AUTHOR AND EDITOR INFORMATION

Section 1 of 10  

• Authors and Editors
• Introduction
• Clinical
• Differentials
• Workup
• Treatment
• Medication
• Follow-up
• Multimedia
• References

INTRODUCTION

Section 2 of 10  

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• Clinical
• Differentials
• Workup
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Background

Subglottic stenosis (SGS) is a narrowing of the subglottic airway (see Media file 1), which is housed in the cricoid cartilage. The subglottic airway is the narrowest area of the airway because it is a complete, nonexpandable, and nonpliable ring, unlike the trachea, which has a posterior membranous section, and the larynx, which has a posterior muscular section. The term SGS implies a narrowing that is created or acquired, although the term is applied to both congenital lesions of the cricoid ring (see Media files 3-5) and acquired SGS (see Media file 2, Media files 6-10, Media files 12-16, and Media file 30).

Acquired SGS is the most common acquired anomaly of the larynx in children and the most common abnormality requiring tracheotomy in children younger than 1 year. Correction of this abnormality requires expanding the lumen of the cricoid area to increase airflow and decrease obstructive breathing. Surgical correction of SGS has been performed with various techniques over the years.

Early in the 20th century, acquired SGS was usually related to trauma or infection from syphilis, tuberculosis, typhoid fever, or diphtheria. Also, children often had tracheotomies placed that caused laryngeal stenosis. In this era, attempted laryngeal dilation failed as a treatment for SGS.

Acquired SGS increasingly occurred in the late 1960s through the 1970s, after McDonald and Stocks introduced long-term intubation as a treatment method for neonates in need of prolonged ventilation for airway support.¹ The increased incidence of SGS focused new attention on the pediatric larynx, and airway reconstruction and expansion techniques were developed.

Surgery without cartilage expansion

In 1971, Rethi and Rhan described a procedure for vertical division of the posterior lamina of the cricoid cartilage with Aboulker stent placement. A metal tracheotomy tube was attached to the Aboulker stent with wires, and the anterior cartilaginous incision was closed. In 1974, Evanston and Todd described success with a castellated incision of the anterior cricoid cartilage and upper trachea, which was sewn open, and a stent made of a rolled silicone sheet was placed in it for 6 weeks. In 1980, Cotton and Seid described a procedure in which tracheotomy is avoided called the anterior cricoid split (ACS).² The procedure was designed for use in neonates (usually, those born prematurely) with anterior SGS or SGS who had airway distress after extubation. The cricoid ring was divided anteriorly and a laryngofissure was created in an attempt to expand the airway without a tracheotomy. Holinger et al also described success with this procedure in 1987.³

Surgery with cartilage-grafting reconstruction

In 1974, Fearon and Cotton described the successful use of cartilage grafts to enlarge the subglottic lumen in African green monkeys and in children with severe laryngotracheal stenosis.⁴ All augmentation materials were evaluated, including thyroid cartilage, septal cartilage, auricular cartilage, costal cartilage, hyoid bone, and sternocleidomastoid myocutaneous flaps. After significant work, it appeared that costal cartilage grafts had the highest success rate.

In the 1980s, Cotton reported his experience with laryngeal expansion with cartilage grafting.^{5, 6, 7, 2} His success rates depended on degree of stenosis. More severe forms of stenosis required multiple surgical procedures. Cotton used the Aboulker stent.

In 1991, Seid et al described a form of single-stage laryngotracheal reconstruction in which cartilage was placed anteriorly to expand the subglottis and upper trachea to avoid a tracheotomy.⁸

In 1992, Cotton et al described a 4-quadrant cricoid split, along with anterior and posterior grafting.⁹ In 1993, Zalzal reported 90% decannulation with any degree of SGS with his first surgical procedure.¹⁰ Zalzal customized the reconstruction on an individual basis, and most patients received Aboulker stents for stabilization.

Cricotracheal resection

In 1993, Monnier described partial cricotracheal resection with primary anastomoses for severe SGS because grade III and grade IV SGS (ie, severe SGS) often requires multiple (3-4) surgical augmentations for decannulation.¹¹ In 1997, Stern described his experience with the procedure, reporting a decannulation rate higher than 90% for primary and rescue cricotracheal resection.¹²

Pathophysiology

The pathophysiology of congenital SGS is a malformed cricoid cartilage in utero. The cause of congenital SGS is in utero malformation of the cricoid cartilage.

The etiology of acquired SGS is related to trauma of the subglottic mucosa. Injury can be caused by infection or mechanical trauma, usually from endotracheal intubation but also from blunt, penetrating, or other trauma. Historically, acquired SGS has been related to infections such as tuberculosis and diphtheria. Over the past 40 years, the condition has typically been related to mechanical trauma.

Factors implicated in the development of SGS include the size of the endotracheal tube relative to the child's larynx, the duration of intubation, the motion of the tube, and repeated intubations. Additional factors that affect wound healing include systemic illness, malnutrition, anemia, and hypoxia.

Local bacterial infection may play an important role in the development of SGS. Gastroesophageal reflux (GER) may play an adjuvant role in the development of SGS because it causes the subglottis to be continually bathed in acid, which irritates and inflames the area and prevents it from correctly healing. A systemic or GI allergy may cause the airway to be more reactive, creating a greater chance of developing stenosis.

Acquired SGS is often caused by endotracheal intubation. Mechanical trauma from an endotracheal tube, as it passes through or remains for long periods in the narrowed neonatal and subglottic airway, can lead to mucosal edema and hyperemia. These conditions then can progress to pressure necrosis of the mucosa. These changes have been observed within a few hours of intubation and may progress to expose the perichondrium of the cricoid cartilage. Infection of the perichondrium can result in a subglottic scar.

This series of events can be hastened if an oversized endotracheal tube is used. Always check for an air leak after placing an endotracheal tube because of the risk of necrosis of the mucosa, even in short surgical procedures. This practice is common among anesthesiologists. Usually, the pressure of the air leak should be less than 20 cm of water, so that no additional pressure necrosis occurs in the mucosa of the subglottis.

Frequency

United States

No known frequency has been reported for congenital SGS; the incidence of acquired SGS has greatly decreased over the past 40 years.

