The Journal of Craniofacial Surgery
Brief Clinical Studies
26. Park EM, Park YM, Gwak YS. Oxidative damage in tissues of rats exposed to cigarette smoke. Free Radic Biol Med 1998;25:79Y86 27. Belch JJ, McArdle BM, Burns P, et al. The effects of acute smoking on platelet behaviour, fibrinolysis and haemorheology in habitual smokers. Thromb Haemost 1984;51:6Y8 28. Kimoto S, Cheng SL, Zhang SF, et al. The effect of glucocorticoid on the synthesis of biglycan and decorin in human osteoblasts and bone marrow stromal cells. Endocrinology 1994;135:2423Y2431 29. Nordahl J, Mengarelli-Widholm S, Hultenby K, et al. Ultrastructural immunolocalization of fibronectin in epiphyseal and metaphyseal bone of young rats. Calcif Tissue Int 1995;57:442Y449 30. Weiss RE, Reddi AH. Synthesis and localization of fibronectin during collagenous matrix-mesenchymal cell interaction and differentiation of cartilage and bone in vivo. Proc Natl Acad Sci USA 1980;77:2074Y2078 31. Murakami J, Nishida T, Otori T. Coordinated appearance of beta 1 integrins and fibronectin during corneal wound healing. J Lab Clin Med 1992;120:86Y93 32. Pitaru S, Aubin JE, Bhargava U, et al. Immunoelectron microscopic studies on the distributions of fibronectin and actin in a cellular dense connective tissue: the periodontal ligament of the rat. J Periodontal Res 1987;22:64Y74 33. Kurihara S, Enlow DH. An electron microscopic study of attachments between periodontal fibers and bone during alveolar remodeling. Am J Orthod 1980;77:516Y531 34. Johnson RB. A classification of Sharpey’s fibers within the alveolar bone of the mouse: a high-voltage electron microscope study. Anat Rec 1987;217:339Y347 35. Magro-Ernica N, Magro-Filho O, Rangel-Garcia I. Histologic study of use of microfibrillar collagen hemostat in rat dental sockets. Braz Dent J 2003;14:12Y15 36. Devlin H. Early bone healing events following rat molar tooth extraction. Cells Tissues Organs 2000;167:33Y37 37. Kurita K, Hashimoto Y, Takei T, et al. Changes in collagen types during the healing of rabbit tooth extraction wounds. J Dent Res 1985;64:28Y32
Technical Strategies for En Bloc Resection and Immediate Reconstruction in Hemangioma of the Frontal Bone
& Volume 25, Number 4, July 2014
calvaria is often needed to ensure a free margin, but it will leave a bony defect that requires reconstruction. Although curettage and covering of the lesion with alloplastic material are a simple treatment option for hemangioma of the skull, it does not always prevent recurrence. Hence, complete resection is needed. As our technical strategies for reconstruction, we organize a split calvarial bone graft if a defect is near the frontal sinus and calcium phosphate cement if it is somewhat far from the sinus. Key Words: Hemangioma of the skull, radical resection, reconstruction, split calvarial bone graft, calcium phosphate cement
H
emangiomas of bone are rare lesions comprising only 0.7% of all osseous tumors and 10% of primary benign tumors of the skull.1 Hemangioma of the skull is commonly encountered as a solitary lesion.2,3 Although very rarely,4 there exists the type of lesion that is often mistaken for a hamartomatous malformation or endothelial tumor.5 It originates in the diploe of the skull, grows gradually, and, if undetected sooner, invades the inner as well as the outer table. It is a benign tumor, but growth persists and may destroy the inner core by invading deep into the cranium.6,7 This type of lesion is often noticed as a bony bulk in the head and is detected first as a punched-out lesion on plain radiograph.2,3,5 Although its definitive diagnosis must be made histopathologically, a physician needs to diagnose it differentially to distinguish this particular type from other similar lesions, as a misdiagnosis may lead to malpractice. Typical findings on plain radiograph and computed tomography (CT) are usually reliable. Although this is a benign lesion, recurrence must be avoided. Once a lesion is diagnosed as this type, radical resection and immediate reconstruction are recommended.3 Although curettage and covering of the lesion with alloplastic material are a simple option for hemangioma of the skull, this combination is not usually sufficient to prevent recurrence.7 The only option to ensure the avoidance of recurrence is complete resection.
