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Intramedullary cavernous angioma of the spinal cord in a pediatric patient, with multiple cavernomas, familial occurrence and partial spontaneous regression: case report and review of the literature

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Abstract

Object

The authors present their experience with the 17th pediatric intramedullary cavernoma reported in English literature.

Methods

The patient firstly underwent surgery for a left frontal cavernoma when he was 2 years old. Also the child’s mother was operated for a C2–C3 intramedullary cavernoma. He grew up normally and the radiological follow-up was negative for other brainstem cavernous malformations. When he was 11 years old he complained a worsening tetraparesis. A cerebral and spinal magnetic resonance (MR) imaging revealed the presence of a C1 intramedullary cavernoma and a pontine cavernoma. He underwent surgery for the cervical lesion, which was completely removed. The postoperative course was regular with a total recovery from the neurological deficit. The boy underwent a radiological follow-up, monitoring the pontine lesion, which spontaneously regressed when he was 19 years old.

Conclusion

The rarity of the pediatric intramedullary cavernoma, the familial occurrence, and the spontaneous regression of the pontine cavernoma make this case very peculiar.

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References

  1. Abid R, Carlier R, Idir AB, David P, Hurth M, Doyon D (1993) Brain and spinal cord cavernoma. Value of MRI and review of the literature. Apropos of a case. J Radiol 74(11):563–567

    PubMed  CAS  Google Scholar 

  2. Anson JA, Spetzler RF (1993) Surgical resection of intramedullary spinal cord cavernous malformations. J Neurosurg 78:446–451

    PubMed  CAS  Google Scholar 

  3. Bakir A, Savas A, Yilmaz E, Savaş B, Erden E, Cağlar S, Sener Ö (2006) Spinal intradural–intramedullary cavernous malformation. Pediatr Neurosurg 42:35–37

    Article  PubMed  Google Scholar 

  4. Balousek P, Ammirati M (1996) Exophytic cavernous malformations of the cervical spinal cord. Acta Neurochir (Wien) 138:890–892

    Article  CAS  Google Scholar 

  5. Barnwell SL, Dowd CF, Davis RL, Edwards MSB, Gutin PH, Wilson CB (1990) Cryptic vascular malformations of the spinal cord: diagnosis by magnetic resonance imaging and outcome of surgery. J Neurosurg 72:403–407

    PubMed  CAS  Google Scholar 

  6. Bergstrand A, Hook O, Lindvall H (1964) Vascular malformations of the spinal cord. Acta Neurol Scand 40:169–183

    Article  PubMed  CAS  Google Scholar 

  7. Bicknell JM, Carlow TJ, Kornfeld M, Stovring J, Turner P (1978) Familial cavernous angiomas. Arch Neurol 35:746–749

    PubMed  CAS  Google Scholar 

  8. Bourgouin PM, Tampieri D, Johnston W, Steward J, Melancon D, Ethier R (1992) Multiple occult vascular malformations of the brain and spinal cord: MRI diagnosis. Neuroradiology 34:110–111

    Article  PubMed  CAS  Google Scholar 

  9. Cantore GP, Delfini R, Cervoni L, Innocenzi G, Ramundo EO (1995) Intramedullary cavernous angiomas of the spinal cord: report of six cases. Surg Neurol 43:448–452

    Article  PubMed  CAS  Google Scholar 

  10. Cantore GP, Missori P, Santoro A (1999) Cavernous angiomas of the brain stem. Intra-axial anatomical pitfalls and surgical strategies. Surg Neurol 52:84–94

    Article  PubMed  CAS  Google Scholar 

  11. Cardon T, Wryemblewsky P, Lesoin F, Brunaud V, Destee A (1992) Angiome caverneux de la moelle cervicale. Rev Neurol 18:152–154

    Google Scholar 

  12. Clatterbuck RE, Moriarity JL, Elmaci I, Lee RR, Bretter SN, Rigamonti D (2000) Dynamic nature of cavernous malformations: a prospective magnetic resonance imaging study with volumetric analysis. J Neurosurg 93:981–986

    PubMed  CAS  Google Scholar 

  13. Cosgrove GR, Bertrand G, Fontaine S, Robitaille Y, Melancon D (1988) Cavernous angiomas of the spinal cord. J Neurosurg 68:31–36

