Spinal dermal sinus coinciding with an infected giant epidermoid cyst in an infant presenting with constipation: a case report
Keywords:
constipation; infections; neoplasms; neural tube defect; surgical procedures
Abstract
Early diagnosis of spinal tumors is a prerequisite for achieving satisfactory neurological recovery. However, rare diseases tend to have a long diagnostic course.1 In addition, the clinical presentation of spinal tumors in most children is not apparent, moreover, spinal tumors may not show clinical symptoms if they are located in the lumbar spine and sized no more than one vertebra.2,3 Clinical presentations caused by tumors in the lumbar spine may include weakness of both limbs, impaired urination, and constipation.
References
1. Benito-Lozano J, Arias-Merino G, Gómez-Martínez M, Ancochea-Díaz A, Aparicio-García A, Posada de la Paz M, et al. Diagnostic Process in Rare Diseases: Determinants Associated with Diagnostic Delay. Int J Environ Res Public Health 2022;19:6456. DOI: https://doi.org/10.3390/ijerph19116456.
2. Spacca B, Giordano F, Donati P, Genitori L. Spinal tumors in children: long-term retrospective evaluation of a series of 134 cases treated in a single unit of pediatric neurosurgery. Spine J. 2015;15:1949–55. DOI: https://doi.org/10.1016/j.spinee.2015.04.012.
3. Hartman J, Granville M, Jacobson RE. Two cases with incidental finding of large asymptomatic intradural lumbar tumors. Cureus. 2018;10:e3446. DOI: https://doi.org/10.7759/cureus.3446.
4. Beechar VB, Zinn PO, Heck KA, Fuller GN, Han I, Patel Aj, et al. Spinal epidermoid tumors: case report and review of the literature. Neurospine. 2018;15:117-122. DOI: https://doi.org/10.14245/ns.1836014.007.
5. Cavus G, Istemen I, Acik V, Bilgin E, Arslan A, Millet H, et al. Spinal dermoid and epidermoid tumors: clinical series of 15 cases. Ann Med Res. 2018;25:667-72. DOI: https://doi.org/10.5455/annalsmedres.2018.06.114.
6. Acharyya S, Chakravarty S. Congenital dermal sinus with extensive intramedullary expansion and an infected spinal epidermoid cyst in an infant. J Family Med Prim Care. 2018;7:1103–5. DOI: https://doi.org/10.4103/jfmpc.jfmpc_158_18.
7. Katz-Lavigne M, Smith WG. Spinal epidermoid tumours following neonatal lumbar puncture: a review of the evidence. Paediatr Child Health. 2013;18:241-5. DOI: https://doi.org/10.1093/pch/18.5.241.
8. Radmanesh F, Nejat F, El Khashab M. Dermal sinus tract of the spine. Childs Nerv Syst. 2010;26:349-57. DOI: https://doi.org/10.1007/s00381-009-0962-z.
9. Ramnarayan R, Dominic A, Alapatt J, Buxton N. Congenital spinal dermal sinuses: poor awareness leads to delayed treatment. Childs Nerv Syst. 2006;22:1220-4. DOI: https://doi.org/10.1007/s00381-006-0073-z.
10. Richter T, Nestler-Parr S, Babela R, Khan ZM, Tesoro T, Molsen E, et al. Rare disease terminology and definitions - A systematic global review: Report of the ISPOR Rare Disease Special Interest Group. Value Health. 2015;18:906-14. DOI: https://doi.org/10.1016/j.jval.2015.05.008
11. Ackerman LL, Menezes AH. Spinal congenital dermal sinuses: a-30-year experience. Pediatrics. 2003;112:641-7. DOI: https://doi.org/10.1542/peds.112.3.641.
12. Song YH, Xia ZY, Qiu SS, Gao PP, Yang B, Bao N. Surgical treatment of congenital dermal sinus: An experience of 56 cases. Pediatr Neurosurg. 2021;56:416-23. DOI: https://doi.org/10.1159/000515515.
13. Singh I, Rohilla S, Kumar P, Sharma S. Spinal dorsal dermal sinus tract: An experience of 21 cases. Surg Neurol Int. 2015;6:S429-34. DOI: https://doi.org/10.4103/2152-7806.166752.
