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Swallowing outcomes in children after slide tracheoplasty
International Journal of Pediatric Otorhinolaryngology 108 (2018) 85–90
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International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Swallowing outcomes in children after slide tracheoplasty b,∗
a
a
T b
b
Alexandra J. Stewart , Colin R. Butler , Nagarajan Muthialu , Debbie Sell , Joseph Marchant , Richard J.D. Hewitta, Martin J. Elliotta a b
The National Service for Severe Tracheal Disease in Children, Great Ormond Street Hospital, Great Ormond Street, London, WC1N 3JH, UK Department of Speech and Language Therapy, Great Ormond Street Hospital, Great Ormond Street, London, WC1N 3JH, UK
A R T I C L E I N F O
A B S T R A C T
Keywords: Swallowing Dysphagia Children Slide tracheoplasty Tracheal stenosis Congenital
Introduction: Slide tracheoplasty is now considered gold standard treatment for long segment congenital tracheal stenosis. Outcomes are typically focused upon airway patency. Dysphagia is often reported in children undergoing cardiothoracic surgery, but not specifically after slide tracheoplasty. This study was carried out to describe the nature and prevalence of dysphagia following slide tracheoplasty for long segment congenital tracheal stenosis. Methods: Retrospective case note review was conducted on a series of patients who underwent swallow evaluation following slide tracheoplasty between 2006 and 2014. A clinical swallow assessment was carried out by a Speech and Language Therapist with videofluoroscopic evaluation of swallowing where indicated. Logistic regression assessed the impact of gender, feeding history, weight, tracheal diameter, stenting and co-morbidities on the likelihood of having post-operative dysphagia. Results: 43 out of 83 slide tracheoplasty patients underwent swallow evaluation. Dysphagia was identified in 30 (70%) of 43 patients. Videofluoroscopy was undertaken in 22 of these patients. All patients who had a videofluoroscopy presented with altered swallow physiology. Aspiration risk was confirmed in 15 patients with frank aspiration seen in 9. Pre-operative history of dysphagia was present in 9 patients. There were two cases of vocal fold palsy. The presence of a stent was the strongest predictor of post-operative dysphagia with an odds ratio of 10.6 (95% CI 1.2–92.8). Conclusions: This study documents a high prevalence of post-operative dysphagia in a pediatric population following slide tracheoplasty. In most cases there was no history suggestive of dysphagia pre-operatively. Swallowing needs to be assessed after slide tracheoplasty and longitudinal studies are required.
1. Introduction Long segment congenital tracheal stenosis (LSCTS) is a rare, lifethreatening condition, usually presenting in the first year of life, associated with the presence of complete cartilaginous rings in the trachea [1–3]. Several surgical treatments have been proposed in the past, including patch tracheoplasty [4], augmentation with costal cartilage [5] or tracheal autograft [6]. Morbidity and mortality were high and follow up short [7]. Slide tracheoplasty (STP) has now become the treatment of choice, providing lower mortality, morbidity and cost of care [1], [3]. Dysphagia has been frequently described in children undergoing thoracic surgery [8–14] with and without vocal cord paralysis [8,11–15]. There are no reports of dysphagia following STP or other surgical repair of LSCTS in the literature, despite the potential risk to both vocal fold innervation and the intimate morphologic relationship
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between the shortened trachea and the oesophagus. The identification of dysphagia in children undergoing STP is of clinical importance as secondary aspiration can occur and is a cause of recurrent pneumonia [16]. Silent aspiration, where the normal protective cough reflex is absent, is even more important to identify, so to prevent chronic lung parenchyma damage [17–19]. Our clinical observations in children undergoing swallow evaluation has suggested children have a degree of dysphagia leading to an increased aspiration risk. We describe swallowing outcomes in a consecutive series of children who underwent swallow assessment following surgical repair for congenital tracheal stenosis between 2006 and 2014. 2. Materials and methods The project was registered with the hospital's Research and
Corresponding author. E-mail address: [email protected] (A.J. Stewart).
https://doi.org/10.1016/j.ijporl.2018.02.029 Received 25 October 2017; Received in revised form 14 February 2018; Accepted 15 February 2018 Available online 21 February 2018 0165-5876/ Crown Copyright © 2018 Published by Elsevier B.V. All rights reserved.
International Journal of Pediatric Otorhinolaryngology 108 (2018) 85–90
A.J. Stewart et al.
Development department. Institutional review determined that ethical approval was not required due to the retrospective case note review methodology. Case note review for 43 consecutively referred children undergoing STP and Speech and Language Therapy swallow assessment between June 2006 and May 2014 was conducted. Forty patients underwent STP over the same period but were not referred to Speech and Language Therapy. Seven of these patients died following surgery. We present findings from the swallow assessment cohort only (n = 43).
Table 2 Co-morbidities.
2.1. Surgical procedure
Cardio-vascular anomalies
n
Non-cardiovascular anomalies
n
Tetralogy of Fallot Left pulmonary artery sling Atrial septal defect Ventricular septal defect Hypoplastic aortic arch Subaortic stenosis Right aortic arch Total anomalous pulmonary venous drainage Absent left pulmonary artery Vascular ring
4 24 4 5 1 1 1 1
Trisomy 21 Ex-prematurity Hydrocephalus Duodenal atresia VACTERL CHARGE association Imperforate anus Single lung
2 2 1 1 2 1 3 2
Slide tracheoplasty was performed as previously described [20]. Briefly all operations were via a median sternotomy with mobilization of the trachea and a slide tracheoplasty performed over the stenosed segment. Where possible, associated cardiopulmonary anomalies were corrected at the same time. All operations were performed on cardiopulmonary bypass.
once the Radiologist and SLT agreed that the child had taken sufficient volume for the goals of study to be achieved [22].
