CASE REPORT
Multicentric Calvarial Hemangioma with Dural Invasion: A Case Report
2026 年 13 巻 p. 265-269
詳細
2026 年 13 巻 p. 265-269
Calvarial hemangiomas are rare benign vascular tumors originating from the skull bones, most commonly observed in the frontal and parietal bones of the cranium. Although most cases are asymptomatic, they can present with headache, cosmetic deformity, or, rarely, neurological deficit. The lesions are usually solitary; multicentric involvement and dural invasion are exceedingly rare phenomena. Here, we report a 22-year-old male patient who presented with frontal headache and a palpable scalp swelling. Imaging revealed two separate expansile lytic lesions in the right and left frontal bones with no connection between them. Surgical resection was planned. Intraoperatively, the lesion on the left side demonstrated findings highly suggestive of dural invasion, and both lesions were completely resected. This case highlights the rarity of the combination of multicentric involvement with dural invasion in the literature.
Hemangiomas are benign vascular lesions most often seen in the skin, soft tissues, and vertebral bodies. They constitute a rare subset of primary bone tumors, accounting for roughly 0.2% of all bone tumors, with calvarial involvement being an even rarer subcategory.1-5)
Clinically, patients may present with headache, a palpable mass, or cosmetic deformity. Radiologically, calvarial hemangiomas appear as expansile lytic lesions; on plain radiographs and computed tomography (CT), the characteristic "sunburst" or "wagon-wheel" appearance can support the diagnosis, although this typical pattern is not observed in every case.5,6) Surgical treatment is recommended for lesions that are symptomatic, show growth, cause cosmetic concerns, or pose diagnostic uncertainty. En bloc resection with appropriate reconstruction (cranioplasty) is the cornerstone of treatment, and in selected cases, preoperative embolization may be utilized to reduce intraoperative bleeding risk.1,7,8)
Calvarial hemangiomas are most often solitary. Multicentric occurrence and especially dural invasion are extremely rare; therefore, cases in which these two features are observed together carry a distinct importance in the literature.9-12)
A 22-year-old male presented with a frontal-region headache and a palpable swelling on the forehead. On physical examination, a firm, immobile mass approximately 2 × 1 cm in size was palpated in the left frontal region. Neurological examination was normal.
Cranial CT revealed expansile, lytic lesions involving both frontal bones in a multicentric distribution (Fig. 1). On magnetic resonance imaging (MRI), the lesions appeared hypointense on T1-weighted sequences and hyperintense on T2-weighted sequences, with homogeneous enhancement after contrast administration (Fig. 2).
(a) Preoperative non-contrast CT; (b) Postoperative non-contrast CT.
CT: computed tomography
(a) Preoperative contrast-enhanced MRI; (b) Postoperative contrast-enhanced MRI.
MRI: magnetic resonance imaging
Through a bifrontal skin incision, a single bone flap was elevated, and both lesions were resected en bloc (Fig. 3). Intraoperatively, no discernible dissection plane was identified between the left-sided lesion and the underlying dura mater. Therefore, the involved dural segment beneath the left frontal lesion was resected en bloc together with the lesion, followed by duraplasty using a galeal graft. This was followed by cranioplasty with polymethyl methacrylate. The postoperative course was uneventful, and at the 3-year follow-up, there was no evidence of recurrence or residual lesion.
Intraoperative macroscopic view of the excised lesion and bone fragments.
Calvarial hemangiomas are rare benign vascular bone tumors and have been reported more frequently in women. They can occur in the vertebrae, long bones, and skull.3,4) Within the cranium, they most commonly involve the frontal and parietal bones; occipital and temporal bone involvement is less frequent.1,2)
Diagnosis is usually made incidentally or after the patient develops symptoms such as headache, a palpable mass, or cosmetic deformity. In rare cases, signs of increased intracranial pressure or neurological deficits may arise.1,7)
In the present case, the patient was male and had a lesion in the frontal region, as is commonly reported, but uniquely, it was bilaterally located. The presenting complaint of a palpable lesion with associated cosmetic deformity was likewise consistent with the literature. No signs of elevated intracranial pressure or neurological deficit were present.
Radiologically, calvarial hemangiomas appear as expansile lytic skull lesions. A "sunburst" or "wagon-wheel" appearance on plain X-ray or CT is characteristic and can aid diagnosis, but a typical pattern may not be seen in every case.5,6) Therefore, the differential diagnosis should include lesions such as meningioma, aneurysmal bone cyst, osteoblastoma, eosinophilic granuloma, and metastases.6,13) In the present case, a sunburst pattern was absent, whereas internal radiating trabecular patterns compatible with a wagon-wheel appearance were observed on plain skull radiography and axial imaging (Figures S1 and S2). The wagon-wheel pattern, although not universally present, is considered a characteristic radiological feature of calvarial hemangiomas.