14. Mete M, Umur AS, Duransoy KY, Barutcuoglu M, Umur N, Gurgen SG, et al. Congenital dermal sinus tract of the spine: Experience of 16 patients. J Child Neurol. 2014;29:1277-82. DOI: https://doi.org/10.1177/0883073813520501.
15. Mehta DV. Magnetic resonance imaging in paediatric spinal dysraphism with comparative usefulness of various magnetic resonance sequences. J Clin Diagn Res. 2017;11:TC17–22. DOI: https://doi.org/10.7860/JCDR/2017/30134.10393.
16. Choi SJ, Yoon HM, Hwang JS, Suh CH, Jung AY, Cho YA, et al. Incidence of occult spinal dysraphism among infants with cutaneous stigmata and proportion managed with neurosurgery: A systematic review and meta-analysis. JAMA Netw Open. 2020;3:e207221. DOI: https://doi.org/10.1001/jamanetworkopen.2020.7221.
17. Merzeau N, Poli-Merol ML, Jolly D, Bourelle S, Jolly C. Occult spinal dysraphism: Utility of cutaneous lesions as a screening tool for early diagnosis in a series of 31 cases. Remed Open Access. 2017;2:1045.
18. Tawfik NA, Ahmed AT, El-Shafei TE, Habba MR. Diagnostic value of spinal ultrasound compared to MRI for diagnosis of spinal anomalies in pediatrics. Egypt J Radiol Nucl Med. 2020;51:18. DOI: https://doi.org/10.1186/s43055-020-0131-7.
19. Alamdaran SA, Mohammadpanah N, Zabihian S, Esmaeeli M, Ghane F, Feyzi A. Diagnostic value of ultrasonography in spinal abnormalities among children with neurogenic bladder. Electron Physician 2017;9:4571-6. DOI: https://doi.org/10.19082/4571.
20. Kriss VM, Desai NS. Occult spinal dysraphism in neonates: assessment of high-risk cutaneous stigmata on sonography. Am J Roentgenol. 1998;171:1687-92. DOI: https://doi.org/10.2214/ajr.171.6.9843314.
21. Venkataramana NK. Spinal dysraphism. J Pediatr Neurosci. 2011;6:S31-40. DOI: https://doi.org/10.4103/1817-1745.85707.
22. Yin H, Zhang D, Wu Z, Zhou W, Xiao J. Surgery and outcomes of six patients with intradural epidermoid cysts in the lumbar spine. World J Surg Oncol. 2014;12:50. DOI: https://doi.org/10.1186/1477-7819-12-50.
23. van Mill MJ, Koppen IJN, Benninga MA. Controversies in the management of functional constipation in children. Curr Gastroenterol Rep. 2019;21:23. DOI: https://doi.org/10.1007/s11894-019-0690-9.
24. Zusman NL, Radoslovich SS, Smith SJ, Tanski M, Gundle KR, Yoo JU. Physical examination is predictive of cauda equina syndrome: MRI to rule out diagnosis is unnecessary. Global Spine J. 2020:2192568220948804. DOI: https://doi.org/10.1177/2192568220948804.
25. Bevan R, Leach P. Infected congenital cervical dermal sinuses leading to spinal cord abscess: two case reports and a review of the literature. Childs Nerv Syst. 2021;37:225-8. DOI: https://doi.org/10.1007/s00381-020-04778-1.
26. Emma-Naeni P, Mahdavi A, Ahmadi H, Baradaran N, Nejat F. Brain abscess as a manifestation of spinal dermal sinus. Ther Clin Risk Manag. 2008;4:1143-7. DOI: https://doi.org/10.2147/tcrm.s2533.
27. Prasad GL, Hedge A, Divya S. Spinal intramedullary abscess secondary to dermal sinus in children. Eur J Pediatr Surg. 2019;29:229-38. DOI: https://doi.org/10.1055/s-0038-1655736.
28. Mattar MAB, Kassem M, Sabry AM. Complicated congenital dermal sinus: Diagnosis and management. Interdiscip Neurosurg. 2020;21:100739. DOI: 10.1016/j.inat.2020.100739.
