Spinal Dermoid and Epidermoid Cyst: An Institutional Experience and Clinical Insight into the Neural Tube Closure ModelsFunding None.
Objectives The spinal dermoid and epidermoid cysts (SDECs) are rare entities comprising less than 1% of pediatric intraspinal tumors. The present study aims to extrapolate the clinicoradiological data, in order to identify the most plausible neural tube closure model in human and provide a retrospective representation from our clinical experience.
Materials and Methods We collected the details of all histologically proven, newly diagnosed primary SDECs who underwent excision over the past 20 years. Secondary or recurrent lesions and other spinal cord tumors were excluded. Surgical and follow-up details of these patients as well as those with associated spinal dysraphism were reviewed. Clinical and radiological follow-up revealed the recurrence in these inborn spinal cord disorders.
Results A total of 73 patients were included retrospectively, having a mean age of 22.4 ± 13.3 years, and 41 (56.2%) cases fell in the first two decades of life. Twenty-four (32.9%) dermoid and 49 (67.1%) epidermoid cysts comprised the study population and 20 of them had associated spinal dysraphism. The distribution of SDECs was the most common in lumbosacral region (n = 30) which was 10 times more common than in the sacral region (n = 3). Bladder dysfunction 50 (68.5%) and pain 48 (65.7%) were the most common presenting complaints. During follow-up visits, 40/48 (83.3%) cases showed sensory improvement while 11/16 (68.7%) regained normal bowel function. There was no surgical mortality with recurrence seen in eight till the last follow-up.
Conclusions The protracted clinical course of the spinal inclusion cysts mandates a long-term follow-up. The results of our study support the multisite closure model and attempt to provide a retrospective reflection of neural tube closure model in humans by using SDECs as the surrogate marker of neural tube closure defect.
24. März 2021 (online)
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- 1 Roux A, Mercier C, Larbrisseau A. Dube LJ, Dupuis C, Del Carpio R. Intramedullary epidermoid cysts of the spinal cord. Case report. J Neurosurg 1992; 76 (03) 528-533
- 2 Van Allen MI, Kalousek DK, Chernoff GF. et al. Evidence for multi-site closure of the neural tube in humans. Am J Med Genet 1993; 47 (05) 723-743
- 3 Golden JA, Chernoff GF. Anterior neural tube in the mouse: fuel for disagreement with the classical theory. Clin Res 1983; 31: 127A
- 4 Juriloff DM, Harris MJ, Tom C, MacDonald KB. Normal mouse strains differ in the site of initiation of closure of the cranial neural tube. Teratology 1991; 44 (02) 225-233
- 5 van Straaten HWM, Peeters MCE, Hekking JWM, van der Lende T. Neurulation in the pig embryo. Anat Embryol (Berl) 2000; 202 (02) 75-84
- 6 O’Rahilly R, Müller F. Neurulation in the normal human embryo. Ciba Found Symp 1994; 181: 70-82 discussion 82-89
- 7 Nakatsu T, Uwabe C, Shiota K. Neural tube closure in humans initiates at multiple sites: evidence from human embryos and implications for the pathogenesis of neural tube defects. Anat Embryol (Berl) 2000; 201 (06) 455-466
- 8 Shikata J, Yamamuro T, Mikawa Y, Kotoura Y. Intraspinal epidermoid and dermoid cysts. Surgical results of seven cases. Arch Orthop Trauma Surg 1988; 107 (02) 105-109
- 9 Netsky MG. Epidermoid tumors. Review of the literature. Surg Neurol 1988; 29 (06) 477-483
- 10 Van JC Gilder, Schwartz HG. Growth of dermoids from skin implants to the nervous system and surrounding spaces of the newborn rat. J Neurosurg 1967; 26 (01) 14-20
- 11 Golden JA, Chernoff GF. Intermittent pattern of neural tube closure in two strains of mice. Teratology 1993; 47 (01) 73-80
- 12 Sakai Y. Neurulation in the mouse: manner and timing of neural tube closure. Anat Rec 1989; 223 (02) 194-203
- 13 Nishimura H. Prenatal versus postnatal malformations based on the Japanese experience on induced abortions in the human being.. In: Blandau RJ, ed. Aging gametes. Basel: Karger 1975: 349-368
- 14 Gonzalvo A, Hall N, McMahon JHA, Fabinyi GC. Intramedullary spinal epidermoid cyst of the upper thoracic region. J Clin Neurosci 2009; 16 (01) 142-144
- 15 Cincu R, Lázaro JF, Liesa JL, Callizo JR. Dorsal intramedullary spinal epidermoid cysts: report of two cases and review of literature. Indian J Orthop 2007; 41 (04) 395-397
