Vs Double-Stage Laryngotracheal Reconstruction

ORIGINAL ARTICLE Single- vs Double-Stage Laryngotracheal Reconstruction

Lee P. Smith, MD; Karen B. Zur, MD; Ian N. Jacobs, MD

Objective: To compare single-stage laryngotracheal re- respectively. Patients who underwent ssLTR and dsLTR construction (ssLTR) and double-stage LTR (dsLTR). were further divided into early and late groups based on whether the posterior graft was sutured in place (early) Design: Retrospective medical record review. or not (late). Overall and operation-specific decannulation rates were 100% and 89%, respectively, for the early Setting: Tertiary care children’s hospital. single-stage group and 100% and 92% for the late group. Regarding the dsLTR group, overall and operation- Patients: Seventy-one patients underwent 84 proce- specific decannulation rates were 88% and 42%, respec- dures (22 ssLTRs and 62 dsLTRs). tively, for the early group and 95% and 79% for the late group. Among all groups, there was no significant differ- Intervention: Review of preoperative disease severity ence in overall decannulation rates (PϾ.05). Single- and surgical outcomes for patients who underwent ssLTR vs dsLTR. stage LTR offered an increased rate of operation-specific decannulation over dsLTR (PϽ.05). However, that dif- Main Outcome Measure: Operation-specific and over- ference was not significant between the late ssLTR and Ͼ all decannulation rates. the late dsLTR groups (P .05).

Results: Regarding ssLTRs, the mean grade of subglot- Conclusion: Careful assessment of preoperative dis- tic stenosis was 2.1 and the overall and operation- ease severity and overall medical status will help sur- specific decannulation rates were 100% and 91%, geons choose between ssLTR and dsLTR, maximizing pa- respectively. The mean grade of subglottic stenosis for tient outcomes for both modalities. double-stage procedures was 2.9, and the overall and operation-specific decannulation rates were 93% and 68%, Arch Otolaryngol Head Neck Surg. 2010;136(1):60-65

ROM ITS EARLIEST DESCRIP- tage of ssLTR is immediate decannulation tions in the 1970s, expan- at the time of reconstruction (or avoid- sion laryngotracheal recon- ance of a tracheotomy altogether). This re- struction (LTR) with cartilage moves a potential source of infection and grafting was performed as a addresses the stoma site concurrent with 2-stage,F or double-stage, procedure.1 By defi- LTR. In addition, prolonged stenting, with nition, in double-stage LTR (dsLTR), the tra- its potential complications, is also avoided. cheostomy tube is kept in place at the con- These advantages must be weighed against clusion of the procedure. If the procedure the potential for airway complications in the is successful, the patient is decannulated perioperative period and the complica- during a separate hospital admission, usu- tions that may result from prolonged ally several weeks or months later. In 1988, endotracheal intubation.11,12 Prescott2 presented perhaps the first de- Although good data regarding surgi- scription of single-stage LTR (ssLTR) when cal outcomes for ssLTR vs dsLTR remain he described the results of 5 children who elusive, a consensus has emerged in the had their tracheostomy tube removed at the literature regarding when to perform each time of LTR. Prescott2 advocated stenting procedure. Most researchers6,8,11 advo- with an endotracheal tube for approxi- cate dsLTR in children with complex mul- Author Affiliations: Division of mately 10 days postoperatively. Since then, tilevel stenosis, significant neurologic defi- Pediatric Otolaryngology, 3-10 The Children’s Hospital of multiple researchers have confirmed the cits, significant lung disease, or anatomy Philadelphia, Philadelphia, feasibility of ssLTR, outlining their results that makes reintubation technically diffi- Pennsylvania. and refining the indications. The advan- cult (craniofacial or vertebral anoma-

