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J Neurosurg Case Lessons
J Neurosurg Case Lessons
J Neurosurg Case Lessons
Journal of Neurosurgery: Case Lessons
2694-1902
American Association of Neurological Surgeons

10.3171/CASE2477
CASE2477
InfectionInfectionPituitary-SurgeryPituitary SurgerySkull-BaseSkull BaseTechniqueTechniqueSurgical-TechniqueSurgical TechniqueCase Lesson
Late meningitis and a nonabsorbable stent in recurrent Rathke’s cleft cyst: illustrative case
Serioli Simona MD 12
Mastropasqua Vincenzo MD 2
De Rosa Giorgia MS 2
Ebrahimian Romina MS 2
Mattogno Pierpaolo MD, PhD 3
Rigante Mario MD 4
Gaudino Simona MD 5
Maria Corsello Salvatore MD 67
Lauretti Liverana MD 23
Murri Rita MD 8
Gessi Marco MD, PhD 9
Olivi Alessandro MD 23
Doglietto Francesco MD, PhD 23
1 Division of Neurosurgery, Department of Medical and Surgical Specialties, Radiological Sciences, and Public Health, Spedali Civili of Brescia, University of Brescia, Brescia, Italy
2 Neurosurgery, Università Cattolica del Sacro Cuore, Rome, Italy
3 Neurosurgery, Fondazione Policlinico A. Gemelli IRCCS, Rome, Italy
4 Otorhinolaryngology and Head-Neck Surgery, Fondazione Policlinico A. Gemelli IRCCS, Università Cattolica del Sacro Cuore, Rome, Italy
5 Department of Radiological Sciences, Fondazione Policlinico A. Gemelli IRCCS, Università Cattolica del Sacro Cuore, Rome, Italy
6 Unit of Endocrinology, Department of Translational Medicine and Surgery, Fondazione Policlinico A. Gemelli IRCCS, Università Cattolica del Sacro Cuore, Rome, Italy
7 Unicamillus, Saint Camillus International University of Medical Sciences, Rome, Italy
8 Infectious Disease, Fondazione Policlinico A. Gemelli IRCCS, Università Cattolica del Sacro Cuore, Rome, Italy
9 Neuropathology, Fondazione Policlinico A. Gemelli IRCCS, Università Cattolica del Sacro Cuore, Rome, Italy
Correspondence Francesco Doglietto: Università Cattolica del Sacro Cuore, Fondazione Policlinico Universitario Agostino Gemelli IRCCS, Rome, Italy. francesco.doglietto@unicatt.it.
INCLUDE WHEN CITING Published September 23, 2024; DOI: 10.3171/CASE2477.

Disclosures Prof. Doglietto reported personal fees for consulting from Brainlab outside the submitted work.

23 9 2024
23 9 2024
8 13 CASE247707 2 2024
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© 2024 the authors
2024
the authors
https://creativecommons.org/licenses/by-nc-nd/4.0/ CC BY-NC-ND 4.0 (http://creativecommons.org/licenses/by-nc-nd/4.0/)

BACKGROUND

The management of recurrent Rathke’s cleft cysts (RCCs) remains challenging. The off-label application of steroid-eluting bioabsorbable or nonabsorbable intracystic stents has been recently described. Early outcomes and complications of this treatment have been described as well, but long-term data are lacking.

OBSERVATIONS

A 31-year-old woman who had undergone 3 prior transsphenoidal surgeries for recurrent RCC at other institutions was admitted for methicillin-susceptible Staphylococcus aureus meningitis. Five years earlier, a nonabsorbable stent had been placed in the cyst using a microsurgical transsphenoidal approach. RCC recurrence had been radiologically evident after 2 years but was clinically asymptomatic. Acute cerebrospinal fluid leakage and other causes of meningitis were ruled out; the stent was occluded. After antibiotic therapy and resolution of the meningitis, the patient underwent endoscopic removal of the catheter, excision of the cyst, and reconstruction with a nasoseptal flap. At the 18-month follow-up, she remained neurologically intact, under replacement therapy, and with no signs of recurrence.

