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Bilateral meningoencephaloceles with cerebrospinal fluid rhinorrhea after facial advancement in the Crouzon syndrome.

Panuganti BA, Leach M, Antisdel J - Allergy Rhinol (Providence) (2015)

Bottom Line: 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.Clinicians should consider CSF shunt placement and carefully weigh the advantages of the transcranial approach versus endonasal, endoscopic techniques.

View Article: PubMed Central - PubMed

Affiliation: Department of Otolaryngology, Head and Neck Surgery, Division of Rhinology and Sinus Surgery, Saint Louis University School of Medicine, St. Louis, Missouri, USA.

ABSTRACT

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.

No MeSH data available.


Related in: MedlinePlus

Ethmoid encephaloceles after facial advancement surgery. Orbital CT, showing bilateral soft-tissue masses extending into the nasal cavities through skull base defects overlying the patient's ethmoid sinuses.
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Figure 2: Ethmoid encephaloceles after facial advancement surgery. Orbital CT, showing bilateral soft-tissue masses extending into the nasal cavities through skull base defects overlying the patient's ethmoid sinuses.

Mentions: We present the case of a 19-year-old African American woman with Crouzon syndrome and Graves disease. In 2011, she underwent a subcranial Le Fort III midface osteotomy and advancement to address issues related to her midface hypoplasia, including nasal obstruction, exorbitism, and malocclusion. The procedure was performed without any immediate postoperative complications. However, within a year of this procedure, the patient developed near-constant clear rhinorrhea and frequent sinus pain and pressure. She was referred to our department for consideration of endoscopic orbital decompression due to her exophthalmos secondary to Graves disease. Rigid nasal endoscopy demonstrated large intranasal pulsatile masses that were concerning for bilateral ethmoid encephaloceles (Fig. 1A). A computed tomography (CT) of the sinuses revealed multisinus opacification and bilateral soft-tissue masses that extended into the nasal cavities through bony defects overlying her ethmoid sinuses (Fig. 2). A CT from 2009, performed before the Le Fort III osteotomy, showed an intact skull base without evidence of an intranasal mass or sinusitis.


Bilateral meningoencephaloceles with cerebrospinal fluid rhinorrhea after facial advancement in the Crouzon syndrome.

Panuganti BA, Leach M, Antisdel J - Allergy Rhinol (Providence) (2015)

Ethmoid encephaloceles after facial advancement surgery. Orbital CT, showing bilateral soft-tissue masses extending into the nasal cavities through skull base defects overlying the patient's ethmoid sinuses.
© Copyright Policy - open-access
Related In: Results  -  Collection

License
Show All Figures
getmorefigures.php?uid=PMC4541637&req=5

Figure 2: Ethmoid encephaloceles after facial advancement surgery. Orbital CT, showing bilateral soft-tissue masses extending into the nasal cavities through skull base defects overlying the patient's ethmoid sinuses.
Mentions: We present the case of a 19-year-old African American woman with Crouzon syndrome and Graves disease. In 2011, she underwent a subcranial Le Fort III midface osteotomy and advancement to address issues related to her midface hypoplasia, including nasal obstruction, exorbitism, and malocclusion. The procedure was performed without any immediate postoperative complications. However, within a year of this procedure, the patient developed near-constant clear rhinorrhea and frequent sinus pain and pressure. She was referred to our department for consideration of endoscopic orbital decompression due to her exophthalmos secondary to Graves disease. Rigid nasal endoscopy demonstrated large intranasal pulsatile masses that were concerning for bilateral ethmoid encephaloceles (Fig. 1A). A computed tomography (CT) of the sinuses revealed multisinus opacification and bilateral soft-tissue masses that extended into the nasal cavities through bony defects overlying her ethmoid sinuses (Fig. 2). A CT from 2009, performed before the Le Fort III osteotomy, showed an intact skull base without evidence of an intranasal mass or sinusitis.

Bottom Line: 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.Clinicians should consider CSF shunt placement and carefully weigh the advantages of the transcranial approach versus endonasal, endoscopic techniques.

View Article: PubMed Central - PubMed

Affiliation: Department of Otolaryngology, Head and Neck Surgery, Division of Rhinology and Sinus Surgery, Saint Louis University School of Medicine, St. Louis, Missouri, USA.

ABSTRACT

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.

No MeSH data available.


Related in: MedlinePlus