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 Table of Contents  
CASE REPORT
Year : 2015  |  Volume : 27  |  Issue : 2  |  Page : 286-290

Cavernous hemangioma in the floor of oral cavity masquerading as a ranula


1 Department of Oral Medicine and Radiology, Faculty of Dental Sciences, SGT University, Gurgaon, Haryana, India
2 King George Medical University, Lucknow, Uttar Pradesh, India
3 Hazaribag College of Dental Sciences, Hazaribag, Jharkhand, India

Date of Submission03-Dec-2014
Date of Acceptance15-Oct-2015
Date of Web Publication21-Nov-2015

Correspondence Address:
Puneeta Vohra
Department of Oral Medicine and Radiology, Faculty of Dental Sciences, SGT University Hospital, Budhera, Gurgaon - 122505, Haryana
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/0972-1363.170161

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   Abstract 

A painless, bluish, submucosal swelling on one side of the floor of the mouth usually indicates the presence of a ranula. Rarely, such a swelling may be caused by an inflammatory disease process in a salivary gland, a neoplasm in the sublingual salivary gland, hemangiomas, a lymphatic nodular swelling, amyloidosis, or embryologic cysts/dermoid cyst. We report a 35-year-old female patient with swelling in the floor of her mouth that was clinically diagnosed as a ranula due to negative diascopy, the site of swelling, and the age of patient. Because of a strong clinical suspicion of a ranula, diagnostic methods such as angiography and magnetic resonance imaging (MRI) were not used in our case. A preoperative diagnosis was not truly established. We report a case of hemangioma in the floor of the mouth masquerading as a ranula. Although a rarity, vascular malformations should always be ruled out by using digital subtraction angiography and MRI studies before going for surgical intervention.

Keywords: Floor of oral cavity, hemangioma, ranula


How to cite this article:
Vohra P, Chandar VV, Patil R, Verma S. Cavernous hemangioma in the floor of oral cavity masquerading as a ranula. J Indian Acad Oral Med Radiol 2015;27:286-90

How to cite this URL:
Vohra P, Chandar VV, Patil R, Verma S. Cavernous hemangioma in the floor of oral cavity masquerading as a ranula. J Indian Acad Oral Med Radiol [serial online] 2015 [cited 2019 Nov 13];27:286-90. Available from: http://www.jiaomr.in/text.asp?2015/27/2/286/170161


   Introduction Top


Submucosal swelling in the floor of the mouth often represents a disease process of the salivary glands (e.g., sublingual, submandibular, minor). Typically, a chronic, painless, one-sided swelling on the floor of the mouth that is bluish and soft indicates a ranula. Rarely, such a swelling may be associated with an inflammatory disease process in a salivary gland, a benign or malignant neoplasm in the sublingual salivary gland, lymphatic nodular swelling, or embryologic cysts. Such processes are thought to arise from tissue normally located in the regions mentioned; so the source may be, for example, vascular, neurogenic, dentigerous, or skeletal. This case report describes a patient with swelling in the floor of her mouth that was clinically diagnosed as a ranula. During surgery, however, a vascular tumor was suspected, and histologic testing confirmed that the lesion was a cavernous hemangioma.


   Case Report Top


A 35-year-old female patient reported to the outpatient department of KM Shah Dental Hospital with swelling in the right side of the floor of the mouth since 3 months. Patient was relatively asymptomatic 3 months back. Swelling was accidentally noted by a dentist while extracting a grossly carious 36. She ignored the swelling initially; but as the swelling was gradually increasing in size and causing intermittent pain, she visited a cancer hospital. Investigations were done and they had sent the patient to the OPD of the dental hospital for opinion. Her symptoms were medium-grade with intermittent pain, and there was difficulty in chewing, speech, and tongue movements. The size of the swelling was unaltered before or after taking meals. She had no difficulty in swallowing and reported no bleeding from the mouth. She was a housewife, vegetarian, with normal sleep and appetite, and used to clean her teeth once daily with tooth brush and paste in the morning. No deleterious oral habits were reported. No extraoral swelling in the orofacial and neck region was present [Figure 1]. Right submandibular lymph node was enlarged, tender, palpable, firm in consistency and non-fixed. Intraoral clinical examination confirmed the presence of a clinically missing 36 and a carious 48. Gingiva was reddish pink, firm in consistency, resilient, and at normal position and contour; stippling was present and bleeding on probing was not present; oral hygiene and periodontal status were fair.
Figure 1: Extraoral view

