Idiopathic posterior laryngeal web on an adult patient
- 1 1st Otorhinolaryngology Dpt, National and Kapodistrian University of Athens School of Medicine, Athens, Attiki, Greece
- 2 Otolaryngology Dpt, Sotiria General Hospital of Chest Diseases of Athens, Athens, Attiki, Greece
- Correspondence to Georgios Chrysovitsiotis; chrysovi@gmail.com
Abstract
Posterior laryngeal webs are uncommon pathologies that are usually acquired at some point in adult life. Prior and prolonged intubation is the leading cause for developing such lesions. In certain rare cases of posterior laryngeal webs, no identifiable cause can be associated with the development of this pathology. We present a case with such an idiopathic lesion. Surgery is the treatment of choice. Several techniques and modifications can be used, to achieve maximum airway release and, at the same time, ensure that restenosis will be avoided. Our patient recurred following initial treatment and a more complex revision surgery was necessary. Special attention should be given in differentiating these lesions from abductor vocal cord paralysis. Furthermore, careful investigation of the cause and individualisation of patient treatment are crucial.
Background
Laryngeal webs are rare entities that usually occur as congenital lesions, due to incomplete recanalisation of the primitive larynx, which takes place during the seventh to eighth week of gestation.1 Congenital laryngeal webs usually comprise of glottic lesions and are more common in the anterior commissure. On the contrary, posterior laryngeal webs are almost always acquired due to a number of causes, with prior intubation being the leading one.
Posterior laryngeal webs in patients with no identifiable cause, termed idiopathic are sporadically reported in the literature. Surgery remains the mainstay of treatment and approaches and techniques are generally individualised, with the least possible morbidity in mind. Recurrence is a common adversity. However, there is limited evidence, regarding recurrence rates comparison between patients with idiopathic laryngeal webs and those with a known cause for it.
We present a case of an adult patient with idiopathic posterior laryngeal web stenosis that recurred after initial surgical treatment and required a more complex revision surgical procedure.
Case presentation
A 68-year-old male patient presented to the outpatients clinic, with an approximate 5-year history of dyspnoea and mild respiratory stridor. The symptoms had deteriorated over the past 2 years and exacerbated by physical activity. The patient did not report any dysphonia or aspiration symptoms. He was a former smoker (ceased smoking 20 years ago), with a long-standing history of coronary artery disease and atrial flutter. The patient reported no history of trauma and had never undergone surgery or intubation. Pantoprazole was included in his standard heart medication. The patient claimed that it was prescribed as a gastrointestinal bleeding prophylaxis, due to oral anticoagulant and antiplatelet medication. He reported no symptoms of gastro-oesophageal reflux disease (GERD) nor had he ever been tested for it.
Investigations
The patient underwent meticulous clinical examination and an indirect laryngoscopy with a flexible fibrescope was conducted. A membranous tissue was revealed in the supraglottis, extending from the interarytenoid fold and connecting the overlying mucosa of both arytenoid cartilages. The arytenoids appeared enlarged with impaired mobility and the supraglottic airway was significantly narrowed (figure 1A). No lesions were found in the glottis (figure 1B). True vocal cord abduction was slightly impaired. There was no apparent subglottic stenosis. No asymmetry or protrusion was demonstrated on the rest of the larynx or pharynx. A CT scan was ordered and the supraglottic tissue, with the corresponding stenosis was demonstrated. No subglottic or tracheal stenosis was shown neither was any other submucosal mass.
Photographs from indirect laryngoscopy with flexible fibrescope, at presentation (outpatient clinic setting). (A) The membranous tissue of the supraglottis is visible, connecting the overlying mucosa of both arytenoid cartilages and extending from interarytenoid fold to both aryepiglottic folds. (B) The lesion does not extend to the true vocal cords and no subglottic stenosis is apparent.
Treatment
The findings were thoroughly explained to the patient and surgical treatment was suggested. The patient was informed about the operative procedure and its potential risks and complications, including the possibility of an emergency tracheotomy and informed consent was obtained. The patient was placed in supine position and was successfully intubated with a 6 mm endotracheal tube. A Zeitels laryngoscope was inserted and the larynx was examined with rigid 0° and 30° laryngeal endoscopes. The tissue was palpated with a probe and found to be tense and thick. The cricoarytenoid joints were not fixated and no subglottic stenosis was noted. Followingly, a laser microlaryngoscopy was conducted. A full thickness wedge excision of the interarytenoid tissue was performed using carbon dioxide (CO2) laser, resulting in a satisfactory size increase of the supraglottic lumen. No intraoperative complications were noted and the patient was successfully extubated.
Initial postoperative period was uneventful and the patient’s stridor was significantly improved. He was discharged on the third postoperative day and was scheduled for regular follow-up. Histopathological examination of excised tissue showed hyperplastic laryngeal mucosa and fibrous tissue, with no signs of malignancy or dysplasia. The patient showed improvement of his symptoms and the surgical result was satisfactory on both 1-month and 3-month follow-up examinations. Six months postoperatively, however, the patient’s symptoms had recurred. Indirect laryngoscopy once again revealed an interarytenoid tissue, having almost the same extent as the one previously diagnosed and treated (figure 2A).
(A) Indirect laryngoscopy photograph of postoperative restenosis, at 6-month follow-up examination. Recurrence of the interarytenoid tissue is shown, having almost the same extent as the initial one. (B) Indirect laryngoscopy photograph at 6 months following revision surgery. The trapdoor flap is visible and no scaring restenosis has occurred.
