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 Table of Contents  
CASE REPORT
Year : 2015  |  Volume : 2  |  Issue : 1  |  Page : 77-81

Unusual site of capillary hemangioma: Practitioner's dilemma!


Department of Oral Medicine and Radiology, Jaipur Dental College, Jaipur, Rajasthan, India

Date of Web Publication14-Jan-2015

Correspondence Address:
Dr. Vela Desai
B- 406, Trimurti Apartment, Malviya Nagar, Model Town, Jaipur - 302 017, Rajasthan
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/2348-3334.149355

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  Abstract 

Hemangioma is a common tumor characterized by the proliferation of blood vessels. It is often congenital in nature and usually but not invariably follows a benign course. Hemangioma is often present at birth, but may become more apparent later in life. Capillary hemangioma is known to have a significant predilection for lips, tongue, buccal mucosa, and palate. This case report describes a hemangioma at an unusual site which is seldom reported earlier in the published literature. Despite being benign in nature, hemangiomas are of clinical importance to the dental professionals as they may pose serious bleeding risk. Because of their uncommon clinical presentations they may mimic other gingival lesions; hence, dental practitioners need to be aware of these lesions.

Keywords: Capillary hemangioma, diagnosis, histopathology, incisive papilla


How to cite this article:
Desai V, Narang P, Varma B, Maghu S. Unusual site of capillary hemangioma: Practitioner's dilemma!. CHRISMED J Health Res 2015;2:77-81

How to cite this URL:
Desai V, Narang P, Varma B, Maghu S. Unusual site of capillary hemangioma: Practitioner's dilemma!. CHRISMED J Health Res [serial online] 2015 [cited 2019 Oct 21];2:77-81. Available from: http://www.cjhr.org/text.asp?2015/2/1/77/149355


  Introduction Top


The term hemangioma includes a heterogeneous group of clinically benign vascular lesions that have similar histologic features. [1] Some authors believe that this lesion particularly the congenital one, is not a true neoplasm, but rather a developmental anomaly or hamartoma. [2]

Hemangiomas are the most common tumors of infancy occurring in 5-10% of children less than 1 year of age. [3] Occasionally, older individuals are affected. [1]

It was first described by Sznajder et al., in 1973 under the term hemorrhagic hemangioma. [1] Hemangioma are localized in the craniofacial area in 60% of the cases, but oral involvement is rare. [4] It commonly affects lips, tongue, buccal mucosa, andpalate. [2] In addition, confusion with other conditions can occur since it may mimic other lesions clinically, radiographically, and histopathologically. The differential diagnosis of hemangioma includes pyogenic granuloma, chronic inflammatory gingival hyperplasia (epulis), irritational fibroma, telangectasia, and evensquamous cell carcinoma. [1]

The treatment of hemangioma represents a clinical challenge. [4] This article reports a 35-year-old female with capillary hemangioma of the oral cavity at an unusual site that mimicked areactionary gingival condition.


  Case Report Top


A 35-year-old female reported to the Outpatient department of Oral Medicine and Radiology, Jaipur Dental College and Hospital, with the chief complaint of pain on mastication for thelast 1 year associated with swelling on the back of upper front teeth. The patient initially noticed a small growth on the palatal aspect of maxillary central incisors which gradually increased to the present size. No history of trauma or any long-term medication was reported.

Detailed dental, medical, and family history was found to be noncontributory to the condition. Intraoral examination revealed reddish-pink solitary, well-defined, nonulcerated growth present over the incisive papilla palatal to maxillary central incisors, covering three-quarter of the crown. The lesion was approximately 2 × 1 cm in size, the maxillary central incisors were displaced distally leading to midline diastema.

On palpation, inspectory findings were confirmed. The lesion was firm in consistency, nonfluctuant, nontender, and pedunculated [[Figure 1]a-c]. No bleeding or pus discharge was associated, but there was impingement of mandibular central incisors over the occlusal surface of the swelling.
Figure 1: (a-c) Soft tissue mass over incisive papilla region

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No blanching was revealed on diascopic examination. Oral hygiene was fair with only mild amount of stains and calculus. Oral prophylaxis was considered as the first choice of treatment. It was provisionally diagnosed as irritational fibroma with pyogenic granuloma being one of the differential diagnosis.