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©2010 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 10/03/2021 lies). There is also a trend toward performing dsLTR in patients with higher grades of stenosis (Cotton-Myer grade Table 1. Surgical Procedures III or IV),3,5,8 although we and others13-16 would advo- cate for partial cricotracheal resection (pCTR) in select Procedures, No. children with severe grade III or IV subglottic stenosis Type of Procedure (N=84) (SGS) whose disease is separated from the larynx by an ssLTR adequate margin. In this study, we attempt to carefully ACCG 16 APCCG 1 define our surgical results for ssLTR and dsLTR to help PCCG 3 surgeons choose between these surgical modalities. ATAG 1 AACG 1 METHODS Subtotal 22 dsLTR ACCG 8 Institutional review board approval was obtained to perform a APCCG 37 retrospective medical record review of all patients who under- PCCG 11 went LTR with cartilage grafting at a tertiary care children’s hos- ATAG 1 pital (The Children’s Hospital of Philadelphia) between Feb- ACCG-PCS 3 ruary 1, 2000, and April 15, 2008. All the operations were ACCG-PSS 2 performed by 1 of 2 senior surgeons (K.B.Z. or I.N.J.). Pa- Subtotal 62 tients who were treated endoscopically or who underwent cricotracheal or tracheal resection were excluded from this study. Abbreviations: A, anterior; ACG, auricular cartilage graft; CCG, costal In an effort to compare the outcomes for ssLTR vs dsLTR, de- cartilage graft; ds, double stage; LTR, laryngotracheal reconstruction; P, posterior; PCS, posterior cricoid split; PSS, posterior scar split (division of tails relating to preoperative disease severity and location, sur- scar tissue that had replaced the posterior cricoid); ss, single stage; gical procedure performed, stent type, and duration of stent- TAG, thyroid ala graft. ing or intubation were recorded. As previously defined by Hartnick et al,5 operation-specific and overall decannulation rates were recorded. Briefly, operation-specific decannulation performed (8 ssLTRs and 12 dsLTRs). Five patients who refers to the rate at which an open surgical procedure is asso- underwent revision had their first procedure performed at ciated with subsequent decannulation or extubation without another institution. One child was unavailable for fol- the need for further open airway surgery. The overall decan- low-up 3 months after reconstruction. This child was in nulation rate includes patients who subsequently receive an ad- the early dsLTR group and was considered a surgical fail- ditional open surgical reconstruction.5 The number of postop- ure (operation specific and overall). Three patients from erative endoscopic procedures and significant complications the late dsLTR group required major endoscopic proce- were also identified. dures before decannulation. Two patients required a car- A reconstruction was defined as single stage if the patient bon dioxide laser partial right arytenoidectomy, and one did not have a tracheostomy tube in place at the conclusion of the procedure. This included patients who had their tracheos- of them also received carbon dioxide laser treatment of the tomy tube removed at the time of surgery and patients who did lingual tonsils. One patient required multiple postopera- not have a tracheostomy tube present preoperatively. All pa- tive balloon dilations, with microdebrider removal of a por- tients who underwent ssLTR remained nasotracheally intu- tion of the anterior costal cartilage graft that had pro- bated after their procedure. Patients who underwent dsLTR had lapsed into the airway. These 3 patients were considered a tracheostomy tube present at the conclusion of their proce- operation-specific surgical successes. One patient was ex- dure. In all cases, these patients had a tracheostomy tube pre- cluded from the analysis. This child acutely lost her air- operatively. way and expired 1 day after dsLTR. Patients who underwent ssLTR and dsLTR were further sub- Two patients from the dsLTR group had a postopera- divided into early and late groups. In late 2003, we began in- tive pneumothorax (2.4%); one resulted from pleural in- serting and securing posterior grafts without sutures, as previously described.17 Patients operated on before this technical jury at the rib graft harvest site and resolved without a advancement were included in the early group and after in the chest tube. The other patient had subcutaneous emphy- late group regardless of whether they had a posterior graft placed. sema and a pneumothorax identified on postoperative day All patients who underwent dsLTR in the early group had an 2 that required neck exploratory surgery with replace- Aboulker stent, and almost all (93%) in the late group had a ment of an extruded Penrose drain and a chest tube. Two cylindrical Silastic (Dow Corning Corp, Midland, Michigan) patients from the dsLTR group had significant postop- stent placed. As we previously described,18 this stent was fash- erative neck infections (2.4%). One of them required in- ioned by cutting one limb off of a Montgomery T-tube (Hood cision and drainage early in the postoperative period, and Laboratories, Pembroke, Massachusetts). Single-stage LTRs the other had resorption of an anterior costal cartilage and dsLTRs were otherwise performed similar to previous graft. Both of these patients failed their respective sur- descriptions.11 gical reconstructions, and one of them was decannulated after a revision procedure. The other patient remains de- RESULTS pendent on a tracheotomy tube 3 years postoperatively. One patient who underwent ssLTR required urgent rein- Eighty-four (22 single- and 62 double-stage) LTRs with car- tubation for symptomatic stridor 2 days after extubation. tilage grafting were performed on 71 patients during the This patient responded well to corticosteroid therapy and study (Table 1). Mean patient age at the time of surgery remains decannulated and asymptomatic 2 years after his was 51.6 months, and median age was 41 months (age LTR. The remainder of the patients who underwent ssLTR range, 5-212 months). Twenty revision procedures were had no significant complications.

