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Location of airway obstruction in term and preterm infants with laryngomalacia
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American Journal of Otolaryngology–Head and Neck Medicine and Surgery 33 (2012) 437 – 440 www.elsevier.com/locate/amjoto
Location of airway obstruction in term and preterm infants with laryngomalacia☆,☆☆ Eelam Adil, MD, MBA⁎, Terrence Rager, MD, Michele Carr, DDS, MD, PhD Division of Otolaryngology–Head and Neck Surgery, Pennsylvania State College of Medicine, Hershey, PA, USA Received 9 October 2011
Abstract
Objective: The aim of this study was to describe and compare the airway findings in term and preterm infants with laryngomalacia. Methods: A retrospective review of 130 patients diagnosed as having laryngomalacia at a tertiary referral center between July 2004 and August 2009 was conducted. Medical records were reviewed for demographic data, supraglottic and glottic airway findings, concomitant airway lesions, and the need for intervention. Results: The mean gestational age and age at diagnosis was 36 and 15 weeks, respectively. Combined posterior and anterior supraglottic collapse was the most common finding (31%). Posterior collapse alone occurred in 25%, anterior collapse in 14%, and lateral collapse in 10%. Twelve percent of patients had all 3 sites of collapse. Forty-one percent of patients had a secondary airway lesion, with tracheomalacia being the most common. Preterm infants had significantly higher rates of reflux and more sites of collapse than did term infants (P b .0001). Eight patients required an intervention for their symptoms. Conclusions: Children with laryngomalacia tend to have more than 1 area of supraglottic collapse, and more than one third have a secondary lesion. All patients who required an intervention had more than 1 area of collapse, and 63% of these patients had a secondary airway lesion. Our high incidence of secondary lesions is similar to recent reports. © 2012 Elsevier Inc. All rights reserved.
1. Introduction The term laryngomalacia'was introduced in 1942 by Chevalier Jackson. It refers to collapse of the supraglottic larynx, creating a narrow airway and turbulent airflow. Laryngomalacia is very common and accounts for 65% to 75% of stridor in infants [1]. It may be an isolated finding or associated with other neurologic disorders such as cerebral palsy. ☆
Conflicts of interest: None. These data were partially presented at the American Society of Pediatric Otolaryngology Annual Meeting in Las Vegas, NV, on April 30, 2010. ⁎ Corresponding author. Division of Otolaryngology–Head and Neck Surgery, H091 Pennsylvania State University, Milton S. Hershey Medical Center, Hershey, PA 17033, USA. Tel.: +1 717 531 6718; fax: +1 717 531 6160. E-mail address: [email protected] (E. Adil). ☆☆
0196-0709/$ – see front matter © 2012 Elsevier Inc. All rights reserved. doi:10.1016/j.amjoto.2011.10.021
There are several laryngomalacia classification schemes proposed in the literature, but none of them are universal. Olney et al [2] categorized laryngomalacia according to the area of supraglottic collapse and method of supraglottoplasty. Type 1 laryngomalacia involves prolapse of the mucosa overlying the arytenoid cartilages. In type 2, there are foreshortened aryepiglottic folds. Posterior displacement of the epiglottis is type 3. Although this classification system is useful, it fails to take into account that there may be more than 1 area of collapse. Another classification strategy proposed in the literature groups patients according to their underlying pathophysiology [3]. In this study, type I laryngomalacia is characterized by a foreshortened aryepiglottic fold. Type 2 disease has redundant soft tissue in the supraglottis, whereas type 3 laryngomalacia is caused by other etiologies such as underlying neuromuscular disorders. In this study, we review our experience with laryngomalacia. The goals are to identify the most common area(s) of
438
E. Adil et al. / American Journal of Otolaryngology–Head and Neck Medicine and Surgery 33 (2012) 437–440
