Management of Recurrent Cerebrospinal Fluid Rhinorrhea Caused by Sequential, Anatomically Separated Skull Base Defects-A Case-Based Systematic Review.

OBJECTIVE: Recurrent cerebrospinal fluid (CSF) rhinorrhea caused by sequential, anatomically separated skull base defects is rarely reported in the literature. Neither management nor etiology has been sufficiently investigated. We present an illustrative case and a systematic review of the literature regarding etiology, diagnostics, and management of this rare phenomenon. METHODS: A systematic literature search looking for articles reporting sequential CSF leaks with multiple skull base defects was performed. Data from included articles were descriptively reported, and the quality of the included studies was assessed with Grading of Recommendations Assessment, Development and Evaluation. RESULTS: A 71-year-old woman with posttraumatic CSF rhinorrhea and left-sided CSF otorrhea due to a left-sided horizontal fracture of the petrous bone presented at our institution. After initial surgical repair and a 10-week symptom-free interval, CSF rhinorrhea recurred. Imaging revealed a preexisting contralateral meningoencephalocele of the lateral sphenoid recess causing recurrent CSF rhinorrhea most likely after initial traumatic laceration. The defect was successfully treated. A literature search identified 366 reports, 6 of which were included in the systematic review with a total of 10 cases. Quality was deemed good in 8 of 10 cases. The most common location for primary and sequential CSF leaks was along the sphenoid bone (4/10 and 5/10 patients, respectively). All publications except one reported the presence of a meningo (encephalo)cele as cause of the sequential CSF leak. CONCLUSIONS: Occurrence of recurrent CSF rhinorrhea due to an anatomically separated sequential skull base lesion remains a rare phenomenon. Reassessment of imaging studies and a structured diagnostic workup to detect sequential CSF leaks independent of the primary lesion should is recommended.

Carolyn's window approach for spontaneous frontal sinus meningoencephalocele.

Spontaneous meningoencephaloceles (MECs) are sparsely documented in the literature. Those occurring in the frontal sinus are an exceedingly rare entity. MECs are commonly associated with cerebrospinal fluid (CSF) rhinorrhoea. CSF rhinorrhoea is frequently misdiagnosed, causing delays in diagnosis and management. The subsequently increased risk of bacterial meningitis can be life-threatening to patients. We report the case of a woman in her late 70s with a spontaneous frontal sinus MEC, presenting with a 6-month history of CSF rhinorrhoea. The patient was successfully treated using the novel Carolyn's window approach endoscopically; 9-month follow-up revealed no skull-base breach. Our case emphasises the importance of considering MEC as a differential diagnosis for clear rhinorrhoea and demonstrates successful repair through a novel surgical approach.

Spontaneous rhinorrhea and idiopathic intracranial hypertension: a complex and challenging association.

INTRODUCTION: Spontaneous CSF leak is a known complication of idiopathic intracranial hypertension (IIH). Patients with CSF rhinorrhea present a unique challenge within the IIH population, as the occurrence of a leak can mask the typical IIH symptoms and signs, complicating the diagnosis. Treatment of leaks in this population can also be challenging, with the risk of rhinorrhea recurrence if intracranial hypertension is not adequately treated. OBJECTIVE: The aim of this narrative review was to examine current literature on the association between spontaneous CSF rhinorrhea leaks and IIH, focusing on key clinical features, diagnostic approaches, management strategies, and outcomes. MATERIAL AND METHODS: A literature search was executed using the PubMed and Scopus databases. The search was confined to articles published between January 1985 and August 2023; extracted data was then analysed to form the foundation of the narrative review. RESULTS: This search yielded 26 articles, comprising 943 patients. Average age was 46.8 ± 6.5 years, and average body mass index was 35.8 ± 4.8. Most of the patients were female (74.33%). Presenting symptoms were rhinorrhea, headaches and meningitis. The most common imaging findings were empty sella and encephalocele. The standard treatment approach was endoscopic endonasal approach for correction of CSF rhinorrhea leak, and shunt placement was also performed in 128 (13%) patients. Recurrences were observed in 10% of cases. CONCLUSIONS: The complex relationship between spontaneous CSF leaks and IIH is a challenge that benefits from multidisciplinary evaluation and management for successful treatment. Treatments such as endoscopic repair, acetazolamide, and VP/ /LP shunts reduce complications and recurrence. Personalised plans addressing elevated intracranial pressure are crucial for successful outcomes.

