September 2019 HSC Section 1 Congenital and Pediatric Problems

International Journal of Pediatric Otorhinolaryngology 115 (2018) 10–18

E. Cole et al.

o A symptom was considered to have resolved after fi rst injection if it was not mentioned in the documentation for the clinic visit immediately following the fi rst injection and did not recur for the remainder of the study period or until after additional interven- tion (second injection or formal repair). • The results of all swallow studies (CSE, FEES, and MBS) with thin, nectar-thickened, or honey-thickened liquids o Abnormal fi ndings during CSE included congestion, coughing, choking, desaturations, distress, wet vocal quality, anterior loss, and vomiting. o Findings of interest on FEES and MBS included penetration (without aspiration) and aspiration (with or without cough). o The results of swallow studies occurring prior to fi rst injection (pre) and after fi rst injection but before any additional surgical intervention (post) were extracted. o Only results from patients with the same swallowing exam both before and after injection were evaluated (i.e. both pre- and post- CSE, FEES and/or MBS). Categorical data were reported as n (%) and compared between age groups using Chi-squared or Fisher's exact test with Holm's correction for 3 comparisons (0 – 3 months vs 4 – 11 months, 0 – 3 months vs 12 – 36 months, and 4 – 11 months vs 12 – 36 months). Due to variability in the length of follow-up (time from fi rst injection to last clinic visit), the odds of undergoing additional operative intervention (second injection or formal repair) over time was evaluated using the log-rank test. Continuous variables were evaluated for normality using the Shapiro- Wilk test. When not normally distributed, data were presented as median (range) and comparisons between age groups were performed using Wilcoxon rank-sum tests followed by Holm's correction. Normally distributed data were presented as mean ± standard deviation (SD) and compared between age groups using t-tests with Holm's correction. McNemar exact signi fi cance probability tests were used to compare swallow studies before and after fi rst injection. Agreement between the fi ndings of the three di ff erent swallow studies before and after the fi rst injection was compared using the kappa statistic and McNemar exact signi fi cance probability. Analyses were performed using Stata (StataCorp, College Station, TX). Unless otherwise speci fi ed, p < 0.05 was considered signi fi cant. A total of 102 patients were included in the study. Demographic characteristics, previous management, airway comorbidities, and ages at fi rst injection laryngoplasty are summarized in Table 1 . Most (72/ 102, 70.6%) patients were male. Median gestational age at birth was 37 weeks (range 24 – 41 weeks), and 27 out of 97 (27.8%) patients with available information had been admitted to the neonatal intensive care unit. GERD was present in 98/102 (96.1%) patients. Medical manage- ment including thickening and GERD medications were used in 94/102 (92.2%) and 97/102 (95.1%) patients, respectively. Nine out of 102 (8.8%) patients received G-tubes, and 10/102 (9.8%) underwent su- praglottoplasty (6 concurrently with injection, 3 prior to injection, and 1 both prior to and concurrently with injection). Comorbid lar- yngomalacia, tracheomalacia, and subglottic stenosis were present in 40/102 (39.2%), 9/102 (8.8%), and 14/102 (13.7%) patients, respec- tively. Patients were grouped based on age at fi rst injection laryngoplasty: 0 – 3 months (n = 15), 4 – 11 months (n = 46), and 12 – 36 months (n =41). Fig. 1 A compares demographic characteristics between pa- tients in these age groups; there were no statistically signi fi cant dif- ferences. Fig. 1 B compares ages at fi rst symptoms and diagnosis 2.3. Statistical analysis 3. Results 3.1. Demographics

Table 1 Demographics. Male, n (%)

72/102 (70.6%)

WGA, median (range) a

37 (24 – 41)

NICU, n (%) b

27/97 (27.8%)

NICU Days, median (range)

14 (2 – 132)

GERD, n (%)

98/102 (96.1%)

GERD Interventions Thickened, n (%)

94/102 (92.2%) 97/102 (95.1%) 9/102 (8.8%) 10/102 (9.8%) 40/102 (39.2%) 9/102 (8.8%) 14/102 (13.7%)

GERD Meds, n (%)

G-tube, n (%)

Airway Comorbidities Supraglottoplasty, n (%) Laryngomalacia, n (%) Tracheomalacia, n (%)

Subglottic Stenosis, n (%)

First Injection Months of Age at First Symptoms, median (range) c Months of Age at Diagnosis, median (range) d Months from Symptoms to Diagnosis, median (range) e Months of Age at First Injection, median (range) Months from Symptoms to First Injection, median (range) c Months from Diagnosis to First Injection, median (range) d Months from First Injection to Last Visit, median (range)

1 (0 – 26)

7.5 (0 – 29)

3 (0 – 24)

10 (0.4 – 36) 6 (0 – 24) 1 (0 – 14) 11 (1 – 71)

Second Injection, n (%)

14/102 (13.7%)

Months of Age at Second Injection, mean (SD) Months from First to Second Injection, median (range)

20.3 (8.3) 8.5 (3 – 28)

Repair, n (%)

27/102 (26.5%)

Months of Age at Repair, mean (SD)

20.9 (8.3) 6 (2 – 26)

Months from First Injection to Repair, median (range)

Second Injection or Repair, n (%)

35/102 (34.3%)

Months from First to Second Injection or Repair, median (range)

6 (2 – 28)

Revision, n (%)

1/102 (1.0%)

Months of Age at Revision

59 43

Months from Repair to Revision

Abbreviations: WGA=Weeks Gestational Age (at birth); NICU=Neonatal Intensive Care Unit; GERD=Gastroesophageal Re fl ux Disease; G- tube =Gastrostomy tube.

a Available for 75/102 patients. b Available for 97/102 patients. c Available for 82/102 patients. d Available for 98/102 patients. e Available for 80/102 patients.

between age groups. Median age at fi rst symptoms was older (median 3 months, range 0 – 26 months) for those in the 12 – 36-month group compared with the 0 – 3-month (median 0 months, range 0 – 1 months, z =2.80, p = 0.005) and 4 – 11-month (median 1 month, range 0 – 9 months, z =2.67, p =0.008) groups. As expected, median age at di- agnosis was youngest for patients in the 0 – 3-month group (median 2 months, range 0 – 3 months), followed by the 4 – 11-month (median 5 months, range 1 – 10 months) and 12 – 36-month (median 16 months, range 3 – 29 months) groups (p < 0.001 for comparisons between all age groups). Patients in the 4 – 11-month group experienced longer times from fi rst symptoms to diagnosis and from fi rst symptoms to fi rst injection than patients in the 0 – 3-month age group (p < 0.004 for all comparisons). Similarly, patients in the 12 – 36-month group experi- enced longer times from fi rst symptoms to diagnosis and from fi rst symptoms to fi rst injection than those in the 4 – 11-month or 0 – 3 month groups (p < 0.02 for all comparisons). Median ages at fi rst injection were 3 months (range 0.4 – 3 months), 8 months (range 4 – 11 months), and 18 months (range 12 – 36 months) in the 0 – 3, 4 – 11, and 12 – 36- month groups, respectively. Median time from fi rst injection to the last clinic visit (length of follow-up) for all patients was 11 months (range 1 – 71 months).

3.2. Additional operative management

Overall, 14/102 (13.7%) patients underwent a second injection and

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