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|LETTER TO EDITOR
|Year : 2016 | Volume
| Issue : 6 | Page : 1347-1351
Extracranial temporal cavernous hemangioma: Differential diagnosis, and a review of literature
Mazda K Turel1, Tim-Rasmus Kiehl2, Fred Gentili1
1 Division of Neurosurgery, Toronto Western Hospital, Toronto, Ontario, Canada
2 Department of Laboratory Medicine and Pathobiology, University of Toronto, Toronto, Ontario, Canada
|Date of Web Publication||11-Nov-2016|
Mazda K Turel
Division of Neurosurgery, Toronto Western Hospital, Toronto, Ontario
Source of Support: None, Conflict of Interest: None
|How to cite this article:|
Turel MK, Kiehl TR, Gentili F. Extracranial temporal cavernous hemangioma: Differential diagnosis, and a review of literature. Neurol India 2016;64:1347-51
A small swelling in the temporal region is often benign and managed conservatively unless it causes local pain, shows an increase in size or becomes cosmetically disfiguring. The differential diagnosis of pathology in this region is quite varied and includes soft tissue tumors such as lipoma, schwannoma, dermoid, mesenchymal angiolipoma, and spindle cell hemangioendothelioma; vascular malformations such as cavernous hemangiomas and arterio-venous malformations (AVMs); and malignant lesions such as liposarcomas and angiosarcomas. Occasionally, extracranial extensions of intracranial meningiomas/hemangiopericytomas or osteoma of the calvarium can present in a similar fashion. Enlarged lymph nodes, myositis ossificans, and temporal arteritis complete the list.
We discuss the case of a 61-year-old female patient with an intramuscular cavernous hemangioma of the temporalis muscle and review varying presentations and the differential diagnosis. Due their rarity and vague presentations, this entity presents significant diagnostic challenges.
This 61-year-old female patient presented with a small swelling in the left temporal region of her head 4 years previously. She was initially asymptomatic and her family physician, who suspected that this was a lipoma, recommended a conservative approach. More recently, she noticed a significant growth in the size of the lesion over 1 year, which prompted a referral. Apart from the increasing size of the lesion, she was otherwise asymptomatic. Examination revealed a smooth, nonpulsatile, nontender, immobile globular swelling in the left temporal region. It was firm and noncompressible. There was no audible bruit. On examination, she had no neurological deficit.
Magnetic resonance imaging (MRI) revealed a 3.5 cm × 2.5 cm × 4.5 cm lesion within the left temporalis muscle. There was no involvement of the underlying bone or scalp. The lesion was hypointense on T1-weighted images and hyperintense on T2-weighted images and showed heterogeneous enhancement with administration of gadolinium [Figure 1]. There was neither restriction of diffusion on the diffusion-weighted imaging nor was there evidence of hemorrhage on susceptibility-weighted imaging [Figure 2]. An angiogram ruled out an AVM, but showed evidence of minimal neovascularity of the mass. There were two small areas of contrast blush in the superior and inferior aspect of the lesion that appeared in the arterial phase and persisted, slightly enlarging into the venous phase. However, the bulk of the lesion did not demonstrate a vascular blush [Figure 3]. The preoperative diagnosis was that of a cavernous hemangioma. Surgery was recommended both for the confirmation of diagnosis as well as cosmesis. A frontal-temporal skin flap was turned forward to the orbital rim. The tumor was then seen and felt within the temporalis muscle, from which it was dissected en bloc, excising it radically. There was no infratemporal or orbital extension. Postoperatively, the patient developed a left frontalis paresis that had significantly improved at 4 weeks. The postoperative computed tomography (CT) scan done showed no residual lesion [Figure 4].
|Figure 1: (a) T2-weighted axial, (b) T1-weighted coronal with gadolinium, and (c) T1-weighted plain sagittal magnetic resonance imaging of the head showing a 4.5 cm T1-weighted hypointense, T2-weighted hyperintense, and heterogeneously enhancing well-defined intramuscular lesion in the left temporalis muscle|
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|Figure 2: (a) Susceptibility-weighted imaging (b) diffusion-weighted imaging and (c) apparent diffusion coefficient axial images showing neither evidence of hemorrhage within the lesion nor restriction of diffusion|
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|Figure 3: The digital subtraction angiogram with (a) left internal carotid artery injection showing a normal study. The left external carotid injection in the (b) arterial phase shows small areas of tumor blush in the superior and inferior aspects of the tumor (white arrows). These areas persist in the (c) venous phase of the angiogram showing a slight enlargement|
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|Figure 4: The postoperative (a) coronal and (b) axial images do not show any residual tumor|
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Histopathology showed a proliferation of large, thick-walled vascular channels with foci of lymphoid inflammation and pieces of striated muscle. This was consistent with a cavernous hemangioma [Figure 5].
