Skip to main content

Advertisement

SpringerLink
Log in

Spontaneous bleeding into a suprasellar cavernous angioma of a neonate: case report and literature review

  • Case Report
  • Published:
Child's Nervous System Aims and scope Submit manuscript

Abstract

Introduction

Cavernous angiomas (CA) are congenital intraparenchymal vascular malformations that contain sinusoidal spaces lined by a single-layer endothelium, separated by collagenous stroma with no intervening brain parenchyma. Despite the congenital origin of CA, they rarely present in the neonatal and prenatal period. In this paper, we present a case report of a neonatal suprasellar CA that presented with a bleed. We also present a literature review focusing on specific features of intracranial CA in the neonatal and fetal age groups.

Case report

A 27-day-old neonate presented with a left eye ptosis for 2 days, followed by a generalized seizure. A head computed tomography revealed a suprasellar hematoma with intraventricular and subarachnoid extension. Brain magnetic resonance imaging revealed hemorrhages of various ages. Magnetic resonance angiography did not reveal any vascular malformation. Surgical exploration of the suprasellar mass revealed a capsulated dense hematoma. Postoperatively, the neonate was weaned of artificial ventilation over a protracted period and remained hemiparetic with signs of third nerve palsy. Pathology revealed a CA. CA presenting as a suprasellar bleed with subarachnoid and intraventricular extension is very rare especially among neonates. To the best of our knowledge, 20 cases of CA have been reported in the neonatal and fetal period in the English literature.

Summary

Neonatal CA in general and suprasellar location in particular are extremely rare lesions. Neonatal/fetal CA seems to present more aggressively and have a worse prognosis compared to those presenting at a later age.

This is a preview of subscription content, log in via an institution to check access.

Access this article

Log in via an institution

Price excludes VAT (USA)
Tax calculation will be finalised during checkout.

Instant access to the full article PDF.

Institutional subscriptions

Fig. 1
Fig. 2
Fig. 3
Fig. 4

References

  1. Acciarri N, Galassi E, Giulioni M, Pozzati E, Grasso V, Palandri G, Badaloni F, Zacchelli M, Calbucci F (2009) Cavernous malformations of the central nervous system in the pediatric age group. Pediatr Neurosurg 45:81–104

    Article  PubMed  Google Scholar 

  2. Aiba T, Tanaka R, Koike T, Kameyama S, Takeda N, Komata T (1995) Natural history of intracranial cavernous malformations. J Neurosurg 83:56–59

    Article  CAS  PubMed  Google Scholar 

  3. Al-Kaabi A, Yanofsky R, Bunge M, Hyman J, Rafay MF (2009) Diffuse hemangiomatosis with predominant central nervous system involvement. Pediatr Neurol 40:54–57

    Article  PubMed  Google Scholar 

  4. Arnstein LH, Boldrey E, Naffziger HC (1951) A case report and survey of brain tumors during the neonatal period. J Neurosurg 8:315–319

    Article  CAS  PubMed  Google Scholar 

  5. Azam M, O'Donovan DJ (2009) Intracranial cavernous hemangioma and seizures in a newborn infant. J Pediatr 155:298

    Article  PubMed  Google Scholar 

  6. Bergeson PS, Rekate HL, Tack ED (1992) Cerebral cavernous angiomas in the newborn. Clin Pediatr (Phila) 31:435–437

    Article  CAS  Google Scholar 

  7. Bristot R, Santoro A, Fantozzi L, Delfini R (1997) Cavernoma of the cavernous sinus: case report. Surg Neurol 48:160–163

    Article  CAS  PubMed  Google Scholar 

  8. Bruni P, Massari A, Greco R, Hernandez R, Oddi G, Chiappetta F (1994) Subarachnoid hemorrhage from cavernous angioma of the cauda equina: case report. Surg Neurol 41:226–229

    Article  CAS  PubMed  Google Scholar 

  9. Buonaguidi R, Canapicci R, Mimassi N, Ferdeghini M (1984) Intrasellar cavernous hemangioma. Neurosurgery 14:732–734

    Article  CAS  PubMed  Google Scholar 

  10. Burn S, Gunny R, Phipps K, Gaze M, Hayward R (2007) Incidence of cavernoma development in children after radiotherapy for brain tumors. J Neurosurg 106:379–383

