Biphasic Development of Focal Cerebral Hyperperfusion After Revascularization Surgery for Adult Moyamoya Disease Associated With Autosomal Dominant Polycystic Kidney Disease.

Abstract

BACKGROUND:

Cerebral hyperperfusion (CHP) syndrome is a potential complication of superficial temporal artery-middle cerebral artery (STA-MCA) anastomosis for moyamoya disease (MMD), but its biphasic and delayed development is extremely rare.

CASE REPORT:

A 47-year-old woman with autosomal dominant kidney disease (ADPKD) presented with transient ischemic attacks due to MMD, and underwent left STA-MCA anastomosis. N-isopropyl-p-[123I] iodoamphetamine single-photon emission computed tomography (123IMP-SPECT) 1 day after surgery revealed asymptomatic CHP at the site of anastomosis. Strict blood pressure control and minocycline hydrochloride relieved CHP at postoperative day 7. However, 2 days later, the patient complained of sensory aphasia, and 123IMP-SPECT demonstrated significant focal CHP at the site of anastomosis accompanying high-intensity signal on magnetic resonance (MR) imaging of fluid attenuated inversion recovery (FLAIR) in her left temporal lobe near the site of anastomosis. We continued strict blood pressure control and additionally administered free radical scavenger (Edaravone) and antiepileptic agents, which gradually improved sensory aphasia. MR imaging and 123IMP-SPECT also confirmed the amelioration of the FLAIR-high lesion and focal CHP in her left temporal lobe. Two months later, the patient underwent right STA-MCA anastomosis without complications.

CONCLUSIONS:

Although the underlying mechanism is unknown, biphasic development of focal CHP after revascularization surgery in an MMD patient with ADPKD is unique. Due to the potential vulnerability of the systemic vessels in ADPKD, it is conceivable that intrinsic vascular wall fragility in MMD could be enhanced by ADPKD and have partly led to this rare complication.

• Authors: Ryosuke Tashiro, Miki Fujimura, Hidenori Endo, Toshiki Endo, Kuniyasu Niizuma, Teiji Tominaga
• Journal: Journal of stroke and cerebrovascular diseases : the official journal of National Stroke Association (J Stroke Cerebrovasc Dis) Vol. 27 Issue 11 Pg. 3256-3260 (Nov 2018) ISSN: 1532-8511 [Electronic] United States
• PMID: 30093201 (Publication Type: Case Reports, Journal Article)
• Copyright: Copyright © 2018. Published by Elsevier Inc.

Chemical References

• Anticonvulsants
• Antihypertensive Agents
• Free Radical Scavengers
• Radiopharmaceuticals
• Iofetamine

Topics

• Anastomosis, Surgical
• Anticonvulsants (therapeutic use)
• Antihypertensive Agents (therapeutic use)
• Cerebral Angiography
• Cerebral Revascularization (adverse effects)
• Cerebrovascular Circulation (drug effects)
• Female
• Free Radical Scavengers (therapeutic use)
• Humans
• Iofetamine (administration & dosage)
• Magnetic Resonance Imaging
• Middle Aged
• Middle Cerebral Artery (physiopathology, surgery)
• Moyamoya Disease (complications, diagnostic imaging, physiopathology, surgery)
• Polycystic Kidney, Autosomal Dominant (complications, diagnosis)
• Postoperative Complications (diagnostic imaging, drug therapy, etiology, physiopathology)
• Radiopharmaceuticals (administration & dosage)
• Temporal Arteries (physiopathology, surgery)
• Tomography, Emission-Computed, Single-Photon
• Treatment Outcome