Endoscopic Repair of Cerebrospinal Fluid Rhinorrhea Due to Trauma by a Bamboo Stick in a Pediatric Patient.

Cerebrospinal fluid rhinorrhea (CSFR) is a condition in which the cerebrospinal fluid flows out of the nasal cavity due to rupture of the arachnoid, dura, and nasal membranes because of bone defects in the skull base. The authors report a rare case of CSFR in a 2-year-old girl who experienced trauma in the nasal cavity by a bamboo stick. She underwent endoscopic repair for the CSFR. During surgery, a bulged vesicle was observed at the left cribriform plate with a small amount of cerebrospinal fluid draining from the surrounding area. Postoperative recovery was good. Endoscopic CSFR repair in pediatric patients is minimally invasive, effective, and safe as demonstrated in this case. Prevention of CSFR in children is important. Parents and caretakers of children need to be more aware, and potentially dangerous objects should not be kept within reach of children.

How I do it? Endoscopic endonasal transpterygoid repair of sphenoid lateral recess cerebrospinal fluid leak after previous unsuccessful transcranial surgery.

BACKGROUND: Direct access to the sphenoid lateral recess offers the best chance of sealing spontaneous cerebrospinal fluid (CSF) rhinorrhea caused by lateral sphenoid encephaloceles of the Sternberg canal defect. METHOD: We present a case of spontaneous left-sided sphenoid lateral recess CSF leak after previous unsuccessful transcranial surgery managed with an endoscopic endonasal transpterygoid approach (EETA). An anatomical-based step-by-step illustration of the EETA was presented in the surgical video. CONCLUSION: This case demonstrates the value of endoscopic endonasal transpterygoid corridor in the exposure and manipulation of the sphenoid lateral recess.

Correlation between Pneumatization Variants of Paranasal Sinuses and Skull Base and Spontaneous Cerebrospinal Fluid Rhinorrhea: A Case-Control Study.

INTRODUCTION: Spontaneous cerebrospinal fluid rhinorrhea (SCSFR) is the most common type of cerebrospinal fluid leakage and may cause serious cerebral complications. The aim of this research was to investigate the relationship between the degree of pneumatization variants of the paranasal sinus and skull base and the incidence of SCSFR. METHODS: In total, 131 patients with SCSFR were analyzed, and 50 patients suffering from the nasal septal deviation were selected as controls. The pneumatization of the paranasal sinus and skull base was observed by CT scan. RESULTS: Among the 137 fistulas, 55 (40.15%) were found in the ethmoid sinus. The incidences of Onodi cells (27.27 vs. 8%) and type 3 lateral recess of the sphenoid sinus (LRSS, 70.37 vs. 22%) in the SCSFR subgroups were significantly higher than those in the control group (p < 0.05). Moreover, the occurrence of SCSFR was linearly correlated with the classification of Onodi cells and LRSS (p < 0.05). There was no significant difference in the incidence of frontal cells, anterior clinoid process pneumatization, and posterior clinoid process pneumatization between the SCSFR patients and the controls. CONCLUSION: The most common site of SCSFR is the ethmoid sinus. The excessive pneumatization of the Onodi cell and LRSS increases the risk for the occurrence of SCSFR in the ethmoid sinus and sphenoid sinus, respectively. The possible association between the paranasal sinus ontogeny and SCSFR pathophysiology needs further studies.

High-Resolution Computed Tomography as an Initial Diagnostic and Localization Tool in Patients with Cerebrospinal Fluid Rhinorrhea: A Meta-Analysis.

