In the early twentieth century, John Winslow wrote that there was no more difficulty in laryngology than treating chronic stenosis of the larynx and trachea. Winslow described cases as “excessively rebellious to treatment” and treatment requiring “patience, persistence, self-sacrifice and skill on the part of the surgeon” and “discomfort or even suffering by the patient.” Three decades later, Chevalier Jackson wrote that curing patients required perseverance over a period of time rarely as short as 3 months and as long as 7 years. Significant strides in surgical technique have been made; this article chronicles the development of laryngotracheal reconstruction in children.
Early history
Documentation of surgical intervention for laryngotracheal stenosis can be found as early as 1871. L. Von Schroetter (Vienna) is credited with devising two methods of dilation—one using metal “olives” after placement of a tracheostomy and the other using hollow hard rubber tubes (known as Schroetter’s bougies) that were inserted through the mouth . In 1885, O’Dwyer (New York) described similar dilation techniques that gained widespread use in the United States. A so-called intubation tube was introduced daily and left in position for approximately 30 minutes. After tube removal, a tracheal canula, or tracheotomy tube, was placed.
In 1903, William Simpson (New York) described the placement of a hard rubber intubation tube “using considerable force” in a 32-year-old woman who had undergone tracheotomy placement 5 months earlier because of a narrowed subglottic airway after a severe cold. The intubation tube remained in place for 4 years and 25 days “with the exception of a few days” and “occasionally during attacks of coughing…the patient had learned the art of pushing the tube back in place with her finger and exerting pressure.” Simpson believed that the outcome was successful because of the pressure exerted by the tube, which caused “absorption” of the tissue that narrowed the airway. Subsequent to his experience with this patient, Simpson had concluded that: (1) the larynx could tolerate a “long continued pressure”; (2) the application of a continuous pressure was superior to the temporary introduction of dilating instruments; and (3) an intubation tube could be worn with relative comfort “indefinitely.”
Although current understanding of the effects of pressure exerted on tracheal mucosa differs significantly from that expressed by Simpson, his work is important in that it provided evidence that a stent could remain in place in the subglottic space for an extended length of time. Additionally, it offered an alternative to repeated dilation procedures.
Development of open surgical approaches
Open approaches for the treatment of subglottic stenosis can be traced as far back as the late 1800s and early 1900s. Killian (Germany) and Sargnon and Barlatier (Lyons) described an operation referred to as a laryngotracheostomy . This procedure involved the creation of a median vertical fissure of the larynx and trachea after a median vertical skin incision. A temporary or permanent fistula was maintained and increasingly larger soft rubber tubes were repeatedly inserted through the tracheostomy site. In 1907, John Winslow (United States) reported his experience with this technique in an 11-year-old patient who had a “fibrous ridge extending obliquely downward from beneath the left chord.” Winslow performed the initial procedure using chloroform and subsequent procedures using cocaine. Although the number of dilations is unknown, the tracheotomy tube was removed and the stoma closed within 6 months. As such, Winslow’s experience reinforced the usefulness of pressure within the airway lumen generated by the rubber tubes. He wrote, “My experience convinces me that we have in laryngotracheostomy an enormous advance in the treatment of intractable and hitherto often incurable stenosis…By means of it, we can control directly the area of necrosis caused by pressure of the dilator.” Winslow also provided a photograph of a special T tube made of hard rubber, which he had devised for insertion through the tracheostomy fistula in particularly difficult cases.
Reports of open procedures for subglottic stenosis in children were rare in the early twentieth century. Laurens of Paris provides one possible explanation for this. In his series of 25 patients reported in 1924, 14 of the 25 children who underwent open procedures died. The cause of death usually was related to infection, most often in the lungs.
In 1927, M. F. Arbuckle (St. Louis) described his experience with three children, aged 3, 4, and 6 years, all of whom had been treated unsuccessfully with repeated intubations after diphtheria or a streptococcal throat infection. He referred to this procedure as an “open operation and skin graft.” The larynx and subglottic larynx were exposed after a median vertical skin incision and a median vertical incision in the airway. Rather than dilate the stenotic segment, Arbuckle excised all visible scar tissue and placed a Thiersch skin graft over the denuded area within the lumen of the airway. This graft was held in place with a rubber balloon, which had a string at the inferior end that was brought out through the tracheostomy site. The balloon was removed after 8 days. In one case, however, it was coughed out only a few hours after the operation. A new balloon then was reinserted; this second balloon was held in place using a stay suture that was passed from the outside, transfixing the larynx, graft, and balloon.