supraglottic collapse, compare airway findings in term and preterm infants, and evaluate the incidence of secondary airway lesions in our population. 2. Materials and methods A retrospective chart review was performed at a tertiary care center on 217 consecutive patients with a diagnosis of laryngomalacia between June 2004 and August 2009. Patients with incomplete records, an undocumented flexible fiberoptic, or direct laryngoscopy examination, or those diagnosed by airway fluoroscopy alone were excluded. This decision was based on the poor sensitivity (27%–34%) of airway fluoroscopy in diagnosing laryngomalacia when compared with airway endoscopy [4,5]. A total of 130 patients met the inclusion criteria (54 were female and 76 were male). The Penn State University Institutional Review Board approved this study, and guardians gave consent during their initial office visit for the use of records for medical research. Evaluation of each patient was dependent on their presentation. Stable patients had a flexible fiberoptic laryngoscopy, airway fluoroscopy, and a barium swallow. Those patients with abnormal fluoroscopic examinations or persistent stridor despite negative examinations underwent a direct laryngoscopy and bronchoscopy (DLB) in the operating room under general anesthesia. Patients who presented with cyanotic episodes, gray spells, and failure to thrive; intubated patients; and those with any other acute signs/symptoms underwent a DLB. During laryngoscopy, vocal fold mobility, the site of supraglottic collapse, edema, erythema, and other lesions were noted. A total of 105 patients underwent flexible fiberoptic laryngoscopy. Fifty-five patients had a DLB, and 108 patients had an airway fluoroscopy. Patients were classified as having posterior collapse if the arytenoids themselves or the overlying mucosa obstructed the airway during inspiration. Lateral collapse included shortened aryepiglottic folds or mucosa that prolapsed medially into the airway. A retropositioned epiglottis or one that prolapsed into the airway was classified as anterior collapse. Reflux was documented if patients had a positive barium swallow, symptoms consistent with reflux, or laryngeal findings such as edema and/or erythema that could be attributed to reflux. Secondary airway lesions were documented based on fluoroscopy and/or DLB. Descriptive statistics and χ 2 analysis were completed using the Statistical Package for the Social Sciences version 17 (SPSS, Chicago, IL).
3. Results 3.1. Demographics The mean gestational age was 36.3 weeks, with a range of 24 to 42 weeks. Thirty-eight percent of patients were preterm (Table 1). The mean age at diagnosis was 3.9 months (range, 0.5–15 months). Nine patients had a history of intubation,
and 6 patients had another congenital anomaly (2 had Down syndrome; 1, atrial septal defect; 1, patent ductus arteriosus; 1, cleft palate; and 1, Apert syndrome). The male/female ratio was 1.4:1. 3.2. Supraglottic findings Thirty-three patients (25%) had isolated posterior supraglottic collapse (Fig. 1). Lateral collapse alone was seen in 13 patients (10%), and anterior collapse alone was noted in 18 patients (14%). Posterior and anterior collapse were frequently noted together (n = 40). Lateral and anterior or posterior collapses were the least frequent findings (n = 4 and 7, respectively). Fifteen patients had collapse in all 3 areas. Sixty-seven percent of patients had reflux. 3.3. Secondary airway lesions Forty-one percent of patients had a secondary airway lesion. All these patients had a DLB for their definitive diagnosis. Tracheomalacia (n = 25) was the most common finding, followed by subglottic stenosis (n = 12). Vocal fold immobility was noted in 7 patients. 3.4. Preterm infants The incidence of reflux, secondary lesions, and number of sites of collapse were compared between preterm infants (ie, born before 37 weeks' gestation) and term infants. Preterm infants were found to have a significantly higher incidence of reflux and more sites of supraglottic collapse (P b .0001). There was no significant difference in the number of secondary lesions (P = .431). 3.5. Additional procedures There were 8 patients who required an airway procedure in addition to DLB, and all of them were preterm. Supraglottoplasty was performed in 3 patients: 1 patient had anterior and posterior collapse and tracheomalacia, 1 had Table 1 Airway findings in preterm infants with laryngomalacia Gestational No. of Site(s) of collapse Reflux Secondary age (wk) patients lesions Posterior Lateral Anterior 24 25 26 27 28 29 30 31 32 33 34 35 36
6 1 2 3 4 2 2 2 3 2 2 11 9
6 1 2 3 4 2 2 2 3 2 2 11 9
2 0 0 1 2 1 1 0 2 0 1 2 2
6 1 2 2 2 2 2 2 2 2 1 10 8
6 1 2 3 4 1 2 2 3 2 2 10 7
6 1 2 0 2 0 0 1 2 0 0 3 2
Note that most patients in each age category had reflux and more than 1 site of supraglottic collapse.
