Abstract
Study design
Eighteen consecutive patients with adult-onset intradural spinal teratoma underwent surgical treatment in our center from 1998 to 2013.
Background and purpose
Teratoma is defined as a neoplasm composed of elements derived from three germ cell layers (ectoderm, endoderm and mesoderm). Intraspinal teratoma is extremely rare and accounts for 0.2–0.5% of all spinal cord tumors. Moreover, teratoma occurs primarily in neonates and young children. Adult-onset intradural spinal teratoma is even rare. The aim of this study was to discuss the clinical characteristics, diagnosis and therapeutic strategies of adult-onset intradural spinal teratoma.
Methods
This retrospective study included 18 consecutive adult patients with intradural teratoma who were surgically treated in our center between 1998 and 2013. The clinical features, pathogenesis, diagnostic strategies and surgical outcomes were discussed. Neurological function outcomes were evaluated by the JOA scoring system.
Results
Of the 18 included patients, 4 patients received subtotal resection and the other 14 patients received total resection. All the 18 cases were diagnosed with mature teratoma. The mean follow-up period was 79.7 (median 60.5; range 27–208) months. Local recurrence occurred in two of the four patients who underwent subtotal resection and in no patient who underwent total resection. The neurologic status improved in 16 cases and remained unchanged in the other two patients.
Conclusions
Adult-onset intradural spinal teratoma is extremely rare. To the best of our knowledge, this is the largest series of patients with this disease. Despite the slow-growth and indolent nature, radical resection remains the recommended treatment to reduce tumor recurrence.
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References
Sharma MC, Jain D, Sarkar C, Suri V, Garg A, Singh M, Mahapatra AK, Sharma BS (2009) Spinal teratomas: a clinico-pathological study of 27 patients. Acta Neurochir (Wien) 151:245–252. doi:10.1007/s00701-009-0212-x (discussion: 252)
Nonomura Y, Miyamoto K, Wada E, Hosoe H, Nishimoto H, Ogura H, Shimizu K (2002) Intramedullary teratoma of the spine: report of two adult cases. Spinal Cord 40:40–43
Virchow R (1863) Die Krankhaften Geschwulste, vol 1. Hirschwald, Berlin, p 514
Gowers WR, Horsley V (1888) A case of tumour of the spinal cord. Removal; Recovery. Med Chir Trans 71(377–430):11
Sloof JL, Kernohan JW, MacCarty CS (1964) Primary intramedullary tumors of the spinal cord and filum terminale. Philadelphia WB Saunders, pp 1–20
Smoker WR, Biller J, Moore SA, Beck DW, Hart MN (1986) Intradural spinal teratoma: case report and review of the literature. AJNR Am J Neuroradiol 7:905–910
Garrison JE, Kasdon DL (1980) Intramedullary spinal teratoma: case report and review of the literature. Neurosurgery 7:509–512
Allsopp G, Sgouros S, Barber P, Walsh AR (2000) Spinal teratoma: is there a place for adjuvant treatment? Two cases and a review of the literature. Br J Neurosurg 14:482–488
Poeze M, Herpers MJ, Tjandra B, Freling G, Beuls EA (1999) Intramedullary spinal teratoma presenting with urinary retention: case report and review of the literature. Neurosurgery 45:379–385
Hosoi K (1931) Intradural teratoid tumors of the spinal cord: report of a case. Arch Pathol 11:875–883
Sullivan BH (1948) Intraspinal teratoma with report of a case. Brooklyn Hosp J 6:142–145
Furtado D, Marques V (1951) Spinal teratoma. J Neuropathol Clin Neurol 10:384–393
Dereymacker A (1954) Medullar compression by congenital tumors or cysts. Acta Neurol Belg 54:874–889
Bakay L (1956) Case records of the Massachusetts General Hospital: case 42502. N Engl J Med 266:1153–1157