• In the late 1960s, when endotracheal intubation and long-term ventilation for premature infants began, the incidence of acquired SGS was as high as 24% in patients who required such care.
• In the 1970s and 1980s, estimates of the incidence of SGS were 1-8%.
• In 2000, Choi reported that the incidence of SGS had remained constant at the Children's National Medical Center in Washington DC, accounting for approximately 1-2% of the children who had graduated from the neonatal ICU (NICU).¹³
• Walner reported that, among 504 neonates who were admitted to the level III NICU at the University of Chicago in 1997, 281 were intubated for an average of 11 days; over a 3-year period, no patients developed SGS.¹⁴
• In 1996, a report from France also described no incidence of SGS in the neonatal population who were intubated with very small endotracheal tubes (2.5 mm internal diameter) in attempts to prevent trauma to the airway.

International

International frequency is the same as that of the United States.

Mortality/Morbidity

Patients can die if they have significant SGS that is left untreated. Difficulty breathing and exercise intolerance can occur with mild, moderate, or severe SGS.

Race

No racial predilection is noted.

Sex

Equal sex distribution is noted.

Age

SGS is observed more often in premature infants because they may require mechanical ventilation for other system or pulmonary problems secondary to their prematurity. The mechanical ventilation can result in airway trauma and, potentially, SGS.

CLINICAL

Section 3 of 10  

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• Clinical
• Differentials
• Workup
• Treatment
• Medication
• Follow-up
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History

Children with subglottic stenosis (SGS) have an airway obstruction that may manifest in several ways.

• In neonates, SGS may manifest as stridor and obstructive breathing after extubation that requires reintubation. At birth, intubation in most full-term neonates should be performed with a 3.5-mm pediatric endotracheal tube. If a smaller-than-appropriate endotracheal tube must be used, narrowing of the airway may be present, which could suggest SGS.
• The stridor in SGS is usually biphasic. Biphasic stridor can be associated with glottic, subglottic, and upper tracheal lesions. Inspiratory stridor is usually associated with supraglottic lesions; expiratory stridor is usually associated with tracheal, bronchial, or pulmonary lesions.
• The level of airway obstruction varies depending on the type or degree of SGS. In mild SGS, only exercise-induced stridor or obstruction may be present. In severe SGS, complete airway obstruction may be present and may require immediate surgical intervention.
• Depending on the severity, SGS can cause patients to have decreased subglottic pressure and a hoarse or a weak voice. Hoarseness or vocal weakness can also be associated with glottic stenosis and vocal cord paresis or paralysis.
• Always assess the history of gastroesophageal reflux disease (GERD). If present, always evaluate GERD prior to surgical intervention. A child who eventually has a diagnosis of SGS often has a history of either laryngotracheal trauma or intubation and ventilation. Frequently, these patients were born prematurely, have bronchopulmonary dysplasia, and may require oxygen administration. The degree of pulmonary disease and the amount of oxygen the child requires may affect the ability to perform decannulation. Prior to surgical intervention, the child should not require a substantial oxygen supplementation.

Physical

A child's physical examination varies depending on the degree of SGS present.

• Perform a complete head and neck evaluation.
• Evaluate the child's initial overall appearance, including the following:
• • Comfort level
  • Presence of increased breathing difficulty, especially during periods of heightened emotion
  • Presence of suprasternal, substernal, or intracostal retractions
  • Presence of any nasal flaring
• Evaluate the child's voice.
• • Presence and nature of stridor
  • Abnormal or normal speaking voice
• Evaluate the child's neurological status.
• In the presence of tracheotomy, evaluate the patient's breathing while tracheotomy is occluded.
• Auscultate the child's lung field and neck to elicit any airway obstructive symptoms and to evaluate the status of their pulmonary function.
• Identify associated facial abnormalities such as cleft palate, choanal atresia, retrognathia, and facial deformities.

Causes

The cause of congenital SGS is in utero malformation of the cricoid cartilage.

The etiology of acquired SGS is related to trauma of the subglottic mucosa. Injury can be caused by infection or mechanical trauma, usually from endotracheal intubation but also from blunt, penetrating, or other trauma. Historically, acquired SGS has been related to infections such as tuberculosis and diphtheria. Over the past 40 years, the condition has typically been related to mechanical trauma.

Factors implicated in the development of SGS include the size of the endotracheal tube relative to the child's larynx, the duration of intubation, the motion of the tube, and repeated intubations. Additional factors that affect wound healing include systemic illness, malnutrition, anemia, and hypoxia.

Local bacterial infection may play an important roll in the development of SGS. GER may play an adjuvant role in the development of SGS because it causes the subglottis to be continually bathed in acid, which irritates and inflames the area and prevents it from correctly healing. A systemic or GI allergy may cause the airway to be more reactive, creating a greater chance of developing stenosis.

DIFFERENTIALS

Section 4 of 10  

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Other Problems to be Considered

Consider any lesion that can cause stridor, including subglottic tumor (benign or malignant), subglottic hemangioma, glottic stenosis, tracheal stenosis, edema from gastroesophageal reflux (GER), and acute infection (eg, croup).

WORKUP

Section 5 of 10  

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Lab Studies

• Specific laboratory studies are not required in patients with subglottic stenosis (SGS).

Imaging Studies

• The criterion standard for evaluation of the airway is direct laryngoscopy and direct bronchoscopy.
• Certain radiographic examinations can help in obtaining a diagnosis and determining the severity of the disease. Usually, the initial radiographic study used to evaluate a child with airway obstruction is anteroposterior and lateral plain neck radiography. Frequently, in a child with SGS, the subglottis appears narrowed and peaked; this is often described as a steeple sign. In a patient with a thin web SGS, lateral plain film radiography may reveal a faint line.
• Fluoroscopy is often performed in children with symptoms of airway obstruction.
• • Fluoroscopy can be used to diagnose lesions of the larynx and trachea.
  • When a barium-enhanced esophagram is added to the procedure, vascular malformations, along with gastroesophageal reflux disease (GERD), may be ruled out.
• CT scans and MRIs are not often used in the primary evaluation of SGS.

