Delayed Manifestation of Sacral Clear Cell Meningioma Distent Metastasis or Multifocal Disease?


Study design: The authors report on a clear cell meningioma (CCM) of the sacrum revealed 17 years after a removal of a spinal lesion. A review of 29 spinal CCM previously reported was done. Objective: To focus on clinical and biological behaviour of CCM. Summary of background data: CCM is a rare subtype characterrized by its inordinately aggressive clinical course despite its benign appearance. The tumour shows proclivity for the cerebellopontine angle and cauda equine region. Recurrence and metastasis have been commonly reported. Method: A 26 year-old man presented with low back pain related to a lytic lesion of the sacrum. He had a history of an intradural extramedullary meningioma of the cervical spine removed when he was 9 year-old. CT scan revealed a sacral osteolytic lesion with anterior and lateral extension. Open biopsy revealed a clear cell meningioma. That was the same pattern of the tumour removed 17 years before. Results: Our patient was managed conservatively by palliative radiation therapy. At 2 years follow-up, he experienced improvement of pain and walking. Conclusion: CCM is an aggressive tumour with a high risk of metastases through cerebrospinal fluid. A follow up with spinal MRI should be performed.

Share and Cite:

H. Nouri, L. Abid, M. Ouertatani, D. Hentati, M. Mestiri and H. Jaafoura, "Delayed Manifestation of Sacral Clear Cell Meningioma Distent Metastasis or Multifocal Disease?," Surgical Science, Vol. 2 No. 6, 2011, pp. 297-302. doi: 10.4236/ss.2011.26064.

Conflicts of Interest

The authors declare no conflicts of interest.


