Management of nasopharyngeal stenosis post-tonsillectomy using the Madame Butterfly technique: A case report with novel approach

Abstract

Background

Nasopharyngeal stenosis (NPS) is an uncommon but significant complication that arise following tonsillectomy in adults, leading to manifestations including obstructive sleep apnea and dysphagia. This condition is often rare, underscoring the need for awareness and effective management strategies.

Case presentation

We present a case involving a 48-year-old female who developed nasopharyngeal stenosis (NPS) 15 months after undergoing a tonsillectomy. Her medical history was unremarkable, with no identifiable predisposing factors. She experienced respiratory distress, snoring, and dysphagia due to significant nasopharyngeal obstruction caused by fibrotic adhesions. The Madame Butterfly flap technique was employed, facilitating effective reconstruction while minimizing complications typically associated with skin grafts.

Discussion

The existing literature on NPS in adults is limited, particularly regarding standardized management protocols. Current treatment modalities exhibit considerable variability; however, few have undergone rigorous investigation within adult populations. The Madame Butterfly technique is particularly noteworthy for its ability to achieve both functional and aesthetic repair without the drawbacks associated with traditional grafting methods. This case underscores the necessity for comparative studies involving larger sample sizes to determine optimal management strategies for NPS.

Conclusion

This case exemplifies the successful implementation of the Madame Butterfly technique in managing NPS following tonsillectomy, with no recurrence observed during a six-month follow-up period. Given the rarity of NPS and the lack of established protocols, there is an urgent need for updated postoperative guidelines to mitigate this complication. Future research should prioritize the development of standardized management approaches to improve surgical outcomes for patients affected by NPS.

Highlights

• •Unique Technique: This report presents the Madame Butterfly graft for managing nasopharyngeal stenosis post-tonsillectomy.
• •Instructional Video: Our manuscript includes a rare instructional video demonstrating the surgical procedure for enhanced learning.
• •Successful Outcomes: The patient showed significant symptom relief with no stenosis recurrence at six-month follow-up, highlighting efficacy.
• •Rare Condition Awareness: The report stresses the need to recognize nasopharyngeal stenosis in adults and improve management strategies.
• •Call for Research: Findings indicate a need for larger studies to compare surgical techniques for optimal management of nasopharyngeal stenosis.

1. Introduction

Nasopharyngeal stenosis (NPS) is a relatively rare condition characterized by varying degrees of narrowing within the nasopharynx, specifically between the choana and the oropharynx, resulting from pathological processes. This complication arises from the development of scar tissue, which can present in several forms, including circumferential narrowing, webs, or bands.

The etiology of NPS is often linked to underlying diseases or traumatic events, particularly following surgical interventions such as uvulopalatopharyngoplasty, tonsillectomy, adenoidectomy, and pharyngeal reconstruction for velopharyngeal insufficiency [2].

The clinical manifestations associated with the development of nasopharyngeal adhesions encompass nasal obstruction, accumulation of nasal secretions, hyponasal speech, anosmia, rhinorrhea, and sleep apnea. Additionally, these symptoms may lead to complications such as acute otitis media and sinusitis, which are secondary to nasopharyngeal stenosis [3,4].

Historically, NPS was predominantly associated with tertiary syphilis affecting the oral cavity, pharynx, and palate, which were treated with caustic agents. Despite the availability of a wide array of therapeutic interventions, including surgical excision, the likelihood of recurrence remains significant. This report delineates the clinical presentation and management strategies employed for a patient diagnosed with partial NPS [5].

The occurrence of NPS predominantly affects the pediatric population following tonsillectomy, with or without adenoidectomy. In contrast, there is a paucity of documented cases in the adult population within the existing literature [[6], [7], [8]].

Nasopharyngeal stenosis can be addressed through a range of surgical interventions, which include the administration of triamcinolone injections, manual dilation techniques, surgical division accompanied by skin grafting, the utilization of local flaps such as pharyngeal or palatal mucosal flaps, as well as free flap procedures [8,9].

The “Madame Butterfly” procedure is a surgical intervention developed to rectify complications associated with lower eyelid blepharoplasty, specifically targeting eyelid retraction that may result in a condition referred to as “scleral show.” This approach functions as both a cosmetic and functional repair technique, circumventing the necessity for skin grafts, which frequently result in unsatisfactory aesthetic outcomes [22]. However, to the best of our knowledge, this method has not been employed to address NPS following tonsillectomy.

