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Table of Contents
ORIGINAL ARTICLE
Year : 2019  |  Volume : 9  |  Issue : 2  |  Page : 51-56

Associated airway anomalies and their impact in patients with laryngomalacia: A retrospective review


Department of Laryngology (E.N.T), Affiliated to Deenanath Mangeshkar Hospital and Research Centre, Pune, Maharashtra, India

Date of Submission29-Jan-2020
Date of Acceptance15-Jun-2020
Date of Web Publication14-Aug-2020

Correspondence Address:
Subash Bhatta
Deenanath Mangeshkar Hospital and Research Centre, Erandawane, Pune - 411 004, Maharashtra
India
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Source of Support: None, Conflict of Interest: None


DOI: 10.4103/jlv.JLV_2_20

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   Abstract 


Introduction: Laryngomalacia is the most common cause of stridor in newborn constituting 50%–75% of all airway anomalies. It is graded as mild, moderate, and severe on the basis of presenting features. It has been shown that 12%–64% of laryngomalacia present with other associated airway anomalies. Here, we have tried to find out the incidence of associated anomalies with laryngomalacia and their impact on the patient's management and prognosis. Materials and Methods: We reviewed patients with laryngomalacia visiting laryngology clinic from January 2014 to January 2019. The incidence and type of associated anomalies; their impact on management protocol and hospital stay; and their association with mild, moderate, and severe grades of laryngomalacia were evaluated. Results: A total of 63 patients were included in the study. Most of the patients presented with severe laryngomalacia (34/63 [53.9%]). Out of them, associated anomalies were present in 23/63 (36%) patients, more common in severe laryngomalacia (16/34 [47.05%]). Tracheomalacia was the most common anomaly (9/63 [14.28%]). The management protocol was changed on the basis of the associated anomalies. The six out of nine patients with tracheomalacia underwent tracheostomy and watchful waiting was done for the other three. The bilateral vocal cords palsy patients underwent partial arytenoidectomy, direct laryngoscopic dilatation was done for all cases of subglottic stenosis, the vallecular cyst patient underwent excision of the cyst and for the patient with laryngeal cleft repairing of the cleft was done after freshening the margins of the cleft. Patients with associated anomalies had statistically significant increased duration of hospital stay (11.35 days) as compared to patients without these anomalies (7.5 days). Conclusion: It can be concluded that associated anomalies are present in a significant number of patients with laryngomalacia and can result in the modification of management protocol and increase in the duration of hospital stay.

Keywords: Associated anomalies, laryngomalacia, stridor, tracheomalacia


How to cite this article:
Gandhi S, Bhatta S, Ganesuni D, Ghanpur AD. Associated airway anomalies and their impact in patients with laryngomalacia: A retrospective review. J Laryngol Voice 2019;9:51-6

How to cite this URL:
Gandhi S, Bhatta S, Ganesuni D, Ghanpur AD. Associated airway anomalies and their impact in patients with laryngomalacia: A retrospective review. J Laryngol Voice [serial online] 2019 [cited 2020 Sep 29];9:51-6. Available from: http://www.laryngologyandvoice.org/text.asp?2019/9/2/51/291926




   Introduction Top


Congenital anomalies of larynx are of rare occurrence with uncertain incidence, with figures quoting about 1 in 10,000–1 in 50,000 live births.[1] Laryngomalacia is the most common congenital anomaly.[1],[2],[3],[4],[5],[6],[7] There are no data to suggest the exact prevalence of laryngomalacia in the newborns, however it has been shown that laryngomalacia constitutes about 50%–75% of all congenital airway anomalies.[8] High-pitched fluttering inspiratory stridor is the most common presenting feature of laryngomalacia. Diagnosis is confirmed by flexible nasopharyngolarygoscopy.

Along with these airway features and endoscopic findings, laryngomalacia may present with various other signs and symptoms. On the basis of these signs and symptoms, laryngomalacia is divided into mild, moderate, and severe grade, as shown in [Table 1].[9]
Table 1: Grades of laryngomalacia

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There may be presence of associated airway abnormalities along with laryngomalacia, which could impact management and its outcome. The exact incidence of these anomalies is not known, but it has been suggested to be present in about 12%–64% in various studies.[5],[7],[10],[11],[12],[13] These anomalies may present anywhere in the airway, so the flexible laryngoscopy may sometimes be unable to detect them. Hence, every case of laryngomalacia should be evaluated by direct laryngoscopy under general anesthesia (GA).[6],[7],[12],[14] Although there are studies which suggest that the mere presence of these anomalies does not impact the treatment protocol, decision of further management should be based on the severity of the condition and videolaryngoscopic findings.[7],[15]

In our hospital, detailed evaluation was done for all patients with laryngomalacia. If any medical comorbidities were suspected, then the patients were referred to the respective subspecialty for further evaluation and management. After addressing the medical comorbidities, the patients were evaluated in the laryngology clinic. In our clinic, flexible endoscopy was done for every patient who had come with difficulty in breathing or noisy breathing for 3 weeks or more. Then, grading was done on the basis of their presenting features and managed accordingly. Surgical fitness under GA was taken for every patient who was planned for surgery.