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©2010 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 10/03/2021 eration-specific failures (1 each from the early and late Table 2. Preoperative Characteristics of the 71 Patients groups); both were successfully treated with a vocal cord by Type of LTR lateralization procedure, only one of whom actually required interval replacement of the tracheostomy tube. Characteristic ssLTR dsLTR Overall and operation-specific decannulation rates were Procedures, No. 22 62 93% and 68%, respectively, for the dsLTR group (88% Patient age, mean, mo 57 50 and 42% for early patients and 95% and 79% for late pa- Revision procedures, No. (%) 8 (36) 12 (19) tients). Among all groups, there was no significant differ- SG stenosis grade, mean (SD) 2.1 (0.7) 2.9 (0.4) Ͼ Site of stenosis, No. (%) ence in overall decannulation rates (P .05). Single-stage Tracheal 6 (27) 0 reconstruction offered an increased rate of operation- Glottic 2 (9) 6 (10) specific decannulation compared with dsLTR (PϽ.05). SG 7 (32) 20 (32) However, no significant difference in operation-specific SG and tracheal 3 (14) 1 (2) decannulation was seen when patients in the late ssLTR SG and glottic 4 (18) 23 (37) group were compared with those in the late dsLTR group SG and supraglottic 0 2 (3) Ͼ Supraglottic and tracheal 0 1 (2) (P .05). Although patients who underwent late dsLTR Supraglottic and glottic 0 1 (2) had a significantly higher operation-specific decannula- Glottic and SG and tracheal 0 2 (3) tion rate compared with those with early dsLTR (PϽ.05), Supraglottic and glottic and SG 0 6 (10) no such difference was seen between the 2 ssLTR groups Multilevel stenosis, No. (%) (PϾ.05). Patients in the ssLTR and dsLTR groups un- Bilevel 7 (32) 28 (45) derwent approximately 4 postoperative airway endosco- Trilevel 0 8 (13) Vocal cord status, No. (%) pies with no statistically significant differences between Mobile bilaterally 15 (68) 20 (32) the groups. The dsLTR group had significantly fewer in- Immobile bilaterally 2 (9) 5 (8) patient perioperative hospital days than did the ssLTR Reduced mobility bilaterally 5 (23) 35 (56) group (PϽ.05). Immobile unilaterally 0 1 (2) Surgical results were further subdivided by grade of Reduced mobility unilaterally 0 1 (2) SGS. Twenty ssLTRs and 55 dsLTRs were performed on patients who had SGS. Table 4 illustrates operation- Abbreviations: ds, double stage; LTR, laryngotracheal reconstruction; SG, subglottic; ss, single stage. specific and overall decannulation rates for these patients subdivided by Cotton-Myer grade of SGS. The overall decannulation rate for all patients who underwent Table 2 outlines the preoperative characteristics of ssLTR was 100%. The operation-specific decannulation patients who underwent ssLTR or dsLTR in this series. rate for grade I was 100%, for grade II was 91%, and for Eight of the 22 ssLTRs (36%) and 12 of the 62 dsLTRs grade III was 100%. No ssLTRs were performed on pa- (19%) were revisions, a difference that was not signifi- tients with grade IV SGS. Regarding patients with dsLTR, cant (PϾ.05). Mean age at the time of surgery was 57 the overall decannulation rate for grade II was 89%, for months for patients who underwent ssLTR and 50 months grade III was 93%, and for grade IV was 100%. The op- for those who underwent dsLTR, a difference that was eration-specific decannulation rate was 67% for grade II, not significant (PϾ.05). For patients who had SGS, the 70% for grade III, and 0% for grade IV. No double-stage mean Cotton-Myer grade for the ssLTR group was 2.1 procedures were performed on patients with grade I SGS. and for the dsLTR groups was 2.9, a difference that was The Fisher exact test was used to compare operation- significant (PϽ.05). In contrast to patients who under- specific and overall decannulation rates for patients with went ssLTR, most patients (58%) who underwent dsLTRs grade II or grade III SGSs, and no significant difference had multilevel stenosis. Twenty-eight patients (45%) had between patients who underwent ssLTR and dsLTR was stenosis at 2 different levels of the airway, and 8 pa- identified (PϾ.05). tients (13%) were actually affected at 3 different levels. Seven patients (32%) in the single-stage group were affected at 2 levels, and none had disease at more than 2 COMMENT levels. ␹2 Analysis revealed that patients in the double- stage group were significantly more likely to have mul- Children with airway stenosis may present with disease tilevel stenosis (PϽ.05). Sixty-eight percent of patients of varying severity and locations in the airway. These pa- who underwent single-stage surgery had normal vocal tients also often have significant comorbid conditions in- cord mobility preoperatively vs 32% of patients who had cluding neurologic, pulmonary, cardiac, and gastroin- dsLTR, a difference that was significant (PϽ.05). Based testinal disorders. In addition to endoscopic techniques, on grade of SGS, number of airway subsites affected, and there are multiple options for airway reconstruction in- vocal cord function, patients who underwent dsLTR had cluding ssLTR, dsLTR, ss-pCTR, and ds-pCTR. Pediat- significantly worse preoperative disease than did those ric airway surgeons must carefully consider which of these who underwent ssLTR. modalities is optimal for achieving safe and expeditious The surgical results for ssLTR vs dsLTR are outlined surgical reconstruction for each patient. in Table 3. Regarding ssLTRs, overall and operation- In recent years, several studies3,5,8 have looked at sur- specific decannulation rates were 100% and 91%, respec- gical outcomes after ssLTR and dsLTR. Saunders et al,8 tively (100% and 89% for early patients and 100% and in 1999, provided the first comparison of ssLTR vs dsLTR. 92% for late patients). Two patients were considered op- In their article, the overall decannulation rate was 91.4%