Other Tests

• Investigate any indication of GERD. Walner showed that children with SGS have a 3-fold increase in GERD compared with the general pediatric population.¹⁵
• Currently, the best test in evaluating for GER is dual-channel pH probe testing. One probe is placed above the lower esophageal sphincter, and another is placed at the area of the cricopharyngeus near the larynx.
• Walner and Cotton recommend treating GER for 1 month before and 12 months after airway reconstructive surgery, even if only mild disease is present.^{15, 16}
• If moderate or severe GERD is diagnosed, start medical therapy and confirm disease resolution with another pH probe test prior to surgery.
• Do not perform laryngeal reconstruction until GER has resolved.
• If reconstruction is being considered, pediatric laryngologists frequently perform tests to rule out GER, even in the absence of symptoms, because the disease may affect the outcome.
• One concern is that an "allergic" esophagitis may occur and may affect the outcome of surgery.
• • To evaluate for this entity, a esophagogastroduodenoscopy (EGD) is performed with biopsies of the proximal and distal esophagus, stomach, and duodenum.
  • If more than 15 eosinophils are found in the mucosa per high power field, the patient may have "allergic" esophagitis.
  • Evaluation and treatment for GERD must have taken place prior to this evaluation because reflux may elicit eosinophils as well.
  • If "allergic" esophagitis is discovered, then treatment with weeks to months of oral steroids or orally applied inhaled steroids is performed to help diminish the affects of the disease and possibly allow for a better success rate of laryngeal reconstruction.

Procedures

In a child with mild or moderate airway obstruction, perform flexible fiberoptic nasopharyngoscopy and laryngoscopy in the clinic or the emergency department (ED). If extreme airway obstruction is observed or if an active supraglottic infectious process is suspected in a young child, flexible endoscopy may be deferred in favor of formal rigid bronchoscopy in the operating room (OR). However, flexible fiberoptic nasopharyngoscopy may be performed in a controlled setting in the OR because determination of the nature of the supraglottis and glottis in awake, unsedated patients is crucial.

The procedures are described as follows:

• Flexible fiberoptic nasopharyngoscopy and laryngoscopy
• • During flexible fiberoptic nasopharyngoscopy and laryngoscopy, topical anesthesia and decongestion can be accomplished in older infants and children with topical Afrin and lidocaine. A 3-mm endoscope can be used, even in an infant. Pass the endoscope into both nasal cavities to access pyriform aperture stenosis, midnasal stenosis, choanal atresia or stenosis, lesions of the nose and nasopharynx, and the adenoid pad.
  • Pass the endoscope into the superior oropharynx and hypopharynx. The hypopharynx and larynx can be assessed. Identify the structure and position of the supraglottis. Evaluate the epiglottis and arytenoids for malacia or stenosis. Evaluate the position and movement of the true vocal cords. Evaluate edema or erythema of the true vocal cords, epiglottis, and arytenoids.
• Flexible endoscopy
• • This can be performed with the patient in the supine or sitting position. The supine position often results in the obstruction of certain supraglottic processes. If the goal is to obtain the best visualization of the true vocal cords and supraglottis, place a child (even an infant) in the sitting position with his or her neck extended.
  • If the child is older, the voice can be evaluated, and videostroboscopy can be performed to assess the vocal cord waveform and vocal cord mobility.
  • Occasionally, the subglottis can be visualized with flexible endoscopy; however, rigid laryngoscopy and bronchoscopy are the safest procedures and offer the best visualization for the subglottis and tracheobronchial tree.
• Rigid laryngoscopy and bronchoscopy
• • Rigid laryngoscopy and bronchoscopy is the best single test for evaluating airway obstruction in children. The otolaryngologist must have knowledge of the pediatric airway, and the OR must have adequate bronchoscopes and telescopes of various sizes. Prepare all equipment for bronchoscopy, including laryngoscopes, light sources, video documentation equipment, telescopes, and bronchoscopes prior to the child's arrival in the OR. Throughout the procedure, maintain good communication between anesthesiologists, surgical nursing staff, and physicians, so that any potential airway obstruction can be quickly assessed and addressed.
  • Do not further injure the pediatric airway. This point is of paramount importance. Use the smallest bronchoscope or telescope alone for evaluation of the subglottis in a child who does not require ventilation throughout the procedure. This practice allows good visualization without iatrogenic injury to the area. If ventilation is required throughout the evaluation, use a bronchoscope-telescope combination.
  • If a child has a tracheotomy or is not in extreme distress, the child can breathe spontaneously and inhale oxygen and anesthetics through an endotracheal tube in the pharynx while the airways are visualized with a laryngoscope and large telescope. Frequently, the true vocal cords are anesthetized with lidocaine prior to evaluation to help prevent laryngospasm.
  • Determine the size of the child's airway by using endotracheal tubes. Myers and Cotton have established a scale for SGS severity that is based on the child's age and the size of the endotracheal tube that can be placed in the airway with an air leak pressure of less than 20 cm of water.
  • Evaluate the subglottis and glottis for fixation, scarring, granulation, edema, paralysis or paresis, and other abnormalities. Evaluate the distance and caliber of the stenosis. Apply the Myers and Cotton staging system only to circumferential SGS. Glottic stenosis and SGS often occur together and must be considered when reconstruction is planned.
  • Evaluate the maturity of the stenosis. If a firm white scar is present, the stenosis is mature. If the stenosis has a granular or erythematous appearance, GERD, viral infection, allergic esophagitis, or another inflammatory process may be present.
  • Examine the area below the subglottis into the trachea and bronchi for secondary lesions. The suprastomal area is important because pathological stenosis or malacia can influence the choice of surgical procedure. In severe SGS, viewing the suprastomal area requires the passage of a tiny telescope through a narrow subglottis or a telescope or bronchoscope through a tracheotomy site, if available.

Staging

Myers and Cotton devised a classification scheme for grading circumferential subglottic stenosis from I-IV. The scale is based on a percentage of stenosis established by the age of the patient and the size of the endotracheal tube that can be placed in the airway with an air leak less than 20 cm of water pressure.

The percentage of stenosis is evaluated by using endotracheal tubes of different sizes. The largest endotracheal tube that can be placed with an air leak less than 20 cm of water pressure is recorded and evaluated against a scale that has previously been constructed by Myers and Cotton. This grading system mainly applies to circumferential stenosis and does not apply to other types of SGS or combined stenoses, although it can be used to obtain a rough estimate.

