal. The swelling was a central firm mass with soft, poorly delineated margins. It appeared to be extending deep into the lower margin of the mandible and was irreducible. There was no palpable thrill. Intraoral and bidigital examinations were unremarkable.
A clinical diagnosis of sialolithiasis was made.
MRI revealed an enlarged submandibular gland with a dilated submandibular duct containing a calculus measuring 3 × 3 mm at the distal opening of the duct adjacent to the genioglossus muscle in the floor of the mouth [Figure 2]. In addition there were multiple intraglandular calculi (8-10 in number) involving the gland and the hilum, with the largest one measuring 8 × 6 mm. There was evidence of hyperintensity with extension into the left parapharyngeal space and along the floor of the mouth suggesting an inflammatory cause and a possible small abscess. Fat stranding was seen around the gland.
Excision of the gland via a left submandibular approach was done. Intraoperatively the gland was sightly enlarged but the superficial lobe appeared otherwise normal. A dense network of large thinly walled vascular channels was encountered deep into the gland while dissecting the deep lobe. These channels were filled with phleboliths and extended posteriorly to the parapharyngeal space, where they were merging among muscle fibres. The entire gland was excised along with the adjacent vascular channels up to their intramuscular course. The excised specimen was sent for histopathology.
The histopathology report was that of a vascular hamartoma of the gland with numerous dilated vascular channels of varying sizes and thicknessess admixed with adipose tissue and surrounded by normal salivary gland tissue. Multiple phleboliths were noted in the vascular channels [Figure 3]. The ducts were dilated, but no significant inflammation or fibrosis was noted.
The postoperative period was uneventful and on follow-up the patient had no significant complaints.
| Discussion|| |
Salivary gland obstruction caused by vascular malformation with phleboliths is relatively uncommon and may mimic sialolithiasis.  Phleboliths or vascular calculi can be found in venous malformations of many adult patients, however, the clinical and radiographic appearance of phleboliths may resemble sialolithiasis, making the diagnosis more difficult.  This is particularly so in cases where the vascular lesion is located within the deeper tissues of the gland without a cutaneous component as in the present patient.
The diagnosis of vascular malformations containing phleboliths in submandibular gland, especially in adults, becomes quite difficult, as these lesions are rare and no confirmative noninvasive investigation exists. A plain x-ray may show multiple calcified pheleboliths, ultrasonography reveals heterogeneous, hypoechoic lesions with calcifications, a CT scan shows a tumor with enhancing quality of blood vessels. Phleboliths and sialoliths may appear similar on a contrast CT scan. MRI demonstrates hyperintensity on T1-weighted images and isointensity with muscle on T2-weighted images. Sialography and angiography have been mentioned and they can be done to determine the etiology.  Nuclear medicine imaging with Technetium-99m ( 99m Tc)-red blood cell scintigraphy has also been reported to be useful in differentiating sialadenitis from hemangiomas.  In the present case, MRI suggested sialolithiasis, which was consistent with the clinical suspicion, and the possibility of venous malformation was not considered until the excision of the gland was done.
Lesions such as venous vascular malformation or vascular hamartomas should be considered in the differential diagnosis whenever focal calcifications are seen radiologically, as it could represent phleboliths. Doppler applications are mandatory in all focal lesions of salivary glands. ,, We did not consider this possibility hence a Doppler was not undertaken for the patient.
As far as submandibular gland hemangiomas are concerned, the treatment of choice is surgical resection. Other modalities of treatment include laser, cryotherapy, embolization, and corticosteroids. 
Although most radiopacities detected on radiographs of the orofacial region may be sialoliths, other differentials such as calcified lymph nodes, tuberculosis of the salivary gland itself, calcified lymph nodes, calcified atherosclerotic plaques in major blood vessels and phleboliths must be kept in mind. ,
The present case is of importance on two accounts: First the submandibular gland is a very uncommon site for vascular hamartomas in adults and second the clinical and radiological features were that of submandibular sialolithiasis. The stone that was palpable was due to a phlebolith.
| Conclusion|| |
In conclusion, the differential diagnosis of vascular malformation with phleboliths as a cause of salivary gland obstruction should be considered whenever sialolithiasis is suspected as well as on encountering radiopacities in the orofacial region. A Doppler examination may be necessary in such a cases to avoid a misdiagnosis.
| References|| |
Chuang CC, Lin HC, Huang CW. Submandubular cavernous hemangiomas with multiple phleboliths masquerading as sialolithiasis. J Chin Med Assoc 2005;68:441-3.
Su YX, Liao GQ, Wang L, Liang YJ, Chu M, Zheng GS. Sialoliths or phleboliths? Laryngoscope 2009;119:1344-7.
Groppo ER, Glastonbury CM, Orloff LA, Kraus PE, Eisele DW. Vascular malformation masquerading as sialolithiasis and parotid obstruction: A case report and review of the literature. Laryngoscope 2010;120(Suppl 4):S130.
Lee SH, Lee KS, Cho YU, Kim KR. Hemangioma with phleboliths, misdiagnosed as silaoliths in submandibular gland. J Korean Surg Soc 2002;63:160-2.
Li X. Rare cavernous hemangioma of the hypopharynx with numerous phleboliths. J Laryngol Otol 1990;104:162-3.
McMenamin M, Quinn A, Barry H, Sleeman D, Wilson G, Toner M. Cavernous hemangioma in the submandibular gland masquerading as sialadenitis: Case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 1997;84:146-8.
Cankaya H, Unal O, Ugras S, Yuca K, Kiriº M. Hemangioma with phleboliths in the sublingual gland: As a cause of submental opacity. Tohoku J Exp Med 2003;199:187-91.
EL-Hakim IE, EL-Khashab MM. Cavernous haemagioma of the submandibular salivary gland. Int J Oral Maxilllofac Surg 1999;28:58-9.
Bradley M, Stewart I, King W, Metreweli C. The role of ultrasound and 99mTc RBC scintigraphy in diagnosis of the salivary gland hemangioma. Br J Oral Maxillofac Surg 1991;29:164-6.
Bar T, Zagury A, London D, Shacham R, Nahlieli O. Calcifications simulating sialolithiasis of the major salivary glands. Dentomaxillofac Radiol 2007;36:59-62.
Huang TC, Dalton JB, Monsour FN, Savage NW. Multiple, large sialoliths of the submandibular gland duct: A case report. Aust Dent J 2009;54:61-5.
[Figure 1], [Figure 2], [Figure 3]