Keywords
frontal bone - intraosseous hemangioma - skull - surgical management
Importance
Intraosseous cavernous hemangiomas are rare, benign tumors.[1]
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[18] These lesions represent 0.2% of primary cranial bone tumors and predominantly present
as solitary lesions in the frontal or parietal bone.[1]
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[32] They often involve the outer table and diploic space, while sparing the inner table,
and are treated with en bloc surgical resection and cranioplasty.[2]
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[20] Few reports have described intraosseous cavernous hemangiomas extending beyond the
inner table or with substantial involvement of the bifrontal calvarium.[2]
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[24] We present a case highlighting the surgical management of a frontal hemangioma with
intradural extension, including a staged reconstruction aimed at optimizing postoperative
cosmesis.
Clinical Presentation
An otherwise healthy 38-year-old female presented for evaluation of a large, painless
mass on the forehead that slowly progressed in size over 7 months. Physical examination
revealed a large, palpable, nontender mass in the frontal region. She was worked up
with a noncontrasted computed tomography (CT) head demonstrating a 5.5 × 3.2 × 5.6 cm
well-circumscribed mass with an internal sunburst pattern of trabecular thickening,
expanding through the inner and outer tables of the right anterior frontal bone ([Fig. 1]). A contrast-enhanced magnetic resonance imaging (MRI) of the lesion was obtained
for better characterization, which demonstrated a heterogeneously enhancing lesion
with T2 hyperintensity causing significant mass effect on the underlying brain parenchyma
([Fig. 2]).
Fig. 1 Noncontrasted computed tomography (CT) preoperative coronal (A) and sagittal (B) images displaying well-circumscribed mass with trabecular thickening.
Fig. 2 Preoperative T1-weighted postcontrast magnetic resonance imaging demonstrating large
heterogeneously enhancing extra-axial lesion with mass effect on the underlying brain
parenchyma. Representative coronal (A) and sagittal images (B).
While the lesion was favored to represent an intraosseous hemangioma, metastasis remained
on the differential. A CT of the chest, abdomen, and pelvis was obtained. The chest
CT revealed a 2.4 × 3.3 cm right lateral breast mass most consistent with a hamartoma
on follow-up mammogram. Given the size of the frontal lesion and associated mass effect
on the underlying brain parenchyma, surgical resection of the lesion was recommended.
She underwent evaluation for preoperative embolization to assist with resection of
the lesion. During the diagnostic cerebral arteriogram, the anterior falcine artery
was found to be the lesion's major blood supply and was secondarily supplied by a
branch of the right superior temporal artery (STA). The anterior falcine artery had
an anastomosis with the right ophthalmic artery, therefore endovascular embolization
was not further pursued given the risk of postprocedural blindness.
The patient was taken to the operating room for surgical intervention. A bicoronal
incision was made ([Fig. 3]), and the branch of the STA found angiographically to be supplying the lesion was
cauterized. After adequate exposure was obtained, a 5-mm diamond burr was used to
create a full-thickness trough around the lesion. The tumor was then elevated and
noted to be extending intradurally with adherence to the underlying brain parenchyma
([Fig. 4]). The bulk of the tumor was removed to better visualize the intradural component,
as it was deemed unsafe to remove the mass in an en bloc fashion ([Fig. 5]). The remainder of the tumor was gently dissected from the underlying cortex and
removed in a piecemeal fashion. A dural patch was sewn in and a temporary titanium
mesh cranioplasty was placed. The final pathologic diagnosis confirmed intraosseous
hemangioma.
Fig. 3 Intraoperative exposure of large frontal bone lesion utilizing a bicoronal incision.
Fig. 4 Intraoperative elevation of frontal bone lesion revealing intradural extension of
tumor and adherence to underlying brain parenchyma.
Fig. 5 Intraoperative photos display resected bulk of frontal bone tumor (A) and the resection cavity with the exposed underlying brain parenchyma (B).
Postoperatively, a thin-cut CT was obtained to design a custom polymethyl methacrylate
implant. The patient returned to the operating room 3 weeks later for removal of the
titanium mesh and placement of the permanent implant ([Fig. 6]).
Fig. 6 Postoperative computed tomography (CT) coronal (A) and sagittal (B) images after placement of permanent implant 3 weeks after the initial resection.
Discussion
Intraosseous cavernous hemangiomas are benign vascular tumors. They present as firm,
solitary masses that slowly progress over months or years.[1]
[2]
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[9]
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[15]
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[19]
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[25]
[28]
[33]
[34] Intracranial extension of a calvarial based intraosseous hemangioma is an infrequent
occurrence.[1]
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[20] Few prior reports have noted expansion of these lesions beyond the dural plane.[2]
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[24] Park et al and Khanam et al describe patients with intracranial expansion loosely
attached to the dura, while Peterson et al describe a patient with extension through
the dura and anastomoses with the thin-walled vessels on the surface of the cerebellum.[8]
[11]
[13] Further, Nasi et al describe a case with complete erosion of the dural plane and
invasion of the subdural space.[2] Similar to our patient, these cases consisted of a progressive mass with dural involvement
and mass effect. To our knowledge, no specific factors have been identified that correlate
with internal expansion of these lesions.
The treatment of choice is en bloc resection with 5 to 10 mm wide bone margin for
the primary goals of limiting painful progression, improving cosmesis, and avoiding
potential complications such as hemorrhage.[1]
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[25]
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[35] Utilization of preoperative embolization can be considered to aid surgical resection
by decreasing intraoperative blood loss.[7]
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[20]
[25]
Following surgical resection, the remaining cranial defect must be reconstructed which
is particularly challenging with large lesions and those involving the forehead. We
propose a staged approach as an option to optimize postoperative cosmesis, particularly
for lesions involving the forehead. Several preoperative and technical nuances were
helpful in this case. The first is preoperative angiography, which while unable to
embolize the main anterior cerebral artery feeder, identified the branch of the STA
that supplied substantial blood flow to the lesion. Given that the authors typically
spare the STA if at all possible, this led to a more efficient surgical case when
it was sacrificed during lesion exposure. Second, the use of a diamond drill bit to
decrease intraoperative blood loss. Given the intrinsically hemorrhagic nature of
these lesions, use of a diamond bur provided hemostatic capacity while allowing for
debulking of the lesion.
Conclusion
The patient did very well postoperatively with a good cosmetic and neurologic outcome.
An MRI obtained at 6 months postoperatively demonstrated no evidence of tumor recurrence.
While rare, intraosseous cavernous hemangiomas can involve the inner table and have
intradural expansion making an en bloc resection a more challenging surgical treatment
strategy. In these cases, removal in a piecemeal fashion should be considered to minimize
the risk of injury to the underlying brain parenchyma and vascular structures. Proper
reconstruction is critical for achieving a good cosmetic outcome, and a staged reconstruction
with a custom-made implant can be considered for large lesions and those involving
the forehead.
