|Year : 2019 | Volume
| Issue : 3 | Page : 159-163
Reddish-blue lesion of the tongue
Manveen Kaur Jawanda1, Ravi Narula2, Vineet Sharma3, Priya Gupta1
1 Department of Oral Pathology, Luxmi Bai Institute of Dental Sciences and Hospital, Patiala, Punjab, India
2 Department of Oral Surgery, Guru Nanak Dev Dental College and Research, Sunam, Punjab, India
3 Department of Conservative Dentistry, Luxmi Bai Institute of Dental Sciences and Hospital, Patiala, Punjab, India
|Date of Web Publication||3-Jul-2019|
Manveen Kaur Jawanda
#9, Doctors Colony, Bhadson Road, Patiala - 147 004, Punjab
Source of Support: None, Conflict of Interest: None
Although hemangioma is considered as the most common soft-tissue tumors of the head and neck, it is relatively rare in the oral cavity and uncommonly encountered by dental professionals. The occurrence of hemangiomas on the tongue is extremely rare. Changes in blood flow dynamics within hemangioma result in thrombus and phleboliths. This article presents a relatively rare and unusual case of cavernous hemangioma of the dorsum of the tongue with phleboliths in a 55-year-old male.
Keywords: Cavernous hemangioma, hemangioma, phlebolith
|How to cite this article:|
Jawanda MK, Narula R, Sharma V, Gupta P. Reddish-blue lesion of the tongue. Indian J Dent Sci 2019;11:159-63
| Introduction|| |
The diagnosis of tongue lesions is a challenge for any clinician. The tongue is a complex area of the mouth, with a variety of native tissue types that give rise to a plethora  of pathological conditions, both benign and malignant [Table 1]. A painless, soft, reddish-blue mass on the tongue indicates the presence of lymphangioma, hemangioma, neurofibroma, pyogenic granuloma, chronic inflammatory gingival hyperplasia (epulis), and even squamous cell carcinoma.
Although hemangioma is considered as the most common soft-tissue tumors of the head and neck, it is relatively rare in the oral cavity and uncommonly encountered by  dental professionals. Changes in blood flow dynamics within hemangioma result in thrombus and phleboliths.
After an extensive search of the English-language literature, only a few cases of head-and-neck hemangiomas with phleboliths were properly documented. Around seven cases have been reported in buccal mucosa or cheek;,,,,,, ten cases were in the parotid, submandibular, and sublingual glands;,,,,,,,,, one case was in the floor of the mouth;, one case was in the hypopharynx; four cases were on the masseter;,,, one case was in the parapharyngeal space; and one was at the base of the tongue. Here, we report another relatively rare and unusual case of cavernous hemangioma of the dorsum of the tongue with phleboliths in a 55-year-old male.
| Case Report|| |
A 55-year-old male has been reported to our institute for the evaluation of a painless mass on the posterolateral surface of the tongue. The patient noticed the swelling 2.5 years back and complained of bleeding from the site 1.5 months back. The patient was unaware of the exact duration of the swelling. The patient appeared to be of normal built with no defect in stature or gait and with no relevant medical history. Intraoral examination revealed a growth measuring about 1.0 cm × 1.0 cm, with a reddish-blue discoloration of the overlying mucosa at the right posterolateral surface of the tongue. The growth appeared to be sessile with no attachment to the underlying muscles [Figure 1]. The lesion showed well-defined borders along with intact surface. The swelling was soft to palpate, and no pulsations, bruits, or thrills were heard. A differential diagnosis of lymphangioma, hemangioma, and pyogenic granuloma was given based on the clinical findings. The mass was excised and sent for histopathological examination. The healing was uneventful. Gross specimen was oval, firm with a pebbly surface [Figure 2].
|Figure 1: Clinical photograph showing a reddish-blue lesion on the posterolateral surface of the tongue|
Click here to view
The histopathological examination revealed numerous endothelium-lined, large, dilated blood sinuses beneath the keratinized stratified squamous epithelium [Figure 3]. In few vascular spaces, the lumina appeared to be blocked by eosin-colored fibrinous material, suggestive of thrombus along with calcium deposits [Figure 4]a and [Figure 4]b. Microscopically, the calcifications were consistent with phleboliths [Figure 4]c. Correlating clinical findings with histopathological features, a confirmatory diagnosis of cavernous hemangioma of the tongue with phleboliths was made.