    PubMed  CAS  Google Scholar 

  14. Cristante L, Hermann HD (1998) Radical excision of intramedullary cavernous angiomas. Neurosurgery 43:424–431

    Article  PubMed  CAS  Google Scholar 

  15. Deutsch H, Jallo GI, Faktorovich A, Epstein F (2000) Spinal intramedullary cavernoma: clinical presentation and surgical outcome. J Neurosurg 1 93:65–70

    CAS  Google Scholar 

  16. Deutsch H, Shrivistava R, Epstein F, Jallo GI (2001) Pediatric intramedullary spinal cavernous malformations. Spine 26(18):427–431

    Article  Google Scholar 

  17. Fazi S, Menei P, Mercier P, Dubas F, Guy G (1992) Cavernomas of the spinal cord: report of two patients. Br J Neurosurg 6:149–152

    PubMed  CAS  Google Scholar 

  18. Feider HK, Yuille D (1991) An epidural cavernous hemangioma of the spine. AJNR 12:243–244

    PubMed  CAS  Google Scholar 

  19. Fontaine D, Lot D, George B (1999) Intramedullary cavernous angioma. Resection by oblique corpectomy. Surg Neurol 51:435–442

    Article  PubMed  CAS  Google Scholar 

  20. Furuya K, Sasaki T, Susuki I, Kim P, Saito N, Kirino T (1996) Intramedullary angiographically occult vascular malformations of the spinal cord. Neurosurgery 39:1123–1132

    Article  PubMed  CAS  Google Scholar 

  21. Gagel O, Meszaros A (1947) Zur Frage der myelopathia necroticans. Arch Psych Neurol 179:423–429

    Article  Google Scholar 

  22. Gao PY (1989) Magnetic resonance imaging of occult intracranial malformations. Chung Hua I Hsueh Tsa Chih 69:375–377

    PubMed  CAS  Google Scholar 

  23. Goran A, Carlson DJ, Fischer RG (1964) Successful treatment of intramedullary angioma of the cord. J Neurosurg 21:311–314

    PubMed  CAS  Google Scholar 

  24. Gordon CR, Crockard HA, Symon L (1995) Surgical management of spinal cord cavernoma. Br J Neurosurg 9:459–464

    Article  PubMed  CAS  Google Scholar 

  25. Guthkelch AN (1948) Haemangiomas involving the spinal epidural space. J Neurol Neurosurg Psychiatry 11:199–210

    PubMed  CAS  Google Scholar 

  26. Hadlich R (1903) Ein Fall von Tumor cavernous des Rückenmarks mit besonderer Berücksichtigung der neueren Theorien über die Gene des Cavernoms. Virchows Arch 172:429–441

    Article  Google Scholar 

  27. Harrison MJ, Eisenberg MB, Ullman JS, Oppenheim JS, Camins MB, Post KD (1995) Symptomatic cavernous malformations affecting the spine and spinal cord. Neurosurgery 37:195–205

    Article  PubMed  CAS  Google Scholar 

  28. Heimberger K, Schnabert G, Koos W et al (1982) Spinal cavernous haemangioma (intradural-extramedullary) underlying repeated subarachnoid haemorrhage. J Neurol 226:289–293

    Article  PubMed  CAS  Google Scholar 

  29. Hernandez D, Moraleda S, Royo A, Martinez M, Garcia J, Vazquez MJ (1999) Cavernous angioma of the conus medullaris as a cause of paraplegia. Spinal Cord 44:428

    Google Scholar 

  30. Hillman J, Bynke O (1991) Solitary extradural cavernous hemangiomas in the spinal canal. Report of five cases. Surg Neurol 36:19–24

    Article  PubMed  CAS  Google Scholar 

  31. Jallo GI, Freed D, Zareck M, Epstein F, Kothbauer K (2006) Neurosurg Focus 21(1):E10

    Article  PubMed  Google Scholar 

  32. Jellinger K (1978) Pathology of spinal vascular malformations and vascular tumors. In: Pia HW, Djindjian R (eds) Spinal angiomas: advances in diagnosis and therapy. Springer, Berlin

    Google Scholar 

  33. Kitahara T, Miyasaka Y, Ohwada T, Yada K, Mera H (1982) An operated case of cervical spontaneous hematomyelia. Neurol Surg 10:675–679

    CAS  Google Scholar 

  34. Lee KS, Spetzler RF (1990) Spinal cord cavernous malformations in a patient with familial intracranial cavernous malformations. Neurosurgery 26:877–880

