Congenital Lobar Emphysema: Case Report of Bilateral Involvement

May, Romulus L.; Meese, Ernest H.; Timmes, Joseph J.

doi:10.1016/s0022-5223(19)33369-0

Cited by 18 publications

(7 citation statements)

References 2 publications

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“…Congenital lobar emphysema (CLE) as a rare condition has an unknown etiology and commonly present in the neonatal age [5]; our case is a 17 day neonate and we could not determine the possible etiology in the patient. The condition is more prevalent among the males, and commonly seen in neonatal age group as documented in most literatures, the case under presentation is a male neonate, thereby conforming to that documented in most literatures [5,9,12]. The patient presented with features of respiratory distress; these features are those documented among patients with CLE in most literatures [5][6][7][8].…”

Section: Discussionsupporting

confidence: 56%

“…Congenital lobar emphysema is usually unilateral affecting the upper left lobe in about 43%, followed by the middle right lobe in 32%, with bilateral involvement of the lobes of the lung been reported [9,11,12]. Congenital lobar emphysema is often associated with congenital heart disease with a 12-20% concomitant rate, the presence of congenital heart disease especially in infants with respiratory distress symptom should be considered [1,13].…”

Section: Introductionmentioning

confidence: 99%

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Congenital Lobar Emphysema in a 17-Day-Old Male Neonate: The Plain Radiographic Features and a Case Report

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et al. 2022

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Congenital lobar emphysema (CLE) is a rare respiratory disorder which allows air to enter the lungs but can’t escape causing over-inflation of the lobes of the lung. This condition (CLE) is commonly detected in the newborn or young infants but some cases are diagnosed in adulthood. Congenital lobar emphysema is a rare congenital malformation with a prevalence of 1 in 20,000 to 1 in 30, 000, and more prevalent in males, with a male to female ratio of about 3:1. This is a 17 day old male neonate that presented with respiratory difficulty and .had a supine chest radiograph that showed an overinflated left lung field more on the upper and middle zones with flattening of the left hemi diaphragm, marked shift of the heart and mediastinum to the right with some degree of loss of volume involving the right lung. We report the radiographic findings of this case due the rare nature of congenital lobar emphysema.

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Section: Discussionsupporting

confidence: 56%

Section: Introductionmentioning

confidence: 99%

Congenital Lobar Emphysema in a 17-Day-Old Male Neonate: The Plain Radiographic Features and a Case Report

Mb¹,

Au²,

Ih³

et al. 2022

JTCS

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“…There are various reasons described for CLE, and the principal mechanism is that the affected bronchus allows passage of air on inspiration but only limited expulsion of air on expiration leading to over-expansion of the affected lobe [1][2][3][4][5][6][7][8]. This air trapping may be due to (a) dysplastic bronchial cartilages creating a ball valve effect, (b) endobronchial obstruction from mucus plug or extensive mucosal proliferation and infolding, (c) extrinsic compression of bronchi from aberrant cardiopulmonary vasculature or enlarged cardiac chambers, and (d) diffuse bronchial abnormalities which may or may not related to infections [1][2][3][4][5][6][7][8]. However, in approximately 50% of cases, the aetiology is unknown [7][8][9][10][11][12].…”

Section: Discussionmentioning

confidence: 99%

“…Congenital lobar emphysema (CLE) is a rare developmental anomaly of the lung characterized by over-distension and air trapping in the affected lobe secondary to bronchomalacia or absent cartilage [1][2][3][4][5][6][7][8][9][10][11][12][13][14][15][16]. It presents as respiratory distress due to ventilation-perfusion mismatch because of compression atelectasis on the ipsilateral or the contralateral side [1][2][3][4]. Bilateral involvement is exceptionally rare and has been reported only in 13 scattered case reports and series with 22 cases reported [1][2][3][4][5][6][7][8][9][10][11][12][13][14][15][16].…”

Section: Introductionmentioning

confidence: 99%

“…It presents as respiratory distress due to ventilation-perfusion mismatch because of compression atelectasis on the ipsilateral or the contralateral side [1][2][3][4]. Bilateral involvement is exceptionally rare and has been reported only in 13 scattered case reports and series with 22 cases reported [1][2][3][4][5][6][7][8][9][10][11][12][13][14][15][16]. Of these, 5 were bilobar and 15 were bilateral involvement (Table 1).…”

Section: Introductionmentioning

confidence: 99%

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Bilateral congenital lobar emphysema: a rare entity and a therapeutic challenge

2022

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Background Bilateral congenital lobar emphysema is an extremely rare condition, and detailed workup of these patients is very important. Operative intervention is usually based on radiological, biochemical, and clinical details. Bronchoscopy gives an added advantage in cases with diagnostic dilemmas and makes the treatment more evidence based. We here present three cases and review the literature for this rare entity. Material and methods We retrospectively collected data about all the cases of bilateral CLE operated by the authors at various centres over the period of 3 years from January 2016 to December 2018. Results We managed three cases of bilateral CLE, and all three had unique details based on which treatment was planned. We operated three cases of this very rare entity and compared our findings with 23 cases from the literature reviewed. Conclusion We propose that bronchoscopy should be an essential component in the management of all cases of CLE. It helps in defining pathology, the severity of the disease, and the decision on which side to be operated first and avoids unnecessary simultaneous lobectomies. We recommend case selection on basis of radiological, biochemical, clinical, and bronchoscopy criteria. Based on this, an approach of sequential lobectomies is less risky and should be advocated in most of the patients.

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