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Original Article |

Laryngeal Anatomic Differences in Pediatric Patients With Severe Laryngomalacia FREE

Scott C. Manning, MD; Andrew F. Inglis, MD; Jason Mouzakes, MD; Jeffrey Carron, MD; Jonathan A. Perkins, DO
[+] Author Affiliations

Financial Disclosure: None.


Arch Otolaryngol Head Neck Surg. 2005;131(4):340-343. doi:10.1001/archotol.131.4.340.
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Published online

Objectives  To compare the aryepiglottic (AE) length in pediatric patients who have severe laryngomalacia (SL) and are undergoing aryepiglottoplasty with the AE length of a convenience sample of control patients without laryngomalacia.

Design  Prospective case-control study.

Setting  A tertiary-care pediatric hospital.

Results  The mean AE fold length–glottic length ratio for patients with SL (0.380) was significantly lower than the mean ratio for controls (0.535) (P = .004 in 2-sample t test with unequal variance). For patients with SL, the aryepiglottoplasy procedure resulted in an average AE length increase–glottic length ratio of 0.330. Seven of the patients with SL were also diagnosed as having an underlying neurologic condition, and 18 had a diagnosis of gastroesophageal reflux disease. Two patients with SL required a tracheotomy for treatment of persistent airway obstruction.

Conclusions  In this series, patients with SL had lower AE fold length–glottic length ratios and more frequent occurrence of neuromuscular tone abnormalities (especially gastroesophageal reflux) than controls. These 2 findings may be related in that low intrauterine tone might contribute to anatomic underdevelopment.

Figures in this Article

Laryngomalacia is the most common cause of stridor in infants and young children. It is characterized by inspiratory prolapse of supraglottic structures in varying patterns, including medial prolapse of the aryepiglottic (AE) folds and anterior prolapse of arytenoid mucosa.1 Proposed explanations of laryngomalacia have included intrinsic structural anomalies of laryngeal cartilages, lack of coordinated muscular laryngeal dilation, and abnormalities of supraglottic gross anatomy, including short AE folds. An estimated 5% to 10% of patients with laryngomalacia show progressive worsening of airway obstruction to the point of failure to thrive, usually between ages 2 and 4 months.2

Starting in the early 1980s, improved outcomes in cases of severe laryngomalacia (SL) were reported from surgical interventions designed to increase the size of the supraglottic inlet. However, information regarding laryngeal anatomic measurements is lacking in the laryngomalacia literature.35 The purpose of the present study is to compare laryngeal measurements in patients with SL undergoing supraglottoplasty with those of controls undergoing laryngoscopy for other reasons.

Data were collected prospectively from a convenience sample of 24 patients with SL undergoing aryepiglottoplasty from 1998 to 2003 at Children’s Hospital and Regional Medical Center, Seattle, Wash (institutional review board No. 04-0516-01). The indications for aryepiglottoplasty were history of laryngomalacia with poor weight gain or weight loss (failure to thrive) and increasing airway symptoms including stridor, retractions, and choking during feeding. All patients with SL had laryngomalacia diagnosed by flexible laryngoscopy without anesthesia in the outpatient clinic or inpatient hospital setting prior to scheduling aryepiglottoplasty. Control patients were a convenience sample of 8 infants and young children without laryngomalacia or other craniofacial or neurological diagnoses who were undergoing laryngoscopy for other reasons (such as recurrent croup).

The anesthesia technique for laryngoscopy consisted of spontaneous ventilation with intravenous propofol and topical 4% lidocaine applied to the larynx. An infant or pediatric Lindholm laryngoscope was suspended at the level of the vallecula, and the larynx was visualized with a microscope or a 4-mm, 0° Hopkins telescope. Oxygen was infused through the side port of the laryngoscope. Glottic length measurements were taken from the anterior border of the interarytenoid muscle to the anterior commissure. The AE fold measurements were made from the most anterior arytenoid cartilage to the closest posterior lateral edge of the epiglottic cartilage (Figure). Measurements were made either with wires bent at the ends in half-millimeter increments or with alligator forceps (then referencing the opening to a millimeter ruler). All measurements were taken at least 3 times, and the average value was recorded. The AE fold measurements were repeated after the surgical procedure. A statistical analysis of the AE fold length–glottic length ratios was performed with Stata 8.2 software (Stata Corp, College Station, Tex) using a 2-sample t test with unequal variances.

Place holder to copy figure label and caption
Figure.

An alligator forcep opening along the aryepiglottic fold with the posterior blade contacting the most anterior arytenoid cartilage and the anterior blade contacting the most proximal edge of the epiglottic cartilage is used to measure the free aryepiglottic fold length.