The patient was subjected for intraoral periapical (IOPA) and occlusal radiographic investigations which showed only displacement of central incisors [[Figure 2]a and b].
Figure 2: (a and b) Intraoral periapical (IOPA) and occlusal radiograph showing displacement of maxillary central incisors

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As the lesion was well-localized, surgical excision was considered as the treatment of choice and a written consent was taken from the patient and relevant blood investigations were carried out before the treatment. The patient was recalled after 7 days for suture removal and the healing was uneventful. The excised specimen was sent for histopathological evaluation.

The hematoxylin and eosin (H and E) stained soft tissue section showed highly cellular stroma which was composed of numerous endothelial lined capillaries with variable diameter lumen. Numerous endothelial cells were proliferating into the connective tissue. The intervening connective tissue was very sparse. The overlying epithelium was parakeratinized stratified squamous epithelium which showed acanthosis at places [[Figure 3]a and b].
Figure 3: (a and b) The H and E stained soft tissue section showed highly cellular stromacomposed of numerous endothelial lined capillaries. The overlying epithelium was parakeratinized stratified squamous epithelium which showed a canthosis at places H and E = Hematoxylin and eosin

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With these findings capillary hemangioma was the final diagnosis. Patient was recalled for periodic examination which revealed uneventful healing and no recurrence of lesion for the next 6 months [Figure 4].
Figure 4: Postoperative after 6 months

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  Discussion Top


The term hemangiomahas traditionally been used to describe a variety of developmental vascular anomalies. They display a rapid growth phase with endothelial cell proliferation, followed by gradual involution. [3]

Unlike our case, a congenital hemangioma is present from birth, whereas a traumatic (acquired) hemangioma is a type of inflammatory hyperplastic lesion. [5] The condition may be right for certain inflammatory hyperplasia-like lesions with many patent capillaries to develop significant blood flow during the inflammatory hyperplasia stage. Such capillary system remains after the irritant has been eliminated and the inflammation subsides. The resultant lesion is usually nodular, bluish-red, bleeds easily, and may blanch on pressure. Capillary hemangioma represents an arrest in the development of the mesenchyme primordial in the undifferentiated capillary network stage. [6],[7]

They are much more common in females than the males in a ratio of 3:1 to 5:1. [3] Clinically hemangioma are characterized as a soft mass, smooth or lobulated, sessile or pedunculated, and seen in any size from a few millimeters to several centimeters with the color of the lesion ranging from pink to red purple and tumor blanches on application of pressure. They are generally painless. [1] They are firm and rubbery to palpation and the blood cannot be drawn by applying pressure. [3] In the present case no such classical features were present except for the gender.

The most common vascular proliferation of the oral mucosa is the pyogenic granuloma. This is a reactive lesion that develops rapidly, bleeds easily, and is usually associated with the inflammation and ulceration. Clinically it is often lobulated, pedunculated, and red to purple in color and it may be hormone sensitive. [3]

Hemangioma may also be confused with the vascular lesions of the face or oral cavity, like  Sturge- Weber syndrome More Details More Details. [8] They are often located in the territory of the branches of the trigeminal nerve and do not undergo spontaneous involution like hemangiomado. Ocular and cerebral vascular lesions may be found in such cases. [3]

Radiographs are advised to rule out bony destruction suggestive of central variety of hemangioma or calcifications. But in this case there were no signs of any bony involvement radiographically. Only slight displacement of maxillary central incisors was appreciable.

In proliferative hemangiomas the degree of vascularity can be evaluated by color-coded duplex sonography. An indication for magnetic resonance imaging (MRI) is only in deep hemangiomas in critical topographic regions such as the orbit or the parotid space. [4]

Precise diagnosis of the type of vascular lesion is important because it may influence treatment considerably.

Histopathologically, capillary hemangioma exhibits a progression from a densely cellular proliferation of endothelial cells in early stages to a lobular mass of well-formed capillaries in mature phase, often resembling the pyogenic granuloma without inflammatory feature. [1]

Complication may occur in 20% of hemangiomas. Most common problem is ulceration which may occur with or without secondary infection. Although hemorrhage may be noted, significant blood loss does not usually occur. Hemorrhage that occurs in crucial areas can be associated with significant morbidity. [3] No such complications were observed in our case.

