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Child's Nervous System
Published in: Child's Nervous System 11/2007

01-11-2007 | Case Report

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

Authors: Antonio Santoro, Manolo Piccirilli, Giacoma Maria Floriana Brunetto, Roberto Delfini, Giampaolo Cantore

Published in: Child's Nervous System | Issue 11/2007

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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.
Literature
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):563–567PubMed
2.
Anson JA, Spetzler RF (1993) Surgical resection of intramedullary spinal cord cavernous malformations. J Neurosurg 78:446–451PubMed
3.
Bakir A, Savas A, Yilmaz E, Savaş B, Erden E, Cağlar S, Sener Ö (2006) Spinal intradural–intramedullary cavernous malformation. Pediatr Neurosurg 42:35–37PubMedCrossRef
4.
Balousek P, Ammirati M (1996) Exophytic cavernous malformations of the cervical spinal cord. Acta Neurochir (Wien) 138:890–892CrossRef
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 72:403–407PubMed
6.
Bergstrand A, Hook O, Lindvall H (1964) Vascular malformations of the spinal cord. Acta Neurol Scand 40:169–183PubMedCrossRef
7.
Bicknell JM, Carlow TJ, Kornfeld M, Stovring J, Turner P (1978) Familial cavernous angiomas. Arch Neurol 35:746–749PubMed
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 34:110–111PubMedCrossRef
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 43:448–452PubMedCrossRef
10.
Cantore GP, Missori P, Santoro A (1999) Cavernous angiomas of the brain stem. Intra-axial anatomical pitfalls and surgical strategies. Surg Neurol 52:84–94PubMedCrossRef
11.
Cardon T, Wryemblewsky P, Lesoin F, Brunaud V, Destee A (1992) Angiome caverneux de la moelle cervicale. Rev Neurol 18:152–154
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 93:981–986PubMed
13.
Cosgrove GR, Bertrand G, Fontaine S, Robitaille Y, Melancon D (1988) Cavernous angiomas of the spinal cord. J Neurosurg 68:31–36PubMed
14.
Cristante L, Hermann HD (1998) Radical excision of intramedullary cavernous angiomas. Neurosurgery 43:424–431PubMedCrossRef
15.
Deutsch H, Jallo GI, Faktorovich A, Epstein F (2000) Spinal intramedullary cavernoma: clinical presentation and surgical outcome. J Neurosurg 1 93:65–70
16.
Deutsch H, Shrivistava R, Epstein F, Jallo GI (2001) Pediatric intramedullary spinal cavernous malformations. Spine 26(18):427–431CrossRef
17.
Fazi S, Menei P, Mercier P, Dubas F, Guy G (1992) Cavernomas of the spinal cord: report of two patients. Br J Neurosurg 6:149–152PubMed
18.
Feider HK, Yuille D (1991) An epidural cavernous hemangioma of the spine. AJNR 12:243–244PubMed
19.
Fontaine D, Lot D, George B (1999) Intramedullary cavernous angioma. Resection by oblique corpectomy. Surg Neurol 51:435–442PubMedCrossRef
20.
Furuya K, Sasaki T, Susuki I, Kim P, Saito N, Kirino T (1996) Intramedullary angiographically occult vascular malformations of the spinal cord. Neurosurgery 39:1123–1132PubMedCrossRef
21.
Gagel O, Meszaros A (1947) Zur Frage der myelopathia necroticans. Arch Psych Neurol 179:423–429CrossRef
22.
Gao PY (1989) Magnetic resonance imaging of occult intracranial malformations. Chung Hua I Hsueh Tsa Chih 69:375–377PubMed
23.
Goran A, Carlson DJ, Fischer RG (1964) Successful treatment of intramedullary angioma of the cord. J Neurosurg 21:311–314PubMed
24.
Gordon CR, Crockard HA, Symon L (1995) Surgical management of spinal cord cavernoma. Br J Neurosurg 9:459–464PubMedCrossRef
25.
Guthkelch AN (1948) Haemangiomas involving the spinal epidural space. J Neurol Neurosurg Psychiatry 11:199–210PubMed
26.
Hadlich R (1903) Ein Fall von Tumor cavernous des Rückenmarks mit besonderer Berücksichtigung der neueren Theorien über die Gene des Cavernoms. Virchows Arch 172:429–441CrossRef
27.
