Highlights
- •A case of CCJ pial AVF causing postoperative medullary and spinal cord edema.
- •Existence of arteriovenous shunts in the medulla in addition to the CCJ pial AVF.
- •Surgical removal of the varix as another draining route
- •The cause of edema is concentration of the drainage into the anterior spinal vein.
- •High flow AVF can induce angiogenesis and secondary arteriovenous shunts.
Abstract
Objectives
Pial arteriovenous fistulas (pAVFs) are direct connections between the pial artery
and vein without an intervening nidus. We report a rare case of craniocervical junction
(CCJ) pAVF causing medullary and spinal cord edema resulting from surgical removal
of the varix with remnant shunt after coil embolization.
Case description
A 16-year-old man presented with subarachnoid hemorrhage. Digital subtraction angiography
revealed a CCJ pAVF with multiple fistulas at the 2 varices (varix A and varix B),
which was fed by the bilateral lateral spinal arteries and anterior spinal artery
(ASA), and drained into the median posterior vermian vein with varix (varix C) and
anterior spinal vein (ASV). Varices A and B were embolized using coils, but the shunts
remained in varix C. Then, varix C was surgically removed. After this operation, medullary
and spinal cord edema occurred. Digital subtraction angiography showed the ASV drainage
responsible for edema. Finally, surgical removal of varices A and B was performed.
However, arteriovenous shunts, supplied by the ASA and drained into the ASV via the
intrinsic vein, were found in the medulla oblongata and coagulated, resulting in disappearance
of edema.
Conclusions
Edema was probably caused by concentration of drainage from the arteriovenous shunt
in the medulla oblongata into the ASV by surgical removal of varix C acting as another
draining route. High flow AVF can induce angiogenesis and secondary arteriovenous
shunt. Precise analysis of the angioarchitecture is important to treat such cases
without complications.
Keywords
Abbreviations:
ASA (anterior spinal artery), ASV (anterior spinal vein), CCJ (craniocervical junction), LSA (lateral spinal arteries), MR (magnetic resonance), pAVF (pial arteriovenous fistula), PICA (posterior inferior cerebellar artery), PSV (posterior spinal vein)To read this article in full you will need to make a payment
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Article info
Publication history
Published online: November 30, 2022
Accepted:
October 16,
2022
Received in revised form:
October 2,
2022
Received:
June 23,
2022
Identification
DOI: https://doi.org/10.1016/j.jstrokecerebrovasdis.2022.106852
Copyright
© 2022 Elsevier Inc. All rights reserved.
