A Rare Case Report of Intraosseous Capillary Hemangioma in the Mandible

Ghom S, Gawali M, Moitra PN, Bansod S, Dhobley A and Banerjee A

Published on: 2024-02-20

Abstract

Intraosseous hemangioma is a benign vascular neoplasm that is mostly seen in vertebrae, maxillofacial bones, and long bones. Intraosseous hemangioma is rarely seen on jaw bones compared to other skeletal bones and usually occurs in the cavernous form. Capillary intraosseous hemangioma of the jaws is an uncommon form of intraosseous hemangioma and has not been thoroughly described so far. In this study, a case of capillary intraosseous hemangioma of the mandible has been presented with a relevant literature review.

Keywords

Capillary Hemangioma, Hemangioma, Intraosseous, Vascular Malformation

Introduction

Hemangioma is a vascular malformation or neoplasm that originates from endothelial cells. Hemangiomas are generally classified into capillary, cavernous, mixed, and sclerosing types according to the type of blood cells they are composed of [1,2]. Hemangiomas are mostly seen in the head and neck region and are thought to originate from endothelial progenitor cells [3]. Oral capillary hemangiomas consist small, thin-walled capillary-like vessels that are lined with a layer of endothelial cells and are usually localized on oral soft tissues [1]. Clinically, capillary hemangiomas are soft, small-sized, lobulated, or solitary lesions with a colour range between red and blue due to the profundity of the lesion. They tend to diminish in size after a proliferative postnatal phase [4]. Oral capillary hemangiomas are rarely seen in adults and are mainly localized on the oral mucosa or gingiva [1,5]. The intraosseous form of capillary hemangioma is an uncommon entity and is reported to be seen in the metacarpal, frontal, and temporal bones [6-8]. In this report, an unusual case of intraosseous capillary hemangioma of the mandible has been presented with radiological and histological features.

Case Report

he patient reported to the department of oral medicine and radiology with the chief complaint of swelling in the left lower region of the jaw. On examination, the patient had diffused tender swelling in the parasymphysis region of the left mandible. Intraoral examination revealed generalized attrition. The oral mucosa was intact, and there was no cervical lymphadenopathy. The patient was advised to go for CBCT. CBCT revealed periapical granuloma with 33 and 34, and it also showed radiolucency in the body region of the mandible in close proximity with the mental foramen. The sclerotic boundaries of the lesion were prominently revealed. The lesion appeared to be hypodense, with a hyperdense particulate appearance scattered at the center of the lesion. The lingual cortical bone and inferior border of the mandible appeared to be intact. The buccal cortical bone showed areas of destruction. The maximum antero-posterior diameter of the lesion was 17.60 mm, the superior-inferior diameter of the lesion was approximately 12.22 mm, and the medio-lateral diameter was found to be 11.11mm. Extraction of 33 and 34 was done under local anesthesia, followed by curettage of the socket. Then an attempt was made to remove the large lesion in the body of the mandible. However, profuse bleeding was encountered during the procedure, and considering the fact that it was difficult to remove the lesion with such profuse bleeding, it was decided to abandon the surgery for the enucleation of the lesion under local anesthesia by extraoral approach. The patient was taken for surgery after two days under general anesthesia. The patient was hypertensive, so hypotensive anesthesia was used. An extraoral submandibular incision was placed, and dissection was done until the body of the mandible was preserved, preserving the marginal mandible. The lesion was completely enucleated with the help of electrocautery, and the mental nerve and the inferior alveolar nerve were preserved during dissection. Hematemesis was achieved, and layer-wise closure was done using 3.0 absorbable polyglactin 910. The skin was closed with a 4.0 Nylon suture, and a rubber dam was placed. The patient had constant pus discharge through the rubber dam for the next twenty days; hence, constant debridement with saline and betadine was done for the next twenty days until there was no drainage. The healing was uneventful. The patient was periodically observed after the treatment for six months, and no recurrence was noted.

Figures: Captured CBCT Images Demonstrate Capillary Hemangioma in All Three Planes.

Figure: Intraoperative Image After the Removal of The Lesion, I.E., Capillary Hemangioma.

Figure: Follow-Up After Six Months.

Histopathological Report

Figure: A H&E-Stained Section Reveals Numerous Budding Endothelial Cell-Lined Capillaries Scattered Throughout the Connective Tissue Stroma. Interspersed Between Are Plasma Lymphocytic Cell Infiltration, Chiefly Lymphocytes And A Few Polymorphonuclear Neutrophils. The Capillaries Are Engorged with Rbcs and Other Blood Elements (4X).

Figure: The High-Power View Shows Small and Occasionally Large Endothelial Cell-Lined Capillaries Engorged with Rbcs. Also Scattered in The Stroma Are Lymphocytes (40X).

Discussion

Soft tissue hemangiomas are considered common, specifically in children under the age of 10 [9,10]. Intraosseous hemangioma of the jaws is extremely rare; it shows no symptoms and is discovered on routine radiographs. Intraosseous hemangiomas are commonly located in the vertebrae, spine, and calvaria, less frequently in skull bones and long bones [11]. It is also difficult to give a diagnosis based on clinical symptoms and radiographs [12]. Pain and swelling are presented in a few cases; intraosseous hemangioma of the jaws is mostly asymptomatic. Patients might experience numbness in the lips and mental region [13]. Intraosseous capillary hemangioma of the jaws has been mentioned and published in the literature since 1974 [14]. 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 are no malignancy features and the imaging shows hypervascularity and avid enhancement [15]. After reading the literature, radiological findings for intraosseous hemangiomas include:

1.A 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.Honeycomb or sunburst appearance.

5.Rarely, a radiodensity area [16,17]. Treatment of intraosseous capillary hemangioma differs according to the dimension and size of the lesion. If the lesion causes no esthetic problem but is 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 [18,19].

Conclusion

Due to the rarity of intraosseous capillary hemangiomas, a gradual approach toward a definite diagnosis is required by excluding other similar bony lesions. The gold standard for a correct diagnosis is the association of histological examination, CT scan, and angiography. The Treatment modality should be based on the patient’s age, clinical features, and the lesion’s extension.

Conflict of Interest

The authors declare no conflicts of interest.

Funding

Self-funding

Journal of Oral and Dental Health Research