This study reports a case of an adult patient who experienced the formation of adhesions following a recent tonsillectomy. The patient presented with primary symptoms of respiratory distress, obstructive sleep apnea, dysphagia, and snoring. Our patient subsequently underwent a Madame Butterfly free everted palatal flap procedure (Also known as Madame Butterfly Procedure).

2. Methods

This manuscript was written in line with SCARE criteria [23].

3. Case presentation

A 48-year-old female patient of Iranian descent presented with respiratory distress, dysphagia, snoring, and sleep apnea. Notably, she had developed a tendency to sleep with her mouth open, which began one month after her tonsillectomy performed 15 months prior to her admission.

The patient's symptoms began one month following her tonsillectomy. Initially mild, the respiratory distress and dysphagia progressively worsened over time. The patient reported difficulty swallowing and frequent episodes of snoring, which were disruptive to her sleep and that of others. Despite these symptoms, she did not experience any fever or significant weight loss. Concerned about her deteriorating condition, she sought medical attention.

The patient had a surgical history notable only for the tonsillectomy performed 15 months earlier. There were no significant medical or surgical conditions reported prior to this surgery. She had no history of allergies or chronic medication use. Also, the patient's personal history was largely unremarkable, with no known exposure to infectious diseases or environmental toxins. Family history did not reveal any hereditary conditions or significant health issues that could relate to her current symptoms.

During the physical examination conducted at the facility, it was observed that the nasopharyngeal region was completely obstructed by adhesive tissue, leaving only a 0.5–1 cm area posterior to the uvula unaffected (Fig. 1). This condition resulted in the patient exhibiting symptoms of respiratory distress, characterized by increased effort in breathing and audible snoring during sleep. Consequently, further imaging techniques were deemed unnecessary. In addition, vital signs were stable: temperature at 36.5 °C, blood pressure at 120/80 mmHg, pulse rate at 80 beats per minute, and respiratory rate at 18 breaths per minute. No cyanosis or pallor was observed. The assessment of the oral cavity indicated the presence of fibrotic and adhesive tissue bands formed at the oropharyngeal region, resulting in the formation of a small circular opening. All laboratory results were normal, and the hemoglobin level was 13.8 g/dL (normal range: 12.0–15.5 g/dL). Hence, these findings indicated no immediate hematological or inflammatory concerns. The final diagnosis was confirmed as post-tonsillectomy nasopharyngeal adhesion leading to obstructive sleep apnea and dysphagia.

Fig. 1.

The adhesive lesion was diagnosed via nasoendoscopy following a thorough physical examination of the nasopharyngeal space. (Please refer to our instructive full video).

The adhesive lesion was diagnosed via nasoendoscopy following a thorough physical examination of the nasopharyngeal space. (Please refer to our instructive full video).

4. Surgical procedure and management

4.1. Stage one: assessment and confirmation of adhesion

An intraoral examination was conducted to evaluate the oropharynx following a previous tonsillectomy. The findings indicated that the posterior pillar was adherent to both the posterior oropharynx and the posterior margin of the soft palate. Notably, the posterior aspect of the uvula exhibited a single orifice, measuring approximately 0.5 to 1 cm in diameter. To ascertain whether this orifice was connected to the nasopharynx or represented a blind-ended space, a black Nelaton tube was employed, guided by nasoendoscopy through each nostril. This procedure confirmed that both Nelaton tubes successfully traversed the inferior choanae and emerged from the posterior orifice of the uvula.

4.2. Stage two: surgical preparation and flap creation

Upon confirming that the orifice communicated with the oropharynx, we proceeded with the Madame butterfly palatal flap technique. Initial markings for the flap were established, followed by the administration of local anesthesia using 2 % Lidocaine hydrochloride and 1:80,000 Epinephrine for bleeding control (Fig. 2).

Fig. 2.

The margins for Madame butterfly flap were designed (please refer to our video).

The margins for Madame butterfly flap were designed (please refer to our video).

