Orbital lymphatic-venous malformation with concomitant spontaneous orbital arteriovenous fistula: case report

Abstract

<jats:p>The authors describe the case of an 11-year-old boy with no history of prior trauma, who experienced acute proptosis and visual loss while showering. Diagnostic carotid artery angiography revealed an orbital lymphatic-venous malformation (LVM) concomitant with an intraorbital arteriovenous fistula. The patient was treated with transvenous coil and <jats:italic>N</jats:italic>-butyl cyanoacrylate glue embolization, combined with direct percutaneous glue injection into the LVM followed by excision. There was good clinical and radiological response without recurrence at the 2-year follow-up. Diagnostic carotid angiography should be considered in cases of orbital LVM, as a concomitant arteriovenous fistula might be present in rare cases.</jats:p>

Orbital Lymphangioma: A Case Report and Review of Management Modalities

“…Recently, conservative treatment has become an alternative for managing orbital lymphangioma, which causes no visual deficits or cosmetic issues [8,12]. Based on previous reports, nonsurgical strategies for orbital lymphatic venous malformations are sclerosing therapy, including ethanol, OK-432, sodium tetradecyl sulfate (Sotradecol, Mylan), morrhuate sodium, doxycycline, bleomycin, and pingyangmycin [3,14,28].…”

“…Based on previous reports, nonsurgical strategies for orbital lymphatic venous malformations are sclerosing therapy, including ethanol, OK-432, sodium tetradecyl sulfate (Sotradecol, Mylan), morrhuate sodium, doxycycline, bleomycin, and pingyangmycin [3,14,28]. Other Lately, the use of oral sildenafil for the treatment of microcystic lesions has been reported [8]. Sildenafil reduces contractility of vascular smooth muscles and causes cystic lesions to collapse [29,30].…”

“…Surgical management can be combined with preoperative intralesional injection of sclerosing agents or liquid polymers such as n-butyl cyanoacrylate glue or fibrin glue to aid in dissection and improve hemostasis [8]. Fibrin glue is a useful aid during resection and debulking surgery and stabilizes the removed structures [31].…”

“…Lymphangioma can also infiltrate diffusely into surrounding vital structures, such as the optic nerve. Because of its nature, as well as location and connection to adjacent vital structures of the orbit, management can be challenging and depends on the clinical presentation [7,8]. Here we report a case of orbital lymphangioma presented as acute proptosis and discuss different features along with management of this malformation.…”

Orbital Venous Malformation Accompanied by Arteriovenous Fistula

“…A similar case has been reported that an orbital lymphaticovenous malformation was accompanied by an arteriovenous fistula. In that case, coil embolization and sclerotherapy were performed followed by resection [ 5 ]. The patient in the present case had a relatively well-demarcated OVM but without surgical intervention of the intraorbital lesion, it was successfully regressed after intracranial feeder vessel cauterization.…”