Journal of Neuro-Oncology 13: 5%61, 1992. © 1992KluwerAcademic Publishers. Printedin the Netherlands. Clinical Study
Primary intraosseous meningioma: case report Hiroshi Ito, Hiroshi Takagi, Nobuyuki Kawano 1 and Kenzoh Yada 1 From the Department of Neurosurgery, Yamato City Hospital, Yamato city, Kanagawa, Japan; 1Department of Neurosurgery, Kitasato University School of Medicine, Sagamihara city, Kanagawa, Japan
Key words: meningioma, ectopic meningioma, skull neoplasm, histogenesis, differential diagnosis Summary A case of 72-year-old Japanese woman with a rare intraosseous meningioma is presented. The tumor was located in the right frontoparietal region, involving the coronal suture. The tumor was excised totally and the pathological diagnosis was meningioma. Similar cases reported in the past literature are reviewed and the possible histogenetic mechanism of the tumor is discussed.
Introduction Generally, meningiomas originate and grow on the inside of the dural envelope. Meningiomas rarely originate within the skull, which they are called primary intraosseous meningioma [2, 10, 17, 19], or calvarial meningioma [14--16, 21, 24]. We report here one of such cases. Preoperative differential diagnostic problems and the possible histogenetic mechanism of intraosseous meningioma are discussed.
Case report A 72-year-old Japanese woman visited our hospital in March, 1987, complaining of a subcutaneous mass in the right frontoparietal area. She first noticed a hard lump with slight tenderness five years ago, and the mass had increased its size in the last few years.
Examination She showed no physical and neurological abnormalities, except for a subcutaneous hard mass
(10 x 7 x 2 cm) in her right fronto-parietal region. The mass had an unclear margin and irregular surface, and was continuous with the calvarium. The overlying skin showed no abnormalities. Results of laboratory examinations were all within normal range.
Radiological examination Plain skull X-ray showed a osteolytic lesion of the skull (10 x 7 cm) with a relatively obscure margin. It was located in the right fronto-parietal region involving the coronal suture (Fig. 1). X-ray tomogram revealed that the mass was located between the inner and outer tables of the skull. Scintigrams using Tc-99 diphosphonate and Ga disclosed a high accumulation in the lesion. The right common carotid angiogram demonstrated that the small branches of the superficial temporal artery were the feeders of the mass. Computed tomography (CT) showed a distinctive intraosseous mass which markedly expanded the inner table of the skull. The mass was enhanced strongly by the contrast medium. No mass was recognized in the subdural space (Fig. 2). Considering of the possibility of metastatic bone tumor,
58
Fig. 1. Plain lateral X-ray showing a osteolytic lesion in the fronto-parietal area, involving the coronal suture.
Fig. 2. Axial computed tomographic (CT) scan showing a mass between the inner and outer tables of the skull. There is no tumor u n d e r n e a t h the dura mater. right: Bone window CT scan demonstrating the bony lesion clearly. The thinned inner table is showed (arrow).
59
Fig. 3. Lightphotomicrographof the tumor showinga typicalmeningotheliomatousmeningiomawithwhorl formations.No malignant features are present. H&E, X200.
whole body scintigram was performed, which showed no abnormal accumulations.
indicated that the tumor had been removed completely. Electron microscopic findings of the tumor confirmed the tumor as meningioma.
Operation Discussion
The scalp and subcutaneous tissues were normal. The outer table of the skull was thinned and elevated, and the tumor partly infiltrated the overlying periosteum. The tumor was resected totally, including the surrounding normal bone. The inner surface of the removed bone was smooth and intact. The dura mater was easily separated from the bone and its outer surface showed no infiltration of the tumor.