The excision of scarred tissue and oropharyngeal mucosa was performed in the submucosal plane, ensuring the preservation of the underlying muscular layer. Careful detachment of the scarred tissue at the orifice's edge was executed, and the muscular component of the flap was sutured with Vicryl 3-0 thread to minimize the remaining mucosal area. The flap was designed with a width of 2 cm to ensure adequate tissue coverage while maintaining sufficient blood supply. Its length ranged from 3 to 4 cm to adequately cover the defect, tailored to our specific surgical needs. The thickness of the flap was maintained at 5–8 mm, providing the necessary vascularity and support. Furthermore, the rectangular shape of the flap was chosen, ensuring that the base was wider than the tip to optimize blood flow and enhance healing.

4.3. Stage three: mucosal repositioning

After gathering the muscle layers, the nasopharyngeal mucosa was repositioned and sutured to the mucosa at the periphery of the flap; in other words, the mucosal pharynx was everted and secured with sutures utilizing the tips of the previously harvested butterfly flap, employing 3–0 vicryl thread. This step is critical for ensuring proper healing and functionality of the newly established connection.

4.4. Stage four: placement of endotracheal tubes

Following the successful suturing of the fibrotic region, two uncuffed endotracheal tubes (ETT), size five, were introduced through each nostril into the oropharyngeal space. The proximal ends of these tubes were sutured together, and a Silastic sheet was affixed anteriorly to these tubes via suturing (See Fig. 3). This configuration is intended to maintain space and prevent the formation of new adhesions in the nasopharynx after the removal of the ETTs.

Fig. 3.

Final stage where uncuffed ETTs and Silastic sheets were inserted onto nostrils and fixed for two weeks.

4.5. Stage five: final adjustments and retention

The proximal segments of the ETTs were trimmed and sutured together, with sterile gauze placed beneath these sutures to protect against potential damage and infection in the columella region. The ETTs, along with the Silastic sheet secured to them, were retained for a duration of two weeks to facilitate healing. The justification for employing uncuffed ETTs is predicated on the notion that these tubes can mitigate the risk of further stenosis resulting from the pressure effects associated with inflated ETTs, which may potentially lead to necrosis. Furthermore, the application of silastic sheets functions to establish an intermediary between the floor and the roof of the nasopharyngeal space, thereby preventing the formation of additional adhesive tissue.

4.6. Stage six: removal of tubes and silastic sheet

After a two-week period, both endotracheal tubes and the Silastic sheet were carefully removed from the patient's oropharyngeal cavity under general anesthesia. This step concludes the surgical intervention aimed at effectively managing post-tonsillectomy adhesions.

4.7. Patient follow-up

The patient was scheduled for a follow-up appointment two weeks' post-surgery, at which time the tubes were removed, and no recurrence of stenosis was observed. Additionally, the patient was advised to return for an extended follow-up period of six months if any respiratory distress, dysphagia, or sleep apnea develops. The patient subsequently confirmed the absence of any recurrence of stenosis. However, after an additional one-month follow-up conducted through an interview at our clinic, the patient reported experiencing minimal fluid regurgitation. In conclusion, the patient was instructed to adhere to a soft diet and to utilize Mometasone nasal drops at a concentration of 0.05 %, with the recommended dosage being four drops administered every twelve hours for a duration of one month. This treatment protocol is designed to mitigate the post-surgical inflammatory response and to prevent the development of adhesions.

5. Discussion

Nasopharyngeal stenosis (NPS) represents a rare yet notable complication that may arise following tonsillectomy, especially in adult populations, where its occurrence is less frequently reported in comparison to pediatric cases. The underlying pathophysiological mechanisms of NPS typically involve the development of scar tissue as a consequence of surgical trauma, which can result in varying levels of airway obstruction and related symptoms, including dysphagia and sleep apnea [[10], [11], [12]].

The prevalence of nasopharyngeal stenosis (NPS) in adult populations is not well-defined, as the majority of existing literature predominantly addresses pediatric cases. A historical analysis has shown that NPS is primarily observed in children following tonsillectomy and adenoidectomy, with relatively few instances reported in adults. This disparity may contribute to the under-diagnosis or misdiagnosis of NPS in adult patients, highlighting the importance for clinicians to consider this condition when adults present with respiratory distress or dysphagia subsequent to tonsillectomy [13].

The formation of scar tissue subsequent to surgical procedures is affected by a multitude of factors, including the specific surgical techniques employed, the unique healing responses of individuals, and possible genetic predispositions. The emergence of fibrotic tissue may lead to considerable anatomical alterations, which can result in complications such as obstructive sleep apnea and chronic sinusitis. A comprehensive understanding of these mechanisms is essential for the formulation of preventive strategies and the enhancement of surgical outcomes [[14], [15], [16], [17], [18]].

