0
Selected Reports |

Cerebrospinal Fluid Leak and Meningitis Associated With Nasal Continuous Positive Airway Pressure Therapy* FREE TO VIEW

Tomasz J. Kuzniar, MD, PhD; Benjamin Gruber, MD, PhD; Gökhan M. Mutlu, MD, FCCP
Author and Funding Information

*From the Division of Pulmonary and Critical Care Medicine (Drs. Kuzniar and Mutlu), Northwestern University Feinberg School of Medicine, Chicago; and Otolaryngology–Head and Neck Surgery (Dr. Gruber), Mercy Hospital and Medical Center, Chicago, IL.

Correspondence to: Tomasz J. Kuzniar, MD, PhD, Sleep Disorders Center, Mayo Clinic, 200 First Street SW, Rochester, MN 55905; e-mail: kuzniar.tomasz@mayo.edu



Chest. 2005;128(3):1882-1884. doi:10.1378/chest.128.3.1882
Text Size: A A A
Published online

Clear rhinorrhea is a common symptom in patients with obstructive sleep apnea (OSA) and may worsen with continuous positive airway pressure therapy. Clear rhinorrhea can also be the presenting symptom of cerebrospinal fluid (CSF) leak, which is evidence of a communication between the subarachnoid space and the nasal cavity or sinuses. While CSF leak has been reported to occur with nasal continuous positive airway pressure (nCPAP) therapy following trauma to the skull base, its association with OSA and nCPAP therapy in the absence of trauma has not been previously described. We report two patients with OSA in whom CSF leak developed following the institution of nCPAP therapy. In one patient, the rhinorrhea was complicated by meningitis. Both patients underwent successful repair of their defects. One patient successfully restarted nCPAP therapy, while the other refused it.

Figures in this Article

Complications of nasal continuous positive airway pressure (nCPAP) are usually local and related to either the mask interface or dry air. Cerebrospinal fluid (CSF) leak is a dangerous but rare complication of nCPAP occurring only with concomitant trauma to the skull base. Herein, we report two patients with obstructive sleep apnea (OSA) in whom, after institution of nCPAP, increased clear rhinorrhea developed, which led to the diagnosis of CSF leak.

An obese, 54-year-old woman with history of moderate OSA (apnea-hypopnea index of 15.5 events/h) presented with complaints of clear, watery rhinorrhea and postnasal drip for 3 months. Rhinorrhea was more prominent when she leaned forward. She denied any headache, fever, or chills. She has been compliant with nCPAP of 10 cm H2O since it was prescribed 3 years previously.

The patient denied any history of sinusitis, allergic rhinitis, facial trauma, or previous nasal or sinus surgery. CT of the sinuses showed an air-fluid level in the left sphenoid sinus with extensive pneumatization of the sinuses. She was prescribed nasal steroids and antibiotics.

She presented to the emergency department 2 weeks later with mental status changes and headache, and subsequently was admitted with the diagnosis of meningitis. A CT cisternogram was obtained (Fig 1 ), and it showed a CSF leak into the left sphenoid sinus. After successful treatment of the meningitis with antibiotics, the patient was discharged and underwent elective surgical repair of the CSF leak via nasal sinus endoscopy. Postoperatively, she had no recurrence of the CSF leak for approximately 1 year. The patient refused to restart nCPAP therapy.

A 63-year-old woman with history of allergic rhinitis, recurrent sinusitis, and nasal polyposis treated with endoscopic sinus surgery 10 years prior to presentation, and recent diagnosis of moderate OSA (apnea-hypopnea index of 18 events/h) presented to her otolaryngologist with a 2-month history of increased clear rhinorrhea. Although she intermittently had rhinorrhea before, she noticed copious amounts of clear secretion within 2 weeks following the introduction of nCPAP at 12 cm H2O. She reported that the fluid had a “salty” taste.

Nasal endoscopic examination did not show any source for discharge, defect, or encephalocele. The CT cisternogram showed a suspicious area in the lateral aspect of the cribriform plate, adjacent to area of the previous sinus surgery, but no frank extravasation of contrast was noted.

Based on patient’s continued symptoms, she underwent revision endoscopic surgery. Intraoperatively, there was a soft area in the lateral lamella of the cribriform, anterior to the sphenoid sinus os, corresponding to the suspicious area on the CT cisternogram further supporting the presence of CSF leak. The area was patched with a temporalis fascia graft. Recovery after the repair was uneventful, and the rhinorrhea resolved completely.