|Figure 5: (a) Low power and (b) high power images showing proliferation of large, thick-walled vascular channels with foci of lymphoid inflammation and pieces of striated muscle, consistent with a cavernous hemangioma|
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Less than 1% of hemangiomas are intramuscular and only 14% occur in the head and neck.,,, The masseter and trapezius muscles are the most frequently affected sites., Involvement of the orbicularis oris, mentalis, digastric and mylohyoid muscles has also been reported. Intramuscular hemangiomas within the temporalis muscle are extremely rare. Histologically they are classified as capillary (small vessel), cavernous (large vessel), and mixed, depending on the predominant vascular channels that they are composed of. Minor trauma, hormonal status, or excessive muscle contraction is believed to play a role in their growth by inducing changes in the blood flow. They have been reported to grow to a giant size, cross tissue planes, and can rarely have an intracranial extension, producing a mass effect on the brain. [Table 1] discusses the clinical, radiological, and pathological differential diagnosis of these lesions within the temporalis muscle.
|Table 1: Characteristics of cavernous hemangioma of the temporalis muscle and its common differential diagnosis|
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While in most patients they present as a painless, progressive swelling within the temporalis muscle causing facial asymmetry, there are reports of cases presenting with significant pain and temporo-mandibular joint dysfunction. They can change in size with a Valsalva maneuver due to large venous channels within. They are also known to occasionally develop several years after blunt trauma to the head. The diagnosis is more straightforward when there is an associated cutaneous change, but this is uncommon.
While classically described as soft and compressible, cavernous hemangiomas can occasionally be firm and mistaken for soft tissue tumors. Rarely, they can be pulsatile and may have a bruit mimicking an AVM. Itosaka et al., have described a case where repeated hemorrhages within the tumor resulted in spontaneous regression of the hemangioma. While a long-standing history of a slowly growing painless mass suggests a benign pathology, a short history of a rapidly growing lesion could signal something more sinister and should not be managed expectantly.
MRI is the investigation of choice in defining the nature of the tumor and in differentiating its vascular nature from soft tissue elements. These lesions are most often well-circumscribed within the muscle, isointense on T1-weighted images, and hyperintense on T2-weighted images. Occasionally, a hypointense rim similar to what is seen in cavernous malformations of the brain is also visible on the T2-weighted images. Some authors suggest that a serpiginous pattern on T2-weighted images is characteristic. The heterogeneity on enhancement is due to thrombosis, hemosiderin deposition, and calcification. Phleboliths can sometimes be seen inside the lesion as low-intensity areas. Lipomas, angiolipomas, and dermoids are usually hyperintense on T1W images, whereas schwannomas and meningiomas have a more uniform enhancement. A CT scan may show remodeling of the underlying skull in long-standing cases.
Angiography is helpful to differentiate cavernous hemangiomas from an AVM. AVMs will have either a compact or a diffuse intramuscular nidus and the presence of early draining veins. The main arterial feeders are branches of the superficial temporal and internal maxillary artery, and the draining veins are commonly the facial and external jugular. Cavernous malformations may be angiographically occult (involuting type) or they may show uniform hypervascularity (proliferating type). There may be separate visible areas of blush due to pooling of stagnant blood, as seen in our case. Angiolipomas, on the other hand, show coarse neovascularity and mottled staining during the capillary phase. These findings should guide in the diagnosis, avoiding the need for an open biopsy, which could lead to unwarranted hemorrhage.
Hemangiomas are benign and not known to metastasize, but can have local recurrence due to infiltration of muscle and soft tissue. Capillary hemangiomas are more aggressive than the cavernous and mixed types. The objective of surgery should be total removal, preferably in an en bloc fashion. Care should be taken to prevent injury to the temporal and auricular branches of the facial nerve because if permanent, this complication could be more disfiguring than the swelling itself. Ohyama et al., used an electric nerve stimulator where the facial nerve ran over the skin as well as subfacially to avoid this complication. Steroids, sclerosing agents, and radiation are other proposed treatment modalities but are not consistently effective.
Due to the wide spectrum of diagnostic possibilities, a soft tissue swelling in the temporal region requires careful evaluation with an MRI as well as angiography, as the diagnosis has a significant impact on the management of these lesions. The definitive diagnosis is usually made by histopathological evaluation. Careful surgical planning and total removal usually result in a good result.
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Conflicts of interest
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[Figure 1], [Figure 2], [Figure 3], [Figure 4], [Figure 5]