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ssLTR dsLTR

All Early Late All Early Late Procedures, No. 22 91362 19 43 SGS grade, mean 2.1 2.0 2.1 2.9 2.9 2.9 Hospital days, mean, No. 13.5 13.9 13.3 7.0 8.4 6.4 Days with stent/ETT, mean, No. 6.8 7.8 6.2 21.2 31.8 16.8 Postoperative MLB, mean, No. 4.0 4.7 3.5 4.0 4.4 3.8 Operation-specific decannulation rate, % 91 89 92 68 42 79 Overall decannulation rate, % 100 100 100 93 88 95

Abbreviations: ds, double stage; ETT, endotracheal tube; LTR, laryngotracheal reconstruction; MLB, microlaryngoscopy with bronchoscopy; SGS, subglottic stenosis; ss, single stage.

Table 4. Surgical Decannulation Rates Separated by Grade of SGS

Decannulation Rate

ssLTR dsLTR SGS Gradea Patients, No. Operation Specific, % Overall, % Patients, No. Operation Specific, % Overall, % I 4 100 100 0 NA NA II 11 91 100 9 67 89 III 5 100 100 44 70 93 IV 0 NA NA 2 0 100

Abbreviations: ds, double stage; LTR, laryngotracheal reconstruction; NA, not applicable; SGS, subglottic stenosis; ss, single stage. a The degrees of subglottic stenosis are as follows: I, less than 70%; II, 70% to 90%; III, 91% to 99%; and IV, 100%.