Background and Objectives: This study was performed to investigate the utility of high-resolution computed tomography (HRCT) for the initial localization of cerebrospinal fluid rhinorrhea. Methods: HRCT data regarding the point of cerebrospinal fluid leakage (as confirmed in the operating room), collected up to December 2022, were extracted from five databases. The risk of bias of the included studies was assessed using the Quality Assessment of Diagnostic Accuracy Studies 2 tool. Results: The search revealed eight relevant studies with a total of 254 patients. The diagnostic odds ratio of the imaging studies was 10.0729 (95% confidence interval [CI]: 2.4486; 41.4376; I2 = 54.1%). The area under the summary receiver operating characteristic curve was 0.8. Sensitivity, specificity, the negative predictive value, and the positive predictive value were 0.7550 (95% CI: 0.6163; 0.8553; I2 = 69.8%), 0.8502 (95% CI: 0.5986; 0.9557, I2 = 49.3%), 0.4106 (95% CI: 0.2418; 0.6035; I2 = 59.0%), and 0.9575 (95% CI: 0.8955; 0.9834; I2 = 27.7%), respectively. Conclusions: HRCT can be used to accurately localize cerebrospinal fluid rhinorrhea because it shows bony defects in high detail. However, it has limited utility for the evaluation of active leakage, and localization is difficult in the presence of coexisting lesions.

Evaluating the Efficacy of Topical Intranasal Fluorescein for Localizing CSF Rhinorrhea: A Systematic Review.

OBJECTIVE: To assess existing literature on topical intranasal fluorescein (TINF) for the diagnosis and localization of nasal cerebrospinal fluid (CSF). STUDY DESIGN: Systematic review. METHODS: A 6-database literature search was conducted to identify articles providing insight into TINF for the diagnosis and treatment of sinonasal CSF leak. Demographic characteristics, technical details, efficacy, and safety data were extracted and analyzed. Methodological quality was assessed using Methodological Items for Non-Randomized Studies (MINORS) criteria. RESULTS: All studies reported a diagnostic accuracy rate ≥96%. There were no major complications reported for any patient (n=99) with either 5% or 10% fluorescein use. The MINORS instrument of methodological quality indicated that the assessed studies were of moderate quality (7.29 out of a maximum score of 24). CONCLUSION: This systematic review indicates that TINF is an easy, safe, inexpensive, and sensitive approach for the diagnosis and treatment of sinonasal CSF leakage. For these reasons, it may be especially well suited for resource-limited clinical scenarios.

Spontaneous cerebrospinal fluid rhinorrhea associated with an incidental pituitary adenoma.

Spontaneous cerebrospinal fluid (CSF) rhinorrhea presenting as the sole symptom of untreated pituitary adenoma is rare, with only 15 cases having been reported in the English literature. All these untreated pituitary adenoma contributing to spontaneous CSF rhinorrhea were diagnosed by the preoperative neuroimaging. Herein, we described an extraordinary rare patient with a pituitary microadenoma, presenting with spontaneous CSF rhinorrhea as the sole symptom. However, this pituitary microadenoma was only found incidentally at surgery, not preoperatively. To the best knowledge of us, this is the first reported case of spontaneous CSF rhinorrhea associated with an untreated pituitary adenoma diagnosed at surgery.

Efficacy of Imaging Methods in the Detection and Diagnosis of Cerebrospinal Fluid Rhinorrhea.