    Article  PubMed  CAS  Google Scholar 

  35. Lindboe CF, Nordal HJ (1985) Multiple neurilemmomas of the cauda equina, cavernous hemangioma of the spinal cord and degeneration of the lateral corticospinal tracts in a man with the clinical diagnosis of multiple sclerosis. Clin Neuropathol 4:260–264

    PubMed  CAS  Google Scholar 

  36. Lopate G, Black JT, Grubb RL (1990) Cavernous hemangioma of the spinal cord: report of two unusual cases. Neurology 40:1791–1793

    PubMed  CAS  Google Scholar 

  37. Lunardi P, Acqui M, Ferrante L, Fortuna A (1994) The role of intraoperative ultrasound imaging in the surgical removal of intramedullary cavernous angiomas. Neurosurg 34:520–523

    Article  CAS  Google Scholar 

  38. Marconi F, Parenti G, Giorgetti V, Puglioli M (1995) Spinal cavernous angioma producing subarachnoid hemorrhage. Case report. J Neurosurg Sci 39:75–80

    PubMed  CAS  Google Scholar 

  39. Mastronardi L, Ferrante L, Scarpinati M, Gagliardi FM, Celli P, Fortuna A (1991) Intradural extramedullary cavernous angioma. Case report. Neurosurgery 29:924–926

    Article  PubMed  CAS  Google Scholar 

  40. McCormick WF (1996) The pathology of vascular (“arteriovenous”) malformations. J Neurosurg 24:807–816

    Google Scholar 

  41. McCormick PC, Michelsen WJ, Post KD, Carmel PW, Stein BM (1988) Cavernous malformations of the spinal cord. Neurosurgery 23:459–463

    Article  PubMed  CAS  Google Scholar 

  42. Mehdorn HM, Stolke D (1991) Cervical intramedullary cavernous angioma with MRI-proven haemorrhages. J Neurol 238:420–426

    Article  PubMed  CAS  Google Scholar 

  43. Meyer O, Kohler B (1917) Uber eine auf kongenitaler Basis entstandene cavernomahnliche Bildung des ruckenmarks. Frank Z Pathol 20:37–56

    Google Scholar 

  44. Moriuchi S, Shimizu K, Yamada M, Sakurai M, Mogami H (1989) A case of repeated hematomyelia caused by cavernous hemangioma in the spinal cord. No Shinkei Geka 17:477–479

    PubMed  CAS  Google Scholar 

  45. Nagashima C, Masuda t, Nagashima R, Enomoto K, Watabe T, Morita H, Takahama M (1996) Spinal intramedullary cavernous angioma associated with hematomyelia: case report with sequential MRI follow-up and histological verification of hematoidin deposits. No Shinkei Geka 24:1125–1132

    PubMed  CAS  Google Scholar 

  46. Nagib MG, O’Fallon MT (2002) Intramedullary Cavernous angiomas of the spinal cord in the pediatric age group: a pediatric series. Pediatr Neurosurg 36:57–63

    Article  PubMed  Google Scholar 

  47. Nonogaki Y, Ishii M, Oda N, Nagashima C (1992) Coexistence of intracranial and spinal cavernous angiomas: case report. No Shinkei Geka 20:1277–1281

    PubMed  CAS  Google Scholar 

  48. Odom GL, Woodhall B, Margolis G (1957) Spontaneous hematomyelia and angiomas of the spinal cord. J Neurosurg 14:192–202

    PubMed  CAS  Google Scholar 

  49. Ogilvy CS, Louis DN, Ojemann RG (1992) Intramedullary cavernous angiomas of the spinal cord: clinical presentation, pathological features and surgical managements. Neurosurg 31:219–230

    Article  CAS  Google Scholar 

  50. Padovani R, Acciarri N, Giuliani M, Pantieri R, Foschini MP (1997) Cavernous angiomas of the spinal district. Surgical treatment of 11 patients. Eur Spine J 6:298–303

    Article  PubMed  CAS  Google Scholar 

  51. Padovani R, Tognetti F, Proietti D et al (1982) Extrathecal cavernous hemangioma. Surg Neurol 18:463–465

    Article  PubMed  CAS  Google Scholar 

  52. Pagni CA, Canavero S, Forni M (1990) Report of a cavernoma of the cauda equina and review of the literature. Surg Neurol 33:124–131

