Spontaneous carotid-cavernous fistula in osteogenesis imperfecta

Case report

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✓ A case of spontaneous carotid-cavernous fistula is presented in a patient suffering from osteogenesis imperfecta tarda type I. The patient was operated on under local anesthesia by intraluminal occlusion of the cavernous carotid artery with a Fogarty catheter, in accordance with Prolo's technique, with total remission except for homolateral loss of vision due to postglaucoma atrophy.

The pathogenetic phenomena described in the literature are reviewed, with the suggestion that osteogenesis imperfecta be included with Ehlers-Danlos disease, Marfan's syndrome, and pseudoxanthoma elasticum, as a connective tissue disease with increased weakness of vessel walls that can produce a spontaneous carotid-cavernous fistula. Reports of cases operated on by intraluminal occlusion with various types of balloon-tipped catheters are also reviewed. The good results obtained place this technique among the elective ones for the treatment of carotid-cavernous fistula.

Article Information

Address reprint requests to: José R. Boixadós, Neurosurgery Service, Fundación Jiménez Díaz, Universidad Autónoma de Madrid, Madrid 3, Spain.

© AANS, except where prohibited by US copyright law.

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Figures

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    Right carotid angiography with compression of the left carotid artery shows filling of the carotid-cavernous fistula and both hemispheres.

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    Left: Left carotid angiography with early drainage of the carotid-cavernous fistula into the contralateral carotid-cavernous sinus (SC), both jugular veins (Y), the ophthalmic vein (O), and the sagittal sinus (SLS) from the sphenoparietal sinus (E), and frontal (F) and parietal (P) veins. Right: Lateral projection showing the fistula (FCC) filling from the left carotid artery (C), and early drainage through the jugular (Y), ophthalmic (O), temporal (T), and frontal (F) veins, and sphenoparietal sinus (E).

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    Postoperative skull film showing the location of the balloon and catheter filled with contrast medium.

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