Hypersomnolence redeveloped shortly after the patient was instructed to discontinue nCPAP, as the CSF leak was attributed to nCPAP. Consequently, continuous positive airway pressure was restarted 8 weeks after surgery using an oral interface (Oracle; Fisher & Paykel, Laguna Hills, CA), to which she responded well. The patient remained asymptomatic after 6 months of observation.

The most common side effects of nCPAP therapy are related to the interface between the continuous positive airway pressure equipment and patient’s face (nasal bridge irritation, eye irritation) or direct effect of the dry air and pressure on the upper airway (ie, rhinitis, oropharyngeal dryness, earache, epistaxis).1Rhinorrhea is a common complaint occurring in 30 to 50% of patients with OSA and is frequently exacerbated by nCPAP therapy.2

Clinical symptoms of CSF leak include clear rhinorrhea, which may worsen with leaning forward; orthostatic headache; and meningitis.3The most common causes of CSF leak are trauma and surgery involving the skull base.4 “Spontaneous” CSF leak without an identifiable cause occurs in 2 to 40% of patients.4 The majority of spontaneous leaks are associated with elevated intracranial pressure (ICP) suggested by development of an arachnoidocele into sella turcica.4

OSA may predispose to the development of CSF leak. Obesity, a major risk factor for OSA, is also risk factor for CSF leak.5Apnea may elevate the ICP up to 30 to 40 cm H2O by increasing central venous pressure and vasodilation in cerebral vessels in response to hypoxemia and hypercapnia.6 However, the prevalence of increased ICP among patients with OSA is unknown.

There are several possible mechanisms through which nCPAP may affect the ICP in patients with OSA. While positive pressure may increase ICP and CSF pressure by decreasing venous return, and increasing central venous and longitudinal vertebral venous pressures during mechanical ventilation,7 these unwanted effects of positive pressure ventilation can conceivably be offset by its beneficial effects on apnea-related changes in ICP. Furthermore, application of positive pressure to the upper airway may also decrease transdural gradient of pressure and lower the amplitude of transdural pressure swings.

In the absence of prior instrumentation, dura and bony structures of the skull base provide a tight barrier between the subarachnoid space and the adjacent air-containing cavities. Nonetheless, the cribriform plate has been implicated in the development of CSF leak, as its thin structure makes it vulnerable to the effects of positive pressure. In fact, congenital fistulas across the cribriform plate are thought to be the cause of spontaneous CSF leaks.8 While these defects may be congenital, they may also be acquired during prior surgery involving air-containing cavities or through presence of an erosive process involving the dura.89 Conceivably, nCPAP may unmask a subclinical defect/communication between subarachnoid space and the aerated spaces that are exposed to positive pressure such as nasal cavity, sinuses, and middle ear, and consequently cause CSF leak. While temporal relationship between the initiation of nCPAP and onset of rhinorrhea in patient 2 suggests such a causal relationship, this is difficult to postulate in patient 1. CT cisternogram results further supported this possibility in patient 2. Nevertheless, we believe the most interesting aspect of these cases is the nonspecificity of the clear rhinorrhea, which could have been easily attributed to nCPAP therapy and resulted in overlooking of the symptom and dismissal of the real cause, the CSF leak.

Although patients with OSA are at risk for CSF leak, it appears to be a rare complication. As clear rhinorrhea can be the only symptom of CSF leak and therefore can be easily overlooked, history of the prior sinus or mastoid surgery should be sought in each patient considered for nCPAP therapy, and clinicians should maintain a low threshold for further investigation.

Abbreviations: CSF = cerebrospinal fluid; ICP = intracranial pressure; nCPAP = nasal continuous positive airway pressure; OSA = obstructive sleep apnea

This work was supported in part by the American Heart Association and American Lung Association.