Tetsuji Uemura, MD,* Kiwako Sawai, MD,* Mamoru Kikuchi, MD,* Jun Masuoka, MD,Þ Toshio Matsushima, MDÞ Abstract: Hemangioma of the skull is a benign solitary tumor, often found in the frontal or parietal area. A hemangioma lesion typically involves the outer table rather than the inner, but its complete removal at the diploe level is difficult. Full-thickness resection at the From the *Departments of Plastic and Reconstructive Surgery and †Neurosurgery, Saga University, Saga, Japan. Received April 17, 2013. Accepted for publication April 29, 2013. Address correspondence and reprint requests to Tetsuji Uemura, MD, Department of Plastic and Reconstructive Surgery, Faculty of Medicine, Saga University, 5-1-1 Nabeshima, Saga City, Saga 849-8501, Japan; E-mail: [email protected] The authors report no conflicts of interest. Copyright * 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0b013e31829ad605
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FIGURE 1. Patient 1.A, Preoperative frontal view showing bony bulk in the right side of the forehead. B, Sunburst appearance seen on plain radiograph (white circle). C, Computed tomographic view showing clear honeycomb shadow. D, Three-dimensional CT view shows the honeycomb mass. E, Gross findings showing fleshy tumor bulk in the right side of the forehead. F, Gross inspection showing hemangioma with a cavernous vascular lesion. G, Hemangioma of the skull shown by microscopy. H, Outer table of split calvarial bone as applied to bony defect at resected site on the forehead. I, Three-dimensional CT view showing considerable stability of the reconstructed site fixed by absorbable plate (at 2 postoperative months). J, Lower view showing flat surface of the reconstructed forehead site (at 2 postoperative months).
* 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery
& Volume 25, Number 4, July 2014
We describe its treatment: en bloc resection and immediate reconstruction in 2 consecutive Japanese patients for technical strategies.
PATIENT 1 A 34-year-old woman presented with a tumorous lesion in the right side of the forehead (Fig. 1A). The tumor, 1.5 2.0 cm, grew gradually during a period of about 2 years. Plain radiograph showed a honeycomb appearance (Fig. 1B); CT showed a honeycomb shadow, suggesting an intraosseous hemangioma of the skull (Fig. 1C), and three-dimensional CT view shows a honeycomb mass (Fig. 1D). The tumor, made up of fleshy bulk in the frontal bone (Fig. 1E), was resected en bloc with an intact area of 3 mm from its margin (Fig. 1F). Histopathologic examination showed a typical hemangioma of the skull (Fig. 1G); the resection-created defect was reconstructed with the outer table of split calvarial bone harvested from the same-side frontoparietal area measuring 3 3 cm and fixed by absorbable plate (Fig. 1H). The harvested outer table was applied to the bony defect at the resected site on the forehead (Fig. 1I). Two months have passed with no problems (Fig. 1J).
PATIENT 2 A 51-year-old woman presented with an asymptomatic hard tumor in the right side of the forehead. The tumor, 2.0 2.5 cm, grew gradually during a period of about 1 year. Plain radiograph showed a punched-out lesion in the right side of the forehead (Fig. 2A). Computed tomography clearly showed a honeycomb shadow in an intraosseous lesion in the frontal bone (Fig. 2B), and threedimensional CT view shows a honeycomb mass (Fig. 2C). The inner table of the skull was intact, and a hemangioma of the skull was diagnosed. The tumor was resected en bloc. The bony defect was reconstructed immediately using calcium phosphate cement (Fig. 2D). Histopathologic examination showed a typical hemangioma of the skull (Fig. 2E). The postoperative course was uneventful. Four years have passed with no problems. No features suggestive of recurrence have been noted in the meantime.
DISCUSSION The incidence of hemangiomas is about 0.5% to 1% of all benign skeletal tumors. It is usually found in the vertebral column.3 However, hemangioma of the skull is seen in about 10% of bony
FIGURE 2. Patient 2. A, Preoperative plain radiograph showing punched-out lesion. B, Computed tomographic view showing typical honeycomb shadow: clear indication of hemangioma of the skull. C, Three-dimensional CT view shows the honeycomb mass. D, Three-dimensional CT view shows calcium phosphate cement for bony defect (at 4 postoperative years). E, Hemangioma of the skull shown by microscopy.
