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R.L. Flores et al. / Journal of Cranio-Maxillo-Facial Surgery 43 (2015) 1614 e 1619

Table 3 Paired t -test of numeric variables between mandibular distraction osteogenesis successes and failures. Failure by tracheostomy Failure by AHI Any failure

Any failure รพ deceases

No

Yes

p value

No

Yes

p value

No

Yes

p value

No

Yes

p value

74

7

68

6

65

13

-

64

16

n

e

e

e

3.03

2.50

0.006 0.031 0.707

Birth weight (kg)

2.97

2.46

0.067 0.001 0.417

3.00

2.69 0.307

3.02

2.61 0.053

29.9

70.5

30.8

51.7

0.037

29.6

49.3

Age (days) Weight (kg)

32.2

43.0

0.423

3.33

3.75

3.43

3.16 0.419

3.39

3.44 0.898

3.38

3.28

AHI: apnea-hypopnea index. Signi fi cant values ( p < 0.05) are listed in bold.

anomalies, GER, intact palate, airway anomalies, and pre-operative intubation. However, when failure is de fi ned as limited improve- ment in AHI, there are no variables statistically associated with failure. This suggests that multifactorial or unanalyzed variables are in fl uencing failure in this unanalyzed and previously unreported sub-population. As reported previously, laryngomalacia is not associated with failure of MDO across any of the analyzed variables ( p < 0.05) ( Tholpady et al., 2015 ). When this supraglottic disease is separated from other airway anomalies, a clear difference can be seen be- tween the two variables. Non-laryngomalacia airway anomalies are associated with failure by tracheostomy and so should still be approached with the knowledge that MDO will not be successful at a higher rate. The analysis of this patient population provides the basis for score creation, much like the GILLS score ( Rogers et al., 2011;

Abramowicz et al., 2012 ). The score is a well-known predictor of success of tongue-lip adhesion (TLA) in the RS population. It has identi fi ed G ER, I ntubation pre-operatively, L ate operation, L ow birth weight, and S yndromic diagnosis as important predictors of success; fewer than three of these predicts a 100% successful TLA. Of these variables GER and intubation pre-operatively were iden- ti fi ed as being important in this mandibular distraction study. Low birth weight was not shown to be signi fi cant, but approached sig- ni fi cance in the deceased population ( p < 0.06). Syndromic status was not signi fi cant. Interestingly, using paired t -test analysis, a breakpoint was identi fi ed between successful and unsuccessful MDO with regard to the age at performance of distraction. RS patients below 30 days of age at the time of distraction were more likely to be successful than children older than 2 months. The reasons for this age dif- ference could be many, but this is similar to the GILLS score in that

Table 4 Sensitivity, speci fi city, positive and negative predictive value for the combinations of the six variables with corresponding receiver operating characteristic curve scores. Combination of variables analyzed a GAITO GIANTO GIT OAINT OTIG TINGO TING TONI Failure by tracheostomy Speci fi city 100 100 85.7 100 100 100 85.7 100 Negative predictive value 22.6 19.4 54.5 28 31.8 25 31.6 43.8 Positive predictive value 100 100 98.6 100 100 100 98.4 100 Sensitivity 67.6 60.8 93.2 75.7 79.7 71.6 82.4 87.8 Area under ROC curve 0.93 (0.86 e 1) 0.94 (0.87 e 1) 0.93 (0.86 e 1) 0.94 (0.88 e 1) 0.95 (0.9 e 1) 0.96 (0.9 e 1) 0.92 (0.82 e 1) 0.96 (0.92 e 0.99) Failure by AHI Speci fi city 83.3 83.3 33.3 66.7 83.3 83.3 50 50 Negative predictive value 18.5 16.1 28.6 18.2 27.8 21.7 21.4 23.1 Positive predictive value 97.9 97.7 94 96.2 98.2 98 95 95.1 Sensitivity 67.6 61.8 92.6 73.5 80.9 73.5 83.8 85.3 Area under ROC curve 0.75 (0.5 e 1) 0.75 (0.49 e 1) 0.73 (0.53 e 0.94) 0.71 (0.47 e 0.96) 0.77 (0.53 e 1) 0.75 (0.51 e 1) 0.72 (0.5 e 0.95) 0.72 (0.5 e 0.94)

Any failure Speci fi city

84.6 36.7

84.6 32.4

53.8

76.9

84.6 52.4

84.6 42.3

61.5 47.1

69.2 56.3

Negative predictive value Positive predictive value

70

40

95.8

95.5

91.2

94.3

96.5

96.2

91.8

93.5

Sensitivity

70.8

64.6

95.4

76.9

84.6

76.9

86.2

89.2

Area under ROC curve

0.8 (0.64 e 0.97) 0.8 (0.63 e 0.97) 0.8 (0.66 e 0.95) 0.79 (0.63 e 0.95) 0.83 (0.67 e 0.99) 0.81 (0.65 e 0.98) 0.78 (0.63 e 0.94) 0.81 (0.66 e 0.96)

Any failure including all deaths Speci fi city 75

81.3 36.1

43.8 63.6

68.8

68.8

68.8 39.3

50

56.3 56.3

Negative predictive value Positive predictive value

38.7

44

50

42.1

92

93.3

87.1

91.1

91.5

90.6

87.1

89.2

Sensitivity

70.8

64.6

93.8

78.5

83.1

73.8

83.1

89.2

Area under ROC curve

0.74 (0.58 e 0.91) 0.77 (0.63 e 0.92) 0.72 (0.57 e 0.87) 0.77 (0.63 e 0.91) 0.73 (0.56 e 0.9) 0.76 (0.61 e 0.9) 0.75 (0.62 e 0.88) 0.75 (0.61 e 0.9)

AHI: apnea-hypopnea index; ROC: receiver operating characteristic. a The combinations of variables analyzed are drawn from the following: G astroesophageal re fl ux; A ge > 30 days; N eurologic anomaly; airway anomalies O ther than lar- yngomalacia; I ntact palate; and pre-operative intubation.

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