Schmiegelow (Copenhagen) was particularly concerned about the disadvantages of repeated dilation procedures that could go on indefinitely and result in a patient’s giving up before obtaining a definitive cure. In 1929, he published an article documenting what he touted as a “new” method of surgical treatment for stenosis of the larynx that he had been using since 1910. In Schmiegelow’s procedure, the larynx and stenotic segment were exposed via a laryngotracheal fissure. He advocated the entire removal of any strictures present by means of scissors, knives, or punch-forceps so that the lumen of the stenosis could be made as normal as possible and the largest possible drain could be introduced. He specifically commented that it was best to use an India rubber drain slightly wider than the lumen, so that the drain could create a certain pressure against the walls of the larynx. The India rubber stent or drain, as he called it, was fixed into position using a silver wire that was passed through the soft tissues of the neck, the thyroid cartilage, and the drain. This was left in place for weeks or months depending on the specific needs of individual patients.
Schmiegelow reported the use of this operative procedure in treating 12 patients (three ranging in age from 1 to 3 years, six ranging in age from 4 to 6 years, and three ranging in age from 10 to 14 years). He described his results as excellent. Although his “new” procedure did not use a skin graft, it was not significantly different from the procedure described earlier by Arbuckle. It is possible that Schmiegelow may have been unaware of the article published by Arbuckle several years earlier. Alternatively, given that his earliest operation took place in 1910, he may have been vying for credit in describing such a procedure.
Development of open surgical approaches
Open approaches for the treatment of subglottic stenosis can be traced as far back as the late 1800s and early 1900s. Killian (Germany) and Sargnon and Barlatier (Lyons) described an operation referred to as a laryngotracheostomy . This procedure involved the creation of a median vertical fissure of the larynx and trachea after a median vertical skin incision. A temporary or permanent fistula was maintained and increasingly larger soft rubber tubes were repeatedly inserted through the tracheostomy site. In 1907, John Winslow (United States) reported his experience with this technique in an 11-year-old patient who had a “fibrous ridge extending obliquely downward from beneath the left chord.” Winslow performed the initial procedure using chloroform and subsequent procedures using cocaine. Although the number of dilations is unknown, the tracheotomy tube was removed and the stoma closed within 6 months. As such, Winslow’s experience reinforced the usefulness of pressure within the airway lumen generated by the rubber tubes. He wrote, “My experience convinces me that we have in laryngotracheostomy an enormous advance in the treatment of intractable and hitherto often incurable stenosis…By means of it, we can control directly the area of necrosis caused by pressure of the dilator.” Winslow also provided a photograph of a special T tube made of hard rubber, which he had devised for insertion through the tracheostomy fistula in particularly difficult cases.
Reports of open procedures for subglottic stenosis in children were rare in the early twentieth century. Laurens of Paris provides one possible explanation for this. In his series of 25 patients reported in 1924, 14 of the 25 children who underwent open procedures died. The cause of death usually was related to infection, most often in the lungs.
In 1927, M. F. Arbuckle (St. Louis) described his experience with three children, aged 3, 4, and 6 years, all of whom had been treated unsuccessfully with repeated intubations after diphtheria or a streptococcal throat infection. He referred to this procedure as an “open operation and skin graft.” The larynx and subglottic larynx were exposed after a median vertical skin incision and a median vertical incision in the airway. Rather than dilate the stenotic segment, Arbuckle excised all visible scar tissue and placed a Thiersch skin graft over the denuded area within the lumen of the airway. This graft was held in place with a rubber balloon, which had a string at the inferior end that was brought out through the tracheostomy site. The balloon was removed after 8 days. In one case, however, it was coughed out only a few hours after the operation. A new balloon then was reinserted; this second balloon was held in place using a stay suture that was passed from the outside, transfixing the larynx, graft, and balloon.
Schmiegelow (Copenhagen) was particularly concerned about the disadvantages of repeated dilation procedures that could go on indefinitely and result in a patient’s giving up before obtaining a definitive cure. In 1929, he published an article documenting what he touted as a “new” method of surgical treatment for stenosis of the larynx that he had been using since 1910. In Schmiegelow’s procedure, the larynx and stenotic segment were exposed via a laryngotracheal fissure. He advocated the entire removal of any strictures present by means of scissors, knives, or punch-forceps so that the lumen of the stenosis could be made as normal as possible and the largest possible drain could be introduced. He specifically commented that it was best to use an India rubber drain slightly wider than the lumen, so that the drain could create a certain pressure against the walls of the larynx. The India rubber stent or drain, as he called it, was fixed into position using a silver wire that was passed through the soft tissues of the neck, the thyroid cartilage, and the drain. This was left in place for weeks or months depending on the specific needs of individual patients.
