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Treatment of Central Hemangioma with Embolization in the Mandible
J Oral Med Pain 2023;48:169-173
Published online December 30, 2023;  https://doi.org/10.14476/jomp.2023.48.4.169
© 2023 Korean Academy of Orofacial Pain and Oral Medicine

Hae-In Choi1 │Gyeong-Yun Kim1 │Dong-Ho Shin1 │Ji-Su Oh1 │ Seong-Yong Moon1 │Jae-Seek You1 │Ji-Won Ryu2

1Department of Oral and Maxillofacial Surgery, School of Dentistry, Chosun University, Gwangju, Korea
2Department of Oral Medicine, School of Dentistry, Chosun University, Gwangju, Korea
Correspondence to: Hae-In Choi
Department of Oral and Maxillofacial Surgery, School of Dentistry, Chosun University, 309 Pilmun-daero, Dong-gu, Gwangju 61452, Korea
E-mail: beautyluby@hanmail.net
https://orcid.org/0000-0002-2425-9506
Received December 1, 2023; Revised December 11, 2023; Accepted December 11, 2023.
This is an open-access article distributed under the terms of the Creative Commons Attribution Non-Commercial License (https://creativecommons.org/licenses/by-nc/4.0) which permits unrestricted non-commercial use, distribution, and reproduction in any medium, provided the original work is properly cited.
Abstract
Hemangioma is a lesion characterized by vascular proliferation of endothelial origin, manifesting neoplastic features. The occurrence of central hemangioma in the oral and maxillofacial region is exceptionally rare, and in two-thirds of cases, it is predominant in the mandible rather than the maxilla. The main symptoms and signs associated with central hemangiomas include pulsation, bone expansion, bruit, teeth displacement, and root resorption of the adjacent teeth. Bleeding may manifest periodically from the sulcus surrounding the affected teeth, particularly when accompanied by hypermobility in the primary dentition. One of the most noteworthy complications is the potential for severe bleeding during tooth extraction or surgical procedures conducted in proximity to unrecognized hemangiomas. Such situations may pose a life-threatening risk. Taking this into consideration, we present two cases of central hemangiomas in adolescent patients who sought consultation, with subsequent embolization performed by the Department of Radiology in Chosun University Hospital.
Keywords : Adolescent; Emobolization; Hemangioma; Mandible
INTRODUCTION

Hemangiomas, which are characterized by vascular proliferation of endothelial origin and demonstrate neoplastic features, constitute a rare subset of intraosseous or central hemangiomas, comprising 1% of primary bone tumors. Hemangiomas are predominantly observed in the vertebrae and craniofacial skeletal regions, with a notable presence in the metaphysis of long bones, and their occurrence in the oral and maxillofacial regions is particularly infrequent. Within this context, two-thirds of cases manifest in the mandible rather than the maxilla [1]. The third decade has been commonly reported as the peak age of incidence; however, conflicting literature suggests an occurrence in the second decade, notably with a twofold higher incidence in female than in male [2]. When central hemangiomas manifest in the maxilla and mandible, they often present clinically asymptomatically and with a variety of radiographic appearances, which makes it challenging to distinguish them from other lesions such as ameloblastoma, odontogenic myxoma, odontogenic keratocyst, central giant cell granuloma, or traumatic bone cyst. Radiographically, primarily in panoramic radiographs or cone-beam computed tomography (CBCT), hemangiomas may exhibit multilocular radiolucent appearances or honeycomb patterns while also adopting unilocular configurations or spoke-like and sunray-like patterns [3]. This diversity underscores the risk of misdiagnosis, potentially leading to unexpected and substantial bleeding during surgical procedures or biopsy. Taking these challenges into consideration, we present two cases of central hemangiomas in two adolescent patients who sought consultation and subsequently underwent embolization at the Department of Radiology in Chosun University Hospital.

This study was approved by the Institutional Review Board of Chosun University Dental Hospital (CUDH IRB-2311-021), and the committee waived the need for written informed consent.