E. Adil et al. / American Journal of Otolaryngology–Head and Neck Medicine and Surgery 33 (2012) 437–440
Fig. 1. Venn diagram depicting the percentage of patients with each type of supraglottic collpase. Note that most patients had combined anterior and posterior collapse.
lateral and posterior collapse, and 1 had collapse at all 3 sites. Three patients underwent subglottic dilation for their secondary lesions, and 2 patients had a tracheotomy. 4. Discussion Laryngomalacia is the most common cause of stridor in infants. Symptoms typically appear within the first 2 weeks of life and peak at 6 months of age. Most cases resolve spontaneously, and 75% of patients will have no stridor by 18 months [2]. Other congenital anomalies are reported in up to 47% of patients with laryngomalacia, although only 5% of our population had another anomaly. Surgical intervention is generally recommended for patients with failure to thrive, apnea, cyanosis, or pectus excavatum [6,7]. The cause of laryngomalacia is unknown, but there are 3 theories: (1) neuromuscular immaturity, (2) misshapen supraglottic cartilage, and/or (3) reflux [8-12]. Neuromuscular immaturity may play a larger role in preterm infants who were all found to have more than 1 area of supraglottic collapse (Table 1). When compared with full-term infants, preterm newborns have significantly more areas of supraglottic collapse (P b .0001). However, they also have a significantly higher incidence of reflux (92%; P b .0001), indicating that the etiology of laryngomalacia in this population is likely multifactorial. Eighty patients in our study had posterior collapse in isolation or combined with another area of collapse. Seventy-two of these patients had a barium swallow, and 68 (94%) of these were positive for reflux. Thirty-six patients without posterior collapse also had a barium swallow, but only 19 (53%) of these were positive for reflux. This supports the hypothesis that reflux is an important factor in the development of laryngomalacia in some patients because most reflux-associated mucosal
439
changes are seen in the posterior larynx. In addition, it has implications for treatment. All patients with isolated posterior collapse (n = 33) received a proton pump inhibitor or H2 blocker and did not require any further surgical intervention. The authors recognize that a barium swallow is not the criterion standard for diagnosing reflux; however, this is a retrospective review, and most patients included in this study did not undergo dual-probe pH monitoring. There have been no studies that describe the incidence of isolated vs combined supraglottic collapse in laryngomalacia. In this study, we found that 51% of patients had more than 1 area of collapse. Combined posterior and anterior laryngeal collapse was the most frequent finding (Fig. 1), but previous classification schemes do not account for multiple areas of collapse [2,13-15]. This is important because all patients in our study who required an airway procedure in addition to DLB had more than 1 area of collapse, so this can be a predictor of who may require an airway intervention. In addition, knowing the site(s) of collapse helps with the communication of airway findings and surgical planning. The decision to perform a DLB in patients with typical laryngomalacia is controversial [16-19]. In the last 2 years, there have been several articles indicating that the incidence of secondary airway lesions is higher than previously reported [2,16]. The most recent study from Northwestern University reported that 58% of patients who underwent a supraglottoplasty had a secondary airway lesion [18]. Another article from the University of Cincinnati showed that 51.7% of patients had a secondary airway lesion, the most common of which was subglottic stenosis followed by tracheomalacia [19]. However, the incidence of secondary airway lesions may have been overestimated because these institutions are commonly referred children with complex airway anomalies. In our review, 41% of patients had a secondary airway lesion, with tracheomalacia being the most common. Preterm infants did not have a significantly higher incidence of secondary lesions when compared with fullterm infants. All patients who required a second airway procedure were preterm and had more than 1 area of supraglottic collapse. 5. Conclusion As in previous studies, we found a male predilection for laryngomalacia [2,13]. In addition, most children were born at or near term. Preterm infants with laryngomalacia had a significantly higher incidence of reflux and more than 1 site of supraglottic collapse. The incidence of secondary airway lesions was 41%, which is less than that in recent studies, but still supports the notion that otolaryngologists should have a low threshold for performing a DLB. References [1] Ahmad SM, Soliman AM. Congenital anomalies of the larynx. Otolaryngol Clin North Am. 2007;40:177-91, viii.