2.2. Clinical swallow assessment
2.4. Statistical analysis
Clinical swallow assessments were carried out by a specialist pediatric Speech and Language Therapist and involved the following:
Data were analysed using SPSS version 21.0 (IBM-SPSS, Inc, Armonk, NY). Predictive modelling was conducted using logistic regression. Gender, pre-operative report (history) of dysphagia, weight, tracheal diameter, stenting and co-morbidities (extracardiac, intracardiac and non cardiac) were the independent variables analysed. Due to the small numbers identified with vocal fold palsy this was not included in the logistic regression model.
(1) Feeding history: Parental information was gathered regarding feeding method, length of mealtimes/feeds, growth, coughing, stridor or other changes to respiration during or after feeding and previous chest infections. (2) Dysphonia screening assessment: An auditory perceptual assessment of voice and/or cry was carried out to screen for dysphonia. All children underwent routine endoscopic airway evaluation (bronchoscopy ± microlaryngobronchoscopy). Further dynamic vocal fold evaluation using flexible nasendoscopy or ultrasound was conducted if dysphonia was identified. (3) Swallow assessment: This involved observation of secretion management and spontaneous saliva swallows. If secretion management was adequate, observational swallow assessment coupled with cervical auscultation on age appropriate food/fluid was carried out. Table 1 lists the clinical indicators used to determine the presence of aspiration risk related to dysphagia [21].
1 1
3. Results Forty three patients (23 male), with a median age of 6 months (range 0.16–35 months) underwent clinical swallow assessment following STP. Comorbidities are outlined in Table 2. Eighteen patients had an associated intracardiac pathology, 11 of which were repaired at the time of STP. Twenty eight patients had an extracardiac pathology, 25 of which were repaired at the time of STP. Seven patients had no associated cardiac pathology. 3.1. Clinical swallow assessment
2.3. Videofluoroscopy swallow study (VFSS)
3.1.1. Pre-operative feeding history Only one patient was non-orally fed pre-operatively. A history of consistent coughing during oral feeds was reported in 9 patients (21%), one of whom was admitted to pediatric intensive care with aspiration related respiratory infection pre-STP. Eight patients (19%) had a history of vomiting with feeding. One underwent gastrostomy insertion and Nissen's fundoplication 2 years prior to STP but was fully orally fed immediately prior to STP.
Videofluoroscopy was carried out when indicators of aspiration risk were identified on clinical swallow assessment. Where there were overt signs of aspiration, a VFSS was contraindicated. Videofluoroscopy was carried out jointly by a Pediatric Radiologist and a Speech and Language Therapist. A fluoroscopy unit (Siemens Polystar digital unitSiemens AG, Erlangen, Germany) connected to a high definition medical quality DVD recorder was used. Studies were undertaken according to the hospital's VFSS protocol [22]. Lateral images were obtained at 15 pulses/second. Each patient was given food and/or liquid boluses mixed with barium sulphate contrast (EZPaque 100% wpv). Where aspiration or penetration were seen on thin fluids, thickened fluids were assessed. Likewise, where aspiration risk was identified on thickened fluids, puree consistency solids were assessed if age appropriate. Fatigue testing was included in all studies. The VFSS was discontinued
3.1.2. Clinical swallow assessment A clinical swallow assessment was carried out a median of 13 days post-operatively (range 2–265). Variability existed due to length of post-operative intubation and timing of referral to Speech and Language Therapy. Dysphagia was identified on clinical swallow assessment in 30 of 43 patients (70%). Clinical indicators of swallow dysfunction were: cough (25 patients, 58%), wet breath sounds (18 patients, 42%), multiple swallows (4 patients, 9%) and general poor swallow coordination (4 patients, 9%). One patient was identified with disorganised feeding secondary to gastro-esophageal reflux but did not present with clinical indicators of pharyngeal stage dysphagia.
Table 1 Clinical indicators of aspiration risk. Coughing or choking during swallowing Inability to handle own oral secretions Noisy, “wet” upper airway sounds after individual swallows or increasing noisiness over course of feeding Multiple swallows to clear a single bolus Apnea during swallowing History of frequent upper-respiratory infections or pneumonias
3.1.3. Perceptual voice assessment Post-operative dysphonia was evident in five patients. This was evaluated endoscopically in 4 patients and via laryngeal ultrasound 86
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Table 3 Results for VFSS investigations. Patient
History of dysphagia
Days post-surgery to VFSS
VFSS description Delayed swallow trigger
Epiglottic undercoating
Laryngeal penetration
Aspiration – + (silent) thin and thickened fluid – – + (silent) thin fluid + (silent) thin fluid
1. 2.
– –
29 98
+ +
+ +
+thin fluids +
3. 4. 5. 6.
– – – –
30 57 44 223
– + + +
– – + +
7.
–
47
+
+ thin fluid + thin fluid + + thickened fluid smooth puree +
8. 9. 10. 11. 12.
– – + – –
31 72 21 26 15
+ + + + +
13. 14. 15. 16. 17. 18. 19. 20. 21. 22.
+ – – + – + – – + –
64 266 12 449 1241 196 57 21 8 33
+ + + – + + + + + +
+ – + + thin fluid + thickened fluid smooth puree + + + – + – – – + +
+ + thin fluid – + – + thin fluid
+ thin fluid – – + (silent) thin fluid – –
+ + + – + thickened – – – + thin +
+ + + – – – – – – +
(silent) thin and thickened fluid thin thin
(silent) thin
Legend: + present, - absent.
post-operatively (range 8–1241days) (Table 3). Significant delay between clinical assessment and VFSS assessment occurred in 1 patient (patient no. 16) due to poor VFSS compliance. Two patients identified with dysphagia on clinical assessment had clear indicators of aspiration such that VFSS was contraindicated and were then referred to the local hospital for ongoing assessment and management. Six patients with dysphagia evident on clinical assessment were discharged to their referring hospital before VFSS could be conducted. Altered swallow physiology was evident in 20 out of 22 patients. The predominant feature of the dysphagia was a delayed swallow trigger, evident in 20 patients. Aspiration was seen in 9 (21%) patients. Silent aspiration (without a cough response) occurred in 6 of these patients. Aspiration risk as a result of laryngeal penetration was evident in a further 4 (9%) patients. Epiglottic undercoating (food or fluid passing underneath the epiglottis during swallowing but not entering the larynx) was seen in a further 3 patients. Of the 9 patients with a history suggestive of aspiration risk: 3 had confirmed aspiration on post-operative VFSS, 2 had no risk on VFSS, 2 had no indicators of swallow dysfunction on clinical assessment so did not undergo VFSS. The remaining 2 underwent VFSS at the referring hospital.