Brief Clinical Studies
tumors that originate in the skull.5 Women are generally more affected in the ratio from 2:1 to 4:1.2,5 The peak incidence in terms of age is around the third to fifth decades.2 The lesion is often located in the frontal, parietal, or temporal bone as a solitary lesion. It is less common in the occipital, sphenoidal, or petrous bone. It usually grows slowly and invades the outer table, showing frontal or parietal bulk in the affected site. Sometimes the lesion grows inward and destroys the inner table of the skull, exposing itself intracranially.6,7 A hemangioma is histologically cavernous and capillary. Microscopically, there are multiple engorged vascular elements interspersed within thin-walled dilated spaces in the abundant trabecular bone.3 Patients usually complain of a painless bulk of the skull, and headache may at times accompany the complaint.4 Our 2 cases had no symptoms of headache, nor were there neurological findings. Only the bulk in the affected site was noticed. Radiographic findings are crucial in making the diagnosis.3,8,12 A punched-out lesion will be seen on a plain view. A honeycomb shadow is also sometimes seen in a plain view, but the sunburst appearance and honeycomb shadow are typically detected in a tangential plain view. If CTunequivocally shows the honeycomb appearance, and the inner table of the skull is invaded, then it necessitates resection. Curettage followed by covering of the lesion with alloplastic material is perhaps an optimal mode of treatment for hemangioma of the skull. But it is not usually sufficient to prevent recurrence.7 The tumor is benign, and it requires en bloc radical resection to ensure no recurrence. Also, as the tumor is located in the skull, a second operation should somehow be avoided. As complete resection is feasible, intracranial surgery is acceptable. In case a bony defect is created, reconstruction is carried out with autogenous bone, including calvarial bone.8 Alloplastic materials such as calcium phosphate cement and methylmethacrylate are alternatives.9 Some surgeons are reluctant to use those materials because they are foreign to the human system and have a risk of causing infection. As our technical strategies of materials for reconstruction, we organize a split calvarial bone graft if a defect is near the frontal sinus, and calcium phosphate cement if it is somewhat far from the sinus. In our 2 cases, the postoperative course was uneventful, and no revision in the morphologic contour was needed. No recurrence was suspected in either case with a follow-up period of 2 months in the first case and of 4 years in the second.
REFERENCES 1. Wyke BD. Primary hemangioma of the skull: a rare cranial tumor. Am J Roentgenol Radium Ther Nucl Med 1945;61:302Y314 2. Clauser L, Mandrioli S, Polito J, et al. Surgical techniques for the removal of forehead hemangioma. J Craniofac Surg 2006;17:702Y704 3. Satoh K, Ohtsuka T, Abe T, et al. Haemangioma of the forehead: radiographic diagnosis and definitive surgical treatment in a Japanese series. Scand J Plast Reconstr Surg Hand Surg 2009;43:75Y81 4. Peterson DL, Murk SE, Story JL. Multifocal cavernous hemangioma of the skull: report of a case and review of the literature. Neurosurgery 1992;30:778Y782 5. Bastug D, Ortiz O, Schochet SS. Hemangiomas in the calvaria: imaging findings. AJR Am J Roentgenol 1995;164:683Y687 6. Hoffmann DF, Israel J. Intraosseous frontal hemangioma. Head Neck 1990;12:160Y163 7. Hook SR, Font RL, McCrary JA, et al. Intraosseous capillary hemangioma of the frontal bone. Am J Ophthalmol 1987;103:824Y827 8. Tessier P. Autogenous bone grafts taken from the calvarium for facial and cranial applications. Clin Plast Surg 1982;9:531Y540 9. Gosain AK, Persing JA. Biomaterials in the face: benefits and risks. J Craniofac Surg 1999;10:404Y414
* 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
e403
Brief Clinical Studies