OBJECTIVE: To assess the diagnostic efficacy of various imaging methods in patients with suspected cerebrospinal fluid (CSF) rhinorrhea. DATA SOURCES: The PubMed, EMBASE, SCOPUS, Web of Science, Cochrane Trials, and Google Scholar databases were searched up to December 2021. REVIEW METHODS: Diagnostic accuracy was compared among seven radiological methods: computed tomography (CT), CT cisternography (CTC), magnetic resonance imaging (MRI), magnetic resonance cisternography (MRC), CT + MRI, radionuclide cisternography, and intrathecal gadolinium (Gd)-MRC. Sensitivity, specificity, and accuracy were used as outcomes of the analysis. Both a traditional pairwise meta-analysis and a network meta-analysis were performed. RESULTS: Twenty-three trials were included in the analysis. The results of a network meta-analysis performed on a network consisting of seven diagnostic methods showed that all imaging modalities had greater diagnostic accuracy than CT, with the exception of CTC, which had lower sensitivity. Only intrathecal Gd-MRC was significantly superior to other imaging methods with regard to sensitivity and accuracy. Gd-MRC also showed the greatest surface under the cumulative ranking curve values for all of the outcomes (sensitivity: 0.9200; specificity: 0.8364; accuracy: 0.8920). CONCLUSION: This network meta-analysis demonstrates that intrathecal Gd-MRC is the most useful diagnostic method to detect CSF rhinorrhea. Laryngoscope, 133:1281-1287, 2023.

Prolactinoma extension as a contributing factor in dopamine agonist-induced CSF rhinorrhea: a systematic review of the literature.

BACKGROUND: Dopamine agonist-induced cerebrospinal fluid (CSF) rhinorrhea is an uncommon treatment-related complication arising in 6.1% of prolactinoma patients treated with dopamine agonists. Locally invasive prolactinomas may create CSF fistulae through formation of dural and osseous skull base defects. Tumor shrinkage secondary to dopamine agonist therapy unmasks skull base defects, thus inducing CSF rhinorrhea. In these cases, repair of the leak may be achieved through collaborative surgical intervention by rhinologists and neurosurgeons. Multiple variables have been investigated as potential contributors to the risk of CSF rhinorrhea development in medically treated prolactinoma patients, with little consensus. OBJECTIVE: The primary aim of our study was the characterization of risk factors for CSF rhinorrhea development following dopamine agonist treatment. METHODS: A systematic review of the literature was conducted to identify cases of CSF rhinorrhea following dopamine agonist treatment of prolactinoma. The clinical history, radiographic findings and treatment outcomes are discussed. RESULTS: Fifty-four patients with dopamine agonist-induced CSF rhinorrhea were identified across 23 articles published from 1979 to 2019. Description of diagnostic imaging [computed tomography (CT)/magnetic resonance imaging (MRI)] was not provided for 18/54 subjects. For the 36 cases that described prolactinoma appearance on CT or MRI, invasion of the cavernous sinuses was reported in 13 (36.1%) and invasion of the sphenoid sinus was reported in 18 (50%). CONCLUSION: Based on our systematic review, we propose that CT findings of osseous erosion of the sella or the anterior skull base may predict dopamine agonist-induced CSF rhinorrhea. We recommend obtaining a thin-slice CT of the sinuses in cases with MRI evidence of sphenoid involvement.

[Diagnostic value of computed tomographic cisternography and magnetic resonance hydrography in cerebrospinal fluid rhinorrhea].

Objective:To evaluate the accuracy of preoperative computed tomographic cisternography（CTC） and magnetic resonance hydrography（MRH）in the diagnosis of cerebrospinal fluid（CSF） rhinorrhea. Methods:Retrospective analysis was made on the data of 38 patients diagnosed as cerebrospinal rhinorrhea who completed preoperative HRCT, CTC and MRH examinations in the Department of Otolaryngology Head and Neck Surgery of Tianjin Huanhu Hospital from October 2016 to January 2022. The diagnostic accuracy of preoperative imaging examinations was compared according to the leak location found during operation. Results:Among all the 38 cases with CSF rhinorrhea, the detection rates of HRCT, CTC, and cranial MRH were 31.58%, 89.47%, and 60.53%, respectively. The accuracy of CTC was significantlyhigher than that of MRH （ χ²=8.49, P=0.007）, and the accuracy of MRH was significantly higher than that of HRCT （χ²=6.41, P=0.01）. Subgroup analyses were performed according to the precise positioning of cerebrospinal fluid rhinorrhea leakage. In patients with cerebrospinal fluid rhinorrhea located in ethmoid sinus, the accuracy of CTC（80.95% vs. 14.29%，P<0.01）and MRH（52.38% vs. 14.29%，P=0.02）were significantly higher than that of HRCT, but the accuracy but the difference between CTC and MRH between CTC and MRH was not statistically significant （P=0.10）. The accuracy of CTC was significantly higher than that of MRH （100.00% vs. 61.54%, P=0.04） and HRCT （100.00% vs. 53.85%, P<0.01） in patients with CSF rhinorrhea located in sphenoid sinus. However, there was no significant difference in the accuracy between MRH and HRCT （ P=1.00）. However, There was no significant difference in the accuracy of CTC, MRH and HRCT in patients with cerebrospinal fluid rhinorrhea located in frontal sinus. The accuracy of CTC and MRH was 84.62% and 57.69% respectively in 26 patients who failed to detect a CSF rhinorrhea by HRCT, and the difference was not statistically significant（χ²=4.59, P=0.06）. Conclusion:The comprehensive application of the multiple imaging methods has important guiding significance for the accurate treatment and prognosis evaluation. CTC and MRH could improve the accuracy of the localization diagnosis of cerebrospinal fluid rhinorrhea.