• The system contains 4 grades, as follows:
• • Grade I - Obstruction of 0-50% of the lumen obstruction
  • Grade II - Obstruction of 51-70% of the lumen
  • Grade III - Obstruction of 71-99% of the lumen
  • Grade IV - Obstruction of 100% of the lumen (ie, no detectable lumen)
• Evaluate the subglottis and the glottis for any fixation, scarring, granulation, edema, paralysis or paresis, or other abnormalities.
• Evaluate the distance and the caliber of the stenosis.
• Only apply the Myers-Cotton staging system to circumferential SGS. Often, glottic stenosis and SGS occur together and must be considered when planning reconstruction.

TREATMENT

Section 6 of 10  

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Medical Care

No medical therapy for mature subglottis stenosis (SGS) is known. If a granular or immature SGS is noted (see Media file 6), treatment of the inflammatory process with oral or inhaled steroids sometimes can decrease the severity of disease. Findings from animal studies have shown that treatment with antibiotics and steroids can help improve an immature or granular SGS; however, the optimal treatment duration is unknown. Evaluate each case on an individual basis. Once SGS is mature, medical therapy is almost always unsuccessful. However, suspected gastroesophageal reflux (GER) must receive aggressive medical treatment preoperatively and postoperatively for optimal surgical results.

Surgical Care

Indications

Perform surgical repair of SGS if the child has SGS and is symptomatic.

Typically, children with grade I (see Media file 4) or mild grade II stenosis do not require surgical intervention. Children with these conditions may have intermittent airway symptoms, especially when infection or inflammation causes mucosal edema.

Surgical intervention may be avoided if periods of airway obstruction are rare and can be treated on an inpatient or outpatient basis with anti-inflammatory and vasoconstrictive agents, such as oral, intravenous, or inhaled steroids and inhaled epinephrine (racemic treatment). If children with these conditions continue to have intermittent or persistent stridor and airway obstructive symptoms when they are well, or if they frequently become ill, surgical intervention may be necessary.

Development of upper respiratory symptoms during routine infections can indicate whether a child with SGS requires surgical reconstruction. Viral infections of the upper respiratory tract can create swelling in any area of the respiratory epithelium from the tip of the nose to the lungs. If a child with SGS has a cold, bronchitis, or both but does not have significant symptoms of stridor or upper airway obstruction, the airway may be large enough to tolerate stress, and reconstruction may not be needed. A history of recurrent croup suggests SGS.

Occasionally, older children have exercised-induced airway obstruction. At evaluation, these children may have grade I or grade II SGS. Expansion of the airway with cartilage augmentation may allow them to lead a healthy and active lifestyle.

Children with grade III (see Media file 3, Media file 12) or grade IV SGS need surgical management.

Endoscopic procedures

For mild or granular SGS, investigators have reported success with serial endoscopic dilation with or without steroid injections. Healy popularized the use of the carbon dioxide laser as an option for soft circumferential SGS. This procedure involves making incisions in 4 quadrants, followed by dilation. This technique is best used in conjunction with steroids when an immature or granular SGS is present. Normally, use of a laser causes recurrence of the scar in a mature stenosis; however, in unusual types of mature SGS (eg, spiraling SGS), improvement may be accomplished with a few serial carbon dioxide laser excisions (see Media files 7-10).

Although controversial (no good placebo-controlled studies have been performed to back up results), mitomycin-c has been reportedly used after CO₂ lasering of a mature SGS with presumed improved results over lasering alone, with or without the use of oral steroids. The author has used topical mitomycin-c after lysing an immature stenotic web with seeming improved results over lysing and oral steroids alone.

Open reconstruction of subglottic stenosis

Base the approach to open reconstruction of SGS on the location and degree of scarring. Reconstruction often may be unnecessary for SGS classified as grades I and II on the Myers-Cotton scale (ie, as much as 70% obstruction of the subglottic airway). When surgery is necessary on the basis of the severity of symptoms, perform an open reconstruction in mature circumferential SGS. The surgical technique depends on adjacent areas of scarring and on the location and appearance of SGS. For severe SGS, classified as grades III and IV (ie, >70% luminal obstruction), laryngeal expansion is almost always necessary.

The goals of open reconstruction are decannulation or resolution of symptoms, with preservation of the voice by expanding the subglottic airway and stabilizing the expanded frame.

For SGS classifies as grade II (50-70% stenosis), surgical reconstruction depends on many factors, including symptoms, environment, and associated medical conditions.

Various procedures for treating SGS include the following: (1) anterior cricoid split (ACS); (2) single staged procedure: anterior cartilaginous grafting with costal, thyroid, or auricular cartilage; (3) multistaged procedure applying (a) anterior and posterior cartilage grafting, usually with costal cartilage, (b) anterior cartilaginous grafting with a posterior cricoid split and stent placement, (c) posterior grafting with costal cartilage, and (d) anterior and posterior costal cartilage with lateral cricoid splits; and (4) cricotracheal resection.

