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Unilateral Hyperlucent Lung: the Case for Investigation

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Unilateral Hyperlucent Lung: the Case for Investigation Thorax: first published as 10.1136/thx.35.10.745 on 1 October 1980. Downloaded from Thorax, 1980, 35, 745-750 Unilateral hyperlucent lung: the case for investigation SHEILA A McKENZIE, D J ALLISON, M P SINGH, AND S GODFREY From the Department of Paediatrics and Neonatal Medicine and Department of Diagnostic Radiology, Hammersinith Hospital, London ABSTRACT Seventeen children with unilateral hyperlucent lungs were referred for investigation. Of the 11 who had a referring diagnosis of possible Macleod's syndrome only two were shown to have post-viral bronchiolitis. Three of the 11 had conditions that required surgical treatment and a further two with bronchiectasis were treated medically. To avoid confusion we suggest that Macleod's syndrome is reserved exclusively for children with post-viral bronchiolitis. Radio- isotopic regional lung function studies were useful in the investigation of the subjects from three points of view. Firstly, they distinguished children with primary perfusion abnormalties and normal ventilation, secondly, they defined the extent of altered respiratory function, and thirdly, they were able to distinguish compensatory emphysema from congenital lobar emphysema. As bronchography and bronchoscopy may be hazardous in small children with poor respiratory reserve, such regional studies may be useful in indicating which patients do not require further invasive investigation. copyright. Of the many causes of unilateral hyperlucent lung aetiological diagnosis was uncertain at the time of in children, congenital lobar emphysema and referral; 11 were referred with a possible diagnosis foreign body inhalation are among the most com- of Macleod's syndrome. We describe here the re- mon. In general neither of these conditions is sults of our investigations and suggest a line of http://thorax.bmj.com/ difficult to diagnose and the affected lung, or lobe, investigation for children presenting in this way. is usually hyperinflated. There is, however, a small group of children with hyperlucent lungs in Methods whom the diagnosis is not immediately apparent. Unilateral hyperlucent lungs which are normal or The age of patients, history, presenting symptoms small in size are occasionally seen on radiographs and signs, chest radiograph appearances, and in children who present with longstanding respir- initial diagnoses are shown in table 1. Seven chil- atory symptoms. Macleod's or Swyer-James syn- dren were known to have congenital heart disease on September 23, 2021 by guest. Protected drome' 2 presents in this way and is now believed but it was not clear whether their respiratory to follow respiratory infections such as measles,3 symptoms and radiographic appearances were adenovirus,4 and mycoplasma.3 5 Bronchograms related entirely to this or whether they had ad- show poor filling of the peripheral bronchi and ditional pulmonary disease. Taking the group as a histologically the bronchi and bronchioles show whole the diagnosis of Macleod's syndrome had evidence of chronic inflammatory changes. Some been considered at some time in 11 children. At clinicians nevertheless prefer to use the term the time of referral, four of these children had Macleod's syndrome simply to describe the radio- small hyperlucent lungs, six had lungs of normral graphic appearances and do not imply any size, and one (patient 12) had a large hyperlucent aetiology. lung which had appeared of normal size previously. Over the past three years 17 children with uni- All children had chest radiographs and fluor- lateral hyperlucent lungs have been referred to us oscopy and underwent regional lung function for investigation. In many of these children the studies. These were carried out using nitrogen-13. Both bolus inhalation and bolus injection studies Address for reprint requests: Dr SA McKenzie, Department ofPaedi- were carried out.6 The distribution of gas and atrics and Neonatal Medicine, Hammersmith Hospital, Du Cane Road, London W12 OHS blood throughout both lung fields was determined. The clearance of gas from the lungs after bolus 745 Thorax: first published as 10.1136/thx.35.10.745 on 1 October 1980. Downloaded from 746 Sheila A McKenzie, D J Allison, M P Singh, and S Godfrey Table 1 Patients' ages, presenting symptoms, chest radiographic appearances, and initial diagnosis Patient Age Presenting symptons/signs Chest radiographic appearance Referring/initial diagnosis and history 1 2 yr Chronic respiratory failure. Small hyperlucent left lung ?Macleod's syndrome "Measles" age 1 yr. High adenoviral titres 2 9 mo Wheezing since age 6 wk Changes suggestive of right middle lobe and ?Macleod's syndrome lower lobe disease up to 6 mo. Age 9 mo, hyperlucent