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Abstract

Laryngomalacia is the most common condition causing stridor in neonates. Surgical management is rarely necessary except in cases of severe airway compromise and failure to thrive. Three cases of severe laryngomalacia treated with CO2 laser assisted supraglottoplasty is described




Introduction

Laryngomalacia is the most common condition causing stridor in neonates and represents 60- 75% of all cases of congenital laryngeal anomalies. [1] It is characterized by the presence of inspiratory stridor that is usually self-limiting and resolves before the age of two years.  Surgical management is necessary in cases of severe airway compromise. We present three cases of severe laryngomalacia treated with CO2 laser assisted supraglottoplasty with excellent results.  


Case Report

Case 1
08 months old male child, a full term normal delivery presented with history of noisy breathing since birth which had increased in the past 03 months and was aggravating on crying and feeding. The child was 05 Kg which was below the normal weight for age (normal for age 8.5 Kg; less than 3rd percentile). On examination there was a harsh inspiratory stridor with sternal and intercostal retraction. Fibreoptic laryngoscopy under dissociation anaesthesia showed a large floppy epiglottis with collapsing aryepiglottic folds into the endolarynx during inspiration (Fig 1).  No other associated abnormalities in the larynx and trachea were detected. The child underwent CO2 Laser assisted supraglottoplasty under general anaesthesia which involved periodic removal of endotracheal tube and use of laser during the apnoeic phase to facilitate better visualisation and application of CO2 laser. Bilateral aryepiglottic folds were cut by using �V� shaped incision (Fig 2). Postoperatively the child was on ventilator support for 48 hrs and then extubated as laryngeal edema subsides by then. The child showed marked reduction in stridor postoperatively with complete recovery and absence of stridor in one month.  The child reviewed after 06 months showed no stridor with significant weight gain. 
Case 2
40 days old male child, a full term normal delivery, a case of meningomyelocele and stridor since birth had multiple episodes of respiratory distress, apnoeic spells leading to cyanosis and subsequent intubation. Multiple attempts at extubation was unsuccessful and the child was referred to our institute on ventilator support. On examination the child was on mechanical ventilation with Wt. - 2.43 Kg (Normal for age 3.7 Kg; 5th percentile). Fibreoptic laryngoscopy under anaesthesia showed a large floppy epiglottis with collapsing aryepiglottic folds, oedematous supraglottis with bilateral mobile vocal cords. MRI showed lumbosacral unrptured meningomyelocele with Arnold Chiari malformation with cerebellar tonsillar herniation. The child underwent microlaryngoscopy and CO2 Laser assisted supraglottoplasty under anaesthesia. Postoperatively child was on ventilator for 48 hrs and then extubated uneventfully. The stridor and apneic spells subsided. After one month the child underwent surgery for meningomyelocoele and recovered well with no stridor.
Case 3
01 year old male child, a full term delivery born by Caesarean section presented with noisy breathing since birth with recurrent chest infection and exacerbation of stridor of two months duration.  On examination the child was 6.24 Kg (Normal for age 10 Kg; less than 3rd percentile), had severe pectus excavatum, severe inspiratory stridor with sternal and intercostal recession. Fibreoptic laryngoscopy under anaesthesia floppy epiglottis with supraglottic indrawing during inspiration. Microlaryngoscopy and CO2 Laser assisted bilateral supraglottoplasty was done with excellent results. The patient is on regular followup with no stridor and significant weight gain.           
Discussion

	Laryngomalacia is the most common cause of congenital stridor caused by collapse of the major components of the supraglottis ie the epiglottis, aryepiglottic folds and aytenoid mucosa. Although a congenital lesion, it manifests as an inspiratory stridor by the age of 4-6 weeks when the inspiratory flow rates are high enough to generate the sounds. The symptoms aggravate till the age of 4 to 6 months and then gradually decrease and resolve by the age 18 to 24 months.[2] The airway obstruction is usually well tolerated and self limiting with only 5 -15 % of patients with severe condition needing intervention.[3] 

	The exact etiology is still unknown with various theories describing anatomical, histological and neurologic abnormalities of the supraglottic structures. Lack of coordination or neuromuscular immaturity and recently gastroesophageal reflux have been considered as causative factors.[4] The higher incidence of gastroesophageal reflux has been hypothesized due to the more negative intrathoracic pressures necessary to overcome the inspiratory obstruction in laryngomalacia.

The infant has a characteristic inspiratory stridor which aggravates in supine position, during crying or agitation, upper respiratory tract infection episodes and sometimes during and after feeding, but improves with neck extension and in prone position. In most cases a detailed history, physical examination and flexible fibreoptic laryngoscopy would suffice to diagnose laryngomalacia. In severe cases requiring surgical intervention a thorough evaluation of the tracheobronchial tree including direct laryngoscopy and bronchoscopy is essential. [5]

Anatomical features on which clinical diagnosis of laryngomalacia should be confirmed by laryngoscopy include flaccid epiglottis prolapsing backwards during inspiration, poorly supported arytenoids that prolapse forward during inspiration and short aryepiglottic folds. Based on these factors laryngoamalcia can be classified as Type 1: aryepiglottic folds are tightened or foreshortened, Type 2: redundant soft tissue in any area of the supraglottic region and Type 3: associated with neuromuscular disease and gastroesophageal reflux. It can also be defined by the direction of supraglottic collapse as type A (posterolateral), type B (complete), and type C (anterior).