[1] D. Louis, Ohgaki, H. Wiestler, et al., “WHO Classification—Tumours of the Nervous System,” 4th Edition, International Agency for Research on Cancer, Lyon, 2007, pp. 164-172.
[2] S. Zorludemir, B. W. Scheithauer, T. Hirose, et al., “Cler Cell Meningioma: A Clinicopathologic Study of a Potentially Aggressive Variant of Meningioma,” The American Journal of Surgical Pathology, Vol. 19, No. 5, 1995, pp. 493-505. doi:10.1097/00000478-199505000-00001
[3] K. W. Ko, D. H. Nam, D. S. Kong, et al., “Relationship between Malignant Subtypes of Meningioma and Clinical Outcome,” Journal of Clinical Neuroscience, Vol. 14, No. 8, 2007, pp. 747-753. doi:10.1016/j.jocn.2006.05.005
[4] M. Prinz, S. Patt, T. Mitrovics, et al., “Clear Cell Meningioma: Report of a Spinal Case,” General & Diagnostic Pathology, Vol. 141, No. 3-4, 1996, pp. 261-267.
[5] R. N. N. Holtzman and S. C. Jorkman, “Nondural-Based Lumbar Clear Cell Meningioma,” Journal of Neurosurgery, Vol. 84, No. 2, 1996, pp. 264-266. doi:10.3171/jns.1996.84.2.0264
[6] J. Pimental, A. Fernandes, A. E. Pinto, et al., “Clear Cell Meningioma Variant and Clinical Aggressiveness,” Clinical Neuropathology, Vol. 17, No. 3, 1998, pp. 141-146.
[7] C. Cances, Y. Chaix, C. Karsenty, et al., “Clear Cell Meningioma: Recurrent Intraspinal Tumor in a Child,” Archives de Pédiatrie, Vol. 5, No. 7, 1998, pp. 758-762. doi:10.1016/S0929-693X(98)80062-6
[8] A. Dubois, A. Sevely, S. Boetto, et al., “Clear Cell Meningioma of the Cauda Equine,” Neuroradiology, Vol. 40, No. 11, 1998, pp. 743-747. doi:10.1007/s002340050676
[9] H. Matsui, M. Kanamori, Y. Abe, et al., “Multifocal Clear Cell Meningioma in the Spine: A Case Report,” Neurosurgical Review, Vol. 21, No. 2-3, 1998, pp. 171-173. doi:10.1007/BF02389326
[10] G. I. Jallo, K. F. Kothbauer, V. M. Silvera, et al., “Intraspinal Clear Cell Meningioma: Diagnosis and Management: Report of Two Cases,” Neurosurgery; Vol. 48, No. 1, 2001, pp. 218-222.
[11] K. B. Yu, M. K. Lim, H. J. Kim, et al., “Clear Cell Meningioma: CT and MR Imaging Findings in Two Cases Involving the Spinal Canal and Cerebellopontine Angle,” Korean Journal of Radiology, Vol. 3, No. 2, 2002, pp. 125-129. doi:10.3348/kjr.2002.3.2.125
[12] R. Boet, H. K. Ng, S. Kumta, et al., “Lumbosacral Clear-Cell Meningioma Treated with Subtotal Resection and Radiotherapy,” Journal of Clinical Neuroscience, Vol. 11, No. 4, 2004, pp. 432-436. doi:10.1016/j.jocn.2003.10.014
[13] P. I. Liu, G. C. Liu, K. B. Tsai, et al., “Intraspinal Clear Cell Meningioma: Case Report and Review of Literature,” Surgical Neurology, Vol. 63, No. 3, 2005, pp. 285-289. doi:10.1016/j.surneu.2004.03.013
[14] S. S. Dhall, L. M. Tumialan, D. J. Brat, et al., “Spinal Intradural Clear Cell Meningioma Following Resection of a Suprasellar Clear Cell Meningioma. Case Report and Recommendations for Management,” Journal of Neurosurgery, Vol. 103, No. 3, 2005, pp. 559-563. doi:10.3171/jns.2005.103.3.0559
[15] D. Jain, M. C. Sharma, C. Sarkar, et al., “Clear Cell Meningioma, an Uncommon Variant of Meningioma: A Clinicopathologic Study of Nine Cases,” Journal of Neuro-Oncology, Vol. 81, No. 3, 2007, pp. 315-321. doi:10.1007/s11060-006-9237-7
[16] C. B. Colen, M, Rayes, J. McClendon Jr., et al., “Pediatric Spinal Clear Cell Meningioma. Case Report,” Journal of Neurosurgery Pediatrics, Vol. 3, No. 1, 2009, pp. 57-60.
[17] T.-T. Wu, L.-J. Bian, Z. Li, Y. Li, B.-N. Luo and Q. Huang, “Clear Cell Meningioma with Anaplastic Features: Case Report and Review of Literature,” Pathology—Research and Practice, Vol. 206, No. 5, 2010, pp. 349-354. doi:10.1016/j.prp.2009.06.015
[18] T. Inoue, K. Miyamoto, H. Kodama, et al., “Cervical Clear Cell Meningioma Mimicking a Vertebral Metastasis,” Journal of Clinical Neuroscience, Vol. 12, No. 6, 2005, pp. 685-687. doi:10.1016/j.jocn.2004.08.019
[19] J. A. Heth, P. Kirby, A. H. Menezes, et al., “Intraspinal Familial Clear Cell Meningioma in a Mother and Child,” Journal of Neurosurgery, Vol. 93, No. 2, 2000, pp. 317-321.
[20] A. Oviedo, D. Pang, J. Zovikian, et al., “Clear Cell Meningioma: Case Report and Review of the Literature,” Pediatric and Developmental Pathology, Vol. 8, No. 3, 2005, pp. 386-390. doi:10.1007/s10024-005-0119-3
[21] N. Epstein, S. Drexler and J. Shneider, “Clear cell Meningioma of the Cauda Equine in an Adult. Case Report and Literature Review,” Journal of Spinal Disorders & Techniques, Vol. 18, No. 6, 2005, pp. 539-543. doi:10.1097/01.bsd.0000173314.98401.b5
