Authors: Kensuke Ikeda, Kuniaki Saito, Nobuyoshi Sasaki, Keiichi Kobayashi, Motoo Nagane
Categories: Case Lesson, adult brainstem gliomas, aspiration prevention surgeries, palliative care, tracheostomy, ALS = amyotrophic lateral sclerosis, FLAIR = fluid-attenuated inversion recovery, QOL = quality of life
Source: Journal of Neurosurgery: Case Lessons
Doi: 10.3171/CASE25284
Adult patients with brainstem gliomas have a poor prognosis, with a median survival of approximately 1 year. Although patients often remain conscious until the end of life, lesions affecting the medulla oblongata can cause severe dysphagia and increase the risk of aspiration and suffocation. These symptoms significantly reduce quality of life (QOL) during advanced stages. While aspiration prevention surgery is often performed for severe dysphagia in other conditions, its efficacy in adult patients with brainstem gliomas remains unclear.
This report presents 2 cases of adult patients with malignant brainstem gliomas who develoved progressive dysphagia. In case 1, glottic closure was performed as an aspiration prevention surgery; however, postoperative complications delayed the resumption of oral feeding, and the patient died due to tumor progression. In case 2, total laryngectomy was performed as an aspiration prevention surgery, which facilitated long-term oral intake and resulted in a good functional outcome, despite subsequent disease progression.
Aspiration prevention surgery may benefit selected adult patients with malignant brainstem gliomas. However, potential complications must be carefully considered. Further studies with larger sample sizes are needed to assess the impact of this intervention on QOL and clinical outcomes.
https://thejns.org/doi/10.3171/CASE25284
Keywords: adult brainstem gliomas, aspiration prevention surgeries, palliative care, tracheostomy
ABBREVIATIONS: ALS = amyotrophic lateral sclerosis, FLAIR = fluid-attenuated inversion recovery, QOL = quality of life
Adult patients with brainstem gliomas have a poor prognosis, with a median survival of approximately 1 year.^1^^,^^2^ When lesions extend to the medulla oblongata, they may affect the lower cranial nerves and the respiratory center, leading to dysphagia, dysarthria, and eventually respiratory paralysis.^3^ Conversely, when the lesion remains confined to the brainstem, higher cerebral functions are often preserved, and patients may remain conscious until the final stages of illness. For patients who remain alert, the inability to eat orally and the fear of suffocation can severely impair quality of life (QOL), posing significant challenges in palliative care.^4^
Surgical interventions to prevent aspiration are commonly performed to reduce the risk of aspiration pneumonia and suffocation associated with severe dysphagia. While various reports have discussed the indications and techniques for such procedures, no prior study has specifically evaluated their effectiveness in adult patients with malignant brainstem gliomas.^5^
In this report, we present 2 illustrative cases of adult patients with malignant brainstem gliomas who underwent aspiration prevention surgery. The aim is to assess the potential benefits and risks of this intervention in this unique clinical population. This research was approved by the Medicine Research Ethics Committee of Kyorin University School of Medicine. The necessary informed consent was obtained in this study.
A 54-year-old male had a history of right hemiparesis diagnosed 13 years earlier. MRI revealed a tumor in the medulla oblongata, and biopsy confirmed a low-grade glioma. He underwent radiation therapy (54 Gy in 30 fractions), which initially led to tumor shrinkage. Three years prior to admission, tumor progression was noted, and maintenance chemotherapy with temozolomide was initiated. The patient presented to our hospital with worsening dysphagia, dysarthria, and right hemiparesis. On admission, his Glasgow Coma Scale score was E4V5M6. MRI revealed further tumor enlargement (Fig. 1). Bevacizumab monotherapy was initiated on the 2nd day of hospitalization, with the second dose administered on the 16th day.
FIG. 1. Case 1. Postadmission axial (A), sagittal (B), and coronal (C) contrast-enhanced MR images revealing multiple contrast-enhancing lesions in the medulla. Corresponding axial (D), sagittal (E), and coronal (F) FLAIR MR images demonstrating high signal intensity in the medulla.