Graphic Jump Location

The exact type of aryepiglottoplasty varied by attending staff physician. The senior author (S.C.M.), divided the membranous AE folds bilaterally with microlaryngeal scissors. The other attending surgeons (A.F.I. and J.A.P.) divided the AE folds with a carbon dioxide laser and varying degrees of conservative excision of “redundant” AE fold mucosa and portions of the cuneiform and corniculate cartilages. None of the patients in this series underwent epiglottoplasty with fixation to the base of tongue.

Results for patients with SL and controls are summarized in Table 1 and Table 2. The mean AE fold length–glottic length ratio for patients with SL (0.380) was significantly lower than the mean ratio for controls (0.535) (P = .004) (Table 3). The increases in AE fold lengths after surgery are also listed in Table 1. The average AE fold length increase–glottic length ratio for the patients with SL was 0.330. (The procedure resulted in about a one-third increase in AE fold length on average.) Sixteen of the patients with SL underwent simple excision of the AE folds, and the remaining 8 underwent laser division with some additional removal of AE fold and arytenoid mucosa. There were no differences in postsurgical AE fold measurements or in patient outcomes based on technique.

Table Graphic Jump LocationTable 1. Findings for Patients With Severe Laryngomalacia
Table Graphic Jump LocationTable 2. Findings for Control Patients
Table Graphic Jump LocationTable 3. Aryepiglottic Fold–Glottic Length Ratio Statistical Findings

Eighteen of the 24 patients with SL had a diagnosis of gastroesophageal reflux disease (GERD), although only 6 of the 18 had undergone a contrast study or pH probe. The remaining patients received a clinical diagnosis of GERD based on a history of vomiting, choking during feeding, and nocturnal distress. All patients with a diagnosis of GERD had been treated prior to surgery, usually with ranitidine before the year 2000 and with proton-pump inhibitors more recently. Patient 6 (trisomy 1q) eventually underwent an endoscopic Nissen procedure for control of severe GERD, and patient 21 (CHARGE syndrome [coloboma, hearing deficit, choanal atresia, retardation of growth, genital defects {in male patients}, and endocardial cushion defect]) underwent placement of a gastrostomy tube, which remains in place.

Seven of the patients with SL had a diagnosis of neurologic disorder other than GERD, including trisomy 21, trisomy 1q, CHARGE syndrome, Cri du Chat (chromosome 5p) syndrome, cerebral palsy, and seizure disorder. The mean AE fold length–glottic length ratio for the 7 patients with neurologic diagnoses (0.40) did not differ significantly from that of the 17 patients without associated neurologic issues (0.37).

All patients without a neurologic diagnosis improved clinically in both airway and GERD status and were discharged home with weight gain, although a few required a few days of postoperative nasogastric or nasoduodenal feeding before their airway status stabilized. None of those patients has been readmitted for failure-to-thrive issues, although 4 were lost to follow-up after their families moved from the region. Four of the patients without neurologic diagnoses have been observed for more than 1 year (because of treatment for new diagnoses), and all had no airway or reflux symptoms at the time of last visit.

Patients 6 (trisomy 1q) and 21 (CHARGE syndrome) underwent a tracheotomy because of continued upper airway obstruction. The airway status of patient 6 improved significantly following the fundoplication procedure, and the tracheotomy tube was successfully removed 1 year later. Patient 21 has a left vocal cord paralysis following ligation of a patent ductus arteriosis and remains with a tracheotomy tube in place. Patient 24 presented with a history of sudden airway obstruction during athletic endeavors at age 6 years. The parents subsequently recalled a clinical diagnosis of laryngomalacia at age 1 month made by their primary care physician. Symptoms of exercise-induced shortness of breath resolved completely following aryepiglottoplasty.

Amin and Isaacson6 in a recent review of patients with laryngomalacia point out that Sutherland and Lack7 proposed more than 100 years ago that abnormal anatomy contributed to laryngomalacia. Many subsequent authors have observed abnormalities, including “short” AE folds in cases of laryngomalacia.1,810 Some authors have proposed that intrinsic abnormalities of laryngeal tissues might explain laryngomalacia, but histologic studies of biopsy specimens have failed to demonstrate chondropathy or other ultrastructural abnormalities.9

The hypothesis that lack of coordinated neuromuscular dilation of the supraglottic airway leads to laryngomalacia was similarly first proposed more than 100 years ago based on animal studies.11 Laryngomalacia has been related to the sleep state,6 brain injury,12 and neurologic disorders including seizure disorder and cerebral palsy. Several authors have noted poorer results of therapeutic intervention when a history of associated neurologic conditions is present.13

Severe laryngomalacia has long been associated with gastroesophageal reflux diagnosed either clinically, by bronchial lavage with lipid-laden macrophages, radiographically, or by pH probe.1,9,10,14,15 Hadfield et al10 demonstrated improvement in reflux measured by pH probe following successful intervention for airway obstruction with aryepiglottoplasty in a group of patients with laryngomalacia. Presumably, the negative inspiratory pressures generated by upper airway obstruction can aggravate reflux. Laryngomalacia and reflux may also be associated because both may represent effects of general low muscle tone in the upper aerodigestive tract.

