Patient outcomes after neonatal tracheotomy: A retrospective case-control study.

To the Editor: Although reports of neonatal upper airway obstruction in patients in neonatal intensive care units (NICUs) are common, neonatal tracheotomy is rare, possibly due to the small number of patients and the even smaller number of well-equipped hospitals to do a successful operation in neonates. However, despite the extensive efforts of qualified, skilled neonatologists and pediatric otorhinolaryngologists in performing a neonatal tracheotomy, some neonates did not undergo this surgery. When a neonate meets the indications of tracheotomy but his/her parent refuses to do the operation, he/she is facing a higher risk to be discharged against medical advice (DAMA) as per their parents' wish. To explore the preliminary evidence of neonatal tracheotomy in China, as well as to find out the outcome difference between neonates who under went tracheotomy and those who did not undergo tracheotomy when they met the surgical indications, we conducted a retrospective study. Patients were treated in Beijing Children's Hospital. Patients were categorized into the tracheotomy group or non-tracheotomy group according to whether their parents signed the tracheotomy consent and finally completed the tracheotomy. This study was approved by the Ethics Committee of Beijing Children's Hospital, Capital Medical University (No. [2021]-E-139-R) and was registered in the Chinese Clinical Trail Registry (No.ChiCTR2100050720). All parents/guardians of participants provided written informed consent. The patients were admitted due to upper airway obstruction from January 2016 to August 2021. The inclusion criteria were: (1) the age at admission was ≤44 weeks of corrected age (for preterm infants) or ≤28 days of age (for term infants), and (2) the patients met the indications of tracheotomy after careful assessments made through group discussions among neonatologists, otorhinolaryngologists, and radiologists. Patients facing medical conflicts or lawsuit were excluded from this study. Discharge criteria for patients were: (1) enteral feeding at ≥140 mL∙kg-1∙day-1, well-tolerated for ≥3 days, (2) no antibiotics were needed, (3) vital sign stability for ≥3 days, and (4) blood Na+/K+/gas/blood urea nitrogen level or liver function within 48 h before discharge was normal or slightly abnormal but required no medical intervention. If the infant was discharged without meeting the above discharge criteria, the patient was considered DAMA in this study. Statistical analysis was performed using SPSS (IBM, Chicago, IL, USA). Values of P <0.05 were considered statistically significant. Normally distributed continuous data were expressed as mean ± standard deviation and compared using the t-test. Skewed continuous data were expressed as median (P25, P75 ) and compared using the non-parametric Mann–Whitney U test. Categorical data were expressed as frequencies (%). The Chi-squared test was used to compare two sets of independent samples, whereas the likelihood Chi-squared test was used to compare three sets of independent samples. The pre- and postoperative weight gain rates were compared using the non-parametric Wilcoxon rank-sum test. The associations between the weight gain rate and neonate intensive care unit (NICU) outcomes were analyzed using the non-parametric Spearman's correlation coefficient test (ρ). In our study, a total of 26 infants were enrolled in this study, implying 1.96/1000 admissions (26/13,263). The tracheotomy and non-tracheotomy groups comprised 14 and 12 patients, and the median age at admission was 16.0 (6.25–25.0) days and 14.0 (2.5–21.5) days, respectively (P = 0.471). There were no significant differences in demographic data between the two groups [Table 1]. All patients required intubation (19/26, 73.1%) or nasal continuous positive airway pressure support (7/26, 26.9%). Except for one patient for whom the cause of upper airway obstruction symptoms could not be confirmed, the other 25 infants were diagnosed at a median age of 14 (4–23) days. There were eight and six infants with malformation(s) in organs other than those of the respiratory tract in the tracheotomy and non-tracheotomy groups, respectively (P = 0.716). The top two diagnoses in the tracheotomy group were bilateral vocal cord paralysis (7/14, 50%) and congenital anomaly/defect of the throat/larynx (3/14, 21.4%), and those in the non-tracheotomy group were bilateral vocal cord paralysis (6/12, 50%) and vocal cord/subglottic masses (3/12, 25%). There were no significant differences in the diagnosis between the two groups [Table 1]. The top three diagnoses and malformations in organs other than respiratory tract of each patient were shown in Supplementary Table 1, https://links.lww.com/CM9/B499. Table 1 - Comparison between infants with and without tracheomtomy who meet the indications. Descriptions Tracheotomy group (n = 14) Non-tracheotomy group* (n = 12) χ 2/Z/t values P values Gestational age (weeks) 37.70 ± 3.80 38.99 ± 1.83 1.087 0.288 Birth weight (g) 2823.57 ± 948.89 3320.83 ± 378.76 1.699 0.102 Male 9 7 0.097 0.756 SGA 5 3 0.188 0.665 Onset within 24 h after birth 12 11 0.224 0.636 Corrected age on admission (weeks) 42.22 ± 5.03 40.95 ± 1.87 -1.129 0.270 Weight on admission (g) 3032.14 ± 569.50 3290.83 ± 429.22 1.289 0.210 Ventilation support 0.042 0.838 Intubated on SIMV 10 9 SIMV and then/or NCPAP 14 12 Diagnosis 7.564 0.109 Bilateral vocal cord paralysis 7 6 Vocal cord/subglottic mass 2 3 Laryngotracheomalacia 2 1 anomaly/defect of throat/larynx 3 0 Unknown 0 2 Age on tracheotomy indications assessed (days) 15.00 (4.75, 25.75) 14.00 (4.00, 25.00) -0.384 0.701 Corrected age at discharge (weeks) 47.49 ± 4.98 43.21 ± 2.62 -2.414 0.024 Discharge weight (g) 3502.14 ± 532.88 3535.83 ± 468.49 0.282 0.780 Death in hospital 1 0 0.891 0.345 DAMA 2 8 5.441 0.020 Total NICU stay (days) 37.0 ± 12.2 15.9 ± 7.4 5.229 <0.001 Total follow-up rate (%) 88.9 30.0 24.116 <0.001 Proportion of follow-up At 3 months 13/13 5/12 7.838 <0.001 At 6 months 10/12 2/10 6.455 0.011 At 12 months 9/11 2/8 4.024 0.045 Data are shown as mean±standard deviation, median (P25, P75), or n (%).*Patients in this group met the indications of tracheotomy but parents did not sign the consent for operation.DAMA: Discharge against medical order; NCPAP: Nasal continuous positive airway pressure; NICU: Neonatal intensive care unit; SGA: Small for gestational age; SIMV: Synchronized intermittent mandatory ventilation. For infants in the tracheotomy group, the median age at surgery was 33.00 (21.50–44.25) days, and the mean weight at surgery was 3187.86 ± 552.74 g. The ventilation period before and after tracheotomy was 19.0 (10.5–34.0) days and 0.86 (0.06–3.25) days, respectively (P <0.001). The pre- and post-operative weight gain rate were 1.66 (-0.16 to 5.54) g∙kg-1∙d-1 and 4.69 (2.30–9.32) g∙kg-1∙d-1, respectively (Z = -1.977; P = 0.048). The pre-operative weight gain rate correlated negatively with the postoperative NICU stay (ρ = -0.732; P = 0.02), and the presence of pneumonia upon admission correlated positively with the total NICU stay (ρ = 0.568; P = 0.027). One patient died in the NICU on the 17th postoperative day due to severe multi-organ congenital defects. The DAMA rate of the tracheotomy group was significantly lower than that of the non-tracheotomy group (2/14 vs. 8/12; P = 0.02). The total follow-up rates in the tracheotomy vs. non-tracheotomy groups were 88.9% and 30.0%, and the follow-up rates at three months, six months, and 12 months were 100% vs. 41.7%, 83.3% vs. 20.0%, and 81.9% vs. 25.0%, respectively (all P <0.05) [Table 1]. Owing to the high rate of patients lost to follow-up, no data regarding the post-discharge mortality could be obtained for the non-tracheotomy group. Among 13 discharged tracheotomy infants, there were two infants died at the age of three months and nine months due to severe pulmonary infection and septicemia, two other infants were lost to post-discharge follow-up at six months and 12 months of age, four infants underwent de-cannulation at ages ranging from seven months to 14 months, and five infants were still wearing the tracheal cannulation (current age ranges from 15 months to 66 months). Among nine infants in tracheotomy group who were still been followed-up all had language disorders, There are four infants had growth retardation and three infants had delayed neurodevelopmental. Among the