Schmiegelow reported the use of this operative procedure in treating 12 patients (three ranging in age from 1 to 3 years, six ranging in age from 4 to 6 years, and three ranging in age from 10 to 14 years). He described his results as excellent. Although his “new” procedure did not use a skin graft, it was not significantly different from the procedure described earlier by Arbuckle. It is possible that Schmiegelow may have been unaware of the article published by Arbuckle several years earlier. Alternatively, given that his earliest operation took place in 1910, he may have been vying for credit in describing such a procedure.
The Jackson era
Before the 1930s, the majority of cases of subglottic stenoses in children resulted from an inflammatory process alone or in combination with an iatrogenic cause . Many children suffered from diphtheria or other infections of the throat and were subjected to repeated and often traumatic dilations. Chevalier Jackson (Philadelphia) cautioned against the placement of a “high tracheotomy” which consisted of a tube placed near or just below the cricoid cartilage. LeJeune and Owens (New Orleans) noted that 90% of their cases were caused by disease or trauma. They wrote that this clinical entity was one of “tissue destruction and its complications: infections, overproduction of scar, contracture and distortion.” They further emphasized that correction of subglottic stenosis was proper management of wound healing. Surgeons now understood the deleterious effects of inflammation and injury to the subglottic and tracheal mucosa with resultant scar tissue formation.
Lejeune and Owens continued to emphasize the importance of complete dissection of all scar, granulation, cartilage, and osseous tissues (at the time of the laryngofissure) that obstructed the laryngeal segment followed by immediate application to the denuded area of a Thiersch graft over a stent. Their rationale was as follows: The most satisfactory healing results from surgically sterile incised wounds with closely approximated borders and conversely, the poorest results are obtained in chronically infected wounds with gaping borders, continually subjected to irritation and infection. In the former, healing by primary intention occurs with the production of a minimum amount of scar and distortion, whereas in the later, healing occurs by secondary intention with the production of an abundance of scar, contracture and distortion.
Although their recommendations with respect to removal of scar tissue are consistent with current knowledge, this work represents a notable change in surgical thinking, given its focus on the importance of the wound healing process in the surgical management of subglottic stenosis.
In an article published in 1936, Jackson made a bold statement regarding subglottic stenosis in children, commenting that almost all cases of congenital and cicatricial stenosis of the larynx were curable. He further maintained that the wearing of a cannula would dominate the entire life of a child and almost certainly create a permanent inferiority complex. In most cases, Jackson favored placement of an elastic rubber core mold, changed weekly. He noted that these molds increase slightly in size as the effect of their contact expanded the tissues; additionally, they become loose as the result of the retraction of the walls of the stenosed larynx under the elastic pressure of the core molds.
In regards to patients who had total laryngeal atresia, Jackson admonished that the cricoid cartilage should not be cut, stating that when no fistula exists, one must be made by perforation or laryngofissure. He advised first performing a tracheotomy and then passing a perforator (a sewing needle–like device with an eye to accommodate a piece of thread) through the mouth and the atretic area to create a lumen. In his words, “above and beyond all things a division of the cricoid cartilage is to be avoided. It is the only complete ring in the airway. The ring can be made to enlarge by growth if elastic core mold dilation is used.” He emphasized that future laryngeal growth would be retarded or arrested if the cricoid cartilage itself, excluding the underlying mucosa, was disturbed.
Jackson’s 1936 article also presented an early description of a tube or stent attached directly to a tracheostomy tube. Sidney Israel (Houston) stated in the discussion section of Jackson’s article that he had “devised a method of attaching a rubber tube to the familiar Jackson tracheotomy with a vertical post.” He held that its use was “very satisfactory and as it did not become detached it served the purpose very well.”
In 1938, Edward Looper (Baltimore) published a case report describing the use of the hyoid bone as an autogenous material for grafting in airway reconstruction in an adult who suffered a severe injury to his larynx after a railroad accident. Looper stressed that this had not been attempted in children and that it should not be used in them. He maintained that the hyoid as a graft has advantages over other material, such as cartilage from a rib, an ear, or some other body part. The concept of using a rib or ear cartilage as a grafting material for the surgical management of subglottic stenosis had thus at least been considered, if not performed, as early as the 1930s in the United States. The use of a two-stage procedure with rib cartilage as a graft to close an open tracheal wound was reported in the German medical literature as early as 1896 .