Intraosseous vascular lesions are rare conditions. They are most commonly seen in the vertebral column and skull; nevertheless, the mandible is a quite rare location. In this report, we present a case of intraosseous cavernous hemangioma in the mandible and discuss the clinical and radiological features. A 28-year-old male patient attended to our clinic with a complaint of painless swelling of mandible. Clinical evaluation revealed a bone-hard, smooth-surfaced, immobile mass in the left mandibular lingual area. The patient was evaluated with panoramic and occlusal radiography and computed tomography. The lesion surgically excised and pathological examination revealed an intraosseous cavernous hemangioma. Follow-up imaging 1 year later with cone beam computed tomography revealed recurrence of the lesion. The conclusion of this paper; when a bone hard, well-shaped mass was seen in the mandible, the possibility of intraosseous hemangioma must be remembered and before surgical procedure detailed radiographic evaluation should be performed

Derya, Yildirim

Elif, Bilgir

Gulperi, Kocer

Sevgi, Bozova

Journal of Clinical and Experimental Dentistry

PubMed

Repositori d'Objectes Digitals per a l'Ensenyament la Recerca i la Cultura

J Clin Exp Dent. 2017;9(1):e153-6.                                                                                                                                                                          Intraosseous hemangioma in mandiblee153Journal section: Oral Medicine and Pathology                            Publication Types: Case ReportIntraosseous Cavernous Hemangioma in the Mandible: A Case ReportBilgir Elif 1, Yildirim Derya 2, Kocer Gulperi 3, Bozova Sevgi 41 PhD, Research Assistant; Suleyman Demirel University, Faculty of Dentistry, Department of Dentomaxillofacial Radiology, Isparta, Turkey2 DDS, PhD, Associated Professor; Suleyman Demirel University, Faculty of Dentistry, Department of Dentomaxillofacial Radio-logy, Isparta, Turkey3 DDS, PhD, Associated Professor; Suleyman Demirel University, Faculty of Dentistry, Department of Oral and Maxillofacial Surgery, Isparta, Turkey4 MD, Specialist Doctor of Medicine; Ministry of Health Turkish Public Hospitals Institution Association of Antalya Public Hos-pitals Serik State Hospital, Department of Pathology, Antalya/TurkeyCorrespondence:Suleyman Demirel UniversityFaculty of DentistryDepartment of Dentomaxillofacial Radiology, Turkeybilgirelif04@hotmail.comReceived: 23/11/2015Accepted: 02/03/2016Abstract Intraosseous vascular lesions are rare conditions. They are most commonly seen in the vertebral column and skull; nevertheless, the mandible is a quite rare location. In this report, we present a case of intraosseous cavernous he-mangioma in the mandible and discuss the clinical and radiological features. A 28-year-old male patient attended to our clinic with a complaint of painless swelling of mandible. Clinical evaluation revealed a bone-hard, smooth-surfaced, immobile mass in the left mandibular lingual area. The patient was evaluated with panoramic and occlusal radiography and computed tomography. The lesion surgically excised and pathological examination revealed an intraosseous cavernous hemangioma. Follow-up imaging 1 year later with cone beam computed tomography re-vealed recurrence of the lesion. The conclusion of this paper; when a bone hard, well-shaped mass was seen in the mandible, the possibility of intraosseous hemangioma must be remembered and before surgical procedure detailed radiographic evaluation should be performed.Key words: Hemangioma, intraosseous, mandible, cavernous, cbct.doi:10.4317/jced.52864http://dx.doi.org/10.4317/jced.52864IntroductionPrimary intraosseous hemangioma is a benign, slow growing neoplasm that comprises 1% of all benign tu-mors arising from the bones. Hemangiomas mostly arise from the soft tissues, and intraosseous hemangiomas are uncommon (1-4). The sites most commonly involved are the vertebral column and the calvarium in the maxi-llofacial region. They have been reported in the man-dible, maxilla, and the nasal bones (5-7). Intraosseous hemangioma usually occurs in adults, and many patients are