[22] H. C. Park, M. J. Sohn, E. Y. Kim, et al., “Spinal Clear Cell Meningiomas Presented with Progressive Parapresis in Infancy,” Child’s Nervous System, Vol. 16, No. 9, 2000, pp. 607-610. doi:10.1007/PL00007302
[23] S. Carra, P. Drigo, M. Gadriman, et al., “Clear Cell Meningioma in a 22 Months-Old Male; a Case Report and Literature Review,” Pediatric Neurosurgery, Vol. 34, No. 5, 2001, pp. 264-267.
[24] M. H. Chen, S. J. Chen, S. M. Lin, et al., “A Lumbar Cleare Cell Meningioma with Foraminal Extension in a Renal Transplant Recipient,” Journal of Clinical Neuroscience, Vol. 11, No. 6, 2004, pp. 665-667. doi:10.1016/j.jocn.2003.10.024
[25] S. K. Ludwin and F. K. Conley, “Malignant Meningioma Metastatizing through the Cerebrospinal Pathways,” Journal of Neurology, Neurosurgery & Psychiatry, Vol. 38, No. 2, 1975, pp. 136-142. doi:10.1136/jnnp.38.2.136
[26] P. M. Son, M. Sacher, S. W. Strenger, et al., “Benign Metastasizing Meningiomas,” American Journal of Neuroradiology, Vol. 8, No. 1, 1987, pp. 127-130.
[27] A. T. kimura, T. Orita, O. Hayashida, et al., “Malignant Meningiomas Metastatizing through the Cerebrospinal Pathway,” Acta Neurologica Scandinavica, Vol. 85, No. 5, 1992, pp. 368-371. doi:10.1111/j.1600-0404.1992.tb04063.x
[28] K. Kamiya, T. Inagawa and R. Nagasako, “Malignant Intraventricular Meningioma with Spinal Metastasis through the Cerebrospinal Fluid,” Surgical Neurology, Vol. 32, No. 3, 1989, pp. 213-218. doi:10.1016/0090-3019(89)90181-X
[29] Y. Y. Lee, W. W. Hsu, T. J. Huang, et al., “Metastatic Meningioma in the Sacrum: A Case Report,” Spine, Vol. 27, No. 4, 2002, pp. E100-E103. doi:10.1097/00007632-200202150-00022
[30] W. C. Peh and Y. W. Fan, “Case Report: Intraventricular Meningioma with Cerebelopontine Angle and Drop Metastases,” The British Journal of Radiology, Vol. 68, No. 808, 1995, pp. 428-430. doi:10.1259/0007-1285-68-808-428
[31] C. G. Riley, “Metastatizing Meningeal Tumour Case Report,” The New Zealand Medical Journal, Vol. 73, 1974, pp. 210-214.
[32] F. J. Wippold II, J. G. Smimoitopoulos and T. K. Pilgram, “Lesions of the Cauda Equine: A Clinical Pathology Review from the Armed Forces Institute of Pathology,” Clinical Neurology and Neurosurgery, Vol. 9, No. 4, 1997, pp. 229-234. doi:10.1016/S0303-8467(97)00095-4
[33] J. L Karasik and S. Mullens, “A Survey of Metastatic Meningiomas,” Journal of Neurosurgery, Vol. 40, No. 2, 1974, pp. 206-212. doi:10.3171/jns.1974.40.2.0206
[34] R. Wasserkurg, E. Peyser and C. Lichtig, “Extracranial Bone Metastases from Intracranial Meningiomas,” Surgical Neurology, Vol. 12, No. 6, 1979, pp. 480-484.
[35] M. D. Conrad, C. Schonauer, I. Pelissou-Guyotat, et al., “Recurrent Lumbosacral Metastases from Intracranial Meningioma. Report of a Case and Review of the Literature,” Acta Neurochirurgica, Vol. 143, No. 9, 2001, pp. 935-937. doi:10.1007/s007010170024
[36] S. A. Enam, S. Abdulrauf, B. Mehta, et al., “Metastasis in Meningioma,” Acta Neurochirurgica, Vol. 138, No. 10, 1996, pp. 1172-1178. doi:10.1007/BF01809747
[37] M. C. Chamberlain and M. J. Glantz, “Cerebrospinal Fluid- Dessiminated Meningioma,” Cancer, Vol. 103, No. 7, 2005, pp. 1427-1430. doi:10.1002/cncr.20926
[38] T. T. Lee and H. J. Landy, “Spinal Metastases of Malignant Intracranial Meningioma,” Surgical Neurology, Vol. 50, No. 5, 1998, pp. 437-441. doi:10.1016/S0090-3019(97)00348-0
[39] P. D. Delgado-Lopez, V. Martin-Velasco, J. M. Castilla- Diez, et al., “Metastatic Meningioma to the Eleventh Dorsal Vertebal Body: Total En Bloc Spondylectomy. Case Report and Review of the Literature,” Neur- ocirurgia, Vol. 17, No. 3, 2006, pp. 240-249.
[40] S. W. Strenger, Y. P. Huang and V. P. Sachdez, “Malignant Meningioma within the Third Ventricle: A Case Report,” Neurosurgery, Vol. 20, No. 3, 1978, pp. 465-468. doi:10.1227/00006123-198703000-00019
[41] W. Lee, K. H. Chang, G. Choe, et al., “MR Imaging Features of Clear Cell Meningioma with Diffuse Lep- tomeningeal Seeding,” American Journal of Neuror- adiology, Vol. 21, No. 1, 2000, pp. 130-132.
[42] S. Fuentes, P. Metellus, C. Bouvier, et al., “Metastatic Meningioma to the First Thoracic Vertebral Body. A Case Report and Review of the Literature,” Neurochirurgie, Vol. 48, No. 1, 2002, pp. 53-56.

Copyright © 2022 by authors and Scientific Research Publishing Inc.

Creative Commons License

This work and the related PDF file are licensed under a Creative Commons Attribution 4.0 International License.