Systematic Review: Evaluating the Efficacy of Intrathecal Fluorescein for Localizing Cerebrospinal Fluid Rhinorrhea.

OBJECTIVES: A defect in the skull base can result in leakage of cerebrospinal fluid (CSF) out of the subarachnoid space into the sinonasal cavities, termed CSF rhinorrhea. Patients presenting with CSF rhinorrhea often require surgical repair, typically performed endoscopically. Successful surgical intervention is highly reliant on accurate identification of the leak site. Identification can be enhanced by the administration of intrathecal fluorescein (IF) via a lumbar drain before surgery. The objective of this systematic review is to better characterize the benefits and limitations of IF administration. METHODS: A systematic search was conducted for literature documenting the use of IF for CSF leak localization. The results of this search were subjected to initial review, followed by full-text evaluation of selected texts, and final inclusion based on predetermined selection criteria. Primary outcomes were specificity and sensitivity of IF administration in identifying CSF leak sites. Secondary outcomes included administration technique and safety data. RESULTS: A total of 25 studies representing 3801 instances of IF administration met inclusion criteria. When injected slowly for up to 30 minutes at doses of <50 mg, IF administration results in minimal complications. A total of 25 mg doses (92.36%) of IF appear more sensitive than 10 mg (71.88%) doses of IF while maintaining a similar safety profile. Mixing fluorescein with double distilled water instead of CSF or saline may decrease waiting times before fluorescein can be identified and increase its safety profile. CONCLUSIONS: Intrathecal fluorescein may play an important role in the identification of CSF leak site during endoscopic repair.

Diagnostic Value of Radioisotope Cisternography Using 111In-DTPA in a Patient with Rhinorrhea and Purulent Meningitis.

Cerebrospinal fluid (CSF) leakage is a rare condition. Prompt diagnosis and early treatment of CSF leakage minimizes the risk of severe complications such as bacterial meningitis. Different diagnostic modalities are used to detect the site of CSF leakage but often with unreliable results. The literature offers limited evidence-based guidance on the diagnostic approach for rhinorrhea. Correct localization of the defect is the mainstay for successful surgical treatment. Herein, we describe a case of recurrent meningitis due to cranio-nasal fistula and rhinorrhea successfully localized with radioisotope cisternography (RIC). We provide a detailed and practical overview of the RIC procedure and compare different imaging modalities used to detect the site of CSF leakage.

Endoscopic Endonasal Reconstruction Using a Pedicled Middle Turbinate Flap for Spontaneous Cerebrospinal Fluid Rhinorrhea.

