Abstract
We report the case of a 37-year-old male with Cowden disease that caused a gradual neurological deficit because of rupture of a brain stem cavernous hemangioma. Removal of the hemangioma and hematoma was performed with an infrafacial triangle approach. Nine months after the operation, magnetic resonance imaging showed abnormal vessels on the cerebellar surface. Digital subtraction angiography showed a dural arteriovenous fistula (dAVF) from part of the meningeal artery to the ectatic inferior vermian vein with cortical reflux. After embolization, surgical obliteration of the dAVF was performed. Surgical findings showed neovascularization in the thickened dura, in which dural vessels shunted to cerebellar vessels through adhesion between the dura mater and cerebellar surface. Therefore, the thickened dura was removed with the cerebellar surface. This case suggests that postoperative angiogenesis may cause arteriovenous fistula in patients with Cowden disease.
Keywords:
Cowden disease; PTEN; cavernous hemangioma; dural arteriovenous fistula; vascular endothelial growth factor.
Published by Elsevier Inc.
MeSH terms
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Adult
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Angiography, Digital Subtraction
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Biopsy
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Brain Stem Neoplasms / complications
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Brain Stem Neoplasms / diagnosis
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Brain Stem Neoplasms / genetics
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Brain Stem Neoplasms / surgery*
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Central Nervous System Vascular Malformations / diagnosis
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Central Nervous System Vascular Malformations / etiology*
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Central Nervous System Vascular Malformations / therapy
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Cerebral Angiography / methods
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Dura Mater / blood supply*
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Embolization, Therapeutic
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Hamartoma Syndrome, Multiple / complications*
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Hamartoma Syndrome, Multiple / diagnosis
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Hamartoma Syndrome, Multiple / genetics
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Hemangioma, Cavernous, Central Nervous System / complications
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Hemangioma, Cavernous, Central Nervous System / diagnosis
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Hemangioma, Cavernous, Central Nervous System / genetics
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Hemangioma, Cavernous, Central Nervous System / surgery*
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Hematoma / diagnosis
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Hematoma / etiology
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Hematoma / surgery*
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Humans
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Magnetic Resonance Imaging
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Male
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Mutation
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Neovascularization, Pathologic
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Neurosurgical Procedures / adverse effects*
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PTEN Phosphohydrolase / genetics
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Reoperation
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Rupture, Spontaneous
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Treatment Outcome
Substances
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PTEN Phosphohydrolase
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PTEN protein, human