    Article  PubMed  CAS  Google Scholar 

  53. Ramos F, Toffol B, De Aesch B, Jan M (1990) Hydrocephalus and cavernoma of the cauda equina. Neurosurgery 27:139–142

    Article  PubMed  Google Scholar 

  54. Richardson RR, Cerullo LJ (1979) Spinal epidural cavernous hemangioma. Surg Neurol 12:266–268

    PubMed  CAS  Google Scholar 

  55. Rigamonti D, Hadley MN, Drayer BP et al (1988) Cerebral cavernous malformations.Incidence and familial occurrence. N Engl J Med 319:343–347

    Article  PubMed  CAS  Google Scholar 

  56. Russell DS, Rubenstein LJ (1989) Pathology of tumours of the nervous system, edn 5. Williams and Wilkins, Baltimore, pp 730–736

    Google Scholar 

  57. Rutka JT, Brant-Zawadsky M, Wilson CB, Rosenblum ML (1988) Familial cavernous malformations. Diagnostic potential of magnetic resonance imaging. Surg Neurol 29:467–474

    Article  PubMed  CAS  Google Scholar 

  58. Sabin HI, Daniel S, Wild AM, Symon L (1989) Cavernous angioma of the thoracic spinal cord with intra- and extramedullary components. Br J Neurosurg 3:123–126

    PubMed  CAS  Google Scholar 

  59. Saito N, Yamakawa K, Sasaki T, Saito I, Takakura K (1989) Intramedullary cavernous angioma with trigeminal neuralgia: a case report and review of the literature. Neurosurgery 25:97–101

    Article  PubMed  CAS  Google Scholar 

  60. Santoro A, Innocenzi G, Bellotti C, Cancrini A, Delfini R, Cantore GP (1998) Total removal of an intramedullary cavernous angioma by transthoracic approach. Case report. Ital J Neurol Sci 19:176–179

    Article  PubMed  CAS  Google Scholar 

  61. Santoro A, Piccirilli M, Bristol R, di Norcia V, Salvati M, Delfini R (2005) Extradural spinal cavernous angiomas: report of seven cases. Neurosurg Rev 28(4):313–319

    Article  PubMed  Google Scholar 

  62. Santoro A, Piccirilli M, Frati A, Salvati M, Innocenzi G, Ricci G, Cantore G (2004) Intramedullary spinal cord cavernous malformation: report of ten new cases. Neurosurg Rev 27(2):93–98

    Article  PubMed  Google Scholar 

  63. Schroder JM, Brunngraber CV (1964) Über ein intramedullares cavernoses Angiom. Acadt Neurochirurg (Wien) 12:632–641

    Article  Google Scholar 

  64. Schultze F (1912) Weiterer Beitrag zur Diagnose und operativen Behandlung von Geschwülsten der Rückenmarkshäute und des Rüückenmarks. Deutsche Medizinische Wochenszeitschrift 38:1676–1679

    Article  Google Scholar 

  65. Scott RM, Barnes P, Kupsky W, Adelman LS (1992) Cavernous angiomas of the central nervous system in children. J Neurosurg 76:38–46

    PubMed  CAS  Google Scholar 

  66. Spetzger U, Gilsbach JM, Bertalanffy H (1995) Cavernous angiomas of the spinal cord. Clinical presentation, surgical strategy and postoperative results. Acta Neurochir 134:200–206

    Article  CAS  Google Scholar 

  67. Stone JL, Lichtor T, Ruge JR (1995) Cavernous angioma of the upper cervical spinal cord. A case report. Spine 15:1205–1207

    Article  Google Scholar 

  68. Szojchet A (1968) Metameric spinal cord and skin hemangiomas. Case report. J Neurosurg 29:199–201

    PubMed  CAS  Google Scholar 

  69. Tu YK, Liu HM, Chen SI, Lin SM (1999) Intramedullary cavernous haemangiomas: clinical features, imaging diagnosis, surgical resection and outcome. J Clin Neurosci 6:212–216

    Article  PubMed  Google Scholar 

  70. Turjman E, Joly D, Monnet O, Faure C, Doyon D, Froment JC (1995) MRI of intramedullary cavernous haemangiomas. Neuroradiology 37:297–302

    Article  PubMed  CAS  Google Scholar 

  71. Tyndel FJ, Bilbao JM, Hudson AR, Colapinto EU (1985) Hemangioma calcificans of the spinal cord. Can J Neurol Sci 12:321–322

    PubMed  CAS  Google Scholar 

  72. Ueda S, Saito A, Inomori S et al (1987) Cavernous angioma of the cauda equina producing subarachnoid hemorrhage. Case report. J Neurosurg 66:134–136

