Endoscopic transoral complete paediatric vallecular cyst excision and correction of retroversion of the epiglottis to improve outcomes

Introduction

Vallecular cyst is an important cause of upper airway obstruction in neonates and infants. Surgical intervention is the primary treatment approach for this condition. Various surgical techniques have been proposed, such as marsupialization, deroofing, laser ablation, coblation, and microdebrider (1). The manifestations of vallecular cyst typically occur within the initial few weeks after birth. The diagnosis can be obtained through flexible endoscopy, supported by imaging such as neck ultrasound (US) and magnetic resonance imaging (MRI), which help exclude other lesions like lingual thyroid or thyroglossal duct cyst, and confirmed by direct laryngoscopy under general anaesthesia (GA). Common symptoms include feeding difficulties, noisy breathing, sleep disturbances, and failure to thrive (2). It is not uncommon for these early symptoms to initially be misattributed to laryngomalacia, which is the most prevalent cause of noisy breathing among infants (3).

The underlying mechanisms by which vallecular cysts contribute to airway constriction and feeding problems resemble those seen in severe cases of Laryngomalacia. These include mass compression on the airways resulting in narrowing effects, dynamic supraglottic collapse, retroversion of the epiglottis, swallowing dysfunction, increased intrathoracic negative pressure as well as reflux (4).

The surgical outcomes of vallecular cyst excision can be variable, with postoperative stridor, respiratory distress, and even the need for intubation and intensive care unit (ICU) admission being not uncommon occurrences. Therefore, it is essential to refine the surgical technique and address all relevant anatomical abnormalities to minimize postoperative respiratory complications (3,5,6).

The author observed that the vallecular cyst can lead to backward movement of the epiglottis particularly during inspiration and even after surgical removal of the cyst, this issue may persist and contribute to ongoing airway obstruction. Therefore, the author believes it is important to address this anatomical abnormality simultaneously with surgery to effectively treat the condition.

The purpose of this study is to share our clinical experience in treating vallecular cysts at a tertiary paediatric hospital and outline the surgical technique utilized to enhance patient outcomes.

Methods

Study population

The study was reported according to the STROBE reporting guidelines and the SUPER reporting guidelines (https://www.theajo.com/article/view/10.21037/ajo-24-63/rc). A cohort of patients who underwent endoscopic excision of vallecular cysts were included from April 2021 to March 2023 at Cairo University Children’s Hospital. Evaluative data were collected in a retrospective fashion from patients with vallecular cyst surgically treated by a single surgeon. Cases with other airway anomalies or significant comorbidities, children older than 6 months, and those experiencing recurrent cysts after previous surgery done elsewhere were excluded from the study population. Such exclusion criteria were applied to ensure a relatively homogenous study population and limit the impact of cofounding factors. All surgical procedures were performed by the author as well as an assistant who held the scope using a standardized technique for all cases.

Preoperative assessment

The preoperative assessment encompassed gathering a comprehensive medical history including symptoms such as noisy breathing, feeding challenges, sleep disturbances and inadequate weight gain. The outpatient flexible endoscopy confirmed the presence of a vallecular cyst and the behaviour of the epiglottis and supraglottic structures was observed. Additional diagnostic imaging was not needed due to the distinct appearance of the smooth-surfaced superficial exophytic cyst in its characteristic location. Swallowing assessment revealed dysphagia due to poor respiratory support and disruption of the coordination between swallowing and breathing. Patients in this cohort didn’t require specific preoperative preparation. After discussing the surgical procedure with the parents, surgery was scheduled for the patient. Consent for the use of anonymous surgical images and videos was obtained from all patients.

Surgical technique

• The surgery is done under GA in the operating room with the anaesthetist in theatre.
• Patients were intubated with an age-appropriate endotracheal tube (ETT) using a video laryngoscope to enhance visualization.
• Using head ring and shoulder roll, patients were placed in suspension using a paediatric laryngoscope (Karl Storz 8587A Lindholm Operating Laryngoscope, Tuttlingen, Germany).
• All procedures were performed by the senior author, with an assistant positioned on the left side, holding a zero-degree 4 mm Hopkins Rod endoscope and camera, allowing the primary surgeon to operate using both hands.
• A thorough inspection of the pharynx and larynx was performed to confirm the diagnosis, assess the cyst, its extensions, and any concomitant anomalies (Figure 1, Video 1).
  

