Central retinal artery occlusion occurring 30 years after successful revascularization surgery for moyamoya disease: cas
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CASE REPORT - PEDIATRIC NEUROSURGERY
Central retinal artery occlusion occurring 30 years after successful revascularization surgery for moyamoya disease: case report Madeline B. Karsten 1
&
Cristiano Oliveira 2 & Alan Z. Segal 3 & R. Michael Scott 1
Received: 26 March 2020 / Accepted: 15 July 2020 # Springer-Verlag GmbH Austria, part of Springer Nature 2020
Abstract We report a case of central retinal artery occlusion (CRAO) leading to unilateral blindness occurring in a moyamoya patient 30 years after successful pial synangiosis when she was 6 years old. Imaging studies at the time of the CRAO revealed total occlusion of the ipsilateral cervical and intracranial internal carotid artery, a vessel shown to be patent on MRI/MRA studies for decades previously. This case demonstrates that long-term follow-up of operated moyamoya patients may reveal late secondary complications, of which physicians, patients, and families need to be aware. Keywords Central retinal artery . Occlusion . Moyamoya . Pediatrics . Ischemia
Abbreviations CRAO Central retinal artery occlusion MMD Moyamoya disease
ipsilateral extracranial and intracranial internal carotid artery which had remained patent on MRI/MRA on sequential studies for three decades previously.
Introduction
Case report
Moyamoya disease (MMD) is a progressive arteriopathy of the intracranial internal carotid arteries and their proximal branches resulting in cerebral ischemia and stroke [24, 26]. Ocular ischemia secondary to MMD has been rarely reported [2, 11, 16, 22]. In this paper, we present a case of unilateral central retinal artery occlusion (CRAO) in a 38-year-old female occurring 30 years after her cerebral revascularization surgery for MMD; this ophthalmic complication appeared to be due to spontaneous total occlusion of the
This 6-year-old female, who was born with rectal atresia and a cloacal anomaly, suffered a stroke under general anesthesia administered for a urologic procedure. A CT scan at that time showed a new stroke in the right hemisphere and an old stroke in the left hemisphere. An arteriogram demonstrated typical findings of MMD, with severe stenosis/occlusion of her right internal carotid artery just above its exit from the cavernous sinus. She underwent pial synangiosis on the right side at Boston Children’s Hospital in 1989 and on the opposite side in 1990. She had no further strokes and remained neurologically stable for the following 30 years. Her only maintenance medication was aspirin, 81 mg per day. Multiple ophthalmologic examinations over three decades revealed no evidence of ocular ischemia. In July of 2019, she experienced a painless reduction of vision in her left eye after bending over and standing back up. Initially, she was able to see hand motion in her left eye. One day later, however, she lost all sight in that eye and sought medical attention. Urgent CT and MRI revealed no changes from prior studies. There was no evidence of intracranial hemorrhage, acute ischemic events, or orbital mass. The MRA study, however, revealed a
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