LESSONS

The authors report the first case of late meningitis due to a nonabsorbable stent positioned for the treatment of a recurrent RCC. Currently, data on the long-term outcomes, failure rates, and complications of stents for RCC are limited.

https://thejns.org/doi/10.3171/CASE2477

case report
intracystic stent
meningitis
nonabsorbable device
recurrent Rathke’s cleft cyst
ABBREVIATIONS

CRP = C-reactive protein
CSF = cerebrospinal fluid
DI = diabetes insipidus
EETS = endoscopic endonasal transsphenoidal
MRI = magnetic resonance imaging
MSSA = methicillin-susceptible Staphylococcus aureus
MST = microscopic sublabial transsphenoidal
RCC = Rathke cleft cyst.
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pmcRathke’s cleft cysts (RCCs) are benign lesions of the sellar region arising from epithelial residuals in the pars intermedia of the pituitary gland and are found in 12%–33% of autopsies.1, 2 They are usually silent, especially when smaller than 3 mm; hence, they are typically found incidentally.2–4 When cases are symptomatic, the most common presentation is characterized by headache, visual field deficit, diabetes insipidus (DI), and anterior hypopituitarism.1–5

Surgical treatment is indicated for symptomatic patients or for patients with progressive cyst enlargement.2, 5 The endoscopic endonasal transsphenoidal (EETS) approach is the preferred technique to partially remove the cyst wall, drain its contents, and properly reconstruct the sella, according to the occurrence and the entity of intraoperative cerebrospinal fluid (CSF) leakage.2, 6

Despite surgical intervention, recurrence rates vary from 14% to 39.6%, with risk factors including cyst size, suprasellar localization, and metaplasia signs on histopathology.7–9 More aggressive removal of the cyst capsule could reduce the recurrence rate but can lead to higher rates of complications, including hypopituitarism, DI, CSF leakage, and meningitis.4–10 Recently, in a few cases, the off-label use of a bioabsorbable steroid stent, positioned between the cyst and the sphenoid sinus, has been described and proven effective in preventing the early recurrence of RCC; however, these stents tend to completely dissolve after 3 months.11–14 Nonabsorbable stents have also been used in transsphenoidal surgery and have proven to be a safe and effective option at the 1-year follow-up, as detailed in a recent case report featuring the endoscopic approach.15 Nonetheless, data on the long-term efficacy of stents in RCC, the failure rates, and the late complications are limited.

Herein, we describe in detail the case of a 31-year-old patient with a history of multiple recurrences of RCC, who presented with meningitis 5 years after the placement of a nonabsorbable intracystic stent through a microscopic sublabial transsphenoidal (MST) approach.

Illustrative Case

A 31-year-old woman presented to our emergency department with signs and symptoms of meningitis. She had a history of recurrent RCC, first diagnosed at the age of 21 years and had undergone 3 MST resections over the subsequent 5 years at another institution. At the third MST surgery, a nonresorbable intralesional stent with a cross shape was placed to drain the cystic contents into the sphenoid sinus (Fig. 1A–C). After the surgery, the patient developed transient DI, hypopituitarism, and secondary amenorrhea, which were treated with replacement therapy. Three-month postoperative brain magnetic resonance imaging (MRI) documented a complete collapse of the cystic lesion (Fig. 1D–F). At the 18-month radiological follow-up, recurrence of the cyst was evident in the sellar region, although the patient was clinically stable. Three months prior to presentation, MRI had documented further progression of the cyst, with cranial displacement of the pituitary stalk but limited mass effect on the chiasm (Fig. 1G–I). FIG. 1. Preoperative coronal T2-weighted (A) and coronal (B) and sagittal (C) T1-weighted contrast-enhanced MRI performed before intracystic stent placement, showing the recurrent RCC. Postoperative coronal T2-weighted (D) and coronal (E) and sagittal (F) T1-weighted contrast-enhanced MRI after positioning the nonresorbable stent, with the complete collapse of the lesion. Follow-up sagittal T2-weighted (G) and coronal (H) and sagittal (I) T1-weighted contrast-enhanced MRI 3 months prior to presentation showed further development of the RCC.