  Figure 1 Excision of the cyst. (A) Examination of a large vallecular cyst after intubation and laryngeal suspension. (B) After needle aspiration for cyst decompression and demonstration of cyst extensions. (C) After complete excision by cold steel. Note the retroversion of the epiglottis.
• Topical adrenaline (1:10,000) was applied to minimize bleeding.
• A 22-gauge needle was used to aspirate the cyst (Figure 1, Video 2).
• The cyst is then completely excised using microlaryngoscopic instruments (Figure 1, Video 3).
• Following cyst removal, the ETT was withdrawn, and the level of anaesthesia reduced to allow for adequate spontaneous respiration and assessment of dynamic airway obstruction.
• Additional steps to address the epiglottis included:
  • The base of the excised cyst on the tongue was gently cauterized (Figure 2, Video 4).
    

    Figure 2 Addressing retroversion of the epiglottis. (A) Cauterized base of tongue (cyst). (B) Point 1 (mid-point) cauterization LSE. (C) Point 2 left lateral cauterization LSE. (D) Point 3 right lateral cauterization LSE. (E) Divided AE folds. (F) Epiglottis retroversion corrected. AE, aryepiglottic; LSE, lingual surface of epiglottis.
  • Three-point cauterization of the lingual surface of the epiglottis. One in the middle and two on both sides. This step was done, and efficiency was judged by the anteversion of the epiglottis till the anterior commissure was seen without changing the position of the laryngoscope. A laryngeal suction diathermy was used (Figure 3) for this purpose with a power setting of 10 watts for 2–3 seconds (Figure 2, Videos 5-7 ).
    

    Figure 3 Laryngeal suction diathermy.
• Division of aryepiglottic (AE) folds to help epiglottis release was then carried out (Video 8).
• Haemostasis was then achieved using adrenaline 1:10,000 on neuro patties applied topically and cauterization if necessary.

Postoperative course

All patients routinely received adrenaline nebulizer in the recovery room and two doses of dexamethasone (0.2 mg/kg/dose) postoperatively at 8 and 16 hours. Following their surgery, swallow assessment included parent interview, thorough history, physical examination, and feeding observation. Pharyngeal phase was specifically evaluated for adequacy of triggering, evacuation and airway protection. This assessment involved both a clinical evaluation conducted at the patient’s bedside as well as functional endoscopic evaluation of swallowing when necessary.

Demographic data, presenting symptoms, and operative times were extracted from patient records. Postoperative outcomes, including the need for intubation or ICU admission, postoperative complications, hospital stay duration, feeding outcomes, and recurrence, were collected from medical records.

All patients were scheduled for outpatient flexible endoscopy at 6 and 12 weeks post-surgery to monitor their progress. After 3 months, the parents were advised to schedule an appointment if they had any concerns or experienced symptoms suggestive of recurrence.

Statistical methods

Data were coded and entered using the Statistical Package for Social Sciences (SPSS) version 28 (IBM Corp., Armonk, NY, USA). Data was summarized using mean, standard deviation (SD), median, minimum, and maximum.

Ethical approval

This study was a retrospective review of patients treated at Cairo University Children’s Hospital. This study was approved by the scientific committee of the Paediatric Otolaryngology Department and the research ethics committee of Faculty of Medicine, Cairo University (N-279-2023). All data were anonymous and it’s impossible to identify patients from this report. All procedures performed in this work were in accordance with the Declaration of Helsinki and its subsequent amendments. Written informed consent was obtained from the patients for publication of this manuscript and any accompanying images and videos.

Results

A total of eleven patients underwent surgical removal of vallecular cysts using the aforementioned technique. The gender distribution among these patients and the preoperative symptoms were outlined in Tables 1,2. All individuals were born at full term without any complications during their neonatal period. The preoperative awake transnasal flexible endoscopy evaluation demonstrated that all patients had documented evidence of retroversion of the epiglottis in addition to the presence of the vallecular cyst. The surgical procedures had a mean duration of 78 minutes, with a range of 55 to 125 minutes. After the surgery, all patients were successfully extubated. The mean duration of hospitalization was 1.5 days, with a range from a single overnight stay to a maximum of three nights. Specifically, six patients were discharged the following day, four remained hospitalized for two nights, and one patient required a three-night stay. No patient needed intubation or ICU admission after the surgery. The statistical description of the study participants was demonstrated in Table 2.

As regards surgical complications, no major issues were documented postoperatively. Specifically, no incidents of primary or secondary bleeding or need for respiratory support.

