Current Management and Outcome of Tracheobronchial Malacia and Stenosis Presenting to the Paediatric Intensive Care Unit

Home , Bronchomalacia, Subglottic stenosis, Tracheobronchomalacia, Tracheomalacia

Intensive Care Med 52001) 27: 722±729 DOI 10.1007/s001340000822 NEONATAL AND PEDIATRIC INTENSIVE CARE

David P.Inwald Current management and outcome Derek Roebuck Martin J.Elliott of tracheobronchial malacia and stenosis Quen Mok presenting to the paediatric intensive care unit

Abstract Objective: To identify fac- but was not related to any other fac- Received: 10 July 2000 Final Revision received: 14 Oktober 2000 tors associated with mortality and tor. Patients with stenosis required a Accepted: 24 October 2000 prolonged ventilatory requirements significantly longer period of venti- Published online: 16 February 2001 in patients admitted to our paediat- latory support 5median length of Springer-Verlag 2001 ric intensive care unit 5PICU) with ventilation 59 days) than patients tracheobronchial malacia and with malacia 539 days). stenosis diagnosed by dynamic con- Conclusions: Length of ventilation Dr Inwald was supported by the Medical Research Council. This work was jointly trast bronchograms. and bronchographic diagnosis did undertaken in Great Ormond Street Hos- Design: Retrospective review. not predict survival. The only factor pital for Children NHSTrust, which re- Setting: Tertiary paediatric intensive found to contribute significantly to ceived a proportion of its funding from the care unit. mortality was the presence of com- NHSExecutive; the views expressed in this Patients: Forty-eight cases admitted plex cardiac and/or syndromic pa- publication are those of the authors and not to our PICU over a 5-year period in thology. However, patients with necessarily those of the NHSexecutive. whom a diagnosis of tracheobron- stenosis required longer ventilatory chial malacia or stenosis was made support than patients with malacia. by dynamic contrast bronchography D.P.Inwald 5)) 51994±1999). Key words Tracheal stenosis ´ Portex Anaesthesia, Intensive Therapy Interventions: Conservative man- Bronchomalacia ´ Tracheomalacia ´ and Respiratory Medicine Unit, Institute of Child Health, agement, tracheostomy and long- Tracheobronchomalacia ´ 30 Guilford Street London WC1N 1EH, term ventilation, surgical correction, Bronchogram ´ Ventilation ´ UK internal or external airway stenting. Mortality E-mail: [email protected] Measurements and results: Record- Phone: +44-20-74059200 ing of clinical details, length of inva- Fax: +44-20-78298634 sive ventilation and appearance at M.J.Elliott contrast bronchography. Five Department of Cardiothoracic Surgery, groups of patients were defined: iso- Great Ormond Street Hospital for lated primary airway pathology Children NHSTrust, Great Ormond Street, 5n = 7), ex-premature infants London WC1N 3JH, UK 5n = 11), vascular rings 5n = 9), complex cardiac and/or syndromic pa- D.Roebuck Department of Diagnostic Imaging, thology 5n = 17) and tracheo- Great Ormond Street Hospital for oesophageal fistulae 5n = 4). The Children NHSTrust, Great Ormond Street, overall mortality was 29%. Median London WC1N 3JH, UK length of invasive ventilation in survivors was 38 days and in patients Q.Mok who died 45. Mortality was highest Paediatric Intensive Care Unit, Great Ormond Street Hospital for in the patients with complex cardiac Children NHSTrust, Great Ormond Street, and/or syndromic pathology London WC1N 3JH, UK 5p = 0.039 Cox regression analysis) 723