CASE REPORT

1. Case 1

In July 2023, a 15-year-old male patient presented to Chosun University Dental Hospital with a cystic lesion situated between the right mandibular second premolar (#45) and first molar (#46). Despite a history of Kawasaki disease 13 years prior, the patient had no concurrent medical conditions. There were no clinical symptoms, and the patient was referred to the pediatric dentistry department due to the incidental discovery of a cystic lesion on panoramic radiography. The radiograph revealed an unilocular radiolucent lesion in proximity to the apex of #45 and 46 (Fig. 1). Subsequent CBCT illustrated indicated a unilocular radiolucent lesion with partial sclerotic bone changes and an ill-defined border, extending from the periapical region of #45 and 46 to the lingual plate of the inferior mandibular canal (Fig. 2). Although #45 displayed an open apex and a negative response to electric pulp testing, #46 exhibited a positive response to high electrical current. Consequently, the provisional diagnosis considered was either a simple bone cyst or a periapical cyst associated with #45. However, during attempted biopsy under local anesthesia involving the removal of the buccal bone below #46, we encountered a marked tendency for severe bleeding, which prompted the cessation of the surgical procedure. At that point, we discontinued the surgery. Hemostasis was achieved via the application of Surgicel and compression, and the wound was subsequently closed using a suture. Considering the observed bleeding tendency, we suspected the possibility of a central hemangioma. We then consulted the Department of Radiology at our institution, which led to a comprehensive assessment. Subsequently, we performed embolization using 50-125 μm SMART GEL (PLmicromed) after we confirmed the blood supply from the inferior alveolar artery, originating from the right external carotid artery and the internal maxillary artery (Fig. 3). The following day, we reopened the wound for curettage under general anesthesia. Notably, we observed no significant bleeding. Bone grafting was performed, and the patient exhibited satisfactory healing without no additional or delayed bleeding events (Fig. 4).



2. Case 2

In January 2021, an 11-year-old female patient was referred from a local clinic due to an extensive radiolucent lesion in the left mandible. The patient had no significant medical history. Radiographically, a multilocular lesion was observed with scalloped margins extended from the left mandibular first premolar (#34) to the midline of the mandible. The lesion exhibited well-defined and hyperostotic borders with lingual bone expansion (Fig. 5). The patient did not manifest clinically specific symptoms but reported dull, throbbing pain upon palpation of the left mandibular first premolar (#34), second molar (#36), and deciduous second molar (#75). The provisional diagnosis leaned toward an odontogenic keratocyst. Taking into consideration the young age of the patient’s and her limited cooperation, we decided to perform the decompression procedure under general anesthesia. However, a vascular lesion was observed during surgery at the extraction sites of #75 and #36, accompanied by a significant tendency for bleeding (Fig. 6). The surgery was subsequently halted, and we sought an immediate consultation with the Department of Radiology, which confirmed the blood supply of this lesion from the maxillary and facial arteries originating from the left external carotid artery. A microguidewire and microcatheter were advanced to the maxillary artery, and embolization was performed via the inferior alveolar artery using eight Concerto coils (Medtronic, Inc.) (Fig. 7A). Follow-up examination conducted 1 year 6 months later, encompassing both panoramic radiography and CT scans, showed no evidence of recurrence, and favorable bone healing was evident (Fig. 7B).


DISCUSSION

Mandibular central hemangiomas often exhibit a slow growth pattern and remain asymptomatic for extended durations. However, in certain instances, rapid enlargement may occur, resulting in facial asymmetry, particularly in young individuals. This phenomenon can affect the eruption and alignment of permanent dentition [4]. The main symptoms and signs associated with central hemangiomas include pulsation, bone expansion, bruit, displacement of teeth, and resorption of adjacent teeth. Bleeding may manifest periodically from the sulcus around the affected teeth, especially accompanied by hypermobility in the primary dentition. A notable complication is the risk of severe bleeding during tooth extraction or surgical procedures conducted in close proximity to an unrecognized hemangioma, which poses a potential life-threatening situation [5]. Central hemangiomas in the maxillofacial region tend to be more prevalent in young individuals. In cases such as the ones described above, children may experience poorer prognoses than adults do because of their lower circulatory volume and immature defense mechanisms against acute bleeding [6].

However, it must be acknowledged that the symptoms and signs mentioned above might not manifest in every case. Many cases are asymptomatic in clinical practice, which means that dental clinicians must rely solely on panoramic radiography or CBCT images for diagnosis. This reliance often leads to misinterpretation, because these lesions may be mistaken for more prevalent tumors in the maxillofacial region, such as ameloblastoma, odontogenic myxoma, central giant cell granuloma, odontogenic keratocysts, or traumatic bone cysts. Clinicians’ unpreparedness in encountering the potential for severe bleeding when approaching these lesions can be a source of embarrassment. Our cases similarly lacked clinical symptoms, with one presenting an unilocular radiolucent lesion resembling a traumatic bone cyst or a periapical cyst associated with #45 and the other exhibiting a scalloped-margin multilocular lesion, raising the suspicion of odontogenic keratocyst. Typically, central hemangiomas exhibit radiographic features such as small honeycomb patterns, large soap bubble patterns, spoke-like patterns, or sunray-like patterns [3]. Although we have presented only two cases here, the radiographic features of these patients did appear to align with typical features of central hemangioma, which posed challenges in the diagnosis. In the second case, severe bleeding during surgery under general anesthesia necessitated immediate consultation with the Department of Radiology for embolization. Relying solely on limited panoramic X-ray or CBCT images to accurately differentiate vascular lesions is challenging. Magnetic resonance angiography (MRA) is considered the optimal imaging tool for diagnosing vascular lesions. If central hemangioma is suspected, it is imperative to obtain an MRA to visualize intra-arterial angiography and evaluate the relevant vascular supply branches before surgery [7]. However, performing MRA for many patients with nonspecific radiographic findings that raise suspicion of central hemangioma may not be cost-effective in clinical practice. In such cases, a practical alternative when central hemangioma is suspected might be the use of a relatively low-cost imaging tool before MRA, such as ultrasonography.