ABSTRACT: Although endoscopic skull-base reconstruction protocols to reduce cerebrospinal fluid (CSF) leakage are reported, the most effective management strategies have not been determined. We describe the successful repair of a spontaneous CSF leak using a vascularized middle turbinate flap (MTF) via an endonasal endoscopic approach and also discuss the effective reconstruction with other available pedicled flaps. An 11-year-old girl had a 5-month history of intermittent CSF rhinorrhea. Endoscopic endonasal skull base reconstruction was performed using the pedicled MTF technique, which sufficiently covered the unilateral cribriform plate and ethmoidal fovea including suspicious leakage site. Middle turbinate flaps may be good for repairing spontaneous CSF leaks, which commonly have small, low-flow CSF fistulas around a cribriform plate. As spontaneous CSF leaks are known to have a higher recurrence rate, MTF may be advantageous because more of the normal structures are retained.

[The value of magnetic resonance hydrography in the localization of fistula in patients with cerebrospinal fluid rhinorrhea].

Objective:To evaluate the application value of magnetic resonance hydrography in determine the leak location of patients with cerebrospinal fluid rhinorrhea. Methods:Clinical data of 58 patients with cerebrospinal fluid rhinorrhea treated in Tianjin Huanhu Hospital from February 2015 to February 2021 were analyzed retrospectively. The leak position was judged by three methods: nasal sinus coronal CT, magnetic resonance hydrography and magnetic resonance hydrography combined with nasal sinus coronal CT. The consistency of the leak position among groups determined by different imageological examination pre-operation was compared with the real leak position found during the operation. Results:The positive rate of magnetic resonance hydrography combined with coronal CT in paranasal sinuses（91.4%） was significantly higher than that of magnetic resonance hydrography（74.1%）（P>0.05）. The positive rate of magnetic resonance hydrography（74.1%） was higher than that of CT（43.1%）（P>0.01）. Conclusion:The comprehensive application of imaging examination technology has important clinical guiding significance for the correct diagnosis of cerebrospinal fluid rhinorrhea. Magnetic resonance hydrography combined with coronal CT of paranasal sinus can effectively find the leak location, which was suggested as the first choice for preoperative localization of cerebrospinal fluid rhinorrhea.

Management of postoperative CSF rhinorrhea after retrosigmoid craniotomy: how I do it.

BACKGROUND: Cerebrospinal fluid (CSF) rhinorrhea as a complication of retrosigmoid craniotomy does not occur often today. This complication is primarily associated with the petrous bone drilling during surgery. METHOD: The management of this complication is shown by the example of the patient with a trigeminal schwannoma located in posterior cranial fossa operated through resrisigmoid craniotomy. Three steps of management and surgical stages of petrous bone plasty are shown. CONCLUSION: CSF rhinorrhea after retrosigmoid craniotomy is a preventable complication: petrous bone pneumatization should be evaluated preoperatively. If the air cells are open, primary plasty of the defect should be performed.

Long-Term Follow-up of Patients Managed Conservatively for Acute Traumatic CSF Rhinorrhea.

OBJECTIVE: Conservative management of acute traumatic cerebrospinal fluid rhinorrhea (TCR) results in cessation of the leak in most patients. The objective of this study was to estimate the incidence of recurrent cerebrospinal fluid (CSF) rhinorrhea and meningitis in these patients on long-term follow-up and to determine the risk factors associated with them. METHODS: Data on 50 patients with acute TCR who were successfully treated with conservative management between 2013 and 2015 and had long-term follow-up was retrieved from our head injury database. Patient variables were analyzed to determine the risk factors associated with recurrence of CSF rhinorrhea and meningitis. RESULTS: All patients in our series developed CSF rhinorrhea within 48 hours of trauma. The mean duration of follow-up was 6.3 ± 1.3 years. CSF rhinorrhea recurred in 16 (32%) patients, 15 (93.8%) of whom developed it within 3 years of trauma. Meningitis occurred in 5 (10%) patients and 1 died. Sphenoid sinus fractures and features of raised intracranial pressure on computerized tomography of the brain at admission were significantly associated with the development of meningitis. There were no risk factors identified for the recurrence of CSF rhinorrhea. CONCLUSIONS: Patients with acute TCR in whom rhinorrhea subsides with conservative therapy have the highest risk for recurrence of leak or meningitis within 3 years of the trauma. Therefore, we recommend that these patients be counselled about the need for periodic follow-up for several years.