26. Park EM, Park YM, Gwak YS. Oxidative damage in tissues of rats exposed to cigarette smoke. Free Radic Biol Med 1998;25:79Y86 27. Belch JJ, McArdle BM, Burns P, et al. The effects of acute smoking on platelet behaviour, fibrinolysis and haemorheology in habitual smokers. Thromb Haemost 1984;51:6Y8 28. Kimoto S, Cheng SL, Zhang SF, et al. The effect of glucocorticoid on the synthesis of biglycan and decorin in human osteoblasts and bone marrow stromal cells. Endocrinology 1994;135:2423Y2431 29. Nordahl J, Mengarelli-Widholm S, Hultenby K, et al. Ultrastructural immunolocalization of fibronectin in epiphyseal and metaphyseal bone of young rats. Calcif Tissue Int 1995;57:442Y449 30. Weiss RE, Reddi AH. Synthesis and localization of fibronectin during collagenous matrix-mesenchymal cell interaction and differentiation of cartilage and bone in vivo. Proc Natl Acad Sci USA 1980;77:2074Y2078 31. Murakami J, Nishida T, Otori T. Coordinated appearance of beta 1 integrins and fibronectin during corneal wound healing. J Lab Clin Med 1992;120:86Y93 32. Pitaru S, Aubin JE, Bhargava U, et al. Immunoelectron microscopic studies on the distributions of fibronectin and actin in a cellular dense connective tissue: the periodontal ligament of the rat. J Periodontal Res 1987;22:64Y74 33. Kurihara S, Enlow DH. An electron microscopic study of attachments between periodontal fibers and bone during alveolar remodeling. Am J Orthod 1980;77:516Y531 34. Johnson RB. A classification of Sharpey’s fibers within the alveolar bone of the mouse: a high-voltage electron microscope study. Anat Rec 1987;217:339Y347 35. Magro-Ernica N, Magro-Filho O, Rangel-Garcia I. Histologic study of use of microfibrillar collagen hemostat in rat dental sockets. Braz Dent J 2003;14:12Y15 36. Devlin H. Early bone healing events following rat molar tooth extraction. Cells Tissues Organs 2000;167:33Y37 37. Kurita K, Hashimoto Y, Takei T, et al. Changes in collagen types during the healing of rabbit tooth extraction wounds. J Dent Res 1985;64:28Y32
Technical Strategies for En Bloc Resection and Immediate Reconstruction in Hemangioma of the Frontal Bone
& Volume 25, Number 4, July 2014
calvaria is often needed to ensure a free margin, but it will leave a bony defect that requires reconstruction. Although curettage and covering of the lesion with alloplastic material are a simple treatment option for hemangioma of the skull, it does not always prevent recurrence. Hence, complete resection is needed. As our technical strategies for reconstruction, we organize a split calvarial bone graft if a defect is near the frontal sinus and calcium phosphate cement if it is somewhat far from the sinus. Key Words: Hemangioma of the skull, radical resection, reconstruction, split calvarial bone graft, calcium phosphate cement
H
emangiomas of bone are rare lesions comprising only 0.7% of all osseous tumors and 10% of primary benign tumors of the skull.1 Hemangioma of the skull is commonly encountered as a solitary lesion.2,3 Although very rarely,4 there exists the type of lesion that is often mistaken for a hamartomatous malformation or endothelial tumor.5 It originates in the diploe of the skull, grows gradually, and, if undetected sooner, invades the inner as well as the outer table. It is a benign tumor, but growth persists and may destroy the inner core by invading deep into the cranium.6,7 This type of lesion is often noticed as a bony bulk in the head and is detected first as a punched-out lesion on plain radiograph.2,3,5 Although its definitive diagnosis must be made histopathologically, a physician needs to diagnose it differentially to distinguish this particular type from other similar lesions, as a misdiagnosis may lead to malpractice. Typical findings on plain radiograph and computed tomography (CT) are usually reliable. Although this is a benign lesion, recurrence must be avoided. Once a lesion is diagnosed as this type, radical resection and immediate reconstruction are recommended.3 Although curettage and covering of the lesion with alloplastic material are a simple option for hemangioma of the skull, this combination is not usually sufficient to prevent recurrence.7 The only option to ensure the avoidance of recurrence is complete resection.