Very little information exists regarding the normal glottic and supraglottic dimensions in infants and young children. Litman et al16 recently described measurements of anterior-posterior glottic length and transverse laryngeal dimensions in a group of 99 infants and children undergoing sedated magnetic resonance imaging for a variety of reasons. The rate of increase in measurements was linear with increasing age, and the ratio of glottic length to transverse dimensions remained fairly constant. Of note, the values for glottic lengths for patients in the 1- to 5-month age group was 5 to 8 mm, similar to the findings in the present study.

However, in the present study, the mean AE fold length–glottic length ratio of patients with SL was significantly lower than that of controls without laryngomalacia. Within the group of patients with SL, the ratio for those without associated neurologic conditions did not differ from that of the other patients. The ratio of AE fold length to glottic length was used as a way to standardize measurements in a group of patients with different ages and weights.

In conclusion, “form follows function” is a favorite adage of oral surgeons and orthodondists. Robin sequence with micrognathia associated with dysphagia and poor swallowing coordination17 may be a consequence of poor mandibular growth due to low intrauterine neuromuscular tone. Similarly, the relative AE fold shortening in patients with SL in the present study may be a developmental consequence of low aerodigestive tract muscle tone.

Correspondence: Scott C. Manning, MD, Department of Otolaryngology, University of Washington, 4800 Sand Point Way NE, Seattle, WA 98105-0371 (scott.manning@seattlechildrens.org).

Submitted for Publication: November 4, 2004; accepted December 16, 2004.

Previous Presentations: This study was presented at the American Society of Pediatric Otolaryngology Annual Meeting; May 2, 2004; Phoenix, Ariz.

Acknowledgment: We thank Kristy Seidel, MS, biostatistician, for her work on this article.

Olney  DRGreinwald  JH  JrSmith  RJHBauman  NM Laryngomalacia and its treatment. Laryngoscope 1999;1091770- 1775
PubMed Link to Article
Denoyelle  FMondain  MGresillon  N  et al.  Failures and complications of supraglottoplasy in children. Arch Otolaryngol Head Neck Surg 2003;1291077- 1080
PubMed Link to Article
Lane  RWWeider  DJSteinem  CMarin-Padilla  M Laryngomalacia: a review and case report of surgical treatment with resolution of pectus excavatum. Arch Otolaryngol 1984;110546- 551
PubMed Link to Article
Seid  ABPark  SMKearns  MJGugenheim  S Laser division of the aryeiglottic folds for severe laryngomalacia. Int J Pediatr Otorhinolaryngol 1985;10153- 158
PubMed Link to Article
Zalzal  GHAnon  JBCotton  RT Epiglottoplasty for the treatment of laryngomalacia. Ann Otol Rhinol Laryngol 1987;9672- 76
PubMed
Amin  MRIsaacson  B State-dependent laryngolmalacia. Ann Otol Rhinol Laryngol 1997;106887- 890
PubMed
Sutherland  GALack  HL Congenital laryngeal obstruction. Lancet 1897;2653- 655
Link to Article
Prescott  CA The current status of corrective surgery for laryngomalacia. Am J Otolaryngol 1991;12230- 235
PubMed Link to Article
Chandra  RKGerber  MEHolinger  LD Histological insight into the pathogenesis of severe laryngomalacia. Int J Pediatr Otorhinolaryngol 2001;6131- 38
PubMed Link to Article
Hadfield  PJAlbert  DMBailey  CMLindley  KPierro  A The effect of aryepiglottoplasty for laryngomalacia on gastro-oesophageal reflux. Int J Pediatr Otorhinolaryngol 2003;6711- 14
PubMed Link to Article
Thompson  JTurner  A On the causation of congenital laryngeal stridor of infants. BMJ 1900;21561- 1563
Link to Article
Peron  DLGruffino  DBZenker  DO The redundant aryepiglottic fold: report of a new cause of stridor. Laryngoscope 1988;98659- 663
PubMed Link to Article
Senders  CWNavarrete  EG Laser supraglottoplasty for laryngomalacia: are specific anatomical defects more influential than associated anomalies on outcome? Int J Pediatr Otorhinolaryngol 2001;57235- 244
PubMed Link to Article
Bibi  HKhvolis  EShoseyov  D  et al.  The prevalence of gastroesophageal reflux in children with tracheomalacia and laryngomalacia. Chest 2001;119409- 413
PubMed Link to Article
Halstead  LA Role of gastroesophageal reflux in pediatric upper airway disorders. Otolaryngol Head Neck Surg 1999;120208- 214
PubMed Link to Article
Litman  RSWeissend  EEShibata  DWestesson  P-L Developmental changes of laryngeal dimensions in unparalyzed sedated children. Anesthesiology 2003;9841- 45
PubMed Link to Article
Baudon  JJRenault  FGoutet  JM  et al.  Motor dysfunction of the upper digestive tract in Pierre Robin sequence as assessed by sucking-swallowing electromyography and esophageal manometry. J Pediatr 2002;140719- 723
PubMed Link to Article

