Intraaxial and Extraaxial Cavernous Malformation with Venous Linkage: Immune Cellular Inflammation Associated with Aggressiveness

World Neurosurg. 2019 Nov:131:87-89. doi: 10.1016/j.wneu.2019.07.145. Epub 2019 Jul 26.

Abstract

Background: Intraorbital and intracerebral cavernous malformation (CM) lesions are considered independent entities. Purely cerebral CMs have variable biology with recent evidence depicting inflammation as an important player and a risk factor for aggressiveness. We describe a case of concomitant left intraaxial and extraaxial CMs, linked by the ipsilateral basal vein, where the extraaxial component has developed an aggressive behavior.

Case description: A 35-year-old female patient presented with a rapid and progressive exophthalmos and loss of vision on the left eye. Cranial magnetic resonance and angiography examinations demonstrated a left craniofacial CM and large intraorbital component. The lesion was connected through a large basal vein to a cerebral intraventricular CM. Transconjunctival resection showed typical findings of CM. A complete histopathology and immunostaining analysis was performed and revealed a clear acute lymphomononuclear reaction with a predominant immune cellular inflammation.

Conclusions: A case of intraorbital and extracranial cavernomatous mass, connected to a cerebral intraventricular CM through a large basal vein, has presented with an aggressive course. A complete histopathologic and immunohistochemical analysis of the orbital mass has pictured a clear immune-cellular inflammatory reaction adding to the amounting evidence of association between inflammation and site aggressiveness in the setting of CMs.

Keywords: Cerebral cavernous malformation; Immune response; Inflammation.

Publication types

  • Case Reports

MeSH terms

  • Adult
  • B-Lymphocytes / immunology
  • B-Lymphocytes / pathology
  • Cerebral Angiography
  • Cerebral Veins / diagnostic imaging*
  • Cerebral Ventricle Neoplasms / diagnostic imaging*
  • Female
  • Hemangioma, Cavernous, Central Nervous System / diagnostic imaging*
  • Hemangioma, Cavernous, Central Nervous System / immunology
  • Hemangioma, Cavernous, Central Nervous System / pathology
  • Hemangioma, Cavernous, Central Nervous System / surgery
  • Humans
  • Inflammation / immunology
  • Inflammation / pathology
  • Macrophages / immunology
  • Macrophages / pathology
  • Magnetic Resonance Imaging
  • Neoplasms, Multiple Primary / diagnostic imaging*
  • Neoplasms, Multiple Primary / immunology
  • Neoplasms, Multiple Primary / pathology
  • Neoplasms, Multiple Primary / surgery
  • Orbital Neoplasms / diagnostic imaging*
  • Orbital Neoplasms / immunology
  • Orbital Neoplasms / pathology
  • Orbital Neoplasms / surgery
  • Plasma Cells / immunology
  • Plasma Cells / pathology