|Figure 3: Photomicrograph showing numerous endothelium-lined, large, dilated blood sinuses beneath the keratinized stratified squamous epithelium (H and E, ×10)|
Click here to view
|Figure 4: Photomicrograph showing (a and b) thrombus obliterating entire lumen and endothelial lining of the vessel wall (c) phlebolith with concentric lamellar structure (H and E, ×40)|
Click here to view
| Discussion|| |
Hemangioma (Greek: Haima – blood, angeion – vessel, oma – tumor) by definition can be defined as “a benign tumor of dilated blood vessels.” Hemangioma is often present at birth and extends during growth. However, many vascular lesions regress spontaneously before or during puberty with a few that go undetected for long periods of time until sudden growth induces pain or cosmetic deformity. The relation of trauma to neoplastic growth has always excited interest and is of unquestionable importance. The posterolateral surface of the tongue is more prone to trauma (injury from teeth) as compared to other sites, so it seems reasonable to suppose that tumors occurring in these areas are often considered to result from such injury. In that way, an injury could start a neoplastic process is interesting to consider. It is thought that trauma of this variety could initiate an abnormal arteriovenous communication. Such a communication would suddenly compel small vessels to withstand pressure and carry a volume of blood to which they are unaccustomed. The sudden stretching and dilatation of their walls would, in some way, resemble a chronic irritative process. The endothelium might be stimulated to proliferate to protect the vessel, and this proliferation might get beyond control, resulting in an abnormally differentiated and proliferating mitotic endothelial cell network. Hence, in our case, trauma may have served as an inciting stimulus.
Phleboliths associated with vascular anomalies were initially found in the splenic vein by Canstatt in 1843 Phleboliths are calcified nodules that can be regarded as a characteristic property of venous or cavernous hemangiomas. They may originate from injury to a vessel wall or result from stagnation of the flow of blood. The injury can result in damage to the intima. Healing involves formation of a protective thrombus that may calcify as a part of the healing process. Slowing with stagnation of blood flow also favors thrombus formation. The thrombus organizes with laminar fibrosis and progresses to central necrosis. Subsequently, deposits of calcium, phosphate, and apatite lead to crystallization, and with calcification of the thrombus, a phlebolith evolves.
Although intravascular calcifications are common in the veins of the pelvis, phleboliths are rarely found in vascular lesions of the maxillofacial area., The occurrence of hemangioma on the tongue is extremely rare. The present report is an unusual case of cavernous hemangioma occurring on the tongue with phleboliths.
Hemangiomas usually appear 2–4 weeks after birth, grow rapidly till the age of 6–8 months, and then slowly develop. By the age of 5–8 years, they start to involute and spontaneously regress in 70% of the cases. In the case presented here, the patient reported at a very late age of 55 years, because of the lesion, being on the posterior part of the tongue, it might have gone undetected for such a long time. Although benign, the lesion can progress with unfavorable complications. Complications may be physical (i.e., hemorrhage, infection, or disfigurement) or psychological secondary to cosmetic concerns. In the present case, as the lesion was on the tongue, recurrent bleeding from the lesion was a major concern.
Histologically, the appearance of these lesions depends on the stage of evolution. Early lesions may be very cellular with solid nests of plump endothelial cells and little vascular lumen. Established lesions comprise well-developed, flattened, and endothelium-lined capillary channels of varying sizes in a lobular configuration. Involuting lesions show increased fibrosis and hyalinization of capillary walls with luminal occlusion. In cavernous hemangioma, there is the presence of large, dilated blood sinuses with thin walls, each showing an endothelial lining. The sinusoidal spaces are usually filled with blood, although there might be the presence of lymphatic vessels. Microscopically, phleboliths consist of calculi with characteristic concentric lamination. Histopathological analysis of the present case showed typical features of hemangioma along with phleboliths and numerous thrombi, so we diagnosed this case as cavernous hemangioma with phleboliths.
