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BMJ Case Rep. 2015; 2015: bcr2014208975.
Published online 2015 March 27. doi:  10.1136/bcr-2014-208975
PMCID: PMC4386299

Locally aggressive cavernous haemangioma of the mandible: an unusual presentation

Description

Intraosseous haemangiomas, especially of the jaw bones, are rare benign vasoformative neoplasms of endothelial origin. They usually present as a firm, painless swelling of the jaw bone with or without facial asymmetry.1 Occasionally bleeding from the gingiva, mobility of teeth, pulsations, audible bruits and paraesthesia may occur. Clinically, the lesion may mimic ameloblastoma, arteriovenous malformation, central giant cell granuloma and aneurysmal bone cyst.

Radiographically, a multilocular radiolucency with ill-defined margins is seen. CT, MRI and angiography may be needed to delineate the lesion.2 Since the radiological appearance is not pathognomonic, ameloblastoma, central giant cell granuloma, osteosarcoma, odontogenic myxoma, dentigerous cyst, fibrous dysplasia, keratocystic odontogenic tumour, aneurysmal bone cyst, schwannoma of inferior alveolar nerve and multiple myeloma may be considered in the differential diagnosis.

Histologically, haemangiomas consist of a mass of endothelial cells forming vascular spaces of varying size, interspersed with fibrous connective tissue stroma and bony trabeculae.1

Haemangiomas may occur as a part of a variety of syndromes such as Osler-Weber-Rendu syndrome, Klippel-Trenaunay syndrome, Kasabach-Merritt syndrome, Sturge-Weber syndrome, Parkes Weber syndrome, CLOVES syndrome, Maffucci syndrome, Cobb syndrome, PHACES syndrome and Blue rubber bleb nevus syndrome.3

Large haemangiomas tend to bleed and embolisation of the feeder vessel prior to surgery reduces the risk of haemorrhage. Propanolol is an antihypertensive drug now being used successfully in treating large haemangiomas of the head and neck region, by inducing vasoconstriction and hypoxia, apoptosis of capillary endothelial cells and decreasing the angiogenic factors such as the vascular endothelial growth factor.4

A 23-year-old woman reported with swelling on the right side of the face with a growth in the right mandibular molar region for 3 months. History revealed that she had gradual swelling, mobility and exfoliation of mandibular molar teeth with limitation of mouth opening.

A diffuse swelling and facial asymmetry were seen on the right side, with an inter-incisal distance of 25 mm. An irregular, erythematous, proliferative growth measuring 4×1.5 cm was seen on the right mandibular alveolus (figure 1); it was ulcerated in the centre and non-tender on palpation. CT scan revealed a lytic lesion in the right mandible, with bicortical expansion, perforation and destruction of the cortical plates (figure 2). The lesion was provisionally diagnosed as a benign odontogenic neoplasm and biopsy was performed. Histological evaluation of the lesion revealed numerous large endothelium lined, thin-walled and thick-walled vascular spaces with erythrocytes, with the evidence of a large thick-walled feeder vessel (figure 3). A definitive diagnosis of cavernous haemangioma of mandible was made and subsequent hemimandibulectomy was performed.

Figure 1
An irregular erythematous growth seen on the right retromolar region with surface ulceration.
Figure 2
CT scan showing expansile lesion in the right body of the mandible with bicortical destruction in axial section (A), soft tissue growth in coronal section (B) and irregular bone destruction with bony spicules seen in the right mandibular alveolus in the ...
Figure 3
H&E section showing numerous large endothelium-lined, thin-walled and thick-walled vascular spaces with erythrocytes (A, original magnification ×10), endothelial cell proliferation (B, original magnification ×40), giant cells in ...

It may be emphasised that, occasionally, haemangiomas do not bleed, thereby mimicking common odontogenic/non-odontogenic neoplasms.

Learning points

  • Central haemangiomas of mandible are rare lesions.
  • They present clinically as an expansile swelling with multilocular radiolucency on the radiographs, mimicking various odontogenic and non-odontogenic neoplasms.
  • Central haemangiomas must be included in the differential diagnosis of multilocular jaw lesions.

Footnotes

Competing interests: None.

Patient consent: Obtained.

Provenance and peer review: Not commissioned; externally peer reviewed.

References

1. Eliot CA, Castle ET Intraosseous hemangioma of the anterior mandible. Head Neck Pathol 2010;4:123–5 doi:10.1007/s12105-010-0170-x [PMC free article] [PubMed]
2. Oliveira GG, García-Rozado A, Rey RL Intraosseous mandibular hemangioma. A case report and review of the literature. Med Oral Patol Oral Cir Bucal 2008;13:E496–8. [PubMed]
3. Nosher JL, Murillo PG, Liszewski M et al. Vascular anomalies: a pictorial review of nomenclature, diagnosis and treatment. World J Radiol 2014;6:677–92 doi:10.4329/wjr.v6.i9.677 [PMC free article] [PubMed]
4. Zimmermann AP, Wiegand S, Werner JA et al. Propranolol therapy for infantile haemangiomas: review of the literature. Int J Pediatr Otorhinolaryngol 2010;74:338–42 doi:10.1016/j.ijporl.2010.01.001 [PubMed]

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