Introduction bronchus intermedius and peripheral bronchi). A pressure monitor was connected to the airway circuit via a Y connector and when Tracheobronchomalacia is a condition of dynamic air- collapse was found positive end-expiratory pressure 5PEEP) was applied and opening pressures noted. Airways were defined as way collapse during expiration. In tracheal stenosis, stenosed when the narrowing was fixed and unaffected by PEEP fixed airway narrowing causes airway obstruction in in- up to 25 cmH2O and malacic when the narrowing was dynamic spiration and in expiration. These conditions, first de- and improved with positive airway pressure. Infants with tracheal scribed in 1952 [1], may be primary, when cartilaginous stenosis with distal malacia were classified as tracheal stenosis for rings are congenitally malformed, or secondary, due to the purposes of this analysis since the numbers of patients with degeneration of previously normal cartilage. In neona- both forms of pathology was small 5n = 10). Our bronchograms are not stored as cine-films or videos but as static images, so no at- tal chronic lung disease, development of the tracheo- tempt was made at retrospective blinded severity scoring as this bronchial tree may be abnormal and the cartilage may was felt to be unlikely to yield any useful information. be weak [2]. Extrinsic airway compression by a vascular Data were analysed using SPSS 8.0 for Windows 5SPSS, UK). ring or by enlarged great vessels in the presence of a Analysis of survival and length of invasive ventilation was per- large shunt can result in permanent airway narrowing formed using Cox regression analysis. The Kruskal-Wallis test was [3]. Tracheo-oesophageal fistulae are often associated used to determine whether differences in the length of follow-up in survivors were significant. with malacia of a portion of the trachea at the point of insertion of the fistula. Airway abnormalities may develop in recurrent aspiration syndromes and in acquired cartilaginous disorders such as relapsing polychondritis. Results The severity of disease depends on the length, location and degree of narrowing in the affected airway seg- Of the 48 patients, 19 were female. The median age ments. at diagnosis was 137 days 5interquartile range Although primary isolated tracheobronchial malacia 62±213 days). The number of patients in each group rarely requires ventilatory support and is often self-lim- was as follows: isolated primary airway pathology 57), iting, infants with tracheobronchial malacia or stenosis ex-premature infants 511), vascular rings 59), complex with abnormal bronchograms who require ventilatory cardiac and/or syndromic pathology 517) and tracheo- support have been reported to have a high morbidity oesophageal fistulae 54). The overall mortality was and mortality, often related to airway obstruction and 14/48 529 %). Median length of follow-up in survivors respiratory failure [4]. This has not been our experience. was 439 days and was not significantly different between We therefore reviewed children admitted to our inten- any of the groups 5p = 0.635, Kruskal-Wallis test). Out- sive care unit with tracheobronchial malacia and stenos- come data is presented related to diagnosis and to oper- is, diagnosed at contrast bronchography, to determine ative procedure 5Tables 1 and 2). which factors affected outcome.

Isolated primary airway pathology

Materials and methods Seven patients with primary airway pathology were Following local ethics committee approval, the notes of 48 pa- identified. The median age at diagnosis was 66 days. tients, who had been admitted to our intensive care unit between One term infant was referred for surgical management 1994±1999 and in whom a diagnosis of tracheobronchial malacia of congenital subglottic stenosis, tracheobronchomala- or stenosis had been made at contrast bronchography, were re- cia being diagnosed when weaning difficulties were en- viewed. Information collected included underlying diagnosis, length of invasive ventilation around the time of diagnosis, surgical countered following surgery. Invasive ventilation was interventions, mortality, time between diagnosis and the present defined in this group as the time from first intubation day for survivors and time between diagnosis and death for those for airway obstruction to death or weaning to either who died. Underlying diagnostic categories were defined as isolat- extubation or continuous positive airway pressure ed primary airway pathology or airway pathology associated with 5CPAP) or biphasic positive airway pressure 5BiPAP) neonatal chronic lung disease, complex cardiac and/or syndromic via a tracheostomy. The median length of invasive venti- pathology, vascular rings or tracheo-oesophageal fistulae. Radiographic data were also reviewed. Bronchography was lation of survivors was 60 days. Five patients had isolat- performed as previously described [5]. The patients were lightly se- ed tracheobronchomalacia and two had tracheal stenos- dated, intubated and were breathing spontaneously. The endotra- is with airway malacia distal to the stenotic segment. cheal tube was maintained in a high position with the tip in the sub- Two patients with mild tracheobronchomalacia were ex- glottic region. Opacity was obtained by bolus injections of 0.5±2 ml tubated, one after a cricoid split procedure for congeni- of contrast medium 5Omnipaque, Nycomed, Birmingham, UK), tal subglottic stenosis, the other requiring no interven- injected into the airway via the endotracheal tube through a 3.7 Fr angiography catheter with a single end-hole. Contrast dis- tion. The latter patient was excluded from the statistical persal was obtained by hand ventilation. Up to five affected sites analysis as he had severe combined immune deficiency were identified 5trachea, left main bronchus, right main bronchus, and died from graft versus host disease following bone 724