    PubMed  Google Scholar 

  11. Canevini P, Farneti A, Flauto U (1963) Report of a case of cavernous hemangioma of the dura mater in a 2-day old newborn. Folia Hered Pathol (Milano) 12:163–166

    CAS  Google Scholar 

  12. Denier C, Labauge P, Bergametti F, Marchelli F, Riant F, Arnoult M et al (2006) Genotype-phenotype correlations in cerebral cavernous malformations patients. Ann Neurol 60:550–556

    Article  PubMed  Google Scholar 

  13. Di Rocco C, Iannelli A, Tamburrini G (1996) Cavernomas of the central nervous system in children. A report of 22 cases. Acta Neurochir (Wien) 138:1267–1274, discussion 1273–1264

    Article  Google Scholar 

  14. Escott EJ, Rubinstein D, Cajade-Law AG, Sze CI (2001) Suprasellar cavernous malformation presenting with extensive subarachnoid hemorrhage. Neuroradiology 43:313–316

    Article  CAS  PubMed  Google Scholar 

  15. Hashimoto H, Sakaki T, Ishida Y, Shimokawara T (1997) Fetal cavernous angioma—case report. Neurol Med Chir (Tokyo) 37:346–349

    Article  CAS  Google Scholar 

  16. Hayashi T, Harada K, Honda E, Utsunomiya H, Hashimoto T (1987) Rare neonatal intracerebral hemorrhage. Two cases in full-term infants. Childs Nerv Syst 3:161–164

    Article  CAS  PubMed  Google Scholar 

  17. Hayashi S, Kondoh T, Morishita A, Sasayama T, Sanabria EA, Kohmura E (2004) Congenital cavernous angioma exhibits a progressive decrease in size after birth. Childs Nerv Syst 20:199–203

    Article  PubMed  Google Scholar 

  18. Henrich W, Stupin JH, Buhling KJ, Buhrer C, Bassir C, Dudenhausen JW (2002) Prenatal sonographic findings of thalamic cavernous angioma. Ultrasound Obstet Gynecol 19:518–522

    Article  CAS  PubMed  Google Scholar 

  19. Holden KR, Alexander F (1970) Diffuse neonatal hemangiomatosis. Pediatrics 46:411–421

    CAS  PubMed  Google Scholar 

  20. Honda M, Toda K, Baba H, Yonekura M (2003) Congenital cavernous angioma of the temporal bone: case report. Surg Neurol 59:120–123, discussion 123

    Article  PubMed  Google Scholar 

  21. Huang YC, Tseng CK, Chang CN, Wei KC, Liao CC, Hsu PW (2006) LINAC radiosurgery for intracranial cavernous malformation: 10-year experience. Clin Neurol Neurosurg 108:750–756

    Article  PubMed  Google Scholar 

  22. Hwang JF, Yau CW, Huang JK, Tsai CY (1993) Apoplectic optochiasmal syndrome due to intrinsic cavernous hemangioma. Case report. J Clin Neuroophthalmol 13:232–236

    CAS  PubMed  Google Scholar 

  23. Iwasa H, Indei I, Sato F (1983) Intraventricular cavernous hemangioma. Case report. J Neurosurg 59:153–157

    Article  CAS  PubMed  Google Scholar 

  24. Jooma R, Kendall BE, Hayward RD (1984) Intracranial tumors in neonates: a report of seventeen cases. Surg Neurol 21:165–170

    Article  CAS  PubMed  Google Scholar 

  25. Kan P, Tubay M, Osborn A, Blaser S, Couldwell WT (2008) Radiographic features of tumefactive giant cavernous angiomas. Acta Neurochir (Wien) 150:49–55, discussion 55

    Article  CAS  Google Scholar 

  26. Karmazyn B, Michovitz S, Sirota L, Drozd T, Horev G (2001) Intracranial cavernous hemangioma in a neonate. Pediatr Radiol 31:610–612