Various methods have been used to treat central hemangiomas, including nutritional vessel ligation, embolotherapy, radiotherapy, cryotherapy, sclerotherapy, and surgery. Treatment method selection may hinge on factors such as size, lesion location, and patient age [8]. Central hemangiomas that occur in the jaw are challenging to treat because of their rich collateral blood supply. Simple ligation and embolization of the main vessel may yield only a temporary decrease in the size of the lesion and carry the risk of recurrence, because the blocked vessel rapidly regenerates to bypass blood circulation. Hence, the most reliable approach to treating central hemangiomas is considered to be ligation of the nutritional vessel or embolization prior to surgery to minimize intraoperative bleeding, followed by immediate surgery if feasible [9]. Nevertheless, complete excision of the lesion is challenging, particularly in cases that involve extensive and diffuse bone destruction. Performing segmental mandibulectomy with immediate reconstruction in young patients, as in the aforementioned cases, may result in potential esthetic asymmetry and an increased likelihood of requiring secondary surgery due to ongoing mandibular growth. Furthermore, in younger age groups, implant-based restorative options may not be viable due to the incomplete growth of the mandible, potentially compromising nutritional intake during a crucial developmental period. Therefore, extensive surgery in younger age groups should be approached with caution.

In the first case, the lesion was of modest dimensions and did not require extensive consideration. Conversely, in the second case, the extent of the lesion was considerable, encompassing the left body and ramus of the mandible and closely abutting multiple permanent teeth. In this scenario pursuing surgical intervention would require extractions. Consequently, we made the decision to conduct embolization with eight Concerto coils on the nidus of the lesion, opting for close observation without surgical intervention. Fortunately, there was no recurrence, and we observed favorable bone formation.

In the cases presented, we detailed the clinical narratives of two adolescent patients who exhibited nonspecific radiographic findings on panoramic or CBCT imaging, rendering the clinical suspicion of central hemangiomas challenging. We managed these cases through surgical intervention coupled with embolization or, alternatively, treated them solely with embolization.

CONFLICTS OF INTEREST

No potential conflict of interest relevant to this article was reported.

DATA AVAILABILITY STATEMENT

The datasets used in this study are available from the corresponding author upon reasonable request.

FUNDING

This study was supported by a research fund from Chosun University Dental Hospital, 2021.

AUTHOR CONTRIBUTIONS

Conceptualization: SYM. Data curation: JSO. Formal analysis: JSY. Funding acquisition: HIC. Methodology: DHS. Project administration: GYK. Visualization: JWR. Writing - original draft: HIC. Writing - review & editing: HIC.

Figures
Fig. 1. A unilocular radiolucency is observed on panoramic X-ray at the apical region of #45 and 46.
Fig. 2. Partial hyperostotic change and ill-defined borders were observed on cone-beam computed tomography around the apical areas of #45 and 46, extending to the lingual plate of the mandibular canal.
Fig. 3. The blood supply within the lesion originated from the right external carotid artery and internal maxillary artery, specifically the inferior alveolar artery.
Fig. 4. A follow-up panoramic radiograph obtained 2 months postoperatively revealed bone grafting at the previous lesion site.
Fig. 5. A multilocular lesion was observed radiographically with scalloped margin extending from #34 to the middle of the mandibular ramus.
Fig. 6. Intraoperative clinical photographs revealed the presence of a vascular lesion at the site where #75 and #35 were extracted, accompanied by a significant tendency for bleeding.
Fig. 7. (A) Postoperative panoramic radiograph. Embolization was performed using eight Concerto coils through the inferior alveolar artery. (B) Panoramic radiograph obtained 1 year 6 months after embolization. Bone regeneration was observed with no signs of recurrence.
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