Abstract
Background
Intraspinal epidermoid/dermoid cysts are very rare, benign tumors arising from pathological displacement of epidermal cells into the spinal canal. Literature data about the long-term outcome after microsurgical resection with multimodal intraoperative neurophysiological monitoring (IONM) are lacking. We analyzed one of the largest case series with special regard to intraoperative characteristics and long-term outcome after IONM-aided surgery.
Method
All 12 patients (m:f = 1.4:1) who underwent microsurgical tumor resection with multimodal IONM for intraspinal epidermoid/dermoid tumors between 1998 and 2019 in our university hospital were included. We retrospectively investigated the patients’ characteristics, imaging/surgical parameters, and postoperative long-term outcomes.
Results
Symptomatic tumor manifestation was seen during adulthood in 4 patients (median age 33.0 years) and during childhood in 8 patients (median age 4.3 years). Spinal dysraphism was the most often comorbidity (75%). The most frequent symptoms at diagnosis were spastic pareses (75%), ataxia (58%), and vegetative disorders (42%). Tumors were most often lumbosacral (L1–L5 42%, L5–S3 50%) and intradural-extramedullary (92%). For microsurgical resection, IONM with EMG, SSEPs, and TcMEPs of the limbs and pudendal nerve/anal sphincter was always applied and feasible; intraoperative corrective actions were initiated in three cases due to transient IONM deterioration. None of the patients showed a postoperative deterioration of the neurological status with a gross total resection rate of 92%. Pain situation, McCormick grade, and mJOA Score were improved at long-term follow-up (median 4.8 years).
Conclusions
IONM-aided resection of intraspinal epidermoid/dermoid tumors is feasible both in adult and pediatric cases and enables a satisfying clinical and surgical outcome.
Similar content being viewed by others
Abbreviations
- ca.:
-
Circa
- CNS:
-
Central nervous system
- DWI:
-
Diffusion-weighted imaging
- EMG:
-
Electromyography
- IONM:
-
Intraoperative neurophysiological monitoring
- mJOA:
-
Modified Japanese Orthopedic Association score
- MMC:
-
Myelomeningocele
- MRI:
-
Magnetic resonance imaging
- resp.:
-
Respectively
- SSEPs:
-
Somatosensory evoked potentials
- TcMEPs:
-
Transcranial motor evoked potentials
- WHO:
-
World Health Organization
References
Guidetti B, Gagliardi FM (1977) Epidermoid and dermoid cysts. Clinical evaluation and late surgical results. J Neurosurg 47(1):12–18
Per H, Kumandaş S, Gümüş H, Yikilmaz A, Kurtsoy A (2007) Iatrogenic epidermoid tumor. Late complication of lumbar puncture. J Child Neurol 22(3):332–336
Beechar VB, Zinn PO, Heck KA, Fuller GN, Han I, Patel AJ, Ropper AE (2018) Spinal epidermoid tumors. case report and review of the literature. Neurospine 15(2):117–122
Lunardi P, Missori P, Gagliardi FM, Fortuna A (1989) Long-term results of the surgical treatment of spinal dermoid and epidermoid tumors. Neurosurgery 25(6):860–864
Roux A, Mercier C, Larbrisseau A, Dube LJ, Dupuis C, Del Carpio R (1992) Intramedullary epidermoid cysts of the spinal cord. Case report J Neurosurg 76(3):528–533
Youmans WHR, Kliot M et al (eds) (2004) Youmans neurological surgery. Normal and abnormal embryology the spinal cord and spine, 5th edn. Philadelphia, Saunders
McDonald JV, Klump TE (1986) Intraspinal epidermoid tumors caused by lumbar puncture. Arch Neurol 43(9):936–939
Ziv ET, Gordon McComb J, Krieger MD, Skaggs DL (2004) Iatrogenic intraspinal epidermoid tumor. Two cases and a review of the literature. Spine 29(1):E15–E18
Herman JM, McLone DG, Storrs BB, Dauser RC (1993) Analysis of 153 patients with myelomeningocele or spinal lipoma reoperated upon for a tethered cord. Presentation, management and outcome. Pediatr Neurosurg 19(5):243–249
Mazzola CA, Albright AL, Sutton LN, Tuite GF, Hamilton RL, Pollack IF (2002) Dermoid inclusion cysts and early spinal cord tethering after fetal surgery for myelomeningocele. N Engl J Med 347(4):256–259
Scott RM, Wolpert SM, Bartoshesky LE, Zimbler S, Klauber GT (1986) Dermoid tumors occurring at the site of previous myelomeningocele repair. J Neurosurg 65(6):779–783
Storrs BB (1994) Are dermoid and epidermoid tumors preventable complications of myelomeningocele repair? Pediatr Neurosurg 20(2):160–162
Sachs E, Horrax G (1949) A cervical and a lumbar pilonidal sinus communicating with intraspinal dermoids; report of two cases and review of the literature. J Neurosurg 6(2):97–112
van Aalst J, Hoekstra F, Beuls EAM, Cornips EMJ, Weber JW, Sival DA, Creytens DHKV, Vles JSH (2009) Intraspinal dermoid and epidermoid tumors. Report of 18 cases and reappraisal of the literature. Pediatr Neurosurg 45(4):281–290