Different approaches have been proposed for the treatment of nasopharyngeal stenosis but a unique and standardized management has not yet been presented [19].

It is important to note that not all treatment modalities are surgical in nature, and their implementation is contingent upon various factors, including the severity of stenosis, the patient's age, and overall clinical condition. Among the non-surgical interventions, the localized administration of triamcinolone acetonide has demonstrated efficacy in the management of mild pharyngeal stenosis, primarily due to its ability to promote collagen dissolution and mitigate keloid formation [20]. Nonetheless, surgical interventions tend to yield superior outcomes, with some options encompassing minimally invasive techniques, such as the reduction of pharyngeal scarring through plasma radio-frequency (Coblation) followed by the application of mitomycin C injections [21].

In certain instances, surgical procedures may be conducted utilizing local anesthesia. Some researchers have chosen to implement carbon dioxide scar lysis in conjunction with the application of a nasopharyngeal obturator, which is typically worn for a duration of two to six months. Additionally, local corticosteroid injections or subsequent balloon dilation may be employed to mitigate the recurrence of stenosis [22].

The most favorable outcomes appear to be achieved through the application of muscular-mucosal flaps, particularly in instances of significant stenosis. Various types of flaps have been suggested, each with distinct anatomical placements within the pharyngeal area [19]. In cases of severe stenosis, pharyngeal flap surgeries are typically recommended, as they may enhance patient tolerance relative to the use of nasopharyngeal obturators.

In the surgical management section of our manuscript, we describe the application of the Madame Butterfly Flap procedure in our patient to address the NPS that developed following tonsillectomy.

This particular technique facilitates the reconstruction of the nasopharyngeal region while reducing the likelihood of recurrence [23]. Alternative approaches, including manual dilation and the application of local flaps, have been documented; however, they may not achieve comparable long-term outcomes. The existing literature appears to be limited in its exploration of the most promising approaches for addressing rare complications associated with adult tonsillectomy surgeries. The authors believe that conducting a comparative analysis of these techniques within larger patient cohorts could yield significant insights into the most effective management strategies for NPS.

6. Conclusion

Nasopharyngeal stenosis represents a rare yet significant complication that may occur post-tonsillectomy in adults, potentially resulting in serious health concerns, including obstructive sleep apnea and respiratory distress. The existing literature does not provide definitive guidelines for effective surgical interventions; however, the novel application of a previously established technique referred to as the Butterfly Procedure has demonstrated promising outcomes, with no reported instances of stenosis recurrence within six months following the intervention.

To enhance the comprehension of this condition and to ascertain optimal management strategies, it is essential to undertake additional comparative studies with larger sample sizes. Such research will yield significant insights for surgical societies in the future. Furthermore, there is a notable deficiency in the literature concerning the establishment of standardized management protocols for nasopharyngeal stenosis in the adult population following tonsillectomy.

The following is the supplementary data related to this article.

Video 1

ENT Video.

Informed consent

Written informed consent was obtained from the patient's parents/legal guardian for publication and any accompanying images. A copy of the written consent is available for review by the Editor-in-Chief of this journal on request.

Research registration number

Non applicable.

Consent for publication

Yes.

Ethical consideration

This research has been granted an exemption from the requirement for ethical approval by the university's Ethical Committee.

Declaration of Generative AI and AI-assisted technologies in the writing process

During the preparation of this work the author used Wordvice.AI/Paraphrasing and proofreading tool in order to avoid either plagiarism or grammar errors. After using this tool/service, the author reviewed and edited the content as needed and takes full responsibility for the content of the publication.

Funding

None.

Authors' contribution

Amir Soltaniesmaeili developed the conceptual framework for the study and performed the surgical procedure, while Fateme Farhadi Pour and Alireza Yousefi assisted with the surgical procedures under supervision. Shayan Yousufzai administered the project, prepared the initial draft, reviewed the literature, and revised the manuscript. Hossein Hosseini visualized the data and produced the video. Saeed Farjam managed the data and recorded the entire procedure.

Approval of the research protocol by an Institutional Reviewer Board

Non applicable.

Declaration of competing interest

The authors declare no competing interest.