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E. Adil et al. / American Journal of Otolaryngology–Head and Neck Medicine and Surgery 33 (2012) 437–440
[2] Olney DR, Greinwald JH, Smith RJ, et al. Laryngomalacia and its treatment. Laryngoscope 1999;109:1770-5. [3] Kay DJ, Goldsmith AJ. Laryngomalacia: a classification system and surgical treatment strategy. Ear Nose Throat J. 2006;85:32831, 336. [4] Huntley C, Carr M. Evaluation of the effectiveness of airway fluoroscopy in diagnosing patients with laryngomalacia. Laryngoscope 2010;120:1430-4. [5] Berg E, Naseri I, Sobol SE. The role of airway fluoroscopy in the evaluation of children with stridor. Arch Otolaryngol Head Neck Surg 2008;134:415-8. [6] Roger G, Denoyelle F, Triglia JM, et al. Severe laryngomalacia: surgical indications and results in 115 patients. Laryngoscope 1995;105:1111-7. [7] Zalzal GH, Anon JB, Cotton RT. Epiglottoplasty for the treatment of laryngomalacia. Ann Otol Rhinol Laryngol 1987;96(1 Pt 1):72-6. [8] Belmont JR, Grundfast K. Congenital laryngeal stridor (laryngomalacia): etiologic factors and associated disorders. Ann Otol Rhinol Laryngol 1984;93:430-7. [9] Archer SM. Acquired flaccid larynx. A case report supporting the neurologic theory of laryngomalacia. Arch Otolaryngol Head Neck Surg 1992;118:654-7. [10] Wiggs Jr WJ, DiNardo LJ. Acquired laryngomalacia: resolution after neurologic recovery. Otolaryngol Head Neck Surg 1995;112:773-6.
[11] Giannoni C, Friedman EM, Duncan III NO. Gastroesophageal reflux association with laryngomalacia: a prospective study. Int J Pediatr Otorhinolaryngol 1998;43:11-20. [12] Matthews BL, Little JP, Mcguirt Jr WF, et al. Reflux in infants with laryngomalacia: results of 24 hour double probe pH monitoring. Otolaryngol Head Neck Surg 1999;120:860-4. [13] Holinger LD. Etiology of stridor in the neonate, infant, and child. Ann Otol 1980;89:397-400. [14] Kay DJ, Goldsmith AJ. Laryngomalacia: a classification system and surgical treatment strategy. Ear Nose Throat J. 2006;85:328-31, 336. [15] Shah UK, Wetmore RF. Laryngomalacia: a proposed classification form. Int J Pediatr Otorhinolaryngol 1998;46:21-6. [16] Mancuso RF, Choi SS, Zalzal GH, Grundfast KM. Laryngomalacia: the search for a second lesion. Arch Otolaryngol Head Neck Surg 1996;122:302-6. [17] Bluestone CD, Healy GB, Cotton RT. Diagnosis of laryngomalacia is not enough! Arch Otolaryngol Head Neck Surg 1996;122:1417-8. [18] Schroeder Jr JW, Bhandarkar ND, Holinger LD. Synchronous airway lesions and outcomes in infants with severe laryngomalacia requiring supraglottoplasty. Arch Otolaryngol Head Neck Surg 2009;135: 647-51. [19] Dickson JM, Richter GT, Meinzen-Derr J, et al. Secondary airway lesions in infants with laryngomalacia. Ann Otol Rhinol Laryngol 2009;118:37-43.
