Intraosseous hemangioma of the mandible: A rare case report

Abstract

Introduction and importance

Intraosseous hemangioma is a benign vascular tumour, it is mostly seen in long bones, vertebrae, and rarely on jaw bones, it usually occurs with its cavernous form. Medical literature is lacking a full description of capillary hemangioma of the jaws due to its rareness.

Case presentation

A 68 year old male referred to the dentist for normal checking, without significant medical history nor presuming medications. The panoramic radiograph showed radiolucent lesion in the right mandibular body. Surgery was performed and the excisional biopsy was subjected to a private oral pathology lab for the definite diagnosis, which was capillary hemangioma.

Clinical discussion

Intraosseous hemangioma rarely occurs in the jaws. Normally it is asymptomatic and discovered accidently on radiographs. Radiological findings for intraosseous hemangiomas are not well documented. Treatment of intraosseous capillary hemangioma differs according to the dimension and size of the lesion.

Conclusion

This paper aims to guide the clinician to start including hemangioma in the differential diagnosis for translucent lesions located in the jawbones, when radiographs show the necessity of that, in order to avoid uncontrollable bleeding and even death during any surgical intervention.

Highlights

• •Intraosseous vascular lesions are rare conditions.
• •The origin of central hemangioma is debatable.
• •Some authors believe that hemangioma is a true neoplasm, whereas others state it as a hamartomatous lesion.
• •Accurate diagnosis is necessary to achieve the safest, less traumatic treatment plan.

1. Introduction

Hemangioma is a benign vasoformative neoplasm of endothelial origin. The pathologic forms are divided into capillary, cavernous, mixed and sclerosing hemangioma [1,2]. It is estimated that hemangiomas cover 7 % of all benign tumours. More than 50 % of these lesions locate in the head and neck area, with a higher prevalence in females than males (3:1 ratio) [3]. Endothelial progenitor cells are the blamed to pull the trigger for tumerogenesis in this disorder [2].

Oral capillary hemangiomas are usually located on oral soft tissues, forming small sized and thin walled capillary like vessels [1]. Capillary hemangiomas manifest clinically with a color ranging from red to blue considering the depth of the lesion, which is mostly soft, small sized, lobulated or solitary [4].

Oral capillary hemangiomas are randomly seen in adults, and are mainly localized on the gingiva or oral mucosa [5,6]. The intraosseous type of capillary hemangioma is not normally seen in head and neck area, therefor their incidence, etiology and treatment approach is not well documented in the literature. The intraossoeuos hemangiomas have been reported in the head and neck region as separate case reports in the frontal, metacarpal and temporal bones [7,8].

It is usually asymptomatic although may have signs and symptoms including a slow growing bluish mass, discomfort, pulsating sensation and teeth mobility. [9] Panoramic radiograph, CT – scan and MRI are the most useful radiographic tests.

Differential diagnosis includes: Odontogenic tumours, Odontogenic cysts, Fibrous lesions, Fibrous dysplasia, Cystic lesions, and Aneurismatic bone cyst [10,11].

The significance of a mandibular hemangioma might lies in the possibility of the teeth representing a high risk of bleeding due to any trauma or extraction attempt.

In this article, we report a rare case of intraosseous capillary hemangioma with unspecific radiological features and a quite infrequent location, which was written according to SCARE criteria. [12]

2. Case report

A 68 year-old-male patient referred to a private dental clinic seeking diagnosis of a radiolucent lobulated lesion located in the mandibular right body. The patient had no complains and there was no clinical symptoms. Radiographs showed the approximate location of the lesion under and between the right lower canine and the second lower molar (Fig. 1). Medical history of the patient was taken and there was no correlation to the current situation. The patient was given an appointment and scheduled for surgical enucleation of the lesion.

Fig. 1.

Panoramic radiograph showing a well-defined radiolucency of the lesion in the right mandible

Surgical work was done by an oral surgeon. Operation site was exposed by creating vertical and sulcular flaps. Bone excavators and surgical burrs were used to directly reach the lesion, along with the saline irrigation. Moderate bleeding was surprisingly coming from the lesion. After wide surgical excision of the lesion that was performed by means of intraoral surgical approach under local anesthesia. The surgical specimen was referred to a private oral pathology service. It had a solid structure with bluish-brown color and measured in biggest dimension: 10 × 05 × 03 mm (Fig. 2). Histological examination showed a complete replacement of normal structure of the region with connective tissue contained lobules of capillaries lined by endothelial cells (Fig. 3(1)) along with areas of hemorrhage (Fig. 3(2)) These lobules are separated by a fibrous stroma. No inflammation, neoplastic or malignant activity is detected. Diagnosis was intraosseous capillary hemangioma. Patient was undertaken in follow-up period with 6 months check-ups. After one year the bone reformation was good (Fig. 4).

Fig. 2.

Biopsy on macroscopic examination after fixation.

Fig. 3.

Photomicrography showing lobules of capillaries lined by endothelial cells (H&E; ×100)(H&E; ×400).

Fig. 4.

Panoramic radiograph for 12 months follow-up showing replacing bone loss area with intact new bone.

3. Discussion

Soft tissue hemangiomas are considered common, specifically in children under the age of ten [13,14]. Intraosseous hemangioma of the jaws is extremely rare, it shows no symptoms, it is discovered on routine radiographs. Intraosseous hemangiomas are commonly located in the vertebrae, spine and calvaria, less frequently in skull bones and long bones [15]. It is also difficult to give a diagnosis based on clinical symptoms and radiographs [16]. Pain and swelling are presented in few cases, intraosseous hemangioma of the jaws is mostly asymptomatic. Patients might experience numbness in the lips and mental region [17].

intraosseous capillary hemangioma of the jaws have been mentioned and published to the literature since 1974. [18] Most of the reports concluded that additional tests like FNA and arteriography should be considered when suspecting intraosseous hemangioma, and that despite its rarity, it's important to give the correct diagnosis if there is no malignancy features and the imaging shows hypervascularity and avid enhancement [19]. After reading the literature, Radiological findings for intraosseous hemangiomas include:

• 1.Cyst-like lesion, of varying size and appearance, resembling a unilocular rounded radiolucent lesion.
• 2.Well defined cavity with dense edges and messy inner trabeculation.
• 3.Bone trabeculae radiating from the center to the outer edge of the lesion.
• 4.Honeycombed or sunburst appearance.
• 5.Rarely, a radiodensity area [20,21].

Treatment of intraosseous capillary hemangioma differs according to the dimension and size of the lesion. If the lesion causes no esthetic problem, superficial and asymptomatic, it can be left without treatment. Generally, the suggested treatment for soft and intraosseous capillary hemangioma is surgical excision with the embolization done before the operation [22,23].

4. Conclusion

To date there are less than 120 cases of intraosseous hemagioma of the mandible documented in the literature, capillary hemangiomas are rarely diagnosed and hardly mentioned in the differential diagnosis for similar radiolucent lesions. Despite the fact that hemangioma is a benign neoplasm with considerably good prognosis, it is important to detect hypervascularity accompanied with the absence of malignant histological features to reach the appropriate and correct diagnosis for this lesion.

Consent

Written informed consent was obtained from the patient for publication of this case report and accompanying photographs.

Sources of funding

We have no sponsor.

Registration of research studies

This paper is case report. The authors don't need to register this work.

Guarantor

Corresponding author is the guarantor for this case report.

Provenance and peer review

Not commissioned, externally peer-reviewed.

Patient perspective

Patient was quite happy with the surgical procedure and overall treatment.

Declaration of competing interest

N/A, There is no conflicts of interest.