Figure Jump LinkFigure 1. CT cisternogram showing leakage of contrast material from the subarachnoid space into the left sphenoid sinus.Grahic Jump Location
Strumpf, DA, Harrop, P, Dobbin, J, et al (1989) Massive epistaxis from nasal CPAP therapy.Chest95,1141. [CrossRef] [PubMed]
 
Brander, PE, Soirinsuo, M, Lohela, P Nasopharyngeal symptoms in patients with obstructive sleep apnea syndrome: effect of nasal CPAP treatment.Respiration1999;66,128-135. [CrossRef] [PubMed]
 
Mokri, B, Hunter, SF, Atkinson, JL, et al Orthostatic headaches caused by CSF leak but with normal CSF pressures.Neurology1998;51,786-790. [CrossRef] [PubMed]
 
Mangiola, A, Anile, C, Di Chirico, A, et al Cerebrospinal fluid rhinorrhea: pathophysiological aspects and treatment.Neurol Res2003;25,708-712. [CrossRef] [PubMed]
 
Badia, L, Loughran, S, Lund, V Primary spontaneous cerebrospinal fluid rhinorrhea and obesity.Am J Rhinol2001;15,117-119. [CrossRef] [PubMed]
 
Hanigan, WC, Zallek, SN Headaches, shunts, and obstructive sleep apnea: report of two cases.Neurosurgery2004;54,764-768;discussion 768–769. [CrossRef] [PubMed]
 
Guerci, AD, Shi, AY, Levin, H, et al Transmission of intrathoracic pressure to the intracranial space during cardiopulmonary resuscitation in dogs.Circ Res1985;56,20-30. [CrossRef] [PubMed]
 
Jarjour, NN, Wilson, P Pneumocephalus associated with nasal continuous positive airway pressure in a patient with sleep apnea syndrome.Chest1989;96,1425-1426. [CrossRef] [PubMed]
 
Bamford, CR, Quan, SF Bacterial meningitis: a possible complication of nasal continuous positive airway pressure therapy in a patient with obstructive sleep apnea syndrome and a mucocele.Sleep1993;16,31-32. [PubMed]
 

Figures

Figure Jump LinkFigure 1. CT cisternogram showing leakage of contrast material from the subarachnoid space into the left sphenoid sinus.Grahic Jump Location

Tables

References

Strumpf, DA, Harrop, P, Dobbin, J, et al (1989) Massive epistaxis from nasal CPAP therapy.Chest95,1141. [CrossRef] [PubMed]
 
Brander, PE, Soirinsuo, M, Lohela, P Nasopharyngeal symptoms in patients with obstructive sleep apnea syndrome: effect of nasal CPAP treatment.Respiration1999;66,128-135. [CrossRef] [PubMed]
 
Mokri, B, Hunter, SF, Atkinson, JL, et al Orthostatic headaches caused by CSF leak but with normal CSF pressures.Neurology1998;51,786-790. [CrossRef] [PubMed]
 
Mangiola, A, Anile, C, Di Chirico, A, et al Cerebrospinal fluid rhinorrhea: pathophysiological aspects and treatment.Neurol Res2003;25,708-712. [CrossRef] [PubMed]
 
Badia, L, Loughran, S, Lund, V Primary spontaneous cerebrospinal fluid rhinorrhea and obesity.Am J Rhinol2001;15,117-119. [CrossRef] [PubMed]
 
Hanigan, WC, Zallek, SN Headaches, shunts, and obstructive sleep apnea: report of two cases.Neurosurgery2004;54,764-768;discussion 768–769. [CrossRef] [PubMed]
 
Guerci, AD, Shi, AY, Levin, H, et al Transmission of intrathoracic pressure to the intracranial space during cardiopulmonary resuscitation in dogs.Circ Res1985;56,20-30. [CrossRef] [PubMed]
 
Jarjour, NN, Wilson, P Pneumocephalus associated with nasal continuous positive airway pressure in a patient with sleep apnea syndrome.Chest1989;96,1425-1426. [CrossRef] [PubMed]
 
Bamford, CR, Quan, SF Bacterial meningitis: a possible complication of nasal continuous positive airway pressure therapy in a patient with obstructive sleep apnea syndrome and a mucocele.Sleep1993;16,31-32. [PubMed]
 
NOTE:
Citing articles are presented as examples only. In non-demo SCM6 implementation, integration with CrossRef’s "Cited By" API will populate this tab (http://www.crossref.org/citedby.html).

Some tools below are only available to our subscribers or users with an online account.

Related Content

Customize your page view by dragging & repositioning the boxes below.

CHEST Journal Articles
PubMed Articles
  • CHEST Journal
    Print ISSN: 0012-3692
    Online ISSN: 1931-3543