Harrison MJ, Eisenberg MB, Ullman JS, Oppenheim JS, Camins MB, Post KD (1995) Symptomatic cavernous malformations affecting the spine and spinal cord. Neurosurgery 37:195–205PubMedCrossRef
28.
Heimberger K, Schnabert G, Koos W et al (1982) Spinal cavernous haemangioma (intradural-extramedullary) underlying repeated subarachnoid haemorrhage. J Neurol 226:289–293PubMedCrossRef
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
30.
Hillman J, Bynke O (1991) Solitary extradural cavernous hemangiomas in the spinal canal. Report of five cases. Surg Neurol 36:19–24PubMedCrossRef
31.
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
33.
Kitahara T, Miyasaka Y, Ohwada T, Yada K, Mera H (1982) An operated case of cervical spontaneous hematomyelia. Neurol Surg 10:675–679
34.
Lee KS, Spetzler RF (1990) Spinal cord cavernous malformations in a patient with familial intracranial cavernous malformations. Neurosurgery 26:877–880PubMedCrossRef
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 4:260–264PubMed
36.
Lopate G, Black JT, Grubb RL (1990) Cavernous hemangioma of the spinal cord: report of two unusual cases. Neurology 40:1791–1793PubMed
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 34:520–523CrossRef
38.
Marconi F, Parenti G, Giorgetti V, Puglioli M (1995) Spinal cavernous angioma producing subarachnoid hemorrhage. Case report. J Neurosurg Sci 39:75–80PubMed
39.
Mastronardi L, Ferrante L, Scarpinati M, Gagliardi FM, Celli P, Fortuna A (1991) Intradural extramedullary cavernous angioma. Case report. Neurosurgery 29:924–926PubMedCrossRef
40.
McCormick WF (1996) The pathology of vascular (“arteriovenous”) malformations. J Neurosurg 24:807–816
41.
McCormick PC, Michelsen WJ, Post KD, Carmel PW, Stein BM (1988) Cavernous malformations of the spinal cord. Neurosurgery 23:459–463PubMedCrossRef
42.
Mehdorn HM, Stolke D (1991) Cervical intramedullary cavernous angioma with MRI-proven haemorrhages. J Neurol 238:420–426PubMedCrossRef
43.
Meyer O, Kohler B (1917) Uber eine auf kongenitaler Basis entstandene cavernomahnliche Bildung des ruckenmarks. Frank Z Pathol 20:37–56
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 17:477–479PubMed
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 24:1125–1132PubMed
46.
Nagib MG, O’Fallon MT (2002) Intramedullary Cavernous angiomas of the spinal cord in the pediatric age group: a pediatric series. Pediatr Neurosurg 36:57–63PubMedCrossRef
47.
Nonogaki Y, Ishii M, Oda N, Nagashima C (1992) Coexistence of intracranial and spinal cavernous angiomas: case report. No Shinkei Geka 20:1277–1281PubMed
48.
Odom GL, Woodhall B, Margolis G (1957) Spontaneous hematomyelia and angiomas of the spinal cord. J Neurosurg 14:192–202PubMed
49.
Ogilvy CS, Louis DN, Ojemann RG (1992) Intramedullary cavernous angiomas of the spinal cord: clinical presentation, pathological features and surgical managements. Neurosurg 31:219–230CrossRef
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 6:298–303PubMedCrossRef
51.
Padovani R, Tognetti F, Proietti D et al (1982) Extrathecal cavernous hemangioma. Surg Neurol 18:463–465PubMedCrossRef
52.
Pagni CA, Canavero S, Forni M (1990) Report of a cavernoma of the cauda equina and review of the literature. Surg Neurol 33:124–131PubMedCrossRef
53.
Ramos F, Toffol B, De Aesch B, Jan M (1990) Hydrocephalus and cavernoma of the cauda equina. Neurosurgery 27:139–142PubMedCrossRef
54.
Richardson RR, Cerullo LJ (1979) Spinal epidural cavernous hemangioma. Surg Neurol 12:266–268PubMed
55.
Rigamonti D, Hadley MN, Drayer BP et al (1988) Cerebral cavernous malformations.Incidence and familial occurrence. N Engl J Med 319:343–347PubMedCrossRef
56.
Russell DS, Rubenstein LJ (1989) Pathology of tumours of the nervous system, edn 5. Williams and Wilkins, Baltimore, pp 730–736
57.