American Journal of Otolaryngology–Head and Neck Medicine and Surgery 33 (2012) 437 – 440 www.elsevier.com/locate/amjoto
Location of airway obstruction in term and preterm infants with laryngomalacia☆,☆☆ Eelam Adil, MD, MBA⁎, Terrence Rager, MD, Michele Carr, DDS, MD, PhD Division of Otolaryngology–Head and Neck Surgery, Pennsylvania State College of Medicine, Hershey, PA, USA Received 9 October 2011
Abstract
Objective: The aim of this study was to describe and compare the airway findings in term and preterm infants with laryngomalacia. Methods: A retrospective review of 130 patients diagnosed as having laryngomalacia at a tertiary referral center between July 2004 and August 2009 was conducted. Medical records were reviewed for demographic data, supraglottic and glottic airway findings, concomitant airway lesions, and the need for intervention. Results: The mean gestational age and age at diagnosis was 36 and 15 weeks, respectively. Combined posterior and anterior supraglottic collapse was the most common finding (31%). Posterior collapse alone occurred in 25%, anterior collapse in 14%, and lateral collapse in 10%. Twelve percent of patients had all 3 sites of collapse. Forty-one percent of patients had a secondary airway lesion, with tracheomalacia being the most common. Preterm infants had significantly higher rates of reflux and more sites of collapse than did term infants (P b .0001). Eight patients required an intervention for their symptoms. Conclusions: Children with laryngomalacia tend to have more than 1 area of supraglottic collapse, and more than one third have a secondary lesion. All patients who required an intervention had more than 1 area of collapse, and 63% of these patients had a secondary airway lesion. Our high incidence of secondary lesions is similar to recent reports. © 2012 Elsevier Inc. All rights reserved.
1. Introduction The term laryngomalacia'was introduced in 1942 by Chevalier Jackson. It refers to collapse of the supraglottic larynx, creating a narrow airway and turbulent airflow. Laryngomalacia is very common and accounts for 65% to 75% of stridor in infants [1]. It may be an isolated finding or associated with other neurologic disorders such as cerebral palsy. ☆
Conflicts of interest: None. These data were partially presented at the American Society of Pediatric Otolaryngology Annual Meeting in Las Vegas, NV, on April 30, 2010. ⁎ Corresponding author. Division of Otolaryngology–Head and Neck Surgery, H091 Pennsylvania State University, Milton S. Hershey Medical Center, Hershey, PA 17033, USA. Tel.: +1 717 531 6718; fax: +1 717 531 6160. E-mail address: [email protected] (E. Adil). ☆☆
0196-0709/$ – see front matter © 2012 Elsevier Inc. All rights reserved. doi:10.1016/j.amjoto.2011.10.021
There are several laryngomalacia classification schemes proposed in the literature, but none of them are universal. Olney et al [2] categorized laryngomalacia according to the area of supraglottic collapse and method of supraglottoplasty. Type 1 laryngomalacia involves prolapse of the mucosa overlying the arytenoid cartilages. In type 2, there are foreshortened aryepiglottic folds. Posterior displacement of the epiglottis is type 3. Although this classification system is useful, it fails to take into account that there may be more than 1 area of collapse. Another classification strategy proposed in the literature groups patients according to their underlying pathophysiology [3]. In this study, type I laryngomalacia is characterized by a foreshortened aryepiglottic fold. Type 2 disease has redundant soft tissue in the supraglottis, whereas type 3 laryngomalacia is caused by other etiologies such as underlying neuromuscular disorders. In this study, we review our experience with laryngomalacia. The goals are to identify the most common area(s) of
438
E. Adil et al. / American Journal of Otolaryngology–Head and Neck Medicine and Surgery 33 (2012) 437–440