   Materials and Methods Top


This was a retrospective data analysis to determine the incidence and type of associated anomalies in infants with laryngomalacia and to determine the association of grade of laryngomalacia and associated anomalies. In addition, our objective was to determine whether the presence of associated anomaly led to modification in the management or addition of any extra procedure and to determine the association between the presence of associated anomalies with duration of hospital stay.

The study was carried out in a tertiary care multispecialty hospital in western Maharashtra. Approval was granted by the institutional ethics committee. The data were retrieved from the hospital software from January 2014 to January 2019. Data included details of patients diagnosed with laryngomalacia and who visited laryngology clinic. Data of patients in whom endoscopy was not done were excluded.

The details of patients such as demographic data, gender, age at presentation, chief complaint at presentation, any comorbidities, and provisional and final diagnosis were documented. For medical comorbidities, the patients were first managed in the respective sub specialties. Their notes were reviewed and analyzed. The patients were divided into mild, moderate, and severe grades according to their presenting complaints. The presenting complaints after the appropriate evaluation of medical comorbidities were only taken. The endoscopic videos were analyzed under the supervision of a single consultant laryngologist surgeon and looked for presence of any associated anomalies. Associated airway anomalies were defined as any anomalies other than laryngomalacia along the entire length of airway from supraglottis to carina. Vocal cord palsy was defined as the loss of either unilateral or bilateral vocal cord movement during flexible endoscopy.[5],[13] Subglottic stenosis was defined as a narrowing of the subglottis measured by the sequential uncuffed endotracheal tube and was classified according to Cotton Myer classification.[5] Tracheomalacia was defined as the collapse of the trachea during expiration, reducing the diameter by >50%.[13] In addition, features suggestive of gastroesophageal reflux disease (GERD) were defined as the presence of interarytenoid congestion, cobblestone appearance of posterior pharyngeal wall, and mucosal edema over endolarynx.[16] On the basis of the above definition, presence of various associated anomalies was documented. Intraoperative videos and notes were reviewed, and we tried to find out whether the associated anomalies led to modification in management or not.

To find out the association between duration of hospital stay and presence or absence of associated anomalies, patients with medical comorbidities were omitted as this might bias the findings. Then, we compared the hospital stay in patients with associated anomalies with those without associated anomalies.

Data were analyzed using SPSS version 21 (SPSS Inc., Chicago, IL, USA). Associations were determined using Chi-square test, and independent t-test was used to compare means. P < 0.05 was considered statistically significant.


   Results Top


We analyzed data of 63 patients; there were 35/63 (55.6%) males and 28/63 (44.44%) females. The mean age of patients at presentation was 65.28 days (median age 49 days). The most common presenting complaint was stridor in 58/63 (92.7%) patients. Others were failure to thrive in 26/63 (41.26%) patients, difficulty in swallowing in 25/63 (39.7%), chest retraction in 24/63 (38.09%), respiratory distress in 22/63 (34.92%), aspiration in 16/63 (25.39%), apneic episodes in 16/63 (25.39%), cyanotic spells in 16/63 (25.39%), fainting episodes in 13/63 (20.63%), and snoring in 10/63 (15.87%) patients.

On the basis of symptomatology, the patients were classified into three grades. The grades of laryngomalacia along with associated anomalies are shown in [Table 2].
Table 2: Distribution of associated airway anomalies according to the grade of laryngomalacia

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Using Chi-square test, it was, however, found that the association between associated anomalies and grade of laryngomalacia was not statistically significant (P = 0.152) as shown in [Table 3].
Table 3: Association between grade of laryngomalacia and associated airway anomalies

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Associated anomalies were present in 23/63 (36.5%) patients. The most common anomaly was tracheomalacia present in 9/63 (14.3%) patients. Next was vocal cord palsy present in 7/63 (11.1%) patients. Among these, 6/63 (9.5%) patients had bilateral palsy, while 1/63 (1.6%) had unilateral vocal cord palsy. There were 3/63 (4.8%) cases of sub-glottic stenosis (SGS): two were Grade II and one was Grade I according to Cotton–Myer's classification. GERD was present in 47/63 (74.6%) patients. Except for these defined anomalies, various other anomalies were present as shown in [Table 2] which were vallecular cyst in 3/63 (4.8%) and laryngeal cleft in 1/6 (1.58%) patients.