29. Porat D, Finkel L, Eilbert W. Infected intracranial dermal sinus tract in a child. Am J Emerg Med. 2021;49:206–8. DOI: https://doi.org/10.1016/j.ajem.2021.06.016.
30. Fish DN. Meropenem in the treatment of complicated skin and soft tissue infections. Ther Clin Risk Manag. 2006;2:401-15. DOI: https://doi.org/10.2147/tcrm.2006.2.4.401.
31. Papaevangelou G, Tsitsopoulos PP, Flaris N, Iliadis C, Tsonidis C. Dermal sinus tract of the thoracic spine presenting with intramedullary abscess and cranial nerve deficits. Pediatr Neurosurg. 2015;50:339–43. DOI: https://doi.org/10.1159/000439539.
32. Yasuoka S, Peterson HA, MacCarty CS. Incidence of spinal column deformity after multilevel laminectomy in children and adults. J Neurosurg. 1982;57:441-5. DOI: https://doi.org/10.3171/jns.1982.57.4.0441.
33. Lee JH, Jang JW, Kim SH, Moon HY, Lee JK, Kim SH. Surgical results after unilateral laminectomy for the removal of spinal cord tumors. Korean J Spine. 2012;9:232-8. DOI: https://doi.org/10.14245/kjs.2012.9.3.232.
34. Dobran M, Paracino R, Nasi D, Aiudi D, Capece M, Carrassi E, et al. Laminectomy versus unilateral hemilaminectomy for the removal of intraspinal schwannoma: Experience of a single institution and review of literature. J Neurol Surg. 2021;82. DOI: https://doi.org/10.1055/s-0041-1722968.
35. Sridhar K, Ramamurthi R, Vasudevan MC, Ramamurthi B. Giant invasive spinal schwannomas: definition and surgical management. J Neurosurg. 2001;94Suppl2:210-5. DOI: https://doi.org/10.3171/spi.2001.94.2.0210. PMID: 11302622.
36. Li J, Qian M, Huang X, Zhao L, Yang X, Xiao J. Repeated recurrent epidermoid cyst with atypical hyperplasia. Medicine. 2017;96:e8950. DOI: https://doi.org/10.1097/MD.0000000000008950.
2. Spacca B, Giordano F, Donati P, Genitori L. Spinal tumors in children: long-term retrospective evaluation of a series of 134 cases treated in a single unit of pediatric neurosurgery. Spine J. 2015;15:1949–55. DOI: https://doi.org/10.1016/j.spinee.2015.04.012.
3. Hartman J, Granville M, Jacobson RE. Two cases with incidental finding of large asymptomatic intradural lumbar tumors. Cureus. 2018;10:e3446. DOI: https://doi.org/10.7759/cureus.3446.
4. Beechar VB, Zinn PO, Heck KA, Fuller GN, Han I, Patel Aj, et al. Spinal epidermoid tumors: case report and review of the literature. Neurospine. 2018;15:117-122. DOI: https://doi.org/10.14245/ns.1836014.007.
5. Cavus G, Istemen I, Acik V, Bilgin E, Arslan A, Millet H, et al. Spinal dermoid and epidermoid tumors: clinical series of 15 cases. Ann Med Res. 2018;25:667-72. DOI: https://doi.org/10.5455/annalsmedres.2018.06.114.
6. Acharyya S, Chakravarty S. Congenital dermal sinus with extensive intramedullary expansion and an infected spinal epidermoid cyst in an infant. J Family Med Prim Care. 2018;7:1103–5. DOI: https://doi.org/10.4103/jfmpc.jfmpc_158_18.
7. Katz-Lavigne M, Smith WG. Spinal epidermoid tumours following neonatal lumbar puncture: a review of the evidence. Paediatr Child Health. 2013;18:241-5. DOI: https://doi.org/10.1093/pch/18.5.241.
8. Radmanesh F, Nejat F, El Khashab M. Dermal sinus tract of the spine. Childs Nerv Syst. 2010;26:349-57. DOI: https://doi.org/10.1007/s00381-009-0962-z.