• Anterior cricoid split
• • In 1980, Cotton and Seid described the use of ACS to avoid tracheotomy in neonates with SGS, good pulmonary and cardiac function, and airway obstructive symptoms after extubation.² ACS allows decompression of the edematous submucosal glands of the subglottis and thus, expansion of the airway.
  • Criteria have been developed to identify the children who are likely to benefit from ACS. These include the following:
  • • Patient weight of more than 1500 g
    • Failure to extubate in identified SGS
    • Oxygen requirement of less than 30%
    • No active respiratory infection
    • Good pulmonary and cardiac function
  • Transport the already intubated child from the ICU, and make horizontal incisions over the cricoid cartilage. Divide the strap muscles in the midline, and identify the thyroid cartilage, costal cartilage, and upper tracheal rings (see Media file 11). Place Prolene stay sutures (4-0) around each side of the anterior component of the cricoid ring. Use a double-sided beaver blade to make an incision in the cricoid ring as far as the tracheal rings and the inferior third to half of the laryngeal cartilage. Then, reintubate the child with an endotracheal tube appropriately sized for his or her age. Do not expand the airway more than necessary, since pressure on the mucosa and persistent SGS can result.
  • Loosely close the skin over the wound, and place a rubber band drain. Mark the Prolene sutures in the cricoid as left and right. Generally, leave the nasal tube in place for 7-10 days. If self-extubation occurs, reintubate from above. Should the endotracheal tube protrude through the airway into the neck during reintubation, the stay sutures can be lifted and crossed to block the cricoid split incision and to direct the endotracheal tube down the trachea. If this procedure is unsuccessful, the stay sutures can be pulled up to the neck and opened so that a tracheal or endotracheal tube can be placed in the airway until the child can be returned to the OR for intubation through the mouth.
  • Administer antibiotics and antireflux medication during the intubation period. Begin the administration of steroids 24 hours before extubation and continue for 48 hours afterwards. Usually, the tube can be removed after 7-10 days. If an air leak around the endotracheal tube is present with a pressure of less than 20 cm of water, extubation should be successful. If airway obstruction that is not amenable to medical therapy (including racemic treatments and steroids) occurs after extubation, return the patient to the OR for evaluation, or immediately reintubate in the ICU if necessary. Complications of ACS are unusual and include pneumothorax, pneumomediastinum, subcutaneous emphysema, wound infection, and persistent SGS.
  • Media files 12-13 were obtained in a 4-month-old infant born 3 months premature who required intubation and ventilation for 3 months. She had a grade III SGS and underwent ACS with intubation and ventilation for 1 week in the ICU. Media file 14 shows the subglottis 1 week after extubation. The size of the larynx was determined with an endotracheal tube, and subsequent dilation of the soft mild restenosis is depicted in Media files 14-15. The child received oral steroids for 5 days and underwent follow-up bronchoscopy 2 weeks later (see Media file 16).
• Single-stage laryngotracheoplasty with cartilage expansion
• • In 1991, Seid et al reported the use of single-stage laryngotracheoplasty (LTP).⁸
  • • Their approach to the airway resembles ACS; however, instead of leaving the area anterior to the fibrosis, a piece of costal cartilage was placed.
    • The procedure was performed in 13 patients with SGS grades I-IV. However, the procedure failed in a patient who had complete glottic and subglottic stenosis (grade IV).
    • The researchers indicated that grade IV SGS was a contraindication to single-stage LTP. Two patients had grade III SGS and a successful result.
    • Seid et al stressed the postoperative course in these patients. Instead of leaving the endotracheal tube in place for 7-10 days, they checked the air leak surrounding the endotracheal tube on a daily basis and removed it when the pressure of the leak was less than 20 cm of water.
    • The authors were also concerned about the transient weakness of the extremities caused by neuromuscular blockade and hydrocortisone. They used vecuronium and benzodiazepines for sedation. Aggressive pulmonary toilet was stressed because wandering atelectasis can be present in a patient who is ventilator dependent for as many as 10 days. The authors stressed the repeated use of a full range of passive extremity motions to decrease the likelihood of transient muscle weakness during the period of induced paralysis for long-term intubation.
    • Seid et al believed that selection of patients was critical and that any child with difficulties in addition to SGS (eg, tracheal problems, true vocal cord paralysis) was not a good candidate for single-stage LTP. The procedure could fail after extubation for reasons other than the newly repaired SGS.
  • In 1995, Rothschild et al reviewed the effectiveness and complications of single-stage LTP.¹⁷
  • • In 104 patients from the Children's Hospital of Cincinnati, repair was successful in 86-92%, depending on the year of correction. The authors further reported that they did not use paralysis in their patients during the 5-day to 10-day period of endotracheal tube placement; they used sedation with chlorohydrate and benzodiazepines instead. In fact, if the patient could tolerate nasotracheal intubation without much difficulty, they were allowed to engage in their usual activities, including eating, playing, and walking. A modified cap placed on the endotracheal tube prevents crust formation in the tube and airway during these activities.
    • Rothschild et al believed that younger children require heavier sedation and increased ventilation secondary to decreased respiratory effort. Neuromuscular paralysis usually was avoided. Among their 104 patients, the researchers found neuromuscular weakness in only one. They did not comment about the presence or absence of pulmonary atelectasis.
    • The average duration of endotracheal tube placement in their patients was 9 days. They did not explain why endotracheal tubes were in place longer than 10 days (as long as 26 d) in 37 children. Twenty-three children, however, had a posterior costal cartilage graft, which normally requires the use of stents for at least 2 weeks to stabilize the cartilaginous framework.
  • Seid and Cotton agreed that ICU staff who are knowledgeable and attentive are important to the success and safety of the procedure.
  • In 1991, Lusk et al also described a single-stage LTP in which auricular cartilage is used for reconstruction when an anterior SGS is repaired.¹⁸ Patients had endotracheal tubes in place for 7-10 days, similar to the duration of intubation in patients in whom an ACS was performed. Lusk et al sutured the cricoid to the strap muscles to help maintain airway patency; their success rate was similar to that of other procedures (ie, approximately 80-90%). This author's experience with the use of auricular cartilage has been less successful. If significant anterior SGS is present, use of cartilage that is rigid enough to maintain the splay of the cricoid cartilage is usually necessary to ensure continued expansion after extubation.
  • Zalzal added to the efficiency of the anterior cartilage single-stage procedure by describing the technique of carving the harvested rib into the shape of a boat with flanges on each end (see Media files 17-18).¹⁹
  • • In this technique, cartilage extends outside the lumen of the trachea, over the cricoid and tracheal rings, to help prevent the lumen from prolapsing into the airway (see Media file 19).
    • With this technique (once any air leak is sealed during the surgery), extubation can be performed without fear of the cartilage requiring further stabilization or prolapsing into the airway.
    • After the procedure has been performed and the child has been admitted to the ICU, air leaks from the neck are checked on a daily basis. Usually, the air leak seals within 48-72 hours; extubation can be accomplished with confidence that the graft is stabilized. In this way, children can avoid the complications of long-term intubation mentioned above.
  • The use of a superior section of the thyroid cartilage, as well as the septal cartilage, as grafting material has been reported. These materials, along with the auricular cartilage, usually do not provide much support. Instead, they act mainly as a patch over the divided area of the cricoid region. In these situations, the stent provides most of the force necessary to keep the lumen open while the surrounding area heals. Some of the other types of cartilage can be used in conjunction with ACS to improve that success rate of the procedure, which has traditionally been 70-80%.