Table 4 Logistic regression predicting likelihood of post-operative dysphagia.
Gender History of aspiration Weight (kg) Tracheal diameter (min, mm) Stenting Extracardiac comorbidity Intracardiac comorbidity Non cardiac comorbidity Constant
Wald
P
Odds ratio
95% CI Lower
Upper
6.725 .979 1.844 1.704 4.607 2.089 1.747 .396 .704
.010 .322 .174 .192 .032 .148 .186 .529 .401
.044 .308 1.369 2.873 10.678 .219 .276 .578 .140
.004 .030 .870 .589 1.228 .028 .041 .105
.466 3.176 2.153 14.008 92.816 1.719 1.863 3.191
Table 5 Suggested minimal dataset for future longitudinal studies. Age at time of surgery Weight Gender Comorbidities (cardiac and non-cardiac) Length of stenosis Tracheal diameter Pre-operative history of dysphagia Pre-operative swallow assessment ± VFSS/FEES Number of days intubated Vocal cord palsy Pre-operative tracheo/bronchomalacia Post-operative tracheo/bronchomalacia Stent type and location Post-operative swallow assessment ± VFSS/FEES Neurological status
3.3. Feeding management and one year outcome Feeding management and one year follow up data is presented for the VFSS cohort. Following initial VFSS, oral feeding with normal fluids was commenced in 8/22 patients and with thickened fluids in 10/22 patients. Supplementary naso-gastric tube (NGT) feeding was required in 15 patients. NGT feeding for all fluids was commenced in 4 patients. All patients commenced on age appropriate foods. One year follow-up data (from the time of initial VFSS) was available for 21 of the 22 patients (95%). NGT feeding had ceased for all but two patients. A further 2 patients required gastrostomy insertion to supplement oral feeding. Five of the 10 children requiring thickened fluids post-operatively had progressed to thin fluids (one lost to follow up). Three of the 4
scanning for 1 patient. Two patients were diagnosed with left vocal fold palsy and another with vocal cord nodules. 3.2. Videofluoroscopy assessment VFSS was carried out in 22 patients (51%) at a median of 45.5 days 87
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Fig. 1. Swallow evaluation pathway in long segment congenital tracheal stenosis and slide tracheoplasty. * Or earlier if clinically indicated.
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children requiring NGT for all fluids had progressed onto full volumes of thickened fluids, whilst one had progressed onto full volumes of thin fluids.
subglottic pressure contributes to the dysphagia and aspiration risk in children with tracheomalacia. This is partly supported by the regression analysis, indicating a significant association between the need for stenting and the presence of dysphagia. The stent itself may have a direct physical impact on the oesophagus resulting in secondary pharyngeal dysfunction. However, we hypothesize that the need for stenting is a marker of severe tracheabronchomalacia and it is the tracheomalacia that effects swallowing. It is also possible that the impact of surgery itself leads to significant changes in airway pressures; leading to disruption of afferent pathways which contributes to post-operative dysphagia and aspiration in our population. Another consideration is iatrogenic injury to the recurrent laryngeal nerve. Vocal fold palsy is a well-reported complication of cardiothoracic surgery [8–15] and dysphonia is the most common presenting symptom [35]; apparent on auditory perceptual assessment [36]. Vocal cord palsy can, but does not always, cause dysphagia [8–15]. We have previously reported a low incidence of vocal fold palsy (3%), all of which resolved within 6 months [3]. This current study cohort had 5 patients with dysphonia, 2 with left vocal cord palsies confirmed endoscopically. It seems unlikely that recurrent laryngeal nerve damage is the sole cause of dysphagia in our population. However, in view of the proximity of the recurrent laryngeal nerve to the surgical site, and the imperfect relationship between dysphonia and cord palsy, we would advocate routine assessment of vocal fold function in all children, either endoscopically or via ultrasound. The direct impact of the surgical procedure on swallow physiology cannot be ignored. Tethering of the larynx and swallow dysfunction following surgery has been previously described [37]. Laryngeal mobilization is less likely in surgery for LSCTS, however tracheal mobilization and subsequent tethering may limit normal laryngeal elevation and effect swallow physiology. Contradicting this are studies indicating laryngeal tethering to have no effect in tracheostomized adults [38] and the fact our children appeared to have normal laryngeal excursion on VFSS. It is more likely that during surgery, tracheal mobilization leads to disruption of both autonomic esophageal afferent and efferent pathways, resulting in secondary effects on pharyngeal function. This phenomenon, whilst difficult to confirm, is the subject of ongoing investigation.
