Extracranial arteriovenous malformation and subsequent contralateral cavernous sinus dural arteriovenous fistula showing abducens nerve palsy: illustrative case.

Atsushi Tsukada,Kuniyuki Onuma,Kiyoyuki Yanaka,Maya Takada,Yuji Matsumaru,Nobuyuki Takahashi,Eiichi Ishikawa,Masayuki Sato,Kazuhiro Nakamura

doi:10.3171/case21265

Abstract

BACKGROUNDExtracranial arteriovenous malformations (AVMs) are rare clinical entities and on rare occasions cause neurological symptoms. The authors report a case of an extracranial pterygoid AVM and a subsequent contralateral cavernous sinus dural arteriovenous fistula (dAVF) presenting with abducens nerve palsy.OBSERVATIONSAn 80-year-old woman was referred to the authors’ hospital with left abducens nerve palsy followed by right ophthalmalgia. Magnetic resonance imaging (MRI) showed abnormal vessel staining in the left pterygoid and the right inferior petrosal sinus (IPS). Cerebral angiography revealed a left pterygoid AVM draining into the right IPS via the cavernous sinus (CS). A dAVF in the right CS was also revealed. The right ophthalmalgia disappeared spontaneously, and, 4 months later, the left abducens nerve palsy also disappeared after conservative management. Follow-up MRI showed spontaneous regression of the AVM and dAVF. The disappearance of the dAVF was considered to be due to spontaneous regression of the left pterygoid AVM and the consequent decrease in venous pressure of the CS, and the symptoms eventually disappeared.LESSONSThe authors treated an extremely rare case of extracranial AVM with dramatic changes in vascular structure and symptoms. Understanding of the pathophysiology between symptoms and dynamic changes in the vascular structure is essential for providing the appropriate treatment.

Highlights

Extracranial arteriovenous malformations (AVMs) are rare clinical entities and on rare occasions cause neurological symptoms
The etiology of AVMs and dural arteriovenous fistulas remain unclear, and there are some reports of spontaneous regression.[2,3,4]
We report a case of a left pterygoid AVM draining into the cavernous sinus (CS), which could have contributed to the development of left abducens nerve palsy and the formation of a contralateral CS dural arteriovenous fistula (dAVF)

Summary

Introduction

Extracranial arteriovenous malformations (AVMs) are rare clinical entities and on rare occasions cause neurological symptoms. Arteriovenous malformations (AVMs) can occur anywhere in the body, but extracranial AVMs are rare and, on very rare occasions, can lead to the development of neurological symptoms.[1] The etiology of AVMs and dural arteriovenous fistulas (dAVFs) remain unclear, and there are some reports of spontaneous regression.[2,3,4] We report a case of a left pterygoid AVM draining into the cavernous sinus (CS), which could have contributed to the development of left abducens nerve palsy and the formation of a contralateral CS dAVF.

Results

Conclusion