Table 1 Outcome by primary surgical procedure Procedure No. Diagnosis Further procedures Invasive ventilation Dead Alive Still 5median, days, ventilated survivors only) Aortopexy 2 Tracheomalacia None 38 0 2 0 Division of vascular 1 Tracheal stenosis None 14 0 1 0 ring Slide tracheoplasty 4 Tracheal stenosis Tracheostomy 54), 61 1 3 1 Palmaz stent 51) Patch tracheoplasty 3 Tracheal stenosis Hagl stent and tra- 104 1 2 0 cheostomy 51), Palmaz stent 52) Resection and end- 2 Severe focal tracheo- None 51), Palmaz 17 0 2 0 to-end anastomosis malacia stent 51) Hagl stent 3 Focal tracheomalacia Tracheostomy 51), 12 2 1 1 52), tracheal stenosis Palmaz stent 51) 51) Tracheostomy and 19 Tracheo broncho- None 518), Palmaz 42 8 11 7 long-term ventilation malacia stent 51) No surgical interven- 13 Tracheo broncho- None 29 2 11 0 tion malacia Died on table during 1 Tracheal stenosis None 34 1 0 0 surgery

marrow transplantation. One patient with severe focal Two infants were extubated to room air without any tracheomalacia had a resection and end-to-end anasto- intervention. The remaining nine patients all required mosis and was then extubated to room air. The remain- tracheostomy for long-term ventilation with either ing four patients had a variety of surgical procedures. CPAP or BiPAP. One patient died of viral encephalitis Three of these four patients survived but all continue and was excluded from the statistical analysis. One pa- to require CPAP or BiPAP via a tracheostomy, either tient, who had four major bronchi affected by disease, at home or in hospital. One patient with multiple areas died following an episode of airway obstruction. Of the of malacia affecting all major bronchi died post-opera- seven survivors, three have been weaned from ventila- tively due to airway obstruction. tory support and two have been decannulated. All survivors have been discharged from hospital.

Neonatal chronic lung disease Vascular rings Eleven ex-premature infants 5median gestation 28.5 weeks) with bronchopulmonary dysplasia and one term Nine patients with vascular rings were identified, two baby with chronic lung disease due to meconium aspira- with a double aortic arch and seven with pulmonary ar- tion were identified. The median age at diagnosis was tery slings. One patient had isolated tracheobroncho- 199 days. All had been referred for investigation of their malacia, two had isolated tracheal stenosis and six had continued ventilatory requirements in the absence of se- tracheal stenosis with distal bronchomalacia. All of vere parenchymal lung disease and all had tracheobron- these patients presented in infancy with airway obstruc- chomalacia. Two had been referred for surgical manage- tion, with a median age at diagnosis of 115 days. Length ment of their subglottic stenoses, tracheobronchomala- of invasive ventilation was defined in this group as the cia being diagnosed when weaning difficulties were en- time from first intubation for airway obstruction to countered following cricoid split procedures. Length of death, weaning to extubation or CPAP or BiPAP via a invasive ventilation was defined in this group as the tracheostomy. The median length of invasive ventilation time from first failed extubation 5following the period of survivors was 48 days. of ventilatory support for neonatal lung disease) to All patients required division of vascular rings. Addi- death, weaning to extubation, or CPAP or BiPAP via a tional primary procedures included slide tracheoplasty tracheostomy. The median length of invasive ventilation 53) and pericardial patch tracheoplasty 52), aortopexy in survivors was 23 days. 51) and placement of a Hagl external stent 51). One pa- 725

Table 2 Outcome by diagnosis Diagnosis Airway pathology No. Initial procedure Invasive ventilation Dead Alive Still 5median, days, ventilated survivors only) Primary airway Tracheobroncho- 5 Tracheostomy 52), 50 1 4 1 malacia no intervention 52), resection and end- to-end anastomosis 51) Tracheal stenosis 2 Slide tracheoplasty 51), 96 1 1 1 pericardial patch tracheoplasty 5with Hagl stent) 51) Neonatal chronic Tracheobroncho- 11 Tracheostomy 59), 23 2 9 4 lung disease malacia no intervention 52) Tracheal stenosis 0 Vascular rings Tracheobroncho- 1 Division of ring 14 0 1 0 malacia Tracheal stenosis 8 Aortopexy 51), peri- 55 2 6 1 cardial patch tracheoplasty 52), slide tracheoplasty 53), Hagl stent 51), died on table 51) Complex cardiac Tracheobroncho- 17 Tracheostomy 58), 44 8 9 1 or syndromic malacia Hagl stent 52), resection, end-to-end anastomosis 51) and no intervention 56) Tracheal stenosis 0 Tracheo-oesopha- Tracheobroncho- 4 No intervention 53), 34 0 4 0 geal fistula malacia aortopexy 51) Tracheal stenosis 0