Postoperative swallow assessment did not require videofluoroscopy for any of the patients prior to discharge. One patient temporarily relied on nasogastric tube feeding for 2 days due to inadequate airway protection, but subsequently transitioned back to oral intake and was discharged. The remaining patients did not report significant choking, intolerance, or prolonged feeding times. Flexible endoscopic evaluation of swallowing revealed a timely triggering of the swallowing reflex, with adequate bolus clearance and airway protection. Oral feeding was promptly initiated for these patients, who were then discharged.

Clinical and endoscopic evaluation of all patients at 6 and 12 weeks postoperatively (Figure 4) showed no evidence of cyst recurrence or persistent retroversion of epiglottis or supraglottic collapse. After 12 weeks till date, there has been no reported cases of patients returning with recurrent symptoms.

Figure 4 Flexible nasal endoscopy 6 weeks postoperative.

Discussion

Abercrombie (7) was the first to report laryngeal cyst in 1881 and called it a congenital cyst in a neonate. DeSanto divided such cysts into ductal and saccular cysts (8). In general, ductal cysts are more common and vallecular location is the most commonly encountered site clinically. Hence, vallecular cysts should be taken into consideration when assessing neonates and infants with aerodigestive symptoms (2). These cysts are typically present at birth, resulting in early symptoms (9). Pathologically, the cysts have fibrous walls lined by squamous or respiratory epithelium along with an underlying inflammatory infiltrate and seromucous glands (1,10). Recurrence is a common concern discussed in existing literature, focusing on identifying the most effective surgical techniques to minimize its incidence (1,11,12).

When it comes to diagnosing vallecular cysts, the use of flexible endoscopy is believed to be a valuable tool. This approach aligns with multiple studies that have been conducted (1-5). This procedure is important for the diagnosis and assessment of any associated abnormalities (6). Specifically, it is crucial to carefully observe the movement of the epiglottis as the patient breathes in. Our experience suggests that the retroversion of the epiglottis and its collapse into the laryngeal inlet usually persist after cyst removal and is probably responsible for persistent distress following surgery if left uncorrected. Moreover, Intraoperative meticulous evaluation and dynamic assessment of the airway before and after vallecular cyst excision enable the surgeon to directly observe any persistent epiglottic retroversion, a crucial step in optimizing surgical outcomes.

This study aims to highlight the importance of dealing with the epiglottis retroversion. Additional surgical steps are proposed to correct this abnormality. The author contends that this technical approach enhances postoperative outcomes by mitigating residual patient distress and reducing the demand for scarce paediatric intensive care resources at some medical centres.

The use of endoscope has some advantages over the microscope as it allows better illumination and visualization, a panoramic view of the operative field, better visualization of corners and hidden areas and thus help in complete removal of the cyst. A potential drawback of this proposed surgical approach is that the additional technical steps may extend the overall operative duration. Furthermore, the relatively constrained operating field may limit the ability to simultaneously manoeuvre both the endoscope and surgical instruments through the laryngoscope.

In a study by Wang et al. (3), they explored the use of endoscopic-assisted transoral coblation marsupialization in 165 patients with vallecular cysts. Their findings indicated that coblation offered benefits such as minimal tissue damage, shorter operation time, and low recurrence rate. However, while coblation is a gentle method for tissue removal, it may not allow precise whole cyst excision and may ablate normal tissues accidentally. It is worth noting that the authors relied on US imaging to confirm their diagnosis. In our case, additional imaging was deemed unnecessary due to characteristic cyst site and gross appearance. While the authors note that marsupialization can prevent epiglottic collapse in children with laryngomalacia type III due to the scar between the root of the tongue and epiglottic cartilage, their own study found contrasting results. Specifically, they reported that 6 out of 86 children (7%) remained intubated after the surgery, and the mean hospital stay was 7 days. In contrast, the present study showed no need for intubation and a shorter mean hospital stay of only 1.5 days, which was significantly shorter than other reports as well (5,6,13-15).

In a study conducted by Leibowitz et al. (6), the risk factors of vallecular cyst recurrence were explored in 11 children. It was found that four patients required intubation for a period ranging from 1 to 4 days after the procedure. Another report examined seven patients who underwent transoral complete excision, and they were extubated immediately following the surgery. These patients were then kept under observation overnight in the ICU, with an average length of inpatient stay after cyst removal being 9.5 days (ranging from 1 to 41 days). However, it should be noted that four out of these seven children had shorter postoperative stays of less than 2 days (13).

