Rare Case of Multifocal Intraosseous Cavernous Hemangioma Posing as Malignancy

Clinical Features

A 58-year-old female with no significant medical history presented with complaints of progressive pain in her left hip, right shoulder and chest. The pain was described as dull and persistent, worsening with movement. She also reported mild swelling in the left hip area, with no history of trauma. Despite the absence of systemic symptoms such as fever, weight loss, or night sweats, the localized bone pain raised concern for a possible malignancy.

On physical examination, the patient exhibited tenderness over the right shoulder, left hip, and sternum. No palpable masses or neurological deficits were noted. Given the chronicity and persistent nature of the pain, malignancy was suspected, prompting further investigation. Tumor markers including CEA, CA125, CA199, and AFP, exhibited normal levels.

Imaging Findings

MRI of the right humerus demonstrated well-defined lobulated lytic expansile lesion with areas of high-signal intensity on T2-weighted images without extraosseous soft tissue or pathological fracture however, suggesting a vascular etiology.

A whole-body positron emission tomography-computed tomography (PET/CT) scan [Figure 1] was performed to evaluate for possible malignancy. The PET/CT showed increased fluorodeoxyglucose uptake in the humerus, femur, and sternal lesions. The lesions were expansile and associated with cortical thinning and focal breakdown of borders. The lesions showed necrotic regions and calcifications. However, there was a lack of periosteal reaction or associated infiltrative soft tissue component. This hypermetabolic activity prompted further investigation to distinguish these benign vascular lesions from metastatic disease.[12]

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Figure 1 (a) Fluorodeoxyglucose positron emission tomography-computed tomography (PET/CT) Maximum intensity projection (MIP) shows multiple foci of tracer concentration in humerus, sternum, pelvis, and femur. (b and c) Axial PET/CT and CT image shows expansile lytic lesion in left pelvic bone with areas of necrosis and cortical breakdown, however no periosteal reaction or extralesional soft tissue component noted. Matrix of lesion does not show any osteoid or chondroid component within. (d and e) Sagittal PET/CT and CT image shows expansile lytic lesion in humerus. (f and g) Axial image PET/CT and CT shows similar lesion in sternum

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Pathology Findings

Given the radiologic and PET/CT findings, a biopsy of the femoral lesion was performed. The histological examination confirmed the diagnosis of an intraosseous cavernous hemangioma (OCH), with blood filled spaces lined by endothelial cells [Figure 2]. The absence of atypical cells, mitotic activity, or features suggestive of malignancy ruled out a malignant process. These findings are consistent with the benign vascular nature of the lesion, supporting the diagnosis of an OCH.[2]

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Figure 2 Hematoxylin and eosin microscopy image shows tumor composed of vascular spaces of varying sizes lined by endothelial cells filled with hemorrhage suggestive of Intraosseous cavernous hemangiomas

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Management and Intervention

The diagnosis of multiple OCHs prompted a conservative approach to management. While surgery was not deemed necessary, the patient was started on zoledronic acid, a bisphosphonate, to manage her bone pain and prevent potential fractures, which can be a complication of these vascular lesions. Bisphosphonates, particularly zoledronic acid, have been shown to effectively alleviate pain in benign bone tumors and improve bone mineral density, reducing the risk of fractures associated with weakened bone structure.[13]

Following treatment, the patient experienced significant relief from her symptoms, with a marked reduction in pain and improved mobility.

Follow-up PET/CT imaging after 1 year demonstrated no evidence of progression of the lesions, and the patient’s symptoms remained well-controlled.

Other interventions include can intralesional excision, radiotherapy, or occasionally cement injection for small, well-defined lesions. For larger lesions, more complex procedures such as curettage with graft placement or en bloc resection may be necessary to remove the tumor completely.

Discussion

OCHs are rare, benign lesions that often present with localized bone pain, though they can be asymptomatic. Their radiological appearance may mimic that of malignant bone tumors, particularly on PET/CT scans, as seen in this case. The increased metabolic activity on PET/CT scans can pose a diagnostic challenge, especially in cases involving weight-bearing bones such as the femur and sternum. Histopathological examination remains the gold standard for differentiating between benign vascular lesions and malignancies.[123]

Zoledronic acid, a potent bisphosphonate, has shown efficacy in treating bone pain associated with benign bone tumors, such as hemangiomas. In this case, it was successfully used to manage the patient’s symptoms, and no fractures or complications were noted during the follow-up. This highlights the role of bisphosphonates in managing benign bone lesions and improving quality of life for affected patients.[4]

Conclusion

OCHs, while rare, should be considered in the differential diagnosis of lytic bone lesions, particularly when presenting with pain or swelling. Radiological findings, including PET/CT, may suggest malignancy; however, histopathological confirmation is essential for accurate diagnosis. Zoledronic acid therapy offers an effective treatment for symptom control and prevention of fractures in these benign vascular lesions, as demonstrated in this case.