in fifth decade of life, with a female predominan-ce. The diagnosis can be difficult, and usually neither the history nor the physical findings lead the clinician to suspect an osseous cavernous hemangioma (1,5,8). They usually become symptomatic with slow-growing, painful swelling (3,8). The radiographic presentation of the cavernous hemangioma is nonspecific. The lesion appears as a radiolucency that could have unilocular or multilocular, reticulated, honeycomb, or sunburst appearance when viewed tangentially. These findings Article Number: 52864                http://www.medicinaoral.com/odo/indice.htm© Medicina Oral S. L. C.I.F. B 96689336 - eISSN: 1989-5488eMail:  jced@jced.esIndexed in:PubmedPubmed Central® (PMC)ScopusDOI® SystemElif B, Derya Y, Gulperi K, Sevgi B. Intraosseous cavernous hemangioma ın the mandible: A case report. J Clin Exp Dent. 2017;9(1):e153-6.http://www.medicinaoral.com/odo/volumenes/v9i1/jcedv9i1p153.pdfJ Clin Exp Dent. 2017;9(1):e153-6.                                                                                                                                                                          Intraosseous hemangioma in mandiblee154Fig. 1. Occlusal radiograph revealed a well defined lesion surrounded by a sclerotic margin, containing reactive bone spicules which are characteristic for intraosseous cavernous hemangioma. Fig. 2. Cone beam computed tomography scan shows the in-traosseous cavernous hemangioma. No signs of destruction of the adjacent tissue are present. A) Coronal section showing the bone mass originated from left lingual mandibular region. B) Axial section; the formation of reactive spicula produced by the lesion was indicated by the arrow.Fig. 3. A,B) Histopathologic slide show that blood-filled vascular space between the bone trabeculae.are generally called ‘sunburst appearance’ for skull he-mangioma, ‘corduroy- like appearance’ for vertebral hemangioma, and ‘honeycomb appearance’ for rib he-mangioma. Most cases of the bone hemangioma can be diagnosed with characteristic radiographic findings that reflect the formation of reactive spicula produced by the lesion. However, this classic feature may be absent in many cases, which present only as lytic or expanding dense bone masses (1,6,9). In this report, we presented a male patient on the third decade of life with intraosseous cavernous hemangioma in the mandibleand discussed the clinical and radiological features with characteristic radiographic findings.Case Report A 28-year-old male patient attended to our clinic with a complaint of painless swelling of mandible. The pa-tient first noticed the swelling about two years ago when brushing his teeth. This swelling which has more than doubled in size since the difference has led to difficulty in speaking since 3 months. Intraoral examination re-vealed bone-hard, smooth-surfaced, immobile mass in the left mandibular lingual crest. Computed tomogra-phy (CT) that had taken at her previous visit at another hospital examined,maxillofacial computed tomography demonstrated a 21x13 mm bone mass originated from lingual cortex of mandible. Panoramic and occlusal ra-diography imaging performed. Panoramic radiograph of the patient did not reveal any pathology. Occlusal radio-graphy revealed a well defined lesion with radiopaque contours, containing reactive bone spicules (Fig. 1). Pre-liminary diagnosis was made as torus or intraosseous ca-vernous hemangioma. The lesion surgically was excised and pathological examination revealed an intraosseous cavernous hemangioma. Follow-up examination and imaging 1 year later with cone beam computed tomogra-phy (CBCT) revealed recurrence of the lesion.  