Pathology Histological studies showed that the tumor was a typical benign meningotheliomatous meningioma with whorl formations (Fig. 3). The absence of tumor tissue at the margin of the resected bone
When the skull is occupied with meningioma in the absence of recognizable subdural tumor by CT, a possibility of meningioma en plaque may be raised [20]. However, the operative findings of the present case indicated its primary intraosseous origin. Ectopic meningioma is a well recognized entity, and it has been reported to occur in various sites such as subcutaneous regions [1, 11], paranasal sinuses [3, 6] and orbits [4, 9]. The histogenetic origin of these tumors is speculated to be ectopic embryonal arachnoid cell rests [6, 11, 22], especially those located in the midline, or cells normally distributed around cranial nerves and sensory organs (orbital, aural, nasal, buccal) [7, 11]. Primary intraosseous meningioma occurring within the calvarium seems to offer different histo-
6O Table 1. Reported cases of intraosseous meningioma
Authors
Year
Age
Sex
Location Plain X-ray
New GB 1~ Pendergrass E TM Wagman A zs
1947 1953 1960
Siegel G 21 Waga S24 Azar-Kia 2
1966 1966 1970 1974
McWhorter JM 12 Ito M 8 Hamada H 5 Palma L 16 Rahoria SK19 Pearl GS 17 Ohaegbulam S ~4 Konishi y10 Oka K 1~
1976 1977 1977 1977 1978 1979 1979 1983 1989
our case
1990
40 23 43 69 33 53 40 50 50 58 60 57 42 75 71 18 30 44 31 57 79 73 72
F M F F F M M F F F F F M M F M F F M M F F F
M-F L-F R-FP L-T L-F M-F R-F R-T R-Pte R-P R-F R-FP L-T R-FP L-P L-F R-FT R-F R-FP L-FP M-P M-P R-FP
T u r n e r O A 23
Hyperostosis Hyperostosis Hyperostosis Hyperostosls Hyperostosls Hyperostosls Hyperostosls Hyperostosxs Hyperostosls Hyperostosls Hyperostosis Hyperostosis Osteolysis Osteolysis Osteolysis Osteolysis Hyperostosis Osteolysis Osteolysis Hyperostosis Osteolysis Osteolysis Osteolysis
Involved suture
Histology
Frontonasal Coronal
Bregma Squamous Squamous Sagittal Coronal Coronal Squamous Sagittal Coronal Coronal Bregma Coronal Coronal sagittal sagittal Coronal
meningotheliomatous meningotheliomatous meningotheliomatous meningotheliomatous fibroblastic meningotheliomatous meningotheliomatous
psammomatous meningotheliomatous transitional fibroblastic meningotheliomatous meningotheliomatous fibloblastic psammomatous transitional transitional meningotheliomatous
Abbreviation (location): R-right, L-left, M-midline, F-frontal, P-parietal, Pte-pteryon, T-temporal.
genetic possibilities, as many of the tumors show preferential sites of the occurrence. Out of 23 cases including the present case, 18 cases were well described referring to the location of the tumors. In these 18 cases, all the tumors involved the cranial sutures (Table 1), a fact which has been noted by some authors [2, 21]. There is no answer as yet to the question on why intraosseous calvarial meningiomas preferentially occur at the cranial sutures. The assumption provided by Azar-Kia et al. [2] is interesting, that is a part of the dura containing arachnoid cells may have been trapped in the suture lines during molding of the skull at birth and later develops into meningioma. Alternatively, Turner et al. [23] reported that trauma may play a role in the development of some primary intraosseous meningiomas. As there is no history of head injury in our case, the former may well explain our case. Neuroradiologically, it seems difficult to diagnose intraosseous meningioma preoperatively.
However, although rare, meningioma must be included for differential diagnosis when the lesion involved the cranial suture as discussed before.
References 1. Altinors N, Celtin A, Pak I: Scalp meningioma. Neurosurgery 16: 379-380, 1985 2. Azar-Kia B, Sarwar M, Alan Marc J, Schechter MM: Intraosseous Meningioma, Neuroradiology 6: 246-253, 1974 3. Batsakis JG: Extracranial meningiomas. Ann Otol Rhinol Laryngol 93: 282-283, 1984 4. Farr HW, Gray Jr GF, Vrana M, Panio M: Extracranial meningioma. J Surg Oncol 5: 411-420, 1973 5. Hamada H, Kadota H, Uetsuhara K, Asakura T, Igakura T: A case of extracalvarial meningioma. No Shlnkei Geka 5: 633-639, 1977 6. Ho KL: Primary meningioma of the nasal cavity and paranasal sinuses. Cancer 46: 1442-1447, 1980 7. Hoye SJ, Hoar Jr CS, Murray JE: Extracranial meningioma presenting as a tumor of the neck. Am J Surg 10: 486-489, 1960
61 8. Ito M, Tomizawa H, Takagi E, Ishi S: Extracalvarial meningioma. No Shinkei Geka 5: 465-471, 1977 (Japanese) 9. Kobayashi S, Kyoshima K, Nakagawa F, Sugita K, Maruyama Y: Diploic meningioma. Surg Neurol 13: 277-281, 1980 10. Konishi Y, Kadowaki C, Shiogai T, Ogashiwa M, Takeuchi K, Fukuzumi N: A case of Osteoid osteoma with Intraosseous meningioma. No Shinkei Geka 12: 1403-1408, 1984 (Japanese) 11. Lopez DA, Silvers DN, Helwig EB: Cutaneous meningiomas. Cancer 34: 728-744, 1974 12. McWhorter JM, Ghatak NR, Kelly Jr DL: Extracranial meningioma presenting as lytic skull lesion. Surg Neurol 5: 223-224, 1976 13. New GB, Devine KD: Neurogenic tumors of nose and throat. Arch Otolaryngol 46: 163-179, 1947 14. Ohaegbulam SC: Ectopic epidural calvarial meningioma. Surg Neurol 12: 33-35, 1979 15. Oka K, Hirakawa K, Yoshida S, Tomonaga M: Primary calvarial meningiomas. Surg Neurol 32: 304-310, 1989 16. Palma L, Mercuri S, Ferrante L: Ectopic calvarial meningioma. Surg Neurol 8: 315-318, 1977 17. Pearl GS, Takei Y, Parent AD, Boehm Jr WM: Primary intraosseous meningioma presenting as a solitary osteolytic skull lesion: Case report. Neurosurgery 4: 269-270, 1979 18. Pendergrass EP, Hope JW: An extracranial meningioma
19. 20.