In the following weeks, the patient began to complain of worsening headaches, nausea, and vomiting. Because of the deterioration of neurological symptoms and hyperpyrexia, the patient was taken to the emergency department. On physical examination, she presented with rigor nucalis without other neurological findings. Brain computed tomography excluded signs of hydrocephalus and intracranial bleeding.

A blood test showed an increased level of C-reactive protein (CRP; 160.2 mg/L). The CSF analysis revealed hypoglycorrhachia (13 mg/dL; blood value: 111 mg/dL), high protein levels (180 mg/dL), and the diffuse presence of polymorphonuclear cells (Table 1). The CSF culture was positive for methicillin-susceptible Staphylococcus aureus (MSSA), so the patient was specifically treated with linezolid following a short course of empirical therapy with ceftriaxone, vancomycin, acyclovir, and ampicillin. TABLE 1. Laboratory values: parameters and reference range of CSF and blood analyses in the diagnosis of bacterial meningitis

Test	Result	Reference Range	
Blood			
 CRP	160.2 mg/L	<5 mg/L	
 WBC cell	6.60 × 109/L	4.5–11.0 × 109/L	
 Fibrinogen	555 mg/dL	200–400 mg/dL	
CSF			
 Glycorrhachia	13 mg/dL (blood value: 111 mg/dL)	40–70 mg/dL	
 Protidorrhachia	180 mg/dL	20–45 mg/dL	
 CSF culture	MSSA		
WBC = white blood cell.

Brain MRI documented diffuse leptomeningeal enhancement of the supra- and infratentorial spaces and minimal growth of the cystic lesion without displacement of the intracystic stent (Fig. 2A–C). An endoscopic endonasal evaluation confirmed obstruction of the distal part of the catheter. Superinfection of the stent was then suspected as the source of the meningitis, and after a multidisciplinary evaluation, it was decided to surgically remove it. FIG. 2. Coronal T2-weighted (A) and coronal (B) and sagittal (C) T1-weighted contrast-enhanced MRI at the diagnosis of meningitis. An intracystic stent with a cross shape and minimal increase in the size of the RCC appear in the sellar region. Coronal T2-weighted (D) and coronal (E) and sagittal (F) T1-weighted contrast-enhanced MRI after 18 months. No sign of residual RCC was found.

Once the acute infection was resolved, the patient underwent an EETS approach (Fig. 3A). A pedicled mucosal flap was prepared on the left side, with recovery at the level of the sphenoid (Fig. 3B). CSF leakage from the catheter was evident, as well as the typical purulent-like contents of the RCC. The intracystic stent, consisting of a horizontal and a vertical portion, was removed without complications (Fig. 3C and D). The cyst, which was adherent to the pituitary gland, was emptied, and the capsule was coagulated. After these maneuvers, no CSF leakage was recognized, the walls of the cyst were compacted, and the mucosal flap was used to cover the defect. The histological examination confirmed the diagnosis of RCC, documented inflammatory changes, and ruled out local metaplasia. FIG. 3. Intraoperative endoscopic images of stent removal during the EETS approach. A view (A) of the distal end of the intracystic catheter at the level of the nasal cavity, surrounded by crusting. A pedunculated mucosal flap was created (B). The vertical (C) and horizontal (D) portions of the nonabsorbable device were removed, and the cyst was emptied.

After surgery, the patient had no signs of CSF rhinorrhea but developed transient DI. She was discharged on the 6th postoperative day. Eighteen months after surgery, MRI showed no signs of recurrence (Fig. 2D–F). The patient remains on replacement therapy for anterior hypopituitarism and is neurologically intact.

Patient Informed Consent

The necessary patient informed consent was obtained in this study.