CBCT showed that the lesion was 25.3x14x12.1 mm in size, the bone mass originated from left lingual mandibular region and the formation of reactive spicula produced by the lesion (Fig. 2). Total excision of the lesion and a margin of surrounding uninvolved bone was perfor-med. It was diagnosed again as intraosseous cavernous hemangioma (Fig. 3).DiscussionBenign vascular anomalies can be broadly classified as hemangiomas (vasoproliferative tumors) and vascular malformations. Vascular anomalies often affect the soft tissues of the maxillofacial region but are rarely found in visceral organs or bone. The diagnosis and management of vascular lesions continue to present diagnostic and therapeutic challenges to all. This is in part because of lack of agreement regarding the nosology and classifica-tions of the lesions. Many authors use the term heman-gioma to describe or qualify vascular malformations and vascular anomalies, whereas others continue to use the term cavernous hemangioma for venous malformation and port-wine stain for capillary malformation, venous malformation, and arteriovenous malformations (AVM) (10-13).AVMs are a complex network of intercommunicating arterial and venous structures. The presence of nerve in AVM provides an additional diagnostic criterion that can J Clin Exp Dent. 2017;9(1):e153-6.                                                                                                                                                                          Intraosseous hemangioma in mandiblee155be readily used to differentiate AVMs from hemangio-mas (14).Hemangiomas are benign vascular neoplasms characte-rized by an abnormal proliferation of blood vessels. They may occur in any vascularized tissue including skin, subcutaneous tissue, muscle, and bone. The microsco-pe appearance has two major classifications; cavernous and capillary types. The cavernous hemangioma is com-posed of large thin-walled vessels and sinusoids lined with a single layer of endothelium. However, a small fine vascular network filled with blood forms the capi-llary hemangioma. Capillary hemangiomas are usually present at birth. In contrast, most cavernous hemangio-mas occur in adult hood and almost all intraosseous he-mangiomas of the facial skeleton are to be cavernous type (1,6,10,11). In this report the presented mandibular intraosseous hemangioma case was also a 28-year-old adult patient.The pathogenesis of intraosseous hemangiomas remains unknown. Nonetheless, local trauma may be a cause, because many patients with intraosseous hemangiomas have had a history of local trauma (4), but in our pa-tient, there was no such predisposition for intraosseous hemangioma development.As intraosseous hemangioma tends to grow very slowly, it remains clinically silent until the tumor becomes large. The radiographic appearance of central hemangioma is not pathognomonic and can simulate many other bone lesions. In this case; the formation of reactive spicula produced by the lesion that was observed in the occlusal radiography is the classic radiographic feature for the tumor (10,11). Preoperative radiological studies include conventional radiography, CT and magnetic resonance imaging (MRI) (10,12). CT is considered the most use-ful imaging technique because of its unique characte-rization of trabecular and cortical details, showing the honey comb appearance. MRI is an effective method for identifying the depth of tumor extension into the soft tissues (2,12,13). In our case, CBCT scans revealed a mandibular mass with the formation of reactive spicula, which is characteristic to intraosseous hemangioma.Hemangiomas should be considered in the differential diagnosis of multilocular lesions involving the body of the ramus and body of the mandible. The differential diagnoses for intraosseous cavernous hemangioma in-clude AVM, osteoma, langerhans cell histiocytosis, fi-brous dysplasia, dermoid tumor and multiple myeloma (9-11). Histologically; osteomas may be of two types: compact and cancellous. Periosteal osteomas are mostly cancellous type which is characterized by bony trabe-culae and fibro-fatty marrow enclosing osteoblast with an architecture resembling mature bone. In cavernous hemangioma, microscobic examination shows many dilated, thin walled vascular spaces lined by benign en-dothelial cells containing a large number of red blood cells rather than fibro-fatty marrow space. Fibrous con-nective tissue stroma was seen supporting the spaces interspersed among the trabeculae (6,14). The presen-ce of arteries, arterioles, or both as an integral part of the lesions (as shown by elastic tissue stains) is often used as a diagnostic criterion