Tetsuji Uemura, MD,* Kiwako Sawai, MD,* Mamoru Kikuchi, MD,* Jun Masuoka, MD,Þ Toshio Matsushima, MDÞ Abstract: Hemangioma of the skull is a benign solitary tumor, often found in the frontal or parietal area. A hemangioma lesion typically involves the outer table rather than the inner, but its complete removal at the diploe level is difficult. Full-thickness resection at the From the *Departments of Plastic and Reconstructive Surgery and †Neurosurgery, Saga University, Saga, Japan. Received April 17, 2013. Accepted for publication April 29, 2013. Address correspondence and reprint requests to Tetsuji Uemura, MD, Department of Plastic and Reconstructive Surgery, Faculty of Medicine, Saga University, 5-1-1 Nabeshima, Saga City, Saga 849-8501, Japan; E-mail: [email protected] The authors report no conflicts of interest. Copyright * 2014 by Mutaz B. Habal, MD ISSN: 1049-2275 DOI: 10.1097/SCS.0b013e31829ad605
e402
FIGURE 1. Patient 1.A, Preoperative frontal view showing bony bulk in the right side of the forehead. B, Sunburst appearance seen on plain radiograph (white circle). C, Computed tomographic view showing clear honeycomb shadow. D, Three-dimensional CT view shows the honeycomb mass. E, Gross findings showing fleshy tumor bulk in the right side of the forehead. F, Gross inspection showing hemangioma with a cavernous vascular lesion. G, Hemangioma of the skull shown by microscopy. H, Outer table of split calvarial bone as applied to bony defect at resected site on the forehead. I, Three-dimensional CT view showing considerable stability of the reconstructed site fixed by absorbable plate (at 2 postoperative months). J, Lower view showing flat surface of the reconstructed forehead site (at 2 postoperative months).
* 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
The Journal of Craniofacial Surgery
& Volume 25, Number 4, July 2014
We describe its treatment: en bloc resection and immediate reconstruction in 2 consecutive Japanese patients for technical strategies.
PATIENT 1 A 34-year-old woman presented with a tumorous lesion in the right side of the forehead (Fig. 1A). The tumor, 1.5 2.0 cm, grew gradually during a period of about 2 years. Plain radiograph showed a honeycomb appearance (Fig. 1B); CT showed a honeycomb shadow, suggesting an intraosseous hemangioma of the skull (Fig. 1C), and three-dimensional CT view shows a honeycomb mass (Fig. 1D). The tumor, made up of fleshy bulk in the frontal bone (Fig. 1E), was resected en bloc with an intact area of 3 mm from its margin (Fig. 1F). Histopathologic examination showed a typical hemangioma of the skull (Fig. 1G); the resection-created defect was reconstructed with the outer table of split calvarial bone harvested from the same-side frontoparietal area measuring 3 3 cm and fixed by absorbable plate (Fig. 1H). The harvested outer table was applied to the bony defect at the resected site on the forehead (Fig. 1I). Two months have passed with no problems (Fig. 1J).
PATIENT 2 A 51-year-old woman presented with an asymptomatic hard tumor in the right side of the forehead. The tumor, 2.0 2.5 cm, grew gradually during a period of about 1 year. Plain radiograph showed a punched-out lesion in the right side of the forehead (Fig. 2A). Computed tomography clearly showed a honeycomb shadow in an intraosseous lesion in the frontal bone (Fig. 2B), and threedimensional CT view shows a honeycomb mass (Fig. 2C). The inner table of the skull was intact, and a hemangioma of the skull was diagnosed. The tumor was resected en bloc. The bony defect was reconstructed immediately using calcium phosphate cement (Fig. 2D). Histopathologic examination showed a typical hemangioma of the skull (Fig. 2E). The postoperative course was uneventful. Four years have passed with no problems. No features suggestive of recurrence have been noted in the meantime.
DISCUSSION The incidence of hemangiomas is about 0.5% to 1% of all benign skeletal tumors. It is usually found in the vertebral column.3 However, hemangioma of the skull is seen in about 10% of bony
FIGURE 2. Patient 2. A, Preoperative plain radiograph showing punched-out lesion. B, Computed tomographic view showing typical honeycomb shadow: clear indication of hemangioma of the skull. C, Three-dimensional CT view shows the honeycomb mass. D, Three-dimensional CT view shows calcium phosphate cement for bony defect (at 4 postoperative years). E, Hemangioma of the skull shown by microscopy.