Teng P, Gordon J (1958) Teratoma of the conus medullaris: report of a case. J Neurosurg 15:569–571
Rewcastle NB, Francoeur J (1964) Teratomatous cysts of the spinal canal; with “sex chromatin” studies. Arch Neurol 11:91–99
Hansebout RR, Bertrand G (1965) Intraspinal teratoma simulating protruded intervertebral disc. J Neurosurg 22:374–379
Eneström S, von Essen C (1977) Spinal teratoma. Acta Neurochir (Wien) 39:121–126
Rosenbaum TJ, Soule EH, Onofrio BM (1978) Teratomatous cyst of the spinal canal. Case report. J Neurosurg 49:292–297
Padovani R, Tognetti F, Sanpaolo P, Pozzati E, Gaist G, Kuba I (1982) Intramedullary cystic teratoma. Acta Neurochir (Wien) 62:101–108
Padovani R, Tognetti F, Laudadio S, Manetto V (1983) Teratoid cyst of the spinal cord. Neurosurgery 13:74–77
Ironside JW, Jefferson AA, Royds JA, Taylor CB, Timperley WR (1984) Carcinoid tumour arising in a recurrent intradural spinal teratoma. Neuropathol Appl Neurobiol 10:479–489
Conti P, Conti R, De Luca G (1984) Observations on some rare cases of vertebro-medullar malformations associated with tumors. J Neurosurg Sci 28:81–87
Monajati A, Spitzer RM, Wiley JL, Heggeness L (1986) MR imaging of a spinal teratoma. J Comput Assist Tomogr 10:307–310
Giacomini P, Lunardi P, Liccardo G, Paris L, Sancesario G (1986) Cystic cervico-dorso-lumbar teratoma. Riv Neurol 56:1–8
Pelissou-Guyotat I, Sindou M, Pialat J, Goutelle A (1988) Intramedullary mature teratoma associated with an attached cord and an intradural lipoma. Apropos of a surgically treated case. Review of the literature. Neurochirurgie 34:205–209
Hamabuchi M, Hasegawa R, Murase T (1989) Teratoma of the spinal cord. A case report with CT scans. J Bone Joint Surg Br 71:390–392
Nicoletti GF, Passanisi M, Platania N, Lanzafame S, Albanese V (1994) Intramedullary spinal cystic teratoma of the conus medullaris with caudal exophytic development: case report. Surg Neurol 41:106–111
Caruso R, Antonelli M, Cervoni L, Salvati M (1996) Intramedullary teratoma: case report and review of the literature. Tumori 82:616–620
al-Sarraj ST, Parmar D, Dean AF, Phookun G, Bridges LR (1998) Clinicopathological study of seven cases of spinal cord teratoma: a possible germ cell origin. Histopathology 32:51–56
Koen JL, McLendon RE, George TM (1998) Intradural spinal teratoma: evidence for a dysembryogenic origin. Report of four cases. J Neurosurg 89:844–851
Bloch I, Varga Z, Benini A (1999) Teratoma of the conus medullaris. J Clin Neurosci 6:249–252
Arai Y, Takahashi M, Takeda K, Shitoto K (2000) Adult-onset intradural spinal teratoma in the lumbar spine: a case report. J Orthop Surg 8:69–74
Elmaci I, Dagcinar A, Ozgen S, Ekinci G, Pamir MN (2001) Diastematomyelia and spinal teratoma in an adult. Case report. Neurosurg Focus 10:ecp
Fan X, Turner JE, Turner TM, Elrod JP, Clough JA, Howell EI, Johnson MD (2001) Carcinoid tumor development in an intramedullary spinal cord mature teratoma. AJNR Am J Neuroradiol 22:1778–1781
Hejazi N, Witzmann A (2003) Spinal intramedullary teratoma with exophytic components: report of two cases and review of the literature. Neurosurg Rev 26:113–116
Fernandez-Cornejo VJ, Martinez-Perez M, Polo-Garcia LA, Martinez-Lage JF, Poza M (2004) Cystic mature teratoma of the filum terminale in an adult. Case report and review of the literature. Neurocirugia (Astur) 15:290–293
Ates O, Cayli SR, Koçak A, Alkan A, Onal C, Usta U (2005) Mature spinal teratoma associated with thickened filum terminale. Neurol Med Chir (Tokyo) 45:375–378
Ak H, Ulu MO, Sar M, Albayram S, Aydin S, Uzan M (2006) Adult intramedullary mature teratoma of the spinal cord: review of the literature illustrated with an unusual example. Acta Neurochir Wien 148:663–669 (discussion 669)