for differentiating AVMs from hemangiomas.In most cases soft tissue changes such as blood discharge, bluish discoloration, mobility of the teeth suggest a vascular lesion. When a heman-gioma produces a sunray spiculated bone pattern at its periphery, the appearance may be difficult to differentia-te from an osteogenic sarcoma (10,11,14). In our case, it can be seen many dilated, thin-walled vascular spa-ces lined by benign endothelial cells separated by bone septum and oedematous-fibrous stroma supporting the bloodfilled vascular spaces.The most effective treatment to prevent recurrence of in-traosseous hemangiomas is to remove the tumor comple-tely without any functional deficit, cosmetic deformity, or significant tissue loss (8). The biopsy and surgery of the lesion should be done cautiously because of the risk of severe bleeding. In the past, radiotherapy and scle-rotherapy were the treatment of choice. Radiotherapy may only be reserved for cases in which surgery is not feasible due to the adverse effects such as telangiectasia, retardation of growth of bones and teeth, tissue necro-sis, and malignant degeneration. Embolization and scle-rotherapy are sometimes performed, but the efficacy is palliative in many cases. Preoperative embolization has been reported to reduce the risk of intraoperative severe bleeding. In contrast, other reports have shown that vas-cular manipulation is not necessary if en bloc excision can be performed with a sufficient margin (1,8,10-12).In our case, we assessed by preoperative CBCT scans that complete tumor excision with sufficient margins was possible, and could be performed without severe bleeding.The diagnosis of facial bone hemangiomas is somewhat difficult to make, because the lesion is usually asymp-tomatic, the most affected patients are women in their fourth and fifth decades (3,5,9), but in this report we presented a male patient on the third decade of life with intraosseous cavernous hemangioma. Early diagnosis of intraosseous hemangioma is essential both for preven-ting uncontrollable haemorrhage and even death during biopsy or surgery and to a lesser cosmetic deformity. Therefore, dentists should be aware of the radiological features of this vascular bone lesion (10,11). Differential diagnosis of the lesion could be made after  the histo-pathological evaluation in this case, thus when a bone hard, well-shaped mass was seen in the mandible, the possibility of intraosseous hemangioma must be remem-bered and before surgical procedure detailed radiogra-phic evaluation should be performed.J Clin Exp Dent. 2017;9(1):e153-6.                                                                                                                                                                          Intraosseous hemangioma in mandiblee156References1. Akiyama K, Karaki M, Osaki Y, Takeda J, Mori N. Intraosseous Ca-vernous Hemangioma of the Middle Turbinate. Auris Nasus Larynx. 2011;38:516-8.2. Suzuki Y, Ikeda H, Matsumoto K. Neuroradiological Features of Intraosseous Cavernous Hemangioma--Case Report. Neurol Med Chir (Tokyo). 2001;41:279-82.3. Buhl R, Barth H, Dörner L, Nabavi A, Rohr A, Mehdorn HM. De Novo Development of Intraosseous Cavernous Hemangioma. 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Intraosseous Cavernous Hemangioma: A Rare Presentation in Maxilla. Eplasty. 2014;14:e35.11. Zlotogorski A, Buchner A, Kaffe I, Schwartz-Arad D. Radiologi-cal Features of Central Hemangioma Of The Jaws. Dentomaxillofac Radiol. 2005;34:292-6.12. Yucel E, Akkaya H, Gürkanlar D, Ergun T. Congenital Cavernous Hemangioma of the Skull. Turk Neurosurg. 2011;21:645-7.13. Handa H, Naidu GS, Dara BG, Deshpande A, Raghavendra R. Di-verse imaging characteristics of a mandibular intraosseous vascular lesion. Imaging Sci Dent.2014;44:67-73.14. Soni S, Bhargava A. Revisiting Peripheral Osteoma of the Mandi-ble with Case Series and Review of Literature. Indian J Otolaryngol Head Neck Surg. 2014;66:212-8.Conflict of InterestThe authors declare that they have no conflict of interest.

Intraosseous cavernous hemangioma in the mandible: a case report

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