21. 22. 23. 24.
25.
with no apparent intracranial source: Report of a case. Am J Roentgenol 70: 967-970, 1953 Rahoria SK, Gulati DR: Intraosseous meningioma: A case report. Neurology (India) 26: 79-80, 1978 Russel DS, Rubinstein LF: Pathology of turnouts of the nervous system. Baltimore, Williams & Wilkins (ed) 5, pp 461, 1989 Siegel GJ, Anderson PJ: Extracalvarial meningioma. J Neurosurg 25: 83-86, 1966 Suzuki H, Gilbert EF, Zimmerman B: Primary extracranial meningioma. Arch Pathol 84: 202-206, 1967 Turner OA, Laird AT: Meningioma with traumatic etiology. Neurosurgery 24, 96-98, 1966 Waga S, Nishikawa M, Ohtsubo K, Kamijyo Y, Handa H: Extracalvarial meningiomas: 2 cases. Neurology 20: 368372, 1970 Wagman AD, Weiss EK, Riggs HE: Hyperplasia of the skull associated with intra-osseous meningioma in the absence of gross tumor: Report of three cases. J Neuropathol Exp Neurol 19: 111-115, 1960
Address for offprints: H. Ito, Department of Neurosurgery, Kitasato University School of Medicine, 1-15-1 Kitasato, Sagamihara, Kanagawa 228, Japan
Primary intraosseous meningioma: case report Hiroshi Ito, Hiroshi Takagi, Nobuyuki Kawano 1 and Kenzoh Yada 1 From the Department of Neurosurgery, Yamato City Hospital, Yamato city, Kanagawa, Japan; 1Department of Neurosurgery, Kitasato University School of Medicine, Sagamihara city, Kanagawa, Japan
Key words: meningioma, ectopic meningioma, skull neoplasm, histogenesis, differential diagnosis Summary A case of 72-year-old Japanese woman with a rare intraosseous meningioma is presented. The tumor was located in the right frontoparietal region, involving the coronal suture. The tumor was excised totally and the pathological diagnosis was meningioma. Similar cases reported in the past literature are reviewed and the possible histogenetic mechanism of the tumor is discussed.
Introduction Generally, meningiomas originate and grow on the inside of the dural envelope. Meningiomas rarely originate within the skull, which they are called primary intraosseous meningioma [2, 10, 17, 19], or calvarial meningioma [14--16, 21, 24]. We report here one of such cases. Preoperative differential diagnostic problems and the possible histogenetic mechanism of intraosseous meningioma are discussed.
Case report A 72-year-old Japanese woman visited our hospital in March, 1987, complaining of a subcutaneous mass in the right frontoparietal area. She first noticed a hard lump with slight tenderness five years ago, and the mass had increased its size in the last few years.
Examination She showed no physical and neurological abnormalities, except for a subcutaneous hard mass
(10 x 7 x 2 cm) in her right fronto-parietal region. The mass had an unclear margin and irregular surface, and was continuous with the calvarium. The overlying skin showed no abnormalities. Results of laboratory examinations were all within normal range.
Radiological examination Plain skull X-ray showed a osteolytic lesion of the skull (10 x 7 cm) with a relatively obscure margin. It was located in the right fronto-parietal region involving the coronal suture (Fig. 1). X-ray tomogram revealed that the mass was located between the inner and outer tables of the skull. Scintigrams using Tc-99 diphosphonate and Ga disclosed a high accumulation in the lesion. The right common carotid angiogram demonstrated that the small branches of the superficial temporal artery were the feeders of the mass. Computed tomography (CT) showed a distinctive intraosseous mass which markedly expanded the inner table of the skull. The mass was enhanced strongly by the contrast medium. No mass was recognized in the subdural space (Fig. 2). Considering of the possibility of metastatic bone tumor,
58
Fig. 1. Plain lateral X-ray showing a osteolytic lesion in the fronto-parietal area, involving the coronal suture.
