• Jorge Marcondes de Souza, Camilo Abud Aiex, Nathalie Henriques Silva Canedo, Suelen Adriani Marques, and Blanco Martinez Ana Maria AM Department of Pathology, Federal University of Rio de Janeiro, HUCFF, and Graduate Program of Pathology, Rio de Janeiro, Brazil; Neurodegener.
• Department of Neurosurgery, Federal University of Rio de Janeiro, Neurosurgery Service (HUCFF), and Graduate Program of Surgery Sciences, UFRJ, Rio de Janeiro, Brazil. Electronic address: jormarcondes@gmail.com.
• World Neurosurg. 2019 Nov 1; 131: 87-89.

BackgroundIntraorbital 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 DescriptionA 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.ConclusionsA 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.Copyright © 2019 Elsevier Inc. All rights reserved.

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