Acta Neurochir (2014) 156:2379–2381 DOI 10.1007/s00701-014-2232-4
LETTER TO THE EDITOR - BRAIN TUMORS
Primary intraosseous meningioma on a calvarium from Byzantine Greece Anastasia Sofia Protopapa & Nikolaos Vlachadis & Emmanouel Agapitos & Theodoros Pitsios
Received: 20 August 2014 / Accepted: 4 September 2014 / Published online: 20 September 2014 # Springer-Verlag Wien 2014
Dear Editor, We present a case of primary intraosseous meningioma on a calvarium recovered in Rhodes, Greece, during excavations in the Byzantine elite cemetery, dated to 12th to 14th centuries AD and assigned to a male individual with estimated biological age at death between 20 and 30 years. The skull was examined by macroscopy, endoscopy, under stereoscopic magnification ×10 and was subjected to conventional radiography and computed tomography (CT) imaging analysis. Widespread hyperostosis was determined to affect the right lateral cranial aspect, involving the frontal, sphenoid, temporal, and parietal bones, altering the normal cranial contour and obliterating surface features including the upper and lower temporal lines, inferior coronal, and sphenofrontal sutures. Surface irregularities along the coronal suture include striking lump formations situated superior to the pterion (Fig. 1a). CT scans with bone window settings revealed marked, widespread diploic expansion on the right-hand side of the skull and a singular intradiploic lesion of inhomogeneous hyperostosis encompassing the articulating borders of the frontal and parietal bones (Fig. 1b). Within the right frontal bone, hyper-dense tissue displaying intraosseous spiculation directly involves the outer table and coronal suture and is associated with localized lytic skull changes in the surrounding diploë as well as with an apparently disrupted inner table (Fig. 1c). Additionally, extensive endocranial surface
A. S. Protopapa : N. Vlachadis (*) : T. Pitsios Museum of Anthropology, National and Kapodistrian University of Athens, School of Medicine, 75 Mikras Asias St, 11527 Athens, Greece e-mail: [email protected] E. Agapitos First Department of Pathology, National and Kapodistrian University of Athens, School of Medicine, Athens, Greece
irregularities in the affected area were determined through endoscopic and CT scan examination. Meningiomas represent 15–20 % of all primary intracranial tumors, however occasionally arise in extracranial sites, particularly in the head and neck. Primary intraosseous meningiomas originate within bones, most commonly in association with the craniofacial skeleton and constitute the rarest manifestation among primary extracranial meningiomas [1–3, 5, 9]. Ectopic arachnoid or multipotent mesenchymal cells within bone are considered to provide the origins of these tumors [2, 3, 8, 9]. Most calvarial primary intraosseous meningiomas present as osteoblastic intradiploic areas, whereas mixed and osteolytic variants are less common [2, 3, 8, 9]. Osteoblastic variants typically induce hyperostosis and bone remodeling particularly at the site of tumor origin, while endocranial surface irregularities observed in the region comprising the lesion constitute a key feature in differential diagnosis of intraosseous meningioma against other hyperostosing conditions [2–4, 8, 9]. Association with cranial sutures is common [2, 9]. Primary intraosseous meningiomas exhibit a bimodal age distribution with incidence peaking in the second and in the sixth to seventh decades of life and a female:male sex ratio of approximately 1:1 [7]. Differential diagnosis of primary intraosseous meningiomas includes secondary intraosseous meningiomas, which constitute direct extracranial extensions of a primary intracranial tumor, exhibiting high frequencies compared to their primary counterparts [2, 9]. We regard to the direct, specific association between the lesion and the coronal suture, the presence of localized, intradiploic lytic changes and an apparently fractured “from within” inner table (Fig. 1b) as evidence against the probability of secondary spread from an intracranial primary site [2, 8]. Meningioma en plaque, a morphological variant of intracranial meningiomas developing in a configuration of thin
2380
Acta Neurochir (2014) 156:2379–2381
Fig.
1 a, b, c Byzantine calvarium: Macroscopic view shows widespread hyperostosis on the right lateral aspect of the skull, obliterating normal surface features. Surface irregularities and hyperostotic lumps are prominent along the coronal suture (a). CT axial section on bone window settings discloses marked widening of the diploë and singular inhomogeneous hyperostotic area, specifically encompassing the coronal suture as well as evidence of intradiploic bony destruction (b), as well as an intradiploic lesion comprising hyperdense tissue forming inwardly oriented spicules. Hyperostotic tissue is not distinguished from the outer table. There is evidence of bony destruction in the peripheral diploic tissue as well as a distinctive, apparently disrupted inner table. The involvement of the coronal suture is direct and specific and the altered ectocranial and endocranial contours are disclosed as well (c)
hyperostosis [6]. Colonization by meningioma en plaque provides a diagnosis compatible with our observations in the skull under study, appearing however less probable due to incidence peak in the later decades of life and strong female predilection observed in this type of meningioma [6, 8]. Despite imposed limitations in examining dry bones, exquisite detail in bone alterations constitutes an advantage of the material. The skull bone changes described above are considered indicative of a long-term response to the presumed underlying meningioma, reflecting primarily osteoblastic but also osteolytic processes induced by the intraosseous tumor. Of note, grooves of the meningeal vessels extending across the hyperostotic bone correspond to the left-hand-side pattern of vascular impressions and maintain adequate detail, providing potential evidence of a close relationship between the dura and the endocranial surface, suggesting the absence of an extradural mass. In conclusion, our calvarium displays CT imaging features compatible with intraosseous meningioma, including hyperostosis, endocranial irregularity, and a mixed lesion comprising hyper-dense and hypo-dense components within the right frontal bone, directly involving the coronal suture. Localization of bone alteration and remodeling characteristics as well as the biological age determination suggest the primary origin of the tumor. To our knowledge, the presented Byzantine skull from Rhodes, Greece, is the oldest case of primary intraosseous meningioma documented by CT. Conflicts of interest None.