Fig. 2. Axial computed tomographic (CT) scan showing a mass between the inner and outer tables of the skull. There is no tumor u n d e r n e a t h the dura mater. right: Bone window CT scan demonstrating the bony lesion clearly. The thinned inner table is showed (arrow).
59
Fig. 3. Lightphotomicrographof the tumor showinga typicalmeningotheliomatousmeningiomawithwhorl formations.No malignant features are present. H&E, X200.
whole body scintigram was performed, which showed no abnormal accumulations.
indicated that the tumor had been removed completely. Electron microscopic findings of the tumor confirmed the tumor as meningioma.
Operation Discussion
The scalp and subcutaneous tissues were normal. The outer table of the skull was thinned and elevated, and the tumor partly infiltrated the overlying periosteum. The tumor was resected totally, including the surrounding normal bone. The inner surface of the removed bone was smooth and intact. The dura mater was easily separated from the bone and its outer surface showed no infiltration of the tumor.
Pathology Histological studies showed that the tumor was a typical benign meningotheliomatous meningioma with whorl formations (Fig. 3). The absence of tumor tissue at the margin of the resected bone
When the skull is occupied with meningioma in the absence of recognizable subdural tumor by CT, a possibility of meningioma en plaque may be raised [20]. However, the operative findings of the present case indicated its primary intraosseous origin. Ectopic meningioma is a well recognized entity, and it has been reported to occur in various sites such as subcutaneous regions [1, 11], paranasal sinuses [3, 6] and orbits [4, 9]. The histogenetic origin of these tumors is speculated to be ectopic embryonal arachnoid cell rests [6, 11, 22], especially those located in the midline, or cells normally distributed around cranial nerves and sensory organs (orbital, aural, nasal, buccal) [7, 11]. Primary intraosseous meningioma occurring within the calvarium seems to offer different histo-
6O Table 1. Reported cases of intraosseous meningioma
Authors
Year
Age
Sex
Location Plain X-ray
New GB 1~ Pendergrass E TM Wagman A zs
1947 1953 1960
Siegel G 21 Waga S24 Azar-Kia 2
1966 1966 1970 1974
McWhorter JM 12 Ito M 8 Hamada H 5 Palma L 16 Rahoria SK19 Pearl GS 17 Ohaegbulam S ~4 Konishi y10 Oka K 1~
1976 1977 1977 1977 1978 1979 1979 1983 1989
our case
1990
40 23 43 69 33 53 40 50 50 58 60 57 42 75 71 18 30 44 31 57 79 73 72
F M F F F M M F F F F F M M F M F F M M F F F
M-F L-F R-FP L-T L-F M-F R-F R-T R-Pte R-P R-F R-FP L-T R-FP L-P L-F R-FT R-F R-FP L-FP M-P M-P R-FP
T u r n e r O A 23
Hyperostosis Hyperostosis Hyperostosis Hyperostosls Hyperostosls Hyperostosls Hyperostosls Hyperostosxs Hyperostosls Hyperostosls Hyperostosis Hyperostosis Osteolysis Osteolysis Osteolysis Osteolysis Hyperostosis Osteolysis Osteolysis Hyperostosis Osteolysis Osteolysis Osteolysis
Involved suture
Histology
Frontonasal Coronal
Bregma Squamous Squamous Sagittal Coronal Coronal Squamous Sagittal Coronal Coronal Bregma Coronal Coronal sagittal sagittal Coronal
meningotheliomatous meningotheliomatous meningotheliomatous meningotheliomatous fibroblastic meningotheliomatous meningotheliomatous
psammomatous meningotheliomatous transitional fibroblastic meningotheliomatous meningotheliomatous fibloblastic psammomatous transitional transitional meningotheliomatous
Abbreviation (location): R-right, L-left, M-midline, F-frontal, P-parietal, Pte-pteryon, T-temporal.
genetic possibilities, as many of the tumors show preferential sites of the occurrence. Out of 23 cases including the present case, 18 cases were well described referring to the location of the tumors. In these 18 cases, all the tumors involved the cranial sutures (Table 1), a fact which has been noted by some authors [2, 21]. There is no answer as yet to the question on why intraosseous calvarial meningiomas preferentially occur at the cranial sutures. The assumption provided by Azar-Kia et al. [2] is interesting, that is a part of the dura containing arachnoid cells may have been trapped in the suture lines during molding of the skull at birth and later develops into meningioma. Alternatively, Turner et al. [23] reported that trauma may play a role in the development of some primary intraosseous meningiomas. As there is no history of head injury in our case, the former may well explain our case. Neuroradiologically, it seems difficult to diagnose intraosseous meningioma preoperatively.