Hemangiomas may mimic other lesions clinically and histopathologically. The differential diagnosis includes lymphangioma, neurofibroma, pyogenic granuloma, chronic inflammatory gingival hyperplasia (epulis), telangiectasia, angiosarcoma, squamous cell carcinoma, and other vascular-appearing lesions. The most common vascular proliferation of the oral mucosa is pyogenic granuloma. This is a reactive lesion that develops rapidly, bleeds easily, and is usually associated with inflammation and ulceration. Clinically, it is often lobulated, pedunculated, and red to purple, and it may be hormone sensitive. There are two histological types of pyogenic granuloma of the oral cavity: the lobular capillary hemangioma (LCH) and non-LCN type. LCH is characterized by proliferating blood vessels that are organized in lobular aggregates although superficially the lesion frequently undergoes no specific change, including edema, capillaries dilatation, or inflammatory granulation tissue reaction, whereas the second type consists of highly vascular proliferation that resembles granulation tissue., Histopathologically, the hemangioma exhibits a progression from a densely cellular proliferation of endothelial cells in the early stages to a lobular mass of well-formed capillaries in the mature phase, often resembling the pyogenic granuloma without the inflammatory features. The present case has clinical features of a pyogenic granuloma, but no microscopic features of pyogenic granuloma. Therefore, biopsy of tissue specimen is often necessary for a definitive diagnosis of hemangiomas. The diagnosis of a head-and-neck phlebolith requires its differentiation from other calcifications that occur in the same area. Salivary gland calculi, tonsilloliths, calcified lymph nodes, soft-tissue tumors, calcification of arteries, antrolith, multiple military osteomas of the skin, myositis ossificans, calcified acne, and cysticercosis, all serve to muddy the diagnostic waters.
Hence, precise diagnosis of the type of vascular lesion is important because it may influence treatment considerably. It becomes mandatory for practitioners to correctly manage any such lesions, as the surgical intervention involves invasive procedures, and only the histopathologic report can give a confirmatory diagnosis.
| Conclusion|| |
The case presented here adds to the existing few cases of cavernous hemangioma with phleboliths reported in the English literature. The present case explains the possible etiologic relation of trauma to a vessel wall resulting in an occurrence of the neoplasm at a late age of 55 years. Hence, hemangioma with phleboliths should be considered in the differential diagnosis of swellings of the tongue.
Declaration of patient consent
The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
Financial support and sponsorship
Conflicts of interest
There are no conflicts of interest.
| References|| |
Laskin DM, Giglio JA, Rippert ET. Differential diagnosis of tongue lesions. Quintessence Int 2003;34:331-42.
Enzinger FM, Weiss SW. Soft Tissue Tumors. 3rd
ed. St. Louis: Mosby; 1995. p. 581-6.
Li X. Rare cavernous haemangioma of the hypopharynx with numerous phleboliths. J Laryngol Otol 1990;104:262-3.
Ikegami N, Nishijima K. Hemangioma of the buccal pad with phlebolithiasis: Report of a case. Acta Med Okayama 1984;38:79-87.
Kutluhan A, Bozdemir K, Ugras S. The treatment of tongue haemangioma by plasma knife surgery. Singapore Med J 2008;49:e312-4.
Kanaya H, Saito Y, Gama N, Konno W, Hirabayashi H, Haruna S. Intramuscular hemangioma of masseter muscle with prominent formation of phleboliths: A case report. Auris Nasus Larynx 2008;35:587-91.
Sano K, Ogawa A, Inokuchi T, Takahashi H, Hisatsune K. Buccal hemangioma with phleboliths. Report of two cases. Oral Surg Oral Med Oral Pathol 1988;65:151-6.
Hemmati A. Phleboliths in a hemangioma of the left cheek. Fortschr Geb Rontgenstr Nuklearmed 1965;102:210-1.
Krause D. Multiple phleboliths in a cavernous hemangioma of the cheek. Z Laryngol Rhinol Otol 1964;43:125-7.
Hassani A, Saadat S, Moshiri R, Shahmirzadi S. Hemangioma of the buccal fat pad. Contemp Clin Dent 2014;5:243-6.
] [Full text]
Chuang CC, Lin HC, Huang CW. Submandibular cavernous hemangiomas with multiple phleboliths masquerading as sialolithiasis. J Chin Med Assoc 2005;68:441-3.
Altuǧ HA, Büyüksoy V, Okçu KM, Doǧan N. Hemangiomas of the head and neck with phleboliths: Clinical features, diagnostic imaging, and treatment of 3 cases. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 2007;103:e60-4.