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Examination of area of chief complaint revealed a large, bluish swelling on the right side of the floor of her mouth measuring 3 × 1.5 cm approximately [Figure 2]. Swelling was evident on the right side of the floor of the mouth with respect to 46, 45, 44, and 43, and extended from the lingual surface of the teeth to the midline in the floor of the mouth, not crossing the midline mediolaterally, and from the mesial surface of the first premolar to the distal surface of the first molar anteroposteriorly. The mass was covered with normal mucosa, with no obvious ulceration. Movement of the tongue, which deviated toward the right, was slightly affected. On bimanual and bidigital palpation, the mass was found to be soft, tender, non-pulsatile, and to have a consistent compressibility, but did not blanch on pressure. It was not attached to the lower jaw and not associated with the submandibular salivary gland. However, it was attached to the sublingual salivary gland. The swelling did not extend beyond the submandibular triangle, and the clinical opinion was that it was localized on the floor of the mouth, above the mylohyoid muscle. With detailed clinical examination and history of patient, a provisional diagnosis of ranula was made with a differential diagnoses of hemangioma, lymphangioma, pleomorphic adenoma, and congenital dermoid cyst. Detailed hematological investigation was carried out, which revealed the following: Hemoglobin (Hb)- 8.3 g/dl, erythrocyte sedimentation rate (ESR)- 15 mm/h, WBC count- 7500 cells/mm 3 , random blood sugar (RBS)- 74 mg/dl, and serum creatinine- 0.8 mg/dl. This was followed by fine needle aspiration cytology (FNAC) performed from the swelling on the floor of the mouth. The smears revealed mainly RBCs with a few polymorphs and lymphocytes. No malignant/dysplastic cells were seen. Orthopantomograph (OPG) and maxillary cross-sectional occlusal view were done, but did not reveal any radiographic findings. Ultrasonography (USG) was done, which did not reveal any abnormal blood flow or phleboliths. Plain and contrast computed tomography (CT) neck was done, which revealed a non-enhancing soft-tissue lesion of size 31.3 × 13.9 mm with distinct borders and of density 52-55 HU, with no rim enhancement or any abnormal uptake of contrast evident [Figure 3]. Focus of calcification was noted within the lesion. No bony erosion was seen [Figure 4]. These findings gave an impression which was suggestive of a lymphangioma, dermoid cyst and hemangioma, but did not rule out ranula. After this, the patient was surgically treated, surgical excision of the lesion was done, and the specimen was sent for biopsy [Figure 5] and [Figure 6]. Gross examination of the specimen revealed a mass of 3 × 3 cm in size, brownish black in color, firm in consistency, and with a lobulated surface [Figure 7]. Histopathologic examination revealed irregular dilated vascular spaces lined by flattened endothelial cells in a fibrous connective tissue stroma. Endothelium-lined vascular spaces were engorged with large aggregates of erythrocytes, which confirmed the diagnosis of cavernous hemangioma [Figure 8]. Three years later, the patient is well and has no complications or recurrence.
Figure 2: Intraoral view with bluish swelling in the floor of oral cavity

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Figure 3: Sagittal view of swelling on CT scan

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Figure 4: Axial view on CT scan

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Figure 5: Surgical resection of the mass from the floor of oral cavity

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Figure 6: Post-surgical photograph after removal of sutures

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Figure 7: Pathological sample

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Figure 8: Histomicrograph showing endothelium-lined vascular spaces