tient died of a pulmonary hypertensive crisis during sur- lation was difficult and in some infants with shunts was gery. Three were extubated shortly after surgery. The thought to be due to enlarged great vessels compressing two patients who had pericardial patch tracheoplasties the airway. The median age at diagnosis was 76 days. were extubated and discharged from hospital after Length of invasive ventilation was defined as the time placement of Palmaz stents. The three patients who from first intubation for airway obstruction or cardiac had slide tracheoplasties all required tracheostomy for surgery to death or weaning to either extubation or long-term ventilation. One of these died from airway CPAP or BiPAP via a tracheostomy. The median length obstruction post-operatively and two were discharged of ventilation in survivors was 44 days. Five infants, who home on chronic ventilation but are now off ventilatory are still alive, were extubated and required no interven- support. tion for airway management. Of the remaining 12, airway included tracheostomy for long-term ventilation 58), Hagl external stent 52), resection and end-to-end anasto- Complex cardiac and/or syndromic pathology mosis 51) and no intervention 51). Five patients who had tracheostomies died, two of viral pneumonia, one of hy- Seventeen infants with complex pathology, 15 cardiac 57 pertrophic obstructive cardiomyopathy and two of air- of whom had multiple anomalies) and 2 syndromic, way obstruction. Both patients with Hagl stents died, were identified. These infants had a range of abnormali- one of acute respiratory distress syndrome 5ARDS) fol- ties and most had been admitted to PICU following car- lowing cardiac surgery and the other, who had multiple diac surgical procedures. All had tracheobronchomala- anomalies, of progressive respiratory failure when inten- cia. Tracheobronchomalacia had been suspected in most sive care was withdrawn. One other patient with multiple of these infants when post-operative weaning from venti- anomalies died of progressive respiratory failure when 726

Table 3 Causes of death 5CPAP continuous positive airway pressure, ARDS acute respiratory distress syndrome, RSV respiratory syncy- tial virus) Sex Diagnostic category Airway pathology Interventions Cause of death M Neonatal Tracheobronchomalacia Tracheostomy, CPAP Airway obstruction M Neonatal Tracheobronchomalacia Tracheostomy, CPAP Viral encephalitis M Vascular ring Tracheal stenosis Died on table during surgery Airway obstruction F Vascular ring Tracheal stenosis Division of ring, slide tracheo- Airway obstruction plasty, tracheostomy CPAP F Primary airway Tracheobronchomalacia Extubated to room air Graft versus host disease following bone marrow transplantation F Primary airway Tracheal stenosis External stent, redo stent, Airway obstruction tracheostomy CPAP F Complex cardiac Tracheobronchomalacia Hagl stent, Palmaz stent Airway obstruction, limita- tion of care due to other lesions M Complex cardiac Tracheobronchomalacia Hagl stent ARDSpost cardiopulmonary bypass M Complex cardiac Tracheobronchomalacia No intervention Airway obstruction, limita- tion of care due to other lesions M Complex cardiac Tracheobronchomalacia Tracheostomy CPAP RSV pneumonia F Complex cardiac Tracheobronchomalacia Tracheostomy CPAP Hypertrophic obstructive cardiomyopathy F Complex cardiac Tracheobronchomalacia Tracheostomy CPAP Influenza A pneumonia F Complex cardiac Tracheobronchomalacia Tracheostomy CPAP Airway obstruction M Complex cardiac Tracheobronchomalacia Tracheostomy CPAP Airway obstruction