Out of 63 patients, 16 had some medical comorbidity. In these 16 patients, the average duration of hospital stay was 17.56 days, whereas the average duration of hospital stay in patients without any medical comorbidity (47/63 [74.6%] patients) was 9.14 days. To find out the impact of associated anomalies in the duration of hospital stay, we excluded these 16 patients with medical comorbidity. Thus, data were analyzed in 47/63 (74.6%) patients, diagnosed with laryngomalacia, with or without associated airway anomalies, but without any medical comorbidities. Using independent t-test, it was found that patients with airway anomalies (n = 20) had a significantly longer hospital stay when compared to those without airway anomalies (n = 27) with mean hospital stay of 11.35 ± 5 days versus 7.5 ± 2.9 days, respectively (P = 0.002), with standard error of 1.18 and 0.53, respectively.

Presence of associated anomalies modified the management protocol, and each of them was dealt accordingly. The various management strategies employed are shown in [Table 4].
Table 4: Distribution according to the management for associated anomalies

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All additional surgical procedures were done in the same setting, in which supraglottoplasty was done. The patients with tracheomalacia were followed up regularly, and the development of trachea was assessed. Out of six tracheostomized patients, five could be de-cannulated successfully. Patients with SGS underwent three consecutive dilatations.

Out of the 63 patients, all those with mild disease and 5/19 with moderate disease were managed by conservative management and the remaining 48 patients were operated. The operation carried out was supraglottoplasty under GA with spontaneous ventilation under intermittent apnea technique.


   Discussion Top


Laryngomalacia is the most common congenital anomaly causing stridor. The initial diagnosis is done with flexible fiberoptic endoscope in outpatient department and those planned for surgery were evaluated with rigid endoscopy.

In our study, there were more males (35/63 [55.6%]), which was in accordance with other studies.[9],[17],[18],[19] However, few studies also showed more females presenting with laryngomalacia.[20],[21] As there was only a small difference in the percentage, the gender predominance of the condition cannot be ascertained. We found that the mean age of presentation in our study was 65.28 days. Kusak et al.[22] showed mean age of presentation to be 3.3 months, which is slightly more than that seen in our study. It is well known that stridor is the most common presenting feature of laryngomalacia.[22],[23],[24],[25],[26],[27],[28] Similar finding was seen in our study, with a majority (92.7%) of patients presenting with stridor. The frequency of other symptoms was comparable to that shown by most of the other studies.

Many studies have shown that the mild form of laryngomalacia is the most common presentation, followed by moderate and severe forms.[9],[17],[19] Thompson[27] and Landry and Thompson[28] have stated that mild and moderate laryngomalacia was present in 40% of cases each in their study, whereas in the remaining 20%, it was severe laryngomalacia. Kusak et al.[22] have reported that most of the times, laryngomalacia is mild and self-limiting. Our findings were contrasting to the findings shown by the above studies, showing majority of the patients with severe disease. The explanation to this could be that our hospital is a tertiary referral center, so most of the cases presenting were complex cases referred from other hospitals.

We found that 36.5% of patients showed the presence of another associated airway anomaly. Tracheomalacia was the most common among them. Thompson.[9] in his study had shown that associated anomalies were present in 7.5%–64% of patients with laryngomalacia. The author had mentioned that the technique used for diagnosis could have been the reason behind the large variation. However, he had not mentioned the incidence of the individual type of airway anomalies. In the studies done by Krashin et al.,[10] Rifai et al.,[5] and Yuen et al.,[26] the associated anomalies were present in 7.5%, 7.7%, and 26.9% of patients, respectively. These studies had less percentage of associated anomalies than that in our study. The first one showed tracheal bronchus to be the most common, whereas the second showed subglottic stenosis and tracheomalacia to be the most common followed by bronchomalacia and choanal atresia. The last one showed tracheomalacia to be most common followed by bilateral vocal cord palsy. Schroeder Jr. et al.,[13] Vijayasekaran et al.,[25] and Dickson et al.[7] showed associated anomalies to be present in 58%, 48%, and 51% of laryngomalacia patients, respectively, which was higher in percentage than that shown by our study. The first and the last one showed tracheomalacia to be most common, whereas the second one showed SGS to be the most common followed by tracheomalacia, bronchomalacia, or both.