Synchronous airway lesions (SAL) are estimated to be present in 19-27% patients of laryngomalacia. Tracheomalacia is the commonest lower airway anomaly followed by bronchomalacia, tracheobronchomalacia, vocal fold movement impairment and subglottic stenosis. The presence of SAL needs to be ruled out before performing surgery as they predict need for postoperative airway support, longer hospital stay and poorer outcome.[6]

The severity and the need for intervention in laryngomalacia does not depend on intensity of stridor, but rather on manifestations of the severity of ventilator obstruction like life threatening hypoxia and apnoea, cyanosis, breastfeeding difficulties, failure to thrive, pectus excavatum, pulmonary hypertension, respiratory failure and cor pulmonale.[7]

A modified croup scoring system is useful in assessing the severity of airway compromise and identifying infants at risk of developing complications. [8] Infants with severe laryngomalacia may require endoscopic excision of redundant supraglottic tissue (supraglottoplasty, epiglottoplasty, or aryepiglottoplasty) to prevent the collapse into the laryngeal inlet during inspiration, thereby improving the stridor and decreasing the effort of breathing. Various techniques have been used in the past but conservative precise tissue division or removal appears to be the cornerstone of appropriate surgery with minimal complications. 

Supraglottoplasty has been found to be an effective procedure for severe laryngomalacia and can be performed with cold microsurgical instruments or CO2 laser. [9] The use of laser in addition to be precise and avoiding tracheostomy, has the advantage of minimal damage to surrounding tissues significantly lowering the risk of complications. Supraglottic stenosis and interarytenoid adhesion are complications with greater morbidity than laryngomalacia. There have been various reports of 3-4% complication rates after supraglottoplasty and the need for performing unilateral vs bilateral surgery. [10] Unilateral supraglottoplasty is a more conservative approach but has the disadvantage of too little excision of tissue with minimal improvement of the obstruction requiring a second procedure. 

	Severe laryngomalacia requiring surgical correction is a relatively rare condition which requires multidisciplinary approach and well trained personnel to treat this challenging condition. In our cases, we performed bilateral CO2 laser assisted supraglottoplasty which involved division of the aryepiglottic folds to release the redundant tissue and prevent supraglottic collapse.  The cases are reported to highlight the need to diagnose severe laryngomalacia requiring surgical correction at an earlier stage and use of CO2 laser which helps in remarkable improvement in symptoms of the patients with minimal morbidity.    




References

1.	Sichel JY, Dangoor E, Eliashar R, Halperin D. Management of congenital laryngeal malformation. Am J Otolaryngol 2000;21:22-30.
2.	Botma M, Kishore A, Kubba H, Geddes N. The role of fibreoptic laryngoscopy in infants with stridor. Int J Pediatr Otorhinolaryngol.2000; 55: 17-20.
3.	 HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed?term=%22Olney%20DR%22%5BAuthor%5D" Olney DR,  HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed?term=%22Greinwald%20JH%20Jr%22%5BAuthor%5D" Greinwald JH Jr,  HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed?term=%22Smith%20RJ%22%5BAuthor%5D" Smith RJ,  HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed?term=%22Bauman%20NM%22%5BAuthor%5D" Bauman NM. Laryngomalacia and its treatment.  HYPERLINK "javascript:AL_get(this,%20'jour',%20'Laryngoscope.');" \o "The Laryngoscope." Laryngoscope. 1999 Nov;109(11):1770-5.
4.	Hadfield PJ, Albert DM, Bailey CM, Lindley K, Pierro A. The effect of aryepiglottoplasty for laryngomalacia on gastrooesophageal reflux. Int J Pediatr Otorhinolaryngol 2003; 67:11-4.
5.	Kayamar R, Gray RF. Per oral flexible fiber-optic laryngoscopy in the investigation of children with stridor without respiratory distress. The Journal of Laryngology & Otology 2001; 115: 894-6.
6.	Yuen HW, Tan HK, Balakrishnan A. Synchronous airway lesions and associated
anomalies in children with laryngomalacia evaluated with rigid endoscopy. Int J Pediatr Otorhinolaryngol. 2006;70(10):1779-1784.
7.	Roger G, Denoyelle F, Triglia JM, Garabedian EN. Severe laryngomalacia: surgical indications and results in 115 patients. Laryngoscope 1995;105:1111-7.
8.	Sivan Y, Ben-Ari J, Soferman R, DeRowe A. Diagnosis of Laryngomalacia by Fiberoptic Endoscopy : Awake Compared With Anesthesia-Aided Technique. Chest 2006;130;1412-1418.
9.	 HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed?term=%22Lee%20KS%22%5BAuthor%5D" Lee KS,  HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed?term=%22Chen%20BN%22%5BAuthor%5D" Chen BN,  HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed?term=%22Yang%20CC%22%5BAuthor%5D" Yang CC,  HYPERLINK "http://www.ncbi.nlm.nih.gov/pubmed?term=%22Chen%20YC%22%5BAuthor%5D" Chen YC. CO2 laser supraglottoplasty for severe laryngomalacia: a study of symptomatic improvement.  HYPERLINK "javascript:AL_get(this,%20'jour',%20'Int%20J%20Pediatr%20Otorhinolaryngol.');" \o "International journal of pediatric otorhinolaryngology." Int J Pediatr Otorhinolaryngol. 2007 Jun;71(6):889-95. 
10.	Reddy D K, Matt B H. Unilateral vs Bilateral Supraglottoplasty for Severe Laryngomalacia in Children. Arch Otolaryngol Head Neck Surg. 2001;127:694-699.












Legend to Figures


Fig 1

Fibreoptic laryngoscopic view of the typical findings of collapsing supraglottis in severe laryngomalacia


Fig 2

	CO2 laser assisted bilateral aryepiglottic folds supraglottoplasty.


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