Bilateral Meningoencephaloceles with Cerebrospinal Fluid Rhinorrhea after Facial Advancement in the Crouzon Syndrome

Background Cerebrospinal fluid (CSF) rhinorrhea and encephaloceles are rare complications of craniofacial advancement procedures performed in patients with craniofacial dysostoses (CD) to address the ramifications of their midface hypoplasia including obstructed nasal airway, exorbitism, and impaire...

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Main Authors: Bharat A. Panuganti M.D., Matthew Leach M.D., Jastin Antisdel M.D.
Format: Article
Language:English
Published: SAGE Publishing 2015-06-01
Series:Allergy & Rhinology
Online Access:https://doi.org/10.2500/ar.2015.6.0129
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author Bharat A. Panuganti M.D.
Matthew Leach M.D.
Jastin Antisdel M.D.
author_facet Bharat A. Panuganti M.D.
Matthew Leach M.D.
Jastin Antisdel M.D.
author_sort Bharat A. Panuganti M.D.
collection DOAJ
description Background Cerebrospinal fluid (CSF) rhinorrhea and encephaloceles are rare complications of craniofacial advancement procedures performed in patients with craniofacial dysostoses (CD) to address the ramifications of their midface hypoplasia including obstructed nasal airway, exorbitism, and impaired mastication. Surgical repair of this CSF rhinorrhea is complicated by occult elevations in intracranial pressure (ICP), potentially necessitating open, transcranial repair. We report the first case in otolaryngology literature of a patient with Crouzon syndrome with late CSF rhinorrhea and encephalocele formation after previous LeFort III facial advancement surgery. Objectives Describe the case of a patient with Crouzon syndrome who presented with CSF rhinorrhea and encephaloceles as complications of Le Fort III facial advancement surgery. Review the literature pertaining to the incidence and management of post-operative CSF rhinorrhea and encephaloceles. Analyze issues related to repair of these complications, including occult elevations in ICP, the utility of perioperative CSF shunts, and the importance of considering alternative repair schemes to the traditional endonasal, endoscopic approach. Methods Review of the literature describing CSF rhinorrhea and encephalocele formation following facial advancement in CD, focusing on management strategies. Results CSF rhinorrhea and encephalocele formation are rare complications of craniofacial advancement procedures. Occult elevations in ICP complicate the prospect of permanent surgical repair, potentially necessitating transcranial repair and the use of CSF shunts. Though no consensus exists regarding the utility of perioperative CSF drains, strong associations exist between elevated ICP and failed surgical repair. Additionally, the anatomic changes in the frontal and ethmoid sinuses after facial advancement present a challenge to endoscopic repair. Conclusion Otolaryngologists should be aware of the possibility of occult elevations in ICP and sinonasal anatomic abnormalities when repairing CSF rhinorrhea in patients with CD. Clinicians should consider CSF shunt placement and carefully weigh the advantages of the transcranial approach versus endonasal, endoscopic techniques.
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spelling doaj.art-41ff0a4af8b64b8ea4107de355b8c0d72022-12-21T16:54:11ZengSAGE PublishingAllergy & Rhinology2152-65672015-06-01610.2500/ar.2015.6.0129Bilateral Meningoencephaloceles with Cerebrospinal Fluid Rhinorrhea after Facial Advancement in the Crouzon SyndromeBharat A. Panuganti M.D.0Matthew Leach M.D.1Jastin Antisdel M.D.2Department of Otolaryngology, Head and Neck Surgery, Division of Rhinology and Sinus Surgery, Saint Louis University School of Medicine, St. Louis, MissouriDepartment of Otolaryngology, Head and Neck Surgery, Division of Rhinology and Sinus Surgery, Saint Louis University School of Medicine, St. Louis, MissouriDepartment of Otolaryngology, Head and Neck Surgery, Division of Rhinology and Sinus Surgery, Saint Louis University School of Medicine, St. Louis, MissouriBackground Cerebrospinal fluid (CSF) rhinorrhea and encephaloceles are rare complications of craniofacial advancement procedures performed in patients with craniofacial dysostoses (CD) to address the ramifications of their midface hypoplasia including obstructed nasal airway, exorbitism, and impaired mastication. Surgical repair of this CSF rhinorrhea is complicated by occult elevations in intracranial pressure (ICP), potentially necessitating open, transcranial repair. We report the first case in otolaryngology literature of a patient with Crouzon syndrome with late CSF rhinorrhea and encephalocele formation after previous LeFort III facial advancement surgery. Objectives Describe the case of a patient with Crouzon syndrome who presented with CSF rhinorrhea and encephaloceles as complications of Le Fort III facial advancement surgery. Review the literature pertaining to the incidence and management of post-operative CSF rhinorrhea and encephaloceles. Analyze issues related to repair of these complications, including occult elevations in ICP, the utility of perioperative CSF shunts, and the importance of considering alternative repair schemes to the traditional endonasal, endoscopic approach. Methods Review of the literature describing CSF rhinorrhea and encephalocele formation following facial advancement in CD, focusing on management strategies. Results CSF rhinorrhea and encephalocele formation are rare complications of craniofacial advancement procedures. Occult elevations in ICP complicate the prospect of permanent surgical repair, potentially necessitating transcranial repair and the use of CSF shunts. Though no consensus exists regarding the utility of perioperative CSF drains, strong associations exist between elevated ICP and failed surgical repair. Additionally, the anatomic changes in the frontal and ethmoid sinuses after facial advancement present a challenge to endoscopic repair. Conclusion Otolaryngologists should be aware of the possibility of occult elevations in ICP and sinonasal anatomic abnormalities when repairing CSF rhinorrhea in patients with CD. Clinicians should consider CSF shunt placement and carefully weigh the advantages of the transcranial approach versus endonasal, endoscopic techniques.https://doi.org/10.2500/ar.2015.6.0129
spellingShingle Bharat A. Panuganti M.D.
Matthew Leach M.D.
Jastin Antisdel M.D.
Bilateral Meningoencephaloceles with Cerebrospinal Fluid Rhinorrhea after Facial Advancement in the Crouzon Syndrome
Allergy & Rhinology
title Bilateral Meningoencephaloceles with Cerebrospinal Fluid Rhinorrhea after Facial Advancement in the Crouzon Syndrome
title_full Bilateral Meningoencephaloceles with Cerebrospinal Fluid Rhinorrhea after Facial Advancement in the Crouzon Syndrome
title_fullStr Bilateral Meningoencephaloceles with Cerebrospinal Fluid Rhinorrhea after Facial Advancement in the Crouzon Syndrome
title_full_unstemmed Bilateral Meningoencephaloceles with Cerebrospinal Fluid Rhinorrhea after Facial Advancement in the Crouzon Syndrome
title_short Bilateral Meningoencephaloceles with Cerebrospinal Fluid Rhinorrhea after Facial Advancement in the Crouzon Syndrome
title_sort bilateral meningoencephaloceles with cerebrospinal fluid rhinorrhea after facial advancement in the crouzon syndrome
url https://doi.org/10.2500/ar.2015.6.0129
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