On the 28th day of hospitalization, the patient developed aspiration pneumonia requiring intubation. A tracheostomy was performed on day 34, and he was weaned off the ventilator the following day. The preoperative blood test revealed a serum albumin level of 2.5 g/dL, indicating nutritional deficiencies associated with dysphagia and aspiration pneumonia. Tracheostomy secures the airway and facilitates tracheal suctioning, but it does not completely prevent aspiration pneumonia. In contrast, aspiration prevention surgery could completely prevent aspiration pneumonia, potentially enabling him to resume oral intake at the cost of losing speech. After discussing this with the patient following extubation, he took some time to consider his options and ultimately expressed his desire to resume oral intake, fully understanding that this would result in the loss of speech. Aspiration prevention surgery was performed on day 50, as more than 30 days had passed since the last administration of bevacizumab monotherapy. Given the limited prognosis, a more minimally invasive procedure, glottic closure, was selected. However, postoperative wound infection and suture dehiscence occurred, necessitating total laryngectomy on day 92 (Fig. 2A). He resumed oral intake on day 146, although he lost his speech. His progressive respiratory muscle paralysis, however, required ventilator support again on day 176. He died on day 278 due to tumor progression.
FIG. 2. Sagittal CT images of the neck region obtained in case 1 (A) and case 2 (B) following total laryngectomy. The arrows indicate the trachea, and the asterisks indicate the esophagus. Because of the surgery, the trachea and esophagus were completely separated, helping to prevent aspiration.
A 47-year-old male presented with truncal ataxia and dysarthria. Fluid-attenuated inversion recovery (FLAIR) MRI revealed tumors with high signal intensity in the pons and cerebellum. Biopsy confirmed a high-grade glioma. He received radiation therapy with temozolomide, followed by bevacizumab due to early disease progression. Three years later, he presented with fever and respiratory distress, which occurred 29 days after the last administration of bevacizumab. On admission, he showed dysphagia, dysarthria, and bilateral coordination deficits. The blood test revealed a serum albumin level of 2.5 g/dL, indicating nutritional deficiencies associated with dysphagia. A chest radiograph revealed aspiration pneumonia. Bevacizumab monotherapy was discontinued. After antibiotic treatment, the pneumonia improved. Follow-up MRI revealed progression of the lesions involving the midbrain, pons, and cerebellar vermis (Fig. 3). Nasopharyngolaryngoscopy revealed bilateral vocal cord fixation and severe aspiration. He wished to regain oral intake, fully understanding that this would result in the loss of speech. Aspiration prevention surgery was performed on day 41, as more than 30 days had passed since the last administration of bevacizumab monotherapy (Fig. 2B). Because of his poor nutritional status, similar to case 1, the risk of postoperative complications, such as wound infection and suture dehiscence, was considered high. Therefore, rather than performing the minimally invasive glottic closure, total laryngectomy was chosen for a more reliable separation of the airway and esophagus. He resumed oral intake on day 54, losing his speech, and was discharged on day 109. Bevacizumab monotherapy was continued after discharge, and the disease remained stable. However, 274 days later, he was rehospitalized for tongue ulcers induced by bevacizumab. The ulcers resolved after discontinuation of the drug, and he resumed oral intake. The patient was discharged on day 11 and died 29 days later due to respiratory failure caused by tumor progression.
FIG. 3. Case 2. Postadmission axial (A), coronal (B), and sagittal (C) FLAIR MR images demonstrating high signal intensity in the midbrain, pons, and cerebellar vermis.
The necessary informed consent was obtained in this study.
This report explored the potential benefits of aspiration prevention surgery in adult patients with malignant brainstem gliomas. Although patients with brainstem gliomas generally remain conscious, severe dysphagia increases the risk of aspiration pneumonia and suffocation, making continued oral intake difficult.^3^^–^^7^ These cases suggest that aspiration prevention surgery may be a potentially useful intervention for such patients.
We retrospectively analyzed the records of 15 adult patients with malignant brainstem glioma treated at our institution between May 2014 and May 2023. The median overall survival was 286 days, and the median survival after the onset of dysphagia was 167 days, accounting for approximately 60% of total survival time. These data indicate that adult patients with malignant brainstem glioma live with dysphagia for more than half of their remaining life. Given that difficulty swallowing and fear of suffocation can significantly reduce QOL, appropriate palliative intervention to address dysphagia should be considered.