intensive care was withdrawn. The patient who had a re- Statistical analysis of factors associated with death section and end-to-end anastomosis had further problems with airway obstruction, was extubated following Fourteen 529%) infants died 5Table 3). Twelve died of insertion of a Palmaz stent and is now at home. Of the cardiorespiratory causes, seven of airway obstruction, three survivors with tracheostomies, one has been dec- one during tracheal surgery, two of viral pneumonia, annulated and two remain on CPAP but are at home. one of ARDSfollowing cardiopulmonary bypass and one of hypertrophic obstructive cardiomyopathy 5HOCM). Two infants died of non-cardiorespiratory Tracheo-oesophageal fistula causes, one after bone marrow transplantation and one of viral encephalitis, and were excluded from the statis- Four patients with tracheo-oesophageal fistulae were tical analysis. Cox regression analysis was used to identi- identified. All had tracheomalacia, median age at diag- fy factors associated with death in the remaining 46 pa- nosis being 128 days. Length of invasive ventilation in tients. Patients were defined by diagnostic category this group was defined as the interval from first intuba- 5cardiac and/or syndromic and others), length of inva- tion for airway obstruction to weaning to extubation. sive ventilation, bronchographic diagnosis 5i.e. tracheo- The median length of ventilation was 34 days. Two pa- bronchomalacia or tracheal stenosis) and number of tients were extubated without any airway intervention, major airways affected. Patients with complex cardiac one was extubated following repair of his tracheo- and/or syndromic pathology were at significantly higher oesophageal fistula and one required aortopexy before risk of death than those in any other group 5p = 0.039) extubation was possible. All patients survived and none 5Fig. 1) but other factors were not found to be signifi- required long-term ventilation. A large number of pa- cant. tients with tracheo-oesophageal fistulae who have been treated with aortopexy in our institution have not been included in this analysis as their airway pathology did not require bronchography for diagnosis. 727

cheal stenosis were likely to require longer invasive ventilation 5median 59 days) than patients with tracheobronchial malacia 5median 39 days) and this difference was statistically significant 5p = 0.037) 5Fig. 2).

Discussion Mild tracheobronchial malacia or stenosis classically presents in early childhood with wheeze, cough, stridor and recurrent lower respiratory tract infections. It usu- ally resolves spontaneously as cartilaginous development of the airway occurs [6, 7]. More severe lesions present in early infancy with respiratory failure, epi- Fig.1 Survival curves of different diagnostic groups sodes of profound cyanosis, dying spells, reflex apnoea and inability to wean from mechanical ventilation following preterm delivery or surgical procedures and of- Statistical analysis of factors associated with prolonged ten present to intensive care. Although mortality and invasive ventilation morbidity in the group requiring intensive care are widely assumed to be poor, there has only been one Cox regression analysis was used to identify factors as- large series published to date. In that series, from Mel- sociated with prolonged invasive ventilation. Four pa- bourne, 62 patients with tracheobronchomalacia or tients who died during the initial period of invasive venti- stenosis who presented between 1986 and 1995 were re- lation were excluded from this analysis as length of venti- viewed and those ventilated for longer than 3 weeks lation was limited by death in these infants. Patients were with abnormal bronchograms had a mortality of 100% defined by diagnostic category, number of major airways [4]. However, since then there have been many advanc- affected and bronchographic diagnosis 5stenosis or mala- es in the management of the child with critical airway le- cia). Length of invasive ventilation was not significantly sions, including the use of external [8] and internal [9, influenced by the number of major airways affected or 10] airway stents and advances in tracheal surgery [11, by the diagnostic category. However, patients with tra- 12, 13]. Our experience over the last 5 years suggests