Liu H, Zhang J-N, Zhu T (2012) Microsurgical treatment of spinal epidermoid and dermoid cysts in the lumbosacral region. J Clin Neurosci 19(5):712–717
Ogden AT, Khandji AG, McCormick PC, Kaiser MG (2007) Intramedullary inclusion cysts of the cervicothoracic junction. Report of two cases in adults and review of the literature. J Neurosurg Spine 7(2):236–242
McCormick PC, Torres R, Post KD, Stein BM (1990) Intramedullary ependymoma of the spinal cord. J Neurosurg 72(4):523–532
Benzel EC, Lancon J, Kesterson L, Hadden T (1991) Cervical laminectomy and dentate ligament section for cervical spondylotic myelopathy. J Spinal Disord 4(3):286–295
Odom GL, Finney W, Woodhall B (1958) Cervical disk lesions. J Am Med Assoc 166(1):23–28
Siller S, Szelényi A, Herlitz L, Tonn JC, Zausinger S (2017) Spinal cord hemangioblastomas. Significance of intraoperative neurophysiological monitoring for resection and long-term outcome. J Neurosurg Spine 26(4):483–493
El-Naggar AK (2017) WHO classification of head and neck toumours, 4th edition. World Health Organization classification of tumours, 4th edn. International Agency for Research on Cancer, Lyon
Louis DN, Ohgaki H, Wiestler OD, Cavenee WK (2016) WHO classification of tumours of the central nervous system, revised, 4th edn. World Health Organization classification of tumours. International Agency for Research on Cancer, Lyon
Ghadirpour R, Nasi D, Iaccarino C, Romano A, Motti L, Sabadini R, Valzania F, Servadei F (2018) Intraoperative neurophysiological monitoring for intradural extramedullary spinal tumors. Predictive value and relevance of D-wave amplitude on surgical outcome during a 10-year experience. J Neurosurg Spine 30(2):259–267
Quinones-Hinojosa A, Gulati M, Lyon R, Gupta N, Yingling C (2002) Spinal cord mapping as an adjunct for resection of intramedullary tumors. Surgical technique with case illustrations. Neurosurgery 51(5):1199–1206 discussion 1206-7
Scibilia A, Terranova C, Rizzo V, Raffa G, Morelli A, Esposito F, Mallamace R, Buda G, Conti A, Quartarone A, Germanò A (2016) Intraoperative neurophysiological mapping and monitoring in spinal tumor surgery. Sirens or indispensable tools? Neurosurg Focus 41(2):E18
Harel R, Schleifer D, Appel S, Attia M, Cohen ZR, Knoller N (2017) Spinal intradural extramedullary tumors. The value of intraoperative neurophysiologic monitoring on surgical outcome. Neurosurg Rev 40(4):613–619
Kahraman S, Gocmen S, Alpsan Gokmen MH, Acka G, Pusat S (2019) Intraoperative neurophysiologic monitoring for lumbar intradural schwannomas. Does it affect clinical outcome? World Neurosurg 124:789–792
Morita M, Miyauchi A, Okuda S, Oda T, Aono H, Iwasaki M (2012) Intraspinal epidermoid tumor of the cauda equina region. Seven cases and a review of the literature. J Spinal Disord Tech 25(5):292–298
Acknowledgments
We are grateful to all patients participating in our study
Author information
Authors and Affiliations
Corresponding author
Ethics declarations
Conflict of interest
The authors declare that they have no conflict of interest.
Ethical approval
All procedures performed in studies involving human participants were in accordance with the ethical standards of the institutional and/or national research committee and with the 1964 Helsinki declaration and its later amendments or comparable ethical standards. The study was approved by the local Institutional Review Board of the Ludwig-Maximilians-University Munich and agrees with all standards regarding the use of informed consent according to the guidelines of the local Institutional Review Board of the Ludwig-Maximilians-University Munich (Project No. 19-569).
Informed consent
Informed consent was obtained from participants included in the study according to the guidelines of the local Institutional Review Board of the Ludwig-Maximilians-University Munich (Project No. 19-569).
Additional information
Publisher’s note
Springer Nature remains neutral with regard to jurisdictional claims in published maps and institutional affiliations.
This article is part of the Topical Collection on Spine - Other
Presentation at a conference:
None
Rights and permissions
About this article
Cite this article
Siller, S., Egensperger, R., Szelenyi, A. et al. Intraspinal epidermoid and dermoid cysts—tumor resection with multimodal intraoperative neurophysiological monitoring and long-term outcome. Acta Neurochir 162, 2895–2903 (2020). https://doi.org/10.1007/s00701-020-04446-y
Received:
Accepted:
Published:
Issue Date:
DOI: https://doi.org/10.1007/s00701-020-04446-y