Paterakis KN, Karantanas AH, Barbanis S, Hadjigeorgiou GM, Karavelis A (2006) Cervical spinal cord intramedullary teratoma. Clin Neurol Neurosurg 108:514–517
Caruso R, Colonnese C (2006) Intramedullary teratomas: two case reports and a review of the literature. Zentralbl Neurochir 67:213–218
Kahilogullari G, Erdem A, Heper AO, Erden E (2006) Intramedullary mature cystic teratoma of the conus medullaris. A case report. J Neurosurg Sci 50:55–58
Stevens QE, Kattner KA, Chen YH, Rahman MA (2006) Intradural extramedullary mature cystic teratoma: not only a childhood disease. J Spinal Disord Tech 19:213–216
Tsitsopoulos P, Rizos C, Isaakidis D, Liapi G, Zymaris S (2006) Coexistence of spinal intramedullary teratoma and diastematomyelia in an adult. Spinal Cord 44:632–635
Makary R, Wolfson D, Dasilva V, Mohammadi A, Shuja S (2007) Intramedullary mature teratoma of the cervical spinal cord at C1-2 associated with occult spinal dysraphism in an adult. Case report and review of the literature. J Neurosurg Spine 6:579–584
Mut M, Shaffrey ME, Bourne TD, Jagannathan J, Shaffrey CI (2007) Unusual presentation of an adult intramedullary spinal teratoma with diplomyelia. Surg Neurol 67:190–194
Mohindra S, Chhabra R, Gupta R, Dass Radotra B (2008) Cystic, exophytic teratoma of conus medullaris presenting with chronic renal failure. Surg Neurol 69:81–83 (discussion 84)
Sung KS, Sung SK, Choi HJ, Song YJ (2008) Spinal intradural extramedullary mature cystic teratoma in an adult. J Korean Neurosurg Soc 44:334–337
Han IH, Kuh SU, Chin DK, Kim KS, ** BH, Cho YE (2008) Surgical treatment of primary spinal tumors in the conus medullaris. J Korean Neurosurg Soc 44:72–77
Oh JS, Im SB, Kim BT, Shin WH (2009) Surgical findings of a lumbar mature teratoma accompanying the preoperative intracranial dissemination of Fatty droplets. J Korean Neurosurg Soc 46:409–412
Arvin B, Pohl U, David K (2009) Intramedullary cervical teratoma in an adult. Spine J 9:e14–e18
Ghostine S, Perry E, Vaynman S, Raghavan R, Tong KA, Samudrala S, Johnson JP, Colohan A (2009) The rare case of an intramedullary cervical spinal cord teratoma in an elderly adult: case report and literature review. Spine (Phila Pa) 34:E973–E978. doi:10.1097/BRS.0b013e3181ba0fe5
Ijiri K, Hida K, Yano S, Iwasaki Y (2009) Huge intradural ossification caused by a mature spinal teratoma: case report. Neurosurgery 64:E1200–E1201. doi:10.1227/01.NEU.0000345939.06275.9A (discussion E1201)
Benes V 3rd, Barsa P, Mikulastik J, Suchomel P (2009) Exophytic intramedullary mature teratoma of the conus medullaris: case report and review of the literature. Cent Eur Neurosurg 70:154–160
Gu W, Shang H, ** X, **e J, Zhao W (2010) Intradural lumbar mature teratoma with neuronal and glial tissue component in an adult. Case report. Neurol Med Chir (Tokyo) 50:1112–1115
Jian W, Ying W, Chao Y (2010) Intramedullary spinal teratoma of the conus medullaris: report of two cases. Acta Neurochir (Wien) 152:553–554
Agrawal M, Uppin MS, Patibandla MR, Bhattacharjee S, Panigrahi MK, Saradhi V, Rani JY, Purohit AK, Challa S (2010) Teratomas in central nervous system: a clinico-morphological study with review of literature. Neurol India 58:841–846. doi:10.4103/0028-3886.73740
Moon HJ, Shin BK, Kim JH, Kim JH, Kwon TH, Chung HS, Park YK (2010) Adult cervical intramedullary teratoma: first reported immature case. J Neurosurg Spine 13:283–287. doi:10.3171/2010.3.SPINE09461
Yu J, Qu LM, Li Y, Huang H (2012) Coexistence of spinal teratoma of the conus medullaris and arteriovenous malformation in an adult: a case report. Turk Neurosurg 22:510–514. doi:10.5137/1019-5149.JTN.3961-10.1