Figures

Place holder to copy figure label and caption
Figure.

An alligator forcep opening along the aryepiglottic fold with the posterior blade contacting the most anterior arytenoid cartilage and the anterior blade contacting the most proximal edge of the epiglottic cartilage is used to measure the free aryepiglottic fold length.

Graphic Jump Location

Tables

Table Graphic Jump LocationTable 1. Findings for Patients With Severe Laryngomalacia
Table Graphic Jump LocationTable 2. Findings for Control Patients
Table Graphic Jump LocationTable 3. Aryepiglottic Fold–Glottic Length Ratio Statistical Findings

References

Olney  DRGreinwald  JH  JrSmith  RJHBauman  NM Laryngomalacia and its treatment. Laryngoscope 1999;1091770- 1775
PubMed Link to Article
Denoyelle  FMondain  MGresillon  N  et al.  Failures and complications of supraglottoplasy in children. Arch Otolaryngol Head Neck Surg 2003;1291077- 1080
PubMed Link to Article
Lane  RWWeider  DJSteinem  CMarin-Padilla  M Laryngomalacia: a review and case report of surgical treatment with resolution of pectus excavatum. Arch Otolaryngol 1984;110546- 551
PubMed Link to Article
Seid  ABPark  SMKearns  MJGugenheim  S Laser division of the aryeiglottic folds for severe laryngomalacia. Int J Pediatr Otorhinolaryngol 1985;10153- 158
PubMed Link to Article
Zalzal  GHAnon  JBCotton  RT Epiglottoplasty for the treatment of laryngomalacia. Ann Otol Rhinol Laryngol 1987;9672- 76
PubMed
Amin  MRIsaacson  B State-dependent laryngolmalacia. Ann Otol Rhinol Laryngol 1997;106887- 890
PubMed
Sutherland  GALack  HL Congenital laryngeal obstruction. Lancet 1897;2653- 655
Link to Article
Prescott  CA The current status of corrective surgery for laryngomalacia. Am J Otolaryngol 1991;12230- 235
PubMed Link to Article
Chandra  RKGerber  MEHolinger  LD Histological insight into the pathogenesis of severe laryngomalacia. Int J Pediatr Otorhinolaryngol 2001;6131- 38
PubMed Link to Article
Hadfield  PJAlbert  DMBailey  CMLindley  KPierro  A The effect of aryepiglottoplasty for laryngomalacia on gastro-oesophageal reflux. Int J Pediatr Otorhinolaryngol 2003;6711- 14
PubMed Link to Article
Thompson  JTurner  A On the causation of congenital laryngeal stridor of infants. BMJ 1900;21561- 1563
Link to Article
Peron  DLGruffino  DBZenker  DO The redundant aryepiglottic fold: report of a new cause of stridor. Laryngoscope 1988;98659- 663
PubMed Link to Article
Senders  CWNavarrete  EG Laser supraglottoplasty for laryngomalacia: are specific anatomical defects more influential than associated anomalies on outcome? Int J Pediatr Otorhinolaryngol 2001;57235- 244
PubMed Link to Article
Bibi  HKhvolis  EShoseyov  D  et al.  The prevalence of gastroesophageal reflux in children with tracheomalacia and laryngomalacia. Chest 2001;119409- 413
PubMed Link to Article
Halstead  LA Role of gastroesophageal reflux in pediatric upper airway disorders. Otolaryngol Head Neck Surg 1999;120208- 214
PubMed Link to Article
Litman  RSWeissend  EEShibata  DWestesson  P-L Developmental changes of laryngeal dimensions in unparalyzed sedated children. Anesthesiology 2003;9841- 45
PubMed Link to Article
Baudon  JJRenault  FGoutet  JM  et al.  Motor dysfunction of the upper digestive tract in Pierre Robin sequence as assessed by sucking-swallowing electromyography and esophageal manometry. J Pediatr 2002;140719- 723
PubMed Link to Article

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