3.4. Regression analysis The regression model, consisting of 6 independent variables was not statistically significant (χ2 = 14.7, p = 0.06). However, 2 variables, gender (p = 0.01) and the presence of a stent (p = 0.032), were significant contributors to the model (Table 4). The presence of a stent was the strongest predictor of post-operative dysphagia with an odds ratio of 10.6 (95% CI 1.2–92.8). Male gender reduced the likelihood of postoperative dysphagia (OR 0.044, CI 0.004–0.466). 4. Discussion 4.1. Discussion of results We present swallowing outcomes on a cohort of children undergoing slide tracheoplasty and find that swallowing difficulties postsurgery were prevalent with 30 out of 43 identified as having dysphagia at bedside assessment. In this group 50% (n = 15) were at risk of aspiration either identified clinically (n = 2) or by VFSS (n = 13). Radiological evaluation in the latter group crucially identified 20% (n = 6) to have silent aspiration. The regression model did not generate a statistically significant predictive model for post-operative dysphagia, however, gender and the need for stenting were considered significant variables. The study highlights the relatively high prevalence of swallowing anomalies in children undergoing slide tracheoplasty which is somewhat higher than postoperative dysphagia outcomes in children undergoing open cardiac procedures [23]. As such clinicians involved in the care of these patients should be alert to this problem postoperatively. Identification of dysphagia and the risk of aspiration is of importance since, in the short-term, it can potentially impact the repair of the airway, and in the long-term, can lead to irreversible damage of lung parenchyma [17]. Swallow evaluation includes a detailed preoperative history; however, we found this to be a poor identifier of post-operative swallowing anomalies. Clinical swallow assessment at the bedside is considered more effective at identifying dysphagia and aspiration risk, is readily repeatable and naturalistic, however lacks full diagnostic accuracy [24]. It is typically used as a screening tool before more invasive investigations are performed [25,26]. Instrumental assessment using VFSS or fibreoptic endoscopic evaluation of swallowing (FEES) are both time limited procedures but add information not identifiable at the bedside including: bolus control, pharyngeal bolus clearance and timing of the swallow trigger, and can potentially identify the mechanism for dysphagia and/or aspiration. It is interesting to note that in our cohort, the prevailing VFSS finding was a delayed swallow trigger; only initiated when the bolus reached the cricopharyngeus. In these cases, the pyriform sinuses were filled prior to swallow initiation leading to laryngeal penetration. We advocate a multi-modal approach to feeding evaluation with clinical decision making taking account of all available information. In normal swallowing, an exhale-swallow-exhale pattern is seen [27]. Disruption to this pattern increases the risk of aspiration and is observed in children with both acute and chronic lung disease [27–32]. Evaluation of the coordination of swallow and respiration was outside of the scope of this review but would be important to consider in future studies. Equally important to safe swallowing is regulated positive airway pressure at the subglottis [28]. Change to this is associated with altered swallow function [28], [29]. Altered subglottic pressure is also seen in children with tracheomalacia, who in turn have been shown to have an increased aspiration risk [33,34]. We hypothesize that altered
4.2. Limitations of this study A major limitation is the retrospective nature of this study with reliance on recorded information at the time of surgery, and that our cohort represents a referred population. No information regarding swallow function for the non-referred patients was available. However, even if we assume there are no dysphagic patients in the non-referred group, prevalence of post-operative dysphagia remains high 30/83 (36%). Another significant limitation is a lack of preoperative investigation, meaning conclusions regarding cause and effect must be carefully considered. Dysphagia rates in our cohort may not necessary represent new onset findings, since children could have had pre-existing dysphagia; studies have shown up to 1/3 of children with congenital heart disease to have swallow abnormalities [15] and the possibility of a neurogenic dysphagia in this complex group has not been investigated. Diagnosis of LSCTS frequently occurs in an acute situation necessitating intubation and ventilation, precluding pre-operative swallow assessment. However, we strongly advocate preoperative assessment where possible. It is possible that the prevalence of dysphagia is under-reported in our cohort as not all patients underwent instrumental swallow assessment. In our unit we use clinical swallow assessment to provoke VFSS assessment to limit unnecessary radiation exposure. The diagnostic accuracy of a clinical swallow assessment is questioned in the literature, although for assessment of thin fluids has been found to have high 89
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sensitivity (92–100%) [26]. It is possible that cases of dysphagia were therefore missed. Use of FEES has not been routinely used at our center, predominantly due to issues achieving good compliance with this age group. Where good compliance can be achieved FEES may provide superior instrumental assessment allowing for pre- and post-surgical instrumental swallow assessment for all STP patients. Finally, the relatively small number of participants is acknowledged but is a consequence of the rarity of the condition and procedure, even though we are a national referral center.
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4.3. Conclusions and future plans Given the high prevalence of dysphagia we would advocate swallow evaluation including appropriate instrumental evaluation, when possible, upon a diagnosis of long segment tracheal stenosis. Future studies should be prospective, with extended follow up to enable investigation of the timeframe for resolution of dysphagia. A suggested dataset for such a prospective study is presented in Table 5. We have proposed a swallow evaluation care pathway to guide future management of such children (Fig. 1). Further exploration of the underlying pathophysiology, and the potential role of subglottic pressure changes, present exciting challenges for future studies. Funding This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors. Conflicts of interest None. Acknowledgements The authors would like to thank all clinicians and patients involved in this study. In particular thanks to M Ryan (Speech and Language Therapist) for her involvement in the analysis of videofluoroscopies. Thanks also to R Grant for his advice and support with statistical analysis. References [1] M. Elliott, et al., Slide tracheoplasty, Curr. Opin. Otolaryngol. Head Neck Surg. 16 (1) (2008) 75–82. [2] X. Li, et al., Management of symptomatic congenital tracheal stenosis in neonates and infants by slide tracheoplasty: a 7-year single institution experience, Eur. J. Cardio. Thorac. Surg. 38 (5) (2010) 609–614. [3] C.R. Butler, et al., Outcomes of slide tracheoplasty in 101 children: a 17-year singlecenter experience, J. Thorac. Cardiovasc. Surg. 147 (6) (2014) 1783–1789. [4] N. Fanous, et al., Anterior pericardial tracheoplasty for long-segment tracheal stenosis: long-term outcomes, J. Thorac. Cardiovasc. Surg. 139 (1) (2010) 18–23 discussion 23-5. [5] K. Kimura, et al., Tracheoplasty for congenital stenosis of the entire trachea, J. Pediatr. Surg. 17 (6) (1982) 869–871. [6] M. Elliott, et al., The management of congenital tracheal stenosis, Int. J. Pediatr. Otorhinolaryngol. 67 (Suppl 1) (2003) S183–S192. [7] C.L. Backer, et al., Tracheal surgery in children: an 18-year review of four techniques, Eur. J. Cardio. Thorac. Surg. 19 (6) (2001) 777–784. [8] W.A. Clement, et al., Unilateral vocal cord paralysis following patent ductus arteriosus ligation in extremely low-birth-weight infants, Arch. Otolaryngol. Head Neck Surg. 134 (1) (2008) 28–33.
