Case report: intraosseous capillary hemangioma presenting as an expansile lesion of the mandibular body with sunburst appearance with 2-year follow-up

Introduction

Hemangiomas are benign vascular neoplasms. A hemangioma is characterized histologically as a proliferative collection of endothelial cells that form vascular channels. These vascular spaces are interposed by a stroma of fibrous connective tissue. They can be categorized based on the size of their vascular channels (1,2). The two most common are capillary and cavernous hemangiomas. The capillary subtype is composed of a network of fine, small vessels with prominent stroma and cellularity. The cavernous subtype is formed by larger, thin-walled vessels and sinuses lined with a single layer of endothelium and scant stroma (1,2).

Intraosseous hemangiomas were first reported in 1845 by Toynbee (3). The first published report of an intraosseous hemangioma of the mandible was by Stanley in 1849, followed by Mottrecht in 1867 (4). These rare, benign vascular intraosseous neoplasms account for only 0.2–0.7% of all osseous neoplasms These lesions often present in the second decade of life with a 2:1 female to male ratio (5,6). With regard to mandibular intraosseous hemangiomas, there are fewer than 130 reported cases in the literature (7).

This paper aims to direct the provider towards the inclusion of intraosseous hemangiomas in the differential diagnosis of an asymptomatic mandibular lesion with a sunburst appearance. Included is a discussion of the differential diagnosis, followed by a discussion and treatment choices. We present this case in accordance with the CARE reporting checklist (available at https://fomm.amegroups.com/article/view/10.21037/fomm-24-46/rc).

Case presentation

Patient information

A 56-year-old, healthy, Caucasian female was referred for evaluation of a right mandibular body lesion which had been present for over 6 years. She was followed by a surgeon who opted not to biopsy since there was no clinical change on serial examinations. In the 6 months prior to presentation, patient noted an increase in size of the lesion and was referred to one of the authors (V.B.Z.) for definitive management. The patient reported no symptoms including pain, paresthesia, bruising or discharge of any type. Her primary complaint was noticing slight increase in size of lesion along the inferior border of the mandible over the past 6 months which was also clinically evident. She does not have any similar lesions. She reports no relevant family history.

Clinical findings

Clinical examination revealed a 2 cm × 2 cm bony mass of the right mandibular body with some expansion noted inferiorly and lingually. There was very slight compressibility to the buccal plate appreciated with no discomfort to palpation, ecchymosis, or bruit palpable. None of the patient’s teeth were mobile, and all involved teeth tested vital. No other intraoral lesions or masses were noted. Maximal incisal opening was >40 mm. Cranial nerves II–XII were noted to be normal bilaterally and no cervical lymphadenopathy was appreciated.

Diagnostic imaging

Panoramic film depicted a 3 cm × 3 cm lesion with mixed radiolucent and radiopaque sunburst appearance (Figure 1). Computed tomography (CT) scan without contrast was ordered which demonstrated a permeative lesion of the right mandibular body with thickened trabeculae and erosion of the adjacent cortices and extension beyond these, with associated periosteal reaction. The mass was expansile and displaced the mandibular canal superiorly with minimal impingement upon the canal itself. The adjacent soft tissue exhibited mild thickening without a defined soft tissue mass (Figure 2). Three-dimensional reconstruction images were consistent with the clinical exam showing a lesion at the inferior border of the right mandibular body with inferior and lingual expansion (Figure 3).

Figure 1 Panoramic radiograph at initial presentation showing 3 cm lesion with mixed radiolucent and radiopaque sunburst appearance. L, left.

Figure 2 Sagittal view of right mandibular lesion with sunburst appearance and superior displacement of inferior alveolar nerve canal.

Figure 3 Submental (A) and lateral (B) views of three-dimensional reconstruction images of right mandibular 3 cm × 3 cm intraosseous lesion (black arrow directed at lesion).

Differential diagnosis

Based on the patient’s history, clinical examination, and CT findings, the following differential diagnosis was considered: bone sarcomas [i.e., osteosarcoma (OS), chondrosarcoma (CS), Ewing sarcoma (ES)], osteoblastic metastases, intraosseous vascular anomalies (e.g., hemangioma, or malformations), and odontogenic myxoma.

The most common sarcomas of the jaw are OS, CS, and ES. OS and CS are connective tissue neoplasms arising within bone and represent malignancies of osteoblasts and chondroblasts, respectively. ES can originate from either bone or soft tissue, with bone being the most common site of involvement. Furthermore, the true origin of ES remains unclear and is a subject of debate and today ES is thought to arise from either mesenchymal stem cells or neuroectodermal cells (8-10). The incidence of OS is bimodal with patients either presenting younger than 25 years of age or older than 59 years of age; CS most commonly presents in the 3rd–5th decade of life; and ES mainly affects pediatric patients (8-12). Most studies have reported a greater incidence of OS and ES in the mandible than in the maxilla, with a greater incidence of CS in the maxilla than in the mandible (11,13). The jaw bones account for approximately 6.5% of all OS cases, 5–12% of CS cases, and 1.8–5.2% of ES cases (11,13,14). Granowski-LeCornu et al. reported the most common presenting symptom of an OS of the jaw was swelling (74.5%), following by pain (31.9%) and lower lip numbness (17%) (13). The radiographic presentation of OS, CS, and ES affecting the mandible is characterized by cortical erosion, osteolysis, and the appearance of “sunburst” (or “sunray”) spiculation of the periosteum. Given the patient’s age, new onset swelling, in the setting of a “sunburst” appearance on radiographic imaging, a diagnosis of sarcoma of the jaw, specifically OS was placed high on the differential diagnosis.