Kalani MY, Iyer S, Coons SW, Smith KA (2012) Spinal intradural teratomas: developmental programs gone awry? Neurosurg Focus 33:E1. doi:10.3171/2012.8.FOCUS12230
Jiang H, **ao Z, Zhan X, Chen Q, He M, Lu L (2013) Unusual association of intraspinal extramedullary teratoma with congenital scoliosis in an elderly adult: case report and literature review. Eur Spine J 22(Suppl 3):S306–S310. doi:10.1007/s00586-012-2385-x
Li Y, Yang B, Song L, Yan D (2013) Mature teratoma of the spinal cord in adults: an unusual case. Oncol Lett 6:942–946
Vanguardia MK, Honeybul S, Robbins P (2014) Subtotal resection of an intradural mature teratoma in an adult presenting with difficulty initiating micturition. Surg Neurol Int 5:23. doi:10.4103/2152-7806.127759
Matsuyama Y, Sakai Y, Katayama Y, Imagama S, Ito Z, Wakao N, Yukawa Y, Ito K, Kamiya M, Kanemura T, Sato K, Ishiguro N (2009) Indirect posterior decompression with corrective fusion for ossification of the posterior longitudinal ligament of the thoracic spine: is it possible to predict the surgical results? Eur Spine J 18:943–948. doi:10.1007/s00586-009-0956-2
Park SC, Kim KJ, Wang KC, Choe G, Kim HJ (2010) Spinal epidural teratoma: review of spinal teratoma with consideration on the pathogenesis: case report. Neurosurgery 67:E1818–E1825. doi:10.1227/NEU.0b013e3181f846ca
Onrat ST, Seyman H, Konuk M (2009) Incidence of neural tube defects in Afyonkarahisar, Western Turkey. Genet Mol Res 8:154–161
Devi AR, Rao NA, Bittles AH (1987) Inbreeding and the incidence of childhood genetic disorders in Karnataka, South India. J Med Genet 24:362–365
Larbrisseau A, Renevey F, Brochu P, Decarie M, Mathieu JP (1980) Recurrent chemical meningitis due to an intraspinal cystic teratoma: case report. J Neurosurg 52:715–717
Devlin VJ, Schwartz DM (2007) Intraoperative neurophysiologic monitoring during spinal surgery. J Am Acad Orthop Surg 15:549–560
Kim SM, Kim SH, Seo DW, Lee KW (2013) Intraoperative neurophysiologic monitoring: basic principles and recent update. J Korean Med Sci 28:1261–1269. doi:10.3346/jkms.2013.28.9.1261
Mpayo LL, Liu XH, Xu M, Wang K, Wang J, Yang L (2014) Extramedullary spinal teratoma presenting with recurrent aseptic meningitis. Pediatr Neurol 50:655–657. doi:10.1016/j.pediatrneurol.2014.02.005
Bokhari I, Rehman L, Farooq G, Qureshi A (2016) Postoperative functional outcome of intradural extramedullary spinal tumors through posterior approach. J Coll Phys Surg Pak 26:117–120. doi: 02.2016/JCPSP.117120
Mehta AI, Adogwa O, Karikari IO, Thompson P, Verla T, Null UT, Friedman AH, Cheng JS, Bagley CA, Isaacs RE (2013) Anatomical location dictating major surgical complications for intradural extramedullary spinal tumors: a 10-year single-institutional experience. J Neurosurg Spine 19:701–707
Emami B, Lyman J, Brown A, Coia L, Goitein M, Munzenrider JE, Shank B, Solin LJ, Wesson M (1991) Tolerance of normal tissue to therapeutic irradiation. Int J Radiat Oncol Biol Phys 21:109–122
Schultheiss TE, Kun LE, Ang KK, Stephens LC (1995) Radiation response of the central nervous system. Int J Radiat Oncol Biol Phys 31:1093–1112
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W. Wan and C. Yang contributed equally to this work and should be considered as first authors.
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Wan, W., Yang, C., Yan, W. et al. Adult-onset intradural spinal teratoma: report of 18 consecutive cases and outcomes in a single center. Eur Spine J 26, 1917–1928 (2017). https://doi.org/10.1007/s00586-016-4939-9
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DOI: https://doi.org/10.1007/s00586-016-4939-9