Aspiration prevention surgery is a surgical intervention designed to prevent the entry of saliva, ingested materials, and gastric contents into the airway, thereby reducing the risk of potentially life-threatening complications such as aspiration pneumonia. Tracheostomy is a common procedure for patients with dysphagia, facilitating the suctioning of tracheal secretions and helping to reduce the risk of aspiration pneumonia and suffocation. However, cuffed tracheostomy tubes alone do not provide complete protection against aspiration, and their effectiveness is limited. Since the 1970s, various surgical techniques have been developed to anatomically separate the respiratory and digestive tracts to prevent aspiration. Initially, total laryngectomy, which was performed for advanced laryngeal and pharyngeal cancers, was repurposed as one of the earliest methods for aspiration prevention. In 1972, Habal and Murray introduced the epiglottic flap technique, in which the epiglottis is sutured posteriorly to close the laryngeal inlet, marking the first surgery specifically designed to prevent aspiration.^8^ In subsequent years, less invasive alternatives were developed, including glottic closure, which is now considered a standard aspiration prevention surgery, and central-part laryngectomy. These techniques aim to improve functional outcomes while minimizing surgical morbidity. Aspiration prevention surgery leads to the resumption of oral intake in approximately 78% of patients, while reducing the frequency of suctioning in 85%. These findings support the clinical efficacy of aspiration prevention surgery as an effective strategy for preventing aspiration.^5^^9^
In cases 1 and 2, aspiration prevention surgery reduced the risk of aspiration pneumonia and suffocation and enabled the patients to resume oral intake, albeit at the cost of speech loss. Particularly for younger patients who remain conscious and motivated to eat orally, this procedure may improve satisfaction and contribute to overall QOL. Previous studies have reported on the benefits of aspiration prevention surgery in patients with amyotrophic lateral sclerosis (ALS), especially among younger individuals with good nutritional status and preserved communication abilities before the procedure.^9^^,^^11^ The median age at diagnosis of adult patients with malignant brainstem gliomas (44 years) is younger than that of patients with high-grade gliomas in general (60 years).^11^^,^^13^ In addition, patients with brainstem gliomas often remain conscious and maintain their desire for oral intake.^3^^–^^6^ These findings suggest that aspiration prevention surgery may be particularly beneficial in relatively young, conscious patients with malignant brainstem gliomas.
However, several limitations must be considered. First, although it enables oral intake, the surgery typically results in loss of speech. Glottic closure leads to permanent loss of speech because the vocal cord mucosa is not preserved.^14^ In contrast, a total laryngectomy preserves the vocal cord mucosa and allows for potential recovery of tracheoesophageal speech via tracheoesophageal puncture. However, acquiring this form of speech requires training and time, with a success rate of less than one-third. Given the poor prognosis of adult brainstem glioma patients, there is often insufficient time for this process, making speech recovery highly challenging.^1^^,^^2^^,^^15^
Second, postoperative complications may delay or prevent the resumption of oral intake. In case 1, wound infection and dehiscence required reoperation, which may have been attributed to impaired wound healing due to bevacizumab treatment or nutritional deficiencies caused by prolonged dysphagia. There are no definitive guidelines regarding the recommended interval between bevacizumab treatment and surgery. In our institution, we generally discontinue bevacizumab at least 30 days prior to surgery. However, according to previously published reports, it is recommended that bevacizumab be discontinued at least 60 days before surgery and that treatment be resumed 28 days after surgery.^16^ Glottic closure was performed 34 days after the second dose of bevacizumab monotherapy. The short duration of the bevacizumab discontinuation period may have contributed to the postoperative complications. Previous studies have reported a 10% complication rate in ALS, cerebrovascular disease, and head and neck cancer, but our findings suggest that this rate may not be low in patients with malignant brainstem gliomas.^9^
Third, even if oral intake is restored, it may be compromised by other factors. In case 2, tongue ulcers caused by bevacizumab temporarily impaired oral intake. This highlights that treatment-related side effects—not just dysphagia—can limit postoperative recovery. Therefore, eligibility for aspiration prevention surgery should be carefully evaluated in the context of treatment side effects and the patient’s overall condition.
The choice of surgical approach is another important consideration. Glottic closure, which can be performed under local anesthesia in less than 2 hours, is the least invasive option.^17^ Although suture loosening may occur due to vocal cord motion, it is a relatively low-burden intervention that can effectively prevent aspiration in malignant glioma patients.^18^ A total laryngectomy, although the most invasive approach, completely separates the trachea and esophagus and is widely used in head and neck surgery. It may be appropriate in high-risk cases, such as those with malnutrition or complications following other procedures. The optimal approach may vary depending on institutional preferences and require collaboration with head and neck surgeons.^5^
It is also important to compare aspiration prevention surgery with a tracheostomy, which is a less invasive alternative. While a tracheostomy does not fully separate the respiratory and digestive tracts and therefore cannot completely prevent aspiration pneumonia, it facilitates airway management, prevents suffocation, and improves the clearance of secretions. Moreover, it is easier and faster to perform than aspiration prevention surgery, making it a viable option for patients who do not seek to resume oral intake. With the addition of a speaking valve to a side-port tracheostomy tube, speech is possible, although the risk of aspiration must still be considered.^19^ Treatment decisions should be personalized on the basis of clinical status and patient preferences. This case series does not suggest that aspiration prevention surgery is superior to tracheostomy, but rather highlights its potential as one of the treatment options during the end-of-life phase.