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International Journal of Pediatric Otorhinolaryngology journal homepage: www.elsevier.com/locate/ijporl
Swallowing outcomes in children after slide tracheoplasty b,∗
a
a
T b
b
Alexandra J. Stewart , Colin R. Butler , Nagarajan Muthialu , Debbie Sell , Joseph Marchant , Richard J.D. Hewitta, Martin J. Elliotta a b
The National Service for Severe Tracheal Disease in Children, Great Ormond Street Hospital, Great Ormond Street, London, WC1N 3JH, UK Department of Speech and Language Therapy, Great Ormond Street Hospital, Great Ormond Street, London, WC1N 3JH, UK
A R T I C L E I N F O
A B S T R A C T
Keywords: Swallowing Dysphagia Children Slide tracheoplasty Tracheal stenosis Congenital
Introduction: Slide tracheoplasty is now considered gold standard treatment for long segment congenital tracheal stenosis. Outcomes are typically focused upon airway patency. Dysphagia is often reported in children undergoing cardiothoracic surgery, but not specifically after slide tracheoplasty. This study was carried out to describe the nature and prevalence of dysphagia following slide tracheoplasty for long segment congenital tracheal stenosis. Methods: Retrospective case note review was conducted on a series of patients who underwent swallow evaluation following slide tracheoplasty between 2006 and 2014. A clinical swallow assessment was carried out by a Speech and Language Therapist with videofluoroscopic evaluation of swallowing where indicated. Logistic regression assessed the impact of gender, feeding history, weight, tracheal diameter, stenting and co-morbidities on the likelihood of having post-operative dysphagia. Results: 43 out of 83 slide tracheoplasty patients underwent swallow evaluation. Dysphagia was identified in 30 (70%) of 43 patients. Videofluoroscopy was undertaken in 22 of these patients. All patients who had a videofluoroscopy presented with altered swallow physiology. Aspiration risk was confirmed in 15 patients with frank aspiration seen in 9. Pre-operative history of dysphagia was present in 9 patients. There were two cases of vocal fold palsy. The presence of a stent was the strongest predictor of post-operative dysphagia with an odds ratio of 10.6 (95% CI 1.2–92.8). Conclusions: This study documents a high prevalence of post-operative dysphagia in a pediatric population following slide tracheoplasty. In most cases there was no history suggestive of dysphagia pre-operatively. Swallowing needs to be assessed after slide tracheoplasty and longitudinal studies are required.
1. Introduction Long segment congenital tracheal stenosis (LSCTS) is a rare, lifethreatening condition, usually presenting in the first year of life, associated with the presence of complete cartilaginous rings in the trachea [1–3]. Several surgical treatments have been proposed in the past, including patch tracheoplasty [4], augmentation with costal cartilage [5] or tracheal autograft [6]. Morbidity and mortality were high and follow up short [7]. Slide tracheoplasty (STP) has now become the treatment of choice, providing lower mortality, morbidity and cost of care [1], [3]. Dysphagia has been frequently described in children undergoing thoracic surgery [8–14] with and without vocal cord paralysis [8,11–15]. There are no reports of dysphagia following STP or other surgical repair of LSCTS in the literature, despite the potential risk to both vocal fold innervation and the intimate morphologic relationship
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between the shortened trachea and the oesophagus. The identification of dysphagia in children undergoing STP is of clinical importance as secondary aspiration can occur and is a cause of recurrent pneumonia [16]. Silent aspiration, where the normal protective cough reflex is absent, is even more important to identify, so to prevent chronic lung parenchyma damage [17–19]. Our clinical observations in children undergoing swallow evaluation has suggested children have a degree of dysphagia leading to an increased aspiration risk. We describe swallowing outcomes in a consecutive series of children who underwent swallow assessment following surgical repair for congenital tracheal stenosis between 2006 and 2014. 2. Materials and methods The project was registered with the hospital's Research and
Corresponding author. E-mail address: [email protected] (A.J. Stewart).
https://doi.org/10.1016/j.ijporl.2018.02.029 Received 25 October 2017; Received in revised form 14 February 2018; Accepted 15 February 2018 Available online 21 February 2018 0165-5876/ Crown Copyright © 2018 Published by Elsevier B.V. All rights reserved.
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Development department. Institutional review determined that ethical approval was not required due to the retrospective case note review methodology. Case note review for 43 consecutively referred children undergoing STP and Speech and Language Therapy swallow assessment between June 2006 and May 2014 was conducted. Forty patients underwent STP over the same period but were not referred to Speech and Language Therapy. Seven of these patients died following surgery. We present findings from the swallow assessment cohort only (n = 43).
Table 2 Co-morbidities.
2.1. Surgical procedure
Cardio-vascular anomalies
n
Non-cardiovascular anomalies
n
Tetralogy of Fallot Left pulmonary artery sling Atrial septal defect Ventricular septal defect Hypoplastic aortic arch Subaortic stenosis Right aortic arch Total anomalous pulmonary venous drainage Absent left pulmonary artery Vascular ring
4 24 4 5 1 1 1 1
Trisomy 21 Ex-prematurity Hydrocephalus Duodenal atresia VACTERL CHARGE association Imperforate anus Single lung
2 2 1 1 2 1 3 2
Slide tracheoplasty was performed as previously described [20]. Briefly all operations were via a median sternotomy with mobilization of the trachea and a slide tracheoplasty performed over the stenosed segment. Where possible, associated cardiopulmonary anomalies were corrected at the same time. All operations were performed on cardiopulmonary bypass.
once the Radiologist and SLT agreed that the child had taken sufficient volume for the goals of study to be achieved [22].
2.2. Clinical swallow assessment
2.4. Statistical analysis
Clinical swallow assessments were carried out by a specialist pediatric Speech and Language Therapist and involved the following:
Data were analysed using SPSS version 21.0 (IBM-SPSS, Inc, Armonk, NY). Predictive modelling was conducted using logistic regression. Gender, pre-operative report (history) of dysphagia, weight, tracheal diameter, stenting and co-morbidities (extracardiac, intracardiac and non cardiac) were the independent variables analysed. Due to the small numbers identified with vocal fold palsy this was not included in the logistic regression model.
