Atypical Presentation of Giant Aneurysm in Pediatric Patient with Duane Syndrome

Chao-Hung Kuo; Lynn B McGrath; Joseph A Carnevale; Neena I Marupudi; Jeffrey G Ojemann; Richard G Ellenbogen; Anthony C Wang

doi:10.1016/j.wneu.2018.05.053

Atypical Presentation of Giant Aneurysm in Pediatric Patient with Duane Syndrome

World Neurosurg. 2018 Aug:116:25-28. doi: 10.1016/j.wneu.2018.05.053. Epub 2018 May 17.

Authors

Chao-Hung Kuo¹, Lynn B McGrath², Joseph A Carnevale³, Neena I Marupudi⁴, Jeffrey G Ojemann⁵, Richard G Ellenbogen⁴, Anthony C Wang⁶

Affiliations

¹ Department of Neurological Surgery, University of Washington, Seattle, Washington, USA; Department of Neurosurgery, Neurological Institute, Taipei Veterans General Hospital, Taipei, Taiwan; School of Medicine, National Yang-Ming University, Taipei, Taiwan. Electronic address: chaohungk@gmail.com.
² Department of Neurological Surgery, University of Washington, Seattle, Washington, USA.
³ Department of Neurological Surgery, Weill Cornell Medical Center, New York, New York, USA; Department of Neurosurgery, Warren Alpert Medical School of Brown University, Providence, Rhode Island, USA.
⁴ Department of Neurological Surgery, University of Washington, Seattle, Washington, USA; Department of Pediatric Neurosurgery, Seattle Children's Hospital, Seattle, Washington, USA.
⁵ Department of Neurological Surgery, University of Washington, Seattle, Washington, USA; Department of Pediatric Neurosurgery, Seattle Children's Hospital, Seattle, Washington, USA; Center for Sensorimotor Neural Engineering, University of Washington, Seattle, Washington, USA; Graduate Program in Neuroscience, University of Washington, Seattle, Washington, USA.
⁶ Department of Pediatric Neurosurgery, Seattle Children's Hospital, Seattle, Washington, USA; Ronald Reagan UCLA Medical Center, Santa Monica, California, USA.

PMID: 29777883
DOI: 10.1016/j.wneu.2018.05.053

Abstract

Background: Duane syndrome is a congenital eye movement disorder characterized by congenital malformation of the abducens nucleus. Thrombogenic conditions during development may lead to vascular anomalies in Duane syndrome; however, the presence of a giant aneurysm in this patient population is a rarely documented phenomenon.

Case description: We reported a case of a large cerebral aneurysm in a pediatric patient with Duane syndrome and performed a review of the literature to identify other potential cases and associations. The pathophysiologic hallmarks of Duane syndrome that lead to alterations in the fetal cerebral vasculature and that may form the basis for a potential mechanism for aneurysm formation were reviewed in this study. The patient was an 11-year-old female with Duane syndrome who presented with seizures. Computed tomography and magnetic resonance imaging demonstrated a large, heterogeneously enhancing right temporal mass. Intraoperatively, the mass was revealed to be a partially thrombosed giant middle cerebral artery aneurysm. After surgery, the patient had an uneventful postoperative course without residual aneurysm presented on postoperative angiogram. No clinical or radiographic appearance of recurrent aneurysm was evident at her 6-month follow-up.

Conclusions: The pathophysiology of vascular anomalies with Duane syndrome may be related to thrombogenic conditions during development leading to alterations in cerebral fetal vasculature. Strong consideration for vascular anomaly should be given when evaluating cerebral masses in patients with Duane syndrome.

Keywords: Cerebral aneurysm; Congenital disorder; Duane syndrome; Vasculopathy.

Publication types

Case Reports

MeSH terms

Cerebral Angiography / methods
Child
Duane Retraction Syndrome / diagnosis*
Duane Retraction Syndrome / surgery*
Female
Humans
Intracranial Aneurysm / diagnosis*
Intracranial Aneurysm / surgery*
Magnetic Resonance Imaging / methods
Ocular Motility Disorders / congenital
Treatment Outcome