Maxillofacial metastases are not common and represent 1–2.1% of all oral cancers (15). Within the head and neck region, the posterior mandible is the most common site of involvement for a metastatic lesion (16,17). In 31% of cases of metastasis to the maxillofacial region, the metastatic lesion is the first clinical evidence of the primary tumor (18). While the most common sites of origin of the primary tumor are lung, breast, prostate, and kidney; these metastases to the maxillofacial region could result from malignancies from anywhere in the body (15,19). The most common finding (37.4%) on clinical presentation is a palpable mass (19). Additionally, 19.2% of patients have been reported to present with a neurosensory deficit of the lower lip, and 17.5% of patients presented with pain (15,19). From a radiographic perspective, when the primary tumor arises from the lung or breast, the metastatic site will present as an osteolytic lesion that is radiolucent with ill-defined borders, which could be also mistaken with inflammation/infection (16,20). However, in patients where the primary tumor originates from the prostate, these can present with an osteoblastic or sclerotic lesion and present with a mixed radiopaque-radiolucent or radiopaque lesion with a “sunburst” periosteal reaction (15,21). Being that the patient’s biological sex is female, this eliminates prostate cancer as an option of a primary site for a metastatic tumor, and with breast and lung cancers presenting as osteolytic lesions with radiolucent and ill-defined borders, it is unlikely that the lesion in our patient represents a maxillofacial metastasis.

Vascular anomalies have been classified into two major types: hemangiomas and malformations. Hemangiomas are benign vascular neoplasms that result from endothelial cell proliferation (22,23). Vascular malformations represent progressive ectasia of abnormal vessels and are grouped into arterial, capillary, and venous with or without fistulae and lymphatic anomalies. While 16% of patients with vascular anomalies of the head and neck have been reported to involve the mandible, maxilla, or both (23). For maxillofacial intraosseous hemangiomas, two-thirds of the lesions occurred in the mandible (5). Lesions that involved both the maxilla and mandible were more likely to have soft tissue involvement, while mandibular lesions had the least amount of soft tissue involvement. In one study, 90% of patients had experienced a bleeding episode most commonly from the gingiva and alveolar ridge, while all patients with combined maxillary and mandibular involvement were noted to have had a bleeding episode (23). In the same study, 61% of patients were noted to have facial asymmetry and swelling, and 35% had experienced pain. Of note, thrills, bruits, and pulsations were noted only in arterial vascular malformations, but were absent in all capillary and venous malformations (23). The radiographic appearance of vascular anomalies within the maxilla and mandible most commonly presents with expansile, osteolytic lesion with ill-defined borders with “soap bubble” or “honeycomb” features with the possibility of a “sunburst” periosteal reaction being present (5). Based on the history of the lesion being present for 6 years with recent onset of symptoms and with the radiographic presentation of the lesion, even in the absence of the hallmark vascular signs (e.g., thrill, pulsation, bruit) a vascular anomaly, specifically hemangioma was considered in our differential diagnosis.

Odontogenic myxoma is a slow growing but potentially destructive benign neoplasm of odontogenic mesenchymal origin that is usually diagnosed in the 2nd–5th decades of life (24). The most common location is the posterior mandible with swelling and cortical expansion being the most common presenting symptoms. Radiographically the odontogenic myxoma most commonly presents as a multilocular radiolucency with lacy and netlike septations, classically described as “honeycomb”, “soap bubble”, or “tennis racket” (24). While uncommon, the odontogenic myxoma has been reported to present with a radiographic “sunburst” periosteal reaction (25,26). Therefore, based on our patient’s radiographic presentation of periosteal “sunburst” radiographic appearance a diagnosis of odontogenic myxoma was considered.