This report also raises important questions about the appropriateness of invasive procedures in patients with limited life expectancy. Adult patients with malignant brainstem gliomas have a median survival of approximately only 1 year.^1^^,^^2^ It is therefore essential to carefully consider whether aspiration prevention surgery improves QOL or merely adds treatment burden. In case 1, the duration of oral intake was limited, and complications led to extended hospitalization. Even when indicated, aspiration prevention surgery should be offered only after thorough discussions with patients and families about its benefits, risks, and alignment with palliative care goals, which include symptom relief and dignity preservation. The importance of oral intake varies among individuals, and thus the value of aspiration prevention surgery in palliative care may also differ. Even in cases with poor prognosis, such as malignant brainstem gliomas in adults, it is crucial to present this option as part of palliative care and advanced care planning. Figure 4 shows a visual illustration demonstrating the balance of benefits and risks of aspiration prevention surgery. If the patient’s wishes are clearly expressed and the expected benefits are deemed to outweigh the risks, aspiration prevention surgery may be a reasonable and appropriate option (Fig. 4).
FIG. 4. Benefits and risks of aspiration prevention surgery. Aspiration prevention surgery offers several benefits, including the prevention of aspiration pneumonia and suffocation, the potential for regaining oral intake, and an improvement in QOL. On the other hand, the patient will inevitably lose the ability to speak. Furthermore, in the terminal stages, the procedure may be highly invasive, and even with surgery, there are risks that the patient may not gain significant benefits owing to postoperative complications, progression of the underlying disease, or side effects from chemotherapy. These factors should be carefully weighed, and the treatment plan must ultimately respect the patient’s desires.
It is advisable to consider the indication for aspiration prevention surgery at an early stage of the disease course. Poor nutritional status is associated with a higher risk of postoperative complications following such procedures.^20^ Therefore, it may be preferable to perform aspiration prevention surgery before the patient experiences repeated episodes of aspiration pneumonia, malnutrition, and overall physical decline. In this case series, delayed wound healing owing to bevacizumab monotherapy may have contributed to postoperative suture dehiscence, suggesting that the timing of surgery should also consider the phase of bevacizumab monotherapy. Careful assessment of the disease trajectory and treatment schedule is essential in determining the optimal timing for surgical intervention. To support shared decision-making, it is important to inform patients regarding the option of aspiration prevention surgery early in the clinical course and to explore their preferences in advance, particularly in cases in which progressive neurological decline or prolonged treatment with agents, such as bevacizumab, is anticipated.
Finally, several challenges remain. This case series is limited by a small sample size. Data on postoperative QOL and speech rehabilitation are also lacking. Future studies should address these gaps and explore strategies for risk reduction during chemotherapy, infection prevention, and optimization of wound healing. In the management of dysphagia, further research and development of less invasive approaches, such as swallowing rehabilitation, nutritional therapy, and pharmacological interventions, are warranted.
Aspiration prevention surgery may offer meaningful benefits for selected adult patients with malignant brainstem gliomas, particularly those who wish to continue oral intake. However, careful preoperative assessment is essential, as the procedure may lead to permanent speech loss and carries a risk of postoperative complications. Clinical decision-making should balance potential improvements in QOL against the patient’s overall condition, prognosis, and treatment burden. Further studies with larger cohorts are needed to more fully evaluate complication risks, long-term outcomes, and postoperative QOL.
ChatGPT was utilized exclusively for enhancing the readability and linguistic quality of the text.
Dr. Sasaki reported personal fees from ONO Pharmaceutical and UCB S.A. outside the submitted work.
Conception and Ikeda, Kobayashi, Nagane. Acquisition of Ikeda. Analysis and interpretation of all authors. Drafting the Ikeda, Sasaki. Critically revising the Ikeda, Saito, Sasaki, Nagane. Reviewed submitted version of Ikeda, Sasaki, Nagane. Approved the final version of the manuscript on behalf of all Ikeda. Statistical Ikeda. Administrative/technical/material Kobayashi. Study Saito, Kobayashi, Nagane.
Portions of this work were presented in abstract form at the 41st Meeting of the Japan Society for Neuro-Oncology, Niigata, Japan, December 3–5, 2023.
Kensuke Ikeda: Kyorin University School of Medicine, Tokyo, Japan. rafaga93.mk.bj18@gmail.com.