References
layers along meningeal planes may infiltrate the overlying dura and invade cranial bones, generating extensive skull
1. Ammirati M, Mirzai S, Samii M (1990) Primary intraosseous meningiomas of the skull base. Acta Neurochir 107:56–60 2. Arana E, Diaz C, Latorre FF, Menor F, Revert A, Beltrán A, Navarro M (1996) Primary intraosseous meningiomas. Acta Radiol 37:937– 942 3. Azar-Kia B, Sarwar M, Marc JA, Schechter MM (1974) Intraosseous meningioma. Neuroradiology 6:246–253
Acta Neurochir (2014) 156:2379–2381 4. Daffner RH, Yakulis R, Maroon JC (1998) Intraosseous meningioma. Skeletal Radiol 27:108–111 5. Geoffray A, Lee YY, Jing BS, Wallace S (1984) Extracranial meningiomas of the head and neck. AJNR Am J Neuroradiol 5: 599–604 6. Kim KS, Rogers LF, Goldblatt D (1987) CT features of hyperostosing meningioma en plaque. AJR Am J Roentgenol 149:1017–1023 7. Lang FF, Macdonald OK, Fuller GN, DeMonte F (2000) Primary extradural meningiomas: a report on nine cases and review of the
2381 literature from the era of computerized tomography scanning. J Neurosurg 93:940–950 8. McGuire TP, Palme CE, Perez-Ordonez B, Gilbert RW, Sándor GK (2007) Primary intraosseous meningioma of the calvaria: analysis of the literature and case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 104:e34–e41 9. Tokgoz N, Oner YA, Kaymaz M, Ucar M, Yilmaz G, Tali TE (2005) Primary intraosseous meningioma: CT and MRI appearance. AJNR Am J Neuroradiol 26:2053–2056
LETTER TO THE EDITOR - BRAIN TUMORS
Primary intraosseous meningioma on a calvarium from Byzantine Greece Anastasia Sofia Protopapa & Nikolaos Vlachadis & Emmanouel Agapitos & Theodoros Pitsios
Received: 20 August 2014 / Accepted: 4 September 2014 / Published online: 20 September 2014 # Springer-Verlag Wien 2014
Dear Editor, We present a case of primary intraosseous meningioma on a calvarium recovered in Rhodes, Greece, during excavations in the Byzantine elite cemetery, dated to 12th to 14th centuries AD and assigned to a male individual with estimated biological age at death between 20 and 30 years. The skull was examined by macroscopy, endoscopy, under stereoscopic magnification ×10 and was subjected to conventional radiography and computed tomography (CT) imaging analysis. Widespread hyperostosis was determined to affect the right lateral cranial aspect, involving the frontal, sphenoid, temporal, and parietal bones, altering the normal cranial contour and obliterating surface features including the upper and lower temporal lines, inferior coronal, and sphenofrontal sutures. Surface irregularities along the coronal suture include striking lump formations situated superior to the pterion (Fig. 1a). CT scans with bone window settings revealed marked, widespread diploic expansion on the right-hand side of the skull and a singular intradiploic lesion of inhomogeneous hyperostosis encompassing the articulating borders of the frontal and parietal bones (Fig. 1b). Within the right frontal bone, hyper-dense tissue displaying intraosseous spiculation directly involves the outer table and coronal suture and is associated with localized lytic skull changes in the surrounding diploë as well as with an apparently disrupted inner table (Fig. 1c). Additionally, extensive endocranial surface
A. S. Protopapa : N. Vlachadis (*) : T. Pitsios Museum of Anthropology, National and Kapodistrian University of Athens, School of Medicine, 75 Mikras Asias St, 11527 Athens, Greece e-mail: [email protected] E. Agapitos First Department of Pathology, National and Kapodistrian University of Athens, School of Medicine, Athens, Greece
irregularities in the affected area were determined through endoscopic and CT scan examination. Meningiomas represent 15–20 % of all primary intracranial tumors, however occasionally arise in extracranial sites, particularly in the head and neck. Primary intraosseous meningiomas originate within bones, most commonly in association with the craniofacial skeleton and constitute the rarest manifestation among primary extracranial meningiomas [1–3, 5, 9]. Ectopic arachnoid or multipotent mesenchymal cells within bone are considered to provide the origins of these tumors [2, 3, 8, 9]. Most calvarial primary intraosseous meningiomas present as osteoblastic intradiploic areas, whereas mixed and osteolytic variants are less common [2, 3, 8, 9]. Osteoblastic variants typically induce hyperostosis and bone remodeling particularly at the site of tumor origin, while endocranial surface irregularities observed in the region comprising the lesion constitute a key feature in differential diagnosis of intraosseous meningioma against other hyperostosing conditions [2–4, 8, 9]. Association with cranial sutures is common [2, 9]. Primary intraosseous meningiomas exhibit a bimodal age distribution with incidence peaking in the second and in the sixth to seventh decades of life and a female:male sex ratio of approximately 1:1 [7]. Differential diagnosis of primary intraosseous meningiomas includes secondary intraosseous meningiomas, which constitute direct extracranial extensions of a primary intracranial tumor, exhibiting high frequencies compared to their primary counterparts [2, 9]. We regard to the direct, specific association between the lesion and the coronal suture, the presence of localized, intradiploic lytic changes and an apparently fractured “from within” inner table (Fig. 1b) as evidence against the probability of secondary spread from an intracranial primary site [2, 8]. Meningioma en plaque, a morphological variant of intracranial meningiomas developing in a configuration of thin
2380
Acta Neurochir (2014) 156:2379–2381
Fig.