In 2013, Tsai et al. published a report on their 15-year experience treating vallecular cysts at a single centre. Out of the 28 infants included in the study, 8 required ICU admission for a duration of 1–7 days and 5 needed an ICU stay for 8–14 days. The average length of hospitalization was calculated to be approximately 12.8 days (14).

Prior research has suggested that airway obstruction in patients with vallecular cysts may continue even after the cysts are removed, which has been attributed to factors such as posterior displacement of the larynx (16), redundant epiglottis, or severe reflux (12). Simultaneous aryepiglottoplasty was necessary in two of the six cases reported in this series (16). However, this investigation contends that a systematic evaluation of the dynamic obstruction has not been undertaken previously, and therefore, it represents the inaugural study to investigate the epiglottis behaviour and propose a surgical management strategy.

Table 3 outlines the details from various studies regarding the postoperative course for cases that underwent vallecular cyst excision in different centres. The data include the number of cases, length of postoperative admission, duration of ICU stay, and duration of intubation.

Based on the results of the above-mentioned studies, it appears that most medical centres have only reported small case series, and many patients require intubation and admission to the ICU for an extended period. However, the author proposes a technique that involves a comprehensive endoscopic transoral excision of the cyst as well as three-point cauterizations of the epiglottis and division of AE folds. This approach has shown promise in optimizing surgical outcomes by minimizing postoperative complications such as intubation, ICU stays, and prolonged hospitalization.

A recent study utilizing a reconstructed three-dimensional geometric model of the upper airway, based on two-dimensional medical images of a patient with vallecular cyst, emphasized the significance of addressing the retroversion of the epiglottis to enhance airflow. The authors suggest that the state of the epiglottis must be carefully evaluated to determine the necessity of performing additional supraglottoplasty in patients with vallecular cysts concomitant with laryngomalacia (17). These findings align with the refinements to the surgical approach proposed in the current investigation.

The study has some limitations that should be acknowledged. The data collection was retrospective in nature, which introduces potential selection biases. Additionally, the number of study participants was relatively small without controls for comparison and the report was from a single medical institution.

Despite these limitations, the findings from this cohort provide valuable insights and pave the way for more extensive future multi-centre studies with larger patient cohorts to corroborate the authors’ technical recommendations and assess their efficacy in improving outcomes for patients with vallecular cysts. In addition, these studies should aid in determining the optimal surgical approach for vallecular cysts.

Conclusions

Vallecular cysts should be included in the diagnostic assessment of neonates and infants presenting with stridor and feeding difficulties. Close consideration should be given to dynamic airway assessment, particularly for epiglottic retroversion. Comprehensive surgical removal of the cyst via transoral endoscopic approaches, along with addressing any concurrent anatomical abnormalities such as epiglottic retroversion, may enhance the postoperative course.

Acknowledgments

None.

Footnote

Reporting Checklist: The author has completed the STROBE and the SUPER reporting checklists. Available at https://www.theajo.com/article/view/10.21037/ajo-24-63/rc

Data Sharing Statement: Available at https://www.theajo.com/article/view/10.21037/ajo-24-63/dss

Peer Review File: Available at https://www.theajo.com/article/view/10.21037/ajo-24-63/prf

Funding: None.

Conflicts of Interest: The author has completed the ICMJE uniform disclosure form (available at https://www.theajo.com/article/view/10.21037/ajo-24-63/coif). The author has no conflicts of interest to declare.

Ethical Statement: The author is accountable for all aspects of the work in ensuring that questions related to the accuracy or integrity of any part of the work are appropriately investigated and resolved. This study was approved by the scientific committee of the Paediatric Otolaryngology Department and the research ethics committee of Faculty of Medicine, Cairo University (N-279-2023). All data were anonymous and it’s impossible to identify patients from this report. All procedures performed in this work were in accordance with the Declaration of Helsinki and its subsequent amendments. Written informed consent was obtained from the patients for publication of this manuscript and any accompanying images and videos.

Open Access Statement: This is an Open Access article distributed in accordance with the Creative Commons Attribution-NonCommercial-NoDerivs 4.0 International License (CC BY-NC-ND 4.0), which permits the non-commercial replication and distribution of the article with the strict proviso that no changes or edits are made and the original work is properly cited (including links to both the formal publication through the relevant DOI and the license). See: https://creativecommons.org/licenses/by-nc-nd/4.0/.

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