Cankaya H, Unal O, Ugras S, Yuca K, Kiriş M. Hemangioma with phleboliths in the sublingual gland: As a cause of submental opacity. Tohoku J Exp Med 2003;199:187-91.
Brands T. Hemangioma of the parotid gland with multiple phleboliths. Zentralbl Chir 1969;94:1064-7.
Neuss O. Multiple phleboliths in an angioma of the parotid gland. Z Laryngol Rhinol Otol 1958;37:240-2.
Barták J. Phleboliths in a hemangioma in the parotidomasseteric region. Cesk Stomatol 1967;67:126-8.
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.
Sleiter R, Altermatt HJ, Buser D, Bornstein M. Cavernous hemangioma in the masseter muscle with multiple phlebolith formation: A case report. Schweiz Monatsschr Zahnmed 2002;112:617-23.
Nagpure PS, Jain S, Dhole P, Tyagi V, Singh R, Garg D. Submandibular gland cavernous hemangioma with phlebolith masquerading as sialadenitis. J MGIMS 2008;13:56-8.
Martin OP, Cagigal BP, Rubiales BM, Sanfrutos SB, Gonzalez MR, Hernandez AV. Sublingual cavernous hemangioma. Cir Oral Y Maxilofac 2013;35:44-9.
Baba Y, Kato Y. Hemangioma with phleboliths in the floor of the mouth presenting as a submental swelling: A case report. J Med Cases 2011;2:28-30.
Bernadski I, Khimich AI, Kutcher GD. Racemose facial hemangioma with multiple phleboliths. Stomatologija 1983;62:79.
Schwartz A, Salz N. Cavernous hemangioma associated with phleboliths in the masseter muscle. Acta Radiol 1955;43:233-4.
Mohan RP, Dhillon M, Gill N. Intraoral venous malformation with phleboliths. Saudi Dent J 2011;23:161-3.
Zengin AZ, Celenk P, Sumer AP. Intramuscular hemangioma presenting with multiple phleboliths: A case report. Oral Surg Oral Med Oral Pathol Oral Radiol 2013;115:e32-6.
Cho JH, Joo YH, Kim MS, Sun DI. Venous hemangioma of parapharyngeal space with calcification. Clin Exp Otorhinolaryngol 2011;4:207-9.
Alsadat M. Rare cavernous haemangioma of the oral cavity with numerous phleboliths. JOHOR 2012;1:46-51.
Downing JG, Mallory GK. Cavernous Haemangioma and trauma: Report of a case. Arch Derm Syphilol 1930;22:414-22.
Riordan BO. Phleboliths and salivary calculi. Br J Oral Surg 1974;12:119-31.
Mandel L, Perrino MA. Phleboliths and the vascular maxillofacial lesion. J Oral Maxillofac Surg 2010;68:1973-6.
Apfelberg DP. Treatment of hemangiomas. In: Georgiade GS, Georgiade NG, editors. Textbook of Plastic, Maxillofacial and Reconstructive Surgery. 2nd
ed. Philadelphia, PA: Williams and Wilkins; 1992.
Edgerton MT, Hiebert JM. Vascular and lymphatic tumors in infancy, childhood and adulthood: Challenge of diagnosis and treatment. Curr Probl Cancer 1978;2:1-44.
Dilsiz A, Aydin T, Gursan N. Capillary hemangioma as a rare benign tumor of the oral cavity: A case report. Cases J 2009;2:8622.
Neville BW, Damm DD, Allen CM, Bouquot JE. Oral and Maxillofacial Pathology. 2nd
ed. Philadelphia: W. B. Saunders; 2002. p. 447-9.
Shafer WG, Hine MK, Levy BM. A Textbook of Oral Pathology. 6th
ed. Philadelphia, PA, USA: W. B. Saunders; 1983.
Handa R, Sheikh S, Shambulingappa P, Aggarwal A, Mago J, Salaria S. Capillary hemangioma of palate: A case report. Pakistan Oral and Dental Journal 2013;33:55-7.
Mills SE, Cooper PH, Fechner RE. Lobular capillary hemangioma: The underlying lesion of pyogenic granuloma. A study of 73 cases from the oral and nasal mucous membranes. Am J Surg Pathol 1980;4:470-9.
[Figure 1], [Figure 2], [Figure 3], [Figure 4]