1 a, b, c Byzantine calvarium: Macroscopic view shows widespread hyperostosis on the right lateral aspect of the skull, obliterating normal surface features. Surface irregularities and hyperostotic lumps are prominent along the coronal suture (a). CT axial section on bone window settings discloses marked widening of the diploë and singular inhomogeneous hyperostotic area, specifically encompassing the coronal suture as well as evidence of intradiploic bony destruction (b), as well as an intradiploic lesion comprising hyperdense tissue forming inwardly oriented spicules. Hyperostotic tissue is not distinguished from the outer table. There is evidence of bony destruction in the peripheral diploic tissue as well as a distinctive, apparently disrupted inner table. The involvement of the coronal suture is direct and specific and the altered ectocranial and endocranial contours are disclosed as well (c)
hyperostosis [6]. Colonization by meningioma en plaque provides a diagnosis compatible with our observations in the skull under study, appearing however less probable due to incidence peak in the later decades of life and strong female predilection observed in this type of meningioma [6, 8]. Despite imposed limitations in examining dry bones, exquisite detail in bone alterations constitutes an advantage of the material. The skull bone changes described above are considered indicative of a long-term response to the presumed underlying meningioma, reflecting primarily osteoblastic but also osteolytic processes induced by the intraosseous tumor. Of note, grooves of the meningeal vessels extending across the hyperostotic bone correspond to the left-hand-side pattern of vascular impressions and maintain adequate detail, providing potential evidence of a close relationship between the dura and the endocranial surface, suggesting the absence of an extradural mass. In conclusion, our calvarium displays CT imaging features compatible with intraosseous meningioma, including hyperostosis, endocranial irregularity, and a mixed lesion comprising hyper-dense and hypo-dense components within the right frontal bone, directly involving the coronal suture. Localization of bone alteration and remodeling characteristics as well as the biological age determination suggest the primary origin of the tumor. To our knowledge, the presented Byzantine skull from Rhodes, Greece, is the oldest case of primary intraosseous meningioma documented by CT. Conflicts of interest None.
References
layers along meningeal planes may infiltrate the overlying dura and invade cranial bones, generating extensive skull
1. Ammirati M, Mirzai S, Samii M (1990) Primary intraosseous meningiomas of the skull base. Acta Neurochir 107:56–60 2. Arana E, Diaz C, Latorre FF, Menor F, Revert A, Beltrán A, Navarro M (1996) Primary intraosseous meningiomas. Acta Radiol 37:937– 942 3. Azar-Kia B, Sarwar M, Marc JA, Schechter MM (1974) Intraosseous meningioma. Neuroradiology 6:246–253
Acta Neurochir (2014) 156:2379–2381 4. Daffner RH, Yakulis R, Maroon JC (1998) Intraosseous meningioma. Skeletal Radiol 27:108–111 5. Geoffray A, Lee YY, Jing BS, Wallace S (1984) Extracranial meningiomas of the head and neck. AJNR Am J Neuroradiol 5: 599–604 6. Kim KS, Rogers LF, Goldblatt D (1987) CT features of hyperostosing meningioma en plaque. AJR Am J Roentgenol 149:1017–1023 7. Lang FF, Macdonald OK, Fuller GN, DeMonte F (2000) Primary extradural meningiomas: a report on nine cases and review of the
2381 literature from the era of computerized tomography scanning. J Neurosurg 93:940–950 8. McGuire TP, Palme CE, Perez-Ordonez B, Gilbert RW, Sándor GK (2007) Primary intraosseous meningioma of the calvaria: analysis of the literature and case report. Oral Surg Oral Med Oral Pathol Oral Radiol Endod 104:e34–e41 9. Tokgoz N, Oner YA, Kaymaz M, Ucar M, Yilmaz G, Tali TE (2005) Primary intraosseous meningioma: CT and MRI appearance. AJNR Am J Neuroradiol 26:2053–2056