    PubMed  CAS  Google Scholar 

  73. Van Reeth PC (1952) Contributien a l’etude de l’angiomatose medullaire. Acta Neurol Psych Bel 52:249–270

    Google Scholar 

  74. Vaquero J, Martinez R, Martinez P (1988) Cavernomas of the spinal cord: report of two cases. Neurosurgery 22:143–144

    Article  PubMed  CAS  Google Scholar 

  75. Villani RM, Arienta C, Caroli M (1989) Cavernous angiomas of the central nervous system. J Neurosurg Sci 33:229–252

    Google Scholar 

  76. Vraa-Jensen G (1988) Angioma of the spinal cord. Acta Psychiat Neurol Scand 24:709–721

    Article  Google Scholar 

  77. Vishteh GA, Sankhala S, Anson JA, Zabramsky JM, Spetzler RF (1997) Surgical resection of intramedullary spinal cord cavernous malformations: delayed complications, long-term outcomes and association with cryptic venous malformations. Neurosurgery 41:1094–1101

    Article  PubMed  CAS  Google Scholar 

  78. Visheth GA, Spetzler RF (1999) Radical excision of intramedullary cavernous angiomas. Neurosurgery 44:428

    Article  Google Scholar 

  79. Visheteh AG, Zabramski JM, Spetzler RF (1999) Patients with spinal cord cavernous malformations are at an increased risk for multiple neuraxis cavernous malformations. Neurosurgery 45:30–33

    Article  Google Scholar 

  80. Wood MW, White RJ, Kernohan JW (1957) Cavernous hemangiomatosis involving the brain, spinal cord, heart, skin and kidney. Report of a case. Mayo Clin Proc 32:249–254

    CAS  Google Scholar 

  81. Yasui T, Komiyama M, Iwai Y, Yamanaka K, Matsusaka Y, Morikawa T, Ishiguro T (2005) A brainstem cavernoma demonstrating a dramatic, spontaneous decrease in size during follow-up: case report and review of the literature. Surg Neurol 63:170–173

    Article  PubMed  Google Scholar 

  82. Yong-Kwang T, Hon Man L, Siu-Jau C (1999) Intramedullary cavernous hemangiomas: clinical features, imaging diagnosis, surgical resectiona nd outcome. J Clin Neurosci 6:212–216

    Article  Google Scholar 

  83. Zabramski JM, Wascher TM, Spetzler RF, Johnson B, Golfinos J, Drayer BP, Brown B, Rigamonti D, Brown G (1994) The natural history of familial cavernous malformations. Results of an ongoing study. J Neurosurg 80:422–432

    Article  PubMed  CAS  Google Scholar 

  84. Zentner J, Hassler W, Gawehn J, Schroth G (1989) Intramedullary cavernous angiomas. Surg Neurol 31:64–68

    Article  PubMed  CAS  Google Scholar 

  85. Zevgaridis D, Medele RJ, Hambergr C, Steiger HJ, Reulen HJ (1999) Cavernous haemangiomas of the spinal cord. A review of 117 cases. Acta Neurochir 141:237–245

    Article  CAS  Google Scholar 

  86. Zimmerman RS, Spetzler RF, Lee KS, Zabramski JM, Hargraves RW (1991) Cavernous malformations of the brain stem. J Neurosurg 75:32–39

    PubMed  CAS  Google Scholar 

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Authors and Affiliations

  1. Department of Neurological Sciences-Neurosurgery, University of Rome “La Sapienza”, Neurochirurgia1, Viale del Policlinico, 155, 00185, Rome, Italy

    Antonio Santoro, Manolo Piccirilli, Giacoma Maria Floriana Brunetto & Roberto Delfini

  2. Department of Neurosurgery, INM, Neuromed, Pozzilli, Isernia, Italy

    Giampaolo Cantore

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  1. Antonio Santoro
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  2. Manolo Piccirilli
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  3. Giacoma Maria Floriana Brunetto
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  4. Roberto Delfini
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  5. Giampaolo Cantore
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Correspondence to Antonio Santoro.

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Santoro, A., Piccirilli, M., Brunetto, G.M.F. et al. Intramedullary cavernous angioma of the spinal cord in a pediatric patient, with multiple cavernomas, familial occurrence and partial spontaneous regression: case report and review of the literature. Childs Nerv Syst 23, 1319–1326 (2007). https://doi.org/10.1007/s00381-007-0395-5

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