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be explained by a higher rate of prematurity in these com plex comorbidity cases. Along the same lines, Hoff et al 13 found that infants requiring supraglottoplasty in their first 2 months of life incurred a significantly higher rate of surgical failure and revision surgery. For the purposes of categorizing patients and analyzing outcome in this review, we defined significant comorbidities as cases in volving congenital cardiac disease, neurologic compro mise, or severe gastrointestinal reflux. None of the stud ies had objective measures of cardiac or neurologic compromise degree or of reflux severity defined in their data but simply reported the number of patients in each of these categories. In general, these categories of co morbidities were grouped by the reports to evaluate whether they affected surgical outcome. Only 3 stud ies 11,13,14 had sufficient data to determine outcome by each individual comorbidity separately; therefore, for the pur poses of our analysis, all 3 comorbidities were grouped together. The overall goal of this review was to group the re cent reports on supraglottoplasty outcome to determine the relative risk of surgical failure in children with laryn gomalacia. In our review, we analyzed data on 481 grouped patients. We found a relative risk of 7.14 ( P .001) for supraglottoplasty failure in infants with as sociated comorbidities compared with those without. This relative risk ratio signifies that, overall, the percent prob ability of the need for revision surgery, tracheotomy, or gastrostomy tube placement was 7.14-fold higher in chil dren with associated cardiac anomalies than in those with isolated laryngomalacia. Furthermore, the relative risk of aspiration in these complex comorbidity cases was also found to be significantly higher at 4.33. However, we were able to include only 3 reports with sufficient data to ex trapolate enough information about aspiration out come. 3,12,14 In our review, we were not able to separate the relative risk by specific comorbidity owing to the dis parity in categorization in each report that we reviewed. Similarly, because of inadequate substratification in the literature, we were unable to determine the outcome of supraglottoplasty by the surgical technique used or by the severity of laryngomalacia. Therefore, it is unclear from the published medical evidence whether laryngomala cia severity alone would portend a higher risk of supra glottoplasty surgical failure. Potential long-term complications related to supra glottoplasty include supraglottic stenosis, laryngeal car tilage damage, airway fires, granuloma formation, and death. In our review, most of the reported complica tions included persistent laryngomalacia, airway dis tress, and need for tracheotomy and were included in the surgical failure rates reported. Denoyelle et al, 4 in the larg est series included in this review (136 patients), re ported the development of supraglottic stenosis in 5 cases (3.7%). Other studies included in this review did not re port the incidence of this potential complication. Our study has several limitations. Overall, the level of medi cal evidence in the literature is low (level 4), as each study identified and included in our review was retrospective in nature and as such does not account for potential se lection bias. The exact definition of significant associ ated comorbidity may have varied from study to study,

Risk Ratio (95% CI)

% Weight

Surgical Failure Comorbidities/Isolated

Source

1.54 (0.06-41.55) 8.27 (3.30-20.72) 2.70 (0.76-9.51) 43.89 (2.39-805.63) 7.4 (0.38-142.01) 13.57 (2.68-271.02)

8.7 38.8 36.3

Groblewski et al, 3 2009 Denoyelle et al, 4 2003 Day et al, 11 2012 Hoff et al, 13 2010 Richter et al, 14 2009 Senders and Navarrete, 10 2001

3.5 7.5 5.3

0.03125

512 0.5 2 8 32 128 0.125

Risk Ratio

Figure 1. Forest plot of included studies demonstrating the relative risk ratio for surgical failure of supraglottoplasty in patients with associated comorbidities vs those with isolated laryngomalacia. A risk ratio greater than 1 indicates increased failure probability in those with associated comorbidities; the horizontal line, the 95% CI; and the size of the box, the sample size.

% Weight

Risk Ratio (95% CI)

Aspiration Comorbidities/Isolated

Source

0.94 (0.07-11.97) 7.40 (0.38-142.01) 6.57 (1.22-35.31)

42.4 18.5 39.1

Groblewski et al, 3 2009 Richter et al, 14 2009 Eustaquio et al, 12 2011

0.0625

256 1 4 16 64 0.25

Risk Ratio

Figure 2. Forest plot of included studies demonstrating the relative risk ratio for aspiration after supraglottoplasty in patients with associated comorbidities vs those with isolated laryngomalacia. A risk ratio greater than 1 indicates increased aspiration probability in those with associated comorbidities; the horizontal line, the 95% CI; and the size of the box, the sample size.

COMMENT

Laryngomalacia is the most frequent cause of congeni tal stridor, warranting surgical intervention in up to ap proximately 10% of children with the condition. In these instances, endoscopic supraglottoplasty is the surgical ap proach of choice, with good outcomes expected in most cases. Case series demonstrate little difference in out comes or complications among the different endo scopic instruments used for supraglottoplasty. How ever, children with associated congenital anomalies, such as concomitant neurologic disease, 4,15 cardiac disease, 13 and severe gastroesophageal reflux, 10,14 have been re ported to be at increased risk of surgical failure, requir ing higher rates of revision surgery, tracheotomy, or feed ing tube insertion. The exact mechanisms for why these children seem to be at increased risk after surgery are un known but are likely attributable to multifactorial rea sons. Complex hypotonia, increased work of breathing, distorted central cardiopulmonary function, and laryn geal edema are all likely to contribute to varying de grees in individual patients. Day et al, 11 in a multivariate analysis of 74 patients, reported that the only variable that contributed to supraglottoplasty surgical failure was a history of prematurity, suggesting that failure previ ously attributed to associated congenital anomalies may

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