Brief Clinical Studies
tumors that originate in the skull.5 Women are generally more affected in the ratio from 2:1 to 4:1.2,5 The peak incidence in terms of age is around the third to fifth decades.2 The lesion is often located in the frontal, parietal, or temporal bone as a solitary lesion. It is less common in the occipital, sphenoidal, or petrous bone. It usually grows slowly and invades the outer table, showing frontal or parietal bulk in the affected site. Sometimes the lesion grows inward and destroys the inner table of the skull, exposing itself intracranially.6,7 A hemangioma is histologically cavernous and capillary. Microscopically, there are multiple engorged vascular elements interspersed within thin-walled dilated spaces in the abundant trabecular bone.3 Patients usually complain of a painless bulk of the skull, and headache may at times accompany the complaint.4 Our 2 cases had no symptoms of headache, nor were there neurological findings. Only the bulk in the affected site was noticed. Radiographic findings are crucial in making the diagnosis.3,8,12 A punched-out lesion will be seen on a plain view. A honeycomb shadow is also sometimes seen in a plain view, but the sunburst appearance and honeycomb shadow are typically detected in a tangential plain view. If CTunequivocally shows the honeycomb appearance, and the inner table of the skull is invaded, then it necessitates resection. Curettage followed by covering of the lesion with alloplastic material is perhaps an optimal mode of treatment for hemangioma of the skull. But it is not usually sufficient to prevent recurrence.7 The tumor is benign, and it requires en bloc radical resection to ensure no recurrence. Also, as the tumor is located in the skull, a second operation should somehow be avoided. As complete resection is feasible, intracranial surgery is acceptable. In case a bony defect is created, reconstruction is carried out with autogenous bone, including calvarial bone.8 Alloplastic materials such as calcium phosphate cement and methylmethacrylate are alternatives.9 Some surgeons are reluctant to use those materials because they are foreign to the human system and have a risk of causing infection. As our technical strategies of materials for reconstruction, we organize a split calvarial bone graft if a defect is near the frontal sinus, and calcium phosphate cement if it is somewhat far from the sinus. In our 2 cases, the postoperative course was uneventful, and no revision in the morphologic contour was needed. No recurrence was suspected in either case with a follow-up period of 2 months in the first case and of 4 years in the second.
REFERENCES 1. Wyke BD. Primary hemangioma of the skull: a rare cranial tumor. Am J Roentgenol Radium Ther Nucl Med 1945;61:302Y314 2. Clauser L, Mandrioli S, Polito J, et al. Surgical techniques for the removal of forehead hemangioma. J Craniofac Surg 2006;17:702Y704 3. Satoh K, Ohtsuka T, Abe T, et al. Haemangioma of the forehead: radiographic diagnosis and definitive surgical treatment in a Japanese series. Scand J Plast Reconstr Surg Hand Surg 2009;43:75Y81 4. Peterson DL, Murk SE, Story JL. Multifocal cavernous hemangioma of the skull: report of a case and review of the literature. Neurosurgery 1992;30:778Y782 5. Bastug D, Ortiz O, Schochet SS. Hemangiomas in the calvaria: imaging findings. AJR Am J Roentgenol 1995;164:683Y687 6. Hoffmann DF, Israel J. Intraosseous frontal hemangioma. Head Neck 1990;12:160Y163 7. Hook SR, Font RL, McCrary JA, et al. Intraosseous capillary hemangioma of the frontal bone. Am J Ophthalmol 1987;103:824Y827 8. Tessier P. Autogenous bone grafts taken from the calvarium for facial and cranial applications. Clin Plast Surg 1982;9:531Y540 9. Gosain AK, Persing JA. Biomaterials in the face: benefits and risks. J Craniofac Surg 1999;10:404Y414
* 2014 Mutaz B. Habal, MD
Copyright © 2014 Mutaz B. Habal, MD. Unauthorized reproduction of this article is prohibited.
e403