(1) Feeding history: Parental information was gathered regarding feeding method, length of mealtimes/feeds, growth, coughing, stridor or other changes to respiration during or after feeding and previous chest infections. (2) Dysphonia screening assessment: An auditory perceptual assessment of voice and/or cry was carried out to screen for dysphonia. All children underwent routine endoscopic airway evaluation (bronchoscopy ± microlaryngobronchoscopy). Further dynamic vocal fold evaluation using flexible nasendoscopy or ultrasound was conducted if dysphonia was identified. (3) Swallow assessment: This involved observation of secretion management and spontaneous saliva swallows. If secretion management was adequate, observational swallow assessment coupled with cervical auscultation on age appropriate food/fluid was carried out. Table 1 lists the clinical indicators used to determine the presence of aspiration risk related to dysphagia [21].
1 1
3. Results Forty three patients (23 male), with a median age of 6 months (range 0.16–35 months) underwent clinical swallow assessment following STP. Comorbidities are outlined in Table 2. Eighteen patients had an associated intracardiac pathology, 11 of which were repaired at the time of STP. Twenty eight patients had an extracardiac pathology, 25 of which were repaired at the time of STP. Seven patients had no associated cardiac pathology. 3.1. Clinical swallow assessment
2.3. Videofluoroscopy swallow study (VFSS)
3.1.1. Pre-operative feeding history Only one patient was non-orally fed pre-operatively. A history of consistent coughing during oral feeds was reported in 9 patients (21%), one of whom was admitted to pediatric intensive care with aspiration related respiratory infection pre-STP. Eight patients (19%) had a history of vomiting with feeding. One underwent gastrostomy insertion and Nissen's fundoplication 2 years prior to STP but was fully orally fed immediately prior to STP.
Videofluoroscopy was carried out when indicators of aspiration risk were identified on clinical swallow assessment. Where there were overt signs of aspiration, a VFSS was contraindicated. Videofluoroscopy was carried out jointly by a Pediatric Radiologist and a Speech and Language Therapist. A fluoroscopy unit (Siemens Polystar digital unitSiemens AG, Erlangen, Germany) connected to a high definition medical quality DVD recorder was used. Studies were undertaken according to the hospital's VFSS protocol [22]. Lateral images were obtained at 15 pulses/second. Each patient was given food and/or liquid boluses mixed with barium sulphate contrast (EZPaque 100% wpv). Where aspiration or penetration were seen on thin fluids, thickened fluids were assessed. Likewise, where aspiration risk was identified on thickened fluids, puree consistency solids were assessed if age appropriate. Fatigue testing was included in all studies. The VFSS was discontinued
3.1.2. Clinical swallow assessment A clinical swallow assessment was carried out a median of 13 days post-operatively (range 2–265). Variability existed due to length of post-operative intubation and timing of referral to Speech and Language Therapy. Dysphagia was identified on clinical swallow assessment in 30 of 43 patients (70%). Clinical indicators of swallow dysfunction were: cough (25 patients, 58%), wet breath sounds (18 patients, 42%), multiple swallows (4 patients, 9%) and general poor swallow coordination (4 patients, 9%). One patient was identified with disorganised feeding secondary to gastro-esophageal reflux but did not present with clinical indicators of pharyngeal stage dysphagia.
Table 1 Clinical indicators of aspiration risk. Coughing or choking during swallowing Inability to handle own oral secretions Noisy, “wet” upper airway sounds after individual swallows or increasing noisiness over course of feeding Multiple swallows to clear a single bolus Apnea during swallowing History of frequent upper-respiratory infections or pneumonias
3.1.3. Perceptual voice assessment Post-operative dysphonia was evident in five patients. This was evaluated endoscopically in 4 patients and via laryngeal ultrasound 86
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Table 3 Results for VFSS investigations. Patient
History of dysphagia
Days post-surgery to VFSS
VFSS description Delayed swallow trigger
Epiglottic undercoating
Laryngeal penetration
Aspiration – + (silent) thin and thickened fluid – – + (silent) thin fluid + (silent) thin fluid
1. 2.
– –
29 98
+ +
+ +
+thin fluids +
3. 4. 5. 6.
– – – –
30 57 44 223
– + + +
– – + +
7.
–
47
+
+ thin fluid + thin fluid + + thickened fluid smooth puree +
8. 9. 10. 11. 12.
– – + – –
31 72 21 26 15
+ + + + +
13. 14. 15. 16. 17. 18. 19. 20. 21. 22.
+ – – + – + – – + –
64 266 12 449 1241 196 57 21 8 33
+ + + – + + + + + +
+ – + + thin fluid + thickened fluid smooth puree + + + – + – – – + +
+ + thin fluid – + – + thin fluid
+ thin fluid – – + (silent) thin fluid – –
+ + + – + thickened – – – + thin +
+ + + – – – – – – +
(silent) thin and thickened fluid thin thin
(silent) thin
Legend: + present, - absent.
post-operatively (range 8–1241days) (Table 3). Significant delay between clinical assessment and VFSS assessment occurred in 1 patient (patient no. 16) due to poor VFSS compliance. Two patients identified with dysphagia on clinical assessment had clear indicators of aspiration such that VFSS was contraindicated and were then referred to the local hospital for ongoing assessment and management. Six patients with dysphagia evident on clinical assessment were discharged to their referring hospital before VFSS could be conducted. Altered swallow physiology was evident in 20 out of 22 patients. The predominant feature of the dysphagia was a delayed swallow trigger, evident in 20 patients. Aspiration was seen in 9 (21%) patients. Silent aspiration (without a cough response) occurred in 6 of these patients. Aspiration risk as a result of laryngeal penetration was evident in a further 4 (9%) patients. Epiglottic undercoating (food or fluid passing underneath the epiglottis during swallowing but not entering the larynx) was seen in a further 3 patients. Of the 9 patients with a history suggestive of aspiration risk: 3 had confirmed aspiration on post-operative VFSS, 2 had no risk on VFSS, 2 had no indicators of swallow dysfunction on clinical assessment so did not undergo VFSS. The remaining 2 underwent VFSS at the referring hospital.