infants who had tracheotomy, two were bronchopulmonary dysplasia (BPD) preterm infants. They have been successfully de-cannulated after carrying the tracheal cannulation for 17 months and 14 months, respectively. Both have language disorders, but no problem in motor development and hearing compared to others infants at the same age. One of them has weight and height below the 10th percentile of the same population. In the study, most of the 26 patients were term infants with normal birth weights. Only 14 neonates completed tracheotomy (1.96/1000 NICU admissions), which was less than that reported by Sidman et al (7.5/1000 NICU admissions).[1] Bilateral vocal cord paralysis was the most common indication for tracheotomy. While upper airway obstruction secondary to acute infections, neuromuscular disease, ventilator-associated respiratory failure have been reported to be the commonest indications of pediatric tracheotomy abroad.[2-4] The differences in surgery indication might arise from the differences in the patient population. In our study, among the infants who met indications of tracheotomy, 46.2% could not undergo the surgery because of their parents' refusal. The most reason was parents' uncertainty of the long-term sequelae or poor outcome. Both the DAMA rate and the lost-to-follow-up rate were considerably higher among neonates in the non-tracheotomy group. We considered that a higher DAMA rate and higher lost-to-follow-up rate revealed poor parents' compliance. In China, neonates being DAMA have garnered growing attention in recent years. This might have grave impacts on infants including, negatively influence on their health and even the mortality rate of children aged <5 years. Developing strategies to improve parents' compliance with a recommended tracheotomy of their infants is not solely a medical issue; it rather is a medical–social–ethical–economic issue. The hospital-associated costs and post-discharge burden on the family are high for infants undergoing tracheotomy; moreover, these infants may face more post-discharge problems. Of the 14 infants in the tracheotomy group, five kept wearing the tracheal cannula, with the longest wear time being 66 months; four of them were de-cannulated at ages ranging from 7 months to 14 months, and all had language disorders. It has been reported that the average time for decannulation among the extremely low–birth–weight infants was 3.8 years after the tracheotomy, and speech and language disorders were noted frequently.[5] In our study, the median age at surgery was 33 days after birth. The pre-operative weight gain rate negatively correlated with the postoperative NICU stay, and the post-operative weight gain rate was significantly higher than the pre-operative rate. This indicated compensatory growth after the surgery. Our study also found that pneumonia before tracheotomy correlated positively with the total NICU stay. Among infants who required tracheotomy intervention, intubation and ventilation dependence were common. The longer the ventilation time, the higher the probability of the occurrence of ventilation-associated pneumonia. Hence, timely tracheotomy is beneficial for better outcomes. In western countries, more patients with BPD underwent tracheotomy to prolong ventilation time. Thus far, limited studies have been conducted in China involving preterm infants with BPD who underwent tracheotomy. The two BPD preterm infants in our study both finally underwent de-cannulation and carried tracheal cannulation for 17 months and 14 months respectively. Neurodevelopmental follow-up was satisfactory. Several studies reported that tracheotomy did not increase the risk of neurodevelopmental retardation[2,6] and was beneficial to the growth and development of BPD infants.[3] However, the sample size of this study is small. Exploring the impact of tracheotomy on the prognosis of BPD infants needs further multicenter clinical studies. Acknowledgments We thank the colleagues in the Department of Radiology and Ultrasound at Beijing Children's Hospital, Capital Medical University. Funding This work was supported by a grant from the Capital's Funds for Health Improvement and Research (No. 2022–2–2096). Conflicts of interest None.