9. Ramnarayan R, Dominic A, Alapatt J, Buxton N. Congenital spinal dermal sinuses: poor awareness leads to delayed treatment. Childs Nerv Syst. 2006;22:1220-4. DOI: https://doi.org/10.1007/s00381-006-0073-z.
10. Richter T, Nestler-Parr S, Babela R, Khan ZM, Tesoro T, Molsen E, et al. Rare disease terminology and definitions - A systematic global review: Report of the ISPOR Rare Disease Special Interest Group. Value Health. 2015;18:906-14. DOI: https://doi.org/10.1016/j.jval.2015.05.008
11. Ackerman LL, Menezes AH. Spinal congenital dermal sinuses: a-30-year experience. Pediatrics. 2003;112:641-7. DOI: https://doi.org/10.1542/peds.112.3.641.
12. Song YH, Xia ZY, Qiu SS, Gao PP, Yang B, Bao N. Surgical treatment of congenital dermal sinus: An experience of 56 cases. Pediatr Neurosurg. 2021;56:416-23. DOI: https://doi.org/10.1159/000515515.
13. Singh I, Rohilla S, Kumar P, Sharma S. Spinal dorsal dermal sinus tract: An experience of 21 cases. Surg Neurol Int. 2015;6:S429-34. DOI: https://doi.org/10.4103/2152-7806.166752.
14. Mete M, Umur AS, Duransoy KY, Barutcuoglu M, Umur N, Gurgen SG, et al. Congenital dermal sinus tract of the spine: Experience of 16 patients. J Child Neurol. 2014;29:1277-82. DOI: https://doi.org/10.1177/0883073813520501.
15. Mehta DV. Magnetic resonance imaging in paediatric spinal dysraphism with comparative usefulness of various magnetic resonance sequences. J Clin Diagn Res. 2017;11:TC17–22. DOI: https://doi.org/10.7860/JCDR/2017/30134.10393.
16. Choi SJ, Yoon HM, Hwang JS, Suh CH, Jung AY, Cho YA, et al. Incidence of occult spinal dysraphism among infants with cutaneous stigmata and proportion managed with neurosurgery: A systematic review and meta-analysis. JAMA Netw Open. 2020;3:e207221. DOI: https://doi.org/10.1001/jamanetworkopen.2020.7221.
17. Merzeau N, Poli-Merol ML, Jolly D, Bourelle S, Jolly C. Occult spinal dysraphism: Utility of cutaneous lesions as a screening tool for early diagnosis in a series of 31 cases. Remed Open Access. 2017;2:1045.
18. Tawfik NA, Ahmed AT, El-Shafei TE, Habba MR. Diagnostic value of spinal ultrasound compared to MRI for diagnosis of spinal anomalies in pediatrics. Egypt J Radiol Nucl Med. 2020;51:18. DOI: https://doi.org/10.1186/s43055-020-0131-7.
19. Alamdaran SA, Mohammadpanah N, Zabihian S, Esmaeeli M, Ghane F, Feyzi A. Diagnostic value of ultrasonography in spinal abnormalities among children with neurogenic bladder. Electron Physician 2017;9:4571-6. DOI: https://doi.org/10.19082/4571.
20. Kriss VM, Desai NS. Occult spinal dysraphism in neonates: assessment of high-risk cutaneous stigmata on sonography. Am J Roentgenol. 1998;171:1687-92. DOI: https://doi.org/10.2214/ajr.171.6.9843314.
21. Venkataramana NK. Spinal dysraphism. J Pediatr Neurosci. 2011;6:S31-40. DOI: https://doi.org/10.4103/1817-1745.85707.
22. Yin H, Zhang D, Wu Z, Zhou W, Xiao J. Surgery and outcomes of six patients with intradural epidermoid cysts in the lumbar spine. World J Surg Oncol. 2014;12:50. DOI: https://doi.org/10.1186/1477-7819-12-50.
23. van Mill MJ, Koppen IJN, Benninga MA. Controversies in the management of functional constipation in children. Curr Gastroenterol Rep. 2019;21:23. DOI: https://doi.org/10.1007/s11894-019-0690-9.