Table 4 Logistic regression predicting likelihood of post-operative dysphagia.
Gender History of aspiration Weight (kg) Tracheal diameter (min, mm) Stenting Extracardiac comorbidity Intracardiac comorbidity Non cardiac comorbidity Constant
Wald
P
Odds ratio
95% CI Lower
Upper
6.725 .979 1.844 1.704 4.607 2.089 1.747 .396 .704
.010 .322 .174 .192 .032 .148 .186 .529 .401
.044 .308 1.369 2.873 10.678 .219 .276 .578 .140
.004 .030 .870 .589 1.228 .028 .041 .105
.466 3.176 2.153 14.008 92.816 1.719 1.863 3.191
Table 5 Suggested minimal dataset for future longitudinal studies. Age at time of surgery Weight Gender Comorbidities (cardiac and non-cardiac) Length of stenosis Tracheal diameter Pre-operative history of dysphagia Pre-operative swallow assessment ± VFSS/FEES Number of days intubated Vocal cord palsy Pre-operative tracheo/bronchomalacia Post-operative tracheo/bronchomalacia Stent type and location Post-operative swallow assessment ± VFSS/FEES Neurological status
3.3. Feeding management and one year outcome Feeding management and one year follow up data is presented for the VFSS cohort. Following initial VFSS, oral feeding with normal fluids was commenced in 8/22 patients and with thickened fluids in 10/22 patients. Supplementary naso-gastric tube (NGT) feeding was required in 15 patients. NGT feeding for all fluids was commenced in 4 patients. All patients commenced on age appropriate foods. One year follow-up data (from the time of initial VFSS) was available for 21 of the 22 patients (95%). NGT feeding had ceased for all but two patients. A further 2 patients required gastrostomy insertion to supplement oral feeding. Five of the 10 children requiring thickened fluids post-operatively had progressed to thin fluids (one lost to follow up). Three of the 4
scanning for 1 patient. Two patients were diagnosed with left vocal fold palsy and another with vocal cord nodules. 3.2. Videofluoroscopy assessment VFSS was carried out in 22 patients (51%) at a median of 45.5 days 87
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Fig. 1. Swallow evaluation pathway in long segment congenital tracheal stenosis and slide tracheoplasty. * Or earlier if clinically indicated.
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children requiring NGT for all fluids had progressed onto full volumes of thickened fluids, whilst one had progressed onto full volumes of thin fluids.
subglottic pressure contributes to the dysphagia and aspiration risk in children with tracheomalacia. This is partly supported by the regression analysis, indicating a significant association between the need for stenting and the presence of dysphagia. The stent itself may have a direct physical impact on the oesophagus resulting in secondary pharyngeal dysfunction. However, we hypothesize that the need for stenting is a marker of severe tracheabronchomalacia and it is the tracheomalacia that effects swallowing. It is also possible that the impact of surgery itself leads to significant changes in airway pressures; leading to disruption of afferent pathways which contributes to post-operative dysphagia and aspiration in our population. Another consideration is iatrogenic injury to the recurrent laryngeal nerve. Vocal fold palsy is a well-reported complication of cardiothoracic surgery [8–15] and dysphonia is the most common presenting symptom [35]; apparent on auditory perceptual assessment [36]. Vocal cord palsy can, but does not always, cause dysphagia [8–15]. We have previously reported a low incidence of vocal fold palsy (3%), all of which resolved within 6 months [3]. This current study cohort had 5 patients with dysphonia, 2 with left vocal cord palsies confirmed endoscopically. It seems unlikely that recurrent laryngeal nerve damage is the sole cause of dysphagia in our population. However, in view of the proximity of the recurrent laryngeal nerve to the surgical site, and the imperfect relationship between dysphonia and cord palsy, we would advocate routine assessment of vocal fold function in all children, either endoscopically or via ultrasound. The direct impact of the surgical procedure on swallow physiology cannot be ignored. Tethering of the larynx and swallow dysfunction following surgery has been previously described [37]. Laryngeal mobilization is less likely in surgery for LSCTS, however tracheal mobilization and subsequent tethering may limit normal laryngeal elevation and effect swallow physiology. Contradicting this are studies indicating laryngeal tethering to have no effect in tracheostomized adults [38] and the fact our children appeared to have normal laryngeal excursion on VFSS. It is more likely that during surgery, tracheal mobilization leads to disruption of both autonomic esophageal afferent and efferent pathways, resulting in secondary effects on pharyngeal function. This phenomenon, whilst difficult to confirm, is the subject of ongoing investigation.
