J R Army Med Corps: first published as 10.1136/jramc-138-02-08 on 1 June 1992. Downloaded from

J R A rmy Med Corps 1992; 131: 95-98 Case Report Tracheobronchomalacia A Rare Cause of Respiratory Distress in Children

Lt Col B Moorthy MD, MRCP, RAMC Consultant Paediatrician

Cap! H Robertson MRCP,RAMC Specialist in Paediatrics BMH Rinleln. BFPO 29

SUMMARY: TracheobroDchomalacia is a rare cause of respiratory distress in children. An infant with tracheohron­ cbomalacia is presented. The aetiology, pathophysiology and management are discussed. Case Report A six month old male infant was admitted to the history suggestive of a foreign body or choking and he Children's Ward at the Cambridge Military Hospital did not have any other systemic symptoms. with symptoms of increasing wheeze. He had been Birth history. past history and family history were • symptomatic for nearly two months prior to admission non-contributory. On examination, at the time ofguest. Protected by copyright. ad­ with cough and wheeze for which he had been prescribed mission, he was well grown, apyrexial, acyanotic but in antibiotics by the General Practitioner. There was no respiratory distress. There was no evidence suggestive of http://militaryhealth.bmj.com/ on September 26, 2021 by

Fig 1. X-ray of the chest (expiratory film) shows evidence Fi~ 2. Follow up X-ray done during re-admission (io­ of underinflation of the right with hyperinflation of splratory film) shows hyperinflation of the left lung with the left lung with mediastinal shift. mediastinal shift. J R Army Med Corps: first published as 10.1136/jramc-138-02-08 on 1 June 1992. Downloaded from 96 Tracheobronchomalacia

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-. L(.______~ b. Fig 3. KryptonlTechnetium 99 .entllation/perlusion scan. a. Impaired ventilation of the left lung (4] 070). Right lung (59070) shows patchy abnormalities of the upper lobe and apical segment of the lower lobe. http://militaryhealth.bmj.com/ b. Decreased perfusion oftbe left lung (41 '70) and right lung (59070).

(A. Krypton: 1 anterior 2 & 3 posterior. B'. Technetium: 1 anterior 2 &3 posterior). chronic respiratory disease such as finger clubbing, ray revealed hyperinflation of the left lung with Harrison's sulcus or pigeon chest. Systemic examination mediastinal shift to the right (Fig 2). A diagnosis of was unremarkable, except for diffuse rhonchi and foreign body in the left main was considered, crepitations. and he was transferred to the Hospital for Sick Children, Full blood count and biochemical screen were normal. Great Ormond Street, London, for and Initial chest X-ray showed an under-inflated right lung further respiratory investigations. On examination of

with hyperinflation of the left lung (Fig I), which was the at the Hospital for Sick Children, on September 26, 2021 by thought to be due to mucous plugging. He was started on he had tachypnoea (respiratory rate SO/ minute) in­ oral erythromycin, nebulised salbutamol and regular tercostal recession, bilateral wheeze and decreased air chest physiotherapy. He improved over the next 72 hours entry in the base of the left lung. Bronchoscopy showed and follow-up X-ray of the chest was normal. He was minimal subglottic narrowing, a tracheomalacic segment discharged home on oral salbutamol and erythromycin between middle and distal thirds with about 20070 nar­ with advice to continue regular chest physiotherapy. He rowing, and a 2cm long bronchomalacic segment of the was re-admitted 17 days later, with symptoms of in­ lelt main bronchus leading on to 80-90'10 obstruction. creasing respiratory distress and wheeze. Repeat chest X- Barium swallow and 2 D echocardiogram were normal, J R Army Med Corps: first published as 10.1136/jramc-138-02-08 on 1 June 1992. Downloaded from B Moorthy and H Robertson 97 ruling out vascular ring, double aortic arch or tracheobronchomalacia in these patients has recently pulmonary artery sling. Magnetic Resonance Imaging been reported (9). Tracheobronchomalacia produces a (MRI) of the chest did not reveal any abnormality to flaccid airway, which has an adequate lumen on inspi­ cause the tracheobronchomalacia. A ventilation ration but develops obstruction on expiration. This can perfusion scan showed decreased ventilation and lead to collapse of the airway, stridor and respiratory perfusion on both sides, the left side showing 330/0 of failure, infrequently needing ventilatory support overall ventilation and 41 % of overall perfusion (Fig 3). (10,11,12,13). The child is now nearly three years old and has re­ As compared to adults the tracheobronchial tree in mained symptomatically well, without further respira­ infants is extremely soft and the mucosa is redundant tory distress, requiring hospital admission, the use of especially around the carina (14). During forced expira­ regular bronchodilators or antibiotics. Chest X-ray has tion, crying and coughing, the lumen may become com­ remained normal. He is growing and developing pletely obliterated due to increased intrathoracic pres­ normally. It is probable that with advancing age, the sure exceeding the transbronchial pressure, leading to tracheobronchomalacic segments have become more herniation of the posterior membrane and anterio-pos­ competent, preventing further respiratory complica­ terior narrowing (15). tions. Usually children with primary tracheobron­ chomalacia recover spontaneously and become Discussion asymptomatic by the second year of life (2,7). Children Respiratory distress is a common reason for hospital with congenital heart disease, enlarged pulmonary ar­ admission in childhood. Chest infection, either bacterial teries and secondary will improve with or viral, and bronchial are the two most common surgical correction of the heart defect and arteriopexy causes. Foreign body inhalation and congenital ab­ (4,16). Recently in children with bronchomalacia and respiratory failure, endoscopic placement of expandable normalities of the , such as guest. Protected by copyright. tracheobronchomalacia, are rarer causes and require stents has been found to be useful, thus avoiding com­ flexible bronchoscopy to confirm the diagnosis and for plex high risk surgical procedures such as arteriopexy, removal ofthe foreign body. tracheopexy, external stenting, tracheobronchial re­ Tracheobronchomalacia can be either congenital or construction and pneumonectomy (17,18). acquired (1). Congenital tracheobronchomalacia may be an isolated abnormality, or be associated with other Acknowledgement congenital abnormalities such as , We would like to thank Major D Whitehouse, MRCP, or cleft palate (2). Recently Larsen's RAMC for his suggestions and Mrs P Tracey for kindly syndrome has been associated with tracheobron­ preparing the manuscript. chomalacia (3). Congenital heart disease with enlarged REFERENCES pulmonary arteries can present as bronchomalacia due 1. NUUTINEN J. Acquired tracheobronchomalacia - to external compression (4). bronchological follow-up study. Ann Clin Res Acquired tracheobronchomalacia, an adult disease, 1977; 9: 359-364. usually follows chronic bronchitis, bronchial carcinoma 2. BAXTER J D, DUNBAR J S. . Ann or pulmonary tuberculosis and often progresses with DIal Rhinal Laryngol1964; 72: 1013. http://militaryhealth.bmj.com/ increasing respiratory morbidity and mortality (1). 3. ROCK M J, GREEN C G, PAUL! R M, PETERS M E. 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