Fig.2 Length of invasive ventilation by bronchographic diagnosis 5survivors only) 728 that the majority of infants with these lesions can now tracheostomy and long-term home ventilation [18], survive, even following prolonged ventilation. which improves pulmonary mechanics and prevents dynamic collapse [19, 20] until the airway grows sufficient- ly to allow ventilatory support to be withdrawn. Howev- Diagnosis er, some infants with severe localised proximal disease benefit from surgical management. Our current strategy The most important aspect of diagnosis is a high index is patch tracheoplasty and repair of associated lesions in of suspicion in the groups of infants outlined above. long segment tracheal stenosis and resection with end- The association of airway abnormalities with tracheo- to-end repair in short segment tracheal stenosis. Slide oesophageal fistulae and vascular rings is well recognis- tracheoplasties are now rarely performed. Patients de- ed. However, although there have been reports of tra- veloping restenosis or malacia are treated with internal cheobronchial malacia and stenosis occurring in associa- wire mesh Palmaz stents or external Gore-Tex Hagl tion with both neonatal chronic lung disease [2] and stents and, if necessary, tracheostomy and long-term congenital cardiac disease 5due to either enlarged great ventilation. Mild tracheomalacia, particularly if associ- vessels or conduits) [14, 15, 16], the associations are not ated with a tracheo-oesophageal fistula, is treated with widely recognised and it is likely that many patients aortopexy [21]. Severe short segment tracheomalacia is with clinically important airway pathology remain undi- treated with resection and end-to-end anastomosis and agnosed. long segment severe tracheomalacia is treated with ex- A diagnosis of tracheobronchomalacia requires as- ternal Gore-Tex Hagl stents. Patients developing rest- sessment of the trachea and bronchi throughout the re- enosis or malacia are treated with balloon dilatation, in- spiratory cycle with demonstration of dynamic airway ternal wire mesh Palmaz stents or tracheostomy and collapse in expiration. Contrast bronchography is cur- long-term ventilation. The management of these infants rently the investigation of choice for ventilated infants is evolving and the use of stents rather than surgery as a in our institution [5]. It enables assessment of the tra- primary intervention in malacic airways is likely to in- chea and the more distal bronchi, unlike bronchoscopy, crease if their design improves. and since it is performed in real time with the infant spontaneously breathing, the airways are assessed throughout the respiratory cycle. It is simple to perform Outcome in infants who are already intubated and is also useful in diagnosing fixed airway stenosis. Methods have been In our patient population, survival was significantly af- suggested for quantitative severity scoring of broncho- fected by one factor among those studied ± the existence graphic data [17] but, in our experience, clinical assess- of associated complex cardiac and/or syndromic patholo- ment of the child is still paramount in determining gy. Although most patients died of respiratory causes which infants should be referred for long-term ventila- 5Table 3), included amongst the respiratory deaths in tion and/or surgical management. Bronchoscopy is still the group with complex cardiac and/or syndromic pa- necessary to identify complete tracheal rings and exam- thology are two deaths following withdrawal or limita- ination of the airway under direct vision by the surgeon tion of care and three due to parenchymal lung disease at operation has been more useful than imaging in de- rather than airway obstruction. Clearly the existence of ciding which surgical approach is most suitable for sev- significant non-airway pathology in these patients may eral patients. have contributed to their ªrespiratoryº deaths. Length Distal malacic lesions often present after proximal of invasive ventilation and bronchographic diagnosis lesions have been treated. This is particularly the case had no impact on outcome. Our data are at odds with in infants with neonatal chronic lung disease who may the series from Melbourne in which there was a much be referred for treatment of subglottic stenosis and in in- higher mortality. There are two possible reasons for this. fants with vascular rings following surgery. In these in- Firstly, our patients might be significantly different from fants, areas of distal malacia should, therefore, be sear- those reported in the previous study. Although there ched for prior to any intervention in order to guide man- was a similar increased male to female ratio in both our agement. series 529/48 patients male) and in the Melbourne series 538/62 patients male), more patients in the Melbourne study had congenital cardiac disease 541/62 patients) Management compared to our study 517/48 patients). Secondly, the patients reported from Melbourne are from an earlier ep- Infants requiring intensive care may need a variety of och and thus may not have benefited from recent advanc- management approaches. Treatment should be individ- es in the management of critical airway obstruction. ualised. Infants with very mild disease may require no Length of invasive ventilation in survivors was signif- intervention. Moderate airway malacia often requires icantly longer if tracheal stenosis was present. This re- 729 flects the tendency for these patients to be managed longed ventilation in the presence of major broncho- with tracheal surgery, the need for prolonged ventilation graphic abnormalities. Mortality is more dependent on following operation and the tendency for restenosis re- pathology outside the airway than it was in the past. quiring further interventions. Length of ventilation was The improved outcome in recent years probably reflects not affected by number of affected major bronchi or by advances in intensive care and surgical management, in- underlying diagnosis. cluding new surgical procedures and the use of internal We recognise the need to study respiratory and neu- and external airway stents. The problems of infants rodevelopmental outcome and quality of life in our with critical airway lesions are often extremely complex long-term survivors and their families. This will be the and are best managed by a multidisciplinary team. subject of future research. In conclusion, it is possible for infants with tracheo- Acknowledgements The authors wish to thank Dr. Isky Gordon, bronchial malacia or stenosis to survive episodes of pro- who performed many of the bronchograms described in this study.

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