for the ssLTR group and 61.8% for the dsLTR group. Al- decannulation rates compared with dsLTR. Although though acknowledging that the severity of disease seemed the dsLTR group had significantly worse disease, to be worse in the dsLTR group, the authors suggest that operation-specific decannulation rates were similar ssLTR is the procedure of choice for uncomplicated pe- between the late ssLTR and late dsLTR groups. Further- diatric SGS.8 Hartnick et al5 recognized the problem of more, when we compare patients with similar grades of comparing surgical results in patients with different dis- SGS, only a statistically insignificant trend toward ease severity. In their article, they identified a cohort of improved outcomes was noted for patients who under- patients with isolated SGS, divided them by Cotton- went ssLTR vs dsLTR (Table 4). Myer grade of stenosis, and presented surgical results for Substantially more posterior grafts were performed in ssLTR, dsLTR, and pCTR. Although formal statistical patients who received dsLTR (Table 1). This is an un- analysis was not performed, their data suggest that ssLTR avoidable confounder in a retrospective review of pa- offers higher operation-specific and overall decannula- tients treated in our practice. Placement of a posterior graft tion rates than does dsLTR, even when grade of SGS is is indicated when LTR with anterior graft alone does not considered. The authors acknowledge the unavoidable adequately expand the airway and in cases of isolated pos- confounder with this retrospective review that the very terior glottic stenosis or SGS.19 In our practice, patients decision to perform ssLTR over dsLTR suggests that the with higher-grade stenosis and glottic disease are prefer- surgeon believes that the patient has a higher likelihood entially treated in a double-stage manner. Thus, patients of success.5 More recently, Agrawal et al3 examined their undergoing dsLTR are more likely to have a posterior graft. results with 53 patients who underwent ssLTR and dsLTR. Findings from previous studies3,5,8 suggest improved Similar to previous studies, overall decannulation rates results with ssLTR compared with dsLTR. Recently, in- were higher in the ssLTR group, although formal statis- cremental technical advances, such as the sutureless pos- tical analysis was not performed and disease severity was terior graft, shorter stenting times, use of cylindrical Si- worse in patients who underwent dsLTR.3 lastic stents, and use of fibrin glue have become more Similar to previously published studies,3,5,8 children widely used during LTR.17 We believe that some of these undergoing dsLTR herein had significantly worse dis- advances have more significantly affected the results for ease based on vocal cord function, grade of SGS, and patients undergoing dsLTR, specifically, shorter stent- number of involved airway subsites. This makes direct ing time and use of a cylindrical Silastic stent. Indeed, comparisons regarding success rates challenging. Nev- although this study demonstrated improved outcomes in ertheless, we found no statistically significant differ- patients who underwent late compared with early dsLTR, ences in overall decannulation rates between patients no such difference was seen between the single-stage who underwent ssLTR vs dsLTR. Similar to previous groups. This study is the first, to our knowledge, to dem- studies,5,8 ssLTR offered increased operation-specific onstrate similar outcomes for ssLTR and dsLTR.