    Article  CAS  PubMed  Google Scholar 

  27. Kilic T, Pamir MN, Kullu S, Eren F, Ozek MM, Black PM (2000) Expression of structural proteins and angiogenic factors in cerebrovascular anomalies. Neurosurgery 46:1179–1191, discussion 1191–1172

    Article  CAS  PubMed  Google Scholar 

  28. Kim DG, Choe WJ, Paek SH, Chung HT, Kim IH, Han DH (2002) Radiosurgery of intracranial cavernous malformations. Acta Neurochir (Wien) 144:869–878, discussion 878

    Article  CAS  Google Scholar 

  29. Kojima M, Mabuchi N, Tsuda E, Nagasawa S (1994) The efficacy of MR angiography in detecting small asymptomatic cerebral aneurysms in clinical examination of the brain. Surg Cerebral Stroke 22:181–186

    Google Scholar 

  30. Kon T, Mori H, Hasegawa K, Nishiyama K, Tanaka R, Takahashi H (2007) Neonatal cavernous angioma located in the basal ganglia with profuse intraoperative bleeding. Childs Nerv Syst 23:449–453

    Article  CAS  PubMed  Google Scholar 

  31. Kondziolka D, Lunsford LD, Kestle JR (1995) The natural history of cerebral cavernous malformations. J Neurosurg 83:820–824

    Article  CAS  PubMed  Google Scholar 

  32. Leung KY, Tang MH, Lam TP, Fan YW, Shek TW, Wong KY et al (2004) Prenatal diagnosis of a cavernous angioma associated with intracranial hemorrhage: report of one case and review of the literature. Ultrasound Obstet Gynecol 24:800–803

    Article  CAS  PubMed  Google Scholar 

  33. Liu AL, Wang CC, Dai K (2005) Gamma knife radiosurgery for cavernous malformations. Zhongguo Yi Xue Ke Xue Yuan Xue Bao 27:18–21

    Article  PubMed  Google Scholar 

  34. Lombardi D, Giovanelli M, de Tribolet N (1994) Sellar and parasellar extra-axial cavernous hemangiomas. Acta Neurochir (Wien) 130:47–54

    Article  CAS  Google Scholar 

  35. Lombardi D, Scheithauer BW, Villani RM, Giovanelli M, de Tribolet N (1996) Cavernous haemangioma of the pineal region. Acta Neurochir (Wien) 138:678–683

    Article  CAS  Google Scholar 

  36. Martinez-Lage JF, de la Fuente I, de San R, Pedro J, Fuster JL, Perez-Espejo MA, Herrero MT (2008) Cavernomas in children with brain tumors: a late complication of radiotherapy. Neurocirugia (Astur) 19:50–54

    CAS  Google Scholar 

  37. Maruishi M, Shima T, Okada Y, Nishida M, Yamane K, Okita S (1994) Cavernous sinus cavernoma treated with radiation therapy—case report. Neurol Med Chir (Tokyo) 34:773–777

    Article  CAS  Google Scholar 

  38. Momoshima S, Shiga H, Yuasa Y, Higuchi N, Kawase T, Toya S (1991) MR findings in extracerebral cavernous angiomas of the middle cranial fossa: report of two cases and review of the literature. AJNR Am J Neuroradiol 12:756–760

    CAS  PubMed  Google Scholar 

  39. Moritake K, Handa H, Nozaki K, Tomiwa K (1985) Tentorial cavernous angioma with calcification in a neonate. Neurosurgery 16:207–211

    Article  CAS  PubMed  Google Scholar 

  40. Mottolese C, Hermier M, Stan H, Jouvet A, Saint-Pierre G, Froment JC, Bret P, Lapras C (2001) Central nervous system cavernomas in the pediatric age group. Neurosurg Rev 24:55–71, discussion 72–53

    Article  CAS  PubMed  Google Scholar 

  41. Ng BH, Mulyadi E, Pereira JK, Ghedia S, Pinner J, Mowat D et al (2006) Familial cerebral cavernous haemangioma diagnosed in an infant with a rapidly growing cerebral lesion. Australas Radiol 50:583–590