supraglottic collapse, compare airway findings in term and preterm infants, and evaluate the incidence of secondary airway lesions in our population. 2. Materials and methods A retrospective chart review was performed at a tertiary care center on 217 consecutive patients with a diagnosis of laryngomalacia between June 2004 and August 2009. Patients with incomplete records, an undocumented flexible fiberoptic, or direct laryngoscopy examination, or those diagnosed by airway fluoroscopy alone were excluded. This decision was based on the poor sensitivity (27%–34%) of airway fluoroscopy in diagnosing laryngomalacia when compared with airway endoscopy [4,5]. A total of 130 patients met the inclusion criteria (54 were female and 76 were male). The Penn State University Institutional Review Board approved this study, and guardians gave consent during their initial office visit for the use of records for medical research. Evaluation of each patient was dependent on their presentation. Stable patients had a flexible fiberoptic laryngoscopy, airway fluoroscopy, and a barium swallow. Those patients with abnormal fluoroscopic examinations or persistent stridor despite negative examinations underwent a direct laryngoscopy and bronchoscopy (DLB) in the operating room under general anesthesia. Patients who presented with cyanotic episodes, gray spells, and failure to thrive; intubated patients; and those with any other acute signs/symptoms underwent a DLB. During laryngoscopy, vocal fold mobility, the site of supraglottic collapse, edema, erythema, and other lesions were noted. A total of 105 patients underwent flexible fiberoptic laryngoscopy. Fifty-five patients had a DLB, and 108 patients had an airway fluoroscopy. Patients were classified as having posterior collapse if the arytenoids themselves or the overlying mucosa obstructed the airway during inspiration. Lateral collapse included shortened aryepiglottic folds or mucosa that prolapsed medially into the airway. A retropositioned epiglottis or one that prolapsed into the airway was classified as anterior collapse. Reflux was documented if patients had a positive barium swallow, symptoms consistent with reflux, or laryngeal findings such as edema and/or erythema that could be attributed to reflux. Secondary airway lesions were documented based on fluoroscopy and/or DLB. Descriptive statistics and χ 2 analysis were completed using the Statistical Package for the Social Sciences version 17 (SPSS, Chicago, IL).
3. Results 3.1. Demographics The mean gestational age was 36.3 weeks, with a range of 24 to 42 weeks. Thirty-eight percent of patients were preterm (Table 1). The mean age at diagnosis was 3.9 months (range, 0.5–15 months). Nine patients had a history of intubation,
and 6 patients had another congenital anomaly (2 had Down syndrome; 1, atrial septal defect; 1, patent ductus arteriosus; 1, cleft palate; and 1, Apert syndrome). The male/female ratio was 1.4:1. 3.2. Supraglottic findings Thirty-three patients (25%) had isolated posterior supraglottic collapse (Fig. 1). Lateral collapse alone was seen in 13 patients (10%), and anterior collapse alone was noted in 18 patients (14%). Posterior and anterior collapse were frequently noted together (n = 40). Lateral and anterior or posterior collapses were the least frequent findings (n = 4 and 7, respectively). Fifteen patients had collapse in all 3 areas. Sixty-seven percent of patients had reflux. 3.3. Secondary airway lesions Forty-one percent of patients had a secondary airway lesion. All these patients had a DLB for their definitive diagnosis. Tracheomalacia (n = 25) was the most common finding, followed by subglottic stenosis (n = 12). Vocal fold immobility was noted in 7 patients. 3.4. Preterm infants The incidence of reflux, secondary lesions, and number of sites of collapse were compared between preterm infants (ie, born before 37 weeks' gestation) and term infants. Preterm infants were found to have a significantly higher incidence of reflux and more sites of supraglottic collapse (P b .0001). There was no significant difference in the number of secondary lesions (P = .431). 3.5. Additional procedures There were 8 patients who required an airway procedure in addition to DLB, and all of them were preterm. Supraglottoplasty was performed in 3 patients: 1 patient had anterior and posterior collapse and tracheomalacia, 1 had Table 1 Airway findings in preterm infants with laryngomalacia Gestational No. of Site(s) of collapse Reflux Secondary age (wk) patients lesions Posterior Lateral Anterior 24 25 26 27 28 29 30 31 32 33 34 35 36
6 1 2 3 4 2 2 2 3 2 2 11 9
6 1 2 3 4 2 2 2 3 2 2 11 9
2 0 0 1 2 1 1 0 2 0 1 2 2
6 1 2 2 2 2 2 2 2 2 1 10 8
6 1 2 3 4 1 2 2 3 2 2 10 7
6 1 2 0 2 0 0 1 2 0 0 3 2
Note that most patients in each age category had reflux and more than 1 site of supraglottic collapse.
