Forty-five-months follow-up of a minimally invasive, interdisciplinary treated hemangioma of the mandible with a high risk of severe bleeding – a case report

The present case illustrates a rare example of an intraosseous hemangioma, which has been well-documented with respect to its interdisciplinary treatment and its follow-up history. In the current case, the diagnostic finding was an incidental finding after fabrication of a panoramic x-ray. In 2007, the patient firstly attained knowledge of an intrabony osteolytic process in the left mandible. At that time point as well as ten years later, both panoramic radiographs showed a homogeneous, unilocular, well-defined osteolytic lesion. This is in contrast to the radiological appearance of most of the intraosseous hemangiomas, which is described as soap bubble, honeycomb-like appearance, a sunray appearance, caused by bony trabeculae in the lesion or a poorly defined osteolytic lesion [14, 15]. The variable appearance of central hemangioma in different projections was also described [16]. Differential diagnosis of for example aneurysmal bone cyst, fibrous dysplasia, ameloblastoma, osteoma, osteosarcoma, giant cell lesion, residual cyst, multiple myeloma, and myxoma could not be radiographically distinguished [17]. Therefore, it is not possible to diagnose an intraosseous hemangioma based on dental radiology alone. Clinical findings are also varying. Many patients do not report about any symptoms, as described in the current case. But bony swellings of different size, sometimes causing asymmetry of the face, paraesthesia of lips as well as the mental region, pulsation or discomfort can occur as well [12, 18,19,20]. Symptoms regarding teeth and the alveolar process such as bluish discoloration of the gingiva, mobile or displaced teeth, agenesis of teeth, alteration of the dental arch or seeping gingival bleeding which were also described, were not found in the present case. Moreover, the patient did not report about early eruption of the permanent teeth, which could also be related to central hemangiomas of the jaw [11, 18, 21,22,23,24]. Osteolytic lesions that are of extraordinary or inexplicable origin, as depicted in the current case, should be biopsied and clarified [24]. From a critical point of view, advantages of intraoperative biopsies are to be contrasted with the disadvantages of the same. The main advantage of a biopsy is seen in the histological verification of a diagnosis. The so collected details allow a precise surgical procedure according to the respective pathology. As a possible disadvantage of a biopsy, severe complications could result from the lack of knowledge regarding the lesion. For instance, if an intraosseous hemangioma is present like in the present case and a biopsy would have been taken, serious complications in form of severe bleeding would be possible during surgery or diagnostic confirmation. It was described that patients died after extractions of teeth with hemangioma association or even after puncture biopsy of a hemangioma of the jaw [10]. For this reason, it is important to be as careful as possible during surgery and to gently remove every tissue layer, without touching the lesion itself, as it was performed in the current situation. The vestibular access provided an optimal overview and allowed to depict the mental foramen for estimation of the mandibular nerve pathway. This was also necessary to protect the mental nerve. If hemangiomas, especially extensive ones, are expected because of radiological appearance and clinical signs, angiography before surgery could be a very useful tool for detecting feeding vessels and planning the approach of therapy [25]. In the present case, it was assumed that the lesion would be a sort of cyst. Accordingly, there was no need to apply angiography. Treatment options that provide complete histopathological investigation reach from partial mandibuloectomy to minimally invasive approaches such as curettage or surgical removal of the lesion with or without ligation or embolization of the feeding vessels [11]. The option, to omit any invasive intervention, in particular, in the present case could have led to root resorptions of neighboring teeth [26], a compression of the inferior alveolar nerve accompanied by paresthesia or pain, and a further expansion to the mental foramen, where a removal would be much more complicated or a reduction of the lower jaw stability [27]. However, since the patient was originally asymptomatic for more than ten years, it was an option to left the lesion untreated [28]. Indeed, the consequence, to clarify the diagnosis of the lesion, relied on the fact, that the inferior alveolar nerve was not detectable within the CBCT. The plan of sample taking in order to histologically verify the most probable diagnosis of an infected radicular cyst, was rejected intraoperatively based on the atypical appearance of the lesion. Instead, the decision was made to completely remove the lesion, carefully as recommended by other authors [11, 18].In the present case, the size of the mass as well as its texture allowed for a gentle procedure resulting in a preservation of teeth and the mental nerve resulting in a nearly four years follow-up without recurrence of the intraosseous lesion.

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