The clinical decision for treatment or combination of treatment depends on the location and type of hemangioma and the indications for treatment. Some congenital lesions may undergo spontaneous regression at an early age. If superficial lesions are not an esthetic problem and are not subjectedto masticatory trauma, they may be left untreated. But, small and superficial lesions may be completely excised with relative ease. [1]

For problematic and life-threatening hemangiomas, pharmacologic therapy may be indicated. Corticosteroids are the first line oftherapy and are thought to be angiogenic. Topically clobetasol propionate can be used. [9] Also, systemic corticosteroids help to reduce the size of the lesion and are associated with 70-90% response rate. [3] Prednisone or methylprednisolone is given at initial daily dose of 2-3 mg/kg for 1 month. If response to therapy is positive, patient is continued on this dose for another 2-4 weeks. Daily dose is gradually lowered to 1 mg/kg and generally maintained for upto 4-6 months. Intralesional corticosteroid is effective for small, localized lesions and one to threeinjections are given at 4-6-week interval. A long-acting steroid (e.g. triamcinolone acetonide) can be combined with a short-acting steroid (e.g. cortisone acetate or betamethasone acetate) in a total dose that generally does not exceed 2.5 ml. Intravenously high doses of methylprednisolone is given at a daily dose of 30 mg/kg for 3 days, followed by daily dose of 20 mg/kg for 4 days, then daily dose of 10, 5, 2, and 1 mg/kg for 1 week. Vincristine, mitotic spindle tubule inhibitor, is currently the drug of choice for complicated tumors that are unresponsive to systemic corticosteroids. It is generally delivered through central venous access; an initial weekly dose of 0.05-1 mg/m 2 administered intravenously. This dose is then tapered, increasing the interval between injections depending on clinical response. Treatment is administeredfor 4-6 months. [9] Interferon α-2a is no longer widely used because of the reported risk of spastic diplegia. [3]

Other forms of therapy include radiation therapy (external radiation or radium), sclerosing agents such as sodium morrhuate or psylliate injected into the lesion, CO 2 snow, cryosurgery, [6] electrocoagulation, and laser therapy [1] [Table 1].
Table 1: Different treatment modalities for management of capillary hemangioma

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The prognosis of hemangioma is excellent since it does not become malignant or recur after adequate removal or destruction. In our case, clinically and radiographically, neither the lesion was large nor life-threatening in nature as it didnot show any bony involvement. Hence, it was treated by simple excision under necessary conditions.


  Conclusion Top


Capillary hemangiomas are infrequently seen on the incisive papilla region and may be easily confused with different lesions, particularly with pyogenic granuloma and irritational fibroma. Early detection and biopsy is necessary to determine the clinical behavior. Dental practitioners should be aware of the risks during diagnosis and management and should take necessary precautions duringthe treatment.

 
  References Top

1.
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.  Back to cited text no. 1
    
2.
Rajendran R. Developmental disturbances of oral and paraaoral structures. In: Rajendran R, Sivapathsundram B, editors. Shafer's Textbook of Oral Pathology. 5 th ed., Vol. 1. Noida: Elsevier Publication; 2006. p. 4-5.  Back to cited text no. 2
    
3.
Neville BW, Damm DD, Allen CM, Bouquot JE. Soft tissue tumors. In: Neville BW, Damm DD, Allen CM, Bouquot JE. editors. Textbook of Oral and Maillofacial Pathology. 4 th ed., Vol. 12. Philadelphia; WB Saunders; Elsevier Publication; 2009. p. 468-9.  Back to cited text no. 3
    
4.
Sznajder N, Dominguez FV, Carraro JJ, Lis G. Hemorrhagic hemangioma of gingiva: Report of a case. J Periodontal 1973;44:579-582.  Back to cited text no. 4
    
5.
Jerald J, Ronald B. Lesions of the facial skin. In. Wood NK, Goaz PW. editors. Differential Diagnosis of Oral and Maxillofaciallesions. 5 th ed. St. Louis: Mosby: Elsevier Publication; 1997. p. 548-50.  Back to cited text no. 5
    
6.
Rajendran R. Benign and malignant tumors of oral cavity. In: Rajendran R, Sivapathsundram B, editors. Shafer's Textbook of Oral Pathology. 6 th ed. Noida: Elsevier Publication; 2009. p. 140-1.  Back to cited text no. 6
    
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.  Back to cited text no. 7
[PUBMED]    
8.
Bhansali RS, Yeltiwar RK, Agrawal AA. Periodontal management of gingival enlargement associated with Sturge-Weber syndrome. J Periodontol 2008;79:549-55.  Back to cited text no. 8
    
9.
Sloan GM, Reinisch JF, Nichter LS, Saber WL, Lew K, Morwood DT. Intralesional corticosteroid therapy for infantile hemangiomas. Plast Reconstr Surg 1989;83:459-67.  Back to cited text no. 9
    


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