    Article  CAS  PubMed  Google Scholar 

  42. Nimjee SM, Powers CJ, Bulsara KR (2006) Review of the literature on de novo formation of cavernous malformations of the central nervous system after radiation therapy. Neurosurg Focus 21:e4

    Article  PubMed  Google Scholar 

  43. Notelet L, Houtteville JP, Khoury S, Lechevalier B, Chapon F (1997) Proliferating cell nuclear antigen (PCNA) in cerebral cavernomas: an immunocytochemical study of 42 cases. Surg Neurol 47:364–370

    Article  CAS  PubMed  Google Scholar 

  44. Porter RW, Detwiler PW, Spetzler RF, Lawton MT, Baskin JJ, Derksen PT et al (1999) Cavernous malformations of the brainstem: experience with 100 patients. J Neurosurg 90:50–58

    Article  CAS  PubMed  Google Scholar 

  45. Pozzati E, Acciarri N, Tognetti F, Marliani F, Giangaspero F (1996) Growth, subsequent bleeding, and de novo appearance of cerebral cavernous angiomas. Neurosurgery 38:662–669, discussion 669–670

    Article  CAS  PubMed  Google Scholar 

  46. Prensky AL, Gado M (1973) Angiographic resolution of a neonatal intracranial cavernous hemangioma coincident with steroid therapy. Case report. J Neurosurg 39:99–103

    Article  CAS  PubMed  Google Scholar 

  47. Ravenel SD (1979) Posterior fossa hemorrhage in the term newborn: report of two cases. Pediatrics 64:39–42

    CAS  PubMed  Google Scholar 

  48. Rigamonti D, Drayer BP, Johnson PC, Hadley MN, Zabramski J, Spetzler RF (1987) The MRI appearance of cavernous malformations (angiomas). J Neurosurg 67:518–524

    Article  CAS  PubMed  Google Scholar 

  49. Rigamonti D, Johnson PC, Spetzler RF, Hadley MN, Drayer BP (1991) Cavernous malformations and capillary telangiectasia: a spectrum within a single pathological entity. Neurosurgery 28:60–64

    Article  CAS  PubMed  Google Scholar 

  50. Rigamonti D, Pappas CT, Spetzler RF, Johnson PC (1990) Extracerebral cavernous angiomas of the middle fossa. Neurosurgery 27:306–310

    Article  CAS  PubMed  Google Scholar 

  51. Robinson JR, Awad IA, Little JR (1991) Natural history of the cavernous angioma. J Neurosurg 75:709–714

    Article  CAS  PubMed  Google Scholar 

  52. Roda JM, Carceller F, Perez-Higueras A, Morales C (1993) Encapsulated intracerebral hematomas: a defined entity. Case report. J Neurosurg 78:829–833

    Article  CAS  PubMed  Google Scholar 

  53. Rothbart D, Awad IA, Lee J, Kim J, Harbaugh R, Criscuolo GR (1996) Expression of angiogenic factors and structural proteins in central nervous system vascular malformations. Neurosurgery 38:915–924, discussion 924–915

    Article  CAS  PubMed  Google Scholar 

  54. Russell DS, LJ R (eds) (1977) Pathology of tumors of the nervous system. Williams & Wilkins, Baltimore

    Google Scholar 

  55. Sakai N, Yamada H, Nishimura Y, Shirakami S, Futamura A, Andoh T (1992) Intracranial cavernous angioma in the 1st year of life and a review of the literature. Childs Nerv Syst 8:49–52

    Article  CAS  PubMed  Google Scholar 

  56. Saldana CJ, Zimman H, Alonso P, Mata PR (1991) Neonatal cavernous hemangioma of the dura mater: case report. Neurosurgery 29:602–605

    Article  CAS  PubMed  Google Scholar 

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

    Article  CAS  PubMed  Google Scholar 

  58. Stephenson G, Afshar F (1994) Suprasellar cavernous angioma causing pituitary and hypothalamic dysfunction. Br J Neurosurg 8:225–227