However, although rare, meningioma must be included for differential diagnosis when the lesion involved the cranial suture as discussed before.
References 1. Altinors N, Celtin A, Pak I: Scalp meningioma. Neurosurgery 16: 379-380, 1985 2. Azar-Kia B, Sarwar M, Alan Marc J, Schechter MM: Intraosseous Meningioma, Neuroradiology 6: 246-253, 1974 3. Batsakis JG: Extracranial meningiomas. Ann Otol Rhinol Laryngol 93: 282-283, 1984 4. Farr HW, Gray Jr GF, Vrana M, Panio M: Extracranial meningioma. J Surg Oncol 5: 411-420, 1973 5. Hamada H, Kadota H, Uetsuhara K, Asakura T, Igakura T: A case of extracalvarial meningioma. No Shlnkei Geka 5: 633-639, 1977 6. Ho KL: Primary meningioma of the nasal cavity and paranasal sinuses. Cancer 46: 1442-1447, 1980 7. Hoye SJ, Hoar Jr CS, Murray JE: Extracranial meningioma presenting as a tumor of the neck. Am J Surg 10: 486-489, 1960
61 8. Ito M, Tomizawa H, Takagi E, Ishi S: Extracalvarial meningioma. No Shinkei Geka 5: 465-471, 1977 (Japanese) 9. Kobayashi S, Kyoshima K, Nakagawa F, Sugita K, Maruyama Y: Diploic meningioma. Surg Neurol 13: 277-281, 1980 10. Konishi Y, Kadowaki C, Shiogai T, Ogashiwa M, Takeuchi K, Fukuzumi N: A case of Osteoid osteoma with Intraosseous meningioma. No Shinkei Geka 12: 1403-1408, 1984 (Japanese) 11. Lopez DA, Silvers DN, Helwig EB: Cutaneous meningiomas. Cancer 34: 728-744, 1974 12. McWhorter JM, Ghatak NR, Kelly Jr DL: Extracranial meningioma presenting as lytic skull lesion. Surg Neurol 5: 223-224, 1976 13. New GB, Devine KD: Neurogenic tumors of nose and throat. Arch Otolaryngol 46: 163-179, 1947 14. Ohaegbulam SC: Ectopic epidural calvarial meningioma. Surg Neurol 12: 33-35, 1979 15. Oka K, Hirakawa K, Yoshida S, Tomonaga M: Primary calvarial meningiomas. Surg Neurol 32: 304-310, 1989 16. Palma L, Mercuri S, Ferrante L: Ectopic calvarial meningioma. Surg Neurol 8: 315-318, 1977 17. Pearl GS, Takei Y, Parent AD, Boehm Jr WM: Primary intraosseous meningioma presenting as a solitary osteolytic skull lesion: Case report. Neurosurgery 4: 269-270, 1979 18. Pendergrass EP, Hope JW: An extracranial meningioma
19. 20.
21. 22. 23. 24.
25.
with no apparent intracranial source: Report of a case. Am J Roentgenol 70: 967-970, 1953 Rahoria SK, Gulati DR: Intraosseous meningioma: A case report. Neurology (India) 26: 79-80, 1978 Russel DS, Rubinstein LF: Pathology of turnouts of the nervous system. Baltimore, Williams & Wilkins (ed) 5, pp 461, 1989 Siegel GJ, Anderson PJ: Extracalvarial meningioma. J Neurosurg 25: 83-86, 1966 Suzuki H, Gilbert EF, Zimmerman B: Primary extracranial meningioma. Arch Pathol 84: 202-206, 1967 Turner OA, Laird AT: Meningioma with traumatic etiology. Neurosurgery 24, 96-98, 1966 Waga S, Nishikawa M, Ohtsubo K, Kamijyo Y, Handa H: Extracalvarial meningiomas: 2 cases. Neurology 20: 368372, 1970 Wagman AD, Weiss EK, Riggs HE: Hyperplasia of the skull associated with intra-osseous meningioma in the absence of gross tumor: Report of three cases. J Neuropathol Exp Neurol 19: 111-115, 1960
Address for offprints: H. Ito, Department of Neurosurgery, Kitasato University School of Medicine, 1-15-1 Kitasato, Sagamihara, Kanagawa 228, Japan