3.4. Regression analysis The regression model, consisting of 6 independent variables was not statistically significant (χ2 = 14.7, p = 0.06). However, 2 variables, gender (p = 0.01) and the presence of a stent (p = 0.032), were significant contributors to the model (Table 4). The presence of a stent was the strongest predictor of post-operative dysphagia with an odds ratio of 10.6 (95% CI 1.2–92.8). Male gender reduced the likelihood of postoperative dysphagia (OR 0.044, CI 0.004–0.466). 4. Discussion 4.1. Discussion of results We present swallowing outcomes on a cohort of children undergoing slide tracheoplasty and find that swallowing difficulties postsurgery were prevalent with 30 out of 43 identified as having dysphagia at bedside assessment. In this group 50% (n = 15) were at risk of aspiration either identified clinically (n = 2) or by VFSS (n = 13). Radiological evaluation in the latter group crucially identified 20% (n = 6) to have silent aspiration. The regression model did not generate a statistically significant predictive model for post-operative dysphagia, however, gender and the need for stenting were considered significant variables. The study highlights the relatively high prevalence of swallowing anomalies in children undergoing slide tracheoplasty which is somewhat higher than postoperative dysphagia outcomes in children undergoing open cardiac procedures [23]. As such clinicians involved in the care of these patients should be alert to this problem postoperatively. Identification of dysphagia and the risk of aspiration is of importance since, in the short-term, it can potentially impact the repair of the airway, and in the long-term, can lead to irreversible damage of lung parenchyma [17]. Swallow evaluation includes a detailed preoperative history; however, we found this to be a poor identifier of post-operative swallowing anomalies. Clinical swallow assessment at the bedside is considered more effective at identifying dysphagia and aspiration risk, is readily repeatable and naturalistic, however lacks full diagnostic accuracy [24]. It is typically used as a screening tool before more invasive investigations are performed [25,26]. Instrumental assessment using VFSS or fibreoptic endoscopic evaluation of swallowing (FEES) are both time limited procedures but add information not identifiable at the bedside including: bolus control, pharyngeal bolus clearance and timing of the swallow trigger, and can potentially identify the mechanism for dysphagia and/or aspiration. It is interesting to note that in our cohort, the prevailing VFSS finding was a delayed swallow trigger; only initiated when the bolus reached the cricopharyngeus. In these cases, the pyriform sinuses were filled prior to swallow initiation leading to laryngeal penetration. We advocate a multi-modal approach to feeding evaluation with clinical decision making taking account of all available information. In normal swallowing, an exhale-swallow-exhale pattern is seen [27]. Disruption to this pattern increases the risk of aspiration and is observed in children with both acute and chronic lung disease [27–32]. Evaluation of the coordination of swallow and respiration was outside of the scope of this review but would be important to consider in future studies. Equally important to safe swallowing is regulated positive airway pressure at the subglottis [28]. Change to this is associated with altered swallow function [28], [29]. Altered subglottic pressure is also seen in children with tracheomalacia, who in turn have been shown to have an increased aspiration risk [33,34]. We hypothesize that altered
4.2. Limitations of this study A major limitation is the retrospective nature of this study with reliance on recorded information at the time of surgery, and that our cohort represents a referred population. No information regarding swallow function for the non-referred patients was available. However, even if we assume there are no dysphagic patients in the non-referred group, prevalence of post-operative dysphagia remains high 30/83 (36%). Another significant limitation is a lack of preoperative investigation, meaning conclusions regarding cause and effect must be carefully considered. Dysphagia rates in our cohort may not necessary represent new onset findings, since children could have had pre-existing dysphagia; studies have shown up to 1/3 of children with congenital heart disease to have swallow abnormalities [15] and the possibility of a neurogenic dysphagia in this complex group has not been investigated. Diagnosis of LSCTS frequently occurs in an acute situation necessitating intubation and ventilation, precluding pre-operative swallow assessment. However, we strongly advocate preoperative assessment where possible. It is possible that the prevalence of dysphagia is under-reported in our cohort as not all patients underwent instrumental swallow assessment. In our unit we use clinical swallow assessment to provoke VFSS assessment to limit unnecessary radiation exposure. The diagnostic accuracy of a clinical swallow assessment is questioned in the literature, although for assessment of thin fluids has been found to have high 89
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sensitivity (92–100%) [26]. It is possible that cases of dysphagia were therefore missed. Use of FEES has not been routinely used at our center, predominantly due to issues achieving good compliance with this age group. Where good compliance can be achieved FEES may provide superior instrumental assessment allowing for pre- and post-surgical instrumental swallow assessment for all STP patients. Finally, the relatively small number of participants is acknowledged but is a consequence of the rarity of the condition and procedure, even though we are a national referral center.
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4.3. Conclusions and future plans Given the high prevalence of dysphagia we would advocate swallow evaluation including appropriate instrumental evaluation, when possible, upon a diagnosis of long segment tracheal stenosis. Future studies should be prospective, with extended follow up to enable investigation of the timeframe for resolution of dysphagia. A suggested dataset for such a prospective study is presented in Table 5. We have proposed a swallow evaluation care pathway to guide future management of such children (Fig. 1). Further exploration of the underlying pathophysiology, and the potential role of subglottic pressure changes, present exciting challenges for future studies. Funding This research did not receive any specific grant from funding agencies in the public, commercial, or not-for-profit sectors. Conflicts of interest None. Acknowledgements The authors would like to thank all clinicians and patients involved in this study. In particular thanks to M Ryan (Speech and Language Therapist) for her involvement in the analysis of videofluoroscopies. Thanks also to R Grant for his advice and support with statistical analysis. References [1] M. Elliott, et al., Slide tracheoplasty, Curr. Opin. Otolaryngol. Head Neck Surg. 16 (1) (2008) 75–82. [2] X. Li, et al., Management of symptomatic congenital tracheal stenosis in neonates and infants by slide tracheoplasty: a 7-year single institution experience, Eur. J. Cardio. Thorac. Surg. 38 (5) (2010) 609–614. [3] C.R. Butler, et al., Outcomes of slide tracheoplasty in 101 children: a 17-year singlecenter experience, J. Thorac. Cardiovasc. Surg. 147 (6) (2014) 1783–1789. [4] N. Fanous, et al., Anterior pericardial tracheoplasty for long-segment tracheal stenosis: long-term outcomes, J. Thorac. Cardiovasc. Surg. 139 (1) (2010) 18–23 discussion 23-5. [5] K. Kimura, et al., Tracheoplasty for congenital stenosis of the entire trachea, J. Pediatr. Surg. 17 (6) (1982) 869–871. [6] M. Elliott, et al., The management of congenital tracheal stenosis, Int. J. Pediatr. Otorhinolaryngol. 67 (Suppl 1) (2003) S183–S192. [7] C.L. Backer, et al., Tracheal surgery in children: an 18-year review of four techniques, Eur. J. Cardio. Thorac. Surg. 19 (6) (2001) 777–784. [8] W.A. Clement, et al., Unilateral vocal cord paralysis following patent ductus arteriosus ligation in extremely low-birth-weight infants, Arch. Otolaryngol. Head Neck Surg. 134 (1) (2008) 28–33.
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