    Article  CAS  PubMed  Google Scholar 

  59. Sure U, Butz N, Schlegel J, Siegel AM, Wakat JP, Mennel HD et al (2001) Endothelial proliferation, neoangiogenesis, and potential de novo generation of cerebrovascular malformations. J Neurosurg 94:972–977

    Article  CAS  PubMed  Google Scholar 

  60. Suzuki Y, Shibuya M, Baskaya MK, Takakura S, Yamamoto M, Saito K et al (1996) Extracerebral cavernous angiomas of the cavernous sinus in the middle fossa. Surg Neurol 45:123–132

    Article  CAS  PubMed  Google Scholar 

  61. Takeshita M, Kagawa M, Izawa M, Kitamura K (1986) Hemorrhagic stroke in infancy, childhood and adolescence. Surg Neurol 26:496–500

    Article  CAS  PubMed  Google Scholar 

  62. Tekkok IH, Ventureyra EC (1997) Spontaneous intracranial hemorrhage of structural origin during the first year of life. Childs Nerv Syst 13:154–165

    Article  CAS  PubMed  Google Scholar 

  63. Tirakotai W, Fremann S, Soerensen N, Roggendorf W, Siegel AM, Mennel HD, Zhu Y, Bertalanffy H, Sure U (2006) Biological activity of paediatric cerebral cavernomas: an immunohistochemical study of 28 patients. Childs Nerv Syst 22:685–691

    Article  PubMed  Google Scholar 

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

    Article  CAS  PubMed  Google Scholar 

  65. Vanlieferinghen P, Lemery D, Sevely A, Dechelotte P, Bloom MC, Raynaud EJ (1993) Prenatal diagnosis of congenital cerebral tumors. Apropos of 3 cases. Arch Fr Pediatr 50:39–41

    CAS  PubMed  Google Scholar 

  66. Yamamoto M, Fukushima T, Ikeda K, Nagasaka S, Sakamoto S, Oka K et al (1993) Intracranial cavernous angioma manifesting as subarachnoid hemorrhage—case report. Neurol Med Chir (Tokyo) 33:706–709

    Article  CAS  Google Scholar 

  67. Zentner J, Grodd W, Hassler W (1989) Cavernous angioma of the optic tract. J Neurol 236:117–119

    Article  CAS  PubMed  Google Scholar 

  68. Zhang N, Pan L, Wang BJ, Wang EM, Dai JZ, Cai PW (2000) Gamma knife radiosurgery for cavernous hemangiomas. J Neurosurg 93(Suppl 3):74–77

    PubMed  Google Scholar 

Download references

Author information

Authors and Affiliations

  1. Department of Pediatric Neurosurgery, Dana Children’s Hospital, Tel Aviv University, Tel Aviv, Israel

    Suhas Udayakumaran, Dimitrios Paraskevopoulos, Jonathan Roth & Shlomi Constantini

  2. Neuropathology Unit, Department of Pathology, Tel Aviv Sourasky Medical Center, Tel Aviv University, 6, Weizman St., 64239, Tel Aviv, Israel

    Emanuela Cagnano

Authors
  1. Suhas Udayakumaran
    View author publications

    You can also search for this author in PubMed Google Scholar

  2. Dimitrios Paraskevopoulos
    View author publications

    You can also search for this author in PubMed Google Scholar

  3. Emanuela Cagnano
    View author publications

    You can also search for this author in PubMed Google Scholar

  4. Jonathan Roth
    View author publications

    You can also search for this author in PubMed Google Scholar

  5. Shlomi Constantini
    View author publications

    You can also search for this author in PubMed Google Scholar

Corresponding author

Correspondence to Shlomi Constantini.

Rights and permissions

Reprints and permissions

About this article

Cite this article

Udayakumaran, S., Paraskevopoulos, D., Cagnano, E. et al. Spontaneous bleeding into a suprasellar cavernous angioma of a neonate: case report and literature review. Childs Nerv Syst 27, 303–311 (2011). https://doi.org/10.1007/s00381-010-1161-7

Download citation

  • Received:

  • Accepted:

  • Published:

  • Issue Date:

  • DOI: https://doi.org/10.1007/s00381-010-1161-7

Keywords

Search

Navigation