Discussion

Several surgical techniques have been described and refined to reduce the recurrence rate of RCCs while keeping the risk of new hypopituitarism low. Complete removal and coagulation of the cyst walls are certainly among the most effective strategies, although it is associated with a higher risk of CSF leakage, DI, and hypopituitarism, which have a significant impact on the patient’s quality of life.4–10

Other techniques include marsupialization of the lesion, with wide opening of the anterior wall of the cyst, or fat grafting, to maintain patency of the cyst fenestration.7, 16 Intracystic instillation of pure ethanol has shown limited results with dramatic complications, such as blindness, anosmia, third cranial nerve palsy, and epileptic seizures in the case of subarachnoid diffusion.17 Chemical cauterization with intracystic bleomycin appears to be a promising treatment, although its toxic effect on the brain parenchyma warrants caution.18

Intralesional catheters are relatively novel devices for the treatment of recurrent RCCs, which are commonly used in ear, nose, and throat practice for the treatment of chronic rhinosinusitis or congenital choanal atresia.19–21 Classified into bioresorbable steroid-eluting11–14 and nonresorbable catheters,15 these intracystic stents are reported to work as a means of draining the cyst contents, reducing the rate of relapses after the marsupialization of RCCs.

Observations

The application of steroid-eluting bioabsorbable stents has been described in 4 papers with a total of 10 patients, highlighting promising initial results in the management of recurrent RCCs.11–14 The ability to be reabsorbed within 1–3 months avoids the need for surgical intervention to remove the stent.11–14 Furthermore, the concomitant release of steroids could have a beneficial effect on scar tissue, although the direct effect on the pituitary gland must be considered.11–14 One of the relevant limitations of these studies is the short-term follow-up, except in the study by Wong et al., who documented neither radiological recurrences nor postprocedural or late complications with a mean follow-up of 56 ± 12 months.11

An early application of nonresorbable cystosphenoidal stents for RCC was described by Byrd et al. in 5 patients. In 3 cases, the catheter was removed after 29–34 days, with a mean follow-up of only 6.8 months.22 Recently, Ellens et al. described the application of a tympanostomy T-tube as a nonabsorbable intrasellar stent, very similar to the one that was positioned in our patient, with no signs of clinical or radiological recurrence or complications at 1 year.15 Although nonresorbable stents can provide a long-lasting response, overcoming the limitations of resorbable catheters, one should consider the risks of superinfection, long-term malfunction as in the case presented herein, or potential dislocation with traumatic injury to the adjacent neurovascular structures.

Lessons

Both resorbable and nonresorbable intracystic stents could represent valid therapeutic tools for RCC recurrences, according to the aforementioned initial reports. However, long-term outcomes and potential complications are not yet well known because of the generally small sample sizes and short follow-up times described. The case presented herein documents, for the first time, a late-onset malfunction of a nonabsorbable intracystic catheter, with subsequent acute meningitis, which required the surgical removal of the device. We hypothesize that complications related to the use of intralesional catheters may be more common than currently described in the literature, especially in the case of nonresorbable stents. Therefore, further studies with significantly longer follow-up durations and larger sample sizes are required to evaluate the long-term efficacy and safety of this therapeutic option.

Disclosures

Prof. Doglietto reported personal fees for consulting from Brainlab outside the submitted work.

Author Contributions

Conception and design: Doglietto, Serioli, Gaudino. Acquisition of data: Doglietto, Serioli, Mastropasqua, Corsello, Gessi. Analysis and interpretation of data: Doglietto, Serioli, Mastropasqua, Gessi. Drafting the article: Doglietto, Serioli, De Rosa, Ebrahimian. Critically revising the article: Doglietto, Serioli, Mastropasqua, Mattogno, Rigante, Corsello, Lauretti, Olivi. Reviewed submitted version of manuscript: Doglietto, Serioli, Mattogno, Rigante, Gaudino, Murri. Approved the final version of the manuscript on behalf of all authors: Doglietto. Administrative/technical/material support: Doglietto. Study supervision: Doglietto, Serioli, Rigante, Gaudino.

Correspondence

Francesco Doglietto: Università Cattolica del Sacro Cuore, Fondazione Policlinico Universitario Agostino Gemelli IRCCS, Rome, Italy. francesco.doglietto@unicatt.it.
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