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©2010 American Medical Association. All rights reserved. Downloaded From: https://jamanetwork.com/ on 10/03/2021 The present operation-specific and overall decannu- tomy site. Isolated suprastomal abnormalities, how- lation rates compare favorably with those of previously ever, usually can be handled with a simple tracheosto- published studies.3,5,6,8 Aside from meticulous surgical mal revision procedure before a decannulation trial technique, there are several possible explanations. First, and should not be an indication for ssLTR. Significant some other large medical centers have a disproportion- vocal cord involvement (with or without vocal cord ate number of patients who are seen after failing previ- immobility) or tracheomalacia may be relative contra- ous reconstruction at an outside institution. Thus, a sig- indications to ssLTR because some authors have sug- nificant portion of their patients may be more challenging gested that this leads to worse outcomes.6,8 Partial revision cases.5 Indeed, only 5 of 71 patients (7%) in the CTR should be considered for severe grade III and present series received a previous open airway proce- grade IV SGS, especially in patients whose disease is dure at an outside institution. Second, before perform- separated from the larynx by an adequate margin.13-16 ing LTR, we are careful to ensure that the larynx and tra- Indeed, 2 of the present patients with grade IV SGS chea are prepared to accept a cartilage graft. We will not who failed dsLTR were decannulated after pCTR. perform an airway reconstruction on an “active larynx,” Double-stage LTR may be considered for patients with and we have cancelled operations on the day of surgery grade II and mild grade III stenosis whose disease is because of significant laryngeal inflammation noted dur- separated from the tracheostomy site by an adequate ing microlaryngoscopy. Although we do not restrict LTR margin, particularly if they have significant disease at procedures to the summer months, we make every ef- the level of the vocal cords. Double-stage LTR should fort to ensure that patients do not have a viral upper res- be preferentially performed in children with complex piratory tract infection at the time of reconstruction and multilevel stenosis, significant neurologic deficits, sig- have all patients evaluated by the gastroenterology ser- nificant lung disease, or anatomical features that make vice for laryngopharyngeal reflux disease and eosino- reintubation technically difficult.6,8,11 Surgeons should philic esophagitis. Although not universally agreed on also consider dsLTR for patients with more severe by all researchers,20 we believe that routine preopera- grade III or grade IV disease involving the vocal cords. tive screening for gastroesophageal disease significantly To facilitate expeditious decannulation, surgeons improves outcomes. Recently, we have become even more should consider dsLTR in complex patients with sig- fastidious about our preoperative gastrointestinal tract nificant comorbidities rather than waiting for the evaluation, performing routine blind biopsies of the comorbid conditions to resolve before proceeding with esophagus and impedance probe testing with the pa- ssLTR. tient taking laryngopharyngeal reflux medications. An- In conclusion, pediatric LTR may be approached in a ecdotally, we have noticed an increase in the number of single- or double-stage manner. Although patients in this patients diagnosed as having eosinophilic esophagitis and series who underwent dsLTR had significantly worse dis- have identified several patients with clinically signifi- ease, decannulation rates were similar to those of pa- cant nonacidic laryngopharyngeal reflux. As this article tients who received ssLTR. These results suggest that an suggests, we are careful to select which operation is ap- individualized approach to the treatment of pediatric air- propriate for each individual patient. Finally, at The Chil- way stenosis may maximize decannulation rates and fa- dren’s Hospital of Philadelphia, we are fortunate to be cilitate expeditious decannulation. part of an outstanding team of airway specialists, includ- ing but not limited to nurses, nurse practitioners, speech language pathologists, intensivists, gastroenterologists, and pulmonologists. Submitted for Publication: March 29, 2009; final revi- Our inability to demonstrate significantly better sion received May 18, 2009; accepted September 21, 2009. outcomes for patients who underwent late ssLTR may Correspondence: Ian N. Jacobs, MD, Division of Pedi- result from the relatively small sample size in this atric Otolaryngology, The Children’s Hospital of Phila- study. Nevertheless, we believe that these data can be delphia, Richard D. Wood Center, First Floor, 34th and used to promote an individualized approach to open Civic Center Boulevard, Philadelphia, PA 19104-4399 airway reconstruction that includes ssLTR, dsLTR, and ([email protected]). pCTR. Even if outcomes are similar, ssLTR offers the Author Contributions: All authors had full access to all advantage of immediate decannulation and allows for the data in the study and take responsibility for the in- surgeons to address the stoma site concurrent with tegrity of the data and the accuracy of the data analysis. LTR.11,12 The obvious disadvantages with ssLTR are Study concept and design: Smith, Zur, and Jacobs. Acqui- the complications associated with prolonged endotra- sition of data: Smith. Analysis and interpretation of data: cheal intubation and the need to rely on the newly Smith, Zur, and Jacobs. Drafting of the manuscript: reconstructed airway immediately after extubation, Smith. Critical revision of the manuscript for important increasing the potential for airway complications dur- intellectual content: Smith, Zur, and Jacobs. Statistical ing the perioperative period.11,12 In addition, the peri- analysis: Smith. Administrative, technical, and material operative hospital admission was approximately 6.5 support: Smith, Zur, and Jacobs. Study supervision: Zur days longer for the single-stage group (Table 3). Thus, and Jacobs. ssLTR should be used for moderate-grade SGS (grade Financial Disclosure: None reported. II and mild grade III) in patients without significant Previous Presentation: This study was presented at the neurologic or pulmonary disease. Single-stage LTR is American Society of Pediatric Otolaryngology Annual particularly useful for stenosis involving the tracheos- Meeting; May 30, 2009; Seattle, Washington.

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