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   Discussion Top


Hemangiomas and vascular malformations are benign lesions of blood vessels that appear relatively often in the head and neck region. Hemangiomas particularly favor the head and neck region, with 65% of them occurring in this region, even though the head and neck comprise only 14% of the human body. [1] Various terms and classes have been suggested to describe these types of lesions. The renaissance began in 1982 when Mulliken and Glowacki introduced a "biological" classification that dispensed with the old, confusing terminology and offered a set of clear, concise terms and definitions. [2] They recognized two distinct entities, hemangioma and vascular malformations, on the basis of clinical and biological differences. [2],[3],[4],[5] In accordance with this classification scheme, hemangiomas are characterized by a rapid, primitive stage of development after the initial period of formation, followed by a slow stage of regression. Microscopically, hyperplasia in the endothelium of the basilar membrane is evident early in the primitive stage of development, which is followed by fibrosis with decreased cellularity in the involution phase. [2] Most hemangiomas (70-90%) appear in the first 4 weeks of life, and the regression stage typically begins after age 7. [2],[3],[4] Vascular malformations are present at birth and progress slowly as the infant grows. Histologically, they consist of vascular channels that are covered by normal endothelial cells. Vascular malformations do not regress later in life and after a traumatic injury or secondary to hormonal influences, they swell suddenly and develop rapidly. [3],[5] Hemangiomas and vascular malformations can localize exclusively to the skin or in deeper tissue and involve the skin, at which point the lesion is visible and easily recognized. Rarely, such lesions localize in extremely deep tissues and can remain undiagnosed for years, especially if they do not produce any clinical symptoms. [5],[6] Often, deep hemangiomas that are not perceptible within the first few years of life and begin to regress are discovered only by chance. [2] Development of a phlebolith within the hemangioma from a thrombus may cause symptoms and usually leads to diagnosis of the hemangioma. [7] Similarly, vascular dysplasias that localize to deep tissues are usually diagnosed only after abrupt growth is caused by trauma or hormonal influences. [8] The most common regions of deep localization of hemangiomas are muscle, liver, and bone. [1],[9],[10] Approximately 1% of hemangiomas localize in the muscles of the head and neck, most commonly the masseter and trapezius muscles. [9],[11] They also can localize in the parotid gland, larynx, and paranasal sinus, and few isolated cases of localization in the mentalis muscle have been reported. [10],[12],[13] Adhering to the diagnostic approach for deep vascular lesions can be difficult. Often, control X-ray films are not helpful. However, findings on USG, CT, magnetic resonance imaging (MRI), and angiography can be very useful. [5],[13],[14] Because clinical suspicion was strongly in favor of a ranula due to negative diascopy, the site of swelling, and the age of patient, diagnostic methods such as angiography and MRI were not applied in our case. Obtaining a biopsy specimen from such vascular tumors in the floor of the mouth which is a rare site for hemangioma can also can be challenging. When biopsy is essential to secure histologic proof, measures to prevent hemorrhage must be in place.

Treatment, too, is difficult in such lesions and often has poor results; but in our case, there was no history of reoccurrence. Lesions causing no significant clinical problems may be left alone, but the patient should be kept under regular follow-up. Thorough evaluation and a precise diagnosis help to determine the most effective therapeutic option, which may be surgery, sclerotherapy, pharmacotherapy, interlesional steroids, or irradiation. [5],[6] In our case, surgical intervention was chosen because the initial clinical diagnosis was a ranula. If the correct diagnosis had been made, however, surgery would have been contraindicated because of the clinical problems as well as the dangers of hemorrhage. A multidisciplinary approach is required in treating hemangiomas, as the hemangioma can be associated with liver, spleen, skin, pancreas, brain, and also with syndromes. Referral should be made to general physician, neurosurgeon, and dermatology departments, respectively. Latest investigation modalities include USG, color Doppler, MRI, dynamic gadolinium (Gd)-enhanced MRI, nuclear medicine, RBC-tagged technetium-99m (99mtc), scintigraphy with single-photon emission CT (SPECT), and digital subtraction angiography (DSA). Latest treatment modalities include laser photocoagulation with Nd:YAG, photocoagulation for vascular malformations and hemangiomas in childhood, sclerotherapy by morrhuate sodium and sodium psylliate, carbon dioxide snow, cryotherapy, and compression. [15],[16]

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.