  • Richardson and Inglis performed a prospective study to compare the cricoid split procedure with and without costal cartilage grafting for the treatment of acquired SGS in infants younger than 6 months in whom extubation in the ICU failed.²⁰ The researchers found that results were improved in 90% of patients in whom cartilage was placed between the cricoid rings to expand the airway, compared with 56% in whom cartilage was not placed. This study was prospective and included only 20 patients, but its findings indicate that placing the lumen expander at the time of surgery greatly improves the likelihood that extubation succeeds and adequate airway is maintained.
  • However, Zalzal and Choi pointed out that when the results of laryngotracheal reconstruction was evaluated in 48 patients aged 4 years or younger, success was decreased in children younger than 25 months compared with that of children aged 2-4 years.²¹ (Note that the patients <2 years had SGS that was less severe than that of the older patients.) Zalzal and Choi still recommended laryngotracheal reconstruction in younger patients, because the procedure may aid the child's speech and language development and help prevent tracheotomy complications.
• Anterior and posterior grafting
• • For severe SGS (grade III-IV), anterior and posterior cricoid splitting with costal cartilage grafts placed anteriorly and posteriorly has been effective in expanding the lumen and allowing decannulation. Most authors, including Zalzal and Cotton, agree that when a posterior graft is used, cartilage of sufficient strength must be placed posteriorly to keep the airway expanded.²² Both Zalzal and Cotton have reported success rates higher than 90% with decannulation, frequently achieved with a single procedure. Occasionally, revision surgery is needed.
  • Often, the posterior graft is formed into an ellipse or elongated hexagon and placed so that the perichondrial side of the graft is flush with the mucosa of the posterior subglottic and tracheal wall.
  • Occasionally, flanges can be fashioned on the posterior that can be placed posteriorly and outside the lumen in a manner similar to that of the boat graft (described by Zalzal), which is placed anteriorly. For a posterior graft, sutures to the posterior cartilage split are all placed individually prior to sliding the graft in position (see Media file 20), at which time the sutures can be tied. Seid described using fibrin glue in an animal study to keep a posterior graft in place, avoiding the arduous task of suturing it in.
  • Place the anterior graft in a similar fashion. Construction of the flanges on the anterior graft is not as critical as it is with a single-stage procedure, since children require stents for a minimum of 2 weeks. Usually, stents are used for 4-6 weeks when anterior and posterior grafts are placed and the tracheotomy is maintained. Once the stent is removed, follow-up bronchoscopies are performed to confirm the stenosis has not recurred before the patient is decannulated. Maintenance of a patent airway can be is evaluated with further bronchoscopies.
  • Usually, an Aboulker stent (see Media files 21-22 and Media file 35) or Montgomery T tube (natively as in Media file 25, or cut to fit and used like an Aboulker stent, which are no longer commercially available) is used. Other types of stents also have been used. Often, if the collapse or scar extends into the area of the tracheotomy site, longer-term stent placement is required with an Aboulker stent that is attached to a metal Holinger tracheotomy tube with wire (see Media files 23-24) or a Montgomery T tube. Complications of short-term stent placement (4-6 wk), such as granulation tissue and scarring from the distal end of the short stent, can be prevented with longer-term techniques for stent use.
  • The surgical approach for anterior and posterior grafting is similar to the approach for ACS and anterior cartilage grafting. Specific care for the posterior cricoid split with or without grafting requires visualization of the esophagus after the posterior cricoid cartilage has been incised. During division, take care to spread the cartilage to identify the esophageal mucosa so that no inadvertent injury occurs. Additionally, make the incision in the midline to prevent injury to the recurrent laryngeal nerve and to ensure that an appropriate site is created for placement of the graft.
• Partial cricotracheal resection
• • In Switzerland, Monnier first reported the use of partial cricotracheal resection in 31 pediatric patients with grade III and IV stenosis in whom decannulation with anterior-posterior grafting failed.¹¹ The decannulation rate after cricotracheal resection was 97%. Cotton and others began to evaluate the use of cricotracheal resection because of failures with grade III and grade IV stenoses. Investigators in the "Cincinnati Experience" (ie, Cotton's 20 years of experience) recently reported that decannulation occurred in 90% of children with refractory grade III and IV stenoses.⁶
  • The best candidates for partial cricotracheal resection are patients with severe SGS (grade III-IV) without associated glottic pathologic conditions and with a margin of at least 4 mm in the healthy airway below the vocal folds and above the stenosis. This space allows resection away from the glottic larynx, with anastomosis of healthy mucosa. Expect significant glottic edema to last 4-6 weeks; use a tracheotomy or T tube during the postoperative period to protect the airway until the edema resolves.
  • Perform the procedure with the patient under general anesthesia; the approach to the larynx and trachea is similar to that of other laryngotracheal reconstructive procedures. Vertically enter the airway with the beaver blade in the midline at the level of the cricoid. Make the incision superior to the inferior margin of the thyroid cartilage and inferior to the second tracheal ring. The superior extent of the stenosis can be defined at endoscopy while simultaneously and directly viewing the open wound, so that a precise view of the scarred subglottic segment can be achieved. Make a horizontal cut just above the superior extent of the stenosis, from the anterior aspect to the posterior aspect, stopping at the level of the cricothyroid joint. By staying anterior to the cricothyroid joint at this level, injury to the recurrent laryngeal nerve can be prevented.
  • Make lateral cuts inferior to the cricothyroid joints, and continue inferiorly through the lateral aspects of the cricoid cartilage to expose the posterior cricoid plate. Approach the inferior area of the stenosis, and place stay sutures in the distal normal tracheal segment. Incise the trachea just below the inferior aspect of the stenosis through the anterior lateral portions of the trachea down to the membranous tracheal wall, then dissected this from the esophageal wall at the superior aspect. Connect the superior incision and remove the segment. Next, suture the uninvolved part of the trachea to the anterior thyroid ala and the exposed posterior cricoid plate.
  • During the dissection from the inferior aspect to the superior aspect, take care to dissect in a perichondrial plane over the cricoid to prevent injury to the recurrent laryngeal nerve. If identification of the esophagus is difficult during this portion of the procedure, a palpable dilator can be placed in the esophagus to delineate the esophageal wall. Before anastomosis, remove the scar tissue from the inner aspect of the posterior cricoid plate by using a small curet or drill. Perform a hyoid release procedure to decrease tension at the suture line. In addition, dissect the trachea until 4-5 rings are mobilized to aid in decreasing tension on the suture line. In addition, place 2-3 additional tension-releasing sutures on the thyroid ala and the upper tracheal rings to help release tension from the suture line. Place Proline stitches (0-0) from the chin to the chest of the child to keep the head flexed for a week.
  • In an older child with minimal glottic involvement, a single-stage procedure can be performed with nasotracheal intubation of 7-10 days' duration. In younger children with more severe glottic involvement, a Montgomery T tube can be placed for 4-6 weeks. Take meticulous care to prevent plugging of the T tube and resultant airway obstruction. Stern et al reported good results with decreased morbidity with T tubes in children.²³