E. Adil et al. / American Journal of Otolaryngology–Head and Neck Medicine and Surgery 33 (2012) 437–440
Fig. 1. Venn diagram depicting the percentage of patients with each type of supraglottic collpase. Note that most patients had combined anterior and posterior collapse.
lateral and posterior collapse, and 1 had collapse at all 3 sites. Three patients underwent subglottic dilation for their secondary lesions, and 2 patients had a tracheotomy. 4. Discussion Laryngomalacia is the most common cause of stridor in infants. Symptoms typically appear within the first 2 weeks of life and peak at 6 months of age. Most cases resolve spontaneously, and 75% of patients will have no stridor by 18 months [2]. Other congenital anomalies are reported in up to 47% of patients with laryngomalacia, although only 5% of our population had another anomaly. Surgical intervention is generally recommended for patients with failure to thrive, apnea, cyanosis, or pectus excavatum [6,7]. The cause of laryngomalacia is unknown, but there are 3 theories: (1) neuromuscular immaturity, (2) misshapen supraglottic cartilage, and/or (3) reflux [8-12]. Neuromuscular immaturity may play a larger role in preterm infants who were all found to have more than 1 area of supraglottic collapse (Table 1). When compared with full-term infants, preterm newborns have significantly more areas of supraglottic collapse (P b .0001). However, they also have a significantly higher incidence of reflux (92%; P b .0001), indicating that the etiology of laryngomalacia in this population is likely multifactorial. Eighty patients in our study had posterior collapse in isolation or combined with another area of collapse. Seventy-two of these patients had a barium swallow, and 68 (94%) of these were positive for reflux. Thirty-six patients without posterior collapse also had a barium swallow, but only 19 (53%) of these were positive for reflux. This supports the hypothesis that reflux is an important factor in the development of laryngomalacia in some patients because most reflux-associated mucosal
439
changes are seen in the posterior larynx. In addition, it has implications for treatment. All patients with isolated posterior collapse (n = 33) received a proton pump inhibitor or H2 blocker and did not require any further surgical intervention. The authors recognize that a barium swallow is not the criterion standard for diagnosing reflux; however, this is a retrospective review, and most patients included in this study did not undergo dual-probe pH monitoring. There have been no studies that describe the incidence of isolated vs combined supraglottic collapse in laryngomalacia. In this study, we found that 51% of patients had more than 1 area of collapse. Combined posterior and anterior laryngeal collapse was the most frequent finding (Fig. 1), but previous classification schemes do not account for multiple areas of collapse [2,13-15]. This is important because all patients in our study who required an airway procedure in addition to DLB had more than 1 area of collapse, so this can be a predictor of who may require an airway intervention. In addition, knowing the site(s) of collapse helps with the communication of airway findings and surgical planning. The decision to perform a DLB in patients with typical laryngomalacia is controversial [16-19]. In the last 2 years, there have been several articles indicating that the incidence of secondary airway lesions is higher than previously reported [2,16]. The most recent study from Northwestern University reported that 58% of patients who underwent a supraglottoplasty had a secondary airway lesion [18]. Another article from the University of Cincinnati showed that 51.7% of patients had a secondary airway lesion, the most common of which was subglottic stenosis followed by tracheomalacia [19]. However, the incidence of secondary airway lesions may have been overestimated because these institutions are commonly referred children with complex airway anomalies. In our review, 41% of patients had a secondary airway lesion, with tracheomalacia being the most common. Preterm infants did not have a significantly higher incidence of secondary lesions when compared with fullterm infants. All patients who required a second airway procedure were preterm and had more than 1 area of supraglottic collapse. 5. Conclusion As in previous studies, we found a male predilection for laryngomalacia [2,13]. In addition, most children were born at or near term. Preterm infants with laryngomalacia had a significantly higher incidence of reflux and more than 1 site of supraglottic collapse. The incidence of secondary airway lesions was 41%, which is less than that in recent studies, but still supports the notion that otolaryngologists should have a low threshold for performing a DLB. References [1] Ahmad SM, Soliman AM. Congenital anomalies of the larynx. Otolaryngol Clin North Am. 2007;40:177-91, viii.