Rutka JT, Brant-Zawadsky M, Wilson CB, Rosenblum ML (1988) Familial cavernous malformations. Diagnostic potential of magnetic resonance imaging. Surg Neurol 29:467–474PubMedCrossRef
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 3:123–126PubMed
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 25:97–101PubMedCrossRef
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 19:176–179PubMedCrossRef
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):313–319PubMedCrossRef
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):93–98PubMedCrossRef
63.
Schroder JM, Brunngraber CV (1964) Über ein intramedullares cavernoses Angiom. Acadt Neurochirurg (Wien) 12:632–641CrossRef
64.
Schultze F (1912) Weiterer Beitrag zur Diagnose und operativen Behandlung von Geschwülsten der Rückenmarkshäute und des Rüückenmarks. Deutsche Medizinische Wochenszeitschrift 38:1676–1679CrossRef
65.
Scott RM, Barnes P, Kupsky W, Adelman LS (1992) Cavernous angiomas of the central nervous system in children. J Neurosurg 76:38–46PubMed
66.
Spetzger U, Gilsbach JM, Bertalanffy H (1995) Cavernous angiomas of the spinal cord. Clinical presentation, surgical strategy and postoperative results. Acta Neurochir 134:200–206CrossRef
67.
Stone JL, Lichtor T, Ruge JR (1995) Cavernous angioma of the upper cervical spinal cord. A case report. Spine 15:1205–1207CrossRef
68.
Szojchet A (1968) Metameric spinal cord and skin hemangiomas. Case report. J Neurosurg 29:199–201PubMed
69.
Tu YK, Liu HM, Chen SI, Lin SM (1999) Intramedullary cavernous haemangiomas: clinical features, imaging diagnosis, surgical resection and outcome. J Clin Neurosci 6:212–216PubMedCrossRef
70.
Turjman E, Joly D, Monnet O, Faure C, Doyon D, Froment JC (1995) MRI of intramedullary cavernous haemangiomas. Neuroradiology 37:297–302PubMedCrossRef
71.
Tyndel FJ, Bilbao JM, Hudson AR, Colapinto EU (1985) Hemangioma calcificans of the spinal cord. Can J Neurol Sci 12:321–322PubMed
72.
Ueda S, Saito A, Inomori S et al (1987) Cavernous angioma of the cauda equina producing subarachnoid hemorrhage. Case report. J Neurosurg 66:134–136PubMed
73.
Van Reeth PC (1952) Contributien a l’etude de l’angiomatose medullaire. Acta Neurol Psych Bel 52:249–270
74.
Vaquero J, Martinez R, Martinez P (1988) Cavernomas of the spinal cord: report of two cases. Neurosurgery 22:143–144PubMedCrossRef
75.
Villani RM, Arienta C, Caroli M (1989) Cavernous angiomas of the central nervous system. J Neurosurg Sci 33:229–252
76.
Vraa-Jensen G (1988) Angioma of the spinal cord. Acta Psychiat Neurol Scand 24:709–721CrossRef
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 41:1094–1101PubMedCrossRef
78.
Visheth GA, Spetzler RF (1999) Radical excision of intramedullary cavernous angiomas. Neurosurgery 44:428CrossRef
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 45:30–33CrossRef
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
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 63:170–173PubMedCrossRef
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 6:212–216CrossRef
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 80:422–432PubMedCrossRef
84.
Zentner J, Hassler W, Gawehn J, Schroth G (1989) Intramedullary cavernous angiomas. Surg Neurol 31:64–68PubMedCrossRef
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 141:237–245CrossRef
86.
Zimmerman RS, Spetzler RF, Lee KS, Zabramski JM, Hargraves RW (1991) Cavernous malformations of the brain stem. J Neurosurg 75:32–39PubMed
Metadata
Title
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
Authors
Antonio Santoro
Manolo Piccirilli
Giacoma Maria Floriana Brunetto
Roberto Delfini
Giampaolo Cantore
Publication date
01-11-2007
Publisher
Springer-Verlag
Published in
Child's Nervous System / Issue 11/2007
Print ISSN: 0256-7040
Electronic ISSN: 1433-0350
DOI
https://doi.org/10.1007/s00381-007-0395-5

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