Following consultation with the patient, reoperation was decided and informed consent was obtained. The patient was successfully intubated with a 6 mm endotracheal tube and a set of Jackson laryngoscopes were used along with rigid 0° and 30° laryngeal endoscopes. Initially, the fibrous interarytenoid tissue was excised using CO2 laser. The interarytenoid muscle was not ablated. A postcricoid mucosal advancement flap (PCMAF) was raised cold-steel and was subsequently attached distally, with two figure-of-eight sutures, using 4–0 poliglecaprone-25 sutures. No further excisions or ablations were made and no intraoperative complications occurred. The patient was successfully extubated.
Outcome and follow-up
Immediate postoperative period was uneventful and the patient was discharged on the second postoperative day, without any stridor or dyspnoea. Monthly follow-up examinations were scheduled for the first 6 months. The patient showed no signs of symptoms recurrence or restenosis (figure 2B). The patient is followed up yearly and remains asymptomatic 4 years later.
Discussion
References of posterior laryngeal webs have been significantly fewer before the frequent use of long-term orotracheal intubation. Prolonged intubation is the leading cause of posterior web-based stenosis.2 Endotracheal tube size, cuff pressure, repeated intubations and extubations, as well as ‘push-pull’ phenomena in relation to the mechanical connection to ventilators, are the main factors that increase the risk for intubation related posterior web stenosis.3 4 Several other causes of posterior webs have been described in the literature, including external trauma to the larynx, radiation therapy, GERD, caustic ingestion or inhalation and inflammatory conditions.5 Idiopathic posterior laryngeal web, with or without stenosis, is a term that is used for patients with no history of airway manipulation, treatment or trauma that could be implicated as the cause of their web. In a study conducted by Atallah et al, 25% (n=13) of the patients presented with idiopathic posterior webs. They all had clinical evidence of developing posterior web-based stenosis secondary to severe GERD, as confirmed by laryngoscopy and flexible esophagoscopy, with or without pH-metry.6 Therefore, investigation for GERD should be considered in patients with idiopathic posterior webs and acid reflux should be controlled, as possible, prior to treatment and postoperatively as well.
Patients usually report stridor and dyspnoea, with variable severity. In general, these symptoms occur progressively. Diagnosis can be established during direct or indirect laryngoscopy. A restrictive web-like tissue is recognised, most commonly joining the arytenoids. Arytenoid and vocal cord mobility can be impaired and caution should be exerted in differentiating this condition from abductor vocal cord paralysis. The arytenoids are often large and bulky and concurrent subglottic stenosis can be also noted.7 However, associated subglottic stenosis is far more common in patients with congenital laryngeal webs. The most commonly used system, for categorising lesions in posterior web-based stenosis, was introduced by Bogdasarian and Olson.8 This grading system divides patients with posterior web lesions into four groups, according to the specific pathology found at the time of surgery, ranging from simple vocal process adhesion (grade I) to posterior commissure stenosis with bilateral cricoarytenoid ankylosis (grade IV). However, in this categorisation, it cannot be distinguished if the arytenoid fixation is derived from the interarytenoid web itself or from another cause.
Surgery remains the treatment of choice for posterior laryngeal webs. A number of surgical techniques are described in the literature, either via a transoral or an open approach. The later, however, generally requires a tracheostomy, unless the patient already has one, and decannulation rates can drop to 70% or less.6 Most authors conclude that plain endoscopic lysis of the scar tissue is an adequate treatment for grade I webs. Lesions of higher grade, require more complex procedures, including microtrapdoor flap technique and PCMAF.6 9 The general principle, especially for higher grade lesions, is that re-epithelialisation before scar formation is crucial for a successful surgical result.9 Therefore, by preserving mucosa and using it as a flap to cover denuded surfaces, restenosis is minimised. This was also demonstrated by our patient’s recurrence after the first, more conservative surgery. Despite the fact that endoscopic suturing can be challenging, endoscopic approach, with or without complex flap techniques, offers minimum morbidity or complications and, thus, should always be taken into consideration.
Learning points
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Posterior laryngeal webs are usually acquired, with prior and prolonged intubation being the leading cause.
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Idiopathic posterior laryngeal web is a term used for patients with no identifiable cause for it.
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High association between idiopathic posterior laryngeal webs and gastro-oesophageal reflux disease has been proposed.
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Surgical approach should be individualised according to the lesion’s grade.
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Reepithelialisation before scar formation is crucial for a successful surgical result and for avoiding recurrence.
Footnotes
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Contributors All authors are clinicians that were involved in the diagnosis and treatment of the case. EK was the primary responsible doctor who performed both surgeries and conceptualised this article. GC, SP and SK have reviewed the literature and written a first draft that was edited following review from EK. All authors approved the final version of the manuscript and agree to the BMJ Case Reports’ submission policies.
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Funding The authors have not declared a specific grant for this research from any funding agency in the public, commercial or not-for-profit sectors.
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Disclaimer Case reports provide a valuable learning resource for the scientific community and can indicate areas of interest for future research. They should not be used in isolation to guide treatment choices or public health policy.
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Competing interests None declared.
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Provenance and peer review Not commissioned; externally peer reviewed.
- © BMJ Publishing Group Limited 2021. No commercial re-use. See rights and permissions. Published by BMJ.
References
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