Acknowledgment

The authors are grateful to the Department of Oral Diagnosis, Medicine and Radiology, KM Shah Dental College and Hospital, Vadodara.

Financial support and sponsorship

Nil.

Conflicts of interest

There are no conflicts of interest.

 
   References Top

1.
Shpitzer T, Noyek AM, Witterick I, Kassel T, Ichise M, Gullane P, et al. Noncutaneous cavernous hemangiomas of the head and neck. Am J Otolaryngol 1997;18:367-74.  Back to cited text no. 1
    
2.
Mulliken JB, Glowacki J. Hemangiomas and vascular malformations in infants and children: A classification based on endothelial characteristics. Plast Reconstr Surg 1982;69:412-22.  Back to cited text no. 2
    
3.
Waner M, Suen JY. A classification of congenital vascular lesions. In: Waner M, Suen JY, editors. Hemangiomas and Vascular Malformations of the Head and Neck. San Francisco: Wiley; 1999. p. 1-12.  Back to cited text no. 3
    
4.
Garzon M. Hemangiomas: Update on classification, clinical presentation, and associated anomalies. Cutis 2000;66:325-8.  Back to cited text no. 4
    
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Donald PJ. Vascular anomalies of the head and neck. Facial Past Surg Clin North Am 2001;9:77-92.  Back to cited text no. 5
    
6.
Fordham LA, Chung CJ, Donnelly LF. Imaging of congenital vascular and lymphatic anomalies of the head and neck. Neuroimaging Clin N Am 2000;10:117-36. viii.  Back to cited text no. 6
    
7.
Hessel AC, Vora N, Kountakis SE, Chang CY. Vascular lesion of the masseter presenting with phlebolith. Otolaryngol Head Neck Surg 1999;120:545-8.  Back to cited text no. 7
    
8.
Bizakis JG, Prassopoulos P, Papadakis CE, Kyrmizakis DE, Panayiotides JG, Katsamouris A. Arteriovenous malformation of the neck presented during delivery. Auris Nasus Larynx 2002;29:203-7.  Back to cited text no. 8
    
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Odabasi AO, Metin KK, Mutlu C, Baºak S, Erpek G. Intramuscular hemangioma of the masseter muscle. Eur Arch Otorhinolaryngol 1999;256:366-9.  Back to cited text no. 9
    
10.
Giudice M, Piazza C, Bolzoni A, Peretti G. Head and neck intramuscular haemangioma: Report of two cases with unusual localization. Eur Arch Otorhinolaryngol 2003;260:498-501.  Back to cited text no. 10
    
11.
Broniatowski M. Intramuscular hemangiomas of the masseter and sternomastoid muscles. Ear Nose Throat J 1993;72:303-5.  Back to cited text no. 11
    
12.
Makeieff M, Maurice N, Mondain M, Crampette L, Guerrier B. Intramuscular hemangioma of posterior neck muscles. Eur Arch Otorhinolaryngol 2001;258:28-30.  Back to cited text no. 12
    
13.
Okabe Y, Ishikawa S, Furukawa M. Intramuscular hemangioma of the masseter muscle: Its characteristic appearance on magnetic resonance imaging. ORL J Otorhinolaryngol Relat Spec 1991;53:366-9.  Back to cited text no. 13
    
14.
Clement WA, Graham I, Ablett M, Rawlings D, Dempster JH. Intramuscular hemangioma of the posterior belly of the digastrics muscle failing to highlight on magnetic resonance imaging. Ann Otol Rhinol Laryngol 2002;111:1050-3.  Back to cited text no. 14
    
15.
Cholewa D, Waldschmidt J. Laser treatment of hemangiomas of the larynx and trachea. Lasers Surg Med 1998;23:221-32.  Back to cited text no. 15
    
16.
Silva VA, Lima NL, Mesquita AT, da Silveira EM, Verli FD, de Miranda JL, Santos CR, Marinho SA. Intramuscular hemangioma in lip treated with sclerotherapy and surgery. Case Rep Dent 2011;2011:302451.  Back to cited text no. 16
    


    Figures

  [Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5], [Figure 6], [Figure 7], [Figure 8]



 

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