Calvarial hemangiomas are usually intradiploic in location and grow by expanding the outer table; involvement of the inner table and dura mater is exceedingly rare. Although histopathological confirmation of dural invasion could not be obtained, the intraoperative absence of a cleavage plane between the lesion and the dura mater, together with the necessity for en bloc dural resection and subsequent duraplasty, strongly suggested true dural invasion rather than simple dural adhesion. Only a limited number of cases with dural invasion have been documented in the literature. For example, Ryu et al.9) reported a case of a skull cavernous hemangioma with dural penetration, and Nasi et al.10) described an aggressive case demonstrating intradural invasion.
In recent years, additional reports have emphasized that intradural involvement in large frontal bone hemangiomas can impact surgical strategy and reconstruction planning. Bird et al.11) described a staged reconstruction approach for a massive frontal bone hemangioma with intradural involvement, and Akhter et al.12) reported dural invasion in an intraosseous cavernous hemangioma of the fronto-orbital region. Previously reported cases of calvarial hemangioma with and without dural involvement are summarized in Table 1.
Comparison of Reported Calvarial Hemangioma Cases in the Literature
| Author, year | Age / gender | Bone location | Number of lesions | Dural invasion | Treatment | Follow-up | Recurrence |
|---|---|---|---|---|---|---|---|
| F: female; M: male | |||||||
| Khanam et al. (2), 2001 | — | Frontal | Solitary | None | Surgical excision | — | — |
| Mohindra et al. (8), 2016 | — | Occipital (Torcula) | Solitary | None | Total resection | — | — |
| Nasi et al. (10), 2016 | — | Frontal(Giant) | Solitary | Present(Intradural) | Total resection | — | — |
| Ryu et al. (9), 2018 | 56 / M | Frontal | Solitary | Present | Total resection | 18 months | None |
| Alkan et al. (13), 2018 | — | Frontal | Solitary | None | Surgical excision | — | — |
| Akhter et al. (12), 2019 | 68 / F | Fronto-orbital | Solitary | Present | GTR + Reconstruction | — | — |
| Bird et al. (11), 2022 | — | Frontal(Massive) | Solitary | Present(Intradural) | Resection + Reconstruction | — | — |
| Krishnan et al. (1), 2024 | Series | Frontal / Parietal | Solitary | None | Total resection | 12-36 months | None |
| Present case | 22 / M | Bilateral frontal | Multicentric | Present(Intraoperative) | Total resection | 36 months | None |
Calvarial hemangiomas are typically solitary, and multicentric involvement is much rarer. In the present case, bilateral frontal bone lesions with a multicentric distribution were observed, along with intraoperative findings suggestive of dural invasion. This combination clearly indicates that calvarial hemangiomas cannot always be considered innocuous, localized lesions, and that in multicentric cases, the possibility of dura mater involvement must be taken into account during surgical planning.9-12)
The gold-standard treatment is en bloc resection of the lesion with a margin of healthy bone, followed by reconstruction of the defect via cranioplasty. Since recurrence has been reported after subtotal resection, achieving adequate surgical margins is important. In hypervascular lesions, preoperative embolization may be performed in select cases to reduce the risk of intraoperative hemorrhage.1,8)
In the present case, both lesions were totally excised in a single session using one bone flap, as their locations allowed repair with a single graft from the same approach. Duraplasty with a galeal graft was performed at the site of dural invasion, and the craniectomy defect was reconstructed with polymethyl methacrylate. No recurrence was detected during the follow-up period. This outcome supports that long-term success can be achieved with appropriate surgical technique and reconstruction.8) Table 1 has been reorganized in chronological order according to year of publication.
Although calvarial hemangiomas are rare benign lesions, they can exhibit multicentric involvement and dural invasion. The present case is noteworthy for featuring multicentric calvarial lesions with dural invasion identified intraoperatively based on surgical findings. Evaluating possible dura mater involvement during surgical planning and ensuring en bloc resection with proper reconstruction may improve long-term outcomes.9-12)
Conception or design of the work: Berkay Ayhan, Edip Rencüzoğulları, Mehmet Emre Yıldırım, Data collection: Berkay Ayhan, Edip Rencüzoğulları, Data analysis and interpretation: Mehmet Emre Yıldırım, Edip Rencüzoğulları, Drafting the article: Edip Rencüzoğulları, Critical revision of the article: Berkay Ayhan, Mehmet Emre Yıldırım, Patient management, surgical planning and execution: Berkay Ayhan, Edip Rencüzoğulları, Title Page Other (study supervision, fundings, materials, etc): Berkay Ayhan, Edip Rencüzoğulları, Mehmet Emre Yıldırım, All authors (Berkay Ayhan, Edip Rencüzoğulları, Mehmet Emre Yıldırım) reviewed the results and approved the final version of the manuscript.
All authors have no conflict of interest.
No institutional ethics approval was required for this case report.
Obtained
ChatGPT (OpenAI) was used to assist in refining the language and clarity of the manuscript. All content was reviewed and edited by the authors, who take full responsibility for the accuracy and integrity of the final text.