Consultations

Consult a pediatric gastroenterologist if GER is suspected or present. Consult a pediatric pulmonologist if chronic lung disease or an oxygen requirement is present.

Diet

No special diet is necessary.

Activity

The need for restriction of activity is evaluated on an individual basis.

MEDICATION

Section 7 of 10  

• Authors and Editors
• Introduction
• Clinical
• Differentials
• Workup
• Treatment
• Medication
• Follow-up
• Multimedia
• References

No known medical therapy for mature SGS is recognized. If a granular or immature subglottic stenosis (SGS) is noted (see Media file 6), treatment of the inflammatory process with oral or inhaled steroids sometimes can decrease the severity of disease. Findings from animal studies have shown that treatment with antibiotics and steroids can help improve an immature or granular SGS; however, the optimal treatment duration is unknown.

Drug Category: Corticosteroids

These strong anti-inflammatory agents also have profound metabolic and immunosuppressive effects.

Drug Category: Antibiotics

Empiric antimicrobial therapy must be comprehensive and should cover all likely pathogens in the context of the clinical setting. Antibiotic selection should be guided by blood culture sensitivity whenever feasible.

FOLLOW-UP

Section 8 of 10  

• Authors and Editors
• Introduction
• Clinical
• Differentials
• Workup
• Treatment
• Medication
• Follow-up
• Multimedia
• References

Further Inpatient Care

Children who undergo various laryngotracheal reconstruction procedures may have different follow-up care and courses, depending on the procedure performed.

If a single-stage laryngotracheal reconstruction or anterior cricoid split (ACS) has been performed, bronchoscopy at extubation is not necessarily required; such decisions are left to the surgeon. However, 1-3 weeks after the procedure, bronchoscopy can be used to assess for any complications. Some authors examine the children after laryngotracheal reconstruction only if they have difficulty. The author often performs laryngoscopy and bronchoscopy 1-2 weeks after extubation to evaluate the airway, since granulation tissue often forms in this period (see Media file 26) and can lead to airway obstruction and scarring. A carbon dioxide laser can be used to remove and control the granulation tissue well (see Media files 27-28). Certainly, any time the child has airway obstructive symptoms, bronchoscopy should be considered.

In a child undergoing 2-stage laryngotracheal reconstruction with grafting and stent placement, the tracheotomy remains in place. The length of follow-up is determined by the duration of stent placement and the quality and quantity of symptoms after stent removal. For short-term stent placement (4-6 wk), follow-up is 2 weeks after stent removal.

If this appears satisfactory, bronchoscopy should be performed at 4 weeks. In the interval, capping of the tracheotomy can be performed intermittently to evaluate for obstruction. If the bronchoscopy at 6 weeks is satisfactory, attempted decannulation can be considered. Prior to decannulation, the tracheotomy tube usually is downsized and plugged intermittently. If the child tolerates plugging, a sleep study can be performed, or the child can be decannulated and watched in the ICU or in a regular hospital room while monitored over night, depending on the individual case. Various methods to evaluate adequate airway prior to decannulation are available.

Walner and Cotton recommend repeat endoscopy at 1, 3, 6, 12, and 24 months after reconstructive surgery. This pattern allows long-term evaluation and detection of a recurring stenosis before it reaches a critical stage. Walner and Cotton also recommend capping and downsizing the tracheotomy in the hospital before decannulation.

Further Outpatient Care

See Further Inpatient Care.

In/Out Patient Meds

Occasionally, inhaled steroids are used to help prevent restenosis following reconstruction. Also, treatment for gastroesophageal reflux (GER) may be necessary if it is suspected or proved, and treatment for "allergic" gastritis may be warranted.

Transfer

Transfer patient only when airway is stable and/or protected.

Deterrence/Prevention

When a child is intubated for any reason, check for an air leak. When possible, the water pressure should be less than 20 cm in order to prevent pressure necrosis and subglottic stenosis (SGS). Use appropriate size endotracheal tubes when intubating a child for any reason. Treat intubated patients with antireflux medications.

Complications

Complications of laryngotracheal reconstruction include the following:

• Failure to correctly repair the stenosis occurs more often in severe stenosis than in moderate or mild stenosis. Zalzal and Choi examined 27 patients in whom laryngotracheal reconstruction failed and found that failure was related to the following:²¹
• • Inappropriate choice of graft
  • Inappropriate choice of stent
  • Inappropriate length of stent
  • Inappropriate duration of stent placement
  • Inadequate assessment and endoscopy
  • Poor postoperative follow-up
  • Anterior suprastomal collapse
  • Slipped Aboulker stent
  • Interactive progression of GERD
  • Keloid formation
  • Failure to repair all abnormalities noted at preoperative evaluation
• Injury to recurrent laryngeal nerve has been reported in a single case of cricoid tracheal resection. Avoidance techniques are outlined in Surgical care.
• The voice quality of patients with glottic stenosis and SGS is decreased and never restored to the preoperative state. However, once the SGS is repaired subglottic pressure can be increased to increases volume and improve speech quality (see Media files 29-33). If an anterior laryngeal fissure is required to repair the SGS, voice quality can worsen, even if the anterior cartilage is displaced only mildly. Therefore, if possible, avoid dividing the anterior commissure.
• Complications from laryngotracheal reconstructive surgery itself include pneumothorax, pneumomediastinum, neck wound infection, chest wound infection, and emphysema.
• Complications during the postoperative ICU course can include those of laryngotracheal surgery itself in addition to atelectasis of lung segments, pneumonia, and neuromuscular weakness with the use of paralytic agents and steroids.