- 16 Zavanone M, Guerra P, Rampini PM, Crotti F, Vaccari U. A cervicodorsal intramedullary epidermoid cyst. J Neurosurg Sci 1991; 35: 111-115
- 17 Guidetti B, Gagliardi FM. Epidermoid and dermoid cysts. Clinical evaluation and late surgical results. J Neurosurg 1977; 47 (01) 12-18
- 18 Falavigna A, Righesso O, Teles AR. Concomitant dermoid cysts of conus medullaris and cauda equina. Arq Neuropsiquiatr 2009; 67 (2A) 293-296
- 19 Sun JC, Steinbok P, Cochrane DD. Cervical myelocystoceles and meningoceles: long-term follow-up. Pediatr Neurosurg 2000; 33 (03) 118-122
- 20 Orakdogen M, Turk CC, Ersahin M, Biber N, Berkman Z. Spinal dysraphisms of the cervicothoracic region in childhood. Turk Neurosurg 2009; 19 (04) 400-405
- 21 Myles LM, Steers AJ, Minns R. Cervical cord tethering due to split cord malformation at the cervico-dorsal junction presenting with self-mutilation of the fingers. Dev Med Child Neurol 2002; 44 (12) 844-848
- 22 Pang D, Zovickian J, Oviedo A, Moes GS. Limited dorsal myeloschisis: a distinctive clinicopathological entity. Neurosurgery 2010; 67 (06) 1555-1579 discussion 1579–1580
- 23 Pessoa BL, Lima Y, Orsini M. True cervicothoracic meningocele: a rare and benign condition. Neurol Int 2015; 7 (03) 6079
- 24 Sewell MJ, Chiu YE, Drolet BA. Neural tube dysraphism: review of cutaneous markers and imaging. Pediatr Dermatol 2015; 32 (02) 161-170
- 25 Liu H, Zhang JN, Zhu T. Microsurgical treatment of spinal epidermoid and dermoid cysts in the lumbosacral region. J Clin Neurosci 2012; 19 (05) 712-717
- 26 van Aalst J, Beuls EAM, Cornips EMJ. et al. Anatomy and surgery of the infected dermal sinus of the lower spine. Childs Nerv Syst 2006; 22 (10) 1307-1315
- 27 Kanev PM, Park TS. Dermoids and dermal sinus tracts of the spine. Neurosurg Clin N Am 1995; 6 (02) 359-366
- 28 Mohindra S, Gupta R, Chhabra R. et al. Infected intraparenchymal dermoids: an underestimated entity. J Child Neurol 2008; 23 (09) 1011-1016
- 29 Chen CY, Lin KL, Wang HS, Lui TN. Dermoid cyst with dermal sinus tract complicated with spinal subdural abscess. Pediatr Neurol 1999; 20 (02) 157-160
- 30 van Aalst J, Hoekstra F, Beuls EAM. et al. Intraspinal dermoid and epidermoid tumors: report of 18 cases and reappraisal of the literature. Pediatr Neurosurg 2009; 45 (04) 281-290
- 31 Barkovich AJ, Edwards MS, Cogen PH. MR evaluation of spinal dermal sinus tracts in children. AJNR Am J Neuroradiol 1991; 12 (01) 123-129
- 32 Liu JK, Gottfried ON, Salzman KL, Schmidt RH, Couldwell WT. Ruptured intracranial dermoid cysts: clinical, radiographic, and surgical features. Neurosurgery 2008; 62 (02) 377-384 discussion 384
- 33 Sandalcioglu IE, Gasser T, Asgari S. et al. Functional outcome after surgical treatment of intramedullary spinal cord tumors: experience with 78 patients. Spinal Cord 2005; 43 (01) 34-41
- 34 Wiedemayer H, Sandalcioglu IE, Aalders M, Wiedemayer H, Floerke M, Stolke D. Reconstruction of the laminar roof with miniplates for a posterior approach in intraspinal surgery: technical considerations and critical evaluation of follow-up results. Spine 2004; 29 (16) E333-E342
- 35 Velayutham P, Cherian VT, Rajshekhar V, Babu KS. The effects of propofol and isoflurane on intraoperative motor evoked potentials during spinal cord tumour removal surgery - A prospective randomised trial. Indian J Anaesth 2019; 63 (02) 92-99
- 36 Ali Z, Bithal PK. Intra-operative neurophysiological monitoring. J Neuroanaesth Crit Care 2015; 2: 179-192
- 37 Deletis V, Sala F. Intraoperative neurophysiological monitoring of the spinal cord during spinal cord and spine surgery: a review focus on the corticospinal tracts. Clin Neurophysiol 2008; 119 (02) 248-264
- 38 Kelleher MO, Tan G, Sarjeant R, Fehlings MG. Predictive value of intraoperative neurophysiological monitoring during cervical spine surgery: a prospective analysis of 1055 consecutive patients. J Neurosurg Spine 2008; 8 (03) 215-221
- 39 Constantini S, Miller DC, Allen JC, Rorke LB, Freed D, Epstein FJ. Radical excision of intramedullary spinal cord tumors: surgical morbidity and long-term follow-up evaluation in 164 children and young adults. J Neurosurg 2000; 93 (Suppl. 02) 183-193
- 40 Munshi A, Talapatra K, Ramadwar M, Jalali R. Spinal epidermoid cyst with sudden onset of paraplegia. J Cancer Res Ther 2009; 5 (04) 290-292