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E. Adil et al. / American Journal of Otolaryngology–Head and Neck Medicine and Surgery 33 (2012) 437–440
[2] Olney DR, Greinwald JH, Smith RJ, et al. Laryngomalacia and its treatment. Laryngoscope 1999;109:1770-5. [3] Kay DJ, Goldsmith AJ. Laryngomalacia: a classification system and surgical treatment strategy. Ear Nose Throat J. 2006;85:32831, 336. [4] Huntley C, Carr M. Evaluation of the effectiveness of airway fluoroscopy in diagnosing patients with laryngomalacia. Laryngoscope 2010;120:1430-4. [5] Berg E, Naseri I, Sobol SE. The role of airway fluoroscopy in the evaluation of children with stridor. Arch Otolaryngol Head Neck Surg 2008;134:415-8. [6] Roger G, Denoyelle F, Triglia JM, et al. Severe laryngomalacia: surgical indications and results in 115 patients. Laryngoscope 1995;105:1111-7. [7] Zalzal GH, Anon JB, Cotton RT. Epiglottoplasty for the treatment of laryngomalacia. Ann Otol Rhinol Laryngol 1987;96(1 Pt 1):72-6. [8] Belmont JR, Grundfast K. Congenital laryngeal stridor (laryngomalacia): etiologic factors and associated disorders. Ann Otol Rhinol Laryngol 1984;93:430-7. [9] Archer SM. Acquired flaccid larynx. A case report supporting the neurologic theory of laryngomalacia. Arch Otolaryngol Head Neck Surg 1992;118:654-7. [10] Wiggs Jr WJ, DiNardo LJ. Acquired laryngomalacia: resolution after neurologic recovery. Otolaryngol Head Neck Surg 1995;112:773-6.
[11] Giannoni C, Friedman EM, Duncan III NO. Gastroesophageal reflux association with laryngomalacia: a prospective study. Int J Pediatr Otorhinolaryngol 1998;43:11-20. [12] Matthews BL, Little JP, Mcguirt Jr WF, et al. Reflux in infants with laryngomalacia: results of 24 hour double probe pH monitoring. Otolaryngol Head Neck Surg 1999;120:860-4. [13] Holinger LD. Etiology of stridor in the neonate, infant, and child. Ann Otol 1980;89:397-400. [14] Kay DJ, Goldsmith AJ. Laryngomalacia: a classification system and surgical treatment strategy. Ear Nose Throat J. 2006;85:328-31, 336. [15] Shah UK, Wetmore RF. Laryngomalacia: a proposed classification form. Int J Pediatr Otorhinolaryngol 1998;46:21-6. [16] Mancuso RF, Choi SS, Zalzal GH, Grundfast KM. Laryngomalacia: the search for a second lesion. Arch Otolaryngol Head Neck Surg 1996;122:302-6. [17] Bluestone CD, Healy GB, Cotton RT. Diagnosis of laryngomalacia is not enough! Arch Otolaryngol Head Neck Surg 1996;122:1417-8. [18] Schroeder Jr JW, Bhandarkar ND, Holinger LD. Synchronous airway lesions and outcomes in infants with severe laryngomalacia requiring supraglottoplasty. Arch Otolaryngol Head Neck Surg 2009;135: 647-51. [19] Dickson JM, Richter GT, Meinzen-Derr J, et al. Secondary airway lesions in infants with laryngomalacia. Ann Otol Rhinol Laryngol 2009;118:37-43.