Diagnosis and management

An incisional biopsy was performed under general anesthesia. The surgical site was exposed by a vestibular incision and the region of bony expansion was identified and noted to have yellowish color at its external border. Using surgical burs under copious irrigation and biopsy instrumentation, samples of the lesion were taken including removal of the expansile portion of the lesion, which was of primary esthetic concern to the patient and reason for seeking treatment. Grossly, the specimen was 1.8 cm × 1.4 cm × 0.7 cm, tan-red-brown, and firm. There was no notable bleeding from the biopsy site. Histopathological analysis demonstrated an intraosseous, capillary hemangioma (Figure 4A,4B). Patient was subsequently referred to neurosurgery and underwent a diagnostic cerebral angiogram to characterize the vascular supply to the lesion with possible embolization. On external carotid investigation there was minimal tumor blush with no major vessels supplying the mandibular lesion. Patient was subsequently offered resection and subsequent reconstructive options versus observation due to the lack of major vessel feeders. Patient elected to proceed with observation. The patient was seen and examined postoperatively at 2-week, 6-week, 3-month, 6-month, 12-month, and then 24-month. Over the course of her follow-up visits, there was no clinical change in size or character of the hemangioma.

Figure 4 Histological features of an intraosseous capillary hemangioma of the mandible via H&E staining at 4× (A) and 10× (B) magnification: thin-walled vessels with focal dilation and single layer of endothelial cells. H&E, hematoxylin and eosin.

All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Declaration of Helsinki and its subsequent amendments. Written informed consent for publication of this case report and accompanying images was not obtained from the patient or the relatives after all possible attempts were made.

Discussion

Soft-tissue hemangiomas are relatively common and predominate in children, whereas, intraosseous hemangiomas of the bone are more common in the older populations. Intraosseous hemangiomas are often located in the calvaria and spine (27,28). Peak incidence occurs in the second decade of life, and is more common in women (5). Intraosseous hemangioma of the mandible is exceedingly rare with fewer than 130 cases of mandibular intraosseous hemangiomas having been reported in the literature (7). Though they are benign, their expansile growth within the mandibular bone can lead to significant morbidity, including facial deformities, tooth mobility, and bone resorption. In the jaws, clinical diagnosis is difficult as presenting signs and symptoms are generally nonspecific (e.g., jaw asymmetry, swelling, and pain). Intraosseous hemangioma of the jaws are predominantly asymptomatic (29,30).

Radiographic findings for the intraosseous hemangioma can also vary. Case reports include descriptions of a unilocular radiolucency, a honeycombed area, a sunburst appearance, and rarely a radiodensity (31-33). Being nonspecific, CT provides little additional information relative to plain imaging. CT imaging aids in defining the extent of the lesion and characterizing its relationship with adjacent structures (2,31).

When intraosseous hemangioma of the jaws is suspected, observation may be preferred. Indications for intervention include hemorrhage, cosmetic deformity, or mass effect (2). Acceptable treatments include embolization, radiotherapy, curettage, and even surgical resection. Surgical curettage carries a risk of iatrogenic intraoperative hemorrhage. Embolization carries well-known risks related to vascular procedures (34). However, there has been success in its use when combined with surgical approaches, such as curettage. Yalcin and Ozer describe a case in which preoperative embolization of the feeder vessel followed by curettage of a 6 cm hemangioma yielded a successful result, and mitigated the risk for intraoperative bleeding (35). Radiotherapy, although effective, long-term risk of secondary malignancy makes this option less favorable (36). This option is generally reserved in patients whom cannot tolerate surgery. Complete excision within healthy bone remains the mainstay of treatment for those cases in which surgical intervention is indicated (2,31,36). Ozdemir et al. described a case of intraosseous mandibular hemangioma with en bloc resection with no complication or recurrence over a 24-month follow-up (5).

Conclusions

Intraosseous hemangioma of the mandible is a rare lesion that is often overlooked in the differential diagnosis of lesions secondary to its low incidence, and often-time nonspecific clinical and radiographic findings. The authors acknowledge the limitations of this case presentation with regard to the lack of clinical photos. Nonetheless, this article provides important emphasis on preoperative evaluation, including a history and physical exam that is correlated with radiographic findings. Histopathologic diagnosis is vital to rule out malignancy and provide a correct diagnosis. Multiple treatment modalities may be offered to patients, with observation being preferred when possible.

Acknowledgments

None.

Footnote

Reporting Checklist: The authors have completed the CARE reporting checklist. Available at https://fomm.amegroups.com/article/view/10.21037/fomm-24-46/rc

Peer Review File: Available at https://fomm.amegroups.com/article/view/10.21037/fomm-24-46/prf

Funding: None.

Conflicts of Interest: All authors have completed the ICMJE uniform disclosure form (available at https://fomm.amegroups.com/article/view/10.21037/fomm-24-46/coif). V.B.Z. serves as an unpaid editorial board member of Frontiers of Oral and Maxillofacial Medicine from November 2025 to October 2027. The other authors have no conflicts of interest to declare.

Ethical Statement: The authors are accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. All procedures performed in this study were in accordance with the ethical standards of the institutional and/or national research committee(s) and with the Declaration of Helsinki and its subsequent amendments. Written informed consent for publication of this case report and accompanying images was not obtained from the patient or the relatives after all possible attempts were made.

Open Access Statement: This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the non-commercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). See: https://creativecommons.org/licenses/by-nc-nd/4.0/.

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