Tracheomalacia was diagnosed during direct laryngoscopy using spontaneous breathing. Tracheomalacia in our study was defined as the prolapse of the posterior wall of trachea during expiration with reduction in diameter of >50% of the original diameter, in accordance with the study by Dickson et al.[7] and Yuen et al.[26] We evaluated vocal cord paralysis during flexible endoscopy done in the outpatient department. Vijayasekaran et al.[25] and Schroeder Jr. et al.[13] evaluated similarly, however some of the studies[5],[23] evaluated it with direct laryngoscopy in the operation theater under spontaneous ventilation. To evaluate the SGS, we compared with the size of age-appropriate endotracheal tubes and then classified it into three grades according to Cotton–Myer's classification. SGS was assessed similarly by Dickson et al.[7] in their study. To assess the presence of GERD, we used criteria such as interarytenoid congestion, cobblestone appearance of posterior pharyngeal wall, and mucosal edema over endolarynx. Yuen et al.[26] also used similar subjective findings such as supraglottic edema. In addition, they analyzed patient history to look for the presence of vomiting, choking during feeding, and nocturnal distress. On the other hand, Dickson et al.[7] diagnosed GERD using contrast esophagography, esophagoscopy with biopsy, and Dual pH-metry. We found GERD in 74.6% of our patients. Yuen et al.[26] and Dickson et al.[7] found GERD in 42.3% and 65.6% of patients, respectively. There are no conclusive data suggesting whether GERD is caused by laryngomalacia or GERD causes laryngomalacia. Some studies have suggested that there is increased intrathoracic negative pressure in patients with laryngomalacia, which in turn causes reflux of acid, resulting in GERD.[7],[27] Other studies have suggested that GERD may cause edema of the laryngeal structure, leading to prolapse of mucosa, causing decrease in airway mimicking laryngomalacia.[3],[29] Hence, there is no definitive evidence that would suggest the exact relationship between laryngomalacia and GERD. As a sizeable number of patients were suffering from GERD and also keeping in mind the postoperative adverse effect of GERD, the patients were prescribed antireflux medications for at least 3 weeks irrespective of the presence or absence of GERD. Similar kind of management has been mentioned by other studies also.[16],[30],[31],[32],[33]

We found that due to the presence of associated anomalies, the management approach was modified in our study. Associated anomalies were managed similarly as shown by other studies.[10],[18],[26],[28],[34],[35]

We calculated the duration of hospital stay in patients with associated anomalies and compared them with that of patients without associated anomalies. For doing this, we excluded the patients who were suffering from medical comorbidities as these could prolong the hospital stay and lead to a biased result. There were 25.4% of patients with medical comorbidities with mean duration of hospital stay of 17.56 days. This was in accordance to the study done by Yuen et al.[26] and Rifai et al.,[5] who also showed medical comorbidities to be present in about 20%–30% of patients. Yuen et al.[26] also found significant increase in the duration of hospital stay when medical comorbidities were present alongside laryngomalacia. The duration of hospital stay in our study was significantly more for patients with associated anomalies than for those without anomalies. In contrast to this, Yuen et al.[26] did not find any difference in the duration of hospital stay in these two groups of patients.

We found that associated anomalies were most common in severe laryngomalacia (16/34 [47.05%]) followed by mild (3/10 [30%]) and moderate (4/19 [21.05%]). However, this finding was statistically not significant. Dickson et al.[7] in their study showed highest secondary lesions in severe laryngomalacia (79%), followed by moderate (61.5%) and mild (28.8%). It can be inferred that association of anomalies with laryngomalacia may lead to increased severity of the disease, however the exact cause–effect relationship needs to be explored.


   Conclusion Top


It can be concluded that associated anomalies occur in a significant percentage of patients with laryngomalacia, resulting in the modification of management protocol and increased duration of hospital stay of the patients. Hence, complete evaluation of the airway to find out any such lesion is mandatory for the management of a patient with laryngomalacia.

Financial support and sponsorship

This study was financially supported by the Department of Laryngology (E.N.T.), Deenanath Mangeshkar Hospital and Research Centre.

Conflicts of interest

There are no conflicts of interest.



 
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    Tables

  [Table 1], [Table 2], [Table 3], [Table 4]



 

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