Prognosis

The outcome of laryngotracheal reconstruction depends on its grade and the procedure performed. Most authors report success rates of 80-90% when the patient has undergone successful preoperative evaluation and when the appropriate surgery has been performed (see Media files 33-34). The presence of acute or chronic respiratory illness, GER, or a reactive larynx may decrease the success rate. Choi and Zalzal showed that age can affect success rates; scars are more likely to recur in children younger than 2 years than in others.²⁴

Zalzal noted that, in any child with voice abnormalities before surgery, those abnormalities persisted after surgery.²⁵ Subglottic pressure is required to produce a strong voice. If the narrowed subglottic airway is expanded, subglottic airflow and pressure increase, and the voice usually is stronger (see Media files 29-33). Voice therapy may help relieve nonsevere glottic stenosis over time.

The voice of a patient with SGS, especially those who require reconstruction, may never return to it preoperative state because the following are possible: (1) glottic stenosis, (2) imperfect closure of a laryngofissure through the anterior commissure, and (3) potential vocal cord weakness or tension caused by other laryngeal pathologic conditions. Because reconstructive techniques have improved over the last 20 years, the focus of attention in patients with SGS who require reconstruction has switched from decannulation to decannulation with improved voice outcome.

Patient Education

Teaching parents life-saving maneuvers for a child with a tracheotomy or airway stent following laryngotracheal reconstruction is critical. Also teach parents cardiopulmonary resuscitation (CPR) before their child leaves the hospital.

For further information, see Subglottic Stenosis in Children. For excellent patient education resources, visit eMedicine's Procedures Center. Also, see eMedicine's patient education article Bronchoscopy.

MULTIMEDIA

Section 9 of 10  

• Authors and Editors
• Introduction
• Clinical
• Differentials
• Workup
• Treatment
• Medication
• Follow-up
• Multimedia
• References

Media file 1:  Intraoperative endoscopic view of a normal subglottis.
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Media file 2:  A glottic and subglottic view of a grade III subglottic stenosis in an 18-year-old patient following a motor vehicle accident. The true vocal cords are seen in the foreground. Subglottic stenosis is seen in the center of the picture.
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Media file 3:  Endoscopic view of the true vocal cords in the foreground and the elliptical congenital subglottic stenosis (SGS) in the center of the picture. A close-up view is seen in Media file 5.
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Media file 4:  Subglottic view of very mild congenital subglottic stenosis. Laterally, the area looks only slightly narrow. When endotracheal tubes were used to determine its size, it was found to be 30% narrowed.
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Media file 5:  Endoscopic subglottic view of congenital elliptical subglottic stenosis, a close-up of subglottic stenosis in Media file 3. See progression of treatment with an anterior graft in Media file 11, Media file 17, Media file 19, Media file 20, and Media file 34.
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Media file 6:  An intraoperative view of granular subglottic stenosis in a 3-month-old infant that was born premature, weighing 800 g. The area is still granular following cricoid split. This patient required tracheotomy and eventual reconstruction at age 3 years. True vocal cords are shown in the foreground (slightly blurry).
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Media file 7:  Intraoperative laryngeal view of the true vocal cords of a 9-year-old boy. Under the vocal cords, a spiraling subglottic stenosis can be seen. A close-up view of the stenosis can be seen in Media file 8, and treatment can be seen in Media files 9-10.
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Media file 8:  A close-up view of the stenosis seen in Media file 7. This spiraling subglottic stenosis is not complete circumferentially. Laser therapy was the treatment choice and was successful after 2 laser treatments. Treatment can be seen in Media files 9-10.
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Media file 9:  Continued lasering of the subglottic stenosis seen in Media files 7-8. The reflected red light is the aiming beam for the CO2 laser.
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Media file 10:  Endoscopic view of patient in Media files 7-9 two months postoperative. Some mild residual posterior subglottic stenosis remains, but the child is asymptomatic and the airway is open overall.
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Media file 11:  An intraoperative view of a split cricoid in a patient with elliptical congenital subglottic stenosis (Media file 3, Media file 5). The open airway can be seen in the center of the picture. The wound extends to the inferior one third of the thyroid cartilage. The first 2 tracheal rings also are divided. The graft and its placement are seen in Media file 17, Media file 19, Media file 20, and Media file 34.
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Media file 12:  Preoperative view of a 4-month-old infant with acquired grade III subglottic stenosis from intubation. Vocal cords are in the foreground. A close-up view of the stenosis can be seen in Media file 13, and treatment can be seen in Media files 14-16.
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Media file 13:  An endoscopic subglottic view of a 4-month-old with grade III subglottic stenosis (close-up view of the patient in Media file 12) born premature at 26 weeks' gestation and intubated for 3 months. Treatment can be seen in Media files 14-16.
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Media file 14:  Postoperative view of the patient in Media files 12-13. Following cricoid split, the patient had been intubated for 1 week and extubated for 1 week. Continued treatment can be seen in Media files 15-16.
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Media file 15:  A subglottic view of patient in Media file 14 following dilation with an endotracheal tube to lyse the thin web of scar and a short course (5-day) treatment with oral steroids. Final result can be seen in Media file 16.
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Media file 16:  Postoperative view of a 4-month-old infant with subglottic stenosis following cricoid split (patient seen in Media files 12-15). This picture is 2 weeks after lysis of scar and steroids. Notice very mild recurrence of scaring at the site of previous scar. Overall, the airway is open and patent. The anterior superior area can be seen, with a small area of fibrosis where the cricoid split previously healed.
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Media file 17:  Rib graft for reconstruction of subglottic stenosis carved in boat type anterior graft. The diamond-shaped internal intraluminal component with perichondrium still present is seen on the top section of the rib and the shape of the rib is seen on the backside of the carved out diamond shape. See placement of graft in Media file 19.
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Media file 18:  Anterior rib graft with a diamond shape. Note it measures approximately 1.7 mm in length. Intraluminal site is facing up. Flanges of rib are carved to remain on the outside of the trachea to prevent prolapse into the trachea.
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Media file 19:  An intraoperative aerial view of an anterior cartilage graft in place over the wound. Note external component of the graft still looks like a portion of the rib. The internal component has been carved in a diamond shape. This is an intraoperative photo of the patient seen in Media file 3 and Media file 5. The cartilage graft in Media file 17 was used in this patient for reconstruction. A postoperative photo is seen in Media file 34.
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Media file 20:  An intraoperative side view of the neck with cartilage graft to be placed into the posterior cricoid suspended and having all sutures in position, ready to be tied. All the sutures are placed prior to lowering the graft into position. Then, the sutures are tied. Another view of the graft is seen in Media file 19.