24. Zusman NL, Radoslovich SS, Smith SJ, Tanski M, Gundle KR, Yoo JU. Physical examination is predictive of cauda equina syndrome: MRI to rule out diagnosis is unnecessary. Global Spine J. 2020:2192568220948804. DOI: https://doi.org/10.1177/2192568220948804.
25. Bevan R, Leach P. Infected congenital cervical dermal sinuses leading to spinal cord abscess: two case reports and a review of the literature. Childs Nerv Syst. 2021;37:225-8. DOI: https://doi.org/10.1007/s00381-020-04778-1.
26. Emma-Naeni P, Mahdavi A, Ahmadi H, Baradaran N, Nejat F. Brain abscess as a manifestation of spinal dermal sinus. Ther Clin Risk Manag. 2008;4:1143-7. DOI: https://doi.org/10.2147/tcrm.s2533.
27. Prasad GL, Hedge A, Divya S. Spinal intramedullary abscess secondary to dermal sinus in children. Eur J Pediatr Surg. 2019;29:229-38. DOI: https://doi.org/10.1055/s-0038-1655736.
28. Mattar MAB, Kassem M, Sabry AM. Complicated congenital dermal sinus: Diagnosis and management. Interdiscip Neurosurg. 2020;21:100739. DOI: 10.1016/j.inat.2020.100739.
29. Porat D, Finkel L, Eilbert W. Infected intracranial dermal sinus tract in a child. Am J Emerg Med. 2021;49:206–8. DOI: https://doi.org/10.1016/j.ajem.2021.06.016.
30. Fish DN. Meropenem in the treatment of complicated skin and soft tissue infections. Ther Clin Risk Manag. 2006;2:401-15. DOI: https://doi.org/10.2147/tcrm.2006.2.4.401.
31. Papaevangelou G, Tsitsopoulos PP, Flaris N, Iliadis C, Tsonidis C. Dermal sinus tract of the thoracic spine presenting with intramedullary abscess and cranial nerve deficits. Pediatr Neurosurg. 2015;50:339–43. DOI: https://doi.org/10.1159/000439539.
32. Yasuoka S, Peterson HA, MacCarty CS. Incidence of spinal column deformity after multilevel laminectomy in children and adults. J Neurosurg. 1982;57:441-5. DOI: https://doi.org/10.3171/jns.1982.57.4.0441.
33. Lee JH, Jang JW, Kim SH, Moon HY, Lee JK, Kim SH. Surgical results after unilateral laminectomy for the removal of spinal cord tumors. Korean J Spine. 2012;9:232-8. DOI: https://doi.org/10.14245/kjs.2012.9.3.232.
34. Dobran M, Paracino R, Nasi D, Aiudi D, Capece M, Carrassi E, et al. Laminectomy versus unilateral hemilaminectomy for the removal of intraspinal schwannoma: Experience of a single institution and review of literature. J Neurol Surg. 2021;82. DOI: https://doi.org/10.1055/s-0041-1722968.
35. Sridhar K, Ramamurthi R, Vasudevan MC, Ramamurthi B. Giant invasive spinal schwannomas: definition and surgical management. J Neurosurg. 2001;94Suppl2:210-5. DOI: https://doi.org/10.3171/spi.2001.94.2.0210. PMID: 11302622.
36. Li J, Qian M, Huang X, Zhao L, Yang X, Xiao J. Repeated recurrent epidermoid cyst with atypical hyperplasia. Medicine. 2017;96:e8950. DOI: https://doi.org/10.1097/MD.0000000000008950.
Published
2022-10-27
How to Cite
1.
Saekhu M, Susanto E, Handryastuti S, Ashari S, Nugroho S. Spinal dermal sinus coinciding with an infected giant epidermoid cyst in an infant presenting with constipation: a case report. PI [Internet]. 27Oct.2022 [cited 22Dec.2024];62(5):357-3. Available from: https://paediatricaindonesiana.org/index.php/paediatrica-indonesiana/article/view/2528
Section
Case Report
Copyright (c) 2022 Mohamad Saekhu, Eka Susanto, Setyo Handryastuti, Samsul Ashari, Setyowidi Nugroho
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Received 2020-11-10
Accepted 2022-10-27
Published 2022-10-27
Accepted 2022-10-27
Published 2022-10-27