sniall right lung 3 11 yr Pertussis aged 4 mo and Recurrent collapse/consolidation left lower lobe ?Macleod's syndrome pneumonia. Haemoptysis age I vr in early childhood. Hyperlucency left lower zone age II yr. Lung of normal size 4 6 yr Recurrent chest infections Hyperlucent left lung of normal size ?Macleod's syndrome 5 6 yr Recurrent wheezing Normal until age 3 yr; subsequently small ?Macleod's syndrome hyperlucent left lung 6 2 yr Recurrent chest infections and Hyperlucent left lung of rormal size ?Macleod's syndrome wheezing since age 3 mo 7 4 yr Short of breath on exercise. Hyperlucent left upper zone. Left lung of normal ?Macleod's syndrome Known ventricular septal defect size 8 7 yr Murmur since birth. Valvar Small hyperlucent left lung on routine X-ray ?Macleod's syndrome pulmonary stenosis at cardiac catheterisation. Asymptomatic 9 3 mo Patent ductus arteriosus ligated Hyperlucent large left upper lobe Congenital lobar emphysema age I mo. Persistent tachypnoea 10 4 days Respiratory distress from birth Hyperlucent large left lung Respiratory distress syndrome I1 4 mo Respiratory distress and cardiac Hyperlucent right middle lobe ?Congenital lobar emphysema murmur from birth ?Compensatory emphysema 12 6 mo Tachypnoea during infancy Hyperlucent large left lung which looked to be of ?Macleod's syndrome copyright. normal size on previous films 13 13 mo Endocardial fibroelastosis. Hyperlucent left lung of normal size ?Macleod's syndrome Tachypnoea for six mo 14 12 yr Mustard repair of transposition Hyperlucent left upper zone. Left lung of normal Asthma great vessels age 4 yr. Recurrent size wheezing since 15 5 mo Ventricular septal defect. Referred Hyperlucent left lung. Size normal large or small ?Congenital lobar emphysema http://thorax.bmj.com/ age 2 mo. Persistent tachypnoea on different X-rays ?Compensatory emphysema and wheeze 16 2-5 yr Recurrent chest infection and Hyperlucent left lung of normal size ?Macleod's syndrome wheeze 17 1 yr Cardiac murmur. Known Small hyperlucent right lung Unilateral pulmonary artery. unilateral pulmonary artery. ?Ventilatory function of right lung Poor exercise tolerance before surgery inhalation represented fractional ventilation and of the principal bronchi, the bronchogram was on September 23, 2021 by guest. Protected was determined for each of four areas, right and preceded by bronchoscopy which was performed left upper and lower zones. The clearance of gas in the radiology department during the same from the lungs after the injection study reflected anaesthetic. After bronchoscopy an endotracheal ventilation of perfused areas. Thus any delay in tube was inserted and a pre-curved nylon cannula clearance of 13N suggested gas trapping and any (OD 2-1 mm; Portex Ltd) passed through the tube difference in the rates of clearance after inhalation into the lungs. A suspension of propyliodone BP and injection studies represented ventilation- was used for bronchography (Dionosil Oily, Glaxo perfusion imbalance. Depending on the results of Laboratories Ltd) which was performed under the regional studies further investigations were fluoroscopic control with appropriate films exposed carried out as detailed in table 2. from an under-couch tube (focal spot 06 mm). Ten children in whom a definite diagnosis re- Where the respiratory reserve was adequate bi- mained uncertain after the regional studies were lateral bronchograms were obtained, the lungs referred for bronchoscopy or bronchography or being catheterised selectively in turn. Where ab- both. Bronchography was performed under general normal variations in bronchial calibre were ob- anaesthetic, and where previous investigations had served during fluoroscopy, they were recorded on indicated the possibility of an abnormality of one 70 mm film exposed at three frames per second. Thorax: first published as 10.1136/thx.35.10.745 on 1 October 1980. Downloaded from Unilateral hyperlucent lung: the case for investigation 747 Table 2 Investigations and final diagnoses Patient Other investigations Final diagnosis I Fluoroscopy-mediastinal movement to hyperlucent side on Macleod's syndrome with bilateral disease (on the basis inspiration. Bronchoscopy-no bronchial obstruction seen. of regional studies) Open lung biopsy-chronic round cell infiltrate around bronchioles 2 Bronchogram-narrowed RML and RLL bronchi with poor Macleod's syndrome peripheral filling. RML and RLL lobectomies for repeated pneumonia-chronic round cell infiltrate around bronchioles 3 Bronchoscopy-white tenacious sputum from LLL bronchus. Bronchiectasis Bronchogram-saccular bronchiectasis lingula and LLL 4 Bronchoscopy-compression ofposterior wall of L main Probable abnormal position ofleft main pulmonary bronchus with pulsation in time with cardiac cycle artery (no further investigations permitted) 5 Fluoroscopy-no mediastinal shift. Occlusion of
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